Hidradenocarcinoma: A Case Series From the Scripps Clinic With a Systematic Review of the Literature.

BACKGROUND: Hidradenocarcinoma (HAC) is a rare adnexal carcinoma. To the best of the authors' knowledge, there are no published systematic reviews on HAC. OBJECTIVE: To incorporate a case series from the authors' institution and systematically integrate reported information to provide a reference tool for optimization of diagnosis and management. METHODS: A comprehensive MEDLINE search was conducted from database inception to 2021 using Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. This yielded 225 studies with 165 cases of HAC. References of included articles were also searched. In addition, 9 patients with HAC were identified from the authors' institution over the past 10 years. RESULTS: The mean age of HAC presentation is 60 years with a slight male predilection (60%). The head and neck is the most commonly affected region. Over 36% of cases either presented with metastatic disease or went on to metastasize. The most common treatment type was wide local excision, followed by Mohs micrographic surgery. CONCLUSION: Early detection with accurate histologic interpretation is prudent in all cases of HAC. Wide local excision is the current first-line treatment. However, Mohs micrographic surgery offers complete marginal analysis with evidence of reduced risk of metastasis and better outcomes compared with wide local excision. Currently, there are no National Comprehensive Cancer Network guidelines for the treatment of HAC, and consensus guidelines are limited to tumor and nodal metastasis staging provided by the American Joint Committee on Cancer, eighth edition. Thus, this case series and systematic review integrates important aspects of diagnosis, workup, and management of HAC.

Spiradenoma With Adenoid Cystic Carcinoma-like Changes: A Case Series of This Rare Variant With a Potential Diagnostic Pitfall.

Spiradenomas are benign cutaneous adnexal neoplasms with sweat gland differentiation that can manifest a broad spectrum of histomorphologic appearances. While they show a characteristic histopathologic phenotype and clinical management involves surgical excision with a low risk of recurrence, there have been unusual histopathologic variants of spiradenoma reported, including cases with adenoid cystic carcinoma (ACC)-like changes. Primary cutaneous ACC is a low-grade malignancy presenting as a subcutaneous mass with the potential for local invasion, perineural invasion, and high rates of local recurrence after excision. The diagnosis of spiradenomas with ACC-like features can be challenging, especially when only the ACC-like component is present for evaluation. A retrospective analysis of 21 cases of spiradenoma with ACC-like changes were obtained from large academic institutions, was performed, and summarized below. All cases showed background of conventional spiradenoma, and the ACC-like areas represented a component in all lesions. The percentage of ACC-like component ranged from 5% to 40% in all cases. The ACC-like component was multifocal and without pleomorphism, perineural and/or vascular invasion, necrosis, or increased mitotic activity. MYB translocation and protein expression was studied in 16 cases by fluorescence in situ hybridization, polymerase chain reaction, and immunohistochemistry. All studied cases were negative for MYB / NFIB , MYB L1, and MYB F by fluorescence in situ hybridization and polymerase chain reaction and 3 cases were positive for MYB expression by immunohistochemistry. Our study expands on spiradenomas with ACC-like features that ought to be considered in the differential diagnosis of cutaneous neoplasms such as primary cutaneous ACC. Our results indicate that a thorough histopathologic inspection and strict application of well-defined histologic criteria are necessary to support the diagnosis of this unusual histopathologic variant. These tumors can be difficult to diagnose, and awareness of their histomorphologic spectrum will facilitate definitive diagnosis and avoid misdiagnosis with other conditions.

Low-grade Hidradenocarcinomas: A Clinicopathologic Study of an Unusual Carcinoma That Can Mimic its Benign Counterpart.

Hidradenocarcinomas are rare cutaneous adnexal malignancies with sweat gland differentiation that can show a broad spectrum of histomorphologic appearances, ranging from low to high grade. The diagnosis of low-grade hidradenocarcinoma can be challenging and may be mistaken for benign hidradenomas, especially on superficial and partial samples. We performed a retrospective analysis of 16 low-grade hidradenocarcinomas, obtained from 4 large academic institutions. All neoplasms presented clinically as nodular lesions that ranged in size from 1.5 to 6.0 cm. All patients were adults and their age ranged from 33 to 74 years of age. All cases shared features similar to hidradenomas in the surface and mid portion of the tumors and all tumors had 1 or more histomorphologic clues to malignancy, including the presence of an asymmetric and infiltrative growth pattern (especially at the base of the tumors), perineurial invasion, and a desmoplastic stromal reaction. In the tumors evaluated for immunohistochemistry, the tumor cells were positive for p63, EMA, AE1/AE3, MNF116, and CK7. Three patients underwent sentinel lymph node biopsy, and 2 cases showed metastatic disease to regional lymph nodes. All cases (including the 2 cases that had regional lymph node metastasis), showed no local recurrence or distant metastasis observed after a complete re-excision of the tumors (follow-up range from 6 to 72 mo). Our study highlights the salient clinical and histopathologic features of low-grade hidradenocarcinomas and emphasizes the potential diagnostic pitfalls in distinguishing this entity from other neoplasms. Our results indicate that a combination of thorough histopathologic inspection is necessary to support the diagnosis of this rare neoplasm. These tumors can be exceedingly difficult to diagnose and awareness of the subtle features of low-grade hidradenocarcinoma is of importance are as it remains a diagnostic challenge for practicing pathologists.

