Diagnostic challenge in parathyroid imaging: incidental schwannoma mimicking primary hyperparathyroidism on 18F-fluorocholine PET/CT.

IntroductionNuclear medicine parathyroid imaging is important in the identification of hyperfunctioning parathyroid glands in primary hyperparathyroidism (pHPT), but it may be also valuable before surgical treatment in secondary hyperparathyroidism (sHPT). Parathyroid radionuclide imaging with scintigraphy or positron emission tomography (PET) is a highly sensitive procedure for the assessment of the presence and number of hyperfunctioning parathyroid glands, located either at typical sites or ectopically. The treatment of pHPT is mostly directed toward minimally invasive parathyroidectomy, especially in cases with a single adenoma. In experienced hands, successful surgery depends mainly on the exact preoperative localization of one or more hyperfunctioning parathyroid adenomas. Failure to preoperatively identify the hyperfunctioning parathyroid gland challenges minimally invasive parathyroidectomy and might require bilateral open neck exploration.MethodsOver a decade has now passed since the European Association of Nuclear Medicine (EANM) issued the first edition of the guideline on parathyroid imaging, and a number of new insights and techniques have been developed since. The aim of the present document is to provide state-of-the-art guidelines for nuclear medicine physicians performing parathyroid scintigraphy, single-photon emission computed tomography/computed tomography (SPECT/CT), positron emission tomography/computed tomography (PET/CT), and positron emission tomography/magnetic resonance imaging (PET/MRI) in patients with pHPT, as well as in those with sHPT.ConclusionThese guidelines are written and authorized by the EANM to promote optimal parathyroid imaging. They will assist nuclear medicine physicians in the detection and correct localization of hyperfunctioning parathyroid lesions.

ABSTRACTBackground and aimRadiological imaging is routinely used in patients with primary hyperparathyroidism to localize the abnormal adenoma and to plan surgical approach. We report an unusual cause of false-positive localization on parathyroid sestamibi scan, i.e., not previously described in the literature.Case reportA 66-year-old man with primary hyperparathyroidism showed a discrete persistent focus in the left infraclavicular area during localization using sestamibi scan. Ultrasound of the neck was negative showing only small bilateral thyroid nodules. Single-photon emission and four-dimensional computed tomography (CT) scans showed an intense focus of the tracer uptake and a 1-cm lesion near the left subclavian vein (SCV), corresponding to the infraclavicular hot spot. Initial infraclavicular exploration showed the lesion to be a collapsible saccular varix of the left SCV. Bilateral neck exploration led to the discovery of the actual right parathyroid adenoma beneath an exophytic thyroid nodule. The patient made an uneventful recovery and remains normocalcemic at 1-year follow up.Conclusion and clinical significanceHot spot on parathyroid imaging outside the line of embryological descent should be interpreted with caution. Vascular retention of injected isotope within a saccular varix of the neck vein can give rise to falsepositive results on sestamibi scans. Bilateral neck exploration remains the “gold standard” procedure when localization scans either are negative or turn out to have false-positive findings.How to cite this articleSingaporewalla RM, Negi A, Seow DYB, Chinchure D. An Unusual Cause of Hot Spot on Parathyroid Imaging. World J Endoc Surg 2016;8(2):164-167.

