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Benign adnexal neoplasms are quite a common occurrence in adults, especially in the head and neck region. They raise suspicion for malignancy if there are red flag signs like rapid increase in size, pain, ulceration or recurrence. We hereby report a case of a middle-aged gentleman who consulted our surgical OPD with right-sided neck swelling, which was initially thought to be a dermoid cyst; on further evaluation, found to be a dermal sweat gland tumour with features of nodular hidradenoma. The point that is of interest but coincidence to note is that this swelling was preceded by a minor trauma. Characterising these swellings using simple imaging and pathological investigation modalities is important to study their behavioural pattern and add the same to our existing database. This will also help the treating surgeons to keep in mind the possibility of occurrence of such histologies in soft tissue swellings when they present with uncommon clinical features, instead of brushing them aside as the common epidermal or dermoid cysts. Incidence of malignancy is almost nil in nodular hidradenoma, which when found, is attributed to poor surgical clearance; hence the prudence to operate with adequate clearance is extremely significant in preventing the transformation of a mole into a mountain.

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Solid cystic hidradenoma, or clear cell hidradenoma, is a distinct and histologically rare tumor formed at the sweat glands, found mainly in adults and majorly among women. In this case, a 26-year-old female presented with asymptomatic swelling in her left inguinal area. Similar cases have been discussed in the literature considering the same kind of tumor. The present case is reported owing to the rarity of the type of tumor in terms of size and region of occurrence with the associated condition of pregnancy.

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Clear cell hidradenoma (CCH), a rare yet benign finding, is a tumor that originates from sweat glands. It mainly presents as a slow-growing cystic nodule on the scalp or trunk. We report a case of a 60-year-old man who presented with an abdominal subcutaneous mass. This mass was excised and pathology confirmed the presence of CCH, with cytology negative for malignant cells. Although benign, CCHs cannot be confirmed as such until excision and pathologic analysis, which is the standard treatment of choice.

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Classification of breast tumors has been evolving in the last decade. Uncommon types of breast neoplasms have been increasingly recognized. While the pathogenesis of a subset of these tumors remains to be ascertained, integration of state-of-the-art knowledge from molecular advancements and clinical practice has enhanced our understanding of these diseases, be they unique to the breast or more frequently seen in other organs. Furthermore, these lesions may have diverse clinical outcomes despite of similar histopathologic and immunophenotypic characteristics or even molecular alterations, thus warranting different clinical management. Therefore, recognizing their salient histologic features and judicious use of ancillary studies is essential to reach the correct diagnosis in the pursuit of personalized medicine. This review provides an update on selective special types of breast neoplasms, with emphasis on their salient clinicopathologic features, diagnostic pitfalls, controversies, and recent molecular genetic advances.

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Introducción: El hidradenoma nodular maligno es un tumor maligno de glándula sudorípara ecrinas, poco común, considerada una lesión de diferenciación anexial ecrinas, que generalmente surge de nuevo, aunque se han descrito unos pocos casos surgidos sobre un hidradenoma nodular. Es decir, representa la contrapartida maligna del hidradenoma nodular. Objetivo: Dar a conocer la presentación de un caso, dada la inusual aparición de esta entidad, con revisión de los criterios para su diagnóstico. Caso clínico: Se informa el caso de un hombre de 74 años de edad con una neo formación en la región parietal derecha del cuero cabelludo. Conclusiones: Debemos pensar en un hidradenoma nodular maligno ante un tumor solitario, firme o fluctuante, infrecuente en el cuero cabelludo, con curso agresivo, recurrencias y metástasis ganglionares y confirmar su diagnóstico con el estudio inmunohistoquímico(AU)

Introduction: Malignant nodular hidradenoma is a rare malignant eccrine sweat gland tumor considered a lesion of eccrine adnexal differentiation, which usually arises again, although a few arising cases on nodular hidradenoma have been described. In other words, it represents the malignant counterpart of nodular hidradenoma. Objective: To report a case, given the unusual occurrence of this entity, with a review of the criteria for its diagnosis. Case report: We report the case of a 74-year-old man with a neoformation in the right parietal region of the scalp. Conclusions: We should consider a malignant nodular hidradenoma when faced with a solitary, firm or fluctuant tumor, rare in the scalp, with aggressive evolution, recurrences and lymph node metastasis, and confirm its diagnosis with immunohistochemical study(AU)