Eccrine Hidradenoma of the Nasal Vestibule: A Rare Clinical Entity.

Eccrine hidradenoma is a relatively rare benign tumor of sweat gland origin but with possible malignant transformation. It usually consists of solitary, well-demarcated papules or nodules covered with normal skin. Common sites of involvement are the scalp, face, limbs, and anterior trunk. Although the lining of the nasal vestibule includes hair follicles, sebaceous glands, and sweat glands, an eccrine hidradenoma originating in the nasal vestibule has yet to be reported. Herein, we describe a rare clinical presentation of nasal eccrine hidradenoma, treated successfully using a transnasal endoscopic approach.

Clinicopathological and genomic copy number variation analysis in nodular hidradenoma and hidradenocarcinoma with focus on prognostically important features.

Nodular hidradenoma is a cutaneous adnexal tumor of sweat gland origin, characterized by its diverse but overlapping histomorphologic features with other skin tumors. In addition, distinction of benign hidradenoma and its malignant counterpart hidradenocarcinoma can be challenging, especially in prognostic prediction. We retrospectively reviewed pathological features of 29 cases, including benign nodular hidradenoma (n = 17) and hidradenocarcinoma (n = 12), with clinical follow-up ranging from 18 to 216 months. Genomic copy number variation (CNV) was studied in selected cases (n = 18) by single nucleotide polymorphism microarray. None of the benign hidradenomas (0/17) or low-grade hidradenocarcinomas (0/6) had recurrence or metastasis after complete excision, whereas all 6 high-grade hidradenocarcinomas (6/6) showed locally destructive disease, recurrence, or local metastases. In benign hidradenomas, CNV abnormality was absent in all clear cell hidradenomas (0/5) but was detected in a considerable portion of poroid hidradenoma (3/5), with number of abnormalities ranging 2, 4, and 9. In malignant cases, regardless of morphological classification, both low-grade hidradenocarcinomas demonstrated limited CNV abnormalities in 2 areas (2/2), whereas all high-grade hidradenocarcinomas contained 8 or more CNV abnormalities (6/6). No disease-associated death was recorded in the cohort except one case was lost to follow-up after the development of metastatic disease. Overall, the findings support that genomic CNV abnormalities may serve as a sensitive but less specific tool in detecting malignancy in these tumors, and potentially have a role in predicting clinical behavior particularly in the tumors of nonporoid morphology.

Value of Immunohistochemistry to Differentiate Digital Papillary Adenocarcinoma From Acral Hidradenoma With Papillary Structures.

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.

Direct intracranial invasion of eccrine spiradenocarcinoma of the scalp: a case report and literature review.

BACKGROUND: Eccrine spiradenocarcinoma (SC), also known as malignant eccrine spiradenoma, is a rare malignant cutaneous adnexal neoplasm arising from long-standing benign eccrine spiradenoma. Malignant skin tumors rarely show direct intracranial invasion. However, once the intracranial structure is infiltrated, curative excision with sufficient margins can become extremely difficult, particularly when the venous sinuses are involved. No effective adjuvant therapies have yet been established. Here, we report an extremely rare case of scalp eccrine SC with direct intracranial invasion, which does not appear to have been reported previously. CASE PRESENTATION: An 81-year-old woman presented with a large swelling on the parietal scalp 12 years after resection of spiradenoma from the same site. The tumor showed intracranial invasion with involvement of the superior sagittal sinus and repeated recurrences after four surgeries with preservation of the sinus. The histopathological diagnosis was eccrine SC. Adjuvant high-precision external beam radiotherapy (EBRT) proved effective after the third surgery, achieving remission of the residual tumor. The patient died 7 years after the first surgery for SC. CONCLUSIONS: Scalp SC with direct intracranial invasion is extremely rare. Radical resection with tumor-free margins is the mainstay of treatment, but the involvement of venous sinuses makes this unfeasible. High-precision EBRT in combination with maximal resection preserving the venous sinuses could be a treatment option for local tumor control.

PIGMENTED NODULAR CYSTIC HIDRADENOMA OF THE ANKLE.

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.

A rare case of Hidradenoma of the lower lip.

Hidradenoma are rare benign tumors with eccrine or apocrine differentiation that most often presents as a small, firm, solitary dermal nodule. At times, hidradenoma shows striking cytologic and histologic similarity to other neoplasms, including its malignant counterpart, hidradenocarcinoma. We present the first reported case of benign hidradenoma of the lower lip. The case illustrates important diagnostic features of hidradenoma and discusses the current understanding of its malignant potential and the appropriate management of such lesions.

Hidradenocarcinoma: a rare but challenging diagnosis.

Hidradenocarcinoma is a rare, aggressive cancer arising from sweat glands. Hidradenocarcinoma has no definitive histologic features compared to its benign hidradenoma, thereby posing a diagnostic challenge for clinicians and pathologists. This novel case report demonstrates specific MRI signs that suggested malignant degeneration of hidradenoma.

