Abstract
BackgroundPolymyositis and myocarditis associated with thymoma are exceptionally rare conditions and usually accompanied by myasthenia gravis (MG) and have been recognized as critical conditions. Thymoma-associated multiorgan autoimmunity was reported recently with skin, liver, and intestinal manifestations similar to those seen in graft-versus-host disease.Case presentationA 77-year-old female presented to our department with exacerbation of ptosis and local recurrence of thymoma. Chest computed tomography revealed local recurrence of thymoma. Following 6 month observation, erythema on the extremities and body trunk suddenly appeared. Afterwards, the patient developed progressive muscle weakness and fatigue. We diagnosed as myocarditis and polymyositis. She was transferred to the intensive-care unit and received artificial ventilation. Steroid pulse therapy was induced immediately. The blood test findings were markedly improved, but the symptoms of MG and weakness of the muscles persisted. Various treatment including eculizumab was induced, and the symptoms of MG and weakness of the muscles were improved. On the 136th day of hospitalization, she was discharged.ConclusionWe were able to cure this patient, as we were able to start treatment immediately after the appearance of severe symptoms. An early diagnosis and treatment are important for curing such patients.
Highlights
Polymyositis and myocarditis associated with thymoma are exceptionally rare conditions and usually accompanied by myasthenia gravis (MG)
Thymoma-associated multiorgan autoimmunity (TAMA) was reported recently with skin, liver, and intestinal manifestations similar to those seen in graft-versus-host disease (GVHD) [3]
The present case showed a variety of symptoms, such as GVHD-like skin lesions, liver dysfunction, polymyositis and myocarditis, MG and pure red cell aplasia (PRCA), which were not mentioned in previous reports
Summary
Polymyositis and myocarditis associated with thymoma are exceptionally rare conditions and usually accompanied by myasthenia gravis (MG). Case presentation A 77-year-old female presented to our department with exacerbation of ptosis and local recurrence of thymoma Five years earlier, she had been diagnosed with an anterior mediastinal tumor with a symptom of ptosis and underwent thymectomy for type B2 thymoma of Masaoka stage II disease. Aspartate aminotransferase (AST) and alanine aminotransferase (ALT) levels were significantly increased, and complication of drug-induced or autoimmune hepatitis was suspected She slept in the sitting position, because her dyspnea worsened in the supine position. A myocardial biopsy could not be performed, we diagnosed as polymyositis and myocarditis which is rarely associated with thymoma This diagnosis was valid considering the positive findings of antistriational autoantibodies (anti-titin and anti-Kv1.4), elevation of myosin light chain I and luck of elevation of anti-AChR antibody, which were revealed later. There has been no recurrence of any of her autoimmune diseases, excluding MG, for 1 year
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