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Down syndrome mouse models Ts65Dn, Ts1Cje, and Ms1Cje/Ts65Dn exhibit variable severity of cerebellar phenotypes.
Olson, L E; Roper, R J; Baxter, L L; Carlson, E J; Epstein, C J; Reeves, R H.
Affiliation
  • Olson LE; Department of Physiology, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.
Dev Dyn ; 230(3): 581-9, 2004 Jul.
Article in En | MEDLINE | ID: mdl-15188443
Two mouse models are widely used for Down syndrome (DS) research. The Ts65Dn mouse carries a small chromosome derived primarily from mouse chromosome 16, causing dosage imbalance for approximately half of human chromosome 21 orthologs. These mice have cerebellar pathology with direct parallels to DS. The Ts1Cje mouse, containing a translocated chromosome 16, is at dosage imbalance for 67% of the genes triplicated in Ts65Dn. We quantified cerebellar volume and granule cell and Purkinje cell density in Ts1Cje. Cerebellar volume was significantly affected to the same degree in Ts1Cje and Ts65Dn, despite that Ts1Cje has fewer triplicated genes. However, dosage imbalance in Ts1Cje had little effect on granule cell and Purkinje cell density. Several mice with dosage imbalance for the segment of the Ts65Dn chromosome not triplicated in Ts1Cje had phenotypes that contrasted with those in Ts1Cje. These observations do not readily differentiate between two prevalent hypotheses for gene action in DS.
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Collection: 01-internacional Database: MEDLINE Main subject: Phenotype / Cerebellum / Down Syndrome / Disease Models, Animal Type of study: Diagnostic_studies / Prognostic_studies Limits: Animals / Female / Humans / Male Language: En Journal: Dev Dyn Journal subject: ANATOMIA Year: 2004 Document type: Article Affiliation country: United States Country of publication: United States
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Collection: 01-internacional Database: MEDLINE Main subject: Phenotype / Cerebellum / Down Syndrome / Disease Models, Animal Type of study: Diagnostic_studies / Prognostic_studies Limits: Animals / Female / Humans / Male Language: En Journal: Dev Dyn Journal subject: ANATOMIA Year: 2004 Document type: Article Affiliation country: United States Country of publication: United States