Off-label pasireotide treatment in one insulinoma patient with an atypical presentation and intolerant to diazoxide
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RESEARCH LETTER
Off-label pasireotide treatment in one insulinoma patient with an atypical presentation and intolerant to diazoxide Federica Sileo1,2 Biagio Cangiano1,2 Chiara Cacciatore2 Jessica Amarù3 Federico Gatto4 Manuela Albertelli3 Massimo Falconi5 Giovanni Vitale1,6 Luca Persani 1,2 ●
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Received: 29 April 2020 / Accepted: 24 June 2020 © Springer Science+Business Media, LLC, part of Springer Nature 2020
Neuroendocrine Neoplasms (NENs) are rare, but their incidence is constantly increasing. Insulinoma is a functioning NEN, with an incidence of 1–4 cases/million/year [1]. Despite its benignity in 90% of cases [2], it is a dangerous condition due to hypoglycemic events and the intrinsic risks can be further extended in case of atypical presentation. Surgery is the treatment of choice for insulinoma, but biochemical diagnosis and localization of the lesion can be challenging, and patients may remain exposed to hypoglycemia risks for weeks. In the meantime, a medical control of the disease is required [2]. We present the off-label use of pasireotide for presurgical management of an atypical insulinoma. A 42-year-old man had recurrent episodes of persistent diplopia for seven years. During one of these episodes, he was admitted to an emergency room revealing a profound hypoglycemia (27 mg/dL), with complete resolution of the symptoms after glucose administration. His past medical history was unremarkable, except for autoimmune hypothyroidism and removal of colon polyp. He was overweight
* Luca Persani [email protected] 1
Department of Clinical Sciences and Community Health, University of Milan, Milan, Italy
2
Lab of Endocrine and Metabolic Research, Department of Endocrine and Metabolic Diseases, IRCCS Istituto Auxologico Italiano, Milan, Italy
3
Endocrinology Unit, Department of Internal Medicine and Medical Specialties, University of Genoa, Genoa, Italy
4
Endocrinology Unit, Department of Specialist Medicine, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
5
Division of Pancreatic Surgery, Pancreas Translational & Clinical Research Center, Università Vita Salute, IRCCS San Raffaele Scientific Institute, Milan, Italy
6
Istituto Auxologico Italiano, IRCCS, Laboratorio Sperimentale di Ricerche di Neuroendocrinologia Geriatrica ed Oncologica, Cusano Milanino, Milan, Italy
(27 kg/m2), had negative anti-insulin and anti-GAD antibodies, but underwent an oral glucose tolerance test (OGTT) (glycemia: baseline 77; +120 min: 238 mg/dL) leading to the diagnosis of diabetes mellitus type 2 (DM2) and reactive post-prandial hypoglycemia. He was put on a 1600 kcal diet with morning and afternoon snacks and acarbose 50 mg at principal meals. The patient lost 5 kg (BMI: 24.1 kg/m2) but continued to register several fasting and postprandial hypoglycemic events always accompanied by diplopia. Thereafter, the patient came to our attention. Adrenal and pituitary deficiencies were excluded, and he was monitored for 5 hours after a new OGTT: this test resulted
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