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Nodular hidradenoma is an infrequent benign tumor originating from the proximal portion of the sweat glands, most commonly associated with the apocrine glands. Owing to its variable clinical presentation, correctly diagnosing nodular hidradenoma can be challenging, with several potential conditions in the differential diagnosis to consider. This article presents a healthy 52-year-old woman with an atypical location of nodular hidradenoma, highlighting the critical role of integrating clinical, dermoscopic, and histopathological characteristics for an accurate diagnosis. We discuss the clinical features, dermoscopic findings, histological examination, differential diagnosis, and treatment options for nodular hidradenoma, emphasizing the importance of surgical intervention in preventing potential malignant transformation.

Rationale: This article presents a challenging case involving an elderly male patient with a misdiagnosed intraductal mammary papilloma initially identified as a sweat adenoma through ultrasound imaging. The study aims to explore the histopathology, clinical presentations, and sonographic features of both conditions, emphasizing the contributing factors to the diagnostic misstep., Patient Concerns: A 61-year-old male reported a persistent left breast mass, along with pain and swelling, spanning a 6-month duration., Diagnoses: Ultrasound examination indicated a deep, square, mixed-echo mass in the left nipple, initially suggestive of a sweat adenoma. However, subsequent pathological analysis following resection under general anesthesia confirmed an intraductal papilloma., Intervention: The patient underwent surgical resection of the left breast mass under general anesthesia., Outcome: Post-surgery, the patient exhibited satisfactory recovery; however, regrettably, he was lost to follow-up., Lessons: This study underscores the challenge in differentiating between clear cell sweat adenoma and male intraductal mammary papilloma solely based on ultrasonic characteristics. It emphasizes the susceptibility of ultrasound-based diagnoses to misinterpretation, highlighting the critical need for a comprehensive pathological examination to establish a definitive diagnosis., Competing Interests: The authors have no funding and conflicts of interest to disclose., (Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.)

Abstract: The distinction between digital papillary adenocarcinoma (DPAC) and benign cutaneous adnexal tumors is clinically important and can be challenging. Poroid hidradenoma frequently occurs at acral sites and can show a number of histological features, which overlap with digital papillary adenocarcinoma. Recent work has shown that YAP1-NUTM1 fusions are frequent in poroid hidradenoma and are associated with nuclear protein in testis (NUT) expression by immunohistochemistry. We evaluated the expression of NUT-1 by immunohistochemistry in 4 cases of DPAC and 4 cases of poroid hidradenoma. Three of 4 cases of poroid hidradenoma showed strong NUT-1 expression, with no staining in any of the cases of DPAC. These results suggest that NUT-1 immunohistochemistry may be a useful additional tool in evaluating this differential diagnosis., Competing Interests: The authors declare no conflicts of interest., (Copyright © 2023 Wolters Kluwer Health, Inc. All rights reserved.)

Abstract: Digital papillary adenocarcinoma is a malignant adnexal tumor with a predilection for acral sites. Hidradenoma is a benign solid and cystic sweat gland neoplasm with focal ductal and glandular differentiation and good outcomes. Hidradenomas can occur at acral sites and show papillary structures; for this reason, they are included in the differential diagnosis of digital papillary adenocarcinoma, and immunohistochemistry is a valuable tool in this scenario. We described a case of a 43-year-old man with an epithelial tumor showing papillary structures in the intermediate phalanx of the fourth finger. There was diffuse positivity for p63 and negativity for S100 protein, suggesting that this tumor was an acral hidradenoma with papillary structures., Competing Interests: The authors declare no conflicts of interest., (Copyright © 2022 Wolters Kluwer Health, Inc. All rights reserved.)

Digital papillary adenocarcinoma (DPAC) is a rare tumor of sweat gland origin that preferentially affects the digits and has the potential to metastasize. Its tumor diagnosis can be difficult. Well-differentiated variants of DPAC can be confused with a benign sweat gland tumor, in particular nodular hidradenoma. With the recent detection of HPV42 DNA in DPAC by next-generation sequence analysis, we reasoned that this association could be used for diagnostic purposes. To this end, we performed in situ hybridization for HPV42 on 10 tumors diagnosed as DPAC as well as 30 sweat gland tumors of various histology types, including 8 acral hidradenomas. All DPAC were positive for HPV42. Positive hybridization signals for HPV42 were seen in both primary and metastatic DPACs. All other tumors and normal tissues were negative. This study confirms the association of HPV42 with the tumor cells of DPAC through in situ hybridization. The positive test result in all lesions of DPAC and lack of detection of HPV42 in any of the acral hidradenomas or other sweat gland tumors examined in this series is encouraging for the potential diagnostic utility of the assay. As documented by two scrotal tumors of DPAC, the in situ hybridization test for HPV42 can also help support the rare occurrence of this tumor at a non-acral site., (© 2022. The Author(s), under exclusive licence to United States & Canadian Academy of Pathology.)

