Clinical Genetic Service, Department of Health, Cheung Sha Wan Jockey Club Clinic, Shamshuipo, Kowloon, Hong Kong.
Hong Kong Med J. 2013 Dec;19(6):556-9. doi: 10.12809/hkmj133739.
Despite the advances in the understanding of the molecular basis for oculopharyngeal muscular dystrophy in the last decade, it remains an underdiagnosed disease, especially among the Chinese. In the presence of a positive family history and late-onset ptosis, dysphagia, and proximal muscle weakness (its cardinal features), we suggest that PABPN1 gene analysis should be the first-line investigation to rule out this condition. Muscle biopsy can be reserved for atypical cases. Non-specific mitochondrial changes in the muscle specimens of these patients should be appreciated, so as to avoid diagnostic confusion. It is hoped that greater awareness among medical professionals and judicious use of PABPN1 gene analysis will lead to earlier diagnosis, better management, and avoidance of unnecessary invasive investigations of affected patients.
尽管在过去十年中,人们对眼咽型肌营养不良症的分子基础有了更深入的了解,但它仍然是一种诊断不足的疾病,尤其是在中国。如果存在阳性家族史和迟发性上睑下垂、吞咽困难和近端肌肉无力(其主要特征),我们建议应首先进行 PABPN1 基因分析以排除这种情况。对于不典型病例,可以保留肌肉活检。应注意这些患者的肌肉标本中存在非特异性线粒体变化,以避免诊断混淆。希望通过提高医疗专业人员的认识和明智地使用 PABPN1 基因分析,可以实现更早的诊断、更好的管理,并避免对受影响的患者进行不必要的侵入性检查。