Histological findings and pathologic diagnosis of spiradenocarcinoma: A case series and review of the literature

Huang, Amy; Vyas, Nikki S.; Mercer, Stephen E.; Phelps, Robert

doi:10.1111/cup.13408

Cited by 8 publications

(11 citation statements)

References 26 publications

(34 reference statements)

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“…LGMES, first described by Leonard et al, 1 are rare tumors, mostly located in the head and neck. LGMESs are generally found among elderly patients admitted to clinics with longstanding lesions which show rapid growth 1 2 3 4 5 6 .…”

Section: Discussionmentioning

confidence: 99%

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Lymphangiectatic Variant of Low-Grade Malignant Eccrine Spiradenoma

et al. 2023

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Low-grade malignant eccrine spiradenoma (spiradenocarcinoma) is a rare sweat gland tumor, which usually arises from a pre-existing benign eccrine spiradenoma. This paper presents the case of a 55-year-old male who had a lesion in his right elbow for 10 years. The microscopic examination revealed a well-demarcated, multilobulated tumor in the dermis and subcutis, which presented with many blood-filled vessels and extensive hemorrhage. The tumor was composed of hyperchromatic, round to oval cells with nucleolar prominence, mild to moderate atypia, and increased mitotic index. Additionally, lymphangiectatic appearance was observed in areas with prominent stromal lymphedema. P53 and Ki-67 had high positivity. Surgical excision of the lesion was performed with adequate surgical margins, and the dissected lymph nodes in the axilla were tumor-negative. After 15 months of follow-up, there was no recurrence or distant metastasis.

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Section: Discussionmentioning

confidence: 99%

“…LGMESs have a very good clinical course, total excision and follow-up are sufficient as treatment, and there is no need for a sentinel lymph node biopsy and extended surgical margin 2 3 4 5 6 .…”

Section: Discussionmentioning

confidence: 99%

Lymphangiectatic Variant of Low-Grade Malignant Eccrine Spiradenoma

et al. 2023

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“…One hypothesis is that it acts as the break to G1/S transition led by GLI1-induced oncogenic accumulation of cyclins and CDK4. 24 The morphologic differential diagnosis is broad; it includes vascular neoplasms 13,[15][16][17][18]24,[30][31][32][33] such as glomus tumor/myopericytoma, [30][31][32][33] skin adnexal carcinoma (hidradenocarcinoma, spiradenocarcinoma), 34,35 solitary fibrous tumor (SFT), 36 PEComa, 37,38 myoepithelial tumors, [39][40][41] extraskeletal myxoid chondrosarcoma (EMC), 42,43 and DDLPS. 44 Vascular neoplasms usually express one or more vascular markers (ERG, CD31, CD34) 13,[15][16][17][18]24,[30][31][32][33] and despite the D2-40 immunoexpression in our cases, other vascular markers were all negative.…”

Section: Discussionmentioning

confidence: 99%

“…[30][31][32][33] Skin adnexal carcinoma (e.g., hidradenocarcinoma, spiradenocarcinoma) may show similar morphology and express D2-40, but is also expected to be positive for cytokeratins and EMA. 34,35 SFT may also have a round cell morphology and be located in the subcutaneous tissue, albeit this is rare. 36 Although STAT6 immunoreactivity has a high sensitivity and specificity for SFT, it can also be expressed in a small subset of GLI1-amplified tumors likely related to coamplification of the gene locus.…”

Section: Discussionmentioning

confidence: 99%

Superficial GLI1‐amplified mesenchymal neoplasms: Expanding the spectrum of an emerging entity which reaches the realm of dermatopathology

