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Case of dermatomyositis during treatment with pembrolizumab for lung cancer

culitis. We report a case of primary SS who had been suffering from lower leg ulcers with cutaneous vasculitis and carried APA including aPS/PT antibodies. A 59-year-old woman with no history of abortion who was working as a hairdresser had been suffering from stasis dermatitis for 10 years. Multiple lower leg ulcers had occurred and progressed 5 months before her first visit. At that visit, she had numerous cutaneous ulcers and varicose veins (Fig. 1a–d), and mild hypoesthesia on the lower legs without livedo racemosa. The joint deformities without X-ray changes of rheumatoid arthritis were considered by a rheumatologist to have been caused by the long-term use of elastic bandages. Blood tests showed elevated levels of serum C-reactive protein (3.54 mg/ dL), immunoglobulin (Ig)G (3032 mg/dL), IgA (597 mg/dL), IgM (117 mg/dL) and C3 (154.3 mg/dL). Although her prothrombin time was within normal limits (106.4%), her activated partial thromboplastin time was reduced (76.8%) and her serum Ddimer level was elevated (2.61 lg/mL; normal, 1.0). Neither serum protein C activity (77%) nor protein S activity (170%) decreased. Serum anti-SSA antibody (116.9 index; normal, <7) and anti-cardiolipin antibody (119 U/mL; normal, <10) were positive, whereas perinuclear/cytoplasmic antineutrophil cytoplasmic antibody, rheumatoid factor, anti-CCP, -Sm, -dsDNA, SS-B and -b2 glycoprotein I antibodies, and lupus anticoagulant were negative. aPS/PT-IgG antibody was positive (>200 U/ mL; normal, <12) without aPS/PT-IgM antibody. She had no cryoglobulinemia. Schirmer and gum tests were both positive. A biopsy specimen from the minor salivary gland in the lower lip showed infiltration of lymphocytes around the salivary ducts (Fig. 1e). A biopsy specimen from skin close to an ulcer on the right lower leg showed hemorrhage in the upper dermis and infiltration of lymphocytes, plasma cells and neutrophils with nuclear dust around the dermal blood vessels (Fig. 1f,g). Direct immunofluorescence tests revealed C3 deposits on the vascular walls in the upper dermis (Fig. 1h). Ultrasonography and contrast-enhanced computed tomography showed no thrombi in the lower limb veins. From these results, she was diagnosed with SS with cutaneous vasculitis. We administrated oral prednisolone (30 mg/day) and aspirin (81.5 mg/day) with the topical application of povidone-iodine for the ulcers. Re-epithelialization of the ulcers began 1 week after the administration of these drugs. The high titer of aPS/PT-IgG antibody in our case is noteworthy. A study of Japanese patients showed all six SS patients with cutaneous vasculopathy to carry aPS/PT antibodies. One patient had aPS/PT-IgG antibody and the other five had IgM-class antibodies. Although all four immunohistochemically examined cases with IgM-class antibodies showed IgM deposition around the dermal vessels, one case with IgG-class antibody exhibited no IgG deposition like our case. Our case suggests the importance of thorough clinical investigation in SS patients with leg ulcers. The detection of APA including aPS/PT antibodies may be useful for the diagnosis of cutaneous vasculitis underlying leg ulcers.