A-59-year-old Japanese woman presented a cellulitis-like erythematous skin rash, low-grade fever, and general fatigue, accompanied by a firm swelling of the right parotid gland. She had a history of tick bite on the right lateral neck 2 weeks before. Serum anti-Borrelia burgdorferi antibody was positive by Western blot analysis, and B. burgdorferi was isolated from the skin lesion. Serum amylase level was elevated with predominant salivary gland isozyme; the level returned to normal within 3 weeks following penicillin and tetracycline treatment. Parotitis might be included among the rare complications of Lyme disease affecting the head and neck region.