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[Malignant nodular hidradenoma and multiple nodular hidradenomas in a hypogonadic patient].

BACKGROUND: We report a case of multiple benign nodular hidradenomas associated with malignant nodular hidradenoma in a hypogonadic patient. CASE-REPORT: A 49 year-old man presented 11 benign nodular hidradenomas in the pectoral region. A malignant nodular hidradenoma had recently appeared in this region some distance from the pre-existing benign modular hidradenomas. This patient had peripheral hypogonadism associated with congenital bilateral cryptorchidia responsible for gynecomastia due to "relative hyperestrogenism". Large numbers of estrogen receptors were demonstrated in each of the benign nodular hidradenomas. The malignant nodular hidradenoma mass was unavailable and screening for these receptors was not possible. DISCUSSION: Cases of multiple nodular hidradenoma are extremely rare: we have seen only two. To the best of our knowledge, there have been no reports to date of combined malignant nodular hidradenoma and multiple benign nodular hidradenoma. However, there is histological evidence of transition forms. The coexistence of endocrine anomalies associated with this type of tumour has never been reported. However, the presence of estrogen receptors has occasionally been demonstrated in benign nodular hidradenomas. Our patient's endocrine disease may have played a role in the presentation of multiple hidradenomas, but this cannot be demonstrated.

Adenoma, Sweat Gland↗

[Hidradenoma of the skin. Synoptic reveiw of syringeal hidradenoma, eccrine epithelioma and eccrine basalioma (author's transl)].

An ulcerating and sclerosing skin tumour on the temporal forehead region is described. Histologically the tumour is composed of different structures as cystic hidradenoma, syringoma, ductal hidradenoma and sclerres as cystic hidradenoma, syringoma, ductal hidradenoma and sclerosing basalioma arranged in layers. The tumour appears to be a distinct and diagnosable entity. It exists a major similarity to such tumours reported as syringeal hidradenoma, eccrine epithelioma or eccrine basalioma.

Adenoma, Sweat Gland↗

Ectopic hidradenoma papilliferum: a case report and review of the literature.

Hidradenoma papilliferum is a benign, cystic, papillary tumor that occurs almost exclusively in women on the skin of the anogenital region. Nonanogenital (ectopic) hidradenoma papilliferum are rare. We describe a 72-year-old white man with an enlarging nodule in the region of the right triceps muscle; microscopic examination showed a hidradenoma papilliferum. The median age of patients with ectopic hidradenoma papilliferum is between 1 to 2 decades older than the average age range of lesion onset in patients with anogenital hidradenoma papilliferum. In contrast to anogenital hidradenoma papilliferum, nearly one half of the patients with ectopic hidradenoma papilliferum are men. Ectopic hidradenoma papilliferum occurs most frequently (60%) in the head and neck region. Eighty-five percent of cases are 1.5 cm in the greatest diameter or smaller. The race, clinical features, pathologic features, treatment, and prognosis for hidradenoma papilliferum occurring in anogenital and ectopic locations are similar.

Adenoma, Sweat Gland↗

Papillary hidradenoma: immunohistochemical analysis of steroid receptor profile with a focus on apocrine differentiation.

AIM: To make a quantitative evaluation by image analysis of oestrogen receptors, progesterone receptors, and androgen receptors in papillary hidradenomas and anogenital sweat glands. METHODS: 20 papillary hidradenomas and the anogenital sweat glands detected in surgical specimens selected from 10 vulvectomies for squamous carcinoma, eight haemorrhoidectomies, and one anal polypectomy, all from female patients, were investigated by the avidinstreptavidin peroxidase testing system. RESULTS: 90% of papillary hidradenomas and almost all the anogenital sweat glands showed immunoreactivity for oestrogen receptor and, more weakly, for progesterone receptor, with immunolabelled nuclear area ranging from 10% to 90%. Conversely conventional sweat glands did not show any nuclear staining. Overexpression of androgen receptors occurred in 20% of papillary hidradenomas, with nuclear staining strictly bordering papillary epithelium with apocrine differentiation. There was no immunoreactivity for androgen receptors in anogenital sweat glands. CONCLUSIONS: Oestrogen and progesterone receptors seem to represent reliable markers for differentiating between anogenital sweat glands and conventional sweat glands, and a further link to explain why papillary hidradenomas occur almost exclusively in the female anogenital region. Positivity for oestrogen/progesterone receptors suggests that epithelia either of anogenital sweat glands or of papillary hidradenomas are controlled by ovarian steroid hormones. Androgen receptor nuclear staining of the epithelium with apocrine differentiation in vulvar papillary hidradenoma strengthens its homology with breast duct papilloma.

