Background. Proliferative myositis is a rare, benign, reactive intramuscular lesion of fibroblastic/myofibroblastic origin; an identical lesion in a subcutaneous or fascial location is referred to as proliferative fasciitis. The rapid growth rate and unusual histopathologic features have frequently been mistaken for a malignant process and have promoted unnecessary invasive procedures. Here we present only the third oral case of proliferative myositis, arising from the tongue of a 65-year-old man.Methods and Results. Histologically, the resected lesion was composed of numerous fibroblastic or myofibroblastic spindle cells and variable numbers of large ganglion-like cells infiltrating between and around muscle fascicles, resembling a ''checkerboard'' configuration. A demographic profile of proliferative myositis of the head and neck is also provided, compiled from 19 patients culled from an English-language literature review and this report.Conclusions. Incisional biopsy or fine-needle aspiration biopsy of proliferative myositis of the head and neck should lead to spontaneous resolution and is, therefore, sufficient to render the diagnosis and to provide conservative treatment. Recurrence is extremely rare. V Fibroblastic and myofibroblastic tumors comprise a group of tumefactions exhibiting biologic diversity ranging from reactive, pseudosarcomatous proliferations to intermediate lesions, such as the fibromatoses, to malignant neoplasms, including fibrosarcoma. 1-3 Nodular fasciitis, proliferative fasciitis, and proliferative myositis represent related reparative reactions with many histologic similarities. 2,3 The hallmark feature of these lesions is the alarming growth rate, often masquerading as a malignant process, such as rhabdomyosarcoma or other sarcomas. Although nodular fasciitis is considered relatively common, proliferative fasciitis and proliferative myositis are rare lesions that are identical in appearance with 1 exception: the former is a fascial or subcutaneous process, while the latter is intramuscular.To date, only 2 patients with proliferative myositis have been reported within the oral cavity, both involving the tongue. 4,5 Here we present a third patient with proliferative myositis of the tongue, including microscopic and immunohistochemical findings.