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Searching for novel biomarkers using a mouse model of CLN3-Batten disease.
Timm, Derek; Cain, Jacob T; Geraets, Ryan D; White, Katherine A; Koh, Seung Yon; Kielian, Tammy; Pearce, David A; Hastings, Michelle L; Weimer, Jill M.
Afiliación
  • Timm D; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, South Dakota, United States of America.
  • Cain JT; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, South Dakota, United States of America.
  • Geraets RD; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, South Dakota, United States of America.
  • White KA; The University of South Dakota, Sioux Falls, South Dakota, United States of America.
  • Koh SY; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, South Dakota, United States of America.
  • Kielian T; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, South Dakota, United States of America.
  • Pearce DA; Department of Pathology and Microbiology, University of Nebraska Medical Center, Omaha, Nebraska, United States of America.
  • Hastings ML; Pediatrics and Rare Diseases Group, Sanford Research, Sioux Falls, South Dakota, United States of America.
  • Weimer JM; The University of South Dakota, Sioux Falls, South Dakota, United States of America.
PLoS One ; 13(8): e0201470, 2018.
Article en En | MEDLINE | ID: mdl-30086172
CLN3-Batten disease is a rare, autosomal recessive disorder involving seizures, visual, motor and cognitive decline, and premature death. The Cln3Δex7/8 mouse model recapitulates several phenotypic characteristics of the most common 1.02kb disease-associated deletion. Identification of reproducible biomarker(s) to facilitate longitudinal monitoring of disease progression and provide readouts for therapeutic response has remained elusive. One factor that has complicated the identification of suitable biomarkers in this mouse model has been that variations in animal husbandry appear to significantly influence readouts. In the current study, we cross-compared a number of biological parameters in blood from Cln3Δex7/8 mice and control, non-disease mice on the same genetic background from multiple animal facilities in an attempt to better define a surrogate marker of CLN3-Batten disease. Interestingly, we found that significant differences between Batten and non-disease mice found at one site were generally not maintained across different facilities. Our results suggest that colony variation in the Cln3Δex7/8 mouse model of CLN3-Batten disease can influence potential biomarkers of the disease.
Asunto(s)

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Glicoproteínas de Membrana / Chaperonas Moleculares / Variación Biológica Poblacional / Lipofuscinosis Ceroideas Neuronales Tipo de estudio: Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Animals / Humans / Male Idioma: En Revista: PLoS One Asunto de la revista: CIENCIA / MEDICINA Año: 2018 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos

Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Glicoproteínas de Membrana / Chaperonas Moleculares / Variación Biológica Poblacional / Lipofuscinosis Ceroideas Neuronales Tipo de estudio: Observational_studies / Prognostic_studies / Risk_factors_studies Límite: Animals / Humans / Male Idioma: En Revista: PLoS One Asunto de la revista: CIENCIA / MEDICINA Año: 2018 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Estados Unidos