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Hidradenitis suppurativa in association with keratitis–ichthyosis–deafness syndrome treated with adalimumab

Dear Editor, Hidradenitis suppurativa (HS) is a chronic and debilitating inflammatory skin disease that is characterized by painful lesions in apocrine gland-bearing areas. Treatments for HS include antibiotics, anti-tumor necrosis factor (TNF) drugs, systemic steroids, estrogens, anti-androgens, retinoids and incision. However, despite appropriate treatment, HS recurs frequently. Keratitis– ichthyosis–deafness (KID) syndrome is a rare congenital disorder of the ectoderm that gives rise to keratitis, erythrokeratoderma and neurosensory deafness. In addition to xerosis, HS has rarely been reported as a skin symptom accompanying KID. Antibiotics, acitretin, retinoids and anti-TNF drugs have been reported to treat it but cases of severe HS are usually refractory to treatment. To the best of our knowledge, this is the first case of HS in association with KID treated with adalimumab. A 43-year-old man came to our clinic suffering from a malodorous fistulating tract of the lower extremities and a purulent draining sinus of the groin embedded in keloidal tissue (Fig. 1a). He presented with ichthyosis cutis and diffuse palmoplantar keratoderma (Fig. 1c,d). He had been diagnosed with KID syndrome via sequencing analysis 11 years prior. The sequencing analysis showed a known polymorphism (–34C?T; National Center for Biotechnology Information reference single nucleotide polymorphism ID: 15 9578260), a heterozygous 148G?A transition in connexin 26 resulting in substitution of aspartic acid with asparagine in codon 50. Histological examination of the inguinal skin tissue showed marked perivascular plasmacytic infiltration in the upper and mid-dermis (Fig. 1f,g, h). Clinically and histologically, HS was diagnosed in association with KID syndrome.