Aims: Clear cells formed due to depositions of glycogen or lipids in the cytoplasm commonly occur in various tissues. Adipophilin (ADP), a lipid regulatory protein, is closely related to lipid droplets. This study aims to examine adipophilin expression in clear cells of various skin lesions., Methods: ADP expression was examined using immunohistochemistry in 108 sections from 15 skin lesion types with clear cell histology, namely, sebaceoma (n=16), sebaceous adenoma (n=3), sebaceous carcinoma (n=12), xanthomata cutis (n=10), xanthogranuloma (n=8), Paget's disease (n=10), Bowen disease (n=10), hidradenoma (n=9), atypical lipoma (n=5), superficial lipomatous nevus (n=5), metastatic renal cell carcinoma (n=5), squamous cell carcinoma (n=4), seborrheic keratosis (n=4), dermatofibroma (n=4) and clear cell sarcoma (n=3)., Results: ADP was not expressed in Bowen disease, hidradenoma or seborrheic keratosis. Four expression patterns, foamy, reticular, granular and punctate, were summarised based on their expression in clear cells. Different expression patterns were related to tissue origin and differentiation degree. Foamy expression was commonly observed in lesions with mature sebaceous glands and xanthomas; reticular expression in adipocytes; granular expression in xanthoma, xanthogranuloma and metastatic renal carcinoma and punctate expression in sebaceoma, sebaceous carcinoma, Paget's disease, squamous cell carcinoma and clear cell sarcoma. Furthermore, stronger staining with focal vesicular labelling was noted in sebaceoma than in sebaceous carcinoma. Characteristic labelling was noted, including the circular distribution in Touton giant cells of xanthogranulomas and focal distribution in the clear cells along the edge of necrotic tissue in clear cell sarcoma., Conclusions: ADP is useful in identifying intracytoplasmic lipids and can be used to diagnose skin lesions with clear cell histology, especially in some lesions with characteristic labelling., Competing Interests: Competing interests: None declared., (© Author(s) (or their employer(s)) 2022. No commercial re-use. See rights and permissions. Published by BMJ.)

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., (© 2022 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)

Eccrine acrospiroma is a benign tumor of skin and adnexa arising from eccrine sweat gland epithelium. It is usually solitary, slow growing tumor commonly affecting extremities. Rarely it affects head and neck region, and extremely rare in parotid region. Females are affected more often. Treatment of choice is wide local excision with adequate skin margins. Although benign this tumor is very notorious for recurrence after inadequate resection. We describe here a case of young male patient with recurrent eccrine acrospiroma over parotid region which was managed by wide local excision with primary repair with excellent results.

DICER1 tumor predisposition syndrome is a rare genetic disorder that predisposes individuals to multiple benign and malignant neoplasms. The phenotype is vast and includes pleuropulmonary blastoma (PPB), thyroid nodules, cystic nephroma, Wilms tumor, ovarian Sertoli-Leydig cell tumor, and medulloepithelioma, among others. Herein, we describe a patient with a DICER1 germline pathogenic variant presenting with two neoplasms that are not commonly encountered in the context of DICER1 syndrome. The first tumor is a multiloculated cystic hepatic lesion with a biphasic pattern, composed of cysts lined by bland biliary type (CK19-positive) epithelium surrounded by a condensation of sarcomatous spindled cell proliferation in a myxoid stroma. This neoplasm resembled PPB or cystic nephroma with malignant transformation. The second tumor is a chest nodule consistent with low-grade hidradenocarcinoma. Although it is difficult to speculate with just a single case, these unusual neoplasms occurring in particular at a young age raises the possibility that they can be inherent to, and thus, be part of the DICER1 tumor predisposition syndrome phenotype.

We report a case of clear cell hidradenoma of the perineum that was initially misinterpreted as a papillary urothelial carcinoma, either metastatic or of Bartholin gland origin, on initial excisional biopsy. The misinterpretation may have been due to the pseudopapillary architecture and GATA3-positivity of the biopsy tissue. Clear cell hidradenomas often show a range of histologic growth patterns and cellular differentiation and are one of many tumors that react immunohistochemically with GATA3. Although rare, these tumors can occur in the genital region and can mimic malignant tumors such as metastatic renal cell carcinoma and carcinomas of the genitourinary tract. This report details the morphologic and immunohistochemical pitfalls that make accurate diagnosis of clear cell hidradenoma in this unusual location challenging., Competing Interests: The authors declare no conflict of interest., (Copyright © 2020 by the International Society of Gynecological Pathologists.)

