Background/Purpose: Hematopoietic stem cell transplant (HSCT) is a commonly employed treatment for hematologic malignancies including acute leukemias and lymphomas but can be complicated by graft-versus-host disease (GVHD), an inflammatory syndrome affecting multiple organs. While there have been case reports of post-transplant rheumatic syndromes (e.g. myositis and psoriasis), the incidence of post-HSCT inflammatory arthritis is less clear. We aimed to characterize the clinical phenotypes of patients evaluated for inflammatory arthritis after HSCT and to perform a literature review of reported cases of post-HSCT inflammatory arthritis.

Methods: We analyzed the clinical and demographic data of six patients evaluated in the Division of Rheumatology at Johns Hopkins for inflammatory arthritis with initial symptoms after HSCT for hematologic malignancy. Chart review included evaluation of the patient’s hematologic malignancy treatment history, clinical arthritis phenotype, arthritis symptom severity and disease activity, serum inflammatory markers, autoantibodies, and arthritis treatment outcomes. For the literature review, the PUBMED database was queried with 24 targeted search terms including those related to inflammatory arthritis, rheumatic disease, and joint involvement combined with hematopoietic stem cell transplant and GVHD, and primary reports were reviewed to assess for common presenting clinical features, immunologic history, and response to treatment.

Results: Of the six patients evaluated in our center for concern of inflammatory arthritis presenting after HSCT, five had HSCT for treatment of acute myeloid leukemia, and three had received at least some treatment for GVHD (Table 1). Symptoms of inflammatory arthritis arose 4-72 months after HSCT (Table 1). Clinical phenotype varied in severity from mild to severe as measured by the Clinical Disease Activity Score (CDAI), and did not clearly correlate with timing of onset post-transplant, prior GVHD, or prior history of autoimmune disease (Table 2). The most frequently involved joints were PIPs, MCPs, MTPs, wrists, and ankles. A majority (5/6) of cases did not have positive serologic markers of rheumatoid arthritis (RF, anti-CCP) or other tested autoantibodies (Table 2). A variety of immunosuppressive medications were employed for treatment of arthritis symptoms, and some patients required corticosteroids for management of symptoms. Literature search identified 16 primary case reports and literature reviews of inflammatory arthritis post-HSCT. The two largest case series each reported three cases. Psoriatic arthritis, seropositive rheumatoid arthritis, pseudogout, and spondylarthritis were described in case reports.

Conclusion: De novo seronegative inflammatory arthritis occurs after HSCT, characterized by variable severity of presentation and association with other symptoms of GVHD. A literature review identified a limited number of reports of inflammatory arthritis after HSCT, suggesting that continued research is needed to identify predisposing factors in the post-transplant period and advance understanding of rheumatologic complications arising from immunotherapy treatment of hematologic malignancies.

Table 1. Malignancy and transplant history and timing of arthritis onset after HSCT.

Table 2. Clinical phenotype and laboratory data of patients presenting with post-HSCT inflammatory arthritis

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ACR Meeting Abstracts - https://acrabstracts.org/abstract/inflammatory-arthritis-after-hematopoietic-stem-cell-transplant-a-case-series-and-literature-review/