Folliculosebaceous Cystic Hamartoma on the Nipple

Abstract

Dear Editor, Folliculosebaceous cystic hamartoma (FSCH) is a rare skin lesion comprised of follicular, sebaceous and mesenchymal elements. FSCH mostly involves the head and neck area but some cases involving the upper back, forearm and ear have been reported. However, FSCH located on the nipple is quite rare. Herein, we describe a case of FSCH on the nipple of a 32-year-old woman.A 32-year-old woman visited our clinic to evaluate a single pedunculated soft nodule on the nipple of her right breast. The nodule had been there for 5 years. It was asymptomatic and very slowly growing. The surrounding areola and underlying mammary ducts and breast tissue were unremarkable. There was no history of nipple discharge or bleeding. Her left breast was normal.On physical examination, the lesion was a 0.5 cm · 0.4 cm · 0.3 cm, light brown, soft tumor with a projecting and polypoid configuration, located on the superolateral aspect of the right nipple. There was no evidence of punctum or sinus opening and involvement of ductal orifice was not noticed (Fig. 1). Initial clinical diagnosis was fibroma of the nipple and accessory nipple, and an excisional biopsy was performed.The tumor was totally excised by a 6-mm punch. Histopathological examination revealed mild epidermal proliferation and hyperplasia. Within the dermis, there were multiple hyperplastic sebaceous glands forming nodules around a large, cysticallydilated, follicular infundibular structure lined with stratified squamous epithelium and filled with keratin material. Sebaceous lobules were attached to the cystic structure through sebaceous ducts. The surrounding stroma consisted of concentric, fibrillary bundles of collagen with numerous dilated capillaries and venules. The perifollicular part of the stroma contained large amounts of mucin. There was cleft formation between the perifollicular stroma and the adjacent dermis (Fig. 2). The final diagnosis was folliculosebaceous cystic hamartoma and the lesion has not recurred after total excision.Folliculosebaceous cystic hamartoma was first described in 1991 by Kimura et al.1 and was con-

Giant genital variant of folliculosebaceous cystic hamartoma: successful management by CO2laser and acitretin therapy

“…Moreover, it is tempting to speculate that the large size might, at least in part, be explained by the higher age of the patient and may reflect a possible evolution of FCSH with age. The histological features of our case were highly similar to those of the other described cases of genital FCSH 2–4 . Histologically, irregularly shaped cystic structures were seen in the dermis, and most of them were connected to the surface of the skin.…”

“…FCSH has a striking predilection for the central face and scalp of adults, with the vast majority of lesions presenting as 0·5–1·5 cm exophytic, partly umbilicated nodules 1–3 . Other locations such as the nipple or the genital area have rarely been reported for FSCH 4–6 . We present a patient with a genital variant of FCSH measuring > 15 cm in diameter and involving the whole scrotum.…”

Folliculosebaceous Cystic Hamartoma in a Patient with Neurofibromatosis Type I

“…It is relatively rare. It usually occurs on the central face and scalp, back, upper extremity, genital area and, rarely, the nipple (where there is no hair follicle)3-6. The ages of patients range from 4 to 84 years8.…”

“…Lesions usually present as solitary papules or nodules with a predilection for the central face and scalp2. Other parts such as the trunk, the genital area or rarely the nipple have been reported for FSCH3-6. It is considered by some authors as a variant of sebaceous trichofolliculoma (STF)7.…”

Cutaneous Solitary Neural Hamartoma: Report of an Unusual Case

“…8 Folliculosebaceous cystic hamartomas usually have a mixture of adnexal structures, sometimes with cystic formation, and other mesenchymal elements including mature nerve bundles. [7][8][9][10][11][12][13] Mature nerves have been reported as a component of neuromuscular hamartoma, which includes, as the name implies, striated muscle component as well. This entity is also known as benign triton tumor or neuromuscular choristoma.…”