Anogenital Mammary-like Glands Research Articles

Bilateral anogenital mammary-like glands lactating in the puerperium. A clinical case

This article presents a rare case of a woman having bilateral anogenital mammary-like glands, lactating in the postpartum period, and a surgical solution to the problem. The histology of the removed tissue corresponded to the morphology of the breast during lactation. No complications occurred after surgery.

Transcriptional repressor GATA binding 1 (TRPS1) immunoexpression in normal skin tissues and various cutaneous tumors.

Transcriptional repressor GATA binding 1 (TRPS1) is a transcription factor recently shown to play a role in the development of breast and liver cancer. Here, we evaluate TRPS1 immunoexpression in normal skin tissues and various cutaneous tumors. TRPS1 immunohistochemistry was performed in 109 cases of primary cutaneous tumors and 19 cases of metastatic carcinomas. TRPS1 expression was also evaluated in the normal skin tissues. The normal epidermis was TRPS1-. In contrast, the eccrine apparatus, epithelial compartment of the hair follicles, hair papilla, sebaceous glands, and anogenital mammary-like glands were TRPS1+. In primary cutaneous tumors, TRPS1 positivity varied in poroma (2/3), nodular hidradenoma (4/5), spiradenoma (4/4), cutaneous mixed tumor (5/5), trichilemmal cyst (7/8), proliferating trichilemmal tumor (1/3), pilomatricoma (9/9), sebaceoma (2/5), extramammary Paget disease (13/13), sebaceous carcinoma (2/2), actinic keratosis (3/10), Bowen disease (7/12), and squamous cell carcinoma (1/5) cases. All cases of seborrheic keratosis, basal cell carcinoma, Merkel cell carcinoma, and malignant melanoma were TRPS1-. All metastatic breast carcinoma cases (8/8) were highly positive for TRPS1, while all but one of the other metastatic tumor cases were TRPS1-. TRPS1 immunoexpression was observed in several skin appendages and cutaneous tumors.

Encapsulated papillary carcinoma originating from the vulva anogenital mammary-like glands: a rare case report

Encapsulated papillary carcinoma originating from the vulva anogenital mammary-like glands: a rare case report

Atypical Non-neoplastic Changes in Anogenital Mammary-like Glands Accompanying Invasive Squamous Cell Carcinoma.

Long regarded as ectopic or supernumerary breast tissue, anogenital mammary-like glands (AGMLG) are now considered a normal constituent of the anogenital area. AGMLG are presumed to be the origin for various benign and malignant lesions. Changes in AGMLG compatible with usual ductal hyperplasia and atypical ductal hyperplasia considered as precursor lesions and its presence in specimens can be explained by their role in the pathogenesis of primary extramammary Paget disease. In this report, we presented four cases of invasive squamous cell carcinoma accompanied by non-neoplastic atypical changes in ductal portions of AGMLG compatible with atypical ductal hyperplasia in breast adjacent to the carcinoma. This is a reactive phenomenon similar to that seen in apocrine/eccrine glands adjacent to squamous cell carcinoma. In a limited biopsy specimen, these areas should not be mistaken for adenocarcinoma.

Lesions of anogenital mammary-like glands: Four cases including novel pathologic and immunohistochemical observations

Anogenital mammary-like glands, formerly described as ectopic breast tissue, are currently considered to be normal histologic components of the anogenital region. Anogenital mammary-like glands can give rise to many lesions identical to counterparts in the native female breast. We describe four cases of such lesions, including fibroadenoma, gynecomastia-like hyperplasia, and ectopic mammary-type tissue with a spectrum of usual ductal hyperplasia, apocrine metaplasia, adenosis, and pseudolactational change. All four cases occurred in young women (ages 29–38) who presented with vulvar or perianal masses. Similar to previously reported cases, these lesions shared histologic and immunohistochemical characteristics identical to native female breast lesions. Novel findings in our cases included (1) the first case of gynecomastia-like change to be reported in the perianal area of a female, (2) Immunohistochemical staining identifying a 3-layered epithelium characterized by a population of CK14 and CK5/6 positive and hormone receptor negative superficial luminal cells, and (3) diffuse, strong positivity for GATA3 in all cases. Our study adds to the literature on these rare lesions and highlights findings which may be useful in understanding the pathogenesis and improving the diagnosis of anogenital mammary-like gland lesions.

