The challenging diagnosis of food protein-induced enterocolitis syndrome: A case report series.

The challenging diagnosis of food protein-induced enterocolitis syndrome: A case report series.
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DOI:
10.3389/fped.2022.913278
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发表时间:
2022
影响因子:
2.6
通讯作者:
Gao, Jinzhi
Gao, Jinzhi
中科院分区:
医学3区
文献类型:
--
作者:
Zhao, Caiyan;Chen, Ling;Gao, Jinzhi

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食物蛋白诱导的小肠结肠炎综合征(FPIES)是一种非免疫球蛋白E(IgE)介导的食物过敏。然而,除了呕吐和腹泻,IgE介导的皮肤或呼吸道症状可能是某些FPIES患者的合并症。我们描述了四个不寻常的情况下,新生儿FPIES,其临床表现是可变的和误导。所有患者均出现呕吐、腹泻或其他胃肠道症状,其中3例出现IgE介导的食物过敏。病例1因惊厥入院,随后出现严重脓毒症和坏死性小肠结肠炎(NEC)样表现。例2因皮肤和粘膜严重外渗性皮疹和全身炎症反应被误诊为Stevens-Johnson综合征。病例3有不明原因的胆汁淤积,可能与食物过敏有关。无症状的C反应蛋白升高是病例4中早期FPIES的唯一提示。此外,在上述三例中,血清食物特异性IgG值升高。在排除了引起不适的食物后,4例患者的上述所有临床表现均迅速改善;因此,我们认为所述4例患者最正确的诊断是FPIES。本病例报告系列应进一步引起临床医生对具有可变和非典型症状的FPIES的注意。IgG水平在识别FPIES存在方面的有用性尚不确定。
Food protein-induced enterocolitis syndrome (FPIES) is a type of non-immunoglobulin E (IgE)-mediated food allergy. However, in addition to vomiting and diarrhea, IgE-mediated skin or respiratory symptoms may be comorbidities in some patients with FPIES. We described four unusual cases of neonates with FPIES, whose clinical presentations were variable and misleading. All patients experienced vomiting, diarrhea or other gastrointestinal symptoms, and three of them developed IgE-mediated food allergy. Case 1 was admitted to the hospital with convulsions and then developed severe sepsis and necrotizing enterocolitis (NEC)-like appearance. Case 2 was wrongly diagnosed with Stevens–Johnson syndrome due to a severe extravasation rash of the skin and mucous membranes and a systemic inflammatory response. There was unexplained cholestasis in case 3, which might be attributed to food allergy. Asymptomatic elevation of C-reactive protein was the only hint at early-stage FPIES in case 4. Moreover, there were increased serum food-specific IgG values in three of the above cases. After eliminating the offending food, all of the above clinical manifestations rapidly improved in the four cases; thus, we believe that the most correct diagnosis in the described four cases was FPIES. This case report series should further draw clinicians’ attention to FPIES with variable and atypical symptoms. The usefulness of IgG levels in identifying the presence of FPIES is uncertain.
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