Genetic Animal Models / FMR1

FMR1 fragile X mental retardation 1

Model Species
Drosophila melanogaster
Gene Aliases
AT24755; BcDNA:GM08679; cg6203; CG6203; dfmr; dFMR; dfmr1; dFmr1; dFMR1; dFmrp; dFMRP; DFMRP; dfxr; dFXR; dfxr1; dFXR1; dFXRP; DmelCG6203; dmfr1; EP(3)3517; fmr; FMR; fmr1; FMR1; Fmrp; FMRp; FMRP; FXR
Reports
9
Associated Human Genes
FMR1
Models / Rescue Models
56 / 26
Rescue Model
-
Model Summary

Fmr1 knockout flies show altered circadian behavior and courtship behavior.

External Links
Reports related to FMR1 (9 Reports)
# Type Title Author, Year
1 Primary Drosophila lacking dfmr1 activity show defects in circadian output and fail to maintain courtship interest. Dockendorff TC , et al. (2002)
2 Primary Pharmacological rescue of synaptic plasticity, courtship behavior, and mushroom body defects in a Drosophila model of fragile X syndrome. McBride SM , et al. (2005)
3 Primary Short- and long-term memory are modulated by multiple isoforms of the fragile X mental retardation protein. Banerjee P , et al. (2010)
4 Primary Neural circuit architecture defects in a Drosophila model of Fragile X syndrome are alleviated by minocycline treatment and genetic removal of matr... Siller SS and Broadie K (2011)
5 Primary PDE-4 inhibition rescues aberrant synaptic plasticity in Drosophila and mouse models of fragile X syndrome. Choi CH , et al. (2015)
6 Primary Activity-dependent FMRP requirements in development of the neural circuitry of learning and memory. Doll CA and Broadie K (2015)
7 Primary Inability to activate Rac1-dependent forgetting contributes to behavioral inflexibility in mutants of multiple autism-risk genes. Dong T , et al. (2016)
8 Primary Hyperactive locomotion in a Drosophila model is a functional readout for the synaptic abnormalities underlying fragile X syndrome. Kashima R , et al. (2017)
9 Primary Acamprosate rescues neuronal defects in the Drosophila model of Fragile X Syndrome. Hutson RL , et al. (2018)
Show all Construct Details Show all Phenotypic Profiles

F_Fmr1_10_KO_HM

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Broadie laboratory, University of Utah (PMID 11733059)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Hutson RL , et al. (2018)
Circadian sleep/wake cycle - Not Reported - - Doll CA and Broadie K (2015)
Circadian sleep/wake cycle - Not Reported - - Siller SS and Broadie K (2011)
Motor phenotype General locomotor activity: Ambulatory activity Decreased General observations Third instar larvae Hutson RL , et al. (2018)
Motor phenotype Ataxia: turn angle Increased General observations Third instar larvae Hutson RL , et al. (2018)
Motor phenotype - Not Reported - - Doll CA and Broadie K (2015)
Motor phenotype - Not Reported - - Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: synaptic bouton number Increased Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Increased Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Axonal architecture: axonal branch number Increased Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Dendritic architecture: dendritic tree complexity Increased Immunocytochemistry 1 day post eclosion Doll CA and Broadie K (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: satellite boutons Increased Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Increased Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Increased Immunostaining adult stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Axonal architecture: branch number Increased Immunostaining adult stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Increased Immunostaining adult stage Siller SS and Broadie K (2011)
Neurophysiology - Not Reported - - Hutson RL , et al. (2018)
Neurophysiology Neuron remodeling: Dendritic pruning Increased Immunocytochemistry 1 day post eclosion Doll CA and Broadie K (2015)
Neurophysiology - Not Reported - - Siller SS and Broadie K (2011)
Repetitive behavior - Not Reported - - Hutson RL , et al. (2018)
Repetitive behavior - Not Reported - - Doll CA and Broadie K (2015)
Repetitive behavior - Not Reported - - Siller SS and Broadie K (2011)
Seizure - Not Reported - - Hutson RL , et al. (2018)
Seizure - Not Reported - - Doll CA and Broadie K (2015)
Seizure - Not Reported - - Siller SS and Broadie K (2011)
Sensory - Not Reported - - Hutson RL , et al. (2018)
Sensory - Not Reported - - Doll CA and Broadie K (2015)
Sensory - Not Reported - - Siller SS and Broadie K (2011)
Social behavior - Not Reported - - Hutson RL , et al. (2018)
Social behavior - Not Reported - - Doll CA and Broadie K (2015)
Social behavior - Not Reported - - Siller SS and Broadie K (2011)
Communications - Not Reported - - Hutson RL , et al. (2018)
Communications - Not Reported - - Doll CA and Broadie K (2015)
Communications - Not Reported - - Siller SS and Broadie K (2011)
Developmental profile - Not Reported - - Hutson RL , et al. (2018)
Developmental profile - Not Reported - - Doll CA and Broadie K (2015)
Developmental profile - Not Reported - - Siller SS and Broadie K (2011)
Emotion - Not Reported - - Hutson RL , et al. (2018)
Emotion - Not Reported - - Doll CA and Broadie K (2015)
Emotion - Not Reported - - Siller SS and Broadie K (2011)
Homeostasis - Not Reported - - Hutson RL , et al. (2018)
Homeostasis - Not Reported - - Doll CA and Broadie K (2015)
Homeostasis - Not Reported - - Siller SS and Broadie K (2011)
Immune response - Not Reported - - Hutson RL , et al. (2018)
Immune response - Not Reported - - Doll CA and Broadie K (2015)
Immune response - Not Reported - - Siller SS and Broadie K (2011)
Learning & memory - Not Reported - - Hutson RL , et al. (2018)
Learning & memory - Not Reported - - Doll CA and Broadie K (2015)
Learning & memory - Not Reported - - Siller SS and Broadie K (2011)
Maternal behavior - Not Reported - - Hutson RL , et al. (2018)
Maternal behavior - Not Reported - - Doll CA and Broadie K (2015)
Maternal behavior - Not Reported - - Siller SS and Broadie K (2011)
Molecular profile Gene expression Decreased Quantitative PCR (qRT-PCR) Third instar larvae Hutson RL , et al. (2018)
Molecular profile - Not Reported - - Doll CA and Broadie K (2015)
Molecular profile - Not Reported - - Siller SS and Broadie K (2011)

