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Stéphane Bardet
Stéphane Bardet
Department of Nuclear Medicine and Thyroid Unit, Centre François Baclesse, Caen, France
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Renaud Ciappuccini
Renaud Ciappuccini
Department of Nuclear Medicine and Thyroid Unit, Centre François Baclesse, Caen, France
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Livia Lamartina
Livia Lamartina
Department of Nuclear Medicine and Endocrine Oncology, Gustave Roussy, Villejuif, France
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Sophie Leboulleux
Sophie Leboulleux
Department of Nuclear Medicine and Endocrine Oncology, Gustave Roussy, Villejuif, France
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selpercatinib. Introduction Serum tumour markers are important tools in the management of patients with medullary thyroid carcinoma (MTC) ( 1 ). Serum calcitonin (CT) and carcinoembryonic antigen (CEA) are produced by neoplastic C-cells, and their
Luciana Puleo
Luciana Puleo
Endocrine Unit, Department of Clinical and Experimental Medicine
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Laura Agate
Laura Agate
Endocrine Unit, Department of Clinical and Experimental Medicine
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Irene Bargellini
Irene Bargellini
Department of Vascular and Interventional Radiology
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Giuseppe Boni
Giuseppe Boni
Regional Center of Nuclear Medicine
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Paolo Piaggi
Paolo Piaggi
Endocrine Unit, Department of Clinical and Experimental Medicine
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Claudio Traino
Claudio Traino
Regional Center of Nuclear Medicine
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Tommaso Depalo
Tommaso Depalo
Regional Center of Nuclear Medicine
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Giulia Lorenzoni
Giulia Lorenzoni
Department of Vascular and Interventional Radiology
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Francesca Bianchi
Francesca Bianchi
Regional Center of Nuclear Medicine
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Duccio Volterrani
Duccio Volterrani
Regional Center of Nuclear Medicine
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Sandra Brogioni
Sandra Brogioni
Endocrine Unit, Department of Clinical and Experimental Medicine
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Valeria Bottici
Valeria Bottici
Endocrine Unit, Department of Clinical and Experimental Medicine
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Maurizia Rossana Brunetto
Maurizia Rossana Brunetto
Hepatology Unit, University of Pisa, Pisa, Italy
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Barbara Coco
Barbara Coco
Hepatology Unit, University of Pisa, Pisa, Italy
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Eleonora Molinaro
Eleonora Molinaro
Endocrine Unit, Department of Clinical and Experimental Medicine
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Rossella Elisei
Rossella Elisei
Endocrine Unit, Department of Clinical and Experimental Medicine
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Association (ATA) guidelines, after surgery, serum calcitonin (Ct) and carcino-embryonic antigen (CEA) must be periodically tested in order to assess disease status, particularly when serum Ct is >150 pg/mL ( 2 ). MTC metastasizes most commonly to the liver
Marine Sitbon
Marine Sitbon
Pharmacy Department, Hospital Saint-Louis APHP, Paris, France
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Porhuoy Chou
Porhuoy Chou
Endocrine Oncology Department, Saint-Louis Hospital (AP-HP), Université Paris Cité, Paris, France
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Seydou Bengaly
Seydou Bengaly
Endocrine Oncology Department, Saint-Louis Hospital (AP-HP), Université Paris Cité, Paris, France
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Brigitte Poirot
Brigitte Poirot
Department of Molecular Oncology, Saint-Louis Hospital (AP-HP), Université Paris Cité INSERM U 944, CNRS UMR 7212, Paris, France
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Marie Laloi-Michelin
Marie Laloi-Michelin
Department of Internal Medicine, Hospital Lariboisière APHP, Paris, France
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Laure Deville
Laure Deville
Pharmacy Department, Hospital Saint-Louis APHP, Paris, France
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Atanas Pachev
Atanas Pachev
Radiology Department, Saint-Louis Hospital (AP-HP), Université Paris Cité, Paris, France
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Ahouefa Kowo-Bille
Ahouefa Kowo-Bille
Endocrine Oncology Department, Saint-Louis Hospital (AP-HP), Université Paris Cité, Paris, France
