Papillary eccrine adenoma: Immunohistochemicai study and literature review

A case of papillary eccrine adenoma on the right forearm of a 78-year-old Japanese woman is reported. The tumor was 1.3 cm in diameter, occupying the whole thickness of the dermis. Histologically, the tumor was composed of dilated tubules of various sizes with intraluminal papillary projections, and was surrounded by a fibrous stroma. An immunohistochemical study revealed that the proliferating tubules were composed of a single outermost layer of α-smooth muscle actin-and keratin 14-positive myoepithelial cells, and keratin 8-positive inner cells. This antigen expression pattern was comparable to that of the normal eccrine secretory coil, which indicates that the tumor differentiated toward the secretory coil of an eccrine sweat gland.     

Tubular adenoma of the skin with follicular and sebaceous differentiation: A report of two cases

The main controversies regarding tubular apocrine adenoma and papillary eccrine adenoma are whether they are two distinct entities or are the very same tumor, and if so, which lineage of differentiation (apocrine versus eccrine) it pursues. We report two cases of tubular adenoma with follicular and, in one case, additionally, sebaceous differentiation. The features in both cases indicated apocrine differentiation of the tubular component. One patient was a 60-year-old woman with a 1-year history of a solitary nodule on the scalp. The other patient was a 48-year-old woman with a solitary nodule of unknown duration located on the back. In both patients, the tumors were surgically removed. The patients were alive and well 2.5 and 2 years after surgery, respectively. The histologic features that both cases had in common included the combination of a tubular adenoma, foci of follicular differentiation, and areas of immature squamous metaplasia. In case 1, follicular differentiation was seen in the form of strands of basaloid cells surrounded by a stroma resembling the embryonic perifollicular sheath. Some aggregates of basaloid cells were juxtaposed with small collections of plump fibroblasts, imparting a resemblance to rudimentary follicular germs associated with follicular papillae. Many minute lumens surrounded by more eosinophilic cells were seen within the strands. In case 2, follicular differentiation was seen as several infundibulocystic structures surrounded by isthmic epithelium housing scattered mature sebocytes. In addition, there were areas reminiscent of desmoplastic trichoepithelioma (columnar trichoblastoma). In both cases, the areas with immature squamous metaplasia were represented by solid nodules that were mostly devoid of lumens and a peripheral basal/myoepithelial cell layer. In conclusion, these two cases of cutaneous tubular adenoma with accompanying follicular and sebaceous differentiation give further support to the proposition that the majority of these neoplasms have apocrine differentiation. Rare cases occurring in the sites normally devoid of apocrine glands may represent the eccrine counterpart.     

Papillary eccrine adenoma. A histopathological and immunohistochemical study

A case of papillary eccrine adenoma of the thigh of a 63-year-old woman is reported. Histologically, the tumor was composed of cystic dilated and branching tubular structures with papillary projections into the lumen, surrounded by a fibrous stroma. In addition, in some areas it was composed almost exclusively of solid strands of epithelial cells. The tumoral eccrine differentiation was assessed by immunohistochemical studies. Problems of differential diagnosis with low-grade sweat gland carcinoma and with tubular apocrine adenoma are discussed.     

Papillary Eccrine Adenoma with a Pomegranate-like Appearance

Papillary eccrine adenoma (PEA) is a rare cutaneous tumor which histopathologically presents numerous intradermal tubular structures with inward papillary projections. Only a few cases of PEA have been reported recently. We report a case of PEA of a 58-year-old Japanese man. The marked hyperkeratosis and pits gave the tumor the clinical appearance of a burst-open pomegranate. Compact hyperkeratosis within proliferated epidermis contained spiral ducts mimicking intraepidermal eccrine sweat ducts histopathologically. These keratinous structures were thought to correspond to the pores. Several tubular structures running up to the overlying thickened epidermis were observed in the upper dermis. With these findings and with immunohistochemical studies, we proposed that this tumor originated from eccrine sweat ducts.     

