Case Report

The Journal of the Korean Rheumatism Association 2008; 15(4): 337-342

Published online December 30, 2008

© Korean College of Rheumatology

류마티스관절염으로 오인된 다중심 망상조직구 증식증 1예

이주현1ㆍ윤보영1ㆍ장재호1ㆍ김한성2ㆍ김용훈3ㆍ박혜진4

인제대학교 의과대학 일산백병원 내과학교실1, 병리학교실2, 영상의학교실3, 피부과학교실4

A Case of Multicentric Reticulohistiocytosis Mistaken as Rheumatoid Arthritis

Ju Hyun Lee1, Bo Young Yoon1, Je Ho Chang1, Han Seong Kim2, Yong Hoon Kim3, Hai Jin Park4

Departments of Internal Medicine1, Pathology2, Radiology3, and Dermatology4, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea

Correspondence to : Bo Young Yoon

Abstract

Multicentric reticulohistiocytosis (MRH) is a rare disease affecting joints, skin and internal organs. Yet the cause is still unknown. In most cases, it can be misdiagnosed as rheumatoid arthritis or psoriatic arthritis before typical change in the skin emerges, and precise diagnosis is essential because it may become severe enough to transform and destroy the joints. Therefore, skin or synovial biopsy can confirm the existence of this disease. This particular patient is a 39-year-old male who had been treated for rheumatoid arthritis on a wrist and hand and transferred to this hospital when arthritic pain continued with erythematous papules and nodules on the hands, the outer rims of the ears, hands and elbows. The X-ray examination of both hands revealed multiple marginal erosion of proximal and distal interphalangeal joints and destruction of subchondral bones. periarticular osteoporosis and joint space enlargement is combined, but no new bones were seen to be formed. Biopsy of erythematous nodules on the dorsum of hands showed that an infiltrate histiocytes and multi-nuclear giant cells were aligned irregularly, and immunological chemical staining showed potential for being positive to PAS and CD68. To control pain and regulate activity of the disease, tracing observation and treatment were started from the outside with NSAID, hydroxycholoquine, MTX and prednisolone. A month of treatment did not improve arthritis and skin problems, and increased dose of MTX, prednisolone did improve arthritis a little but not skin problems. Treatment with infliximab (3 mg/kg), a anti-tumor necrosis factor, is in progress, showing improvement in both conditions.

Keywords Multicentric reticulohistiocytosis, Rheumatoid arthritis, Infliximab

Article

Case Report

The Journal of the Korean Rheumatism Association 2008; 15(4): 337-342

Published online December 30, 2008

Copyright © Korean College of Rheumatology.

류마티스관절염으로 오인된 다중심 망상조직구 증식증 1예

이주현1ㆍ윤보영1ㆍ장재호1ㆍ김한성2ㆍ김용훈3ㆍ박혜진4

인제대학교 의과대학 일산백병원 내과학교실1, 병리학교실2, 영상의학교실3, 피부과학교실4

A Case of Multicentric Reticulohistiocytosis Mistaken as Rheumatoid Arthritis

Ju Hyun Lee1, Bo Young Yoon1, Je Ho Chang1, Han Seong Kim2, Yong Hoon Kim3, Hai Jin Park4

Departments of Internal Medicine1, Pathology2, Radiology3, and Dermatology4, Ilsan Paik Hospital, Inje University College of Medicine, Goyang, Korea

Correspondence to:Bo Young Yoon

Abstract

Multicentric reticulohistiocytosis (MRH) is a rare disease affecting joints, skin and internal organs. Yet the cause is still unknown. In most cases, it can be misdiagnosed as rheumatoid arthritis or psoriatic arthritis before typical change in the skin emerges, and precise diagnosis is essential because it may become severe enough to transform and destroy the joints. Therefore, skin or synovial biopsy can confirm the existence of this disease. This particular patient is a 39-year-old male who had been treated for rheumatoid arthritis on a wrist and hand and transferred to this hospital when arthritic pain continued with erythematous papules and nodules on the hands, the outer rims of the ears, hands and elbows. The X-ray examination of both hands revealed multiple marginal erosion of proximal and distal interphalangeal joints and destruction of subchondral bones. periarticular osteoporosis and joint space enlargement is combined, but no new bones were seen to be formed. Biopsy of erythematous nodules on the dorsum of hands showed that an infiltrate histiocytes and multi-nuclear giant cells were aligned irregularly, and immunological chemical staining showed potential for being positive to PAS and CD68. To control pain and regulate activity of the disease, tracing observation and treatment were started from the outside with NSAID, hydroxycholoquine, MTX and prednisolone. A month of treatment did not improve arthritis and skin problems, and increased dose of MTX, prednisolone did improve arthritis a little but not skin problems. Treatment with infliximab (3 mg/kg), a anti-tumor necrosis factor, is in progress, showing improvement in both conditions.

Keywords: Multicentric reticulohistiocytosis, Rheumatoid arthritis, Infliximab

JRD
Oct 01, 2024 Vol.31 No.4, pp. 191~263
COVER PICTURE
Ancestry-driven pathways for SLE-risk SNP-associated genes. The ancestry-driven key signaling pathways in Asians, Europeans, and African Americans were analyzed by enrichr (https://maayanlab.cloud/Enrichr/#libraries) using non-HLA SNP-associated genes. SLE: systemic lupus erythematosus, SNP: single-nucleotide polymorphism, JAK–STAT: janus kinase–signal transducers and activators of transcription, IFN: interferon gamma. (J Rheum Dis 2024;31:200-211)

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