Multidisciplinary approach including receptor radionuclide therapy with 90Y-DOTATOC ([90Y-DOTA0,Tyr3]- octreotide) and 177Lu-DOTATATE ([177Lu-DOTA 0,Tyr3]-octreotate) in ectopic Cushing syndrome from a metastatic gastrinoma: A promising proposal

Maria Vittoria Davì, Lisa Bodei, Marco Ferdeghini, Massimo Falconi, Marco Testoni, Giovanni Paganelli, Cristina Oliani, Vincenzo Lo Cascio, Giuseppe Francia

Research output: Contribution to journalArticle

11 Citations (Scopus)

Abstract

Objective: To present a case of a young woman with Cushing syndrome caused by ectopic production of adrenocorticotropic hormone from a metastatic pancreatic gastrin-secreting endocrine carcinoma, who had a good response to combination peptide receptor radionuclide therapy. Methods: We review the history, physical examination, laboratory investigations, and radiographic findings in this unusual patient. Moreover, the multimodal interventions are described and discussed. Results: In a 38-year-old woman with typical signs of cortisol excess, laboratory studies revealed diabetes mellitus, hypokalemia, and high levels of adrenocorticotropic hormone, plasma cortisol, and urinary cortisol. Abdominal computed tomography showed a 4-cm pancreatic mass and multiple metastatic lesions in the liver, and ectopic Cushing syndrome was diagnosed. Treatment consisted of surgical debulking of the tumor, ketoconazole, somatostatin analogues, chemoembolization of the liver metastatic lesions, and peptide receptor radionuclide therapy with the radiolabeled somatostatin analogues 90Y-DOTATOC ([90Y-DOTA0,Tyr 3]-octreotide) and 177Lu-DOTATATE ([177Lu- DOTA0,Tyr3]-octreotate). The 5 1/2-year follow-up showed positive results, which included complete regression of all clinical and hormonal evidence of the tumor and substantial decrease in the size and number of hepatic metastatic lesions. The patient achieved and still maintains an optimal quality of life. Conclusion: To the best of our knowledge, this is the first report of a multidisciplinary approach including peptide receptor radionuclide therapy with 90Y-DOTATOC and 177Lu-DOTATATE, which proved to be effective in improving clinical outcome in a case of metastatic endocrine carcinoma of the pancreas in conjunction with ectopic Cushing syndrome. In this unusual case, the patient has one of the longest durations of survival in this setting described in the literature.

Original languageEnglish
Pages (from-to)213-218
Number of pages6
JournalEndocrine Practice
Volume14
Issue number2
Publication statusPublished - Mar 2008

Fingerprint

Gastrinoma
Octreotide
Cushing Syndrome
Radioisotopes
Peptide Receptors
Hydrocortisone
Somatostatin
Adrenocorticotropic Hormone
Liver
Carcinoma
Ketoconazole
Hypokalemia
Gastrins
Therapeutics
Islets of Langerhans
Physical Examination
Neoplasms
Diabetes Mellitus
History
Tomography

ASJC Scopus subject areas

  • Endocrinology
  • Endocrinology, Diabetes and Metabolism
  • Medicine(all)

Cite this

Multidisciplinary approach including receptor radionuclide therapy with 90Y-DOTATOC ([90Y-DOTA0,Tyr3]- octreotide) and 177Lu-DOTATATE ([177Lu-DOTA 0,Tyr3]-octreotate) in ectopic Cushing syndrome from a metastatic gastrinoma : A promising proposal. / Davì, Maria Vittoria; Bodei, Lisa; Ferdeghini, Marco; Falconi, Massimo; Testoni, Marco; Paganelli, Giovanni; Oliani, Cristina; Lo Cascio, Vincenzo; Francia, Giuseppe.

In: Endocrine Practice, Vol. 14, No. 2, 03.2008, p. 213-218.

Research output: Contribution to journalArticle

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abstract = "Objective: To present a case of a young woman with Cushing syndrome caused by ectopic production of adrenocorticotropic hormone from a metastatic pancreatic gastrin-secreting endocrine carcinoma, who had a good response to combination peptide receptor radionuclide therapy. Methods: We review the history, physical examination, laboratory investigations, and radiographic findings in this unusual patient. Moreover, the multimodal interventions are described and discussed. Results: In a 38-year-old woman with typical signs of cortisol excess, laboratory studies revealed diabetes mellitus, hypokalemia, and high levels of adrenocorticotropic hormone, plasma cortisol, and urinary cortisol. Abdominal computed tomography showed a 4-cm pancreatic mass and multiple metastatic lesions in the liver, and ectopic Cushing syndrome was diagnosed. Treatment consisted of surgical debulking of the tumor, ketoconazole, somatostatin analogues, chemoembolization of the liver metastatic lesions, and peptide receptor radionuclide therapy with the radiolabeled somatostatin analogues 90Y-DOTATOC ([90Y-DOTA0,Tyr 3]-octreotide) and 177Lu-DOTATATE ([177Lu- DOTA0,Tyr3]-octreotate). The 5 1/2-year follow-up showed positive results, which included complete regression of all clinical and hormonal evidence of the tumor and substantial decrease in the size and number of hepatic metastatic lesions. The patient achieved and still maintains an optimal quality of life. Conclusion: To the best of our knowledge, this is the first report of a multidisciplinary approach including peptide receptor radionuclide therapy with 90Y-DOTATOC and 177Lu-DOTATATE, which proved to be effective in improving clinical outcome in a case of metastatic endocrine carcinoma of the pancreas in conjunction with ectopic Cushing syndrome. In this unusual case, the patient has one of the longest durations of survival in this setting described in the literature.",
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