颅干骺端发育不良导致脑积水和Chiari畸形。

IF 0.6 Q3 MEDICINE, GENERAL & INTERNAL
Leigh Cui, Priya Kadam, Jamie Campbell, Chandrasekaran Kaliaperumal
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引用次数: 0

摘要

我们描述一个病例颅干骺端发育不良(CMD)最初被误诊为颅干骺端发育不良(CDD)。CMD和CDD都是影响颅面骨和管状骨的罕见遗传性疾病,分别由ANKH和SOST基因突变引起,在控制骨矿化方面造成类似的缺陷。该患者为男性,从出生到十几岁一直被纵向跟踪,涉及到CMD的误诊和管理的重要里程碑。我们讨论了相关的调查、基因检测诊断ANKH突变和神经外科治疗,因为患者成功地接受了大孔减压治疗继发性Chiari I畸形。我们称这位患者为“先证者”,因为他是家族中第一个被诊断出患有这种遗传病的人。这项研究强调了正确识别潜在诊断的重要性,因为这可能影响管理。手术干预可能具有挑战性,但可以成功地控制危及生命的并发症。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Craniometaphyseal dysplasia leading to hydrocephalus and Chiari I malformation.

We describe a case of craniometaphyseal dysplasia (CMD) that was initially misdiagnosed as craniodiaphyseal dysplasia (CDD). CMD and CDD are both rare genetic disorders affecting the craniofacial and tubular bones, due to ANKH and SOST gene mutations, respectively, causing similar defects in the control of bone mineralisation.The patient is male, who has been followed longitudinally from birth to his mid-teens, touching on important milestones concerning misdiagnosis and management of CMD. We discuss relevant investigations, diagnosis of ANKH mutation on genetic testing and neurosurgical management, as the patient successfully underwent foramen magnum decompression for secondary Chiari I malformation. We refer to the patient as 'proband' as he is the first in his family diagnosed with a genetic condition.This study highlights the importance of correct identification of the underlying diagnosis as this can affect management. Surgical intervention can be challenging but can successfully manage life-threatening complications of this condition.

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来源期刊
BMJ Case Reports
BMJ Case Reports Medicine-Medicine (all)
CiteScore
1.40
自引率
0.00%
发文量
1588
期刊介绍: BMJ Case Reports is an important educational resource offering a high volume of cases in all disciplines so that healthcare professionals, researchers and others can easily find clinically important information on common and rare conditions. All articles are peer reviewed and copy edited before publication. BMJ Case Reports is not an edition or supplement of the BMJ.
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