Department of Neurology, Peking University First Hospital, Beijing, China.
Department of Neurology, The First Affiliated Hospital of Nanchang University, Nanchang, China.
Acta Neuropathol Commun. 2022 May 31;10(1):79. doi: 10.1186/s40478-022-01383-y.
Recently, inspired by the similar clinical and pathological features shared with fragile X-associated tremor/ataxia syndrome (FXTAS), abnormal expansion of CGG repeats in the 5' untranslated region has been found in neuronal intranuclear inclusion disease (NIID), oculopharyngeal myopathy with leukoencephalopathy (OPML), and oculopharyngodistal myopathy (OPDMs). Although the upstream open reading frame has not been elucidated in OPML and OPDMs, polyglycine (polyG) translated by expanded CGG repeats is reported to be as a primary pathogenesis in FXTAS and NIID. Collectively, these findings indicate a new disease entity, the polyG diseases. In this review, we state the common clinical manifestations, pathological features, mechanisms, and potential therapies in these diseases, and provide preliminary opinions about future research in polyG diseases.
最近,受脆性 X 相关震颤/共济失调综合征 (FXTAS) 相似临床和病理特征的启发,在神经元核内包涵体病 (NIID)、眼咽肌病伴脑白质病 (OPML) 和眼咽远端肌病 (OPDMs) 中发现了 5'非翻译区 CGG 重复序列的异常扩增。尽管 OPML 和 OPDMs 中的上游开放阅读框尚未阐明,但据报道,由扩增的 CGG 重复翻译的多聚甘氨酸 (polyG) 是 FXTAS 和 NIID 的主要发病机制。这些发现共同表明了一种新的疾病实体,即多聚甘氨酸疾病。在这篇综述中,我们阐述了这些疾病的常见临床表现、病理特征、机制和潜在治疗方法,并对多聚甘氨酸疾病的未来研究提供了初步意见。