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

共 50 条

[1] Multifocal lymphangioendotheliomatosis with thrombocytopenia
Petrone, Emma
Santini, Gennaro
Correra, Antonio
Lista, Rita
MINERVA PEDIATRICS, 2022, 74 (04): : 482 - 484
[2] Multifocal lymphangioendotheliomatosis with thrombocytopenia
Aljefri, K.
Charlton, F.
Leech, S.
Taylor, A. E. M.
BRITISH JOURNAL OF DERMATOLOGY, 2014, 171 : 120 - 121
[3] Newborn with multifocal lymphangioendotheliomatosis with thrombocytopenia
Soledad Zegpi, Maria
Zavala, Alejandro
del Puerto, Constanza
Cardenas, Consuelo
Gonzalez, Sergio
INDIAN JOURNAL OF DERMATOLOGY VENEREOLOGY & LEPROLOGY, 2012, 78 (03):
[4] Expanding the Phenotype of Multifocal Lymphangioendotheliomatosis With Thrombocytopenia
Maronn, Mandi
Catrine, Kristina
North, Paula
Browning, Meghen B.
Kerschner, Joseph E.
Noel, Richard
Drolet, Beth A.
Kelly, Michael
PEDIATRIC BLOOD & CANCER, 2009, 52 (04) : 531 - 534
[5] Bevacizumab Treatment in Multifocal Lymphangioendotheliomatosis With Thrombocytopenia
Kline, Ronald M.
Buck, Laura Marcu
PEDIATRIC BLOOD & CANCER, 2009, 52 (04) : 534 - 536
[6] MULTIFOCAL LYMPHANGIOENDOTHELIOMATOSIS WITH THROMBOCYTOPENIA (MLT) WITH PULMONARY HEMORRHAGE
Galletta, Thomas
Wurth, Mark
Young, Lisa
Adams, Denise
Friedman, Debra
PEDIATRIC BLOOD & CANCER, 2019, 66
[7] Orthotopic liver transplant for multifocal lymphangioendotheliomatosis with thrombocytopenia
Yang, Christine H.
Zhou, Shengmei
Alexopoulos, Sophoclis
Wang, Larry
Baron, Howard I.
Genyk, Yuri
Kerkar, Nanda
PEDIATRIC TRANSPLANTATION, 2016, 20 (03) : 456 - 459
[8] Multifocal Congenital Lymphangioendotheliomatosis Without Gastrointestinal Bleeding and/or Thrombocytopenia
Khamaysi, Ziad
Bergman, Reuven
AMERICAN JOURNAL OF DERMATOPATHOLOGY, 2010, 32 (08) : 804 - 808
[9] Multifocal lymphangioendotheliomatosis with thrombocytopenia - A newly recognized clinicopathological entity
North, PE
Kahn, T
Cordisco, MR
Dadras, SS
Detmar, M
Frieden, IJ
ARCHIVES OF DERMATOLOGY, 2004, 140 (05) : 599 - 606
[10] Successful Treatment With Everolimus for Multifocal Lymphangioendotheliomatosis With Thrombocytopenia in an Infant
Hoshino, Yusuke
Arai, Junichi
Kato, Keisuke
Tagawa, Manabu
JOURNAL OF PEDIATRIC HEMATOLOGY ONCOLOGY, 2023, 45 (02) : 95 - 98

← 1 2 3 4 5 →