F_Fmr1_10_KO_HM_Acamprosate-1

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Acamprosate was made as a 100 mM stock solution and added to the standard cornmeal molasses agar as the final concentrations of 10 microM. Adult animals were placed in vials with acamprosate treated food and allowed to lay eggs. Only first generation, third-instar larvae were selected for the study.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: PMID 29317220
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Hutson RL , et al. (2018)
Motor phenotype General locomotor activity: Ambulatory activity Ameliorated General observations Third instar larvae Hutson RL , et al. (2018)
Motor phenotype Ataxia: turn angle Restored General observations Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: synaptic bouton number Restored Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Refractory Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Axonal architecture: axonal branch number Restored Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neurophysiology - Not Reported - - Hutson RL , et al. (2018)
Repetitive behavior - Not Reported - - Hutson RL , et al. (2018)
Seizure - Not Reported - - Hutson RL , et al. (2018)
Sensory - Not Reported - - Hutson RL , et al. (2018)
Social behavior - Not Reported - - Hutson RL , et al. (2018)
Communications - Not Reported - - Hutson RL , et al. (2018)
Developmental profile - Not Reported - - Hutson RL , et al. (2018)
Emotion - Not Reported - - Hutson RL , et al. (2018)
Homeostasis - Not Reported - - Hutson RL , et al. (2018)
Immune response - Not Reported - - Hutson RL , et al. (2018)
Learning & memory - Not Reported - - Hutson RL , et al. (2018)
Maternal behavior - Not Reported - - Hutson RL , et al. (2018)
Molecular profile Gene expression Ameliorated Quantitative PCR (qRT-PCR) Third instar larvae Hutson RL , et al. (2018)

F_Fmr1_10_KO_HM_Acamprosate-2

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Acamprosate was made as a 100 mM stock solution and added to the standard cornmeal molasses agar as the final concentrations of 100 microM. Adult animals were placed in vials with acamprosate treated food and allowed to lay eggs. Only first generation, third-instar larvae were selected for the study.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: PMID 29317220
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Hutson RL , et al. (2018)
Motor phenotype General locomotor activity: Ambulatory activity Refractory General observations Third instar larvae Hutson RL , et al. (2018)
Motor phenotype Ataxia: turn angle Restored General observations Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: synaptic bouton number Restored Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Restored Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neuroanatomy / Ultrastructure / Cytoarchitecture Axonal architecture: axonal branch number Restored Immunohistochemistry Third instar larvae Hutson RL , et al. (2018)
Neurophysiology - Not Reported - - Hutson RL , et al. (2018)
Repetitive behavior - Not Reported - - Hutson RL , et al. (2018)
Seizure - Not Reported - - Hutson RL , et al. (2018)
Sensory - Not Reported - - Hutson RL , et al. (2018)
Social behavior - Not Reported - - Hutson RL , et al. (2018)
Communications - Not Reported - - Hutson RL , et al. (2018)
Developmental profile - Not Reported - - Hutson RL , et al. (2018)
Emotion - Not Reported - - Hutson RL , et al. (2018)
Homeostasis - Not Reported - - Hutson RL , et al. (2018)
Immune response - Not Reported - - Hutson RL , et al. (2018)
Learning & memory - Not Reported - - Hutson RL , et al. (2018)
Maternal behavior - Not Reported - - Hutson RL , et al. (2018)
Molecular profile Gene expression Ameliorated Quantitative PCR (qRT-PCR) Third instar larvae Hutson RL , et al. (2018)

F_Fmr1_10_KO_HM_dfmr1^wt

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene with constitutive expression of Fmr1.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: -

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Doll CA and Broadie K (2015)
Motor phenotype - Not Reported - - Doll CA and Broadie K (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture Dendritic architecture: dendritic tree complexity Restored Immunocytochemistry 1 day post eclosion Doll CA and Broadie K (2015)
Neurophysiology - Not Reported - - Doll CA and Broadie K (2015)
Repetitive behavior - Not Reported - - Doll CA and Broadie K (2015)
Seizure - Not Reported - - Doll CA and Broadie K (2015)
Sensory - Not Reported - - Doll CA and Broadie K (2015)
Social behavior - Not Reported - - Doll CA and Broadie K (2015)
Communications - Not Reported - - Doll CA and Broadie K (2015)
Developmental profile - Not Reported - - Doll CA and Broadie K (2015)
Emotion - Not Reported - - Doll CA and Broadie K (2015)
Homeostasis - Not Reported - - Doll CA and Broadie K (2015)
Immune response - Not Reported - - Doll CA and Broadie K (2015)
Learning & memory - Not Reported - - Doll CA and Broadie K (2015)
Maternal behavior - Not Reported - - Doll CA and Broadie K (2015)
Molecular profile - Not Reported - - Doll CA and Broadie K (2015)

F_Fmr1_10_KO_HM_mino-1

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene with 5 uM of minocycline.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Siller SS and Broadie K (2011)
Motor phenotype - Not Reported - - Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: satellite boutons Refractory Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Refractory Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neurophysiology - Not Reported - - Siller SS and Broadie K (2011)
Repetitive behavior - Not Reported - - Siller SS and Broadie K (2011)
Seizure - Not Reported - - Siller SS and Broadie K (2011)
Sensory - Not Reported - - Siller SS and Broadie K (2011)
Social behavior - Not Reported - - Siller SS and Broadie K (2011)
Communications - Not Reported - - Siller SS and Broadie K (2011)
Developmental profile - Not Reported - - Siller SS and Broadie K (2011)
Emotion - Not Reported - - Siller SS and Broadie K (2011)
Homeostasis - Not Reported - - Siller SS and Broadie K (2011)
Immune response - Not Reported - - Siller SS and Broadie K (2011)
Learning & memory - Not Reported - - Siller SS and Broadie K (2011)
Maternal behavior - Not Reported - - Siller SS and Broadie K (2011)
Molecular profile - Not Reported - - Siller SS and Broadie K (2011)

F_Fmr1_10_KO_HM_mino-2

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene with 10 uM of minocycline.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Siller SS and Broadie K (2011)
Motor phenotype - Not Reported - - Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: satellite boutons Ameliorated Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Refractory Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neurophysiology - Not Reported - - Siller SS and Broadie K (2011)
Repetitive behavior - Not Reported - - Siller SS and Broadie K (2011)
Seizure - Not Reported - - Siller SS and Broadie K (2011)
Sensory - Not Reported - - Siller SS and Broadie K (2011)
Social behavior - Not Reported - - Siller SS and Broadie K (2011)
Communications - Not Reported - - Siller SS and Broadie K (2011)
Developmental profile - Not Reported - - Siller SS and Broadie K (2011)
Emotion - Not Reported - - Siller SS and Broadie K (2011)
Homeostasis - Not Reported - - Siller SS and Broadie K (2011)
Immune response - Not Reported - - Siller SS and Broadie K (2011)
Learning & memory - Not Reported - - Siller SS and Broadie K (2011)
Maternal behavior - Not Reported - - Siller SS and Broadie K (2011)
Molecular profile - Not Reported - - Siller SS and Broadie K (2011)

