Hyperkeratotic lesion of the nipple revealing cutaneous leiomyoma

BACKGROUND: Leiomyoma of the nipple and areola is a rare benign neoplasm. We report the case of a patient with leiomyoma of the nipple presenting as a hyperkeratotic plaque. OBSERVATION: A 23-year-old patient presented with a five year history of a papillomatous, hyperkeratotic, painful plaque originating in her right nipple. Histological examination of a punch biopsy showed hyperkeratosis of the epidermis with dilatation of the lymphatic vessels within the dermis. Surgical excision revealed a proliferation of smooth muscle fibres, leading to diagnosis of leiomyoma. DISCUSSION: The clinical and histological features were initially consistent with idiopathic naevoid hyperkeratosis of areola. However, associated pain is uncommon in idiopathic lesions. This unusual feature led us to surgical excision enabling the diagnosis of leiomyoma. A hyperkeratotic lesion of the nipple may be associated with benign or malignant neoplasms, hamartoma or chronic dermatoses, or it may be idiopathic. In the present case, the hyperkeratotic lesion revealed subareolar leiomyoma. This is an uncommon clinical presentation not previously seen in medical observations, since leiomyoma usually presents as a firm, painful lump in the subareolar region.     

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.     

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.     

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.     

Cutaneous leiomyoma of the male nipple

A case of cutaneous leiomyoma of the male nipple is reported. This is the second case reported by dermatologists to the best of our knowledge.     

面部多发性毛发平滑肌瘤一例

【摘要】 报告1例面部多发性毛发平滑肌瘤。患者男,43岁,右侧面部及额头部红色斑丘疹,伴冬季疼痛20年余,近4年加重。临床表现为粟粒至黄豆大小,高出皮面的丘疹和结节分布于右侧面颊部以及额部右侧,呈淡红色至红色,簇集状,质地坚实,基底稍呈浸润状。冰块实验阳性。患者无其他系统疾病。皮损组织病理检查：表皮大致正常,真皮内见肿瘤细胞不规则聚集或交织成束,肿瘤细胞细长,胞浆嗜酸,胞核两端钝圆或呈雪茄样,免疫组化显示平滑肌强阳性的特征：Desmin和SMA（+）,结合临床诊断多发性毛发平滑肌瘤。由于该患者皮损面积大,未能手术切除,随访。     

Cutaneous leiomyosarcoma originating in a symplastic pilar leiomyoma: a rare occurrence and potential diagnostic pitfall

A case of cutaneous leiomyosarcoma originating in a symplastic pilar leiomyoma is described. The patient was a 60‐year‐old man who presented with a painless nodule on his chest. Thorough clinical examination did not reveal any evidence of tumor elsewhere. A punch biopsy was performed which showed a benign spindle cell neoplasm with focal cellular pleomorphism that had smooth muscle differentiation and was clearly originating from the arrector pili muscle, consistent with a cutaneous symplastic pilar leiomyoma. Immunohistochemical studies were performed and the tumor cells were strongly positive for smooth muscle actin (SMA) and desmin. Given the clinical information of a mass on the chest, clinical evaluation was recommended and a re‐excision was performed. Histologically, the re‐excision showed two distinct populations of cells. The upper portion of the tumor mirrored the initial biopsy; however, the base of the tumor showed hypercellular areas composed of spindle cells with marked pleomorphism and increased number of mitoses. The diagnosis of a cutaneous leiomyosarcoma originating in a symplastic pilar leiomyoma was given. This is the first report of this association of such occurrence reported in the literature. Fons ME, Bachhuber T, Plaza JA. Cutaneous leiomyosarcoma originating in a symplastic pilar leiomyoma: a rare occurrence and potential diagnostic pitfall.     

A case of cutaneous symplastic leiomyoma - a rare variant of cutaneous pilar leiomyoma

We describe the case of a cutaneous symplastic leiomyoma in a 37-year-old woman who presented with a 4-year history of a painful slow growing lesion on the left upper arm. The lesion was excised and subjected to histological examination. A poorly circumscribed lesion was seen in the dermis composed of spindle shaped cells with marked nuclear pleomorphism. No mitotic figures or necrosis were seen. The cells stained strongly positive with desmin and smooth muscle actin, and negative with S100, melan A, MNF116 a mouse monoclonal antibody to cytokeratin and CK5/6. The diagnosis was felt to be in keeping with a cutaneous symplastic leiomyoma, a rarely reported variant of the pilar leiomyoma. Histologically, it shows features similar to the symplastic variant of uterine leiomyoma with cytological atypia, nuclear pleomorphism and minimal mitotic activity. Although the long-term outlook is probably benign, the presence of cytological atypia and mitoses in any spindle cell tumor is generally a concerning feature and warrants long-term follow up.     

Scrotal leiomyoma

Genital leiomyomas of the scrotal skin are extremely rare benign tumors, originating from the tunica dartos of the scrotum. We describe a 55-year-old caucasian male with a 3-year history of a slowly growing asymptomatic scrotal knot which was first clinically regarded as a cyst and finally diagnosed histopathologically as leiomyoma. This typical case demonstrates that leiomyomas should be considered in the differential diagnosis of scrotal tumors.     

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.

     

Association of multiple familial cutaneous leiomyoma with a uterine symplastic leiomyoma

We describe a patient who has familial cutaneous leiomyoma in association with a symplastic uterine leiomyoma. This association has not been described previously.     

