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      Benefits of 11C-methionine PET/MRI and intraoperative fluorescence in treating hyperparathyroidism

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          Abstract

          Multiple endocrine neoplasia 1 (MEN1) is a rare genetic syndrome characterized by the manifestation of tumors in endocrine glands most often in the parathyroid gland (PG). Treatment may involve several parathyroidectomies (PTX), especially in young patients, which increases the risk of postoperative complications. We present a 16-year-old patient with a family history of MEN1 syndrome. The patient started to show biochemical signs of hyperparathyroidism (HPT) and hypercalcemia at the age of 10. One and a half years later a PTX was successfully performed with removal of the two left PGs. However, a rise in plasma parathyroid hormone and ionized calcium was observed 4 years later. Preoperative noninvasive imaging with 99mTc-sestamibi scintigraphy showed no definitive parathyroid adenoma. A 11C-methionine position emission tomography combined with MRI (MET-PET/MRI) was then performed and detected a focus posterior to the lower part of the right thyroid lobe. Intraoperative angiography with fluorescence and indocyanine green dye was used to assess the vascularization of the remaining PGs. The lower right PG was removed. The patient was discharged with normalized biochemical values and without postoperative complications. Recurrence of primary HPT is frequent in MEN1 patients which often necessitates repeated operations. Our case report showed that the use of advanced noninvasive preoperative imaging techniques and intraoperative fluorescent imaging are valuable tools and should be taken into consideration in selected cases to avoid postoperative complications. To our knowledge, this is the first case where MET-PET/MRI has been used to detect parathyroid pathology.

          Learning points:
          • MEN1 patients will develop parathyroid disease, which eventually will lead to surgical treatment with removal of the pathological glands.

          • Preoperatively usage of MRI combined with PET tracers such as 11C-methionine and 18F-Fluorocholine are able to detect parathyroid pathology with a higher sensitivity than conventional imaging.

          • Techniques using intraoperatively angiography with fluorescence and florescent dyes allow surgeons to verify the vascularization of each parathyroid gland.

          • Optimization of noninvasive preoperative imaging techniques and intraoperative fluorescent imaging are valuable tools and should be taken into consideration when performing PTX consecutively in the same patient to avoid postoperative complications.

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          Most cited references12

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          Clinical studies of multiple endocrine neoplasia type 1 (MEN1)

          Multiple endocrine neoplasia type 1 (MEN1) is an autosomal dominant disorder characterized by the combined occurrence of parathyroid, pancreatic islet and anterior pituitary tumours. To facilitate a screening programme for MEN1, we investigated 709 people (364 males and 345 females, age range 1-84 years) from 62 MEN1 families, and 36 non-familial MEN1 patients. Of those investigated, 220 (95 males and 125 females, age range 8-79 years) suffered from MEN1. Parathyroid, pancreatic and pituitary tumours occurred in 95%, 41% and 30% of the patients, respectively. Parathyroid tumours were the first manifestation of MEN1 in 87% of patients, and amongst the pituitary and pancreatic tumours, somatotrophinomas and gastrinomas were more common in patients above the age of 40 years, whilst insulinomas occurred more frequently in patients below the age of 40 years. Biochemical screening indicated that the penetrance of MEN1 by the ages of 20, 35 and 50 years was 43%, 85% and 94%, respectively, and that the development of MEN1 was confined to first-degree relatives in 91% of patients and to second-degree relatives in 9% of patients. These findings have helped to define a proposed screening programme for MEN1.
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            Heterogeneous and low-intensity parathyroid autofluorescence: Patterns suggesting hyperfunction at parathyroid exploration

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              Use of PET tracers for parathyroid localization: a systematic review and meta-analysis

              Purpose The great spatial and temporal resolution of positron emission tomography might provide the answer for patients with primary hyperparathyroidism (pHPT) and non-localized parathyroid glands. We performed a systematic review of the evidence regarding all investigated tracers. Methods A study was considered eligible when the following criteria were met: (1) adults ≥17 years old with non-familial pHPT, (2) evaluation of at least one PET isotope, and (3) post-surgical and pathological diagnosis as the gold standard. Performance was expressed in sensitivity and PPV. Results Twenty-four papers were included subdivided by radiopharmaceutical: 14 studies investigated l-[11C]Methionine (11C-MET), one [11C]2-hydroxy-N,N,N-trimethylethanamium (11C-CH), six 2-deoxy-2-[18F]fluoro-d-glucose (18F-FDG), one 6-[18F] fluoro-l-DOPA (18F-DOPA), and three N-[(18F)Fluoromethyl]-2-hydroxy-N,N-dimethylethanaminium (18F-FCH). The 14 studies investigating MET included a total of 327 patients with 364 lesions. Sensitivity for the detection of a lesion in the correct quadrant had a pooled estimate of 69 % (95 % CI 60–78 %). Heterogeneity was overall high with I2 of 51 % (p = 0.01) for all 14 studies. Pooled PPV ranged from 91 to 100 % with a pooled estimate of 98 % (95 % CI 96–100 %). Of the other investigated tracers, 18-FCH seems the most promising with high diagnostic performance. Conclusions The results of our meta-analysis show that 11C-MET PET has an overall good sensitivity and PPV and may be considered a reliable second-line imaging modality to enable minimally invasive parathyroidectomy. Our literature review suggests that 18F-FCH PET may produce even greater accuracy and should be further investigated using both low-dose CT and MRI for anatomical correlation.

                Author and article information

                Journal
                Endocrinol Diabetes Metab Case Rep
                Endocrinol Diabetes Metab Case Rep
                EDM
                Endocrinology, Diabetes & Metabolism Case Reports
                Bioscientifica Ltd (Bristol )
                2052-0573
                29 July 2020
                2020
                : 2020
                : 20-0053
                Affiliations
                [1 ]Department of Nuclear Medicine and PET-Centre , Aarhus University Hospital, Aarhus, Denmark
                [2 ]Department of Otorhinolaryngology , Aarhus University Hospital, Aarhus, Denmark
                [3 ]Department of Endocrinology and Internal Medicine , Aarhus University Hospital, Aarhus, Denmark
                Author notes
                Correspondence should be addressed to T V Luong; Email: thiluo@ 123456rm.dk
                Author information
                http://orcid.org/0000-0002-5772-4494
                Article
                EDM200053
                10.1530/EDM-20-0053
                7424345
                64004fbf-e14d-4cb5-9487-bca6c2ffd74c
                © 2020 The authors

                This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License..

                History
                : 20 April 2020
                : 09 July 2020
                Categories
                Paediatric
                Female
                White
                Denmark
                Parathyroid
                PTH
                Men1
                Hyperparathyroidism (Primary)
                Hypercalcaemia
                Parathyroid Adenoma
                Hypercalcaemia
                Headache
                PTH
                PET scan
                MRI
                Angiography
                Calcium (serum)
                Sestamibi scan
                Ultrasound scan
                Parathyroidectomy
                Genetics
                Paediatrics
                Novel Diagnostic Procedure
                Novel Diagnostic Procedure

                paediatric,female,white,denmark,parathyroid,pth,men1,hyperparathyroidism (primary),hypercalcaemia,parathyroid adenoma,headache,pet scan,mri,angiography,calcium (serum),sestamibi scan,ultrasound scan,parathyroidectomy,genetics,paediatrics,novel diagnostic procedure,july,2020

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