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Retinopathia Pigmentosa Associated Syndrome in LEW/Ztm-ci2 Mutated Rats
1Gockeln R., 2Lindemann S., 2Loescher W., 3Sistani F., 4Winter R.,
1Medizinische Hochschule Hannover, Augenheilkunde, Elektrophysiologisches Labor (Hannover)
2Tiermedizinische Hochschule Hannover, Pharmakologie und Toxikologie (Hannover)
3Wilhelm-Fresenius-Klinik, Klinik für Augenheilkunde (Wiesbaden)
4Medizinische Hochschule Hannover, Augenheilkunde (Hannover)
Purpose: Genetic animal models (spontaneous mutations, transgenic animals) play an important role in understanding the pathogenesis and in searching for therapeutic options in hereditary diseases of the visual system. Lately, cochlear and vestibular dysfunctions combined with hyperkinetic disorders of the movements could be demonstrated in LEW/Ztm-ci2 mutated rats, which can be inherited in an autosomal recessive manner. An increase of the movement disorders could cause an associated retinopathia pigmentosa (RP) in the mutated rats.
Methods: A Ganzfeld ERG (Roland Consults, Retiscan) was performed in 15 LEW/Ztm-ci2 mutated rats , 15 heterozygous siblings of these animals and 10 LEW-Rats (control group) of different ages.
Results: All LEW/Ztm-ci2 mutated rats and the heterozygous siblings showed, depending on their age a reduced scotopic and photopic ERG compatible with a rod-cone dystrophy. ERG of the control group was normal. Rat chromosome 10 could be shown to be the locus of the genetic defect. The exact locus of the defect is under investigation right now.
Conclusions: The neurological deficits of the LEW/Ztm-ci2 mutated rats are associated with a rod-cone dystrophy and show a new RP associated animal model with an autosomal recessive pattern of inheritance.