F_Fmr1_10_KO_HM_mino-3

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene with 20 uM of minocycline.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Siller SS and Broadie K (2011)
Motor phenotype - Not Reported - - Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: satellite boutons Restored Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Ameliorated Immunostaining third instar larval stage Siller SS and Broadie K (2011)
Neurophysiology - Not Reported - - Siller SS and Broadie K (2011)
Repetitive behavior - Not Reported - - Siller SS and Broadie K (2011)
Seizure - Not Reported - - Siller SS and Broadie K (2011)
Sensory - Not Reported - - Siller SS and Broadie K (2011)
Social behavior - Not Reported - - Siller SS and Broadie K (2011)
Communications - Not Reported - - Siller SS and Broadie K (2011)
Developmental profile - Not Reported - - Siller SS and Broadie K (2011)
Emotion - Not Reported - - Siller SS and Broadie K (2011)
Homeostasis - Not Reported - - Siller SS and Broadie K (2011)
Immune response - Not Reported - - Siller SS and Broadie K (2011)
Learning & memory - Not Reported - - Siller SS and Broadie K (2011)
Maternal behavior - Not Reported - - Siller SS and Broadie K (2011)
Molecular profile - Not Reported - - Siller SS and Broadie K (2011)

F_Fmr1_10_KO_HM_mino-4

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene with 1 mM of minocycline.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Siller SS and Broadie K (2011)
Motor phenotype - Not Reported - - Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Restored Immunostaining adult stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Axonal architecture: branch number Restored Immunostaining adult stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Restored Immunostaining adult stage Siller SS and Broadie K (2011)
Neurophysiology - Not Reported - - Siller SS and Broadie K (2011)
Repetitive behavior - Not Reported - - Siller SS and Broadie K (2011)
Seizure - Not Reported - - Siller SS and Broadie K (2011)
Sensory - Not Reported - - Siller SS and Broadie K (2011)
Social behavior - Not Reported - - Siller SS and Broadie K (2011)
Communications - Not Reported - - Siller SS and Broadie K (2011)
Developmental profile - Not Reported - - Siller SS and Broadie K (2011)
Emotion - Not Reported - - Siller SS and Broadie K (2011)
Homeostasis - Not Reported - - Siller SS and Broadie K (2011)
Immune response - Not Reported - - Siller SS and Broadie K (2011)
Learning & memory - Not Reported - - Siller SS and Broadie K (2011)
Maternal behavior - Not Reported - - Siller SS and Broadie K (2011)
Molecular profile - Not Reported - - Siller SS and Broadie K (2011)

F_Fmr1_10_KO_HM_mmp1_KO

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene and homozygous knockout of mmp1.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Siller SS and Broadie K (2011)
Motor phenotype - Not Reported - - Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Restored Immunostaining adult stage Siller SS and Broadie K (2011)
Neurophysiology - Not Reported - - Siller SS and Broadie K (2011)
Repetitive behavior - Not Reported - - Siller SS and Broadie K (2011)
Seizure - Not Reported - - Siller SS and Broadie K (2011)
Sensory - Not Reported - - Siller SS and Broadie K (2011)
Social behavior - Not Reported - - Siller SS and Broadie K (2011)
Communications - Not Reported - - Siller SS and Broadie K (2011)
Developmental profile Respiratory system development Side Effect General observations adult stage Siller SS and Broadie K (2011)
Developmental profile Mortality/lethality Side Effect General observations adult stage Siller SS and Broadie K (2011)
Emotion - Not Reported - - Siller SS and Broadie K (2011)
Homeostasis - Not Reported - - Siller SS and Broadie K (2011)
Immune response - Not Reported - - Siller SS and Broadie K (2011)
Learning & memory - Not Reported - - Siller SS and Broadie K (2011)
Maternal behavior - Not Reported - - Siller SS and Broadie K (2011)
Molecular profile - Not Reported - - Siller SS and Broadie K (2011)

F_Fmr1_10_KO_HM_TIMP

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

Homozygous knockout of Fmr1 gene and overexpression of TIMP.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Siller SS and Broadie K (2011)
Motor phenotype - Not Reported - - Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: satellite boutons Restored Immunostaining adult stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Ameliorated Immunostaining adult stage Siller SS and Broadie K (2011)
Neuroanatomy / Ultrastructure / Cytoarchitecture Axonal architecture: branch number Restored Immunostaining adult stage Siller SS and Broadie K (2011)
Neurophysiology - Not Reported - - Siller SS and Broadie K (2011)
Repetitive behavior - Not Reported - - Siller SS and Broadie K (2011)
Seizure - Not Reported - - Siller SS and Broadie K (2011)
Sensory - Not Reported - - Siller SS and Broadie K (2011)
Social behavior - Not Reported - - Siller SS and Broadie K (2011)
Communications - Not Reported - - Siller SS and Broadie K (2011)
Developmental profile Respiratory system development Side Effect General observations adult stage Siller SS and Broadie K (2011)
Developmental profile Mortality/lethality Side Effect General observations adult stage Siller SS and Broadie K (2011)
Emotion - Not Reported - - Siller SS and Broadie K (2011)
Homeostasis - Not Reported - - Siller SS and Broadie K (2011)
Immune response - Not Reported - - Siller SS and Broadie K (2011)
Learning & memory - Not Reported - - Siller SS and Broadie K (2011)
Maternal behavior - Not Reported - - Siller SS and Broadie K (2011)
Molecular profile - Not Reported - - Siller SS and Broadie K (2011)

F_Fmr1_11_KO_HT

Model Type: Genetic
Model Genotype: Heterozygous

Mutation:

Heterozygous knockout of Fmr1 gene.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Circadian sleep/wake cycle - Not Reported - - Doll CA and Broadie K (2015)
Motor phenotype General locomotor activity: Larval locomotor behavior Increased General observations third instar larvae Kashima R , et al. (2017)
Motor phenotype - Not Reported - - Doll CA and Broadie K (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Increased Immunostaining third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Dendritic architecture: dendritic tree complexity No Change Immunocytochemistry 1 day post eclosion Doll CA and Broadie K (2015)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Doll CA and Broadie K (2015)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Doll CA and Broadie K (2015)
Seizure - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Doll CA and Broadie K (2015)
Sensory - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Doll CA and Broadie K (2015)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Doll CA and Broadie K (2015)
Communications - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Doll CA and Broadie K (2015)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Doll CA and Broadie K (2015)
Emotion - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Doll CA and Broadie K (2015)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Doll CA and Broadie K (2015)
Immune response - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Doll CA and Broadie K (2015)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Doll CA and Broadie K (2015)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Doll CA and Broadie K (2015)
Molecular profile - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Doll CA and Broadie K (2015)

F_Fmr1_12_KD

Model Type: Genetic
Model Genotype: Wild type

Mutation:

Fmr1 knockdown.

