Department of Pathology, Princess Margaret Hospital, Hong Kong, China.
Clin Genet. 2012 Apr;81(4):394-8. doi: 10.1111/j.1399-0004.2011.01659.x. Epub 2011 Apr 4.
In contrast to the usual slow disease progression in myofibrillar myopathies, patients with Bag3opathy often have a rapidly progressive and more severe phenotype with a worse prognosis. We describe a Chinese patient, born to non-consanguineous parents, who first presented at age 6 with clumsy walking and difficult climbing staircase. With a history of restrictive lung disease previously diagnosed as asthma, she progressed rapidly with proximal myopathy, rigid spine and bilateral tightening of the Achilles tendons requiring surgical elongation. Hypertrophic cardiomyopathy with restrictive physiology was shown by echocardiogram. Moreover, prolonged QT interval was also noted in the patient. Family history was unremarkable yet her father was incidentally found to have prolonged QT interval. Mutation analysis with genomic DNA of the proband showed heterozygous de novo known mutation c.626C>T (p.Pro209Leu) and a germline variation c.772C>T (p.Arg258Trp) in BAG3. Her father was found to be a carrier of c.772C>T. Muscle biopsy findings were suggestive of myofibrillar myopathy on light microscopy and ultrastructural studies. To our knowledge, this is the first Chinese case of Bag3opathy so far reported.
与肌原纤维肌病的常见缓慢疾病进展相反,Bag3 病患者的表型通常具有快速进展和更严重的特点,预后更差。我们描述了一位中国患者,出生于非近亲父母,她在 6 岁时首次出现行走笨拙和难以爬楼梯的症状。她患有先前被诊断为哮喘的限制性肺病,疾病进展迅速,出现进行性近端肌病、僵直脊柱和双侧跟腱紧张,需要手术延长。超声心动图显示肥厚型心肌病伴限制性生理学。此外,还发现该患者 QT 间期延长。家族史无明显异常,但她的父亲偶然发现 QT 间期延长。对先证者的基因组 DNA 进行突变分析显示杂合新生 c.626C>T(p.Pro209Leu)和 BAG3 中的种系变异 c.772C>T(p.Arg258Trp)。她的父亲被发现是 c.772C>T 的携带者。肌肉活检结果在光镜和超微结构研究中提示肌原纤维肌病。据我们所知,这是迄今为止报告的首例中国 Bag3 病病例。