Department of Neurology, Royal North Shore Hospital, St. Leonards, NSW 2065, Australia.
Neuromuscul Disord. 2010 May;20(5):330-4. doi: 10.1016/j.nmd.2010.03.002. Epub 2010 Mar 23.
We report the first Australian families with inclusion-body myopathy, Paget's disease of the bone and frontotemporal dementia (IBMPFD). The clinical characteristics of the two pedigrees are described including a previously undescribed phenotypic feature of pyramidal tract dysfunction in one family member. A novel mutation in the valosin-containing protein (VCP) gene (p.Arg155Leu) was found in one family while the other family had a previously reported mutation (p.Leu198Trp). Our findings broaden the phenotypic spectrum of IBMPFD and further emphasise the resemblance to amyotrophic lateral sclerosis in some cases.
我们报道了首例澳大利亚包涵体肌病、骨 Paget 病和额颞叶痴呆(IBMPFD)的家系。描述了这两个家系的临床特征,包括一个家系中以前未描述的锥体束功能障碍的表型特征。一个家系中发现了一种新型的 VCP 基因突变(p.Arg155Leu),另一个家系则携带了以前报道的突变(p.Leu198Trp)。我们的发现拓宽了 IBMPFD 的表型谱,并进一步强调了在某些情况下与肌萎缩侧索硬化症的相似性。