Background Goltz syndrome, also known as focal dermal hypoplasia (FDH), is a rare genetic disorder that affects ectodermal and mesodermal tissues. The purpose of this study is to report the treatment of papillomatous lesions in the orofacial region of a patient with Goltz syndrome. Case description A 31‐year‐old male patient was attended at Dental School of Federal University of Goiás with the chief complaint of discomfort during speaking and eating, due to the presence of a papillomatous lesion at the labial commissure. During the physical examination, syndrome characteristics were found, such as unilateral anophthalmia; atrophic cutaneous lesions of different intensities of pigmentation, mostly on the arms and neck; sparse hair; ectrodactyly, being the right hand was shaped like a ‘lobster claw’; and body asymmetry. In addition, papillomatous lesions at the right labial commissure and on the dorsum of the tongue were noted, which were subsequently removed with surgical laser. The microscopic diagnosis of the lesions was a squamous papilloma. Conclusion The proposed surgical treatment was well succeeded, and the rarity of the present case also having Goltz syndrome is an uncommon condition in males.
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