PMID- 38152056 OWN - NLM STAT- MEDLINE DCOM- 20240215 LR - 20240221 IS - 2328-9503 (Electronic) IS - 2328-9503 (Linking) VI - 11 IP - 2 DP - 2024 Feb TI - Clinicopathological features of graft versus host disease-associated myositis. PG - 508-519 LID - 10.1002/acn3.51973 [doi] AB - BACKGROUND AND OBJECTIVE: Chronic graft versus host disease (GVHD)-associated myositis targeting skeletal muscle is a relatively rare but potentially debilitating complication following allogeneic hematopoietic stem cell transplantation (HSCT). We reviewed the clinicopathological features of GVHD-associated myositis among patients receiving allogeneic HSCT to elucidate the cellular pathogenesis. METHODS: We retrospectively reviewed clinical data and muscle biopsy results from 17 consecutive patients diagnosed with GVHD-associated myositis at our institution between 1995 and 2019. Immunostaining findings of GVHD-associated myositis were compared to those of patients with anti-tRNA-synthetase antibody-associated myopathy (ASM) (n = 13) and dermatomyositis (DM) (n = 12). RESULTS: The majority of patients with GVHD-associated myositis showed subacute or chronic progression of mild to moderate limb weakness together with elevated serum creatine kinase. These patients also exhibited mild C-reactive protein elevation but were negative for myositis-related autoantibodies. Programmed death-1 (PD-1)-positive cells were observed in muscle interstitium adjacent to myofibers expressing human leukocyte antigen (HLA)-DR. The interstitium was also HLA-DR-positive, similar to biopsy samples from ASM patients but not DM patients. The proportions of HLA-DR-positive muscle fibers and PD-1-positive interstitial cells were significantly higher in GVHD and ASM samples than DM samples. The PD-1-positive cells were mostly CD-8-positive lymphocytes. DISCUSSION: GVHD-associated myositis is characterized by HLA-DR-positive myofibers and infiltration of PD-1-positive lymphocytes. These features distinguish GVHD-associated myositis from DM but not from ASM. CI - (c) 2023 The Authors. Annals of Clinical and Translational Neurology published by Wiley Periodicals LLC on behalf of American Neurological Association. FAU - Kazuta, Tomoyuki AU - Kazuta T AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. AD - Department of Neurology, Chutoen General Medical Center, Kakegawa, Japan. FAU - Murakami, Ayuka AU - Murakami A AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. AD - National Hospital Organization Suzuka National Hospital, Suzuka, Japan. FAU - Noda, Seiya AU - Noda S AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. AD - National Hospital Organization Suzuka National Hospital, Suzuka, Japan. FAU - Hirano, Satoko AU - Hirano S AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. AD - National Hospital Organization Suzuka National Hospital, Suzuka, Japan. FAU - Kito, Hiroshi AU - Kito H AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. AD - National Hospital Organization Suzuka National Hospital, Suzuka, Japan. FAU - Tsujikawa, Koyo AU - Tsujikawa K AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. FAU - Nakanishi, Hirotaka AU - Nakanishi H AD - Department of Neurology, Yokkaichi Municipal Hospital, Yokkaichi, Japan. FAU - Kimura, Seigo AU - Kimura S AD - National Hospital Organization Suzuka National Hospital, Suzuka, Japan. FAU - Sahashi, Kentaro AU - Sahashi K AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. FAU - Koike, Haruki AU - Koike H AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. AD - Division of Neurology, Department of Internal Medicine, Saga University Faculty of Medicine, Saga, Japan. FAU - Katsuno, Masahisa AU - Katsuno M AUID- ORCID: 0000-0001-9453-9311 AD - Department of Neurology, Nagoya University Graduate School of Medicine, Nagoya, Japan. AD - Department of Clinical Research Education, Nagoya University Graduate School of Medicine, Nagoya, Japan. LA - eng GR - JP20H00527/Japan Society for the Promotion of Science/ GR - JP23H00420/Japan Society for the Promotion of Science/ PT - Journal Article PT - Research Support, Non-U.S. Gov't PT - Review DEP - 20231228 PL - United States TA - Ann Clin Transl Neurol JT - Annals of clinical and translational neurology JID - 101623278 RN - 0 (Programmed Cell Death 1 Receptor) RN - 0 (HLA-DR Antigens) SB - IM MH - Humans MH - Retrospective Studies MH - Programmed Cell Death 1 Receptor MH - *Myositis/etiology/diagnosis MH - *Graft vs Host Disease/complications MH - HLA-DR Antigens/metabolism PMC - PMC10863911 COIS- We have no conflict of interest to disclose. EDAT- 2023/12/28 06:42 MHDA- 2024/02/15 06:43 PMCR- 2023/12/28 CRDT- 2023/12/28 03:54 PHST- 2023/12/02 00:00 [revised] PHST- 2023/08/17 00:00 [received] PHST- 2023/12/05 00:00 [accepted] PHST- 2024/02/15 06:43 [medline] PHST- 2023/12/28 06:42 [pubmed] PHST- 2023/12/28 03:54 [entrez] PHST- 2023/12/28 00:00 [pmc-release] AID - ACN351973 [pii] AID - 10.1002/acn3.51973 [doi] PST - ppublish SO - Ann Clin Transl Neurol. 2024 Feb;11(2):508-519. doi: 10.1002/acn3.51973. Epub 2023 Dec 28.