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BACKGROUND: Cysts of the skin are observed frequently and their diagnoses are generally straightforward. However, atypical cystic lesions for which differentiation is indistinct have been noted. METHODS: We examined five cases of trichilemmal cyst with proteinaceous material (TCPM), which required differentiation from sweat duct/gland tumors. We investigated the histopathological findings of TCPMs and evaluated the immunohistochemical expression of cytokeratin (CK) 10, CK13, CK17, CK19, CD8, and CD117. Immunohistochemical analysis was performed on the 5 TCPMs, 10 trichilemmal cysts (TCs), 5 clear-cell hidradenomas, 5 poroid hidradenomas, and cutaneous normal adnexa. RESULTS: Apoptotic cells were present in the cyst wall with a small amount of keratin or calcification in the cavity of TCPMs. The TCPMs and TCs were negative for CK19 and CD117, on the other hand clear-cell hidradenoma and poroid hidradenoma were positive for CK19 and CD117. The restricted positivity for CK10 was detected in the suprabasal layers of the cyst walls of TCPMs and TCs. The immunostaining patterns of TCPMs and TCs were similar to those of normal follicular isthmus. CONCLUSIONS: The histopathological findings with characteristics of TCs and a panel of immunohistochemical antibodies including CD117, CK19, and CK10 contributed to a correct diagnosis of TCPM.

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Clear cell hidradenoma (CCH) is an uncommon adnexal tumor usually arising from eccrine glands and commonly seen on the face and the upper extremities. CCH occurring in the breast is extremely rare. Herein we report a case of MAML2-rearranged CCH of breast with a papillary architecture closely mimicking intraductal papilloma, adenomyoepithelioma and low-grade mucoepidermoid carcinoma, thus representing a source of diagnostic confusion. An overview of salient histologic features and immunophenotypes to distinguish CCH and low-grade mucoepidermoid carcinoma is also integrated into the report.

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Clear cell hidradenoma is a tumor that originates from a sweat gland and typically involves the dermis and subcutaneous tissue. Malignant clear cell hidradenoma is very rare, and surgical excision is usually performed without imaging. There are few reports of the ultrasonographic findings of malignant clear cell hidradenomas. Herein, we present the ultrasonographic characteristics of a malignant clear cell hidradenoma.

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Nodular hidradenoma is an uncommon cutaneous adnexal tumor arising from sweat glands. In the skin, it usually presents as a solitary dermal nodule; excision is curative in most cases. In rare instances, it may present as a breast mass and can mimic breast carcinoma clinically and radiologically, causing diagnostic dilemmas for the treating physician and pathologist. Herein, we discuss a case of nodular hidradenoma in a 20-year-old Hispanic woman as a rapidly growing mass in the breast that mimicked breast carcinoma. We discuss the rare presentation of this uncommon tumor and the differential diagnosis of this entity, as well as the results of our literature review on the topic.

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OBJECTIVES: The purpose of this study was to evaluate the imaging features of clear cell hidradenoma on ultrasonography (US), computed tomography (CT), and magnetic resonance imaging (MRI). METHODS: The radiologic and pathologic databases at 2 medical institutions were searched retrospectively from 2004 to 2016 to identify patients with a diagnosis of clear cell hidradenoma. Ultrasonographic, CT, and MRI features were described, and pathologic specimens were reviewed. RESULTS: There were 5 female and 4 male patients. The mean patient age was 48.9 years (range, 28-70 years). Five patients underwent only US; 2 patients underwent only CT; 1 patient underwent both US and CT; and 1 patient underwent US and MRI. Most of the tumors were located primarily in the subcutaneous fat layer. The mean tumor size was 18.4 mm. On US, 6 masses had a heterogeneous echo texture, including an anechoic portion with protruding echogenic portions. Two masses had multiple septa in the anechoic portion. On color Doppler US, blood flow was both central and peripheral in 5 patients. All 3 cases seen on CT presented as a low-attenuation mass with an enhanced solid internal nodule. On MRI, the mass showed heterogeneous signal intensity on T2-weighted images and enhancement of the peripheral wall and internal solid component on contrast-enhanced T1-weighted images. CONCLUSIONS: Clear cell hidradenoma is usually located in the subcutaneous fat layer, has a well-defined margin, appears as a cystic mass with an internal solid nodule, and occasionally has multiple septa on US, CT, and MRI.