Parathyroid imaging is essential for the detection and localization of hyperfunctioning parathyroid tissue in patients with primary hyperparathyroidism (pHPT). Surgical treatment of pHPT mainly consists of minimally invasive parathyroidectomy (MIP), as a single adenoma represents the most common cause of this endocrine disorder. Successful surgery requires an experienced surgeon and relies on the correct preoperative detection and localization of hyperfunctioning parathyroid glands. Failure to preoperatively identify the culprit parathyroid gland by imaging may entail a more invasive surgical approach, including bilateral open neck exploration, with higher morbidity compared to minimally invasive parathyroidectomy. Parathyroid imaging may be also useful before surgery in case of secondary hyperparathyroidism (sHPT) or hereditary disorders (MEN 1, 2, 4) as it enables correct localization of typically located parathyroid glands, detection of ectopic as well as supernumerary glands. It is now accepted by most surgeons experienced in parathyroid surgery that preoperative imaging plays a key role in their patients' management. Recently, the European Association of Nuclear Medicine (EANM) issued an updated version of its Guidelines on parathyroid imaging. Its aim is to precise the role and the advantages and drawbacks of the various imaging modalities proposed or well established in the preoperative imaging strategy. It also aims to favor high performance in indicating, performing, and interpreting those examinations. The objective of the present article is to offer a summary of those recent EANM Guidelines and their originality among other Guidelines in this domain issued by societies of nuclear medicine physicians or other disciplines.

MP95-08 PRIMARY HYPERPARATHYROIDISM: A SOMETIMES ELUSIVE DIAGNOSIS

Health care consumer organizations and insurance companies increasingly are scrutinizing value when considering reimbursement policies for medical interventions. Recently, members of several American Academy of Otolaryngology-Head & Neck Surgery (AAO-HNS) committees worked closely with one insurance company to refine reimbursement policies for preoperative localization imaging in patients undergoing surgery for primary hyperparathyroidism. This endeavor led to an AAO-HNS parathyroid imaging consensus statement (https://www.entnet.org/content/parathyroid-imaging). The American Head and Neck Society Endocrine Surgery Section gathered an expert panel of authors to delineate imaging options for preoperative evaluation of surgical candidates with primary hyperparathyroidism. We review herein the current literature for preoperative parathyroid localization imaging, with discussion of efficacy, cost, and overall value. We recommend that planar sestamibi imaging, single photon emission computed tomography (SPECT), SPECT/CT, CT neck/mediastinum with contrast, MRI, and four dimensional CT (4D-CT) may be used in conjunction with high-resolution neck ultrasound to preoperatively localize pathologic parathyroid glands. PubMed literature on parathyroid imaging was reviewed through February 1, 2019.

Disclosure: J.P. Suelto: None. G.D. Wassmer: None. Background: Eighty percent of the cases of Primary Hyperparathyroidism is caused by a single adenoma and only about 15% are multiglandular adenoma. Parathyroid imaging like Sestamibi is less sensitive in detecting multiglandular disease. Clinical Case: A 63 year-old female presented with intermittent left lower back pain for 7 years. There was no joint pain, weakness, urinary symptoms, constipation and abdominal pain. In 2019, due to persistence of left back pain, abdominal ultrasound done showed a right non-obstructing nephrolithiasis. Ionized and total calcium were elevated at 1.53 meq/L (NV 1.12-1.32 meq/L) and 11.35 ng/dl (NV 8.42-10.23 ng/dl) respectively with normal creatinine, phosphorus and albumin. PTH was elevated at 116.6 pg/ml (NV 10-65 pg/ml). Primary Hyperparathyroidism probably secondary to parathyroid adenoma was considered. Thyroid ultrasound showed right thyroid cyst but no enlarged parathyroid gland. Parathyroid imaging using Dual Isotope Subtraction Method neither showed evidence of adenoma. Patient was advised parathyroidectomy but did not consent hence started on Cinacalcet. In 2021, ionized calcium was still elevated to 1.58 meq/L with elevated PTH 151 pg/ml and low Vitamin D level 22.76 ng/ml (>30 ng/mL). Patient was given Vitamin D3 2000 IU daily and Cinacalcet was up-titrated to 60mg daily. Sestamibi/SPECT CT showed faint focus of radioactivity in the right thyroidal bed area suspicious for parathyroid adenoma or hyperplasia. She then agreed to have parathyroidectomy. There was no decrease in PTH after removal of the right inferior parathyroid gland (measuring 1.8x0.6x0.6cm, weighs 500mg), hence re-exploration was done. It was only after the removal of the right superior parathyroid gland (measuring 2.8x0.9x0.7cm, weighs 1 gram) when the 5 and 10 minute post excision PTH decreased to 123.91 and 76.01 pg/ml respectively from preincision PTH of 407.19 pg/ml. Histopathology showed Parathyroid adenoma of right inferior and superior parathyroid glands. No further evidence of hypercalcemia was noted at 6-month follow-up. Conclusion: Double adenoma is reported in 2% to 12% of patients with primary hyperparathyroidism. This occurs predominantly as bilateral superior in 45% and on same side of the neck in 18%. Sestamibi scanning is poor in the presence of multiglandular disease. It was observed that the presence of oxyphil cells may cause longer sestamibi retention in parathyroid tissues. Given the same amount of oxyphil cells in ipsilateral adenomas, the tracer may be distributed hence faintly seen on imaging. Addition of SPECT/CT/4D CT may improve the anatomical localization. Another reason for false negative imaging in this patient can be due to use of Cinacalcet which may decrease scintigraphy uptake of parathyroid adenoma. The combination of preoperative imaging localization and intraoperative PTH monitoring permits successful surgical outcome. Presentation: Saturday, June 17, 2023

