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Cutaneous keratocyst arising independently of the nevoid basal cell carcinoma syndrome.

CLINICAL HISTORY AND MICROSCOPIC FINDINGS A 62-year-old man presented with a 2.3-cm asymptomatic cystic lesion on his calf. The specimen exhibited a cystic cavity with light-brown fluid. Microscopic examination revealed a dermal cyst (Fig. 1) lined by a corrugated squamous lining, forming keratin without a granular layer (Fig. 2). No pilar structures, including sebaceous glands or lanugo hairs, were identified on complete serial-sectioning through the block, ruling out the possibility of a cutaneous steatocystoma. The patient had no history of BCCs or jaw cysts, and lacked palmoplantar pitting or dysmorphic features. DISCUSSION The nevoid basal cell carcinoma syndrome (NBCCS) was described by Gorlin et al as a syndrome of multiple BCCs, odontogenic keratocysts (OKCs), skeletal abnormalities, and medulloblastomas. Further anomalies have been associated with the syndrome. The causative gene, patched on chromosome 9q22.3, is a homeobox analog that interacts with sonic hedgehog to control multiple steps in embryonic morphogenesis. Heterozygous patched knockout mice develop mandibular cysts similar to OKCs. Cutaneous keratocysts were first reported in NBCCS by Barr et al in 1986. Two additional cases have been reported. In addition, Leppard stated that cutaneous cysts in NBCCS