Adenoma, Sweat Gland↗

Immunohistochemistry in the differential diagnosis of nodular hidradenoma and glomus tumor.

The histologic distinction between nodular hidradenoma and glomus tumor is an occasional difficult diagnostic problem. Both tumors may show circumscribed aggregates of uniform epithelioid cells, a myxoid stroma, and variable numbers of blood vessels. Especially troublesome are solid cellular hidradenomas without duct-like structures and glomus tumors without a vascular pattern. To develop an immunohistochemical profile useful in this differential diagnosis, 25 selected skin tumors and four normal glomus bodies were studied with antibodies against low molecular-weight cytokeratin (CAM 5.2), epithelial membrane antigen (EMA), carcino-embryonic antigen (CEA), S-100, and vimentin (VIM). The tumors included eight unequivocal hidradenomas, seven unequivocal glomus tumors, and 10 histologically equivocal cases, originally diagnosed as glomus tumors. In all unequivocal glomus tumors and glomus bodies, only VIM was positive. Of the eight unequivocal hidradenomas, three were positive for CAM 5.2, EMA, CEA, S-100, and VIM; two for CAM 5.2 only; one for CAM 5.2, EMA, and S-100; one for CAM 5.2, EMA, and CEA; and one for CEA only. In the histologically equivocal cases, eight were positive for VIM only, characteristic of glomus tumor; and two were positive for CAM 5.2, EMA, CEA, S-100, and VIM, and were reclassified as hidradenomas. The study suggests that morphologic criteria may not always accurately differentiate between hidradenoma and glomus tumor and that in equivocal cases immunohistochemistry may be useful in the differential diagnosis.

Adenoma, Sweat Gland↗

Clear cell hidradenoma associated with the folliculo-sebaceous-apocrine unit. Histologic study of five cases.

Hidradenomas are benign adnexal neoplasms of uncertain derivation. Although most investigators traditionally have considered these neoplasms to exhibit eccrine differentiation, with only occasional reports supporting apocrine derivation, subdivision of hidradenomas into two groups was recently suggested: those with eccrine differentiation (or poroid hidradenomas) and those exhibiting apocrine differentiation (or clear cell hidradenomas). We have observed five cases of clear cell hidradenomas manifesting unquestionable apocrine characteristics excised from sites other than the axilla, and displaying continuity with the epidermis through follicles. Because of the common embryologic derivation of apocrine glands, sebaceous glands, and hair follicles, and their histologic integration as the folliculo-sebaceous-apocrine unit, these findings suggest a possible origin of certain apocrine hidradenomas from pluripotential germinative cells present in follicles.

Acrospiroma↗

Histogenesis of clear cell hidradenoma: immunohistochemical study of keratin expression.

The expression of cytokeratins in 10 cases of clear cell hidradenoma, including 3 cases of solid cystic hidradenoma, were examined using 21 kinds of monoclonal antibodies. We divided them into three histologic patterns: massive nests with a few lumina (M nests), nests with some tubular lumina (L nests), and nests in solid cystic hidradenomas (S nests). All hidradenomas showed similar immunoreactivities to those in the lower dermal ducts or secretory cells of normal eccrine glands. With antibodies against simple epithelial cytokeratins (CKs 7, 8, 18, and 19), however, different immunostaining was noted among the three histologic patterns. Namely, the M nests failed to react to them, although some luminal cells in the L nests revealed a positive staining. Furthermore, a majority of luminal cells in the S nests revealed a positive staining with them. Therefore, we think that the luminal cells in solid cystic hidradenoma mainly differentiate toward the secretory cells, and that the M nests mainly differentiate toward the dermal duct. Those in the L nests are thought to differentiate toward the dermal duct and the secretory cells. The proportion of the differentiation toward luminal cells of dermal ducts to the differentiation toward secretory cells was the main difference among the three nests. In addition, there was no difference in immunophenotypes between clear cells and epidermoid cells in the two kinds of hidradenomas.

Acrospiroma↗

Hidradenoma papilliferum with oxyphilic metaplasia: a clinicopathological study of 18 cases, including detection of human papillomavirus.

