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Efficacy of zoledronic acid in Erdheim-Chester disease: A case report
Lucile Poiroux1, Frédéric Paycha2, Marc Polivka3
1Service de rhumatologie, pôle appareil locomoteur, centre Viggo-Petersen, hôpital Lariboisière, 2, rue Ambroise-Paré, 75010 Paris, France.
Insights
Erdheim-Chester disease, a rare histiocytosis, can cause debilitating bone pain. Zoledronic acid effectively relieved pain in a patient with bone Erdheim-Chester disease, showing sustained efficacy.
Area of Science:
- Histiocytosis
- Oncology
- Rheumatology
Background:
- Erdheim-Chester disease is a rare non-Langerhans cell histiocytosis.
- It involves organ infiltration by specific histiocytes (CD68+, CD1a-).
- Over 90% of cases present with bone involvement, causing pain and disability.
Observation:
- A 65-year-old woman with a history of breast cancer was diagnosed with biopsy-proven Erdheim-Chester disease affecting her bones.
- She experienced significant, difficult-to-treat pain due to bone localizations.
Findings:
- Treatment with zoledronic acid, a bisphosphonate, was initiated.
- The patient reported complete pain relief after three infusions.
- Efficacy was maintained at the one-year follow-up.
Implications:
- Bisphosphonates, such as zoledronic acid, appear to be an effective and safe treatment for bone manifestations of Erdheim-Chester disease.
- This case highlights a potential therapeutic option for managing pain and disability in affected patients.
- Further research into bisphosphonate efficacy in Erdheim-Chester disease is warranted.
Abstract:
Erdheim-Chester disease is rare form of non-Langerhans cell histiocytosis characterized by organ infiltration of CD68+ CD1a- histiocytes. Between 500 and 600 cases have been reported. It is a multifaceted disease ranging from a solely asymptomatic bone to a fatal multisystem pattern. Bone involvement occurs in more than 90% of cases. Although not life-threatening, bone localizations can be responsible of difficult-to-treat pain and disability. Treatment depends on lesion severity. Bisphosphonates have been reported to be efficient and safe in bone involvement. We report a case of a biopsy proven bone Erdheim-Chester disease in a 65-year-old woman with history of breast cancer. Her pain was relieved after 3 perfusions of zoledronic acid and the efficiency remained at one year of follow-up.
