| Literature DB >> 2242177 |
S Wakai1, K Kameda, Y Ishikawa, S Miyamoto, M Nagaoka, M Okabe, R Minami, N Tachi.
Abstract
We report the histopathologic findings of 3 sural nerve biopsies and 1 muscle biopsy from 3 patients with Rett syndrome. The 3 sural nerve biopsies demonstrated a few ultrastructural abnormalities, including the presence of many Pi-granules and mitochondrial changes in the cytoplasm of Schwann cells, occasional bands of Büngner and onion-bulb formations, and mitochondrial alterations in myelinated axons. Morphometric analysis disclosed reduction in the number of large myelinated fibers with normal densities in comparison to those of an age-matched normal control. Light microscopic examination of the biopsied muscle from a 6-year-old patient with Rett syndrome revealed the existence of many small, dark, angulated fibers with NADH-TR staining. Ultrastructural investigation of the muscle confirmed the presence of the dumbbell-shaped mitochondria. Peripheral nerve involvement and the possibility of mitochondrial abnormalities in Rett syndrome were suggested by the results.Entities:
Mesh:
Year: 1990 PMID: 2242177 DOI: 10.1016/0887-8994(90)90028-y
Source DB: PubMed Journal: Pediatr Neurol ISSN: 0887-8994 Impact factor: 3.372