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Introduction: Celiac disease (CD) is an autoimmune enteropathy due to hypersensitivity to consumed gluten. Patients can present with gastrointestinal and systemic symptoms. CD often runs in families. Studies have reported multiple subtypes of CD including at least one seronegative variant. The cutaneous manifestations of CD remain to be delineated; some cases of seropositive CD are associated with dermatitis herpetiformis. We report. Cases: In this study, three patients with a longstanding history of personal and/or familial seronegative CD presented withwho developed basal cell carcinoma (BCC) of the perineum. We hope to inform clinical physicians about the possible relationship between CD and cutaneous malignancies. DiscussionResults: We report three adult patients with a diagnosed or suspected personal history of seronegative CD and a family history of CD, who presented with BCC of the perineum. Discussion: The mechanism by which CD causes BCC remains to be defined. Physicians should consider testing evaluating patients with skin cancer located in the perineum for CD.