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A case of Sjogren's syndrome evolving into multicentric reticulohistiocytosis

Rheumatology & Autoimmunity 2023年第3期3卷 179-184页

作者： Daniel Gonzalez Vinh Lu Madeline Bonnet Rachelle Gietzen Anthony Linfante Enrique Medina Vijaya Murthy Division of Rheumatology University of Texas Medical BranchGalvestonTexasUSA Department of Internal Medicine University of Texas Medical BranchGalvestonTexasUSA University of Texas Medical Branch(UTMB)John Sealy School of Medicine GalvestonTexasUSA Department of Pathology University of Texas Medical BranchGalvestonTexasUSA Department of Dermatology University of Texas Medical BranchGalvestonTexasUSA

Introduction:Multicentric reticulohistiocytosis(MRH)is a rare disease that is known to affect the skin and joints,*** is considered a rare form of non-Langerhans cell histiocytosis(Group C)that can cause destructive inflammatory arthritis involving both the small and large *** eruptions of periungual,“coral beads”and nodules appearing over the distal fingers are considered pathognomonic clues for identifying this *** evaluation of the cutaneous papules typically shows infiltrative histiocytes and multinucleated giant *** no well-established therapies exist to date,a variety of immunosuppressants have been used with varying degrees of *** Description:A 53-year-old Caucasian female patient with a family history of rheumatoid arthritis and a personal history of Sjogren's syndrome presented to the rheumatology clinic complaining of pain in her bilateral hands and *** were several small,papulo-nodular lesions ranging from 1 to 2mm in size noted at the base of her nails.A 4mm punch biopsy of one of the papules from the neck showed dermal infiltration of eosinophilic mononucleated and multinucleated giant cells with“ground glass”appearing cytoplasm consistent with MRH.X-ray of her hands showed periarticular demineralization and erosions surrounding several bilateral proximal and distal interphalangeal joints,and thus tofacitinib in addition to methotrexate,hydroxychloroquine,and dexamethasone(dosed weekly)was started to help control any further articular ***:Our aim is to further support the relation of MRH with autoimmune diseases,including Sjogren's *** diseases have been reported in association with MRH,although a clear association has yet to be made.

关键词： erosive arthritis multicentric reticulohistiocytosis Sjogren's syndrome

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