et al. 2022

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Mesenchymal neoplasms with GLI1 alterations (rearrangements and/or amplification) have been reported recently in several anatomic locations, which include head and neck, soft tissue, and gastrointestinal tract. Herein, to the best of our knowledge, we describe the first three cases of superficial/subcutaneous mesenchymal neoplasm with GLI1 amplification. The neoplasms exhibited low-grade cytologic features with predominant round cell morphology, glomangioma-like areas and a rich background capillary network. There were two to three mitotic figures per 10 HPF and focal necrosis in one case. The tumors exhibited variable expression of CDK4, MDM2, STAT6, D2-40, CD56 and cyclin D1. p16 had strong and diffuse nuclear and cytoplasmic expression in two cases. Numerous other stains were negative. Fluorescence in situ hybridization detected GLI1, DDIT3, and CDK4 coamplification in all cases, while next generation sequencing did not detect a GLI1 gene fusion. The overall features were compatible with a GLI1-amplified mesenchymal neoplasm. In Case 1 a new distant skin lesion appeared 1 month after the surgery exhibiting similar morphology albeit with a higher mitotic index. In Cases 2 and 3, there is no evidence of local recurrence or systemic disease after 8 years and 1 month of follow-up, respectively. These new cases of superficial GLI1-amplified neoplasm expand its clinical spectrum and enter the realm of dermatopathology. The combination of CDK4, cyclin D1, D2-40, and p16 expression with variable MDM2, STAT6, CD56, and S100 immunoreactivity in a low-grade neoplasm with round/ovoid cytomorphology resembling a vascular or adnexal neoplasm may suggest the possibility of GLI1-amplified neoplasm.

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“…1,[19][20][21][24][25][26][27][28][81][82][83][84] Among the 182 cases of spiradenocarcinoma described, 32.3% occurred on the trunk, 31.3% developed on the limbs, 30.7% on the head and neck, and a small number (1.6%) in the genital region. 1,[31][32][33][34][35][36][37][38][39][40][41][42][43][44][45][46][47][48] Interestingly, six cases were preceded by trauma to the location of the lesion, often several years before the onset of symptoms that prompted the pursuit of medical treatment. 8,15,39,50,66,84 Authors of one of these case reports hypothesized that trauma could be implicated in the pathogenesis of spiradenoma.…”

Section: Demographicsmentioning

confidence: 99%

Challenges in diagnosis and management of a spiradenocarcinoma: a comprehensive literature review

Wagner

Jassal

Lee

et al. 2021

ANZ Journal of Surgery

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Background: Spiradenocarcinoma is a rare skin adnexal neoplasm that may behave aggressively. It is often associated with a benign slow-growing spiradenoma that has undergone malignant transformation. Given the paucity of cases in the literature, there is a lack of consensus on treatment. Methods: The terms 'malignant spiradenoma' or 'spiradenocarcinoma' were systematically used to search the PubMed, MEDLINE and Google Scholar databases. A total of 182 cases of spiradenocarcinoma were identified as eligible for this comprehensive literature review. Results: Spiradenocarcinoma was commoner in older age and Caucasian race. In most cases, surgical excision for local disease is the mainstay of treatment. Lymph node dissection is usually reserved for those with suspected or confirmed lymph node metastases. High rates of local recurrence (20.8%), metastasis (37.4%) and mortality (19.1%) were identified, prompting some authors to suggest regular follow up including chest X-rays and liver function tests. Conclusions: Patients with spiradenocarcinoma may benefit from a magnetic resonance imaging and fluorodeoxyglucose-positron emission tomography/computed tomography to establish the extent of disease. We recommend wide local excision as the treatment of choice to achieve surgical margins of ≥1 cm, with node resection to be determined on a case-to-case basis. Regular follow up is important given the high rate of local recurrence, metastasis and mortality. This should include an examination of the regional lymph nodes. Further research is required to refine an evidence-based approach to spiradenocarcinoma.

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Histological findings and pathologic diagnosis of spiradenocarcinoma: A case series and review of the literature

Cited by 8 publications

References 26 publications

Lymphangiectatic Variant of Low-Grade Malignant Eccrine Spiradenoma

Lymphangiectatic Variant of Low-Grade Malignant Eccrine Spiradenoma

Superficial GLI1‐amplified mesenchymal neoplasms: Expanding the spectrum of an emerging entity which reaches the realm of dermatopathology

Challenges in diagnosis and management of a spiradenocarcinoma: a comprehensive literature review

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