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Malignant hidradenocarcinomas are rare soft tissue tumors of sweat gland origin. We present the case of a soft tissue, fungating tumor of 15 years' duration of the medial ankle in an 85-year-old male that exhibited malignant features clinically and radiographically. Subsequent punch biopsy revealed a diagnosis of malignant hidradenocarcinoma. Given the risk of recurrence and the poor radiation and chemotherapy options, the patient initially decided to leave the lesion untreated. However, he soon developed lower extremity cellulitis from the exposed lesion and decided to have the tumor excised, eliminating the source of the infection. In the present case study, we discuss the etiology, clinical and radiographic characteristics, and treatment options for this rare lesion. At the 18-month follow-up visit, he had had no recurrence of the lesion.

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BACKGROUND: Clear cell hidradenoma and cutaneous clear cell renal cell carcinoma (CCRCC) overlap morphologically. The distinction may be difficult in a patient with a history of CCRCC, presenting with a cutaneous nodule, potentially leading to an erroneous diagnosis. We investigated the usefulness of napsin A and paired box gene 8 (PAX-8) with previously studied markers epithelial membrane antigen (EMA), carcinoembryonic antigen (CEA), vimentin and cluster of differentiation marker 10 (CD10) in differentiating CCRCC from hidradenoma. METHODS: We evaluated hidradenomas and cutaneous CCRCCs for immunohistochemical expression of napsin A, PAX-8, EMA, CEA, vimentin and CD10. RESULTS: PAX-8 was expressed in all CCRCCs (8/8) while negative in hidradenomas. Napsin A was negative in both hidradenomas (0/12) and CCRCCs (0/10). EMA showed membranous reactivity in 11 of 12 hidradenomas and 8 of 10 CCRCCs; and highlighted ductal epithelium in 1 of 12 hidradenomas and cystic areas in 4 of 10 CCRCCs. CD10 showed ductal expression in 3 of 12 hidradenomas and membranous staining in 8 of 9 CCRCCs. CEA highlighted ductal epithelium in 11 of 12 hidradenomas while absent in CCRCCs (0/10). Vimentin highlighted neoplastic cells in 8 of 8 CCRCCs and failed to stain the hidradenomas (0/12). CONCLUSION: A conservative immunohistochemical panel including PAX-8, vimentin and CEA allow for easy distinction of CCRCC from hidradenoma, whereas napsin A added no additional value.

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Cutaneous clear cell hidradenoma is an uncommon benign adnexal tumor which is not supposed to metastasize, contrary to its rare malignant counterpart, hidradenocarcinoma. We report the case of a 49-year-old man, who had had a stable inguinal lymph node enlargement for 6 years. An excision was performed and revealed an intra-nodal tumor, made of large clear cells with abundant cytoplasm and round nuclei without atypia or mitosis. The immunohistochemical staining showed diffuse positivity for keratin AE1/AE3, keratin 5/6 and p63, and focal staining with keratin 7, epithelial membrane antigen (EMA) and carcinous epithelial antigen (CEA), which underlined some ductular structures. Tumor cells were negative for renal markers PAX8 and CD10. Ki67 stained less than 1% of tumor cells. A translocation involving MAML2 gene was evidenced by fluorescence in situ hybridization (FISH) analysis. No primary cutaneous tumor was found after extensive examination. Altogether, these results are in favor of an isolated nodal hidradenoma, for which we discuss two hypothesis: a primary nodal lesion, or a 'benign metastasis' of a cutaneous tumor. Cases of morphologically benign hidradenoma with lymph node involvement are exceptional. Our case, similar to every other reported case, was associated with an excellent prognosis, supporting the idea that these patients should not be overtreated.

