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孤立性骨朗格汉斯细胞组织细胞增多症病变单次输注双膦酸盐后完全缓解:一例报告及文献综述

Complete remission after a single bisphosphonate infusion in isolated bone Langerhans cell histiocytosis lesion: a case report and a narrative review of the literature.

作者信息

Kachaner Alexandra, Seror Raphaèle, Aubart Fleur Cohen, Henry Julien, Lazure Thierry, Emile Jean François, Mariette Xavier, Bitoun Samuel

机构信息

Department of Rheumatology, Paris Saclay University, Bicêtre Hospital, AP-HP, FHU Care, Le Kremlin Bicêtre 94270, France.

Internal Medicine Department 2, French National Referral Center for Rare Systemic Diseases and Histiocytosis, Sorbonne University AP-HP, Pitié-Salpêtrière Hospital, INSERM UMRS-1135 Team 7, Paris 75013, France.

出版信息

JBMR Plus. 2024 Apr 20;8(5):ziae043. doi: 10.1093/jbmrpl/ziae043. eCollection 2024 May.

Abstract

Langerhans cell histiocytosis (LCH) is a rare disease with limited treatment options. We present a case involving a 57-year-old woman afflicted with an isolated LCH bone osteolytic lesion. A single bisphosphonate infusion significantly alleviated pain, and follow-up scans via CT, PET-CT, and MRI revealed a substantial recalcification of the lesion. Conducting an extensive literature review, we identified 46 cases documenting the efficacy of bisphosphonates in the context of LCH. These findings have raised interest in bisphosphonate infusion as a simple therapeutic alternative in similar situations, with benefits in terms of bone recalcification and pain control for individuals with LCH.

摘要

朗格汉斯细胞组织细胞增多症(LCH)是一种治疗选择有限的罕见疾病。我们报告了一例病例,患者为一名57岁女性,患有孤立性LCH骨溶解性病变。单次双膦酸盐输注显著缓解了疼痛,通过CT、PET-CT和MRI进行的随访扫描显示病变有大量重新钙化。通过广泛的文献综述,我们确定了46例记录双膦酸盐在LCH背景下疗效的病例。这些发现引发了人们对双膦酸盐输注作为类似情况下一种简单治疗选择的兴趣,对LCH患者在骨重新钙化和疼痛控制方面有好处。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/8555/11032215/fce22711d771/ziae043ga1.jpg

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