Purpose: We report a case of ocular surface squamous neoplasia In a child with xeroderma pigmentosum, treated with excisional biopsy and cryotherapy, in which a map biopsy of noncontiguous conjunctiva was performed to assess the status of the ocular surface. Methods: Case report of patient managed by the Cornea & External Disease service at University of California, Davis Eye Center. Results: Clinical and histopathologic findings of the specimens including p53 staining are described. Conclusion: Scattered ocular surface abnormalities were found in our patient and this may predispose these patients to future recurrences in areas that are not necessarily contiguous with the primary lesion. This finding confirms the need for close follow-up and raises the possibility of adjunctive topical chemotherapy use even for primary lesions in this high-risk patient group.