Reported here are 18 cases of hidradenoma papilliferum with oxyphilic metaplasia. All patients were women ranging in age from 29 to 74 years. Each presented clinically with a small, solitary tumor in the anogenital region. Microscopically, in addition to classic histopathological features, in every case there was oxyphilic metaplasia of the constituent epithelial cells. This finding could be likened to apocrine metaplasia, a term used in breast pathology. Other histopathological findings observed in this series, analogous to benign breast disease, included sclerosing adenosis-like changes, atypical apocrine adenosis-like changes, changes corresponding to usual ductal epithelial hyperplasia, epitheliomatosis with a streaming growth pattern, lamprocyte-like changes, clear cell change of the myoepithelium, foamy histiocyte reaction, and stromal fibrosis. Immunohistochemistry inferred that in the majority of cases oxyphilic metaplasia resulted from more lysosomes, whereas numerous mitochondria were detected in only 3 cases. Using 2 different PCR methods we identified HPV in 4 of 15 cases of hidradenoma with oxyphilic metaplasia. In addition, HPV was detected in 3 of 16 conventional papillary hidradenomas used as a control group. The following HPV types were identified: 16, 31, 33, 53, and 56. The last type was found in 5 cases. More than one HPV type from a single lesion was seen in 5 cases. Our observations are consistent with previous publications noting similarities between tumors of the breast and sweat glands. Oxyphilic metaplasia, areas with solid growth, and changes simulating atypical apocrine adenosis are rare and poorly recognized in hidradenoma papilliferum and may cause diagnostic difficulties; in our cases several submitting pathologists suspected malignancy. A causal role for HPV in hidradenoma papilliferum cannot be confirmed from our results, as the detection rate is too low. The exact role of the HPV in etiology and pathogenesis of this neoplasm has yet to be determined.

Adenoma, Sweat Gland↗

A single lesion demonstrating features of eccrine poroma and poroid hidradenoma.

A case of a single poroma lesion with the features of eccrine poroma and poroid hidradenoma is reported. The patient was a 61-year-old woman with a soft nodule, light brown in color, located on the posterior aspect of the right knee and containing a pigmented papular lesion in its center. The histological features of the papular lesion and the nodule were consistent with those of eccrine poroma and poroid hidradenoma, respectively. The eccrine poroma portion consisted mostly of poroid cells. The number of cuticular cells was slightly greater than that of poroid cells in the poroid hidradenoma portion. The cuticular cells in this portion as well as the poroid cells in both portions were immunohistochemically positive for epithelial membrane antigen. The findings in this case suggest that there is a close relationship between eccrine poroma and poroid hidradenoma and support the view that poroid hidradenoma is a type of poroma.

Acrospiroma↗

Management of a hidradenoma with Mohs micrographic surgery.

BACKGROUND: Hidradenomas are adnexal tumors that may grow to be very large and frequently recur after excision. Malignant transformation and metastatic spread can occur and histologic distinction of benign from malignant hidradenomas can be very difficult. Some tumors with bland histology have demonstrated aggressive behavior. OBJECTIVE: To decide the benefits of management with Mohs micrographic surgery of large hidradenomas. METHODS: The literature was reviewed, including both the clinical course of the tumor and histologic features. The case of our report was interpreted in light of the review. RESULTS: Three stages of Mohs surgery were required for complete removal of the hidradenoma. The patient remains free of recurrence. CONCLUSION: This report highlights the difficulties in distinguishing benign from malignant hidradenomas and emphasizes the benefits of Mohs surgery for large or recurrent tumors.

Adenoma, Sweat Gland↗

Clear cell hidradenoma of the skin-a third tumor type with a t(11;19)--associated TORC1-MAML2 gene fusion.

Recent studies have shown that the t(11;19)(q21;p13) translocation in mucoepidermoid carcinomas and benign Warthin's tumors results in a fusion of the N-terminal CREB-binding domain of the cAMP coactivator TORC1 (a.k.a. MECT1 and WAMTP1) to the Notch coactivator MAML2. Here we show that a third tumor type, clear cell hidradenoma of the skin, also expresses this gene fusion. RT-PCR analysis of a clear cell hidradenoma with a t(11;19)(q21;p13) translocation revealed expression of a TORC1-MAML2 fusion transcript consisting of exon 1 of TORC1 fused to exons 2-5 of MAML2. Because the fusion was only detected in a single case, the frequency of this aberration in clear cell hidradenomas remains unknown. These results demonstrate that the t(11;19) in mucoepidermoid carcinoma, Warthin's tumor, and clear cell hidradenoma targets the same genes and results in identical gene fusions, indicating that at least subgroups of these glandular tumors evolve through activation of the same molecular pathways.