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Clement Dumont
Clement Dumont
Medical Oncology Department, Saint-Louis Hospital (AP-HP), Université Paris Cité, Paris, France
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Cécile N Chougnet
Cécile N Chougnet
Endocrine Oncology Department, Saint-Louis Hospital (AP-HP), Université Paris Cité, Paris, France
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The endocrine secretions of carcinomas can be life-threatening. Medullary thyroid carcinoma (MTC) is a rare cancer that is often associated with cortisol secretion, leading to paraneoplastic Cushing’s syndrome. Mutations of the proto-oncogene RET are driver molecular events in 70% of MTC cases. Here, we report a case of a woman, born in 1956, who was diagnosed with sporadic MTC in 2005, with subsequent relapses treated with focal treatments. In April 2019, she presented with severe and rapidly progressive paraneoplastic Cushing’s syndrome associated with lymph node, lung, liver and bone metastases. A supraclavicular lymph node biopsy revealed a somatic p.M918T (c.2753T>C) mutation in exon 16 of the RET proto-oncogene. The patient began treatment with selpercatinib in September 2019. Clinical efficacy was immediate. Chronic diarrhea disappeared within a few days. Clinical hypercorticism quickly disappeared, with quick improvements in muscle and skin conditions and fatigue. Two months after treatment initiation, urinary free cortisol normalized to 42 µg/24 h. Levels of the tumor markers carcinoembryonic antigen (CEA) and calcitonin also greatly decreased from baseline. After 34 months of treatment, selpercatinib elicits sustained clinical, biological and morphological responses. In summary, this case report illustrates the rapid and long-lasting antisecretory effect of selpercatinib associated with tumor control. As Cushing’s syndrome associated with medullary thyroid cancer is associated with poor prognosis, this case report is very encouraging. In addition, this suggests the potential benefit of molecular testing in all cases of medullary thyroid cancer.
Simona Censi
Simona Censi
Endocrinology Unit, Department of Medicine (DIMED), University of Padua, Padua, Italy
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Jacopo Manso
Jacopo Manso
Endocrinology Unit, Department of Medicine (DIMED), University of Padua, Padua, Italy
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Teresa Benvenuti
Teresa Benvenuti
Endocrinology Unit, Department of Medicine (DIMED), University of Padua, Padua, Italy
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Ilaria Piva
Ilaria Piva
Endocrinology Unit, Department of Medicine (DIMED), University of Padua, Padua, Italy
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Maurizio Iacobone
Maurizio Iacobone
Endocrine Surgery Unit, Department of Surgical, Oncological and Gastroenterological Sciences (DiSCOG), University of Padua, Padua, Italy
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Alberto Mondin
Alberto Mondin
Endocrinology Unit, Department of Medicine (DIMED), University of Padua, Padua, Italy
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Francesca Torresan
Francesca Torresan
Endocrine Surgery Unit, Department of Surgical, Oncological and Gastroenterological Sciences (DiSCOG), University of Padua, Padua, Italy
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Daniela Basso
Daniela Basso
Laboratory Medicine, Department of Medicine (DIMED), University of Padua, Padua, Italy
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Gino Crivellari
Gino Crivellari
Hereditary Tumor Unit, Istituto Oncologico Veneto, IOV - Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Padova, Italy
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Stefania Zovato
Stefania Zovato
Hereditary Tumor Unit, Istituto Oncologico Veneto, IOV - Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Padova, Italy
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Caterina Mian
Caterina Mian
Endocrinology Unit, Department of Medicine (DIMED), University of Padua, Padua, Italy
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necessary if there is lateral lymph node compartment involvement ( 2 ). MTC follow-up is then based on a periodical neck ultrasound (US) and biochemical follow-up, based primarily on calcitonin (Ct) and carcinoembryonic antigen (CEA) measurement. Ct is
Francesca Orsolini
Francesca Orsolini
Department of Clinical and Experimental Medicine, Endocrine Unit, University of Pisa, Pisa, Italy
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Alessandro Prete
Alessandro Prete
Department of Clinical and Experimental Medicine, Endocrine Unit, University of Pisa, Pisa, Italy
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Pierpaolo Falcetta
