Successful Treatment of Topical Tretinoin Cream in a Patient with Unilateral Nevoid Hyperkeratosis of Nipple and Areola

Corresponding author: Hsiu-Cheng Hsu, Department of Dermatology, Chang Gung Memorial Hospital, No. 199, Tunghwa N Rd., Taipei, 105, Taiwan TEL: 886-2-27135211 ext. 3397 FAX: 886-2-27191623 E-mail: [email protected] Funding source: none Confl ict of interest: none declared CASE REPORT A 53-year-old man presented with a 20year history of asymptomatic, progressive pigmented thickening of his left nipple and areola. He denied having any chronic illness. However, a diagnosis of glioblastoma multiforme had recently been made, and he had received three years of combination therapies, including subtotal excision, radiotherapy, chemotherapy, and photodynamic therapy, without any documented subsequent endocrine abnormality. Physical examination revealed a blackish verrucous plaque blanketing the left areola, and extending up the base of the left nipple (Fig. 1A, 1C). On palpation, the involved skin was neither indurated nor tender nor adherent to the underlying structures. There was no discharge from the nipple, and no regional lymphadenopathy, associated warts, ichthyosis, epidermal nevus, or acanthosis nigricans was noted. No family member had similar lesions. Histopathologic examination of a specimen from the left areola showed extensive and marked elongations of rete ridges of the epidermis, inducing filiform acanthosis and papillomatosis, orthokeratotic invaginating hyperkeratosis, and occasional keratotic plugging (Fig. 2). In addition, some anastomosing strands of elongated ridges and ectopic sebaceous glands were seen. The ectopic sebaceous glands were thought to be an accidental finding. The basal layer of the epidermis was hyperpigmented, but there was no melanocytic proliferation. The elongated papillary dermis was mildly edematous, and a sparse perivascular lymphocytic inflammatory infiltrate was found in the dermis. The histopathologic picture was compatible with a diagnosis of nevoid hyperkeratosis of nipple and areola. The patient received tretinoin cream per night topically use for a period of two months. The lesion improved significantly in pigmentation and minimally in thickness (Fig. 1B, 1D). No relapse has been observed during 6 months’ follow-up after discontinuation of topical agent.