Vascular Leiomyoma of the Scalp with a Small Deformity on the Skull Mimicking a Dermoid Cyst

Abstract: We report a vascular leiomyoma of the extracranial scalp with a small deformity on the skull in a child mimicking a dermoid cyst. The tumor was removed surgically, and histologic examination revealed a vascular leiomyoma. A small deformity‐like dimple on the skull improved without evidence of recurrence. The early surgical excision yielded good results.     

Cryosurgery as a Minimally Invasive Alternative Treatment for a Patient with Erosive Adenomatosis of the Nipple

Erosive adenomatosis of the nipple (EAN), also known as nipple adenoma, florid papillomatosis, or papillary adenoma of the nipple, is a benign neoplasm originating from a lactiferous duct of the breast. Although the potential for malignant change is invariably negligible, the nature of the disease is quite intractable despite several treatment methods. Surgical excision is known as the treatment of choice, but this invasive approach is generally not acceptable to the vast majority of patients due to the cosmetic outcomes. Cryosurgery could be an alternative choice to preserve the structure of the nipple-areola complex, though its application has not been studied due to the paucity of cases. A 22-year-old female presented with a unilateral, crater-like erosion of the left nipple with serosanguineous discharge. The skin biopsy revealed proliferation of tubular structures, which corresponded to EAN. She was treated with 4 sessions of cryosurgery (open cryospray with liquid nitrogen) over 6 months, and the skin lesion resolved completely without any recurrence for 12 months. Although further study is required to determine the optimal treatment regimen for EAN, cryosurgery should be considered as an effective option to surgical excision.     

无痛性多发性皮肤平滑肌瘤一例

患者,女,19岁。右上肢多发结节半年,左上肢多发结节1个月。皮肤组织病理示真皮可见束状及团块状平滑肌束。免疫组化证实过度增生的纤维为平滑肌。根据临床表现及组织病理结果,诊断为无痛性多发性皮肤平滑肌瘤。     

Multiple leiomyoma

A case of asymptomatic multiple leiomyoma along with its histopathological features is described.     

Papillomatous lactic-duct adenoma (pseudoPaget's disease of the mamilla)]

Papillomatosis and adenomatosis of the nipple ducts can be interpreted clinically as Paget's disease. Three patients with subareolar papillomatosis and adenomatosis are described. The name papillary adenoma of the nipple ducts (pseudo-Paget's disease has been proposed. The differentiation between the papillary adenoma of the nipple ducts and the genuine Paget's disease needs histological examination. The differential diagnosis is very important, because local excision of the diseased ducts represents the treatment of choice of the papillary adenoma of the nipple ducts. Radical mastectomy is always necessary in genuine Paget's disease since the adjacent lactiferous ducts are involved by the carcinoma.     

Positive correlation of vanilloid receptor subtype1 and prostaglandin E2 expression with pain in leiomyomas

Cutaneous leiomyomas are benign smooth muscle tumors that are occasionally painful. The mechanism of pain related to leiomyoma is not fully understood. To investigate the possible involvement of algoneic factors in pain from cutaneous leiomyomas. We present a case of cutaneous leiomyoma with severe, diffused pain in a large area and collected 10 more specimens of cutaneous leiomyoma with or without pain in patient histories. We immunohistochemiacally examined the expression of algoneic factors: serotonin, histamin, Substance P, PGE2, BDKRB2, VR1 and CGRP. We compared the pain area and expression of algoneic factors to reveal possible correlations. We describe here a patient with a cutaneous leiomyoma 1‐cm in diameter, which caused severe pain diffused throughout an area of 20‐cm around the tumor. The pain completely resolved after surgical excision of the leiomyoma. We observed that the leiomyoma cells expressed CGRP, PGE2 and VR1 in this case. We found a positive correlation between VR1 and PGE2 expression in the leiomyoma cells and areas with pain around the tumors among 11 specimens in total. VR1 and PGE2 might be key algogenic substances in painful leiomyoma.     

Leiomyoma of the breast in the male

A macroscopically not sure classifiable solitary tumour on the nipple was observed in a 68 year old man. Histologically a pure leiomyoma was found. Therapy of choice is the tumour resection, if requested.     

Erosive adenomatosis of the nipple: Conservation of nipple by Mohs micrographic surgery

Erosive adenomatosis of the nipple (EAN) is a rare, benign tumor that resembles Paget's disease and well-differentiated adenocarcinoma. Total excision of the nipple has been the usual treatment modality because of the high incidence of recurrence when removal is incomplete, and complete excision generally mandates a reconstructive procedure. We report a case of a 56-year-old woman with EAN treated with Mohs micrographic surgery (MMS). We suggest that MMS is the first choice of treatment for EAN because it can be effectively used to remove the tumor completely and at the same time preserve the nipple, if performed early enough, before the tumor is widely spread.     

Palisaded and verocay body prominent leiomyoma of deep soft tissue

Leiomyoma of deep soft tissue is a very rare tumor, which is sometimes confused with leiomyosarcoma, histopathologically. We describe a case of palisaded and Verocay-body prominent leiomyoma of deep soft tissue. The lesion was characterized by areas of nuclear palisading with Verocay-body like formation in addition to the features of leiomyoma.