Genital leiomyomas of the male nipple: two cases

BACKGROUND: Genital leiomyoma is a rare benign solitary skin tumor, not painful, developed from smooth muscle. Genital leiomyoma arising from the nipple is extremely rare, especially in males.CASES REPORT: A 47 year-old male had a 2 cm cutaneous plaque with nodules located on the right nipple. This plaque was circumscribed, erythematous, pruriginous and was not painful. The lesion had been noticed by the patient two years ago. A 37 year-old male showed a 1.5 cm cutaneous plaque located on the left nipple. The plaque was slightly erythematous, pruriginous, not painful and had been noticed by the patient 5 years earlier. Histology provided the diagnosis of genital leiomyoma in both cases. No surgical therapy was performed.DISCUSSION: Cutaneous leiomyomas are classified in 3 types regarding their origin: multiple or solitary piloleiomyoma, arising from arectores pilorum muscles, solitary genital leiomyoma, arising from the dartoic, vulvar, or mammillary muscles, and solitary angioleiomyoma, arising from the vein muscles. Clinically, genital leiomyoma is a 1 cm diameter solitary erythematous, firm nodule. According to many authors, genital leiomyoma is asymptomatic, but in the 2 patients, the lesions were pruriginous. Surgical excision is usually performed.     

Nevoid hyperkeratosis of the nipple and areola: a distinct entity

Although nevoid hyperkeratosis of the nipple and areola was initially described in 1923, there are only case reports or reviews about it; no large series have been documented to date. The clinical features of the reported cases in the literature are not uniform, and it is questioned whether nevoid hyperkeratosis of the nipple and areola is a distinct clinicopathologic entity or a clinical presentation of various dermatoses. We describe 7 cases with hyperkeratotic nevoid lesions localized on the nipple and areola with different clinical features. None of them had any other associated dermatologic or systemic disease. Histopathologic examination was performed in 6 patients. Four of them had common histopathologic features suggesting a distinct entity, namely, nevoid hyperkeratosis of the nipple and areola; 2 of them had histopathologic features consistent with seborrheic keratosis. Seborrheic keratosis presents as sharply demarcated papules or plaques, whereas nevoid hyperkeratosis of the nipple or areola presents as a plaque diffusely involving the nipple or the areola.     

Nevoid hyperkeratosis of the areola misinterpreted as mycosis fungoides

Nevoid hyperkeratosis of the nipple and areola is a benign condition with fewer than 70 cases reported in the literature. We report a case of unilateral nevoid hyperkeratosis of the areola with intraepidermal lymphocytes that resembled Pautrier's microabscesses on histological examination. This is the third report of mycosis fungoides-like changes in nevoid hyperkeratosis of the nipple and areola. In addition, this is the first case to present immunohistochemical and T-cell gene rearrangement studies of the intraepidermal lymphocytes. This case highlights a potential histopathological pitfall in the diagnosis of nevoid hyperkeratosis of the nipple and areola.     

Nevoid hyperkeratosis of male breast: Successful treatment with minocycline

Nevoid hyperkeratosis of the nipple and/or areola (NHNA) is an uncommon disease with no definite etiology. NHNA of the male breast is rare in clinical practice. Despite being a benign disease, it is distressing for patients and therapeutically challenging for clinicians. We report a male patient with NHNA who responded favorably to minocycline treatment.     

Vemurafenib‐induced hyperkeratosis of the areola treated with topical adapelene

We present a rare condition, hyperkeratosis of the areola, induced by vemurafenib. Only a few papers have described an association of BRAF inhibitors with hyperkeratosis of the areola and/or nipple. Vemurafenib is a selective BRAF inhibitor used in patients with unresectable or metastatic melanoma who are positive for the V600 mutation. This drug has been associated with numerous cutaneous side effects, both benign and malignant. We report a male patient with vemurafenib‐induced hyperkeratosis of the areola managed successfully with a topical retinoid, and describe for the first time a treatment for this side effect.     

Nevoid hyperkeratosis of the nipple and/or areola: a report of two cases and a review of the literature

Nevoid hyperkeratosis of the nipple and/or areola is a rare and idiopathic disorder with fewer than 50 cases reported in the literature. We report two cases, discuss the clinical and histological findings, and review the literature pertaining to its pathogenesis and therapy. It is hoped that, through this report, nevoid hyperkeratosis of the nipple and/or areola becomes a more recognizable and reported entity, thereby stimulating future studies regarding its pathogenesis and therapy.     

Painful nipple hyperkeratosis secondary to vemurafenib

Vemurafenib is a selected BRAF kinase inhibitor approved for treating metastatic or unresectable melanoma, which has numerous cutaneous side effects unfortunately, including three previously reported cases of asymptomatic areola and/or nipple hyperkeratosis. We present the first case of painful bilateral nipple hyperkeratosis secondary to vemurafenib in an 84‐year‐old woman. She was successfully treated with tretinoin 0.05% cream that allowed her to comfortably continue treatment. With increased awareness of this condition, we found a second case of asymptomatic nipple hyperkeratosis secondary to vemurafenib in our clinic. As this medication gains acceptance for treatment of metastatic melanoma, it is imperative that dermatologists are aware of this potentially uncomfortable side effect that can result in decreased compliance and impaired quality of life.     

