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产前暴露于帕博利珠单抗作为复发性坏死性小肠结肠炎、新生儿糖尿病和外分泌性胰腺功能不全之间的潜在联系:一例报告

Prenatal pembrolizumab exposure as the potential link among recurrent necrotizing enterocolitis, neonatal diabetes mellitus and exocrine pancreatic insufficiency: a case report.

作者信息

Castaldo Pasquale, Altimari Luigi, Capretti Maria Grazia, Maltoni Giulio, Scozzarella Andrea, Paoletti Vittoria, Corvaglia Luigi, Aceti Arianna

机构信息

School of Specialization in Pediatrics, Department of Medical and Surgical Sciences, University of Bologna, Bologna, Italy.

Neonatal Intensive Care Unit, IRCCS Azienda Ospedaliero-Universitaria di Bologna, Via Massarenti 11, Bologna, 40138, Italy.

出版信息

Ital J Pediatr. 2025 Jul 18;51(1):237. doi: 10.1186/s13052-025-02021-8.

Abstract

BACKGROUND

Necrotizing enterocolitis (NEC) is a serious complication predominantly affecting very low birth weight neonates, characterized by high mortality and long-term morbidity. Neonatal diabetes mellitus (NDM) is a rare condition with diverse etiologies, including genetic predisposition and autoimmune mechanisms. To our knowledge, NEC and NDM have never been previously reported together.

CASE PRESENTATION

We report the clinical case of a late preterm infant whose mother had received an immune checkpoint inhibitor treatment (pembrolizumab) for metastatic gastric cancer during pregnancy. The infant suffered from severe and recurrent necrotizing enterocolitis (NEC), neonatal diabetes mellitus (NDM) and exocrine pancreatic insufficiency that required a multidisciplinary approach. Prenatal exposure to pembrolizumab might explain the co-occurrence of the three diseases and the complex and unusual medical history of the child, even if the possibility that these events were independent of each other cannot be totally ruled out.

CONCLUSION

This case suggests the potential association between antenatal pembrolizumab exposure and severe neonatal comorbidities. Furthermore, it highlights the challenge of managing concurrent NEC, NDM and exocrine pancreatic insufficiency in early infancy, emphasizing the need for multidisciplinary care and long-term follow-up to address complex clinical and developmental outcomes.

摘要

背景

坏死性小肠结肠炎(NEC)是一种主要影响极低出生体重儿的严重并发症,其特征为高死亡率和长期发病。新生儿糖尿病(NDM)是一种病因多样的罕见病症,包括遗传易感性和自身免疫机制。据我们所知,此前从未有过NEC和NDM同时出现的报道。

病例报告

我们报告了一例晚期早产儿的临床病例,其母亲在孕期因转移性胃癌接受了免疫检查点抑制剂治疗(帕博利珠单抗)。该婴儿患有严重且复发性坏死性小肠结肠炎(NEC)、新生儿糖尿病(NDM)和外分泌胰腺功能不全,需要多学科方法进行治疗。产前接触帕博利珠单抗可能解释了这三种疾病的同时出现以及该患儿复杂且不寻常的病史,尽管不能完全排除这些事件相互独立的可能性。

结论

本病例提示产前接触帕博利珠单抗与严重新生儿合并症之间可能存在关联。此外,它凸显了在婴儿早期管理同时存在的NEC、NDM和外分泌胰腺功能不全的挑战,强调了多学科护理和长期随访以应对复杂临床和发育结局的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8f51/12275445/fd3563bc3c96/13052_2025_2021_Fig1_HTML.jpg

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