Artificial Gene Fusion↗

Recurrent clear cell hidradenoma of the foot.

BACKGROUND: Clear cell hidradenoma is an uncommon neoplasm without established optimal treatment. OBJECTIVE: Using Mohs micrographic surgery, a recurrent clear cell hidradenoma of the right foot was treated. METHODS: Case report and literature review. RESULTS: Two stages of Mohs micrographic surgery and subsequent repair successfully treated the recurrent clear cell hidradenoma. No recurrence of the neoplasm has been observed at 11 months follow-up. CONCLUSION: Optimal treatment of clear cell hidradenoma is unestablished. Adequate primary local excision is essential. Mohs micrographic surgery should be considered for treatment, especially on recurrent tumors in critical locations.

Acrospiroma↗

Large ulcerated perianal hidradenoma papilliferum in a young female.

BACKGROUND: Hidradenoma papilliferum is an uncommon benign tumor that is located almost exclusively in the vulvar and anal areas. It is usually very small and asymptomatic, and to make a correct diagnosis is clinically very difficult. Occasionally the tumor becomes elevated to form a reddish brown papillary mass, and the surface ulcerates, which may erroneously suggest malignancy. OBJECTIVE: We report a case of a large, perianal hidradenoma papilliferum with suspected malignancy in a young Japanese female. RESULTS: A 22-year-old female had been aware of a perianal nodule for approximately 1 year. Examination of the perianal area revealed a wide pedunculated, reddish nodule with several white maculae. It was ulcerated and bleeding, 2.0 x 1.2 x 0.8 cm in size, and located in the 3 o'clock position. The nodule was totally excised with a narrow margin. The histopathologic diagnosis was hidradenoma papilliferum. No recurrence was observed for 23 months. CONCLUSION: When dermatologists encounter tumors of the anogenital area of adult females, it is important to keep hidradenoma papilliferum in mind as the differential diagnosis. Dermatologists should recognize that the tumor is benign, eliminating the need for wide resection.

Adenoma, Sweat Gland↗

Non-anogenital (ectopic) hidradenoma papilliferum with sebaceous differentiation: a case report and review of reported cases.

Hidradenoma papilliferum is a benign tumor that usually occurs in the female anogenital region. We present a 52-year-old male with a non-anogenital (ectopic) hidradenoma papilliferum on his face. He had had a subcutaneous tumor on his right eyebrow for 30 years. The histological findings included variously shaped cystic and tubular structures in the tumor. The lumina were surrounded by a double layer of cells and showed active decapitation secretion. The lesion contained sebaceous differentiation, which has not been previously documented in the histopathology of hidradenoma papilliferum. We review the published work of non-anogenital (ectopic) hidradenoma papilliferum tumors on the head and neck.

Adenoma, Sweat Gland↗

Nodular hidradenoma of the breast: report of two cases with literature review.

Two cases of nodular hidradenoma of the breast with possibly different origins are reported. Case 1 is of a 58-year-old female with a breast mass in the left, outer lower-quadrant. A histogenetical origin in the skin adnexal glands was suspected due to its superficial location and immunohistochemical findings. Case 2 is of a 44-year-old male with a subareolar nodule and nipple discharge. Histological examination demonstrated that the tumor was located deep in the breast tissue, was surrounded by dilated mammary ducts and exhibited intraductal extensions, which are all features mimicking those of breast cancer. Immunohistochemical positivity against gross cystic disease fluid protein-15 was weakly identified and negativity for endoplasmic reticulum was observed. This case can be interpreted as arising in the mammary ducts. It is well known that various kinds of skin adnexal tumors arise in the breast tissue; however, nodular hidradenoma of the breast is still a rare benign neoplasm. Clinically, nodular hidradenoma of the breast tends to occur in the nipple or subareolar region of the female breast. It should be kept in mind that nodular hidradenoma may occur in mammary ducts and it should be included when differential diagnoses are made of subareolar breast tumors.

Actins↗