Dual-phase computed tomography for localization of parathyroid lesions in children and adolescents with primary hyperparathyroidism

Sharma, Anima; Patil, Virendra; Sarathi, Vijaya; Purandare, Nilendu; Hira, Priya; Memon, Saba Samad; Jadhav, Swati; Karlekar, Manjiri; Lila, Anurag; Bandgar, Tushar

doi:10.1016/j.ando.2023.03.001

Cited by 3 publications

(11 citation statements)

References 42 publications

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“…Despite recent progress in the era of imaging in PHP, CT remains a mostly used and feasible approach as part of a routine exam in real-life medicine [39,40]. Dual-phase (non-enhanced and arterial) CT protocols increase the accuracy of results, in both the adult and paediatric (but less accurate) population with PHP [41]. Of note, CT should be avoided if possible in children, especially as non-radiating techniques are available.…”

Section: Concordance Of Pre-operatory Localisation Studies In Phpmentioning

confidence: 99%

Pitfalls of DualTracer 99m-Technetium (Tc) Pertechnetate and Sestamibi Scintigraphy before Parathyroidectomy: Between Primary-Hyperparathyroidism-Associated Parathyroid Tumour and Ectopic Thyroid Tissue

Carsote,

Stanciu,

Popa

et al. 2023

Medicina

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Diagnosis of primary hyperparathyroidism (PHP) is based on blood assessments in terms of synchronous high calcium and PTH (parathormone), but further management, particularly parathyroid surgery that provides the disease cure in 95–99% of cases, requires an adequate localisation of the parathyroid tumour/tumours as the originating source, with ultrasound and 99m-Technetium (99m-Tc) sestamibi scintigraphy being the most widely used. We aimed to introduce an adult female case diagnosed with PHP displaying unexpected intra-operatory findings (ectopic thyroid tissue) in relation to concordant pre-operatory imaging modalities (ultrasound + dual-phase 99m-Tc pertechnetate and sestamibi scintigraphy + computed tomography) that indicated bilateral inferior parathyroid tumours. A sudden drop in PTH following the removal of the first tumour was the clue for performing an extemporaneous exam for the second mass that turned out to be non-malignant ectopic thyroid tissue. We overviewed some major aspects starting from this case in point: the potential pitfalls of pre-operatory imaging in PHP; the concordance/discordance of pre-parathyroidectomy localisation modalities; the need of using an additional intra-operatory procedure; and the clues of providing a distinction between pathological parathyroids and thyroid tissue. This was a case of adult PHP, whereas triple localisation methods were used before parathyroidectomy, showing concordant results; however, the second parathyroid adenoma was a false positive image and an ectopic thyroid tissue was confirmed. The pre-operatory index of suspicion was non-existent in this patient. Hybrid imaging modalities are most probably required if both thyroid and parathyroid anomalies are suspected, but, essentially, awareness of the potential pitfalls is mandatory from the endocrine and surgical perspectives. Current gaps in imaging knowledge to guide us in this area are expected to be solved by the significant progress in functional imaging modalities. However, the act of surgery, including the decision of a PTH assay or extemporaneous exam (as seen in our case), represents the key to a successful removal procedure. Moreover, many parathyroid surgeons may currently perform 4-gland exploration routinely, precisely to avoid the shortcomings of preoperative localisation.

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Section: Concordance Of Pre-operatory Localisation Studies In Phpmentioning

confidence: 99%

Pitfalls of DualTracer 99m-Technetium (Tc) Pertechnetate and Sestamibi Scintigraphy before Parathyroidectomy: Between Primary-Hyperparathyroidism-Associated Parathyroid Tumour and Ectopic Thyroid Tissue

Carsote,

Stanciu,

Popa

et al. 2023

Medicina

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“…These two patients represent the first of their kind in the pediatric population according to the authors, who identified only four other cases, all in adults [44] (Table 2). Biomedicines 2023, 11, x FOR PEER REVIEW 6 of 27 [46] with germline mutations: 3 with CDC73 and 1 with CASR)…”

Section: Clinical Presentationmentioning

confidence: 99%

“…Szabo Yamashita [27] Single Biomedicines 2023, 11, x FOR PEER REVIEW 6 of 27 [46] with germline mutations: 3 with CDC73 and 1 with CASR)…”

Section: Clinical Presentationmentioning

confidence: 99%

“…El Allali [50] Retrospective study N = 63 patients with PHP who underwent genetic analysis 52% had genetic PHP Younger group (94%) had CASR mutations Older group presented other mutations (MEN1, CDC73, RET, and CDKN1B) Sharanappa [28] Retrospective study (between 1989 [46] with germline mutations: 3 with CDC73 and 1 with CASR)…”