Syringometaplasia: Mucinous and Squamous Variants

The eccrine sweat ducts are normally lined by cuboidal epithelial cells which may rarely undergo metaplasia, i.e. syringometaplasia. Two lesions were observed in which eccrine sweat ducts displayed the mucinous and squamous variants of syringometaplasia. The first lesion clinically and histologically appeared to be a plantar wart. Microscopically, it consisted of a central invagination surrounded by marked epidermal acanthosis and hyperkeratosis. The invagination was lined by keratinocytes admixed with mucin-filled goblet cells. The mucin was positive by the Alcian blue (pH 2.5) and mucicarmine stains. Numerous eccrine sweat ducts led into the invagination and were focally lined by the mucin-laden cells. Recognition of mucinous syringometaplasia is important since it may be confused with primary or metastatic adenocarcinoma of the skin. The second lesion occurred on the outer ear and was clinically believed to be chondrodermatitis nodularis helicis. Microscopically, there were many islands of atypical squamous cells within the papillary and reticular dermis. These epithelial islands represented squamous syringometaplasia since many contained central lumina with eosinophilic cuticles and blended with normal ductal structures. It is important not to confuse this metaplastic change with invasive squamous cell carcinoma. Squamous syringometaplasia may be analogous to necrotizing sialometaplasia, a recently described phenomenon which occurs in minor salivary glands.     

Papillary eccrine adenoma: immunohistochemical studies of keratin expression

Despite various studies, there are serious disagreements about the cellular differentiation of papillary eccrine adenoma. In the present study, 2 specimens of papillary eccrine adenoma were analyzed by immunohistochemical techniques, using a panel of monoclonal antibodies against keratins, to elucidate its differentiation. Histopathologically, the tumor was composed of multiple tubular structures lined by two or more layers of epithelial cells. The luminal cells of the tubules were flattened or cuboidal. The former were noted in large dilated tubules. The latter were usually observed in small-to-moderate-sized tubules, and formed intra-luminal papillary projections in some tubules. Immunohistochemically, there were two kinds of cuboidal cells in the luminal layers of the tubules. Most of the large dilated tubules and some of the small-to-moderate-sized tubules expressed immunophenotypes similar to those of the eccrine dermal duct. The other tubular structures, including the small tubules resembling those of syringoma, expressed immunophenotypes similar to those of the transitional portions between the dermal ducts and the secretory segments of eccrine glands. From the above comparative studies, papillary eccrine adenoma is considered to differentiate towards the dermal duct and the transitional portions between the dermal ducts and the secretory segments of eccrine glands.     

Myoepithelial differentiation in benign sweat gland tumors

One hundred and two cases of benign sweat gland tumors of the skin were studied for the presence of myoepithelial cells specifically identified by a monoclonal antibody to alpha-smooth muscle actin on paraffin-embedded tissues. The monoclonal antibody gave a positive result in 12 of 12 cases of cylindroma, 14 of 16 cases of spiradenoma, 2 of 2 cases of apocrine tubular adenoma (papillary eccrine adenoma), 5 of 5 cases of apocrine hidrocystoma, 5 of 5 cases of hidradenoma papilliferum, and in 10 of 10 cases of syringocystadenoma papilliferum. Rare myoepithelial cells were detected in only 1 of 10 cases of mixed tumor, apocrine type. There was no immunoreactivity for alpha-smooth muscle actin in eccrine hidrocystoma (2 cases), mixed tumor of eccrine type (2 cases), syringoma (7 cases), hidroacanthoma simplex (1 case), eccrine poroma (14 cases), clear cell hidradenoma (15 cases), and in 1 case of eccrine syringofibroadenoma. Our data support the concept that myoepithelial cells are seen in most sweat gland tumors considered to differentiate toward the secretory coil of the normal sweat gland. In contrast, myoepithelial cells are absent in tumors showing differentiation toward the excretory (ductal) component of the gland.     

CD44 Expression in Normal Human Skin and Skin Tumors

CD44 is thought to be a principal cell surface receptor for hyaluronic acid. Although the distribution of hyalulonic acid has been studied, little is known about the distribution of the CD44 molecule in the human skin and skin tumors. This study was undertaken to investigate the distribution of the CD44 molecule in normal human skin as well as in benign and malignant skin tumors. In normal skin, CD44 was expressed on 1) keratinocyte cell surfaces throughout the epidermis except for the granular and horny layers, 2) hair follicular cells, 3) eccrine sweat gland cells, and 4) cell surfaces of dendritic cells in the dermis. In skin tumors, although CD44 was expressed on the tumor cell surface of seborreic keratosis, Bowen's disease, and squamous cell carcinoma as in normal skin, we could not detect any CD44 expression on the cell surface of the tumor cells of basal cell carcinoma. However, CD44 positive dendritic cells were observed in the tumor islands of basal cell carcinoma. Phenotypic analysis suggested that these CD44 positive cells were melanocytes.     

