Literature DB >> 30044355

Sirolimus Treatment of an Infant With Intrathoracic Kaposiform Hemangioendothelioma Complicated by Life-threatening Pleural and Pericardial Effusions.

Lucy Duan1, Samuele Renzi2, Danielle Weidman2, Nicolas Waespe2,3, Rose Chami4, David Manson5, Michaela Cada2, Manuel Carcao2.   

Abstract

Kaposiform hemangioendothelioma (KHE) is a rare infiltrative vascular tumor that may be associated with Kasabach-Merritt Phenomenon (KMP), which is a consumptive coagulopathy with potentially life-threatening thrombocytopenia. Management of KHE and KMP is challenging, and currently, there are no standardized validated treatment protocols. Mammalian target of rapamycin inhibitors have been shown to be effective in the treatment of KHE. We describe a term male who presented as a diagnostic dilemma with life-threatening pleural and pericardial effusions and severe thrombocytopenia. After extensive work-up the etiology for his condition was determined to be KHE with KMP. The patient was commenced on sirolimus and responded well to therapy with resolution of KMP.

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Year:  2020        PMID: 30044355     DOI: 10.1097/MPH.0000000000001268

Source DB:  PubMed          Journal:  J Pediatr Hematol Oncol        ISSN: 1077-4114            Impact factor:   1.289


  6 in total

1.  Kaposiform lymphangiomatosis and kaposiform hemangioendothelioma: similarities and differences.

Authors:  Yi Ji; Siyuan Chen; Suhua Peng; Chunchao Xia; Li Li
Journal:  Orphanet J Rare Dis       Date:  2019-07-05       Impact factor: 4.123

2.  Effective surgical treatment of life-threatening huge vascular anomalies associated with thrombocytopenia and coagulopathy in infants unresponsive to drug therapy.

Authors:  Yaohao Wu; Ronglin Qiu; Lexiang Zeng; Liyang Liang; Jie Zhang; Jiajia Zhou; Wenli Jiang; Jianhang Su; Xiaogeng Deng
Journal:  BMC Pediatr       Date:  2020-04-27       Impact factor: 2.125

Review 3.  Kaposiform hemangioendothelioma: current knowledge and future perspectives.

Authors:  Yi Ji; Siyuan Chen; Kaiying Yang; Chunchao Xia; Li Li
Journal:  Orphanet J Rare Dis       Date:  2020-02-03       Impact factor: 4.123

4.  Case report: Experience of a rare case of rebound of the Kasabach-Merritt phenomenon during sirolimus treatment in kaposiform hemangioendothelioma.

Authors:  Liang Wang; Jing Li; Changhua Wu; Dan Song; Zhuang Liu; Yanli Niu; Jie Zhou; Lei Guo
Journal:  Front Pediatr       Date:  2022-08-05       Impact factor: 3.569

5.  Pregnancy-related Kasabach-Merritt phenomenon with pleural effusion: A case report and literature review.

Authors:  Xiaodan Jiao; Shiwei Kang; Yanping Zhang; Yadong Yuan; Yuan Wang
Journal:  Indian J Dermatol       Date:  2022 Mar-Apr       Impact factor: 1.757

Review 6.  Vascular tumors of the mediastinum.

Authors:  Kristen Paral; Thomas Krausz
Journal:  Mediastinum       Date:  2020-09-30
  6 in total

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