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腭部复杂婴幼儿血管瘤:1例新生儿患者病例报告及文献复习

Complicated Infantile Hemangiomas in the palate: case report of a newborn patient and review of the literature.

作者信息

Pensabene Marco, Cambiaso Chiara, Patti Maria, Sergio Maria, Serra Gregorio, Grasso Francesco, Baldanza Fabio, Giuffre' Mario, Corsello Giovanni, Di Pace Maria Rita

机构信息

Pediatric Surgery Unit, Department of Health Promotion, Mother and Child Care, Internal Medicine and Medical Specialties "G. D'Alessandro", University of Palermo, Palermo, Italy.

Neonatal Intensive Care Unit, Department of Health Promotion, Mother and Child Care, Internal Medicine and Medical Specialties "G. D'Alessandro", University of Palermo, Palermo, Italy.

出版信息

Ital J Pediatr. 2025 Jun 5;51(1):171. doi: 10.1186/s13052-025-02030-7.

DOI:10.1186/s13052-025-02030-7
PMID:40474189
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12142874/
Abstract

BACKGROUND

Infantile hemangiomas (IH) are the most common benign vascular tumors in neonates, with a prevalence of 5-10% in full-term infants. Its occurrence in the palate is rare. While typically self-limiting, complicated IHs can lead to significant morbidity, especially when involving critical structures.

CASE PRESENTATION

We report the case of a full-term newborn presenting with a 2×2 cm exophytic lesion on the hard palate, causing feeding difficulties, anemia, and recurrent bleeding. Early administration of oral propranolol, initiated on the 11th day of life, proved effective in managing this complicated IH. The treatment, started at a dose of 0.5 mg/kg/day and gradually increased to 3 mg/kg/day, resulted in rapid ulceration healing and lesion regression. Follow-up over eight months confirmed the therapy's efficacy and safety, with no adverse effects reported. To contextualize this case, a systematic review of the literature was conducted following PRISMA guidelines, focusing on neonatal IHs of the head and neck treated with beta-blockers. Out of 1052 papers identified between 2015 and 2024, only four relevant studies were included. These cases highlighted propranolol's role as a first-line treatment for complicated IHs, even in neonates. However, the review also emphasized diagnostic challenges, particularly in atypical locations such as the hard palate, which may delay therapy.

CONCLUSIONS

This case and literature review underscore the importance of early diagnosis and a multidisciplinary approach for managing high-risk IHs. While propranolol is generally avoided in neonates younger than 45 weeks of corrected gestational age due to potential adverse effects, our findings suggest that close monitoring in a hospital setting enables its safe administration. This report contributes to the growing evidence supporting propranolol as a safe and effective therapy for neonatal IHs and highlights the need for further research to refine treatment protocols for this vulnerable population.

摘要

背景

婴儿血管瘤(IH)是新生儿中最常见的良性血管肿瘤,足月婴儿的患病率为5%-10%。其发生于腭部较为罕见。虽然通常具有自限性,但复杂的婴儿血管瘤可导致严重的发病率,尤其是当累及关键结构时。

病例报告

我们报告一例足月新生儿病例,其硬腭出现一个2×2 cm的外生性病变,导致喂养困难、贫血和反复出血。出生后第11天开始早期口服普萘洛尔,证明对治疗这种复杂的婴儿血管瘤有效。治疗开始时剂量为0.5 mg/kg/天,并逐渐增加至3 mg/kg/天,导致溃疡迅速愈合和病变消退。八个月的随访证实了该治疗方法的有效性和安全性,未报告不良反应。为了结合该病例进行分析,按照PRISMA指南对文献进行了系统回顾,重点关注接受β受体阻滞剂治疗的头颈部新生儿婴儿血管瘤。在2015年至2024年间确定的1052篇论文中,仅纳入了四项相关研究。这些病例突出了普萘洛尔作为复杂婴儿血管瘤一线治疗药物的作用,即使在新生儿中也是如此。然而,该综述也强调了诊断方面的挑战,特别是在硬腭等非典型部位,这可能会延迟治疗。