Section: Clinical Presentationmentioning

confidence: 99%

“…Szabo Yamashita [27] Single-center retrospective study (between 1994 and 2020) N = 66 with PHP (61% females) Age: ≤21 years old (mean age of 17. [46] with germline mutations: 3 with CDC73 and 1 with CASR)…”

Section: Clinical Presentationmentioning

confidence: 99%

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Pediatric Neuroendocrine Neoplasia of the Parathyroid Glands: Delving into Primary Hyperparathyroidism

Carsote,

Stanciu,

Popa

et al. 2023

Biomedicines

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Our objective was to overview the most recent data on primary hyperparathyroidism (PHP) in children and teenagers from a multidisciplinary perspective. Methods: narrative review based on full-length, English-language papers (from PubMed, between January 2020 and July 2023). Results: 48 papers (14 studies of ≥10 subjects/study, and 34 case reports/series of <10 patients/study). Study-sample-based analysis: except for one case–control study, all of the studies were retrospective, representing both multicenter (n = 5) and single-center (n = 7) studies, and cohort sizes varied from small (N = 10 to 19), to medium-sized (N = 23 to 36) and large (N = 63 to 83); in total, the reviewed studies covered 493 individuals with PHP. Case reports/series (n = 34, N = 41): the mean ages studied varied from 10.2 to 14 years in case reports, and the mean age was 17 years in case series. No clear female predominance was identified, unlike that observed in the adult population. Concerning the assessments, there were four major types of endpoints: imaging data collection, such as ultrasound, 99mTc Sestamibi, or dual-phase computed tomography (CT); gene testing/familial syndrome identification; preoperative findings; and exposure to surgical outcome/preoperative drugs, like cinacalcet, over a 2.2-year median (plus two case reports of denosumab used as an off-label calcium-lowering agent). Single-gland cases (representing 85% of sporadic cases and 19% of genetic PHP cases) showed 100% sensitivity for neck ultrasounds, with 98% concordance with 99mTc Sestamibi, as well as a 91% sensitivity for dual-phase CT, with 25% of the lesions being ectopic parathyroids (mostly mediastinal intra-thymic). Case reports included another 9/41 patients with ectopic parathyroid adenomas, 3/41 with parathyroid carcinomas, and 8/41 subjects with brown tumors. Genetic PHP (which has a prevalence of 5–26.9%) mostly involved MEN1, followed by CDC73, CASR, RET, and CDKN1B, as well as one case of VHL. Symptomatic PHP: 70–100% of all cases. Asymptomatic PHP: 60% of genetic PHP cases. Renal involvement: 10.5% of a cohort with genetic PHP, 71% of sporadic PHP cases; 50% (in a cohort with a mean age of 16.7), 29% (in a cohort with a mean age of 15.2); 0% (in infancy) to 50–62% (in teenagers). Bone anomalies: 83% of the children in one study and 62% of those in two other studies. Gastrointestinal issues: 40% of one cohort, but the data are heterogeneous. Cure rate through parathyroidectomy: 97–98%. Recurrent PHP: 2% of sporadic PHP cases and 38% of familial PHP cases. Hungry bone syndrome: maximum rate of 34–40%. Case reports identified another 7/41 subjects with the same post-parathyroidectomy condition; a potential connection with ectopic presentation or brown tumors is suggested, but there are limited data. Minimally invasive thoracoscopic approaches for ectopic tumors seemed safe. The current level of statistical evidence on pediatric PHP qualifies our study- and case-sample-based analysis (n = 48, N = 534) as one of the largest of its kind. Awareness of PHP is the key factor to benefit our young patients.

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The polar vessel sign: insights from CT imaging analysis in Asian Indian primary hyperparathyroidism

Sharma,

Memon,

Goroshi

et al. 2024

Endocrine

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Dual-phase computed tomography for localization of parathyroid lesions in children and adolescents with primary hyperparathyroidism

Cited by 3 publications

References 42 publications

Pitfalls of DualTracer 99m-Technetium (Tc) Pertechnetate and Sestamibi Scintigraphy before Parathyroidectomy: Between Primary-Hyperparathyroidism-Associated Parathyroid Tumour and Ectopic Thyroid Tissue

Pitfalls of DualTracer 99m-Technetium (Tc) Pertechnetate and Sestamibi Scintigraphy before Parathyroidectomy: Between Primary-Hyperparathyroidism-Associated Parathyroid Tumour and Ectopic Thyroid Tissue

Pediatric Neuroendocrine Neoplasia of the Parathyroid Glands: Delving into Primary Hyperparathyroidism

The polar vessel sign: insights from CT imaging analysis in Asian Indian primary hyperparathyroidism

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