Recurrent Eccrine Acrospiroma of the Parotid Region: A Case Report.

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.

Spiradenoma of the breast: a rare diagnostic pitfall in the evaluation of solid-basaloid breast lesions with a dual cell population.

Breast spiradenoma is extremely rare, with only 4 cases reported previously. We describe an instructive case of breast spiradenoma resembling adenoid cystic carcinoma (AdCC). A 71-year-old woman underwent excisional biopsy of a breast mass after a conclusive diagnosis was unable to be obtained from core needle biopsy showing an AdCC-like pattern. Histopathologically, the lesion demonstrated solid and cribriform foci comprising basaloid cells, luminal cells, and eosinophilic hyaline material, reminiscent of solid-basaloid AdCC, alongside convoluted lumens, stromal edema, lymphocytic infiltration, and c-kit negativity. On molecular analysis, neither MYB fusion genes nor CYLD gene abnormalities were identified. These results were supportive of spiradenoma. Salivary gland- and skin adnexal-type tumors are challenging to diagnose due to morphological overlaps. This case, highlighting histopathological and molecular features, shows that breast spiradenoma can be a diagnostic pitfall among the differential diagnoses of AdCC.

Rapidly Enlarging Hidradenoma of the Eyelid.

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.

Clear Cell Hidradenoma-Basal Cell Carcinoma Collision.

ABSTRACT: A 91-year-old man presented with a tumor on the left temporal area, clinically suspicious of basal cell carcinoma. The histopathologic study showed a central solid-cystic tumor composed by 3 different types of cells (clear or finely granular cells, polygonal cells, and squamoid cells). It had a sclerotic stroma. At the periphery, another tumor composed by smaller interconnected nests was evident. Some nests were separated from the stroma by clefts. The stroma of this second tumor was highly cellular. There was a sharp delimitation between both tumors, with no transitional area. Immunochemistry demonstrated they are different tumor. A diagnosis of clear cell hidradenoma-basal cell carcinoma collision was performed. To the best of our knowledge, this is the first description of this challenging association.

Tubulopapillary hidradenoma-like tumor of the mandible: Two case reports of an underrecognized entity.

Tubulopapillary hidradenoma-like tumor of the mandible is extremely rare, with only 3 cases published in the English-language literature. The clinicopathological characteristics and histogenesis of this tumor are unclear. Few pathologists and perhaps fewer clinicians are familiar with this entity, and it is likely underrecognized and under-reported. Herein, we present two additional cases, both misdiagnosed as malignancies preoperatively and postoperatively by different unwary pathologists. Awareness and knowledge of this enigmatic entity and its clinical and radiographic features, together with careful morphological assessment should enable the correct diagnosis and prevent unnecessary treatment.

Clinicopathologic Characterization of Hidradenoma on Acral Sites: A Diagnostic Pitfall With Digital Papillary Adenocarcinoma.

Hidradenomas are benign sweat gland tumors that typically present as small nodules in adulthood. Their anatomic distribution is wide and rarely includes acral sites. In this setting, reliable separation from digital papillary adenocarcinoma is important, but notoriously difficult. Hematoxylin and eosin-stained sections of 25 hidradenomas on acral skin were retrieved. The clinical presenting features and morphologic findings were recorded, and follow-up was obtained. Immunohistochemistry was performed for AE1/3, CK5/6, EMA, CEA, SMA, S100, p40, and p63. The tumors presented as solitary nodules on the hands (n=17) and feet (n=8) of adults (age range: 20 to 81 y; median: 50 y), with an equal sex distribution. Histologically, the well-circumscribed tumors were lobular, with a solid and cystic growth within dermis. Duct and squamous differentiation and vascularized hyaline stroma were frequent. The majority (n=18) were poroid hidradenomas. Scattered cytologic atypia and mitotic activity (median: 2/10 HPF) were common, and a pseudoinfiltrative growth of strands in a hyaline to sclerotic matrix was noted in 5 tumors. No papillary structures, atypical mitoses, or tumor necrosis were present. Immunohistochemically, all tumors expressed AE1/3, CK5/6, p40, and p63 strongly and diffusely. Luminal differentiation was highlighted by epithelial membrane antigen and carcinoembryonic antigen staining. S100 and SMA staining was absent. Follow-up (1 to 288 mo; median: 61 mo), available for 20 patients, showed no local recurrences and no disease-related mortality. Acral hidradenomas and digital papillary adenocarcinomas share a well-circumscribed dermal growth pattern containing solid, cystic, and tubular areas with mitotic activity and at least focal cytologic atypia. Lack of papillary structures and the diffuse positivity for p40 and p63 in the absence of S100 and SMA expression are helpful features in favor of acral hidradenoma.

Pápula eritematosa pediculada en la rodilla / Erythematous Pedunculated Papule on the Knee

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Utilidad de la ecografía en el diagnóstico diferencial de un nódulo doloroso en el tórax / Value of Ultrasound as a Diagnostic Tool for a Painful Thoracic Nodule

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