结论

该病例及文献综述强调了早期诊断和多学科方法管理高危婴儿血管瘤的重要性。虽然由于潜在的不良反应,通常避免对矫正胎龄小于45周的新生儿使用普萘洛尔,但我们的研究结果表明,在医院环境中密切监测可使其安全给药。本报告为支持普萘洛尔作为新生儿婴儿血管瘤安全有效治疗方法的越来越多的证据做出了贡献,并强调需要进一步研究以完善针对这一脆弱人群的治疗方案。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f483/12142874/1503c7a1a310/13052_2025_2030_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f483/12142874/36ede9d511f8/13052_2025_2030_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f483/12142874/c714dbe7999d/13052_2025_2030_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f483/12142874/1503c7a1a310/13052_2025_2030_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f483/12142874/36ede9d511f8/13052_2025_2030_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f483/12142874/c714dbe7999d/13052_2025_2030_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f483/12142874/1503c7a1a310/13052_2025_2030_Fig3_HTML.jpg

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本文引用的文献

1
Management of Orofacial Infantile Haemangioma: A Case Report.婴幼儿口腔颌面部血管瘤的治疗:病例报告
Case Rep Dent. 2024 Dec 18;2024:4988221. doi: 10.1155/crid/4988221. eCollection 2024.
2
Efficiency of Topical Beta-Blockers for Epistaxis Control in Ulcerated Infantile Hemangioma.外用β受体阻滞剂控制溃疡性婴儿血管瘤鼻出血的疗效
Cureus. 2024 Aug 25;16(8):e67709. doi: 10.7759/cureus.67709. eCollection 2024 Aug.
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A 1-Day-Old Girl With Infantile Hemangioma and Sternal Cleft.1 天女婴,患婴儿血管瘤,胸骨裂。
Chest. 2024 May;165(5):e137-e142. doi: 10.1016/j.chest.2023.12.010.
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Congenital hypopituitarism and multiple midline defects in a newborn with non-familial Cat Eye syndrome.新生儿患非家族性猫眼综合征,表现为先天性垂体功能减退和中线多处缺陷。
Ital J Pediatr. 2022 Sep 8;48(1):170. doi: 10.1186/s13052-022-01365-9.
5
Novel mutations of the ABCA12, KRT1 and ST14 genes in three unrelated newborns showing congenital ichthyosis.三个无关联的先天性鱼鳞病患儿中 ABCA12、KRT1 和 ST14 基因的新型突变。
Ital J Pediatr. 2022 Aug 13;48(1):145. doi: 10.1186/s13052-022-01336-0.
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Quality of life improving after propranolol treatment in patients with Infantile Hemangiomas.普萘洛尔治疗婴幼儿血管瘤患者后生活质量提高。
Ital J Pediatr. 2022 Aug 4;48(1):140. doi: 10.1186/s13052-022-01334-2.
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Interstitial deletions of chromosome 1p: novel 1p31.3p22.2 microdeletion in a newborn with craniosynostosis, coloboma and cleft palate, and review of the genomic and phenotypic profiles.1p 号染色体间区缺失:1 例颅缝早闭、眼眶裂和腭裂新生儿的 1p31.3p22.2 微缺失,并复习相关基因组和表型特征
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Hypertrophic pyloric stenosis masked by kidney failure in a male infant with a contiguous gene deletion syndrome at Xp22.31 involving the steroid sulfatase gene: case report.男性婴儿肥厚性幽门狭窄被肾衰竭掩盖,该婴儿患有 Xp22.31 连续基因缺失综合征,涉及甾体硫酸酯酶基因:病例报告。
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Neuroaxial Infantile Hemangiomas: Imaging Manifestations and Association with Hemangioma Syndromes.神经轴索婴儿血管瘤:影像学表现及与血管瘤综合征的关系。
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