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口服普萘洛尔成功治疗弥漫性婴儿肝血管瘤:病例报告及文献综述

Diffuse infantile hepatic hemangioma successfully treated with propranolol orally: a case report and literature review.

作者信息

Li Zengyan, Wu Zhiming, Dong Youhong, Yuan Xiaojun, Zhang Dongdong

机构信息

Department of Oncology, Xiangyang No. 1 People's Hospital, Hubei University of Medicine, Xiangyang, China.

Department of Orthopedics, Xiangyang No. 1 People's Hospital, Hubei University of Medicine, Xiangyang, China.

出版信息

Front Oncol. 2024 Jan 29;14:1336742. doi: 10.3389/fonc.2024.1336742. eCollection 2024.

Abstract

BACKGROUND

Infantile hepatic hemangioma (IHH) is a common vascular, fast-growing hepatic tumor that is usually accompanied by multiple cutaneous hemangiomas. Diffuse IHH (DIHH) is a rare type of IHH that exhibits many tumors with nearly complete hepatic parenchymal replacement. At present, there is no specific standardized treatment plan for DIHH. Herein, we present the case of a 2-month-old girl with DIHH and without cutaneous hemangioma who achieved complete remission after undergoing propranolol monotherapy.

CASE PRESENTATION

The infant with low birth weight was presented to the pediatric department with a 2-month history of persistent vomiting and feeding difficulty. Ultrasonography and abdominal magnetic resonance imaging revealed hepatomegaly and diffused intrahepatic lesions. A computed tomography-guided percutaneous liver biopsy was performed, and the pathological examination suggested the diagnosis was DIHH. The patient exhibited remarkably response to an increasing dose of oral propranolol, from 0.5 mg/kg to 2 mg/kg every day. The intrahepatic lesions were almost completely regressed after one year of treatment and no distinct adverse reaction was observed.

CONCLUSION

DIHH can induce life-threatening complications that require prompt interventions. Propranolol monotherapy can be an effective and safe first-line treatment strategy for DIHH.

摘要

背景

婴儿肝血管瘤(IHH)是一种常见的、生长迅速的肝脏血管肿瘤,通常伴有多发皮肤血管瘤。弥漫性婴儿肝血管瘤(DIHH)是IHH的一种罕见类型,表现为多个肿瘤几乎完全替代肝实质。目前,对于DIHH尚无特定的标准化治疗方案。在此,我们报告一例2个月大的DIHH女童病例,该患儿无皮肤血管瘤,接受普萘洛尔单药治疗后完全缓解。

病例介绍

该低体重婴儿因持续呕吐和喂养困难2个月就诊于儿科。超声检查和腹部磁共振成像显示肝脏肿大和肝内弥漫性病变。进行了计算机断层扫描引导下的经皮肝活检,病理检查提示诊断为DIHH。患者对口服普萘洛尔剂量从每天0.5mg/kg增加至2mg/kg表现出显著反应。治疗1年后,肝内病变几乎完全消退,未观察到明显不良反应。

结论

DIHH可引发危及生命的并发症,需要及时干预。普萘洛尔单药治疗可为DIHH提供一种有效且安全的一线治疗策略。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f5b9/10859491/6d70176e9e61/fonc-14-1336742-g001.jpg

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