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AIMS: High expression of carbonic anhydrase IX (CA IX) has been reported in clear cell renal cell carcinoma (ccRCC); few studies have reported CA IX expression in other tumours with predominantly clear cell morphology. The aim of study was to examine the expression and diagnostic implications of CA IX in these latter tumours. METHODS AND RESULTS: An immunohistochemical study was performed of 159 tumours with predominantly clear cell morphology. The results showed that, in addition to primary (25/25) and metastatic (10/11) ccRCC, CA IX was also expressed in breast (2/2), pulmonary (3/5) and hepatic (1/4) clear cell carcinoma, urothelial carcinoma with clear cell change (3/6), clear cell meningioma (4/6) and ependymoma (2/3), haemangioblastoma (10/10), and clear cell hidradenoma (5/6). However, while strong and diffuse positivity for CA IX was observed in ccRCC, clear cell breast carcinoma, haemangioblastoma, and clear cell hidradenoma, the other cases showed predominantly focal positivity for CA IX. In particular, CA IX staining was often seen at the periphery of necrotic areas. CONCLUSIONS: Our results indicate that strong and diffuse CA IX expression may be useful for differentiating ccRCC from several clear cell tumours, with the exception of clear cell breast carcinoma, haemangioblastoma, and clear cell hidradenoma.

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Clear cell hidradenoma (CCH) is a benign tumor of skin appendage. This lesion is commonly seen on the head, face, extremities and rarely in the oral cavity. The clinical appearance of this lesion is not specific and differential diagnosis from other lesions, both benign and malignant, can only be made after complete removal of the lesion. Histopathology of these lesions is often confused with tumors of salivary glands because of their striking resemblance. In this case of oral CCH, histopathology was an important aid in the diagnosis and hence, CCH should be considered in the differential diagnosis of lesions of the oral cavity.

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Clear cell hidradenoma (CCH) is an uncommon variant of benign cutaneous adnexal tumors. These tumors are clinically asymptomatic, solitary dermal nodules. They occur most frequently on the scalp, face abdomen and extremities. Growth is slow and malignant change is rare. 45-year-old woman presented with a nodule which had begun 4 years ago as a small nodular asymptomatic lesion and had a central ulceration and a minimal hemorrhagic discharge on her anterior abdomen wall. On dermatologic examination there was a 6.5×5×4 cm non-tender, soft reddish purple nodule, with lobular appearance and ulceration. In the laboratory investigations, all hematologic and biochemical tests were normal. A computed tomography (CT) scan demonstrated a cystic tumor with lobulated contour with contrast enhancement. The lesion was excised totally. In histopathological examination, the tumor was composed of biphasic smaller dark polygonal cells and larger clear cells and coarse nuclear chromatine. There were duct like structures. Immunohistochemical investigation was done for the suspicion of malignancy. Cytoplasm of clear cells and of duct like structures showed PAS-positive and d-PAS resistant staining. There was a positive reaction to epithelial membrane antigen and carcinoembryonic antigen. The mitotic index in Ki 67 examination was low. All these findings confirmed the diagnosis of benign CCH.

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Clear cell hidradenoma is a relatively rare benign cutaneous tumor. Treatment of choice is surgical excision but there have been some reports in Korea of recurrence of clear cell hidradenoma after surgical excision. We report a case of a 33-year-old woman with recurrent clear cell hidradenoma which recurred twice on her left anterior thigh. The first lesion was noticed 8 years ago and excised surgically 6 years ago. Three years after the operation, the lesion recurred on the operation site and was excised again. But 3 months ago the lesion appeared again, so we performed wide, complete excision and made a diagnosis of clear cell hidradenoma.

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PURPOSE: Clear cell hidradenoma, now regarded as an eccrine sweat gland tumor on the basis of its enzyme histochemical and electron microscopic features, occurs as a solitary tumor in most instances. METHODS: A 17 year old male presented with asymptomatic nodule, which had developed on nipple with a four years of history and total excision and purse-string suture was then performed. RESULTS: No recurrence was observed 2 months after excision. Histologically, it showed a well circumscribed tumor composed of characteristic clear epithelial cells which are focally arranged in glandular patterns. CONCLUSION: This case is unique in that the tumor developed on the young male nipple, unusual site and this report emphasizes the benefit of local excision to prevent recurrence of these tumor.

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Malignant clear cell hidradenomas are rare tumors originating from the eccrine sweat gland. Usually, they are malignant from their inception, but some develop from benign nodular hidradenomas. Although there is insufficient evidence in the literature, the recurrence rate may be estimated at about 50%, and the metastasis rate is about 60%, including regional nodes, bone, viscera, and the skin. We report a case of malignant clear cell hidradenoma in a 63-year-old woman, who had a 3-year history of asymptomatic pea sized hard solitary nodules on the right forearm. Despite repetitive surgical excisions, the lesions were recurred 5 times. To our best knowledge this is the first case of malignant clear cell hidradenoma in the Korean dermatologic literature.

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