Parathyroid imaging: Its current status and future role

Thyroid and parathyroid imaging

Parathyroid Imaging

Parathyroid imaging modalities have been used to guide clinicians and surgeons in finding the source of hyperparathyroidism for over 40 years. Primary hyperparathyroidism (PHPT) is generally caused by a parathyroid gland(s) autonomous production of parathyroid hormone (PTH), associated by enlargement of one or more glands. Noninvasive imaging procedures that are used in the management of hyperparathyroidism are anatomical (ultrasound, computer tomography, magnetic resonance imaging) and/or functional (nuclear medicine techniques: planar scintigraphy, single photon emission tomography, positron emission imaging) and/or hybrid imaging.

Parathyroid lipoadenoma is an unusual cause of primary hyperparathyroidism. Only a few earlier reports have documented parathyroid imaging in diagnostic studies of parathyroid lipoadenoma. Our case was a 27-year-old man, who was originally diagnosed with primary hyperparathyroidism. He underwent a Tc-99m sestamibi dual-phase parathyroid imaging study, which revealed a right-inferior parathyroid hyperfunctional lesion. The first operation removed a lesion of 1.5 cm in diameter in the area, and parathyroid lipoadenoma was diagnosed by pathology. However, hypercalcemia persisted. One week later, a Tc-99m sestamibi whole-body scan and another dual-phase imagining were performed, which demonstrated a residual lesion in the inferior part of the right thyroid region, while no ectopic lesion was found. A second surgery was performed and pathological diagnosis was confirmed as parathyroid lipoadenoma again. Our case demonstrated that although hyperfunctional parathyroid lipoadenoma is rare, Tc-99m sestamibi parathyroid imaging is reliable in locating the lesion for surgical purposes. And Tc-99m sestamibi imaging is useful in reoperative and persistent hyperparathyroidism situations as well.

