Genetic characterisation of spontaneous ankylosing arthropathy with unique inheritance from Fas-deficient strains of mice
| Autor: | Masao Ono, Naoko Tanda, Mingcai Zhang, Fumiko Date, Shiro Mori, Hiroshi Furukawa, Masato Nose |
|---|---|
| Jazyk: | angličtina |
| Rok vydání: | 2006 |
| Předmět: |
Male
medicine.medical_specialty Pathology Mice Inbred MRL lpr Genotype Ratón Immunology Ankylosis Quantitative Trait Loci Locus (genetics) Quantitative trait locus urologic and male genital diseases Severity of Illness Index General Biochemistry Genetics and Molecular Biology Mice Rheumatology immune system diseases Internal medicine Arthropathy medicine Inheritance Mode Immunology and Allergy Animals Genetic Predisposition to Disease fas Receptor skin and connective tissue diseases Mice Inbred C3H Ossification business.industry medicine.disease Extended Report Disease Models Animal Female medicine.symptom business Microsatellite Repeats |
| Zdroj: | Annals of the Rheumatic Diseases. 65(10):1273-1278 |
| ISSN: | 0003-4967 |
| Popis: | Background: The spontaneous onset of macroscopic arthropathy in the ankle of the particular F 1 mice descended from two Fas -deficient strains of mice; a mutant substrain of MRL/Mp. Fas lpr (MRL/rpl) and C3H/He. Fas lpr (C3H/lpr) was recently observed. Aim: To histopathologically characterise and genetically interpret the unique inheritance mode of disease in this arthropathy model. Methods: MRL/rpl, C3H/lpr, (MRL/rpl × C3H/lpr; MC) F 1 , (C3H/lpr × MRL/rpl; CM) F 1 and MCF 2 mice were bred under specific pathogen-free conditions. Histopathological grade of arthropathy was determined at 6 months by examination under a light microscope. To search for a linkage locus to the arthropathy, the whole genome of selected 48 male MCF 2 mice with 71 polymorphic microsatellite markers was scanned, followed by quantitative trait locus analysis. Results: The incidence of microscopically defined arthropathy in the male and female MCF 1 groups was 100% and 19.4%, respectively. No incidence was observed in the parental strains, MRL/rpl and C3H/lpr, and in CMF 1 mice. In the MCF 1 mice, the arthropathy mainly affected the ankle joints and was histopathologically characterised by marked entheseal proliferation with chondrocytic differentiation and ossification in the ankle joints, the manifestations similar to ankylosing enthesitis reported previously. An MRL/rpl-derived autosomal dominant susceptibility locus was mapped in the distal of D7Mit68 (60 cM) to the ankylosis onset. Conclusion: The MCF 1 mice stably develop spontaneous ankylosing disorders in the ankle, with a male predominance. The unique inheritance mode of ankylosis is possibly interpreted by the genetic interaction between the autosomal dominant locus and a Y-linked locus. |
| Databáze: | OpenAIRE |
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