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成人先天性氯性腹泻和溃疡性结肠炎重叠误诊为巴特综合征的诊断挑战:病例报告及文献复习。

Diagnostic Challenge of Congenital Chloride Diarrhea and Ulcerative Colitis Overlap in an Adult Misdiagnosed with Bartter Syndrome: Case Report and Literature Review.

机构信息

Department of Medicine, King Abdulaziz Medical City, Ministry of National Guard Health Affairs, Jeddah, Saudi Arabia.

King Abdullah International Research Center, Ministry of National Guard Health Affairs, Jeddah, Saudi Arabia.

出版信息

Am J Case Rep. 2022 Jul 5;23:e936715. doi: 10.12659/AJCR.936715.

DOI:10.12659/AJCR.936715
PMID:35787602
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9272577/
Abstract

BACKGROUND Congenital chloride diarrhea (CCD) is an autosomal recessive disease that is usually diagnosed in early childhood. Mutations in the SLC26A3 gene have been attributed to the primary etiology of disease development. Patients with CCD usually present with electrolyte disturbances, metabolic alkalosis, and chronic diarrhea. Early diagnosis is essential to prevent long-term complications that often require genetic testing. Bartter syndrome is another congenital disorder that has clinical features similar to CCD, which might cause a delay in diagnosis in a few patients. CASE REPORT We describe the case of a 28-year-old man who was misdiagnosed as having Bartter syndrome when he was 5 months old based on the clinical features of hypokalemia, metabolic alkalosis, and a family history of Bartter syndrome. He had multiple admissions with diarrhea and was diagnosed with ulcerative colitis. Unfortunately, the course was complicated by renal failure, and the patient underwent a kidney transplant. Persistent metabolic alkalosis with diarrhea after transplantation was unusual in Bartter syndrome. Therefore, his primary diagnosis was challenged and suspicion of CCD was raised, which was confirmed by genetic testing. CONCLUSIONS CCD is a rare congenital disorder that requires high clinical suspicion and often a genetic test to confirm diagnosis. Here, we report a patient who was misdiagnosed as having Bartter syndrome until early adulthood owing to several misleading factors. We hope by reporting this case it will raise awareness about CCD in high-prevalence areas and the importance of early diagnosis to prevent serious complications.

摘要

背景

先天性氯性腹泻(CCD)是一种常染色体隐性疾病,通常在儿童早期诊断。SLC26A3 基因突变被认为是疾病发展的主要病因。CCD 患者通常表现为电解质紊乱、代谢性碱中毒和慢性腹泻。早期诊断对于预防常需基因检测的长期并发症至关重要。巴特综合征是另一种先天性疾病,其临床特征与 CCD 相似,这可能导致少数患者的诊断延迟。

病例报告

我们描述了一例 28 岁男性的病例,他在 5 个月大时因低钾血症、代谢性碱中毒和巴特综合征家族史的临床特征被误诊为巴特综合征。他因腹泻多次住院,并被诊断为溃疡性结肠炎。不幸的是,病情发展为肾衰竭,患者接受了肾移植。巴特综合征移植后出现持续性代谢性碱中毒伴腹泻并不常见。因此,对他的主要诊断提出了质疑,并怀疑是 CCD,这通过基因检测得到了证实。

结论

CCD 是一种罕见的先天性疾病,需要高度的临床怀疑,通常需要基因检测来确诊。在这里,我们报告了一例患者,由于几个误导因素,直到成年早期才被误诊为巴特综合征。我们希望通过报告这个病例,能够提高高患病率地区对 CCD 的认识,并认识到早期诊断对于预防严重并发症的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ab6/9272577/5308bae599d2/amjcaserep-23-e936715-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ab6/9272577/5308bae599d2/amjcaserep-23-e936715-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7ab6/9272577/5308bae599d2/amjcaserep-23-e936715-g001.jpg

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Clin Transl Gastroenterol. 2021 Nov 18;12(11):e00427. doi: 10.14309/ctg.0000000000000427.
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Inflammatory Bowel Disease in Patients with Congenital Chloride Diarrhoea.先天性氯性腹泻患者的炎症性肠病。
J Crohns Colitis. 2021 Oct 7;15(10):1679-1685. doi: 10.1093/ecco-jcc/jjab056.
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Congenital chloride diarrhea clinical features and management: a systematic review.先天性氯性腹泻的临床特征与管理:系统综述。
Pediatr Res. 2021 Jul;90(1):23-29. doi: 10.1038/s41390-020-01251-2. Epub 2020 Nov 10.
4
Development of Crohn's Disease in a Child With -related Congenital Chloride Diarrhea: Report of the First Case in East Asia and a Novel Missense Variant.一名患有先天性氯腹泻相关疾病的儿童患克罗恩病:东亚首例病例报告及一种新型错义变体
Ann Lab Med. 2021 Mar 1;41(2):255-257. doi: 10.3343/alm.2021.41.2.255.
5
A novel de novo SLC26A3 mutation causing congenital chloride diarrhea in a Japanese neonate.一个导致日本新生儿先天性氯腹泻的 SLC26A3 基因新的从头突变。
Mol Genet Genomic Med. 2020 Nov;8(11):e1505. doi: 10.1002/mgg3.1505. Epub 2020 Sep 20.
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Medicine (Baltimore). 2019 May;98(22):e15928. doi: 10.1097/MD.0000000000015928.
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