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

Author:

Kachaner Alexandra1,Seror Raphaèle1,Aubart Fleur Cohen23,Henry Julien1,Lazure Thierry4,Emile Jean François5,Mariette Xavier1,Bitoun Samuel1

Affiliation:

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

2. Internal Medicine Department 2 , French National Referral Center for Rare Systemic Diseases and Histiocytosis, , INSERM UMRS-1135 Team 7, Paris 75013 , France

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

4. Department of Pathology, Paris Saclay University, Bicêtre Hospital, AP-HP , Le Kremlin Bicêtre 94270 , France

5. Paris-Saclay University, Versailles SQY University, EA4340-BECCOH, Assistance Publique–Hôpitaux de Paris (AP-HP), Ambroise-Paré Hospital, Smart Imaging, Service de Pathologie , Boulogne 92100 , France

Abstract

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.

Publisher

Oxford University Press (OUP)

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