AlSaman Abdulaziz, Tomoum Hoda
Department of Pediatric Neurology, King Fahad Medical City, Riyadh 11525, Kingdom of Saudi Arabia.
J Child Neurol. 2010 Jun;25(6):764-9. doi: 10.1177/0883073809344121. Epub 2010 Mar 1.
The condition, currently known as spinal muscular atrophy with respiratory distress type 1, is an unusual variant of spinal muscular atrophy type 1 that is characterized by early respiratory failure due to diaphragmatic paralysis. The defective gene, the immunoglobulin mu-binding protein 2 (IGHMBP2 gene), of this autosomal recessive disorder is located on chromosome 11q13 and encodes immunoglobulin mu-binding protein 2. The natural history and phenotypic spectrum of the disease are still not clear. The authors present the first genetically proven case of spinal muscular atrophy with respiratory distress type 1 to be reported from Saudi Arabia. The parents are first cousins and the causative gene sequencing revealed mutation in exon 7 reported for the first time in a homozygous form. The clinical scenario of the case is discussed. The findings in the muscle magnetic resonance imaging (MRI) are presented.
这种疾病目前被称为伴有呼吸窘迫的1型脊髓性肌萎缩症,是1型脊髓性肌萎缩症的一种不寻常变体,其特征是由于膈肌麻痹导致早期呼吸衰竭。这种常染色体隐性疾病的缺陷基因是免疫球蛋白μ结合蛋白2(IGHMBP2基因),位于11号染色体q13上,编码免疫球蛋白μ结合蛋白2。该疾病的自然病史和表型谱仍不清楚。作者报告了沙特阿拉伯首例经基因证实的伴有呼吸窘迫的1型脊髓性肌萎缩症病例。父母是近亲,致病基因测序显示外显子7存在首次以纯合形式报道的突变。本文讨论了该病例的临床情况,并展示了肌肉磁共振成像(MRI)的检查结果。