Kwadwo Antwi1, Guillaume Nicolas1,2, Melpomeni Fani1, Tobias Heye1, Francois Pattou3, Ashley Grossman4,5, Philippe Chanson6,7, Jean Claude Reubi8, Aurel Perren8, Beat Gloor9, Deborah R Vogt10, Damian Wild1,2, Emanuel Christ2,11. 1. Clinic of Radiology and Nuclear Medicine, University Hospital Basel, Basel, Switzerland. 2. Center for Neuroendocrine and Endocrine Tumors, University Hospital Basel, Basel, Switzerland. 3. Department of General and Endocrine Surgery, Lille University Hospital, Lille France. 4. Oxford Centre for Diabetes, Endocrinology and Metabolism, University of Oxford, Oxford, United Kingdom. 5. Neuroendocrine Unit, Royal Free Hospital, London, United Kingdom. 6. Assistance Publique-Hôpitaux de Paris, Service d'Endocrinologie et des Maladies de la Reproduction, Hôpital Bicêtre, Le Kremlin-Bicêtre, France. 7. UMR S-1185, Université Paris Sud, Université Paris-Saclay, Le Kremlin-Bicêtre, France. 8. Department of Pathology, University of Bern, Bern, Switzerland. 9. Department of Visceral Surgery, University Hospital of Bern, Inselspital, Bern, Switzerland. 10. Clinical Trial Unit, Department of Clinical Research, University Hospital Basel, University of Basel, Basel, Switzerland. 11. Division of Endocrinology, Diabetology and Metabolism, University Hospital Basel, Basel, Switzerland.
Abstract
CONTEXT: Surgical intervention is advised in patients with multiple endocrine neoplasia type-1 (MEN-1) and nonfunctioning pancreatic neuroendocrine tumors (PanNETs) with a size ≥20 mm. Functioning PanNETs, such as in patients with endogenous hyperinsulinemic hypoglycemia (EHH) due to (one or multiple) insulinomas, should be treated surgically independent of size. Preoperative localization of insulinomas is critical for surgery. OBJECTIVE: To evaluate the feasibility and sensitivity of 68Ga-DOTA-exendin-4 positron emission tomography (PET)/CT in the detection of clinically relevant lesions in patients with MEN-1 and EHH in combination with MRI. DESIGN: Post hoc subgroup analysis of a larger prospective imaging study with 52 patients with EHH. PATIENTS: Six of 52 consecutive patients with EHH and genetically proven MEN-1 mutation were included. INTERVENTIONS: All patients received one 68Ga-DOTA-exendin-4 PET/CT and one MRI scan within 3 to 4 days. Thereafter, surgery was performed based on all imaging results. MAIN OUTCOME MEASURES: Lesion-based sensitivity of PET/CT and MRI for detection of clinically relevant lesions was calculated. Readers were unaware of other results. The reference standard was surgery with histology and treatment outcome. True positive (i.e., clinically relevant lesions) was defined as PanNETs ≥20 mm or insulinoma. RESULTS: In six patients, 37 PanNETs were confirmed by histopathology. Sensitivity (95% CI) in the detection of clinically relevant lesions for combined PET/CT plus MRI, MRI, and PET/CT was 92.3% (64% to 99.8%), 38.5% (13.9% to 68.4%), and 84.6% (54.6% to 98.1%), respectively (P = 0.014 for the comparison of PET/CT plus MRI vs MRI). Postsurgery, EHH resolved in all patients. CONCLUSION: 68Ga-DOTA-exendin-4 PET/CT is feasible in patients with MEN-1 and EHH. The combination with MRI is superior to MRI alone in the detection of insulinomas and may guide the surgical strategy.
CONTEXT: Surgical intervention is advised in patients with multiple endocrine neoplasia type-1 (MEN-1) and nonfunctioning pancreatic neuroendocrine tumors (PanNETs) with a size ≥20 mm. Functioning PanNETs, such as in patients with endogenous hyperinsulinemic hypoglycemia (EHH) due to (one or multiple) insulinomas, should be treated surgically independent of size. Preoperative localization of insulinomas is critical for surgery. OBJECTIVE: To evaluate the feasibility and sensitivity of 68Ga-DOTA-exendin-4 positron emission tomography (PET)/CT in the detection of clinically relevant lesions in patients with MEN-1 and EHH in combination with MRI. DESIGN: Post hoc subgroup analysis of a larger prospective imaging study with 52 patients with EHH. PATIENTS: Six of 52 consecutive patients with EHH and genetically proven MEN-1 mutation were included. INTERVENTIONS: All patients received one 68Ga-DOTA-exendin-4 PET/CT and one MRI scan within 3 to 4 days. Thereafter, surgery was performed based on all imaging results. MAIN OUTCOME MEASURES: Lesion-based sensitivity of PET/CT and MRI for detection of clinically relevant lesions was calculated. Readers were unaware of other results. The reference standard was surgery with histology and treatment outcome. True positive (i.e., clinically relevant lesions) was defined as PanNETs ≥20 mm or insulinoma. RESULTS: In six patients, 37 PanNETs were confirmed by histopathology. Sensitivity (95% CI) in the detection of clinically relevant lesions for combined PET/CT plus MRI, MRI, and PET/CT was 92.3% (64% to 99.8%), 38.5% (13.9% to 68.4%), and 84.6% (54.6% to 98.1%), respectively (P = 0.014 for the comparison of PET/CT plus MRI vs MRI). Postsurgery, EHH resolved in all patients. CONCLUSION: 68Ga-DOTA-exendin-4 PET/CT is feasible in patients with MEN-1 and EHH. The combination with MRI is superior to MRI alone in the detection of insulinomas and may guide the surgical strategy.
Authors: Dirk-Jan van Beek; Sjoerd Nell; Helena M Verkooijen; Inne H M Borel Rinkes; Gerlof D Valk; Menno R Vriens Journal: Surgery Date: 2020-11-19 Impact factor: 4.348
Authors: Maria Luisa Brandi; Sunita K Agarwal; Nancy D Perrier; Kate E Lines; Gerlof D Valk; Rajesh V Thakker Journal: Endocr Rev Date: 2021-03-15 Impact factor: 19.871