Allele Type: Knockdown
Strain of Origin: -
Genetic Background: -

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Circadian sleep/wake cycle - Not Reported - - Doll CA and Broadie K (2015)
Motor phenotype General locomotor activity: Larval locomotor behavior Increased General observations third instar larvae Kashima R , et al. (2017)
Motor phenotype - Not Reported - - Doll CA and Broadie K (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Increased Immunostaining third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Dendritic architecture: dendritic tree complexity Increased Immunocytochemistry 1 day post eclosion Doll CA and Broadie K (2015)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Doll CA and Broadie K (2015)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Doll CA and Broadie K (2015)
Seizure - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Doll CA and Broadie K (2015)
Sensory - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Doll CA and Broadie K (2015)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Doll CA and Broadie K (2015)
Communications - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Doll CA and Broadie K (2015)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Doll CA and Broadie K (2015)
Emotion - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Doll CA and Broadie K (2015)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Doll CA and Broadie K (2015)
Immune response - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Doll CA and Broadie K (2015)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Doll CA and Broadie K (2015)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Doll CA and Broadie K (2015)
Molecular profile - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Doll CA and Broadie K (2015)

F_Fmr1_12_KD_LIMKi

Model Type: RESCUE-Pharmaceutical
Model Genotype: Wild type

Mutation:

LIMK inhibitor rescue of Fmr1 RNAi-mediated knockdown.

Allele Type: Knockdown
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Restored General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Restored Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_13_KO_HM

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Bloomington Drosophila Stock Center
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Dong T , et al. (2016)
Motor phenotype - Not Reported - - Dong T , et al. (2016)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Dong T , et al. (2016)
Neurophysiology - Not Reported - - Dong T , et al. (2016)
Repetitive behavior - Not Reported - - Dong T , et al. (2016)
Seizure - Not Reported - - Dong T , et al. (2016)
Sensory - Not Reported - - Dong T , et al. (2016)
Social behavior - Not Reported - - Dong T , et al. (2016)
Communications - Not Reported - - Dong T , et al. (2016)
Developmental profile - Not Reported - - Dong T , et al. (2016)
Emotion - Not Reported - - Dong T , et al. (2016)
Homeostasis - Not Reported - - Dong T , et al. (2016)
Immune response - Not Reported - - Dong T , et al. (2016)
Learning & memory Olfactory learning and memory No Change Olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Memory of cue: Long term recall Increased Olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Memory of cue: Long term recall Decreased Olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Extinction Increased Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Extinction Increased Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cognitive flexibility: Associative learning Decreased Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Maternal behavior - Not Reported - - Dong T , et al. (2016)
Molecular profile Targeted expression Decreased Western blot Adult stage Dong T , et al. (2016)
Molecular profile Gene expression Decreased Western blot Adult stage Dong T , et al. (2016)

F_Fmr1_13_KO_HM_Rac1-expression

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Ronald L. Davis (The Scripps Research Institute, Jupiter, FL)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Dong T , et al. (2016)
Motor phenotype - Not Reported - - Dong T , et al. (2016)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Dong T , et al. (2016)
Neurophysiology - Not Reported - - Dong T , et al. (2016)
Repetitive behavior - Not Reported - - Dong T , et al. (2016)
Seizure - Not Reported - - Dong T , et al. (2016)
Sensory - Not Reported - - Dong T , et al. (2016)
Social behavior - Not Reported - - Dong T , et al. (2016)
Communications - Not Reported - - Dong T , et al. (2016)
Developmental profile - Not Reported - - Dong T , et al. (2016)
Emotion - Not Reported - - Dong T , et al. (2016)
Homeostasis - Not Reported - - Dong T , et al. (2016)
Immune response - Not Reported - - Dong T , et al. (2016)
Learning & memory Cognitive flexibility: Associative learning Restored Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Maternal behavior - Not Reported - - Dong T , et al. (2016)
Molecular profile - Not Reported - - Dong T , et al. (2016)

F_Fmr1_13_KO_HM_Rac1-inhibition

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Ronald L. Davis (The Scripps Research Institute, Jupiter, FL)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Dong T , et al. (2016)
Motor phenotype - Not Reported - - Dong T , et al. (2016)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Dong T , et al. (2016)
Neurophysiology - Not Reported - - Dong T , et al. (2016)
Repetitive behavior - Not Reported - - Dong T , et al. (2016)
Seizure - Not Reported - - Dong T , et al. (2016)
Sensory - Not Reported - - Dong T , et al. (2016)
Social behavior - Not Reported - - Dong T , et al. (2016)
Communications - Not Reported - - Dong T , et al. (2016)
Developmental profile - Not Reported - - Dong T , et al. (2016)
Emotion - Not Reported - - Dong T , et al. (2016)
Homeostasis - Not Reported - - Dong T , et al. (2016)
Immune response - Not Reported - - Dong T , et al. (2016)
Learning & memory Cognitive flexibility: Associative learning No Adverse Effect Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Maternal behavior - Not Reported - - Dong T , et al. (2016)
Molecular profile - Not Reported - - Dong T , et al. (2016)

F_Fmr1_14_KO_HM

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Bloomington Drosophila Stock Center
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Dong T , et al. (2016)
Motor phenotype - Not Reported - - Dong T , et al. (2016)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Dong T , et al. (2016)
Neurophysiology - Not Reported - - Dong T , et al. (2016)
Repetitive behavior - Not Reported - - Dong T , et al. (2016)
Seizure - Not Reported - - Dong T , et al. (2016)
Sensory - Not Reported - - Dong T , et al. (2016)
Social behavior - Not Reported - - Dong T , et al. (2016)
Communications - Not Reported - - Dong T , et al. (2016)
Developmental profile - Not Reported - - Dong T , et al. (2016)
Emotion - Not Reported - - Dong T , et al. (2016)
Homeostasis - Not Reported - - Dong T , et al. (2016)
Immune response - Not Reported - - Dong T , et al. (2016)
Learning & memory Olfactory learning and memory Decreased Olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Memory of cue: Long term recall Increased Olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Extinction Increased Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Extinction Increased Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cognitive flexibility: Associative learning Decreased Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Maternal behavior - Not Reported - - Dong T , et al. (2016)
Molecular profile Targeted expression Decreased Western blot Adult stage Dong T , et al. (2016)

F_Fmr1_15_CKD_HM_Mushroombody

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

.

Allele Type: Loss-of-function
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Ronald L. Davis (The Scripps Research Institute, Jupiter, FL)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Dong T , et al. (2016)
Motor phenotype - Not Reported - - Dong T , et al. (2016)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Dong T , et al. (2016)
Neurophysiology - Not Reported - - Dong T , et al. (2016)
Repetitive behavior - Not Reported - - Dong T , et al. (2016)
Seizure - Not Reported - - Dong T , et al. (2016)
Sensory - Not Reported - - Dong T , et al. (2016)
Social behavior - Not Reported - - Dong T , et al. (2016)
Communications - Not Reported - - Dong T , et al. (2016)
Developmental profile - Not Reported - - Dong T , et al. (2016)
Emotion - Not Reported - - Dong T , et al. (2016)
Homeostasis - Not Reported - - Dong T , et al. (2016)
Immune response - Not Reported - - Dong T , et al. (2016)
Learning & memory Olfactory learning and memory No Change Olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cued or contextual fear conditioning: Memory of cue: Long term recall Increased Olfactory classical conditioning Adult stage Dong T , et al. (2016)
Learning & memory Cognitive flexibility: Associative learning Decreased Reversal learning of olfactory classical conditioning Adult stage Dong T , et al. (2016)
Maternal behavior - Not Reported - - Dong T , et al. (2016)
Molecular profile - Not Reported - - Dong T , et al. (2016)

