Treatment of hypertrophic osteoarthropathy with zoledronic acid: case report and review of the literature

Objectives: Zoledronic acid (ZA) is rarely used to manage hypertrophic osteoarthropathy (HOA). We report our experience with ZA treatment of a patient with HOA and sarcoidosis who had also undergone lung transplant. We also conducted a literature review of the usefulness of bisphosphonates in HOA.

Methods: We performed a PubMed literature search using keywords HOA, periostitis, bisphosphonate, ZA, sarcoidosis, and lung transplant. A PRISMA flow diagram is presented to depict the data collection process, and a case is reported.

Results: A 62-year-old woman with bilateral lung transplant as a result of severe pulmonary sarcoidosis developed severe limb pain and inflammatory polyarthritis. HOA was diagnosed in the presence of periostitis with the symptoms. Failure of the refractory bone and joint pain to respond to low doses of prednisone, tramadol, or even pamidronate infusion prompted a trial of a single dose of intravenous ZA. Surprisingly, the pain completely resolved without recurrence. A total of 12 cases of HOA treated with bisphosphonates were retrieved from the literature and reviewed.

Conclusions: Bisphosphonates are generally effective therapy for HOA-related pain. ZA may be even more efficacious and longer lasting than pamidronate for management of the bone and joint pain associated with HOA irrespective of the underlying disorders.