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成功治疗儿童肝移植患者肝和皮肤的类脂性肉芽肿病。

Successful treatment of malakoplakia of the liver and skin in a pediatric liver transplant patient.

机构信息

Indiana University School of Medicine, Indianapolis, Indiana, USA.

IU Health Pathology Laboratory, Department of Pathology and Laboratory Medicine, Indiana University School of Medicine, Indianapolis, Indiana, USA.

出版信息

Pediatr Transplant. 2023 Aug;27(5):e14492. doi: 10.1111/petr.14492. Epub 2023 Mar 1.

Abstract

BACKGROUND

Malakoplakia occurs uncommonly at any age, but pediatric reports are exceptionally limited. Malakoplakia appears primarily in the urinary tract, although involvement of essentially all organs has been reported, cutaneous malakoplakia is very uncommon and liver involvement is the rarest.

METHOD

We report the first pediatric case of concurrent hepatic and cutaneous malakoplakia in a pediatric liver transplant recipient. We also provide a literature review for cutaneous malakoplakia cases in children.

RESULT

A 16-year-old male received a deceased-donor liver transplant for autoimmune hepatitis, present with the persistence of the liver mass of unknown etiology and cutaneous plaque-like lesions around the surgical scar. Core biopsies taken from the skin and abdominal wall lesions demonstrated histiocytes containing Michaelis-Gutmann bodies (MGB) revealing the diagnosis. The patient successfully was treated with antibiotics alone for 9 months without surgical intervention or a decrease in immunosuppressive therapy.

CONCLUSION

This case demonstrates the need to include malakoplakia in the differential diagnosis of mass-forming lesions after solid transplantation and increase awareness of this very rare entity in pediatrics.

摘要

背景

弹状肿病罕见发生于任何年龄,但儿科报告极为有限。弹状肿病主要发生于泌尿道,虽然几乎所有器官均有受累的报告,但皮肤弹状肿病非常罕见,肝脏受累则是最罕见的。

方法

我们报告首例在儿科肝移植受者中同时发生肝和皮肤弹状肿病的病例。我们还为儿科皮肤弹状肿病病例提供了文献复习。

结果

一名 16 岁男性因自身免疫性肝炎接受了已故供体的肝移植,目前存在不明病因的肝肿块和手术瘢痕周围的斑块状皮肤病变。从皮肤和腹壁病变处取的核心活检显示含 Michaelis-Gutmann 体(MGB)的组织细胞,提示诊断。该患者单独接受抗生素治疗 9 个月,成功治愈,无需手术干预或减少免疫抑制治疗。

结论

该病例表明,在实体器官移植后形成肿块的病变鉴别诊断中需要包括弹状肿病,并提高对儿科中这一非常罕见实体的认识。

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