Nipple leiomyoma: A rare neoplasm with a broad spectrum of histologic appearances

Cutaneous leiomyomas are rare benign smooth‐muscle tumors. These lesions are distinguished based on their cell of origin and are subclassified as pilar leiomyoma, angioleiomyoma, and genital‐type leiomyoma. Nipple leiomyoma is the least common genital‐type leiomyoma, arising from the dartoic muscle cell of the nipple. Histologic examination of the lesion is necessary for definitive diagnosis, and these uncommon tumors can pose a diagnostic challenge. We describe herein a series of six nipple leiomyomas with a spectrum of histologic appearances.     

Focal nevoid hyperkeratosis of the nipple in a prepubertal girl

Nevoid hyperkeratosis (NH) of the nipple and areola is an extremely uncommon condition in prepubertal children. We report on a prepubertal girl with NH and provide dermoscopic findings that helped to make the diagnosis.     

Genital leiomyoma: surgical excision for both diagnosis and treatment of a unilateral leiomyoma of the male nipple

We describe a rare case of unilateral leiomyoma of the nipple in a man presenting with pruritus of the nipple for 3 months. A conservative surgical excision is performed for diagnosis. Histologic examination and immunostaining confirmed the diagnosis of leiomyoma. The leiomyoma is completely excised, and the patient denies pruritus after surgery. Conservative surgical excision of a persistently hard and pruritic nipple can be effectively used as both diagnostic measure and treatment modality.     

Pseudoepitheliomatous keratotic and micaceous balanitis treated with topical 5‐fluorouracil and liquid nitrogen

Pseudoepitheliomatous keratotic and micaceous balanitis (PKMB) is an uncommon premalignant condition involving the glans penis. We report the case of an 86‐year‐old man who presented with phimosis and pain on retracting his foreskin. He had previously undergone circumcision, which revealed a hyperkeratotic plaque with thin mica‐like scales involving his glans penis. Histology of the lesion showed hyperkeratosis, parakeratosis, epidermal acanthosis and papillomatosis with no evidence of dysplasia. Immunohistochemistry for human papillomavirus was negative. The patient was treated with topical 5‐fluorouracil and liquid nitrogen with clinical improvement. He is now under long‐term surveillance for verrucous carcinoma and squamous cell carcinoma.     

Olmsted综合征与1例文献复习

报告1例Olmsted综合征。男,21岁,因双侧进行性掌跖角化和腔口周围角化性斑片就诊。患者自小曾在当地医院以湿疹、皮炎等治疗,并使用各种药膏(名不详),无明显疗效,病情进行性加重,表现为边缘锐利的残毁性掌跖角化、指(趾)压缩,全身泛发毛囊性角化性灰白色丘疹,甲床充满白色、坚硬的角化性碎片,同时还有乳头乳晕角化过度和普秃。     

Leiomioma de la areola mamaria. Presentación de dos casos

—Cutaneous genital or dartoic leiomyomas are rare benign tumors originating in the smooth muscle fibers of the skin of the scrotum, vulva, nipple and areola. The latter are the less frequently observed.We report two cases of leiomyoma of the areola. Both presented as a solitary, small, nonulcerated nodule. Histopathological features shared similar characteristics: a non-encapsulated, ill-defined tumor in superficial and mid-dermis composed of spindle cells with elongated nuclei and eosinophilic cytoplasm, arranged in interdigitating fascicles. There were no atypia or mitoses. It was remarkable a reactive epidermal hyperplasia overlying the surface of the tumor, as is typically seen in the histiocytomas. Areolar leiomyoma is a benign lesion that is not known to undergo malignant transformation, although it may be confused clinically with carcinoma of the breast.     

双侧巨大型乳头乳晕角化过度症

报告1例双侧巨大型乳头乳晕角化过度症。患者女，24岁。乳晕褐色斑10年，加重半年。患者自幼左侧乳头看不见。体格检查：双侧乳晕粗糙、增厚，呈乳头瘤样增生，右侧尤为明显。组织病理检查示表皮乳头瘤样增生，皮突延长并融合。诊断：乳头乳晕角化过度症。     

Nevoid hyperkeratosis of the nipple and areola: treatment with topical retinoic acid

Nevoid hyperkeratosis of the nipple and areola is a rare dermatosis with unknown etiology, (Perez-Izquierdo JM, Vilata JJ, Sanchez JL, et al. Retinoic acid treatment of nipple hyperkeratosis. Arch Dermatol 1990;126:687-688). Only 40 cases have been reported until 1997 (Alpsoy E, Yilmaz E, Aykol A. Hyperkeratosis of the nipple: report of two cases. J Dermatol 1997;24:43-45). The disease has a benign course and may only be a cosmetic problem. Different modalities have been used in the treatment of NHNA. In our case treatment with topical retinoic acid induced an acceptable response.     

