We herein report a case of therapy-resistant dermatomyositis treated with oral prednisolone and chlorambucil combination therapy. Concurrently, she showed cervical carcinoma in situ(CIS). Initially, we started to treat her with combination oral prednisolone, intramuscular methotrexate, hydroxychloroquin, and removal of cervical CIS. However, our patient failed to respond to these regimens. Thus, we had have another combination treatment of oral prednisolone and chlorambucil. After the treatment of this combination regimen, her recalcitrant dermatomyositis improved dramatically without recurrence. There were no significant adverse side effects with chlorambucil therapy.