A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib Therapy

Introduction: Pituitary metastases (PM) are rare events and to date only very few cases of melanoma PM have been described in literature up to now.Case Presentation: We describe the clinical history of a 33-year-old male patient who underwent surgical excision of an inter-scapular melanoma in 2008....

Full description

Bibliographic Details
Main Authors: Marilda Mormando, Giulia Puliani, Agnese Barnabei, Rosa Lauretta, Marta Bianchini, Alfonsina Chiefari, Michelangelo Russillo, Francesco Cognetti, Luisa Romano, Marialuisa Appetecchia
Format: Article
Language:English
Published: Frontiers Media S.A. 2020-07-01
Series:Frontiers in Endocrinology
Subjects:
Online Access:https://www.frontiersin.org/article/10.3389/fendo.2020.00471/full
_version_ 1819081383996293120
author Marilda Mormando
Giulia Puliani
Giulia Puliani
Agnese Barnabei
Rosa Lauretta
Marta Bianchini
Alfonsina Chiefari
Michelangelo Russillo
Francesco Cognetti
Luisa Romano
Marialuisa Appetecchia
author_facet Marilda Mormando
Giulia Puliani
Giulia Puliani
Agnese Barnabei
Rosa Lauretta
Marta Bianchini
Alfonsina Chiefari
Michelangelo Russillo
Francesco Cognetti
Luisa Romano
Marialuisa Appetecchia
author_sort Marilda Mormando
collection DOAJ
description Introduction: Pituitary metastases (PM) are rare events and to date only very few cases of melanoma PM have been described in literature up to now.Case Presentation: We describe the clinical history of a 33-year-old male patient who underwent surgical excision of an inter-scapular melanoma in 2008. The subsequent follow-up was negative for ~10 years. In September 2018, due to the onset of a severe headache, the patient underwent a brain magnetic resonance imaging, which showed an expansive mass in the saddle and suprasellar region with a maximum diameter of 17 mm. Pituitary function tests and visual field were normal. Worsening of the headache and the appearance of a left eye ptosis led the patient to surgical removal of the lesion in October 2018. The histological examination unexpectedly showed metastasis of the melanoma. Post-operative hormonal assessment showed secondary hypothyroidism and hypoadrenalism, which were both promptly treated, and a mild hypogonadism. Three months after surgery, a sellar MRI showed a persistent, increased pituitary mass (3 cm of diameter); fluorine-18-fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) detected an increased radiopharmaceutical uptake in the sellar region. Due to the persistence of the disease and the evidence of a BRAF V600E mutation, in February 2019, the patient underwent a combined treatment with dabrafenib (a BRAF inhibitor) and trametinib (mitogen-activated extracellular signal-regulate kinase inhibitor). Sellar MRI performed 6 months later showed no evidence of mass in the sellar region. The patient was in a good clinical condition and did not complain of headaches or other symptoms; there were no significant side-effects from the anticancer therapy. After 13 months of treatment, the patient showed no recurrence of the disease on morphological imaging. Anticancer therapy was confirmed, replacement therapies with hydrocortisone and levothyroxine continued and the pituitary-gonadal axis was restored.Conclusion: This is a very interesting case, both for the rarity of the pituitary melanoma metastasis and for the singular therapeutic course carried out by the patient. This is the first case of a pituitary melanoma metastasis with BRAF mutation, successfully treated with the combination of dabrafenib and trametinib after incomplete surgical removal.
first_indexed 2024-12-21T19:59:55Z
format Article
id doaj.art-40fa68f784c74ae8a24b7ee4960e9f21
institution Directory Open Access Journal
issn 1664-2392
language English
last_indexed 2024-12-21T19:59:55Z
publishDate 2020-07-01
publisher Frontiers Media S.A.
record_format Article
series Frontiers in Endocrinology
spelling doaj.art-40fa68f784c74ae8a24b7ee4960e9f212022-12-21T18:51:59ZengFrontiers Media S.A.Frontiers in Endocrinology1664-23922020-07-011110.3389/fendo.2020.00471561967A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib TherapyMarilda Mormando0Giulia Puliani1Giulia Puliani2Agnese Barnabei3Rosa Lauretta4Marta Bianchini5Alfonsina Chiefari6Michelangelo Russillo7Francesco Cognetti8Luisa Romano9Marialuisa Appetecchia10Oncological Endocrinology Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyOncological Endocrinology Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyDepartment of Experimental Medicine, Sapienza University of Rome, Rome, ItalyOncological Endocrinology Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyOncological Endocrinology Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyOncological Endocrinology Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyOncological Endocrinology Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyDivision of Medical Oncology 1, IRCCS Regina Elena National Cancer Institute, Rome, ItalyDivision of Medical Oncology 1, IRCCS Regina Elena National Cancer Institute, Rome, ItalyNuclear Medicine Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyOncological Endocrinology Unit, IRCCS Regina Elena National Cancer