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Multicentric Reticulohistiocytosis with Positive Anticyclic Citrullinated Antibodies Anupama Chauhan, MD; Zhanna Mikulik; and Kevin V. Hackshaw
This is a case report of a woman with multicentric reticulohistiocytosis with positive anticyclic citrullinated antibodies. This patient had been misdiagnosed with rheumatoid arthritis for many years. Recently, she presented with symmetric distal interphalangeal joint destruction and papules along her nail beds. Her clinical presentation, laboratory data, radiographic and histologic findings were all consistent with multicentnc reticulohistiocytosis, not rheumatoid arthntis. This is the first case report of a patient with multicentric reticulohistiocytosis that tested positive for anticyclic citrullinated
antibodies. Key words: multicentric reticulohistiocytosis U anticyclic citrullinated antibodies * distal interphalangeal joints © 2007. From the Division of Immunology/Rheumatology, William Davis Medical Research Center, The Ohio State University College of Medicine and Public Health, Columbus, OH. Send correspondence and reprint requests for J NatI Med Assoc. 2007;99:678-680 to: Dr. Anupama Chauhan, Division of Immunology/Rheumatology, William Davis Medical Research Center, The Ohio State University College of Medicine and Public Health, 480 Medical Center Drive, Columbus, OH 43210-1228; phone: (614) 2938093; fax: (614) 293-5631; e-mail:
[email protected]
INTRODUCTION ulticentric reticulohistiocytosis (MRH) is a rare disorder characterized by infiltration of multinucleated giant cells and histiocytes into various tissues. The typical clinical picture includes skin nodules and a destructive polyarthritis. The first case described was by Goltz and Layman in 1954.' This entity is frequently mistaken for rheumatoid arthritis. Cyclic citrullinated peptide is an artificial molecule similar to fillagrin. Anticyclic citrullinated antibodies (anti-CCPs) can be found in the joints of rheumatoid arthritis patients early in the disease, whereas citrullinated proteins are not found in normal joints.2 Anti-CCPs are also found in psoriatic arthritis, Sjogren's syndrome and juvenile rheumatoid arthritis.3 The specificity of the test for rheumatoid arthritis ranges from 88.9-100%, and the sensitivity ranges from 14.4-96%.3 This case is unique in that the patient presents with MRH in the presence of positive anti-CCPs. M u
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CASE REPORT This patient, a 74-year-old African-American woman, was first seen in our clinic in 2004. The disease started with left heel pain six years prior. The patient was treated with prednisone. Two years later, she developed arthralgias in her knees and neck, and Plaquenil® was added. Within the next two years, she noticed painless, symmetric deformities of her distal interphalangeal joints (DIPs). One year ago, she developed erythematous nodules and papular lesions on her nail beds and left ear. Decreased appetite, fatigue and a 40-pound weight loss occurred in the last two years. Her past medical history included hypertension, hiatal hernia, esophageal stricture and bilateral cataracts. Her past surgical history included a hysterectomy, sinus surgery and esophageal dilation. Her family history included Alzheimer's, cirrhosis and no connective tissue disease. Her physical exam revealed marked deformities in bilateral DIPs and synovitis of the left knee. A nodule was noted on the interphalangeal joint of her left thumb. Erythematous nodular and papular lesions were noted on the dorsum of bilateral hands. Laboratory studies showed an elevated erythrocyte sedimentation rate of 32 (0-29) mm/hr, anemia with hemoglobin of 11.3 and a low albumin at 3.2. Additional studies showed a negative rheumatoid factor, a negative antinuclear antibody, negative extractable nuclear antigens and a positive antiCCP of >100 (