Étude anatomoclinique et immunohistochimique d’une tumeur dérivant des glandes ano-génitales de type mammaire : l’hidradénome papillifère

To specify the clinical, histopathological and immunochemical features in a large series of hidradenoma papilliferum patients and to point out similarities with breast disease. We performed a monocentric retrospective histopathological study. Cases of hidradenoma papilliferum seen at the dermatopathology laboratory of Strasbourg (1977-2017) were included and analyzed with hematoxylin-eosin staining and immunostaining. Diagnosis was confirmed where tubular formations and papillary proliferations were noted and were lined by a luminal layer of columnar cells surrounded by a basal layer of myoepithelial cells. We included 32cases solely involving women of a mean age of 49years. Most cases involved the vulva (67%) or the perianal region (25%). The lesions ranged in size from 3to 25mm. They occurred as solitary, non-ulcerated, skin-colored and cyst-like lesions. They were mostly localized in the mid-dermis. We identified several peculiar morphological variants. Three HP had a structure resembling tubular apocrine papillary adenoma, 3were similar to apocrine cystadenoma, while 9HP, of which 3involved epidermal hyperplasia, were connected with the overlying epidermis and had dense stromal infiltrate in plasma cells, closely resembling syringocystadenoma papilliferum. Sixteen tumors had no features of note. Similarities with breast tumors were revealed through columnar cell hyperplasia (6cases), high mitotic rates (4cases), and atypical nuclei (3cases). Anogenital mammary-like glands were found in the immediate vicinity of 10HP. Myoepithelial cells were immunoreactive for smooth muscle actin and p63, while luminal cells were immunoreactive for CK7, CK8, EMA, and, in a more focused and less consistent manner, for Ber-EP4, CD117, PHLDA1and androgen receptors. Our study highlighted a variety of patterns and cell morphologies, on occasion within a single tumor, identical to breast disease. Histological and immunochemical examination revealed that anogenital mammary-like glands and HP share common features, which militates in favor of a common histogenesis.

Extensive Squamous Metaplasia in Hidradenoma Papilliferum of Vulva: Illusion of Squamous Cell Carcinoma

Hidradenoma papilliferum is a rare, slow-growing, benign, cystic, papillary tumour occurring in middle-aged females, in the anogenital region commonly. It involves the anogenital mammary-like glands and is usually asymptomatic. Simple excision is curative. We present a case of hidradenoma papilliferum of long-standing duration with extensive squamous metaplasia. A post-menopausal lady presented with an exophytic growth on the left labia majora and part of mons pubis. It was initially misdiagnosed on the basis of vulvar biopsy and managed as squamous cell carcinoma of the vulva. The final histopathology revealed an epithelial tumour involving the vulva with extensive squamous metaplasia. The tumour involved mainly the dermis and the subcutaneous tissue. This is the first case in the literature where the diagnosis of hidradenoma papilliferum was confused with squamous cell carcinoma of the vulva due to its clinical presentation and extensive squamous metaplasia. Diagnosis of hidradenoma papilliferum should be kept in mind if a woman presents with vulval papillary or nodular lesions. An expert clinical and histopathological review should be done to avoid misdiagnosis.

Anogenital mammary-like gland adenoma: report of two cases and review of literature

Anogenital mammary-like gland adenoma: report of two cases and review of literature

Tubular Apocrine Adenoma on Vulva: An Unusual Location.

Tubular Apocrine Adenoma on Vulva: An Unusual Location.

BRAF and KRAS mutations in tubular apocrine adenoma and papillary eccrine adenoma of the skin

Tubular apocrine adenoma (TAA) and papillary eccrine adenoma (PEA) are benign sweat gland tumors. Their names imply that they exhibit apocrine and eccrine differentiation, respectively. However, morphologically they are very similar and are often indistinguishable. The molecular pathogenesis of either tumor is poorly understood at present. On the basis of an index case of nipple adenoma that was morphologically reminiscent of cutaneous TAA/PEA and harbored a BRAFV600E mutation, we investigated whether a similar genetic change is also present in TAA/PEA. BRAF, RAS, and PIK3CA mutation analyses, and BRAFV600E-specific immunohistochemistry were performed for 24 TAAs/PEAs, 10 eccrine poromas, 7 apocrine cystadenomas, 2 TAA-like adenomas associated with nevus sebaceus, and one apocrine adenoma probably arising in anogenital mammary-like glands (AGMLGs). The results demonstrated that BRAFV600E mutations were present in TAAs (9/15, 60%) and PEAs (7/9, 78%), but not in other neoplasms. Two additional TAAs harbored KRASG12D mutations. In addition, a KRASG12C mutation was identified in one nevus sebaceus-associated TAA-like adenoma. The speculated AGMLG-related apocrine adenoma had a PIK3CAH1047R mutation. We concluded that activating BRAF and KRAS mutations were commonly present in TAAs/PEAs, indicating that in addition to a morphological resemblance, they are closely related genetically. Therefore, they could be considered to be united as a single entity. By contrast, the apocrine adenoma probably arising in AGMLG harbored a PIK3CA mutation, which is also commonly present in hidradenoma papilliferum. Further studies are necessary to determine whether the pathogenesis of AGMLG-related tumors is similar to breast tumors.