Immunohistochemical demonstration of myoepithelial cells in sweat gland carcinomas

Although myoepithelial cells are detectable in many benign sweat gland tumours, little is known about their role in sweat gland carcinomas. To specifically demonstrate myoepithelial cells, paraffin sections from 46 sweat gland carcinomas were stained, using a standard avidin-biotin-peroxidase complex method, with the monoclonal α-smooth muscle actin antibody 1A4. Myoepithelial cells were not found in adenoid cystic eccrine carcinoma (n=2), malignant nodular hidradenoma (n=2), porocarcinoma (n=4), extramammary Paget's disease (n= 12), sclerosing sweat duct carcinoma (n=4) or in adenosquamous mucoepidermoid carcinoma (n=l). In contrast, myoepithelial cells were demonstrated in two of eight apocrine adenocarcinomas, one of six mucinous eccrine carcinomas and two of seven eccrine adenocarcinomas. In all these tumours myoepithelial differentiation was found in peripheral cells of solid tumour islands, or in basal cells of tubular structures. However, in most areas of the tumours, myoepithelial layers were discontinuous. Cells in the centre of solid tumour nodules, and luminal cells of tubular structures, were negative for α-smooth muscle actin. In analogy to breast tumours, in which malignancy and invasiveness correlate with scattered or absent myoepithelial cells, we suggest that disrupted myoepithelial layers in sweat gland carcinomas may be interpreted as a loss of the invasion barrier.     

Syringoid eccrine carcinoma: report of a case with immunohistochemical analysis of cytokeratin expression

Syringoid eccrine carcinoma is an extremely rare cutaneous malignant tumor, thought to be derived from eccrine sweat apparatus. We report a case of syringoid eccrine carcinoma occurring on the scalp of a 66-year-old woman and analyzed its cytokeratin expression immunohistochemically to clarify its histogenesis. The tumor consisted mainly of numerous small cords and nests extending from the reticular dermis to the subcutaneous tissue, which formed luminal or tubular structures mimicking the nests of syringoma. Immunohistochemical analysis revealed that most tumor cells expressed simple epithelial cytokeratins (CKs 7, 8, 18, 19) suggesting their sweat secretory differentiation, and that a small number of tumor cells showed an expression of stratified epithelial cytokeratins (CKs 5, 14) suggesting their ductal differentiation. We believe that the syringoid eccrine carcinoma of our case may differentiate mainly toward the sweat secretory cells rather than toward the dermal ductal cells.     

Histogenesis of primary localized cutaneous amyloidosis: sequential change of epidermal keratinocytes to amyloid via filamentous degeneration.

Two cases of lichen amyloidosus and 8 cases of macular amyloidosis were examined by electron microscopy. Epidermal keratinocytes showed variable degrees of focal degeneration in the basal or lower Malpighian layer. The primary change was seen in cells which contain fibrillar (30 nm in thickness) cytoplasmic inclusion. The following developments seemed to lead to filamentous degeneration (colloid bodies): (1) aggregation of tonofilaments within the granular or fibrillar cytoplasm, (2) filamentous cells, which are composed of bundles of 7-nm thick filaments surrounded by cell membrane and desmosomes, and (3) filamentous masses composed of bundles or whorls of tightly packed 7-nm thick filaments in the intercellular spaces. At the dermo-epidermal junction, some of the filamentous masses were surrounded by the basal lamina of the epidermis and others were dropping into the dermis. Occasionally, loosened filaments (similar to amyloid filaments) were about to drop into the dermis. Early formation of amyloid islands consisted of electron-dense and electron-light parts. They were located directly beneath the epidermis. In the deeper postion of the papillary dermis and in the upper reticular dermis, the majority were electron-light masses. Electron dense parts were the densely packed 7-nm thick filaments, whereas electron-light parts were the typical straight amyloid filaments. Small tubular filaments were seen in common in the filamentous cells, filamentous masses, and amyloid islands. It is concluded that some of the amyloid substance in primary localized forms of cutaneous amyloidoses derive from the epidermal cells through filamentous degeneration.     