Pierpaolo Falcetta
Department of Clinical and Experimental Medicine, Endocrine Unit, University of Pisa, Pisa, Italy
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Domenico Canale
Domenico Canale
Department of Clinical and Experimental Medicine, Endocrine Unit, University of Pisa, Pisa, Italy
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Fulvio Basolo
Fulvio Basolo
Department of Surgical, Medical and Molecular Pathology and Critical Care Medicine, University of Pisa, Pisa, Italy
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Greta Alì
Greta Alì
Department of Surgical, Medical and Molecular Pathology and Critical Care Medicine, University of Pisa, Pisa, Italy
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Francesca Manassero
Francesca Manassero
Division of Urology, Department of Translational Research and of New Surgical and Medical Technologies, University of Pisa, Pisa, Italy
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Paolo Vitti
Paolo Vitti
Department of Clinical and Experimental Medicine, Endocrine Unit, University of Pisa, Pisa, Italy
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Rossella Elisei
Rossella Elisei
Department of Clinical and Experimental Medicine, Endocrine Unit, University of Pisa, Pisa, Italy
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Eleonora Molinaro
Eleonora Molinaro
Department of Clinical and Experimental Medicine, Endocrine Unit, University of Pisa, Pisa, Italy
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. ( 16 ). Serum tumoral markers of metastatic MTC are both calcitonin (CTN) and carcinoembryonic antigen (CEA). Their levels correlate with the tumor burden, and their doubling time is associated with the outcome of the disease ( 17 ). In this article
Carla Gambale
Carla Gambale
Department of Clinical and Experimental Medicine, Unit of Endocrinology, Pisa University Hospital, Pisa, Italy
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Alessandro Prete
Alessandro Prete
Department of Clinical and Experimental Medicine, Unit of Endocrinology, Pisa University Hospital, Pisa, Italy
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Chiara Romei
Chiara Romei
Department of Diagnostic Imaging, Unit of Radiology, Pisa University Hospital, Pisa, Italy
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Alessandro Celi
Alessandro Celi
Department of Surgery, Medicine, Molecular Biology and Critical Care, Respiratory Pathophysiology Unit, Pisa University Hospital, Pisa, Italy
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Rossella Elisei
Rossella Elisei
Department of Clinical and Experimental Medicine, Unit of Endocrinology, Pisa University Hospital, Pisa, Italy
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Antonio Matrone
Antonio Matrone
Department of Clinical and Experimental Medicine, Unit of Endocrinology, Pisa University Hospital, Pisa, Italy
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/inflammatory disease were described in this CT scan. CTN and carcinoembryonic antigen (CEA) values were 4280 ng/L and 40 µg/L, respectively. The patient started systemic treatment with selpercatinib in April 2021 at the initial dose of 160 mg/BID. CTN and CEA values
Grace Segall
Grace Segall
Eli Lilly and Company, Indianapolis, IN, USA
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Ravinder Singh
Ravinder Singh
Eli Lilly and Company, Indianapolis, IN, USA
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Min-Hua Jen
Min-Hua Jen
Eli Lilly and Company, Indianapolis, IN, USA
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Isaac Sanderson
Isaac Sanderson
Adelphi Real World, Bollington, UK
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Alex Rider
Alex Rider
Adelphi Real World, Bollington, UK
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Katie Lewis
Katie Lewis
Adelphi Real World, Bollington, UK
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Urpo Kiiskinen
Urpo Kiiskinen
Eli Lilly and Company, Indianapolis, IN, USA
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MTC 31 22 Yes 21 (68) 18 (82) No 2 (6) 1 (5) a In patients with known date at initial diagnosis. BMI, body mass index; CEA, carcinoembryonic antigen; ECOG PS, European Cooperative Oncology Group performance
Junyu Tong
Junyu Tong
Department of Nuclear Medicine, Shanghai Tenth People’s Hospital, Tongji University School of Medicine, Shanghai, People’s Republic of China
Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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Maomei Ruan
Maomei Ruan
Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
Department of Nuclear Medicine, Shanghai Chest Hospital, Shanghai Jiao Tong University, Shanghai, People’s Republic of China