Solid-cystic hidradenoma is a benign cutaneous tumor that is most commonly found in the head, neck, trunk, and upper extremity regions of patients in the middle to older aged groups. Presentation on lower extremities and in particular on the foot is uncommon. Nodular hidradenomas represent a dermatological pitfall, being difficult to differentiate from basal cell carcinoma and melanoma. We report on a 53-year-old male patient with a pigmented nodular hidradenoma on his ankle that was surgically removed. We discuss histopathology and differential diagnosis of this eccrine tumor of skin. This is the second reported case in the English literature.

Hidradenoma is a rare benign neoplasm, with few cases reported to involve the eyelid. When affecting the eyelid, hidradenomas may mimic other benign or malignant lesions. Rarely, a benign hidradenoma may transform into a malignant hidradenoma and metastasize. The authors present a case of a benign hidradenoma arising from the eyelid, presenting with rapid growth, ulceration, and bleeding, suggestive of a malignant lesion., Competing Interests: The authors have no financial or conflicts of interest to disclose., (Copyright © 2021 The American Society of Ophthalmic Plastic and Reconstructive Surgery, Inc.)

Malignant eccrine spiradenoma is an extremely rare neoplasm of adnexal origin. It almost always originates from a preexisting long standing eccrine spiradenoma. We present a case of malignant eccrine spiradenoma arising from benign counterpart and having both carcinomatous and sarcomatous differentiation. Here we present a case of a 46 years old lady who presented with a long standing small nodule on her left leg of 7 years ' duration with suddenly increase in size. Grossly the mass was partly solid and partly cystic measuring 11.5 cm in maximum dimension with cystic area forming the deeper plane. On microscopy, the superficial dermis showed well demarcated lobules of benign eccrine spiradenoma. Deeper dermis showed tumor cells with features of malignant transformation having both carcinomatous and sarcomatous component. After wide local excision patient is now doing well. The diagnosis of malignant eccrine spiradenoma requires a thorough histopathological examination of the lesion and requires finding a focus of benign spiradenoma within or adjacent to malignant tumour. Wide local excision and close follow-up for early detection of recurrence and metastasis is the mostly recommended management modality., Competing Interests: None

Adenomyoepithelioma is an extremely rare primary cutaneous neoplasm. Although there is ample evidence on the existence of malignant adenomyoepithelioma in the breast, a malignant counterpart in the skin has not been documented. We report a primary cutaneous adenomyoepithelioma (pcAME) with malignant features arising from a spiradenoma in a 39-year-old female patient. The tumor was solid-cystic in appearance and entirely located in the subcutaneous tissue. Histologically, the tumor displayed foci of adenomatous changes and adenomyoepitheliomatous hyperplasia adjacent to a minute spiradenoma. Gradual increase of architectural complexity, cytologic atypia, mitotic activity, and infiltrative growth were observed in a significant portion of the neoplasm, indicative of transformation to adenomyoepithelioma and subsequently low- to high-grade salivary-type epithelial-myoepithelial carcinoma (EMCA). The intimate dual populations of ductal and myoepithelial cells were highlighted by a panel of immunohistochemical stains in all different components of the tumor. Molecular studies revealed a PIKCA3 mutation, a genetic aberration that has been documented in EMCA, particularly of breast origin. The current case documents for the first time a pcAME with malignant features arising from a spiradenoma and suggests adenomyoepithelioma ex spiradenoma as a possible tumorigenesis pathway of this rare cutaneous tumor.

Eccrine spiradenoma is a rare lesion originating from eccrine sweat glands, with only few cases reported in breast tissue: we here describe for the first time, an eccrine spiradenoma arising in the nipple. An 84 year-old woman with a lesion enlarging her right nipple, showing ulcerations and eczema-like changes of the covering skin, was admitted to our hospital. Surgical excision of the central quadrant with nipple-areola complex was performed, followed by histopathological evaluation which revealed an adenoma with predominantly basaloid epithelial cells. The lesion was composed of tightly packed small and large groups of cells, arranged in diffuse alveolar/pseudorosette formations. The small cells expressed p63 and calponin, while a positive expression of CK7 and CD117 was detected in large cells. After careful and detailed examination, excluding various similar entities, a diagnosis of eccrine spiradenoma has been rendered. Although extremely rare, eccrine spiradenoma should be taken into account in the differential diagnosis of subcutaneous primary breast tumors.