多发小汗腺螺旋腺瘤1例并文献复习

患者男,11岁,颈背部皮下多个结节3年。皮损组织病理示:肿瘤细胞位于真皮内,肿瘤为2种不同细胞组成,中央细胞大,核嗜酸,淡染,外周细胞核小,深染,瘤体可见条索样结构。瘤体外周有纤维包膜。诊断:小汗腺螺旋腺瘤。治疗:手术切除并随访。     

Papillary eccrine adenoma. A tubulopapillary hidradenoma with eccrine differentiation

A slow-growing tumor on the dorsum of the right foot of a 25-year-old female had features that corresponded to papillary eccrine adenoma. The grouping of this entity under the term "tubular apocrine adenoma" in a recent edition of a textbook led to further study of this case in the form of enzyme histochemistry and electron microscopy. The tumor was positive for amylophosphorylase and negative for acid phosphatase, and on electron microscopy showed complex intercellular membrane interdigitation and a possible intercellular canaliculus. No features of apocrine differentiation were found, and we feel that this tumor is distinctly eccrine in its differentiation. The similarity of these two ostensibly separate entities, and the opinion of some authors that intermediate differentiation may be possible in adnexal tumors, would favor them being grouped under the single term of "tubulopapillary hidradenoma," with apocrine or eccrine differentiation as the case may be.     

Eccrine syringofibroadenoma of clear cell variant: an immunohistochemical study

Eccrine syringofibroadenoma is a rare eccrine tumor first described by Mascaro in 1963. The clear cell variant was reported by Fretzin in 1995. We describe a hemorrhagic papule on the knee of a 64-year-old woman. Microscopic examination revealed epidermal-based anastomosing thin epithelial cords with ductal structures surrounded by a fibrovascular stroma. Multiple nests of glycogen-containing clear cells were also present. The tumor cells forming the epithelial cords expressed keratin. The eccrine ductal structures and clear cells additionally expressed gross cystic disease fluid protein-15 (GCDFP-15). Reactivity with antibodies against estrogen receptor or progesterone receptor was negative. These results favor the dual differentiation of the clear cell variant of eccrine syringofibroadenoma toward ductal and secretory portions of the eccrine sweat gland.     

Eccrine angiomatous hamartoma

A 50-year-old Korean man had an erythematous patch on the left side of his chest since birth ( Fig. 1 ). We first regarded this lesion as nevus flammeus because of the resemblance of the lesion to an erythematous patch that blanched with pressure. Physical examination revealed an 8×5 cm erythematous patch with a central profuse sweating zone surrounded by a flame-like non-sweating zone ( Fig. 2 ). The patch was sharply demarcated from the normal skin. Sweating was evoked by physical exercise or emotional stress. There was no history of pain or tenderness for the lesion. The patient had no history of any other vascular disease. Histopathologic examination of the central zone showed an increased number of eccrine glands in the mid and deep dermis. Some dilated vascular channels that were not associated with the eccrine glands were present in the upper dermis and subcutaneous tissue ( Fig. 3 ). Immunohistochemical stain with anti-factor VIII-related antigen showed reactivity on the endothelia of the vascular elements which were not associated with eccrine glands in the upper dermis. Histopathologic examination of the surrounding zone showed non-specific findings, except for the dilated vascular channels in the upper dermis.

Figure 1 Open in figure viewer PowerPoint (A) hyperhidrotic erythematous patch on the left side of the chest     

Microcystic adnexal carcinoma--a light and electron microscopic studies

Microcystic adnexal carcinoma (MAC) in a 48-year-old Japanese male was studied. A firm nodule had appeared on the right side of his upper lip at the age of 38 years and then had gradually enlarged. Light microscopic examinations revealed numerous strands and islands of basaloid cells and keratinous cysts with desmoplastic stroma in the dermis and subcutaneous tissue. Some keratinous cysts showed early calcification and some contained pilar structures. Clear cells resembling sebaceous gland cells were observed in the strands of tumor cells connecting the keratinous cysts. In the middle dermis, there were small ductal structures containing amorphous eosinophilic material. Immunoperoxidase staining for carcinoembryonic antigen was focally positive in the lumina of small ducts, on the ductal lining surface of tumor cells, and in the contents of keratinous cysts. Electron microscopic examinations revealed tumor cells resembling non-keratotic keratinocytes, and containing tonofilaments, mitochondria, and vacuoles. In some parts, tumors were differentiated to various types of sebaceous gland, sweat duct, or sweat secretary segments. Sebaceous gland cells contained lipid droplets and glycogen. Tumors differentiated to sweat duct had a marked similarity to embryo eccrine duct in the lower epidermis because of the presence of multivesicular dense bodies and periluminal filamentous zones and the absence of myoepithelial cells. Tumors differentiated to sweat secretary segments had a similarity to embryo eccrine secretary segments because of the absence of multivesicular dense bodies, periluminal filamentous zones and myoepithelial cells.     