Nipple adenoma in a female patient presenting with persistent erythema of the right nipple skin: case report,review of the literature,clinical implications,and relevancy to health care providers who evaluate and treat patients with dermatologic conditions of the breast skin

Background

Nipple adenoma is a very uncommon, benign proliferative process of lactiferous ducts of the nipple. Clinically, it often presents as a palpable nipple nodule, a visible nipple skin erosive lesion, and/or with discharge from the surface of the nipple skin, and is primarily seen in middle-aged women. Resultantly, nipple adenoma can clinically mimic the presentation of mammary Paget’s disease of the nipple. The purpose of our current case report is to present a comprehensive review of the available data on nipple adenoma, as well as provide useful information to health care providers (including dermatologists, breast health specialists, and other health care providers) who evaluate patients with dermatologic conditions of the breast skin for appropriately clinically recognizing, diagnosing, and treating patients with nipple adenoma.

Case presentation

Fifty-three year old Caucasian female presented with a one year history of erythema and induration of the skin of the inferior aspect of the right nipple/areolar region. Skin punch biopsies showed subareolar duct papillomatosis. The patient elected to undergo complete surgical excision with right central breast resection. Final histopathologic evaluation confirmed nipple adenoma. The patient is doing well 31 months after her definitive surgical therapy.

Conclusions

Since nipple adenoma represents a benign proliferative process of the nipple, complete surgical excision is curative. However, the coexistence of nipple adenoma and ipsilateral or contralateral breast cancer is well reported in the literature. The potential for a direct causal link or association of nipple adenoma and breast cancer cannot be fully excluded.

     

Nevoid hyperkeratosis of the nipple and areola: treatment of two patients with topical calcipotriol.

Nevoid hyperkeratosis of the nipple and areola, which is characterized by verrucous thickening and pigmentation of the nipple or areola, is a rare condition. Different therapeutic options have been used with varying results, but there is no uniformly effective treatment. We describe two patients with hyperkeratosis of the nipple and areola who responded well to topical calcipotriol ointment.     

Psoriasis with a dirt‐adherent: A rare case report

Cutaneous dirt‐adherent disease (CDAD) is a rare psychogenic dermatosis mainly occurring in young Japanese and Chinese women. It mainly occurs on cheeks, forehead, nipple, mammary areola and around mammary areola. To our knowledge, this is the first case of CDAD with the skin lesion of psoriasis rupioides to be reported. In our case, the patient, a 43‐year‐old Chinese man presented with thick, yellowish‐brown adherent crusts on his face with severe painful 6‐days duration. Histopathologic image: Parakeratosis, the epidermis demonstrates regular acanthosis with some thinning of the suprapapillary plates, neutrophils exocytosis are noted. As for the histopathologic diagnosis in his right crus, combined with the clinical manifestation of rupioides‐shaped crusts, film phenomenon and Auspitz's sign, we considered Psoriasis rupioide.     

Two cases of male nipple leiomyoma: idiopathic leiomyoma and gynecomastia-associated leiomyoma

We describe 2 cases of male nipple leiomyoma. A 70-year-old man had a painful subcutaneous tumor on his left nipple of 6 months duration. Histopathology disclosed dermal spindle cells with oval-shaped nuclei forming interlacing bundles with irregular pattern. Glandular elements were absent. The spindle cells were positive to α-smooth muscle actin, desmin, and vimentin. Estrogen receptor (ER) and progesterone receptor (PrR) were negative. We diagnosed this case as male leiomyoma of the nipple. Another patient was a 61-year-old man with gynecomastia induced by spironolactone of 6 months duration. He also had a painful nodule on his left nipple and histopathology disclosed spindle-shaped tumor cells as in the previous patient. The tumor was accompanied by glandular elements in the deep dermis and subcutaneous tissue, which showed apocrine secretion and were positive for α-smooth muscle actin, ER, and PrR. These glandular elements were interpreted as mammary gland. But ER and PrR stain did not show positive results for leiomyoma in the upper dermis. To the best of our knowledge, this is the first report of male idiopathic and gynecomastia-induced leiomyoma with ER and PrR staining.     

Hyperkeratosis of the nipple and areola

Hyperkeratosis of the nipple and areola is a rare condition. We report two cases of hyperkeratosis of the nipple and areola occurring in men with no underlying endocrinopathy or synthetic estrogenic drug therapy. Both patients demonstrated prompt resolution of the hyperkeratosis of the nipples with a keratolytic gel. Because our cases were not associated with ichthyosis or epidermal nevus, they best fit into the category of nevoid hyperkeratosis of the nipples.