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., (© American Society for Clinical Pathology 2019. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.)

Tissue-sparing biopsy techniques are frequently used in clinical practice but can result in misdiagnosis of large, clinically challenging lesions. We present a case of a 70-year-old man with hidradenocarcinoma of the left chin, a diagnosis that was delayed owing to repeated superficial biopsies that were negative for disease. Diagnosis was ultimately obtained via an incisional biopsy. We discuss the clinical features of hidradenocarcinoma and why this diagnosis can be easily missed with superficial biopsies. It is important that dermatologists consider incisional biopsies in the workup of clinically challenging lesions for which malignancy is considered.

Aims: Hidradenoma 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 investigate oncogenic mutations involved in the tumourigenesis of HP. We focused specifically on PIK3CA and AKT1 mutations, which are both reported to be detected in 33% of mammary intraductal papillomas., Methods: In total, seven HP cases were analysed. Clinicopathological analyses and immunohistochemistry for oestrogen receptor, p63, smooth muscle actin (SMA), p53 and β-catenin were performed. Furthermore, PIK3CA , AKT1, BRAF and KRAS hot spot mutations were examined by Sanger sequencing., Results: Morphologically, all HPs had a papillary and tubular architecture with a biphasic pattern of epithelial and myoepithelial cells. Immunohistochemistry revealed that oestrogen receptor expression was restricted to epithelial cells, whereas p63 and SMA were exclusively expressed in myoepithelial cells. The patterns of p53 and β-catenin immunostaining suggested wild-type genotypes. Direct sequencing revealed the presence of somatic PIK3CA mutations (Ex9. c.1633G>A, p.E545K and Ex20. c.3140A>G, p.H1047R) in two of the HPs and an AKT1 (c.49G>A, p.E17K) mutation in one. BRAF and KRAS mutations were not found in any of the HP cases., Conclusions: PIK3CA and AKT1 are frequently mutated in HP tumours (29% and 14%, respectively). PIK3CA/AKT1 pathway alterations in HP further support the hypothesis that HP is the vulvar (anogenital mammary-like gland) analogue of breast intraductal papilloma., Competing Interests: Competing interestsNone declared., (Published by the BMJ Publishing Group Limited. For permission to use (where not already granted under a licence) please go to http://www.bmj.com/company/products-services/rights-and-licensing/.)

An 81-year-old man presented to the dermatology clinic with a painful lesion on his chest. The nodule would occasionally bleed and leak serous fluid for 10 years. Physical examination revealed an unspecified nodule with two superimposed nodules. A deep shave biopsy of the lesion was obtained and expressed a solid-cystic dermal neoplasm that was comprised of an admixture of cell types. Through the presenting clinical and histological features seen, a final diagnosis of nodular hidradenoma was made.

A 46-year-old woman was evaluated for a "recurring papilloma" of the left medial upper eyelid margin. Beneath the papillary lesion medial to the punctum was a 5-mm diameter cutaneous mass thought to be cystic. After excisional biopsy, histopathologic analysis documented the presence of an epidermal keratinizing squamous papilloma surmounting a circumscribed dermal papillary hidradenoma composed of deeply eosinophilic columnar cells. Additionally, there was intraductal proliferation of tumor extending toward a subclinical poral opening through the epidermis. Immunohistochemistry proved the apocrine nature of the benign, non-cystic lesion by virtue of its nuclear androgen receptor and cytoplasmic gross-cystic disease fluid protein-15 positivity, along with its smooth muscle actin-positive myoepithelial layer. This and prior cases establish that apocrine tumors, both benign and malignant, are strictly localized at or near the eyelid margin where only apocrine glands are found. These tumors are more often papillary than solid adenomas, and most exceptionally can be malignant. We review the differential diagnosis of simulating eccrine eyelid tumors. We recommend wide local excision for benign lesions, in view of possible intraductal extension that can be eccentric to the main tumor and the miniscule potential for malignant transformation., (Copyright © 2014 Elsevier Inc. All rights reserved.)

Hidradenocarcinomas are rare, yet highly malignant tumors of eccrine sweat gland origin. Due to its locally aggressive growth and likelihood for metastasis it should be considered as a differential diagnosis especially in case of suspicious intraoperative findings. We report the case of a 73-year-old female patient presenting with a hidradenocarcinoma of the wrist. Treatment of hidradenocarcinomas is similar to the treatment of sarcomas: The first step is an incisional biopsy and obtaining an expert second opinion on the histopathological diagnosis as well as staging. The second step is a clear margin resection of the tumor and the plastic-surgical reconstruction. A long-term follow-up is mandatory to detect potential recurrence or metastasis., (© Georg Thieme Verlag KG Stuttgart · New York.)

