Mroczek Magdalena, Durmus Hacer, Bijarnia-Mahay Sunita, Töpf Ana, Ghaoui Roula, Bryen Samantha, Duff Jennifer, England Eleina, Cooper Sandra T, MacArthur Daniel G, Straub Volker
John Walton Muscular Dystrophy Research Centre, Translational and Clinical Research Institute, Newcastle University and Newcastle Hospitals NHS Foundation Trust, Central Parkway, Newcastle upon Tyne NE1 3BZ, UK.
Department of Neurology, Neuromuscular disorders Unit, Istanbul Faculty of Medicine, Istanbul University, Istanbul, Turkey.
Neuromuscul Disord. 2020 Apr;30(4):310-314. doi: 10.1016/j.nmd.2020.02.006. Epub 2020 Feb 14.
Adenylosuccinate synthase (ADSSL1) is a muscle specific enzyme involved in the purine nucleotide cycle and responsible for the conversion of inosine monophosphate to adenosine monophosphate. Since 2016, when mutations in the ADSSL1 gene were first described to be associated with an adult onset distal myopathy, nine patients with compound heterozygous variants in the ADSSL1 gene, all of Korean origin, have been identified. Here we report a novel ADSSL1 mutation and describe two sporadic cases of Turkish and Indian origin. Many of the clinical features of both patients and muscle histopathology and muscle MRI findings, were in accordance with previously reported findings in the adult onset distal myopathy individuals. However, one of our patients presented with progressive, proximally pronounced weakness, severe muscle atrophy and early contractures. Thus, mutations in ADSSL1 have to be considered in patients with both distal and proximal muscle weakness and across various ethnicities.
腺苷酸琥珀酸合成酶(ADSSL1)是一种肌肉特异性酶,参与嘌呤核苷酸循环,负责将肌苷单磷酸转化为腺苷单磷酸。自2016年首次报道ADSSL1基因突变与成人起病的远端肌病相关以来,已鉴定出9例ADSSL1基因复合杂合变异患者,均为韩国人。在此,我们报告了一种新的ADSSL1突变,并描述了两例分别来自土耳其和印度的散发病例。两名患者的许多临床特征、肌肉组织病理学和肌肉MRI表现均与先前报道的成人起病远端肌病患者的表现一致。然而,我们的一名患者表现为进行性、近端明显无力、严重肌肉萎缩和早期挛缩。因此,对于远端和近端肌肉无力的患者,无论何种种族,都必须考虑ADSSL1基因突变。