Online ISSN: 2187-2988 Print ISSN: 0911-1794
特定非営利活動法人日本小児循環器学会 Japanese Society of Pediatric Cardiology and Cardiac Surgery
Pediatric Cardiology and Cardiac Surgery 37(3): 239-245 (2021)
doi:10.9794/jspccs.37.239

症例報告Case Report

完全大血管転位症に合併した新生児壊死性腸炎後に新生児・乳児食物蛋白誘発胃腸症を発症した1例A Case of Transposition of the Great Arteries that Led to the Development of Non-IgE-Mediated Gastrointestinal Food Allergy after a Neonatal Necrotizing Enterocolitis Complication

順天堂大学医学部小児科Departments of Pediatrics, Juntendo University Faculty of Medicine ◇ Tokyo, Japan

受付日:2021年1月15日Received: January 15, 2021
受理日:2021年4月30日Accepted: April 30, 2021
発行日:2021年11月1日Published: November 1, 2021
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新生児壊死性腸炎(neonatal necrotizing enterocolitis: NEC)は新生児期から管理を要する先天性心疾患(congenital heart disease: CHD)児において重要な合併症の一つである.一方,新生児・乳児食物蛋白誘発胃腸症は嘔吐・下痢・血便・腹部膨満などの消化器症状を呈することが多いが,稀に消化管穿孔やNECなどを合併する重症例も散見する.また,CHDと新生児・乳児蛋白誘発胃腸症の関連についても報告され,特に腸管血流が低下する血行動態の先天性心疾患では新生児・乳児蛋白誘発胃腸症の発症のリスクとされている.症例は完全大血管転位症I型の女児で,経腸管栄養を開始した後に日齢8で血便,ショックを呈し,先天性心疾患に合併したNECと診断した.保存的治療で症状は軽快したが,大血管スイッチ術後に母乳・人工乳を再開した際に消化器症状を認め,血液検査にて末梢血好酸球数増多と牛乳蛋白に対するアレルゲン特異的リンパ球刺激試験(allergen-specific lymphocyte stimulation test: ALST)の陽性,その後に加水分解乳に変更し症状が改善したことから,新生児・乳児食物蛋白誘発胃腸症の関与が考えられた.先天性心疾患例でNECの炎症後,経腸栄養開始に併って消化器症状を認めた際には,新生児・乳児食物蛋白誘発胃腸症の関与も考慮し,診断と治療を進めていくことが重要であると考えられる.

Neonatal necrotizing enterocolitis (NEC) is a serious complication associated with congenital heart disease (CHD). Non-IgE-mediated gastrointestinal food allergy (non-IgE-GI-FA) in neonates and infants manifests as digestive symptoms, including emesis, diarrhea, melena, and abdominal distension, with occasional intestinal perforation and NEC. Previous studies showed the relationship between CHD and non-IgE-GI-FA, which suggests that a decrease in intestinal blood flow can be a risk factor of non-IgE-GI-FA in neonates and infants with CHD. We report the case of a neonate with transposition of the great arteries who developed NEC before an arterial switch operation (ASO) and non-IgE-GI-FA after the operation. The patient presented with bloody stools after the start of formula feeding and was diagnosed with NEC on the basis of abdominal radiography findings. After recovery from NEC, the patient underwent ASO. However, the patient presented with abdominal distension after formula feeding was restarted and was diagnosed with non-IgE-GI-FA on the basis of the findings of eosinophilia and positivity in an allergen-specific lymphocyte stimulation test. This demonstrates that postoperative digestive symptoms impose the possibility of non-IgE-GI-FA in neonates with CHD, especially in those with an NEC complication. Therefore, we infer that if gastrointestinal complications occur in patients with severe CHD, the possibility of non-IgE-GI-FA should be considered.

Key words: transposition of the great artery; non-IgE-mediated gastrointestinal food allergy; neonatal necrotizing enterocolitis; congenital heart disease

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This page was last modified on 2021-12-17T16:14:21.000+09:00


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