Pseudomyogenic hemangioendothelioma of bone treated with denosumab: A case report

Shinya Otani, Turrent Robert Nakayama, Tetsuya Sekita, Toru Hirozane, Naofumi Asano, Kazumasa Nishimoto, Aya Sasaki, Hajime Okita, Hideo Morioka, Masaya Nakamura, Morio Matsumoto

研究成果: Article

抄録

Background: Pseudomyogenic hemangioendothelioma (PMHE) is a rare endothelial neoplasm that involves the bones in only 14% of all cases. The optimal treatment strategy has not been established. We herein report a case of primary PMHE in which denosumab treatment showed activity in both imaging studies and the clinical outcome. Case presentation: A 20-year-old woman presented with worsening pain in her left ankle. Imaging studies showed multifocal fluorodeoxyglucose (FDG)-avid [maximum standardized uptake value (SUVmax), 15.95] osteolytic lesions in the bones of her left lower extremity. While waiting for the definitive pathologic diagnosis of PMHE, denosumab, a human immunoglobulin G2 monoclonal antibody against RANKL, was initiated to treat progressive bone absorption after curettage of one of the lesions. Denosumab induced osteosclerosis around the lesions and pain relief and was discontinued 4 years after its initiation. Although all of the multifocal lesions remained, they all became less FDG-avid (SUVmax, 2.6), and the patient developed no signs of new lesions or distant metastasis. Conclusion: Denosumab plays a certain role in prevention of bone destruction by PMHE through suppression of osteoclast-like giant cells and would be an excellent treatment for bone absorption by PMHE of bone.

元の言語English
記事番号872
ジャーナルBMC Cancer
19
発行部数1
DOI
出版物ステータスPublished - 2019 9 3

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Hemangioendothelioma
Bone and Bones
Bones of Lower Extremity
Osteosclerosis
Pain
Bone Neoplasms
Curettage
Osteoclasts
Giant Cells
Ankle
Immunoglobulins
Therapeutics
Monoclonal Antibodies
Outcome Assessment (Health Care)
Denosumab
Neoplasm Metastasis

ASJC Scopus subject areas

  • Oncology
  • Genetics
  • Cancer Research

これを引用

Pseudomyogenic hemangioendothelioma of bone treated with denosumab : A case report. / Otani, Shinya; Nakayama, Turrent Robert; Sekita, Tetsuya; Hirozane, Toru; Asano, Naofumi; Nishimoto, Kazumasa; Sasaki, Aya; Okita, Hajime; Morioka, Hideo; Nakamura, Masaya; Matsumoto, Morio.

:: BMC Cancer, 巻 19, 番号 1, 872, 03.09.2019.

研究成果: Article

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AU - Nishimoto, Kazumasa

AU - Sasaki, Aya

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AB - Background: Pseudomyogenic hemangioendothelioma (PMHE) is a rare endothelial neoplasm that involves the bones in only 14% of all cases. The optimal treatment strategy has not been established. We herein report a case of primary PMHE in which denosumab treatment showed activity in both imaging studies and the clinical outcome. Case presentation: A 20-year-old woman presented with worsening pain in her left ankle. Imaging studies showed multifocal fluorodeoxyglucose (FDG)-avid [maximum standardized uptake value (SUVmax), 15.95] osteolytic lesions in the bones of her left lower extremity. While waiting for the definitive pathologic diagnosis of PMHE, denosumab, a human immunoglobulin G2 monoclonal antibody against RANKL, was initiated to treat progressive bone absorption after curettage of one of the lesions. Denosumab induced osteosclerosis around the lesions and pain relief and was discontinued 4 years after its initiation. Although all of the multifocal lesions remained, they all became less FDG-avid (SUVmax, 2.6), and the patient developed no signs of new lesions or distant metastasis. Conclusion: Denosumab plays a certain role in prevention of bone destruction by PMHE through suppression of osteoclast-like giant cells and would be an excellent treatment for bone absorption by PMHE of bone.

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