Analyzing the landscape of coeliac crisis in adult and paediatric populations: A systematic review and meta-analysis

IF 3.8 3区医学 Q1 GASTROENTEROLOGY & HEPATOLOGY Digestive and Liver Disease Pub Date : 2025-06-01 Epub Date: 2025-03-17 DOI:10.1016/j.dld.2025.02.011

Arkadeep Dhali , Rick Maity , Hareesha Rishab Bharadwaj , Syed Hasham Ali , Muhammad Hamza Shah , David Surendran Sanders

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Abstract

Background

Coeliac crisis (CC) is a rare, potentially life-threatening manifestation of coeliac disease (CD).

Aims

To comprehensively explore the clinical aspects, management strategies, and outcomes related to CC in both adult and paediatric populations.

Methods

We conducted a comprehensive literature search to identify studies eligible for inclusion up to 28 December 2024. The study protocol was registered in PROSPERO (CRD42024510682).

Results

A total of 46 included articles and 195 patients were analyzed. The median age of presentation was 18 years (range: 8 months to 83 years), predominantly affecting the paediatric population. An overwhelming majority (180 patients, 92.8 %) had CC as their first presentation, whereas the others were previously diagnosed CD cases with poor adherence to gluten-free diet (GFD). Clinically, patients presented with diarrhoea (88.2 %), abdominal pain (72.8 %), vomiting (56.4 %), confusion and lethargy (11.3 %). Electrolyte derangements in the form of hypokalemia (89.7 %) and hyponatremia (64.1 %), along with metabolic acidosis (79.5 %) and hypoalbuminemia (76.9 %), reflected the severity of malabsorption. Treatment with GFD caused clinical improvement in 186 (95.4 %) patients and mostly favourable long-term outcomes.

Conclusion

CC is a poorly understood, life-threatening complication of CD. Given its similarity with infectious conditions, clinicians require a high index of suspicion for recognizing CC to diagnose early and provide proper management.

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分析成人和儿科人群乳糜泻危象的情况：一项系统回顾和荟萃分析。

背景：乳糜泻危象（Coeliac crisis， CC）是一种罕见的、可能危及生命的乳糜泻表现。目的：全面探讨成人和儿科人群中与CC相关的临床方面、管理策略和结果。方法：我们进行了全面的文献检索，以确定2024年12月28日之前有资格纳入的研究。研究方案已在PROSPERO注册（CRD42024510682）。结果：共纳入文献46篇，分析患者195例。中位发病年龄为18岁（范围：8个月至83岁），主要影响儿科人群。绝大多数患者（180例，92.8%）首次表现为乳糜泻，而其他患者先前诊断为乳糜泻，且无谷蛋白饮食（GFD）依从性差。临床表现为腹泻（88.2%）、腹痛（72.8%）、呕吐（56.4%）、精神错乱和嗜睡（11.3%）。电解质紊乱表现为低钾血症（89.7%）和低钠血症（64.1%），以及代谢性酸中毒（79.5%）和低白蛋白血症（76.9%），反映了吸收不良的严重程度。用GFD治疗186例（95.4%）患者的临床改善，大多数患者的长期预后良好。结论：CC是一种鲜为人知的危及生命的CD并发症。鉴于其与感染性疾病的相似性，临床医生需要高度的怀疑指数来识别CC，以便早期诊断并提供适当的治疗。

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来源期刊

Digestive and Liver Disease 医学-胃肠肝病学

CiteScore

6.10

自引率

2.20%

发文量

632

审稿时长

19 days

期刊介绍： Digestive and Liver Disease is an international journal of Gastroenterology and Hepatology. It is the official journal of Italian Association for the Study of the Liver (AISF); Italian Association for the Study of the Pancreas (AISP); Italian Association for Digestive Endoscopy (SIED); Italian Association for Hospital Gastroenterologists and Digestive Endoscopists (AIGO); Italian Society of Gastroenterology (SIGE); Italian Society of Pediatric Gastroenterology and Hepatology (SIGENP) and Italian Group for the Study of Inflammatory Bowel Disease (IG-IBD). Digestive and Liver Disease publishes papers on basic and clinical research in the field of gastroenterology and hepatology. Contributions consist of: Original Papers Correspondence to the Editor Editorials, Reviews and Special Articles Progress Reports Image of the Month Congress Proceedings Symposia and Mini-symposia.