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创伤性自伤性心室撕裂:一例成年史密斯-莱姆利-奥皮茨综合征病例

Traumatic Self-Inflicted Ventricular Laceration: A Case of Smith-Lemli-Opitz Syndrome in an Adult.

作者信息

Beuschel Jennifer J, Ng Grace I, Abaraoha Joanna C, Fortuna Robert J

机构信息

Internal Medicine and Pediatrics, University of Rochester Medical Center, Rochester, USA.

出版信息

Cureus. 2024 Feb 5;16(2):e53613. doi: 10.7759/cureus.53613. eCollection 2024 Feb.

DOI:10.7759/cureus.53613
PMID:38449995
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10916526/
Abstract

Adults with intellectual and developmental disabilities (IDD) are increasingly living into adulthood, highlighting the need for adult clinicians to expand their familiarity with congenital conditions. Smith-Lemli-Opitz syndrome (SLOS) is a rare autosomal recessive inborn error of cholesterol synthesis. SLOS is commonly diagnosed in childhood, but a number of adults with IDD progress into adulthood without a formal diagnosis. We present an 18-year-old male with a history of IDD and altered pain sensation who was hospitalized following a self-inflicted knife injury resulting in a traumatic ventricular septal defect. Over the following 15 years, the patient continued to exhibit self-injurious behaviors. At the age of 33, caregivers consented to further work-up of his intellectual disability, and whole-exome genetic sequencing revealed a diagnosis of SLOS. The clinical course of this patient represents a unique presentation of altered pain sensation, a delayed diagnosis of SLOS into adulthood, and the challenges of providing care to an adult with IDD. The case further highlights the importance of understanding the typical workup and management of genetic and congenital conditions arising in childhood.

摘要

患有智力和发育障碍(IDD)的成年人越来越多地步入成年期,这凸显了成年临床医生需要扩大对先天性疾病的了解。史密斯-勒米-奥皮茨综合征(SLOS)是一种罕见的常染色体隐性遗传性胆固醇合成障碍。SLOS通常在儿童期被诊断出来,但许多患有IDD的成年人在没有正式诊断的情况下进入成年期。我们报告一名18岁男性,有IDD病史且痛觉改变,因自伤刀伤导致创伤性室间隔缺损而住院。在接下来的15年里,该患者继续表现出自伤行为。33岁时,护理人员同意对他的智力残疾进行进一步检查,全外显子基因测序显示诊断为SLOS。该患者的临床病程代表了痛觉改变的独特表现、SLOS成年期延迟诊断以及为患有IDD的成年人提供护理的挑战。该病例进一步凸显了了解儿童期出现的遗传和先天性疾病的典型检查和管理的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/b45d029746ae/cureus-0016-00000053613-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/64222ded18da/cureus-0016-00000053613-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/1fe4b7b33905/cureus-0016-00000053613-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/5156522e1876/cureus-0016-00000053613-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/db9c257623cf/cureus-0016-00000053613-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/b45d029746ae/cureus-0016-00000053613-i05.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/64222ded18da/cureus-0016-00000053613-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/1fe4b7b33905/cureus-0016-00000053613-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/5156522e1876/cureus-0016-00000053613-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/db9c257623cf/cureus-0016-00000053613-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/1f05/10916526/b45d029746ae/cureus-0016-00000053613-i05.jpg

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