Combination csDMARD and Infliximab in Primary Hypertrophic Osteoarthropathy, a Case-Based Review
Burak Okyar1, İbrahim Halil Bilen2, Fatih Yildiz31Adana City Training and Research Hospital, Department of Internal Medicine, Division of Rheumatology, Adana/Turkey2Kahramanmaraş Sütçü İmam University Faculty of Medicine, Department of Internal Medicine, Kahramanmaraş/Turkey
3Kahramanmaraş Sütçü İmam University Faculty of Medicine, Department of Internal Medicine, Division of Rheumatology, Kahramanmaraş/Turkey
An 18-year-old male patient presented with excessive sweating on the palms of the hands and soles of the feet. X-ray showed periostosis in the carpal and tarsal bones. Genetic analysis revealed c.1807C>T(p.Arg603Ter), a homozygous mutation, and PHO was diagnosed. The patient was treated with dexketoprofen, methotrexate, and methylprednisolone for the first six months. No adequate response was obtained; infliximab was added to the existing treatment and continued for another six months. The number of tender and swollen joints decreased. However, a partial response was obtained. After the 12th month, dexketoprofen was discontinued, and celecoxib was added. Arthralgia, hyperhidrosis, acne and seborrhea, and minor joint arthritis responded utterly. COX inhibitors were effective on pachydermia and clubbing but did not affect arthralgia and hyperhidrosis. It seems effective in cases treated with MTX and IFX, especially on pustular lesions, arthralgia, and hyperhidrosis. We responded to almost all clinical phenotypes with combination therapy and put the patient into remission. The literature review shows that the number of patients in whom biologic therapy has been tried is very small. Our case is the first case with an almost complete response to combination therapy, and the second case was treated with infliximab.
Keywords: Primer Hipertrophy Osteoarthropathy, pachyderma periostosis, biological treatmentManuscript Language: English