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Hidradenitis suppurativa (HS) is a debilitating skin disease characterized by painful recurrent nodules and abscesses caused by chronic inflammation. Early events in the development of HS are believed to occur in the folliculopilosebaceous unit; however, the signaling pathways behind this mechanism are unknown. Sphingolipids, such as ceramide, are essential components of the skin and appendages and have important structural and signaling roles.We sought to explore whether the gene expression of enzymes involved in sphingolipid metabolic pathways is altered in HS.A microarray data set including 30 samples was used to compare the expression of sphingolipid-related enzymes in inflammatory skin lesions from HS patients (n = 17) with the expression in clinically healthy skin tissue (n = 13). Differential expression of sphingolipid metabolism–related genes was analyzed using Gene Expression Omnibus 2R.HS lesional skin samples have significantly decreased expression of enzymes generating ceramide and sphingomyelin, increased expression of enzymes catabolizing ceramide to sphingosine, and increased expression of enzymes converting ceramide to galactosylceramide and gangliosides.Limitations of this study include assessing the expression of sphingolipid-related enzymes without assessing the levels of the related sphingolipids.Our study suggests that sphingolipid metabolism is altered in HS lesional skin compared with normal skin.