Papillary Hidradenoma of Vulva: A Rare Case Presentation

Papillary hidradenomas in the vulva arises from specialized anogenital mammary-like glands in and around the intralabial sulcus. These are benign lesions, which present as slow growing, circumscribed cystic masses. Histologically, papillary hidradenomas are characterized by glandular structures and complex branching papillae with fibrovascular stalks. The glands are lined by myoepithelial cells and epithelial secretory cells with a surrounding fibrous stroma. Oestrogen and progesterone receptors are potential markers to differentiate between anogenital sweat glands and conventional sweat glands. We intend to present a case of excoriating papillary hidradenoma of vulva in a 23-year old female with review of literature. Key words: Papillary hidradenoma, Vulva, Benign Tumor.

Anogenital Mammary-Like Glands: A Study of Their Normal Histology With Emphasis on Glandular Depth, Presence of Columnar Epithelial Cells, and Distribution of Elastic Fibers.

The normal histology of anogenital mammary-like glands (AGMLG) has been studied previously, but some aspects, including glandular depth, presence of columnar epithelium resembling columnar cell change/hyperplasia as defined in mammary pathology, and distribution of elastic fibers, have not been previously investigated. To address these issues, we studied 148 AGMLG identified in 133 paraffin blocks sampled from 64 vulvar wide excision or vulvectomy specimens (64 patients, various indications for surgery). The depth of AGMLG ranged from 0.64 to 3.9 mm. Epithelial columnar cell change was noted in 33.1% of all AGMLG, whereas columnar cell hyperplasia was detected in 10.1%. Occasionally, combinations of cuboidal epithelium and columnar cell change were seen within 1 histological section. Of 22 specimens stained for elastic fibers, in only 6 (27.3%) cases were elastic fibers found around glands. Periductal elastic fibers were demonstrated around 3 of the only 5 ducts, which were available for analysis in slides stained for elastic fibers. The depth of AGMLG should be taken into account when planning topical and surgical therapies for lesions derived or evolving from AGMLG. Alterations identical to columnar cell change may represent a normal variation of AGMLG.

Spectrum of Changes in Anogenital Mammary-like Glands in Primary Extramammary (Anogenital) Paget Disease and Their Possible Role in the Pathogenesis of the Disease.

To determine whether a subset of primary extramammary Paget disease (EMPD) may originate in anogenital mammary-like glands (AGMLG), the authors studied 181 specimens of EMPD, detailing alterations in AGMLG. The latter were identified in 33 specimens from 31 patients. All patients were women, ranging in age from 38 to 93 years (median, 65 y). In all cases, lesions involved the vulva and in 1 patient the perianal skin was affected. Histopathologically, AGMLG manifested changes identical to columnar cell change (CCC) (87.1%), usual ductal hyperplasia (22.6%), columnar cell hyperplasia (CCH) (9.7%), oxyphilic (apocrine) metaplasia (6.5%), and atypical duct hyperplasia (3.2%). Four cases (12.9%), in addition to intraepidermal carcinoma, harbored invasive carcinoma. In all 4 of these, AGMLG displayed a range of alterations including ductal carcinoma in situ, CCC, and CCH. Three further cases (9.7%) showed ductal carcinoma in situ without any definite invasive carcinoma. Colonization of AGMLG by neoplastic Paget cells was noted in 6 cases. As CCC and CCH may be encountered in normal AGMLG, these alterations are unlikely to play a significant role in the pathogenesis of the disease. However, by analogy with mammary Paget disease, rare cases of primary EMPD may originate in AGMLG with a subsequent upward migration of the neoplastic cells into the epidermis and possible later breach through the basal membrane. Usual ductal hyperplasia and atypical duct hyperplasia can then be regarded as earlier precursor lesions, linking both ends of the spectrum.

An Immunohistochemical Study of Anogenital Mammary-Like Glands.

Although the normal histology of anogenital mammary-like glands (AGMLG) has been studied, no systemic investigation has been performed on the immunoprofile of these structures. We studied intact AGMLG with a broad panel of antibodies. The immunoprofile of AGMLG is similar to that of a normal breast tissue, and there are similarities to eccrine glands and coils about cytokeratin expression. Our immunohistochemical data may contribute to understanding of the pathogenesis of lesions arising from AGMLG.

