Background:

Hypertrophic osteoarthropathy involves abnormal proliferation of bone at distal extremities. It involves clubbing, periostitis of tubular bones and synovial effusions. Some causes of secondary hypertrophic osteoarthropathy include lung cancer, congenital heart disease, inflammatory bowel disease and cirrhosis. The inflammation associated with intestinal transplant rejection may be another cause of secondary hypertrophic osteoarthropathy.

Methods:

Retrospective review of a transplant recipient who experienced significant bone pain after being treated for acute moderate rejection of her intestinal graft

Results:

A 5 year-old female who had received a liver/small bowel/pancreas transplant was treated for acute moderate rejection of her intestine with steroids, thymoglobulin and infliximab. Approximately two months after her initial presentation, she had increased pain and was unwilling to move or bear weight on her legs. An xray osseous survey showed diffuse periosteal bone formation in the long bones, including the bilateral humeri, ulna, femurs and tibias. There was also involvement in the left fibula and left radius. This was diagnosed as hypertrophic osteoarthropathy and was thought to be related to recent transplant rejection. The patient also had clubbing in her fingertips. Treatments of choice for hypertrophic osteoarthropathy include treatment of underlying disease and symptom management with analgesics, NSAIDS, corticosteroids and bisphosphonates. Due to potential nephrotoxic side effects of NSAIDS with tacrolimus, our patient was treated with oxycodone, methylprednisolone (initial dose: 2 mg/kg/day then gradually tapered over 10 weeks) and pamidronate 1 mg/kg IV X 2 doses three weeks apart. Within a few days after initiating treatment, our patient was able to walk across the room and was more cooperative with her care.

Conclusions:

Moderate to severe rejection intestinal rejection is a potential etiology for the development of hypertrophic osteoarthropathy post-transplant which causes significant pain and discomfort. Treatment modalities that may improve patient symptoms include analgesics, corticosteroids and bisphosphonates.

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