F_Fmr1_16_KO_HT

Model Type: Genetic
Model Genotype: Heterozygous

Mutation:

Heterozygous null dfmr1 mutant dfmr1^3.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Increased General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Increased Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_17_KO_HM

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

Homozygous null dfmr1 mutant dfmr1^113.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Increased General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Increased Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile Protein phosphorylation Increased Western blot third instar larvae Kashima R , et al. (2017)

F_Fmr1_17_KO_HM_LIMKi

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

LIMK inhibitor rescue of homozygous null dfmr1^113 mutant.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Restored General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Ameliorated Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile Protein phosphorylation Ameliorated Western blot third instar larvae Kashima R , et al. (2017)

F_Fmr1_17_KO_HM_puromycin

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Puromycin rescue of homozygous null dfmr1^113 mutant.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Ameliorated General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Ameliorated Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_17_KO_HM_TH251

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

TH251 rescue of homozygous null dfmr1^113 mutant.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Ameliorated General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Ameliorated Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_17_KO_HM_TH255

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

TH255 rescue of homozygous null dfmr1^113 mutant.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Ameliorated General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Ameliorated Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_17_KO_HM_TH263

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

TH263 rescue of homozygous null dfmr1^113 mutant.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Refractory General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Refractory Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_17_KO_HM_wit_KO_HT

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

Heterozygous wit mutant rescue of homozygous dfmr1^113 null mutant.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Restored General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Ameliorated Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_18_KO_HM

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

Null dfmr1 mutant with two mutant alleles, dfmr1^3 and dfmr1^113.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Increased General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Increased Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_18_KO_HM_LIMKi

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

LIMK inhibitor rescue of null mutant dfmr1^3/dfmr1^113.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: -
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Kashima R , et al. (2017)
Motor phenotype General locomotor activity: Larval locomotor behavior Ameliorated General observations third instar larvae Kashima R , et al. (2017)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density: mature boutons Restored Immunostaining third instar larvae Kashima R , et al. (2017)
Neurophysiology - Not Reported - - Kashima R , et al. (2017)
Repetitive behavior - Not Reported - - Kashima R , et al. (2017)
Seizure - Not Reported - - Kashima R , et al. (2017)
Sensory - Not Reported - - Kashima R , et al. (2017)
Social behavior - Not Reported - - Kashima R , et al. (2017)
Communications - Not Reported - - Kashima R , et al. (2017)
Developmental profile - Not Reported - - Kashima R , et al. (2017)
Emotion - Not Reported - - Kashima R , et al. (2017)
Homeostasis - Not Reported - - Kashima R , et al. (2017)
Immune response - Not Reported - - Kashima R , et al. (2017)
Learning & memory - Not Reported - - Kashima R , et al. (2017)
Maternal behavior - Not Reported - - Kashima R , et al. (2017)
Molecular profile - Not Reported - - Kashima R , et al. (2017)

F_Fmr1_1_KO_HM

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3, compared to control wildtype strain w^1118. Generated by imprecise excision of P element (P[EP]3517) in 5' UTR of Fmr1 locus.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle Circadian rhythm Decreased General observations 2-day old adults Banerjee P , et al. (2010)
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Circadian sleep/wake cycle Circadian rhythms: timing/phases of locomotor activity Decreased Beam crossing adult stage Dockendorff TC , et al. (2002)
Circadian sleep/wake cycle Circadian rhythm: Protein expression periodicity No Change Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Circadian sleep/wake cycle Circadian rhythm: Eclosion rhythm Abnormal Eclosion profile analysis pharate adult stage Dockendorff TC , et al. (2002)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Increased Immunostaining third instar larvae Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Decreased Immunostaining 2-day old adults Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture Neuronal number in the brain No Change Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture Neuronal number in the brain No Change Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture Brain morphology No Change Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Increased Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Increased Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Increased Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - Dockendorff TC , et al. (2002)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - Dockendorff TC , et al. (2002)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - Dockendorff TC , et al. (2002)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - McBride SM , et al. (2005)
Sensory Vision: phototaxis No Change Y-maze test adult stage Dockendorff TC , et al. (2002)
Sensory Olfaction No Change Olfactory trap adult stage Dockendorff TC , et al. (2002)
Social behavior Social memory: short-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Mating behavior: Male courtship behavior Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social habituation No Change General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: wing extension Decreased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior: wing extension Decreased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior: proboscis-mediated licking Decreased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior: prior orientation Increased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior: prior orientation Increased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior No Change Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior Decreased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior Decreased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Social behavior Mating behavior: Male courtship behavior Decreased Mating behavior test adult stage Dockendorff TC , et al. (2002)
Communications - Not Reported - - Banerjee P , et al. (2010)
Communications - Not Reported - - McBride SM , et al. (2005)
Communications - Not Reported - - Dockendorff TC , et al. (2002)
Developmental profile Mortality/lethality: embryonic Decreased General observations Unreported Banerjee P , et al. (2010)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Developmental profile Mortality/lethality No Change General observations adult stage Dockendorff TC , et al. (2002)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - Dockendorff TC , et al. (2002)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - Dockendorff TC , et al. (2002)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - Dockendorff TC , et al. (2002)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - Dockendorff TC , et al. (2002)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - Dockendorff TC , et al. (2002)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - McBride SM , et al. (2005)
Molecular profile Regulation of translation No Change Immunohistochemistry adult stage Dockendorff TC , et al. (2002)
Molecular profile Regulation of gene expression No Change RNase protection assay adult stage Dockendorff TC , et al. (2002)

F_Fmr1_1_KO_HM_futsch^N94

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 with futsch^N94.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle Circadian rhythms: timing/phases of locomotor activity No Adverse Effect Beam crossing adult stage Dockendorff TC , et al. (2002)
Motor phenotype - Not Reported - - Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Dockendorff TC , et al. (2002)
Neurophysiology - Not Reported - - Dockendorff TC , et al. (2002)
Repetitive behavior - Not Reported - - Dockendorff TC , et al. (2002)
Seizure - Not Reported - - Dockendorff TC , et al. (2002)
Sensory - Not Reported - - Dockendorff TC , et al. (2002)
Social behavior - Not Reported - - Dockendorff TC , et al. (2002)
Communications - Not Reported - - Dockendorff TC , et al. (2002)
Developmental profile - Not Reported - - Dockendorff TC , et al. (2002)
Emotion - Not Reported - - Dockendorff TC , et al. (2002)
Homeostasis - Not Reported - - Dockendorff TC , et al. (2002)
Immune response - Not Reported - - Dockendorff TC , et al. (2002)
Learning & memory - Not Reported - - Dockendorff TC , et al. (2002)
Maternal behavior - Not Reported - - Dockendorff TC , et al. (2002)
Molecular profile - Not Reported - - Dockendorff TC , et al. (2002)

