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乳糜泻患儿的肠套叠

Intussusception in children with celiac disease.

作者信息

Alhussaini Bakr H

机构信息

Pediatric Department, Jeddah, Saudi Arabia.

Gastroenterology, Hepatology, and Clinical Nutrition Unit, Jeddah, Saudi Arabia.

出版信息

J Family Med Prim Care. 2024 Nov;13(11):5385-5388. doi: 10.4103/jfmpc.jfmpc_719_24. Epub 2024 Nov 18.

DOI:10.4103/jfmpc.jfmpc_719_24
PMID:39722969
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11668390/
Abstract

Celiac disease (CD) is a chronic illness. Blood testing for tissue transglutaminase antibodies is the initial screening test for the diagnosis of CD, and upper gastrointestinal endoscopy and duodenal/jejunal biopsy are used to confirm CD. Intussusception (IS) is the process in which a proximal segment of the bowel invaginates through the lumen of a distal segment. The association between pediatric IS and CD has been described but is still not widely recognized. Herein, we report a case of IS as the first manifestation of CD in a child. A 3-year-old girl presented to the emergency department with a 1-week history of marked abdominal distention and lethargy, but there was no history of fever, bleeding per rectum, or jaundice. A second-degree relative had a family history of CD. Clinical examination: The patient was a lethargic child with pale conjunctiva and bilateral lower limb edema. She was a febrile and had a normal hemodynamic status. The adipose tissue was diminished throughout the patient's body; her weight was 8 kg (<5% weight percentile for girls), and her height was 81 cm (<5% height percentile for girls). Laboratory results included the following: Hb of 9 g/dL, serum ferritin of 10 ng/mL (30-400 ng/mL), normal liver function test results except for hypoalbuminemia at 21 g/L (35-52 g/L), and low blood cholesterol of 0.94 g/L (1.54-2.01 g/L). The patient's blood sugar level was 98 mg/dL, and her renal function test results were normal, with negative septic screening. Abdominal radiography revealed several air-fluid levels, suggestive of an obstruction in the small bowel. Abdominal ultrasonography revealed typical features confirming the diagnosis of IS. Abdominal computed tomography demonstrated an enteroenteric IS with no other signs of organic causes, such as lymphoma or other tumors. Based on the high index of suspicion of CD, a workup confirmed the diagnosis. A gluten-free diet [A1] was started during the hospital course, and the patient improved dramatically regarding her symptoms and was discharged home. In conclusion, this case highlights the association between IS and CD. Intussusception is an emergency condition and usually idiopathic. However, in atypical or recurrent typical presentations underlying causes, we should do proper investigations to initiate appropriate management. [A1]Abbreviations are generally avoided for terms that are not repeated in the text. In this case, this abbreviation is not used elsewhere in the Abstract. Hence, I have deleted the abbreviation. Categories: Pediatrics, Gastroenterology.

摘要

乳糜泻(CD)是一种慢性疾病。检测组织转谷氨酰胺酶抗体的血液检测是诊断CD的初始筛查试验,上消化道内镜检查和十二指肠/空肠活检用于确诊CD。肠套叠(IS)是一段近端肠管通过远端肠管腔套入的过程。小儿IS与CD之间的关联已有描述,但仍未得到广泛认可。在此,我们报告一例以IS为首发表现的小儿CD病例。一名3岁女孩因腹胀和嗜睡1周就诊于急诊科,无发热、直肠出血或黄疸病史。其二级亲属有CD家族史。临床检查:患儿嗜睡,结膜苍白,双下肢水肿。无发热,血流动力学状态正常。全身脂肪组织减少;体重8kg(低于女孩体重百分位数第5%),身高81cm(低于女孩身高百分位数第5%)。实验室检查结果如下:血红蛋白9g/dL,血清铁蛋白10ng/mL(30 - 400ng/mL),肝功能检查结果除白蛋白水平低至21g/L(35 - 52g/L)外均正常,血胆固醇低至0.94g/L(1.54 - 2.01g/L)。血糖水平98mg/dL,肾功能检查结果正常,脓毒症筛查阴性。腹部X线检查显示多个气液平面,提示小肠梗阻。腹部超声检查显示典型特征,确诊为IS。腹部计算机断层扫描显示肠-肠型IS,无淋巴瘤或其他肿瘤等其他器质性病因的迹象。基于对CD的高度怀疑,进一步检查确诊。住院期间开始无麸质饮食,患儿症状明显改善后出院。总之,该病例突出了IS与CD之间的关联。肠套叠是一种紧急情况,通常为特发性。然而,对于非典型或复发性典型表现,我们应进行适当检查以启动恰当治疗。分类:儿科学、胃肠病学

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/46b5/11668390/9bcb2480eb59/JFMPC-13-5385-g003.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/46b5/11668390/bed69a391307/JFMPC-13-5385-g001.jpg
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https://cdn.ncbi.nlm.nih.gov/pmc/blobs/46b5/11668390/9bcb2480eb59/JFMPC-13-5385-g003.jpg

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本文引用的文献

1
Celiac disease: a rare cause of 'postoperative' ileoileal intussusception after surgical reduction of ileocolic intussusception in a toddler.乳糜泻:幼儿回结肠套叠手术复位后“术后”回肠-回肠套叠的罕见病因。
J Surg Case Rep. 2020 Jun 1;2020(5):rjaa073. doi: 10.1093/jscr/rjaa073. eCollection 2020 May.
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Adult intussusception : A 14-year retrospective study of clinical assessment and computed tomography diagnosis.成人肠套叠:一项关于临床评估和计算机断层扫描诊断的14年回顾性研究。
Acta Gastroenterol Belg. 2018 Jul-Sep;81(3):367-372.
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Mass Screening for Celiac Disease Among School-aged Children: Toward Exploring Celiac Iceberg in Saudi Arabia.
学龄儿童乳糜泻的大规模筛查:探索沙特阿拉伯的乳糜泻冰山现象
J Pediatr Gastroenterol Nutr. 2017 Dec;65(6):646-651. doi: 10.1097/MPG.0000000000001681.
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Sao Paulo Med J. 2016 Sep 19;134(5):0. doi: 10.1590/1516-3180.2016.0092220616.
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Childhood intussusception: 17-year experience at a tertiary referral centre in Hong Kong.儿童肠套叠:香港一家三级转诊中心的17年经验
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Association between Celiac Disease and Intussusceptions in Children: Two Case Reports and Literature Review.儿童乳糜泻与肠套叠之间的关联:两例病例报告及文献综述
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A large nationwide population-based case-control study of the association between intussusception and later celiac disease.一项基于全国范围人群的大型病例对照研究,旨在探讨肠套叠与后来的乳糜泻之间的关联。
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Clinical practice. Celiac disease.临床实践。乳糜泻。
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Should intussusception in children prompt screening for celiac disease?儿童肠套叠是否需要筛查乳糜泻?
J Pediatr Gastroenterol Nutr. 2013 Jan;56(1):56-9. doi: 10.1097/MPG.0b013e31826a1099.
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