Case of unilateral epidermal nevi without extracutaneous anomalies

Abstract

Dear Editor, Epidermal nevi are noted as linear tan patches or thin plaques. The linear pattern often follows Blaschko's lines, which are considered to represent patterns of epidermal migration during embryogenesis. Histologically, epidermal nevi exhibit hyperkeratosis and papillomatosis. The condition usually appears at birth or within the first year of life, but on rare occasions, it develops until later in childhood. 1 Here, we report a case of epidermal nevi limited to the right side of the body in an adult male.A 36-year-old Japanese man was referred to our department for evaluation of his unilateral dermatitis that had begun in early childhood and became significant at the age of 15 years. He was first diagnosed with atopic dermatitis and had been treated with topical steroids. He had no family history of genetic disorders. Physical examination showed a striking lateralization diffusely affecting only the right side of the body, with strict midline demarcation. The affected side exhibited ichthyosiform erythroderma partially covered with waxy and yellowish scales (Fig. 1a). He did not exhibit alopecia, neurological abnormalities, limb hypoplasia or skeletal (a) (b) (c) (d) Figure 1. Clinical manifestations and histology of the patient. (a) Ichthyosiform erythroderma partially covered with waxy and yellowish scales with striking lateralization diffusely affecting only the right side of the body with strict midline demarcation at the initial physical examination. He had mild itchy and hyperhidrosis of the right side of the body. (b) Skin biopsy from the right precordium revealed hyperkeratosis and perivascular lymphocytic infiltration (hematoxylin-eosin [HE], original magnification 940). (c,d) Skin biopsy from a thick nodule of the buttock reminiscent of psoriasis (HE, original magnifications: [c] 9100; [d] 9200).

A case of unilateral epidermal nevi complicated by atopic dermatitis treated with dupilumab

“…The disease usually presents at birth or within the first year of life, but rarely later in childhood. We have previously reported adult-onset epidermal nevi localized on the right side of the body which was also complicated by atopic dermatitis (AD) [1] . Here, we report a case of epidermal nevi improved by the use of dupilumab for AD.…”