Coêlho K E, Ramos E S, Felix T M, Martelli L, de Pina-Neto J M, Niikawa N
Departamento de Genetica, Faculdade de Medicina de Ribeirao Preto, Universidade de Sao Paulo, Brazil.
Am J Med Genet. 1998 Apr 28;77(1):12-5. doi: 10.1002/(sici)1096-8628(19980428)77:1<12::aid-ajmg3>3.0.co;2-n.
Spondylocarpotarsal synostosis syndrome (SSS) or congenital synspondylism is a recently delineated clinical entity. At least 15 patients have been reported. We present 3 new patients, 2 of whom were sibs born to first-cousin parents. All of our patients had multiple synostoses involving cervical, thoracic and/or lumbar vertebral bodies and carpal/tarsal bones, scoliosis/lordosis, and short stature. Sensorineural deafness was found in 2 of the 3 patients. Analysis of clinical manifestations suggests clinical variability and genetic heterogeneity in SSS. Of a total of 18 SSS patients, 10 were five pairs of sibs from five families, with first-cousin consanguinity of parents in 3, indicating that at least one type of SS is an autosomal-recessive disorder.
脊椎腕跗骨融合综合征(SSS)或先天性脊柱融合症是一种最近才被明确的临床病症。至少已有15例患者被报道。我们报告3例新患者,其中2例是表亲父母所生的同胞。我们所有的患者都有多个关节融合,累及颈椎、胸椎和/或腰椎椎体以及腕骨/跗骨,有脊柱侧凸/脊柱前凸,且身材矮小。3例患者中有2例发现感音神经性耳聋。对临床表现的分析表明SSS存在临床变异性和遗传异质性。在总共18例SSS患者中,10例是来自5个家庭的5对同胞,其中3对父母为近亲表亲,这表明至少有一种类型的SS是常染色体隐性疾病。