{Reference Type}: Case Reports {Title}: Orofacial Manifestations in a Middle-Aged Woman with Cowden Syndrome: A Case Image. {Author}: de Arruda JAA;Freire CH;Leite TDB;Noce CW;Cavalcante IL;Vogel JO;Mesquita RA;Tenório JR;de Andrade BAB; {Journal}: Head Neck Pathol {Volume}: 17 {Issue}: 4 {Year}: 2023 Dec 21 暂无{DOI}: 10.1007/s12105-023-01586-2 {Abstract}: A 56-year-old Brazilian woman sought dental care, presenting with multiple asymptomatic papillomatous lesions with a coalescent pattern and intermingled cobblestone-like clefts along the alveolar ridge and marginal and attached gingivae. Multiple whitish papules were also observed on the face, neck, and limbs. Incisional biopsies of these lesions were performed. Microscopically, the skin lesion revealed epithelial clear cells and intraepithelial keratinization with areas of orthokeratosis, while the gingival lesions showed a parakeratinized stratified squamous epithelium with collagenous connective tissue. These features were consistent with those of a trichilemmoma and fibroepithelial hyperplasia, respectively. This article illustrates a case of Cowden syndrome (CS), a rare multisystem genetic condition in which both cutaneous and mucosal tissues were affected. Fewer than 40 cases of CS with oral involvement affecting middle-aged adults have been documented hitherto.