Tüysüz Beyhan, Gazioğlu Nurperi, Ungür Savaş, Aji Dolly Yafet, Türkmen Seval
Department of Pediatric Genetics, Cerrahpaşa Medical School, Istanbul University, Istanbul, Turkey.
Pediatr Radiol. 2009 Jan;39(1):84-9. doi: 10.1007/s00247-008-1036-1. Epub 2008 Nov 11.
A 1-month-old boy with shortness of extremities on prenatal US was referred to our department with a provisional diagnosis of achondroplasia. His height was normal but he had short extremities and platyspondyly, premature carpal epiphyses on both hands, and short tubular bones with irregular metaphyses on radiographs. Re-evaluation of the patient at the age of 1 year revealed very short height and premature calcification of the costal cartilages and epiphyses. Spondylometaepiphyseal dysplasia (SMED), short limb-abnormal calcification type was diagnosed. This condition is a very rare autosomal recessively inherited disorder, and most of the patients die in early childhood due to neurological involvement. At the age of 2 years and 5 months, a CT scan showed narrowing of the cervical spinal canal. One month later he died suddenly because of spinal cord injury. In conclusion early diagnosis is very important because the recurrence risk is high and patients may die due to early neurological complications. The time of onset of abnormal calcifications, a diagnostic finding of the disease, is at the age of around 1 year in most patients. When abnormal calcifications are not yet present, but radiological changes associated with SMED are present, this rare disease must be considered.
一名产前超声检查发现四肢短小的1个月大男婴被转诊至我科,初步诊断为软骨发育不全。他身高正常,但四肢短小且椎体扁平,双手腕骨骨骺早熟,X线片显示管状骨短且干骺端不规则。1岁时对该患者进行重新评估,发现身高极矮,肋软骨和骨骺过早钙化。诊断为脊椎骨骺发育不良(SMED),短肢-异常钙化型。这种疾病是一种非常罕见的常染色体隐性遗传疾病,大多数患者因神经受累在幼儿期死亡。2岁5个月时,CT扫描显示颈椎管狭窄。1个月后,他因脊髓损伤突然死亡。总之,早期诊断非常重要,因为复发风险高,患者可能因早期神经并发症而死亡。异常钙化的出现时间是该病的一个诊断依据,大多数患者在1岁左右出现。当尚未出现异常钙化,但存在与SMED相关的放射学改变时,必须考虑这种罕见疾病。