Institute, Rome, ItalyIntroduction: Pituitary metastases (PM) are rare events and to date only very few cases of melanoma PM have been described in literature up to now.Case Presentation: We describe the clinical history of a 33-year-old male patient who underwent surgical excision of an inter-scapular melanoma in 2008. The subsequent follow-up was negative for ~10 years. In September 2018, due to the onset of a severe headache, the patient underwent a brain magnetic resonance imaging, which showed an expansive mass in the saddle and suprasellar region with a maximum diameter of 17 mm. Pituitary function tests and visual field were normal. Worsening of the headache and the appearance of a left eye ptosis led the patient to surgical removal of the lesion in October 2018. The histological examination unexpectedly showed metastasis of the melanoma. Post-operative hormonal assessment showed secondary hypothyroidism and hypoadrenalism, which were both promptly treated, and a mild hypogonadism. Three months after surgery, a sellar MRI showed a persistent, increased pituitary mass (3 cm of diameter); fluorine-18-fluorodeoxyglucose positron emission tomography/computed tomography (18F-FDG PET/CT) detected an increased radiopharmaceutical uptake in the sellar region. Due to the persistence of the disease and the evidence of a BRAF V600E mutation, in February 2019, the patient underwent a combined treatment with dabrafenib (a BRAF inhibitor) and trametinib (mitogen-activated extracellular signal-regulate kinase inhibitor). Sellar MRI performed 6 months later showed no evidence of mass in the sellar region. The patient was in a good clinical condition and did not complain of headaches or other symptoms; there were no significant side-effects from the anticancer therapy. After 13 months of treatment, the patient showed no recurrence of the disease on morphological imaging. Anticancer therapy was confirmed, replacement therapies with hydrocortisone and levothyroxine continued and the pituitary-gonadal axis was restored.Conclusion: This is a very interesting case, both for the rarity of the pituitary melanoma metastasis and for the singular therapeutic course carried out by the patient. This is the first case of a pituitary melanoma metastasis with BRAF mutation, successfully treated with the combination of dabrafenib and trametinib after incomplete surgical removal.https://www.frontiersin.org/article/10.3389/fendo.2020.00471/fullmelanomapituitary melanoma metastasispituitarydabrafenibtrametinibtherapy
spellingShingle Marilda Mormando
Giulia Puliani
Giulia Puliani
Agnese Barnabei
Rosa Lauretta
Marta Bianchini
Alfonsina Chiefari
Michelangelo Russillo
Francesco Cognetti
Luisa Romano
Marialuisa Appetecchia
A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib Therapy
Frontiers in Endocrinology
melanoma
pituitary melanoma metastasis
pituitary
dabrafenib
trametinib
therapy
title A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib Therapy
title_full A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib Therapy
title_fullStr A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib Therapy
title_full_unstemmed A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib Therapy
title_short A Rare Case of Pituitary Melanoma Metastasis: A Dramatic and Prolonged Response to Dabrafenib-Trametinib Therapy
title_sort rare case of pituitary melanoma metastasis a dramatic and prolonged response to dabrafenib trametinib therapy
topic melanoma
pituitary melanoma metastasis
pituitary
dabrafenib
trametinib
therapy
url https://www.frontiersin.org/article/10.3389/fendo.2020.00471/full
work_keys_str_mv AT marildamormando ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT giuliapuliani ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT giuliapuliani ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT agnesebarnabei ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT rosalauretta ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT martabianchini ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT alfonsinachiefari ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT michelangelorussillo ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT francescocognetti ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT luisaromano ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT marialuisaappetecchia ararecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT marildamormando rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT giuliapuliani rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT giuliapuliani rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT agnesebarnabei rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT rosalauretta rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT martabianchini rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT alfonsinachiefari rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT michelangelorussillo rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT francescocognetti rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT luisaromano rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy
AT marialuisaappetecchia rarecaseofpituitarymelanomametastasisadramaticandprolongedresponsetodabrafenibtrametinibtherapy