School of Biology, College of Science, University of Tehran, Tehran, Iran.
Department of Neurology, Tehran University of Medical Sciences, Tehran, Iran.
Am J Med Genet A. 2019 Aug;179(8):1507-1515. doi: 10.1002/ajmg.a.61184. Epub 2019 May 20.
Charcot-Marie-Tooth (CMT) is a common neuropathy, and hereditary motor and sensory neuropathy with proximal predominance (HMSN-P) is a recently described rare neuromuscular disease. Although many genes have been implicated for CMT, TFG is the only known HMSN-P-causing gene. Within the framework of diagnostic criteria, clinical variation is evident among CMT-diagnosed and also HMSN-P-diagnosed individuals. Mutations that cause p.(Pro285Leu) and p.(Gly269Val) in TFG were earlier reported as cause of HMSN-P in two Iranian pedigrees. Here, we report the identification of p.(Gly269Val) in TFG as cause of CMT in a large Iranian pedigree. The clinical features of patients of the three pedigrees are presented and critically compared. Similarities between the two HMSN-P-diagnosed pedigrees with different TFG mutations, and differences between the two differentially diagnosed pedigrees with the same p.(Gly269Val) mutation were evident. The clinical features of the HMSN-P pedigree with the p.(Pro285Leu) and the CMT pedigree with the p.(Gly269Val) mutation were clearly congruent with the respective diagnoses, whereas the features of the HMSN-P-diagnosed pedigree with the p.(Gly269Val) were intermediate between the other two pedigrees. It is therefore suggested that the clinical features of the three Iranian pedigrees with TFG mutations and diagnosed with HMSN-P or CMT represent a continuum.
Charcot-Marie-Tooth (CMT) 是一种常见的周围神经病,遗传性运动感觉神经病伴近端优势(HMSN-P)是一种最近描述的罕见神经肌肉疾病。虽然许多基因与 CMT 有关,但 TFG 是唯一已知的导致 HMSN-P 的基因。在诊断标准的框架内,CMT 诊断和 HMSN-P 诊断个体之间存在明显的临床差异。导致 TFG 中 p.(Pro285Leu)和 p.(Gly269Val)的突变早些时候被报道为两个伊朗家系中 HMSN-P 的病因。在这里,我们报告了在一个大型伊朗家系中 TFG 中 p.(Gly269Val)突变导致 CMT 的鉴定。提出了三个家系患者的临床特征,并进行了批判性比较。具有不同 TFG 突变的两个 HMSN-P 诊断家系之间存在相似性,以及具有相同 p.(Gly269Val)突变的两个不同诊断家系之间存在差异。具有 p.(Pro285Leu)的 HMSN-P 家系和具有 p.(Gly269Val)突变的 CMT 家系的临床特征与各自的诊断明显一致,而具有 p.(Gly269Val)的 HMSN-P 诊断家系的特征则介于其他两个家系之间。因此,建议具有 TFG 突变并诊断为 HMSN-P 或 CMT 的三个伊朗家系的临床特征代表一个连续体。