Study of Selected BRCA1, BRCA2, and PIK3CA Mutations in Benign and Malignant Lesions of Anogenital Mammary–Like Glands

Anogenital mammary-like glands (AGMLGs) are nowadays considered a normal component of the anogenital area. Lesions involving AGMLGs are histopathologically very similar to their mammary counterparts, but the information on molecular biological mechanisms in these vulvar/perianal tumors is scarce. Mutations in the PI3K-AKT cascade have been found in hidradenoma papilliferum. The authors studied selected BRCA1, BRCA2, and PIK3CA mutations in series of benign and malignant neoplasms thought to be associated with AGMLGs, including 9 cases of primary extramammary Paget disease, 3 different cases of mammary-type carcinoma (adenoid cystic like, tubulolobular, and invasive ductal like), and 5 cases of hidradenoma papilliferum. No BRCA mutation was detected, whereas 3 neoplasms yielded PIK3CA mutation, including extramammary Paget disease, mammary-type invasive ductal carcinoma, and tubulolobular carcinoma. Our study expands the spectrum of lesions of AGMLGs harboring mutations in genes encoding the PI3K-AKT cascade. Further studies of the whole BRCA1 and BRCA2 genes using a larger cohort are needed to clarify their role in the pathogenesis of AGMLG lesions.

Molecular alterations in lesions of anogenital mammary-like glands and their mammary counterparts including hidradenoma papilliferum, intraductal papilloma, fibroadenoma and phyllodes tumor.

Lesions affecting anogenital mammary-like glands (AGMLG) are histopathologically very similar to those seen in the breast but whether this morphological similarity is also reflected at the genetic level is unknown. To compare the underlying molecular mechanisms in lesions of AGMLG and their mammary counterparts, we analyzed the mutational profile of 16 anogenital neoplasms including 5 hidradenomas papilliferum (HP), 1 lesion with features of HP and fibroadenoma (FA), 7 FA, 3 phyllodes tumors (PhT)) and 18 analogous breast lesions (6 intraductal papillomas (IDP), 9 FA, and 3 PhT) by high-coverage next generation sequencing (NGS) using a panel comprising 50 cancer-related genes. Additionally, all cases were analyzed for the presence of a mutation in the MED12 gene. All detected mutations with allele frequencies over 20% were independently validated by Sanger sequencing (concordance: 100%). Mutations in PIK3CA, AKT1, MET, ABL1 and TP53 genes were found in lesions of AGMLG and also their mammary counterparts. The PI3K-AKT cascade plays a role in tumors arising at both sites. It appears that some histopathologically similar anogenital and breast lesions develop along similar molecular pathways.

Anogenital mammary-like glands and related diseases. Part 2. Malignant tumors of the anogenital glands

Mammary-like glands are a normal anatomical component of the anogenital region and can give rise to many benign and malignant tumors that morphologically mimic the similar diseases of the breast. The literature review is complemented by a description of 199 cases of malignant tumors of mammary-like glands. The paper presents the clinical and morphological characteristics of various malignant tumors of mammary-like glands, including extramammary Paget's disease, ductal, tubulolobular, adenoid cystic adenocarcinoma, low-grade phyllodes tumor, etc.

Anogenital mammary-like glands and related lesions. Part 1. Benign tumors and tumor-like disorders

Anogenital mammary-like glands represent a normal anatomic constituent of the anogenital area and may give rise to many benign and malignant tumors that morphologically mimic similar breast diseases. The literature review is complemented by a description of 286 cases of benign tumors and tumor-like processes in the mammary-like glands. The paper presents the clinical and morphological characteristics of papillary hidradenoma, fibroadenoma, benign phyllodes tumor, lactating adenoma, sclerosing adenosis, pseudoangiomatous stromal hyperplasia, etc.

PIK3CA and AKT1 mutations in hidradenoma papilliferum

AimsHidradenoma papilliferum (HP) is a benign vulvar neoplasm that arises from anogenital mammary-like glands, and its morphology is similar to mammary intraductal papilloma. The aim of this study was to...

The role of DOG1 immunohistochemistry in dermatopathology.

This study was designed to evaluate the immunoexpression of DOG1, a specific marker expressed in gastrointestinal stromal tumors, in normal skin tissues and cutaneous epithelial tumors. DOG1 immunostaining extent, intensity and pattern were evaluated in 69 cutaneous apocrine/eccrine tumors, 11 sebaceous tumors, 46 follicular tumors, 52 keratinocytic tumors and perilesional normal tissues. In normal tissues, DOG1 was expressed strongly in the intercellular canaliculi of eccrine glands, moderately in the myoepithelial cells of mammary and anogenital mammary-like glands, and weakly or not at all in the periphery of sebaceous lobules and the lower layer of epidermis and follicular infundibulum. All apocrine-type cutaneous mixed tumors showed apical-luminal positivity for DOG1, and 4/9 of these tumors included intercellular canaliculi highlighted by DOG1 immunostaining. Other sweat gland tumors, including hidrocystadenoma, spiradenoma, cylindroma and apocrine carcinoma, also expressed DOG1 focally with an apical-luminal pattern. Although slight membranous positivity for DOG1 was observed in various tumor types, hidradenoma papilliferum exhibited diffuse membranous DOG1 staining in the myoepithelial cells. DOG1 is a novel marker for identifying intercellular canaliculi and is a potential immunomarker of myoepithelial cells specific to mammary glands, anogenital mammary-like glands and tumors originating therein.