Evaluation of the phenotype of primary hyperparathyroidism (PHPT), adherence to International Guidelines for parathyroidectomy (PTx), and rate of surgical cure. From January 2014-January 2016, we performed a prospective, multicenter study in patients with newly diagnosed PHPT. Biochemical and instrumental data were collected at baseline and during 1-year follow-up. Over the first year we enrolled 604 patients (age 61 ± 14years), mostly women (83%), referred for further evaluation and treatment advice. Five hundred sixty-six patients had sporadic PHPT (93.7%, age 63 ± 13 years), the remaining 38 (6.3%, age 41 ± 17 years) had familial PHPT. The majority of patients (59%) were asymptomatic. Surgery was advised in 281 (46.5%). Follow-up data were available in 345 patients. Eighty-seven of 158 (55.1%) symptomatic patients underwent PTx. Sixty-five (53.7%) of 121 asymptomatic patients with at least one criterion for surgery underwent PTx and 56 (46.3%) were followed without surgery. Negative parathyroid imaging studies predicted a conservative approach [symptomatic PHPT: OR 18.0 (95% CI 4.2-81.0) P < 0.001; asymptomatic PHPT: OR 10.8, (95% CI 3.1-37.15) P < 0.001). PTx was also performed in 16 of 66 (25.7%) asymptomatic patients without surgical criteria. Young age, serum calcium concentration, 24h urinary calcium, positive parathyroid imaging (either ultrasound or MIBI scan positive in 75% vs. 16.7%, P = 0.001) were predictors of parathyroid surgery. Almost all (94%) of patients were cured by PTx. Italian endocrinologists do not follow guidelines for the management of PHPT. Negative parathyroid imaging studies are strong predictors of a non-surgical approach. PTx is successful in almost all patients.

SummaryDistinguishing primary hyperparathyroidism (PHPT) from familial hypocalciuric hypercalcaemia (FHH) can be challenging. Currently, 24-h urinary calcium is used to differentiate between the two conditions in vitamin D replete patients, with urinary calcium creatinine clearance ratio (UCCR) <0.01 suggestive of FHH and >0.02 supportive of PHPT. A 26-year-old Caucasian gentleman presented with recurrent mild hypercalcaemia and inappropriately normal parathyroid hormone (PTH) following previous parathyroidectomy 3 years prior. He had symptoms of fatigue and light-headedness. He did not have any other symptoms of hypercalcaemia. His previous evaluation appeared to be consistent with PHPT as evidenced by hypercalcaemia with inappropriately normal PTH and UCCR of 0.0118 (borderline low using guidelines of >0.01 consistent with PHPT). He underwent parathyroidectomy and three parathyroid glands were removed. His calcium briefly normalised after surgery, but rose again to pre-surgery levels within 3 months. Subsequently, he presented to our centre and repeated investigations showed 24-h urinary calcium of 4.6 mmol/day and UCCR of 0.0081 which prompted assessment for FHH. His calcium-sensing receptor (CASR) gene was sequenced and a rare inactivating variant was detected. This variant was described once previously in the literature. His mother was also confirmed to have mild hypercalcaemia with hypocalciuria and, on further enquiry, had the same CASR variant. The CASR variant was classified as likely pathogenic and is consistent with the diagnosis of FHH. This case highlights the challenges in differentiating FHH from PHPT. Accurate diagnosis is vital to prevent unnecessary surgical intervention in the FHH population and is not always straightforward.Learning points:Distinguishing FHH from PHPT with co-existing vitamin D deficiency is difficult as this can mimic FHH. Therefore, ensure patients are vitamin D replete prior to performing 24-h urinary calcium collection.Individuals with borderline UCCR could have either FHH or PHPT. Consider performing CASR gene sequencing for UCCR between 0.01 and 0.02.Parathyroid imaging is not required for making the diagnosis of PHPT. It is performed when surgery is considered after confirming the diagnosis of PHPT.

Evaluación diagnóstica y diagnóstico diferencial del hiperparatiroidismo primario

This article illustrates the complementary nature of preoperative radionuclide parathyroid imaging and intraoperative rapid parathyroid hormone (PTH) assays in primary hyperparathyroid disease. The authors review the literature on these procedures and compare this protocol and its cost-effectiveness with those of the classic four-gland exploration. Preoperative parathyroid imaging with Tc-99m MIBI and intraoperative rapid PTH assays were performed at the time of neck exploration. One of two parathyroid adenomas seen on radionuclide images would have been missed if the authors had relied solely on the initial decrease in PTH assay value to a normal level. Tc-99m MIBI imaging and intraoperative rapid PTH assays are complementary; when used together, they lessen the likelihood that abnormal parathyroid glands will be overlooked. This experience and that of others suggest these combined procedures are cost-effective.