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脊椎骨骺发育不良,短肢-异常钙化型中异常钙化的发病时间。

The time of onset of abnormal calcification in spondylometaepiphyseal dysplasia, short limb-abnormal calcification type.

作者信息

Tüysüz Beyhan, Gazioğlu Nurperi, Ungür Savaş, Aji Dolly Yafet, Türkmen Seval

机构信息

Department of Pediatric Genetics, Cerrahpaşa Medical School, Istanbul University, Istanbul, Turkey.

出版信息

Pediatr Radiol. 2009 Jan;39(1):84-9. doi: 10.1007/s00247-008-1036-1. Epub 2008 Nov 11.

DOI:10.1007/s00247-008-1036-1
PMID:19002453
Abstract

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.

摘要

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。

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本文引用的文献

1
Spondylo-meta-epiphyseal dysplasia, short limbs, abnormal calcification type: a new case with severe neurological involvement.脊椎干骺端发育异常,短肢,异常钙化型:一例伴有严重神经受累的新病例
Pediatr Radiol. 2001 Jan;31(1):19-22. doi: 10.1007/s002470000359.
2
Spondylo-meta-epiphyseal dysplasia, short limb, abnormal calcification type.脊椎骨骺发育异常,短肢,异常钙化型。
Clin Dysmorphol. 1996 Jul;5(3):197-206.
3
Further delineation of spondylo-meta-epiphyseal dysplasia, short limb-abnormal calcification type, with emphasis on diagnostic features.
进一步扩展伴有异常钙化的骨-软骨-干骺端发育不良的突变谱。
J Hum Genet. 2018 Sep;63(9):1003-1007. doi: 10.1038/s10038-018-0473-4. Epub 2018 Jun 8.
4
Spondyloepimetaphyseal dysplasia, short limb-abnormal calcifications type: progressive radiological findings from fetal age to adolescence.短肢-骨发育不良伴异常钙化型脊椎干骺端发育不良:从胎儿期到青春期的进行性影像学发现。
Pediatr Radiol. 2011 Oct;41(10):1298-307. doi: 10.1007/s00247-011-2123-2. Epub 2011 Aug 5.
5
Trafficking defects and loss of ligand binding are the underlying causes of all reported DDR2 missense mutations found in SMED-SL patients.所有在 SMED-SL 患者中发现的 DDR2 错义突变均归因于转运缺陷和配体结合丢失。
Hum Mol Genet. 2010 Jun 1;19(11):2239-50. doi: 10.1093/hmg/ddq103. Epub 2010 Mar 10.
6
Mutations in DDR2 gene cause SMED with short limbs and abnormal calcifications.DDR2基因的突变会导致肢体短小并伴有异常钙化的小眼线虫病。
Am J Hum Genet. 2009 Jan;84(1):80-4. doi: 10.1016/j.ajhg.2008.12.004. Epub 2008 Dec 24.
脊椎干骺端发育不良,短肢-异常钙化型的进一步描述,重点在于诊断特征。
Am J Med Genet. 1993 Feb 15;45(4):488-500. doi: 10.1002/ajmg.1320450419.
4
Spondylo-meta-epiphyseal dysplasia (SMED), short limb-hand type: a congenital familial skeletal dysplasia with distinctive features and histopathology.脊椎-干骺端-骨骺发育不良(SMED),短肢-手型:一种具有独特特征和组织病理学表现的先天性家族性骨骼发育不良。
Am J Med Genet. 1993 Feb 1;45(3):320-6. doi: 10.1002/ajmg.1320450308.