Hidradenomas are adenomatous tumors of sweat gland origin that are usually benign and can occur in any cutaneous skin surface. When malignant, the tumors have high recurrence rates and are often fatal. Because of their infrequency and clinical ambiguity, hidradenomas are often misdiagnosed as other soft-tissue masses. Radiation and chemotherapy have not shown to be of benefit; therefore, wide aggressive excision and lymphadenectomy are currently the treatments of choice. We present the case of a patient with a malignant hidradenoma presenting in her medial heel. After excisional biopsy identified the tumor, the patient was successfully treated with wide excision and delayed closure via vacuum-assisted closure.

Clear cell hidradenocarcinomas are extremely rare neoplasms, with very few well-documented cases reported in the literature. The most common sites are the head and neck regions. These tumors are histologically malignant but are not always aggressive. They are known for recurrence and may metastasize widely. Treatment is wide local resection. We report on a case of clear cell hidradenocarcinoma occurring over the eyelid together with a review of the literature.

Hidradenoma papilliferum is a benign adnexal tumor originating from apocrine glands of skin. It is commonly seen in the anogenital region of middle-aged women and presents as an asymptomatic, slow-growing, solitary nodule. Rarely this tumor occurs at extragenital sites and is called ectopic hidradenoma papilliferum. Although head and neck is a common site for this group, only 3 cases in the upper eyelid have been described. We present the first case of ectopic hidradenoma papilleferum presenting as a cystic swelling in the lower eyelid. A brief review of literature is included.

The authors present a case of seemingly textbook digital osteomyelitis with draining ulceration. Per protocol, specimens were sent to pathology with the intent to verify and identify the offending organisms. Surprisingly, the textbook osteomyelitis returned with a pathological diagnosis of rare sweat gland tumor, malignant eccrine acropsiroma, prompting immediate systemic medical workup and treatment. This case emphasizes the importance of proper laboratory workup of all surgically removed specimens, despite having a "typical" presentation.

Hidradenoma papilliferum (HP) is a rare, small, benign, cystic, papillary, slow growing tumor occurring in anogenital region of adult women. The authors describe a case of large, rapidly enlarging HP of vulva in a 25-year-old woman and review the literature available on this rare condition.

Hidradenomas are rare adnexal tumors. Recently, two groups have been individualized: those with eccrine differentiation called poroid hidradenoma and those with apocrine differentiation called clear-cell hidradenoma or nodular hidradenoma. Herein we report a case of 19-year old woman with a nodular hidradenoma of the scapular region. Our report highlights the clinic-pathological characteristics of these tumors and emphasizes the benefit of complete local excision to prevent risk of recurrence and possible malignant potential.

Background: Malignant eccrine spiradenoma is an aggressive sweat gland tumor with poorly understood behavior and no currently accepted therapeutic regimen., Methods: An individual patient data meta-analysis with Kaplan–Meier survival curves was performed on 72 reported cases of malignant eccrine spiradenoma., Results: In 35 patients with no distant metastasis, local resection resulted in 100% disease-free survival. Of 7 patients with lymph node but no distant metastasis treated with surgical resection and lymph node dissection, 6 patients remained disease-free at final follow-up evaluation. For the 24 cases with confirmed distant metastatic disease, patient survival did not significantly differ between local resection and surgery with adjuvant chemoradiotherapy (P = .8763)., Conclusions: Heightened awareness is recommended among surgeons likely to treat this entity. An aggressive surgical approach is supported in the absence of metastasis. When lymph nodes are not clinically involved, sentinel node may have a role followed by lymph node dissection in patients with a positive node., (Published by Elsevier Inc.)

Poroid neoplasm is a benign sweat gland neoplasm that accounts for 10% of sudoriferous tumors. Poroid hidradenoma is an uncommon variant that usually affects adults, with a peak of incidence in the seventh decade. It is rare in children. We report the first case of poroid hidradenoma presenting in a 13-year-old boy., (© 2011 Wiley Periodicals, Inc.)

Eccrine acrospiroma, better known as eccrine poroma, is a benign adnexal neoplasm of the skin. Its clinical aspect can masquerade as some other nodular and cystic lesions. The current dermoscopy literature offers very few case studies. Moreover, these very few examples present a totally different appearance pattern compared to the one we examined. Its homogeneous blue pattern suggested the better-known nodular malignant melanoma; in fact, this dermoscopic appearance was due to the Tyndall effect.