MedElevator Войти в исследования
← Поиск публикаций

DOI 10.1177/088307389000500311

Медицинская библиотека · Journal of child neurology · 1990

Rett syndrome: a mitochondrial disease?

Eeg-Olofsson O, al-Zuhair A G, Teebi A S, Daoud A S, Zaki M, Besisso M S, Al-Essa M M

СтатьяПеревод пока не подготовлен

Аннотация

Six girls between 2 years 9 months and 15 years of age with Rett syndrome were thoroughly investigated. Blood ammonia levels varied between 42 and 123 mumol/L, and serum lactate concentration was slightly elevated in two girls. Electroencephalograms showed a dysrhythmic pattern during wakefulness; during drowsiness and light sleep, bilateral bursts of spike or multispike-and-wave activity were seen in all but the oldest girl. In one of the younger girls, slight cortical atrophy was found on computed tomographic scan. Muscle biopsy was performed on all girls, and electron microscopy revealed abnormal mitochondria. Physical signs such as somatic hypotrophy with extremely small muscle mass, and unsatisfactory weight gain in spite of good appetite are found in Rett syndrome. These attributes, as well as reports of ornithine carbamoyltransferase deficiency, may support a mitochondrial dysfunction. The mitochondrial changes indicate either a mitochondrial mutation or more probably an X-borne modulator gene mutation. Another genetic possibility discussed is the "metabolic interference" of an X-borne allele. Further delineation of such mitochondrial changes may clarify the causal metabolic defect in Rett syndrome.

Для цитирования: Eeg-Olofsson O, al-Zuhair A G, Teebi A S, Daoud A S, Zaki M, Besisso M S, Al-Essa M M. Rett syndrome: a mitochondrial disease?. Journal of child neurology. 1990. DOI: 10.1177/088307389000500311.

Предметные рубрики MeSH

Подготовка и перевод публикации
Обработка статьи · этапы, ошибки и управление
Загрузка состояния…

История · было → стало

Состояние обновляется каждые 5 секунд, пока блок открыт. Процент показывается только там, где известен объём работы. Пауза останавливает продолжение этапа; текущий запрос к источнику может завершиться.

Тип публикации указан по данным источника. Уровень доказательности автоматически не присваивается.