Metastatic Spiradenocarcinoma Managed With PD-1 Inhibition

Wargo, Jeffrey J.; Carr, David; Plaza, Jose A.; Verschraegen, Claire F.

doi:10.6004/jnccn.2021.7119

Cited by 9 publications

(10 citation statements)

References 6 publications

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“…To our knowledge, this is the first paper to report on the use of immunotherapy for the management of metastatic DEA. On review of the published literature, 5 case reports in the last decade have presented favourable outcomes for immunotherapy used in other subtypes of eccrine carcinoma: two for eccrine porocarcinoma [14, 15], two for eccrine spiradenocarcinoma [7, 16], and one for eccrine carcinoma not otherwise specified [13] (Table 1). Of these, the report by Singh et al shared many similarities with our own case: the authors described an elderly male with eccrine porocarcinoma of the anterior scalp, initially managed with surgical excision and adjuvant radiotherapy, later complicated by multiple distant metastases.…”

Section: Discussionmentioning

confidence: 99%

“…The role of immunotherapy for metastatic non-melanoma skin cancer (NMSC) is an area of active research. Only a handful of case reports of immunotherapy in the setting of eccrine carcinomas are available, the majority for metastatic eccrine porocarcinoma [7,8,[13][14][15][16]. The use of immunotherapy for metastatic DEA has not previously been published.…”

Section: Introductionmentioning

confidence: 99%

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Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy

Fadhil,

Lochhead,

Trinh

et al. 2023

Case Rep Oncol

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Eccrine carcinoma, a subtype of which is ductal eccrine adenocarcinoma (DEA), is a rare cutaneous malignancy. For metastatic eccrine carcinoma, there are very limited data to guide treatment. Conventional chemotherapy is of limited benefit and there is only a small body of evidence for the use of immunotherapy in non-DEA eccrine carcinomas. We report the first case of metastatic DEA treated with a multimodality approach including surgery, radiotherapy, and immunotherapy, with an excellent prolonged response to pembrolizumab, and provide a review of the literature on pathological and management aspects for this rare tumour subtype. A 60-year-old male with a history of pT1N0M0 left scalp DEA, managed 2 years prior with excision and adjuvant radiotherapy, represented with a symptomatic right pontine metastasis. Imaging demonstrated intracranial, pulmonary, and hilar disease; biopsy of the cranial and lung lesions showed metastatic adenocarcinoma, morphologically similar to the previously resected scalp DEA. The patient was treated with stereotactic resections of his pontine metastases and adjuvant cranial radiotherapy, then commenced on immunotherapy with pembrolizumab. The patient has completed 21 months of pembrolizumab with a significant radiological response of the pulmonary and hilar disease and nil evidence of intracranial recurrence or further metastases. In this case report, we provide the first evidence of efficacy of immunotherapy in metastatic DEA, demonstrating an excellent and prolonged response of metastatic DEA to pembrolizumab. Further research is required to better establish the role of immunotherapy within the management protocol for this uncommon but aggressive tumour subtype.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy

Fadhil,

Lochhead,

Trinh

et al. 2023

Case Rep Oncol

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“…Malignant neoplasms arising from spiradenoma, cylindroma, and spiradenocylindroma are rare malignant sweat gland tumors, the majority of which arise in association with a benign spiradenoma precursor (spiradenocarcinoma). [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15] Their histopathologic separation into morphologically low-and high-grade spiradenocarcinomas appears to correlate well with clinical behavior and outcome. 2,3,6,11 While the morphologically high-grade spiradenocarcinomas may be associated with aggressive behavior, including distant metastases and mortality, the morphologically low-grade tumors appear to be indolent, mainly showing increased risk for local recurrence only.…”

Section: Introductionmentioning

confidence: 99%

“…Malignant neoplasms arising from spiradenoma, cylindroma, and spiradenocylindroma are rare malignant sweat gland tumors, the majority of which arise in association with a benign spiradenoma precursor (spiradenocarcinoma) 1–15 . Their histopathologic separation into morphologically low‐ and high‐grade spiradenocarcinomas appears to correlate well with clinical behavior and outcome 2,3,6,11 .…”

Section: Introductionmentioning

confidence: 99%

Distant metastasis from morphologically low‐grade spiradenocarcinoma: A report of two cases

et al. 2023

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Malignant tumors arising from benign eccrine spiradenomas are rare. They are divided by morphology into low-grade and high-grade spiradenocarcinomas, with prognosis and metastatic potential closely linked to their histopathologic features.Tumors with low-grade morphology are known for their indolent behavior, with only two reported instances of metastatic spread. We report herein two further low-grade metastatic spiradenocarcinomas resulting in distant metastasis. Both tumors showed a background of a benign spiradenoma and subtle histopathologic signs of malignant transformation, characterized by loss of the dual-cell population, up to moderate cytological atypia and increased mitotic activity. Both patients developed metastases to the lungs years after the initial presentation, and one showed additional lymph nodal disease. We show that even the morphologically low-grade tumors may rarely show more aggressive behavior. Although often challenging, recognition of the morphologically low-grade malignant spiradenocarcinoma and long-term follow-up of the patients are important to detect metastatic disease.

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“…The constraint scores on average are statistically higher than random samples, indicating that these sequence variations are likely to contribute to CC. Six genes ( PXDN , RTEL1 , ANKRD11 , MAP2K1 , CYLD , and ACAN ) with the de novo variants are involved in human developmental diseases, such as sclerocornea, spondyloepimetaphyseal dysplasia, dyskeratosis congenita, and familial multiple trichoepitheliomata [ 57 , 58 , 59 , 60 , 61 , 62 , 63 , 64 , 65 ]. Furthermore, four genes ( PIK3CA , TLN1 CYLD , and MAP2K1 ) with the damaging variants are linked to bile duct and liver cancer.…”

Section: Introductionmentioning

confidence: 99%

Pathogenesis of Choledochal Cyst: Insights from Genomics and Transcriptomics

Lui

Tam

2022

Genes

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Choledochal cysts (CC) is characterized by extra- and/or intra-hepatic b\ile duct dilations. There are two main theories, “pancreaticobiliary maljunction” and “congenital stenosis of bile ducts” proposed for the pathogenesis of CC. Although family cases or CC associated with other anomalies have been reported, the molecular pathogenesis of CC is still poorly understood. Recent advances in transcriptomics and genomics analysis platforms have unveiled key expression signatures/genes/signaling pathways in the pathogenesis of human diseases including CC. This review summarizes insights from genomics and transcriptomics studies into the pathogenesis of CC, with the aim to improve (i) our understanding of its underlying complex pathomechanisms, and (ii) clinical management of different subtypes of CC, in particular their associated hepatic fibrotic change and their risk of malignancy transformation.

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Metastatic Spiradenocarcinoma Managed With PD-1 Inhibition

Cited by 9 publications

References 6 publications

Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy

Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy

Distant metastasis from morphologically low‐grade spiradenocarcinoma: A report of two cases

Pathogenesis of Choledochal Cyst: Insights from Genomics and Transcriptomics

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