Musculoskeletal graft versus host disease (GVHD) is a rare manifestation of chronic GVHD (cGVHD) following allogeneic hematopoietic stem cell transplantation (allo-HSCT). Left untreated, the disease can cause extensive damage to muscle tissue and joints. We describe a 62-year-old male with musculoskeletal GVHD and generalized muscle pain and stiffness. In addition, we performed a systemic literature review based on published cases of musculoskeletal GVHD between 1983 and 2019. We identified 85 cases, 62% male and 38% female with an age of 4–69 years and median age of 39 years at diagnosis. The majority of patients (72%) also had manifestations of cGVHD in at least one other organ system, most frequently the skin (52%), followed by oropharyngeal mucosa (37%), and pulmonary and gastrointestinal tract (GI tract) (21%). We conclude that, while musculoskeletal cGVHD is a rare complication of allo-HSCT, it remains a serious and debilitating risk that must be considered in patients with muscle pain, muscle weakness, joint stiffness, and tissue inflammation. Early intervention is critical for the patient’s prognosis.
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