Immunohistochemical studies of normal sweat glands, sweat gland tumors and extramammary Paget's diseases. II. Immunohistochemical studies of sweat gland tumors and extramammary Paget's diseases

Localizations of 18 antigens were analyzed in 41 cases with benign sweat gland tumors (13 with eccrine acrospiroma, 4 with eccrine spiradenoma, 2 with hidroacanthoma simplex, 9 with chondroid syringoma, 4 with syringocystadenoma papilliferum, 1 with tubular apocrine adenoma, 1 with papillary eccrine adenoma, 1 with apocrine cystadenoma, 1 with cylindroma, 5 with syringoma), 14 with malignant sweat gland tumors (7 with eccrine porocarcinoma, 3 with eccrine duct carcinoma, 3 with apocrine gland carcinoma, 1 with mucinous carcinoma) and 13 with extramammary Paget's disease. The results I obtained were compared with those in the normal sweat glands for determination of a differentiation of each tumor.     

皮脂腺痣继发肿瘤16例临床与组织病理分析

目的 探讨皮脂腺痣继发肿瘤的组织病理学特征。方法 回顾性分析所收集到的16例皮脂腺痣继发肿瘤患者的临床、病理资料和组织病理学特征。结果 ≥18岁12例,11 ~ 17岁4例。7例伴有乳头状汗管囊腺瘤,4例伴有管状顶泌汗腺腺瘤,3例伴有毛鞘瘤,2例伴有基底细胞癌,汗孔角化样汗孔和真皮导管痣及鲜红斑痣各1例。6例有1个以上的肿瘤,尤以乳头状汗管囊腺瘤伴管状顶泌汗腺腺瘤最多,占有4例。结论 皮脂腺痣基础上有任何继发肿瘤发生,都应密切关注。     

Papillary eccrine adenoma

A 28-year-old man came to us with a solitary skin colored, mildly tender nodule of 6 months duration on the dorsum of the right hand. On histological examination, multiple dilated ducts without apparent continuity with the surface were found in the dermis. These dilated ducts had branching tubules with eosinophilic amorphous material filling most of the lumina. The peripheral cells of the tubules resembled myoepithelial cells, whereas the luminal border cells were cuboidal or low columnar. Papillary projections arising from the inner cells were seen extending into the lumen. These features were diagnostic of a rare tumor, papillary eccrine adenoma.     

Axillary apocrine carcinoma associated with apocrine adenoma and apocrine gland hyperplasia

Apocrine carcinomas represent a rare group of tumors with a potential for destructive local invasion, regional and distant metastases, and are equally common in both sexes. A case of a 79-year-old woman with axillary apocrine carcinoma associated with apocrine adenoma and apocrine gland hyperplasia is presented. To our knowledge, this is the first case diagnosed in a Caucasian and also the first case diagnosed in a female patient. Grossly, the tumor measured 3.2x1.5x1.2 cm and on cut section appeared granular, white to gray-tanned. Microscopically, the tumor was located in the dermis, poorly demarcated, focally necrotic with ulcerated overlying skin. It was predominantly composed of complex, closely packed tubuloglandular structures but in few areas papillary structures were also observed. The cells contained abundant eosinophilic, finely granular cytoplasm with pleomorphic nuclei and showed apocrine-like decapitation. The cytoplasm contained periodic acid Schiff diastase resistant granules. Mitoses were frequent and some were atypical. In one area, the tumor was lobular and composed of tubular structures lined with one layer of uniform cuboidal or columnar eosinophilic cells, indicating a pre-existing apocrine adenoma. Beneath the tumor, in the deep dermis and subcutaneous tissue, hyperplastic apocrine glands were also found. No additional therapy was used, and one year after the surgery the patient was alive and showed no signs of tumor spread. This and previously reported cases suggest that apocrine hyperplasia and apocrine adenoma may represent successive steps in the development of apocrine carcinoma.