A Case of Multifocal Lymphangioendotheliomatosis With Thrombocytopenia and Changes in Coagulopathy

被引：0

作者：

Nagao, Daiki ^{[1
]}

Ozeki, Michio ^{[1
]}

Nozawa, Akifumi ^{[1
,3
]}

Yasue, Shiho ^{[1
]}

Sasai, Hideo ^{[1
]}

Endo, Saori ^{[1
]}

Kato, Takazumi ^{[2
]}

Hori, Yumiko ^{[4
,5
]}

Ohnishi, Hidenori ^{[1
]}

机构：

[1] Gifu Univ, Grad Sch Med, Dept Pediat, Gifu, Japan

[2] Gifu Univ, Grad Sch Med, Dept Digest Surg & Pediat Surg, Gifu, Japan

[3] Tohoku Univ, Sch Med, Dept Med Genet, Sendai, Miyagi, Japan

[4] Osaka Univ, Grad Sch Med, Dept Pathol, Osaka, Japan

[5] Natl Hosp Org, Osaka Natl Hosp, Dept Cent Lab & Surg Pathol, Osaka, Japan

来源：

JOURNAL OF PEDIATRIC HEMATOLOGY ONCOLOGY | 2023年 / 45卷 / 03期

关键词：

vascular anomaly; thrombocytopenia; coagulopathy; lymphatic anomaly; sirolimus;

D O I：

10.1097/MPH.0000000000002597

中图分类号：

R73 [肿瘤学];

学科分类号：

100214 ;

摘要：

Multifocal lymphangioendotheliomatosis with thrombocytopenia is a rare disease characterized by progressive multiple vascular lesions and is accompanied by thrombocytopenia. The precise diagnosis of this disease is frequently difficult because of the heterogeneity of the clinical symptoms. We report a case of a male infant who presented with severe thrombocytopenia induced by local inflammation. In addition, enlargement of the extremities with soft tissue and bone involvement without gastrointestinal bleeding was observed. The thrombocytopenia resolved after a combination therapy of sirolimus and prednisolone. Our finding that plasma angiopoietin-2 concentrations reflected the disease status suggests its utility as a biomarker of Multifocal lymphangioendotheliomatosis with thrombocytopenia.

引用

页码：E384 / E388

页数：5

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