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Department of Nuclear Medicine, Shanghai Chest Hospital, Shanghai Jiao Tong University, Shanghai, People’s Republic of China
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Yuchen Jin
Yuchen Jin
Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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Hao Fu
Hao Fu
Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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Lin Cheng
Lin Cheng
Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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Qiong Luo
Qiong Luo
Department of Nuclear Medicine, Shanghai Tenth People’s Hospital, Tongji University School of Medicine, Shanghai, People’s Republic of China
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Zhiyan Liu
Zhiyan Liu
Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
Department of Pathology, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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Department of Pathology, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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Zhongwei Lv
Zhongwei Lv
Department of Nuclear Medicine, Shanghai Tenth People’s Hospital, Tongji University School of Medicine, Shanghai, People’s Republic of China
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Libo Chen
Libo Chen
Department of Nuclear Medicine, Shanghai Jiao Tong University Affiliated Sixth People’s Hospital, Shanghai, People’s Republic of China
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, helps distinguishing PDTC from DTC ( 38 ). In addition, the negative expression of calcitonin, chromogranin, and carcinoembryonic antigen in PDTC can exclude neuroendocrine tumors, that is, medullary thyroid carcinoma ( 39 ). Likewise, PDTC is not
Takaaki Konishi
Takaaki Konishi
Department of Breast and Endocrine Surgery, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
Department of Clinical Epidemiology and Health Economics, School of Public Health, The University of Tokyo, Tokyo, Japan
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Department of Clinical Epidemiology and Health Economics, School of Public Health, The University of Tokyo, Tokyo, Japan
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Michimasa Fujiogi
Michimasa Fujiogi
Department of Clinical Epidemiology and Health Economics, School of Public Health, The University of Tokyo, Tokyo, Japan
Department of Emergency Medicine, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts, USA
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Department of Emergency Medicine, Massachusetts General Hospital, Harvard Medical School, Boston, Massachusetts, USA
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Nobuaki Michihata
Nobuaki Michihata
Department of Health Services Research, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
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Takayoshi Niwa
Takayoshi Niwa
Department of Breast and Endocrine Surgery, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
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Kojiro Morita
Kojiro Morita
Department of Clinical Epidemiology and Health Economics, School of Public Health, The University of Tokyo, Tokyo, Japan
Department of Health Services, Faculty of Medicine, University of Tsukuba, Tsukuba, Ibaraki, Japan
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Department of Health Services, Faculty of Medicine, University of Tsukuba, Tsukuba, Ibaraki, Japan
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Hiroki Matsui
Hiroki Matsui
Department of Clinical Epidemiology and Health Economics, School of Public Health, The University of Tokyo, Tokyo, Japan
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Kiyohide Fushimi
Kiyohide Fushimi
Department of Health Policy and Informatics, Tokyo Medical and Dental University Graduate School, Tokyo, Japan
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Masahiko Tanabe
Masahiko Tanabe
Department of Breast and Endocrine Surgery, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
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Yasuyuki Seto
Yasuyuki Seto
Department of Breast and Endocrine Surgery, Graduate School of Medicine, The University of Tokyo, Tokyo, Japan
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Hideo Yasunaga
Hideo Yasunaga
Department of Clinical Epidemiology and Health Economics, School of Public Health, The University of Tokyo, Tokyo, Japan
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as serum concentrations of thyroglobulin, calcitonin and carcinoembryonic antigen, are not listed in the database and therefore could not be evaluated. Secondly, the database does not provide detailed surgical information, such as operative time and