F_Fmr1_2_KO_HM

Model Type: Genetic
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Choi CH , et al. (2015)
Circadian sleep/wake cycle - Not Reported - - Banerjee P , et al. (2010)
Circadian sleep/wake cycle Circadian rhythms: timing/phases of locomotor activity Decreased Beam crossing adult stage McBride SM , et al. (2005)
Circadian sleep/wake cycle Circadian rhythms: timing/phases of locomotor activity Decreased Beam crossing adult stage Dockendorff TC , et al. (2002)
Circadian sleep/wake cycle Circadian rhythm: Eclosion rhythm Abnormal Eclosion profile analysis pharate adult stage Dockendorff TC , et al. (2002)
Motor phenotype - Not Reported - - Choi CH , et al. (2015)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype General locomotor activity No Change Beam crossing adult stage McBride SM , et al. (2005)
Motor phenotype - Not Reported - - Dockendorff TC , et al. (2002)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Abnormal Immunostaining Unreported Choi CH , et al. (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Brain morphology Abnormal Immunohistochemistry adult stage McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Dockendorff TC , et al. (2002)
Neurophysiology - Not Reported - - Choi CH , et al. (2015)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - Dockendorff TC , et al. (2002)
Repetitive behavior - Not Reported - - Choi CH , et al. (2015)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - Dockendorff TC , et al. (2002)
Seizure - Not Reported - - Choi CH , et al. (2015)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - Dockendorff TC , et al. (2002)
Sensory - Not Reported - - Choi CH , et al. (2015)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Sensory Vision No Change Y-maze test adult stage McBride SM , et al. (2005)
Sensory Olfaction No Change Olfactory trap adult stage McBride SM , et al. (2005)
Sensory - Not Reported - - Dockendorff TC , et al. (2002)
Social behavior Social memory: short-term social memory Decreased Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Social behavior Social memory Decreased Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Social behavior Social memory: short-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory: long-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Mating behavior: Male courtship behavior Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory Decreased General observations adult stage McBride SM , et al. (2005)
Social behavior Social memory Decreased General observations adult stage McBride SM , et al. (2005)
Social behavior Social memory Decreased General observations adult stage McBride SM , et al. (2005)
Social behavior Social memory Decreased General observations adult stage McBride SM , et al. (2005)
Social behavior Social habituation No Change General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: wing extension Decreased General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: proboscis-mediated licking Decreased General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: prior orientation No Change General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Decreased General observations adult stage McBride SM , et al. (2005)
Social behavior - Not Reported - - Dockendorff TC , et al. (2002)
Communications - Not Reported - - Choi CH , et al. (2015)
Communications - Not Reported - - Banerjee P , et al. (2010)
Communications - Not Reported - - McBride SM , et al. (2005)
Communications - Not Reported - - Dockendorff TC , et al. (2002)
Developmental profile - Not Reported - - Choi CH , et al. (2015)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - Dockendorff TC , et al. (2002)
Emotion Fear response No Change Fear conditioning test: olfactory cue 1 to 3 days old Choi CH , et al. (2015)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - Dockendorff TC , et al. (2002)
Homeostasis - Not Reported - - Choi CH , et al. (2015)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - Dockendorff TC , et al. (2002)
Immune response - Not Reported - - Choi CH , et al. (2015)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - Dockendorff TC , et al. (2002)
Learning & memory Olfactory learning and memory No Change Fear conditioning test: olfactory cue 1 to 3 days old Choi CH , et al. (2015)
Learning & memory Olfactory learning and memory Decreased Fear conditioning test: olfactory cue 3- to 5 days old Choi CH , et al. (2015)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - Dockendorff TC , et al. (2002)
Maternal behavior - Not Reported - - Choi CH , et al. (2015)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - Dockendorff TC , et al. (2002)
Molecular profile Signaling Decreased ELISA Unreported Choi CH , et al. (2015)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - Dockendorff TC , et al. (2002)

F_Fmr1_2_KO_HM_dnc_KO

Model Type: RESCUE-Genetic
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). With dunce mutation, dnc/Y.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Choi CH , et al. (2015)
Motor phenotype - Not Reported - - Choi CH , et al. (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Choi CH , et al. (2015)
Neurophysiology - Not Reported - - Choi CH , et al. (2015)
Repetitive behavior - Not Reported - - Choi CH , et al. (2015)
Seizure - Not Reported - - Choi CH , et al. (2015)
Sensory - Not Reported - - Choi CH , et al. (2015)
Social behavior Social memory: short-term social memory Side Effect Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Communications - Not Reported - - Choi CH , et al. (2015)
Developmental profile - Not Reported - - Choi CH , et al. (2015)
Emotion - Not Reported - - Choi CH , et al. (2015)
Homeostasis - Not Reported - - Choi CH , et al. (2015)
Immune response - Not Reported - - Choi CH , et al. (2015)
Learning & memory - Not Reported - - Choi CH , et al. (2015)
Maternal behavior - Not Reported - - Choi CH , et al. (2015)
Molecular profile - Not Reported - - Choi CH , et al. (2015)

F_Fmr1_2_KO_HM_Hi-LiCl-A

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 50 mM LiCl after eclosion (adult stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior Social memory Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Side Effect General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_Hi-MPEP-A

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 86 microM MPEP after eclosion (adult stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype General locomotor activity No Adverse Effect Beam crossing adult stage McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture Brain morphology Refractory Immunohistochemistry adult stage McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory Vision No Adverse Effect Y-maze test adult stage McBride SM , et al. (2005)
Sensory Olfaction No Adverse Effect Olfactory trap adult stage McBride SM , et al. (2005)
Social behavior Social memory Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Social memory Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Social habituation No Adverse Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: wing extension Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: proboscis-mediated licking Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: prior orientation No Adverse Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Side Effect General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_Hi-MPEP-L

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 86 microM MPEP during development (larval stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype General locomotor activity No Adverse Effect Beam crossing adult stage McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory Vision No Adverse Effect Y-maze test adult stage McBride SM , et al. (2005)
Sensory Olfaction No Adverse Effect Olfactory trap adult stage McBride SM , et al. (2005)
Social behavior Social memory Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Social memory Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Social habituation Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: wing extension Ameliorated General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: proboscis-mediated licking Ameliorated General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: prior orientation No Adverse Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Restored General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_Hi-MPEP-LA

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 86 microM MPEP during development and after eclosion (larval and adult stages).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle Circadian rhythms: timing/phases of locomotor activity Refractory Beam crossing adult stage McBride SM , et al. (2005)
Motor phenotype General locomotor activity No Adverse Effect Beam crossing adult stage McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory Vision No Adverse Effect Y-maze test adult stage McBride SM , et al. (2005)
Sensory Olfaction No Adverse Effect Olfactory trap adult stage McBride SM , et al. (2005)
Social behavior Social memory Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Social memory Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Social habituation Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: wing extension Ameliorated General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: proboscis-mediated licking Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior: prior orientation No Adverse Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Restored General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_Lo-LiCl-A

