迪乔治综合征在 38 岁时被确诊:低资源环境下的挑战与漏诊的影响》。

JCEM case reports Pub Date : 2024-07-24 eCollection Date: 2024-07-01 DOI:10.1210/jcemcr/luae136
William Kuenstner, Suthee Rapisuwon, Leila Shobab
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引用次数: 0

摘要

22q11.2 缺失综合征(22.q11.2 DS)是一种由 22 号染色体微缺失导致的遗传综合征。它的表现多种多样,大多数病例在儿童早期就被诊断出来。我们介绍了一例出生在发展中国家的 38 岁女性病例,她因原发性甲状腺功能减退症来我院就诊。在补充甲状腺素后,她的临床和生化指标均为甲状腺功能正常。我们还发现她在 PTH 偏低的情况下出现了低钙血症,之前曾给她开过降钙素三醇。鉴于患者有腭裂史、面部特征异常、轻度反复鼻窦肺部感染以及内分泌病史(包括身高在第6百分位的矮身材),医生对她进行了基因检测。她被诊断为 TBX1 基因杂合子全基因缺失。额外的基因评估显示,22a11.2 区域有一个 2.6 兆字节的微缺失区段,包含 62 个基因。鉴于患者有快速性心律失常病史,该患者被转诊至心脏科,以评估是否累及心脏。该病例凸显了 22.q11.2 DS 的诊断难题和延迟诊断的影响。
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DiGeorge Syndrome Diagnosed at Age 38: Challenges in Low-resource Settings and Implications of a Missed Diagnosis.

22q11.2 deletion syndrome (22.q11.2 DS) is a genetic syndrome resulting from a microdeletion on chromosome 22. It has a diverse array of manifestations, and most cases are diagnosed early in childhood. We present the case of a 38-year-old female born in a developing country who presented to our clinic to establish care for a history of primary hypothyroidism. She was clinically and biochemically euthyroid on thyroid supplementation. She was also noted to have hypocalcemia in the setting of low PTH, for which the patient was previously prescribed calcitriol. Given a history of cleft palate, abnormal facial features, mild recurrent sinopulmonary infections, and her endocrine history (including short stature with height in the 6th percentile), genetic testing was obtained. She was diagnosed with a heterozygous whole gene deletion of the TBX1 gene. Additional genetic evaluation demonstrated a 2.6-Mb microdeleted segment of the 22a11.2 region encompassing 62 genes. The patient was referred to cardiology for evaluation of cardiac involvement given a history of tachyarrhythmia. This case highlights challenges in diagnosis and the implications of a delayed diagnosis of 22.q11.2 DS.

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