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Erschienen in: Archives of Osteoporosis 1/2022

01.12.2022 | Review

Pseudomyogenic hemangioendothelioma with bone and soft tissue involvement with favorable response to pamidronate: a case report and systematic review of the literature

verfasst von: María Lorena Brance, Nicolás M. Cóccaro, Pablo Roitman, Alejandro Castiglioni, Florencia Agostinis, Mariel Spense, Bárbara Scheitlin, Nicholas Rene, Lucas R. Brun

Erschienen in: Archives of Osteoporosis | Ausgabe 1/2022

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Abstract

Pseudomyogenic hemangioendothelioma (PMH) can be a challenge for diagnosis and might be confused with other tumors, such as epithelioid sarcoma. Here we present a case and a systematic review of the literature to identify and discuss PMH treatment in primary bone involvement. A 25-year-old woman was referred for bone pain (10/10) in the left lower limb. Magnetic resonance imaging (MRI) showed multiple bone lesions (left femur, tibia, patella, ankle, and foot) with well-defined borders without signs of local aggressiveness. Positron Emission Tomography-Computed Tomography (PET-CT) showed multiple metabolic musculoskeletal lesions in the left lower limb. A CT scan–guided biopsy was performed. Histological and immunohistochemical findings confirmed the diagnosis of PMH. After treatment with intravenous pamidronate (90 mg/monthly), the patient had clinical improvement, mild pain 2/10 without the use of non-steroidal anti-inflammatory drugs or opiates. Follow-up was assessed by MRI and PET-CT. PET-CT showed metabolic resolution of most of the bone and muscular lesions and a significant improvement of the femoral lesion. MRI showed that the lesions in the left femur, tibia, and foot had a marked decrease in size without intravenous post-contrast enhancement and smaller lesions had disappeared. After a 3-year follow-up, PET-CT showed no metabolically active images. Literature review identified 31 records including 58 clinical cases of PMH with primary bone involvement and treatment description for qualitative analysis. Most lesions (69%) were treated by local excision or curettage. In addition, amputations were performed in a significant percentage of cases (20.7%). In the last years, mTOR inhibitors (n = 7) and anti-resorptive treatments (n = 4) were considered as alternative treatment options, especially in multifocal lesions.
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Metadaten
Titel
Pseudomyogenic hemangioendothelioma with bone and soft tissue involvement with favorable response to pamidronate: a case report and systematic review of the literature
verfasst von
María Lorena Brance
Nicolás M. Cóccaro
Pablo Roitman
Alejandro Castiglioni
Florencia Agostinis
Mariel Spense
Bárbara Scheitlin
Nicholas Rene
Lucas R. Brun
Publikationsdatum
01.12.2022
Verlag
Springer London
Erschienen in
Archives of Osteoporosis / Ausgabe 1/2022
Print ISSN: 1862-3522
Elektronische ISSN: 1862-3514
DOI
https://doi.org/10.1007/s11657-022-01062-4

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