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 5 mM LiCl after eclosion (adult stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior Social memory Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Side Effect General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_Lo-MPEP-A

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 8.6 microM MPEP after eclosion (adult stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior Social memory Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior Side Effect General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_Lo-MPEP-L

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 8.6 microM MPEP during development (larval stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture Brain morphology Ameliorated Immunohistochemistry adult stage McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior - Not Reported - - McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_LY341495-A

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 400 nM LY341495 after eclosion (adult stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior Social memory Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Social memory Side Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Social habituation No Adverse Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Side Effect General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_LY341495-L

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 400 nM LY341495 during development (larval stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture Brain morphology Restored Immunohistochemistry adult stage McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior - Not Reported - - McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_MPPG-A

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 573 microM MPPG after eclosion (adult stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior Social memory Restored General observations adult stage McBride SM , et al. (2005)
Social behavior Social habituation No Adverse Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Side Effect General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_MTPG-A

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 348 microM MTPG after eclosion (adult stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior Social habituation No Adverse Effect General observations adult stage McBride SM , et al. (2005)
Social behavior Mating behavior: Male courtship behavior Side Effect General observations adult stage McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_MTPG-L

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Rescue with 348 microM MTPG during development (larval stage).

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - McBride SM , et al. (2005)
Motor phenotype - Not Reported - - McBride SM , et al. (2005)
Neuroanatomy / Ultrastructure / Cytoarchitecture Brain morphology Ameliorated Immunohistochemistry adult stage McBride SM , et al. (2005)
Neurophysiology - Not Reported - - McBride SM , et al. (2005)
Repetitive behavior - Not Reported - - McBride SM , et al. (2005)
Seizure - Not Reported - - McBride SM , et al. (2005)
Sensory - Not Reported - - McBride SM , et al. (2005)
Social behavior - Not Reported - - McBride SM , et al. (2005)
Communications - Not Reported - - McBride SM , et al. (2005)
Developmental profile - Not Reported - - McBride SM , et al. (2005)
Emotion - Not Reported - - McBride SM , et al. (2005)
Homeostasis - Not Reported - - McBride SM , et al. (2005)
Immune response - Not Reported - - McBride SM , et al. (2005)
Learning & memory - Not Reported - - McBride SM , et al. (2005)
Maternal behavior - Not Reported - - McBride SM , et al. (2005)
Molecular profile - Not Reported - - McBride SM , et al. (2005)

F_Fmr1_2_KO_HM_ro-20

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Treatment with Ro-20-1724 20 uM.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Choi CH , et al. (2015)
Motor phenotype - Not Reported - - Choi CH , et al. (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Choi CH , et al. (2015)
Neurophysiology - Not Reported - - Choi CH , et al. (2015)
Repetitive behavior - Not Reported - - Choi CH , et al. (2015)
Seizure - Not Reported - - Choi CH , et al. (2015)
Sensory - Not Reported - - Choi CH , et al. (2015)
Social behavior Social memory: short-term social memory Restored Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Social behavior Social memory Restored Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Communications - Not Reported - - Choi CH , et al. (2015)
Developmental profile - Not Reported - - Choi CH , et al. (2015)
Emotion - Not Reported - - Choi CH , et al. (2015)
Homeostasis - Not Reported - - Choi CH , et al. (2015)
Immune response - Not Reported - - Choi CH , et al. (2015)
Learning & memory - Not Reported - - Choi CH , et al. (2015)
Maternal behavior - Not Reported - - Choi CH , et al. (2015)
Molecular profile - Not Reported - - Choi CH , et al. (2015)

F_Fmr1_2_KO_HM_rolipram-1

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Treatment with rolipram 50 uM.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Choi CH , et al. (2015)
Motor phenotype - Not Reported - - Choi CH , et al. (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Refractory Immunostaining Unreported Choi CH , et al. (2015)
Neurophysiology - Not Reported - - Choi CH , et al. (2015)
Repetitive behavior - Not Reported - - Choi CH , et al. (2015)
Seizure - Not Reported - - Choi CH , et al. (2015)
Sensory - Not Reported - - Choi CH , et al. (2015)
Social behavior Social memory: short-term social memory Restored Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Social behavior Social memory Restored Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Communications - Not Reported - - Choi CH , et al. (2015)
Developmental profile - Not Reported - - Choi CH , et al. (2015)
Emotion Fear response No Adverse Effect Fear conditioning test: olfactory cue 1 to 3 days old Choi CH , et al. (2015)
Homeostasis - Not Reported - - Choi CH , et al. (2015)
Immune response - Not Reported - - Choi CH , et al. (2015)
Learning & memory Olfactory learning and memory No Adverse Effect Fear conditioning test: olfactory cue 1 to 3 days old Choi CH , et al. (2015)
Learning & memory Olfactory learning and memory Ameliorated Fear conditioning test: olfactory cue 3- to 5 days old Choi CH , et al. (2015)
Maternal behavior - Not Reported - - Choi CH , et al. (2015)
Molecular profile Signaling Restored ELISA Unreported Choi CH , et al. (2015)

F_Fmr1_2_KO_HM_rolipram-2

Model Type: RESCUE-Pharmaceutical
Model Genotype: Homozygous

Mutation:

Homozygous dfmr1^3 plus one transgenic copy of a genomic fragment containing a frameshift mutation in the dfmr1 open reading frame (dfmr1^FS), compared to control homozygous dfmr1^3 plus one transgenic copy of a wild-type genomic dfmr1 fragment (dfmr1^wt). Treatment with rolipram 500 uM.

Allele Type: Knockout
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Choi CH , et al. (2015)
Motor phenotype - Not Reported - - Choi CH , et al. (2015)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity Ameliorated Immunostaining Unreported Choi CH , et al. (2015)
Neurophysiology - Not Reported - - Choi CH , et al. (2015)
Repetitive behavior - Not Reported - - Choi CH , et al. (2015)
Seizure - Not Reported - - Choi CH , et al. (2015)
Sensory - Not Reported - - Choi CH , et al. (2015)
Social behavior Social memory: short-term social memory Restored Courtship assay 9 days post eclosion Choi CH , et al. (2015)
Communications - Not Reported - - Choi CH , et al. (2015)
Developmental profile - Not Reported - - Choi CH , et al. (2015)
Emotion - Not Reported - - Choi CH , et al. (2015)
Homeostasis - Not Reported - - Choi CH , et al. (2015)
Immune response - Not Reported - - Choi CH , et al. (2015)
Learning & memory - Not Reported - - Choi CH , et al. (2015)
Maternal behavior - Not Reported - - Choi CH , et al. (2015)
Molecular profile - Not Reported - - Choi CH , et al. (2015)

