Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy

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 im...

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Main Authors: Matthew Fadhil, Alistair Lochhead, Hon Trinh, Daniel Brungs
Format: Article
Language:English
Published: Karger Publishers 2023-11-01
Series:Case Reports in Oncology
Subjects:
Online Access:https://beta.karger.com/Article/FullText/533956
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author Matthew Fadhil
Alistair Lochhead
Hon Trinh
Daniel Brungs
author_facet Matthew Fadhil
Alistair Lochhead
Hon Trinh
Daniel Brungs
author_sort Matthew Fadhil
collection DOAJ
description 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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spelling doaj.art-62267dd1c678464bb470e0dd462a15e02023-12-21T10:09:26ZengKarger PublishersCase Reports in Oncology1662-65752023-11-011611415142410.1159/000533956533956Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to ImmunotherapyMatthew Fadhil0Alistair Lochhead1Hon Trinh2Daniel Brungs3Department of Medical Oncology, Wollongong Hospital, Wollongong, NSW, AustraliaSouthern IML Pathology, Wollongong, NSW, AustraliaDepartment of Radiation Oncology, Shoalhaven Cancer Care Centre, Nowra, NSW, AustraliaDepartment of Medical Oncology, Wollongong Hospital, Wollongong, NSW, AustraliaEccrine 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.https://beta.karger.com/Article/FullText/533956eccrine carcinomaductal eccrine adenocarcinomametastasisimmunotherapypembrolizumab
spellingShingle Matthew Fadhil
Alistair Lochhead
Hon Trinh
Daniel Brungs
Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy
Case Reports in Oncology
eccrine carcinoma
ductal eccrine adenocarcinoma
metastasis
immunotherapy
pembrolizumab
title Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy
title_full Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy
title_fullStr Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy
title_full_unstemmed Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy
title_short Metastatic Ductal Eccrine Adenocarcinoma with Excellent Response to Immunotherapy
title_sort metastatic ductal eccrine adenocarcinoma with excellent response to immunotherapy
topic eccrine carcinoma
ductal eccrine adenocarcinoma
metastasis
immunotherapy
pembrolizumab
url https://beta.karger.com/Article/FullText/533956
work_keys_str_mv AT matthewfadhil metastaticductaleccrineadenocarcinomawithexcellentresponsetoimmunotherapy
AT alistairlochhead metastaticductaleccrineadenocarcinomawithexcellentresponsetoimmunotherapy
AT hontrinh metastaticductaleccrineadenocarcinomawithexcellentresponsetoimmunotherapy
AT danielbrungs metastaticductaleccrineadenocarcinomawithexcellentresponsetoimmunotherapy