TY - JOUR
T1 - Screening in asymptomatic SDHx mutation carriers
T2 - added value of 18F-FDG PET/CT at initial diagnosis and 1-year follow-up
AU - Lepoutre-Lussey, C.
AU - Caramella, C.
AU - Bidault, F.
AU - Déandreis, D.
AU - Berdelou, A.
AU - Al Ghuzlan, A.
AU - Hartl, D.
AU - Borget, I.
AU - Gimenez-Roqueplo, A. P.
AU - Dumont, F.
AU - Deschamps, F.
AU - Nascimento, C.
AU - Lumbroso, J.
AU - Guillaud Bataille, M.
AU - Schlumberger, M.
AU - Baudin, E.
AU - Leboulleux, S.
N1 - Publisher Copyright:
© 2015, Springer-Verlag Berlin Heidelberg.
PY - 2015/4/2
Y1 - 2015/4/2
N2 - Purpose: Specific recommendations on screening modalities for paraganglioma (PGL) and phaeochromocytoma (PCC) in asymptomatic SDHx mutation carriers (relatives) are still lacking. We evaluated the added value of 18F-FDG PET/CT in comparison with morphological imaging at initial diagnosis and 1 year of follow-up in this population. Methods: The study included 30 consecutive relatives with a proven SDHx mutation who were investigated by 18F-FDG PET/CT, gadolinium-enhanced magnetic resonance angiography of the head and neck, thoracic/abdominal/pelvic (TAP) contrast-enhanced CT and/or TAP MRI. 123I-MIBG scintigraphy was performed in 20 subjects and somatostatin receptor scintigraphy (SRS) in 20 subjects. The gold standard was based on pathology or a composite endpoint as defined by any other positive imaging method and persistent tumour on follow-up. Images were considered as false-positive when the lesions were not detected by another imaging method or not confirmed at 1 year. Results: At initial work-up, an imaging abnormality was found in eight subjects (27 %). The final diagnosis was true-positive in five subjects (two with abdominal PGL, one with PCC and two with neck PGL) and false-positives in the other three subjects (detected with 18F-FDG PET/CT in two and TAP MRI in one). At 1 year, an imaging abnormality was found in three subjects of which one was an 8-mm carotid body PGL in a patient with SDHD mutaion and two were considered false-positive. The tumour detection rate was 100 % for 18F-FDG PET/CT and conventional imaging, 80 % for SRS and 60 % for 123I-MIBG scintigraphy. Overall, disease was detected in 4 % of the subjects at the 1-year follow-up. Conclusion: 18F-FDG PET/CT demonstrated excellent sensitivity but intermediate specificity justifying combined modality imaging in these patients. Given the slow progression of the disease, if 18F-FDG PET/CT and MRI are normal at baseline, the second imaging work-up should be delayed and an examination that does not expose the patient to radiation should be used.
AB - Purpose: Specific recommendations on screening modalities for paraganglioma (PGL) and phaeochromocytoma (PCC) in asymptomatic SDHx mutation carriers (relatives) are still lacking. We evaluated the added value of 18F-FDG PET/CT in comparison with morphological imaging at initial diagnosis and 1 year of follow-up in this population. Methods: The study included 30 consecutive relatives with a proven SDHx mutation who were investigated by 18F-FDG PET/CT, gadolinium-enhanced magnetic resonance angiography of the head and neck, thoracic/abdominal/pelvic (TAP) contrast-enhanced CT and/or TAP MRI. 123I-MIBG scintigraphy was performed in 20 subjects and somatostatin receptor scintigraphy (SRS) in 20 subjects. The gold standard was based on pathology or a composite endpoint as defined by any other positive imaging method and persistent tumour on follow-up. Images were considered as false-positive when the lesions were not detected by another imaging method or not confirmed at 1 year. Results: At initial work-up, an imaging abnormality was found in eight subjects (27 %). The final diagnosis was true-positive in five subjects (two with abdominal PGL, one with PCC and two with neck PGL) and false-positives in the other three subjects (detected with 18F-FDG PET/CT in two and TAP MRI in one). At 1 year, an imaging abnormality was found in three subjects of which one was an 8-mm carotid body PGL in a patient with SDHD mutaion and two were considered false-positive. The tumour detection rate was 100 % for 18F-FDG PET/CT and conventional imaging, 80 % for SRS and 60 % for 123I-MIBG scintigraphy. Overall, disease was detected in 4 % of the subjects at the 1-year follow-up. Conclusion: 18F-FDG PET/CT demonstrated excellent sensitivity but intermediate specificity justifying combined modality imaging in these patients. Given the slow progression of the disease, if 18F-FDG PET/CT and MRI are normal at baseline, the second imaging work-up should be delayed and an examination that does not expose the patient to radiation should be used.
KW - F-FDG PET/CT
KW - Paraganglioma
KW - Phaeochromocytoma
KW - Screening
KW - Succinate dehydrogenase
UR - http://www.scopus.com/inward/record.url?scp=84943699126&partnerID=8YFLogxK
U2 - 10.1007/s00259-015-3003-z
DO - 10.1007/s00259-015-3003-z
M3 - Article
C2 - 25676472
AN - SCOPUS:84943699126
SN - 1619-7070
VL - 42
SP - 868
EP - 876
JO - European Journal of Nuclear Medicine and Molecular Imaging
JF - European Journal of Nuclear Medicine and Molecular Imaging
IS - 6
ER -