F_Fmr1_3_TG_KI_Fmr1^dQN

Model Type: Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of short isoform of Fmr1 lacking Q/N domain (line 8) in a null background.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle Circadian rhythm Decreased General observations 2-day old adults Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Increased Immunostaining third instar larvae Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity No Change Immunostaining 2-day old adults Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior Social memory: short-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Mating behavior: Male courtship behavior Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile Mortality/lethality: embryonic No Change General observations Unreported Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile Targeted expression No Change Western blot Adult stage Banerjee P , et al. (2010)

F_Fmr1_3_TG_KI_Fmr1^dQN_Fmr1^wt

Model Type: RESCUE-Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of short isoform of Fmr1 lacking Q/N domain (line 8) in a null background, co-expressed with a transgene of wildtype genomic Fmr1.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Restored Immunostaining third instar larvae Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior - Not Reported - - Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)

F_Fmr1_4_TG_KI_Fmr1^dQN

Model Type: Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of short isoform of Fmr1 lacking Q/N domain (line 1B) in a null background.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle Circadian rhythm Decreased General observations 2-day old adults Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Increased Immunostaining third instar larvae Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity No Change Immunostaining 2-day old adults Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior Social memory: short-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Mating behavior: Male courtship behavior Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile Mortality/lethality: embryonic No Change General observations Unreported Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile Targeted expression Increased Western blot Adult stage Banerjee P , et al. (2010)

F_Fmr1_4_TG_KI_Fmr1^dQN_Fmr1^wt

Model Type: RESCUE-Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of short isoform of Fmr1 lacking Q/N domain (line 1B) in a null background, co-expressed with a transgene of wildtype genomic Fmr1.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density Restored Immunostaining third instar larvae Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior - Not Reported - - Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)

F_Fmr1_5_TG_KI_Fmr1^L

Model Type: Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of long isoform of Fmr1 containing Q/N domain (line C51L) in a null background.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior Social memory: short-term social memory No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory: long-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Mating behavior: Male courtship behavior No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)

F_Fmr1_6_TG_KI_Fmr1^L

Model Type: Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of long isoform of Fmr1 containing Q/N domain (line L1) in a null background.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle Circadian rhythm No Change Immunostaining third instar larvae Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Synapse density No Change Immunostaining third instar larvae Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture Anatomical projections and connectivity No Change Immunostaining third instar larvae Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior Social memory: short-term social memory No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory: long-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Mating behavior: Male courtship behavior No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)

F_Fmr1_7_TG_KI_Fmr1^L

Model Type: Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of long isoform of Fmr1 containing Q/N domain (line L32) in a null background.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior Social memory: short-term social memory No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Social memory: long-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Social behavior Mating behavior: Male courtship behavior No Change Courtship assay Adult stage Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)

F_Fmr1_8_TG_KI_Fmr1^L

Model Type: Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of long isoforms of Fmr1 containing Q/N domain (C51L x L1) in a null background.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior Social memory: long-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)

F_Fmr1_9_TG_KI_Fmr1^L

Model Type: Genetic
Model Genotype: Transgenic

Mutation:

Transgenic expression of long isoforms of Fmr1 containing Q/N domain (C51L x L32) in a null background.

Allele Type: LOF Knockin
Strain of Origin: -
Genetic Background: w^1118

Es cell line: -
Model Source: Jongens laboratory, University of Pennsylvania (PMID 12086644)
Phenotypic Profile
Category Entity Value Change Experimental Paradigm Age at Testing Author, Year
Circadian sleep/wake cycle - Not Reported - - Banerjee P , et al. (2010)
Motor phenotype - Not Reported - - Banerjee P , et al. (2010)
Neuroanatomy / Ultrastructure / Cytoarchitecture - Not Reported - - Banerjee P , et al. (2010)
Neurophysiology - Not Reported - - Banerjee P , et al. (2010)
Repetitive behavior - Not Reported - - Banerjee P , et al. (2010)
Seizure - Not Reported - - Banerjee P , et al. (2010)
Sensory - Not Reported - - Banerjee P , et al. (2010)
Social behavior Social memory: long-term social memory Decreased Courtship assay Adult stage Banerjee P , et al. (2010)
Communications - Not Reported - - Banerjee P , et al. (2010)
Developmental profile - Not Reported - - Banerjee P , et al. (2010)
Emotion - Not Reported - - Banerjee P , et al. (2010)
Homeostasis - Not Reported - - Banerjee P , et al. (2010)
Immune response - Not Reported - - Banerjee P , et al. (2010)
Learning & memory - Not Reported - - Banerjee P , et al. (2010)
Maternal behavior - Not Reported - - Banerjee P , et al. (2010)
Molecular profile - Not Reported - - Banerjee P , et al. (2010)
Other FMR1 Rescue Models(26 Models)
F_Fmr1_1_KO_HM 1 Genetic Genetic futsch mutation
F_Fmr1_2_KO_HM 1 Genetic Genetic dunce mutation
F_Fmr1_2_KO_HM 2 Genetic Pharmaceutical Lithium
F_Fmr1_2_KO_HM 5 Genetic Pharmaceutical MPEP
F_Fmr1_2_KO_HM 2 Genetic Pharmaceutical LY-341,495
F_Fmr1_2_KO_HM 1 Genetic Pharmaceutical MPPG
F_Fmr1_2_KO_HM 2 Genetic Pharmaceutical MTPG
F_Fmr1_2_KO_HM 1 Genetic Pharmaceutical Ro-20-1724
F_Fmr1_2_KO_HM 2 Genetic Pharmaceutical Rolipram
F_Fmr1_3_TG_KI_Fmr1^dQN 1 Genetic Genetic dfmr1^wt transgene
F_Fmr1_4_TG_KI_Fmr1^dQN 1 Genetic Genetic dfmr1^wt transgene
F_Fmr1_10_KO_HM 2 Genetic Pharmaceutical Acamprosate
F_Fmr1_10_KO_HM 1 Genetic Genetic dfmr1
F_Fmr1_10_KO_HM 4 Genetic Pharmaceutical Minocycline
F_Fmr1_10_KO_HM 1 Genetic Genetic mmp1^Q112
F_Fmr1_10_KO_HM 1 Genetic Genetic Timp
F_Fmr1_12_KD 1 Genetic Pharmaceutical LIM domain kinase 1
F_Fmr1_13_KO_HM 1 Genetic Genetic Drac1(V12)
F_Fmr1_13_KO_HM 1 Genetic Genetic Drac1(N17)
F_Fmr1_17_KO_HM 1 Genetic Pharmaceutical LIM domain kinase 1
F_Fmr1_17_KO_HM 1 Genetic Pharmaceutical Puromycin
F_Fmr1_17_KO_HM 1 Genetic Pharmaceutical TH251
F_Fmr1_17_KO_HM 1 Genetic Pharmaceutical TH255
F_Fmr1_17_KO_HM 1 Genetic Pharmaceutical TH263
F_Fmr1_17_KO_HM 1 Genetic Genetic wit mutation
F_Fmr1_18_KO_HM 1 Genetic Pharmaceutical LIM domain kinase 1
CNVs associated with FMR1(1 Models)
Xq27.3 12 Deletion-Duplication 20  /  113
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