Clinical Genetics Department, Human Genetics and Genome Research Division, National Research Centre, Cairo 12311, Egypt.
Howard Hughes Medical Institute, University of California, San Diego, La Jolla, CA 92093, USA.
Gene. 2014 Apr 15;539(2):279-82. doi: 10.1016/j.gene.2014.01.070. Epub 2014 Feb 13.
Fukuyama-type congenital muscular dystrophy (FCMD, MIM#253800) is an autosomal recessive disorder characterized by severe muscular dystrophy associated with brain malformations. FCMD is the second most common form of muscular dystrophy after Duchenne muscular dystrophy and one of the most common autosomal recessive diseases among the Japanese population, and yet few patients outside of Japan had been reported with this disorder. We report the first known Egyptian patient with FCMD, established by clinical features of generalized weakness, pseudohypertrophy of calf muscles, progressive joint contractures, severe scoliosis, elevated serum creatine kinase level, myopathic electrodiagnostic changes, brain MRI with cobblestone complex, and mutation in the fukutin gene. In addition, our patient displayed primary microcephaly, not previously reported associated with fukutin mutations. Our results expand the geographic and clinical spectrum of fukutin mutations.
福山型先天性肌肉萎缩症 (FCMD,MIM#253800) 是一种常染色体隐性疾病,其特征为严重的肌肉萎缩症伴有脑部畸形。FCMD 是继杜兴氏肌肉营养不良症之后第二常见的肌肉萎缩症类型,也是日本人中最常见的常染色体隐性疾病之一,但在日本以外的地区,很少有报道过这种疾病。我们报告了首例已知的埃及 FCMD 患者,该患者具有全身性肌无力、小腿假性肥大、进行性关节挛缩、严重脊柱侧凸、血清肌酸激酶水平升高、肌病性电诊断改变、鹅卵石样脑 MRI 以及 fukutin 基因突变等临床表现。此外,我们的患者还表现出原发性小头畸形,这与 fukutin 基因突变以前未被报道过。我们的研究结果扩展了 fukutin 基因突变的地理和临床谱。