Case Report: Nine-year-old with parathyroid adenoma within the piriform sinus

Zenno, Anna; Ramamoorthy, Bhavishya; Hammoud, Dima A.; Quezado, Martha; Zeiger, Martha A.; Jha, Smita

doi:10.3389/fendo.2023.1171052

Cited by 4 publications

(4 citation statements)

References 17 publications

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“…A 91% sensitivity for dual-phase CT for single-gland tumors was revealed in one study [46], in which 25% of the lesions were ectopic parathyroid tumors (mostly intra-thymus, representing one-half of the ectopic presentations) [5]. Case reports included another 9 out of the 41 patients with ectopic parathyroid adenomas [10,11,29,34,68,69,[83][84][85], and 3 parathyroid carcinomas [72,73,78]; moreover, 8 subjects (N = 8/41) had brown tumors [15,36,38,40,71,72,74,77].…”

Section: Trans-pandemic Insights Into Paediatric Phpmentioning

confidence: 99%

“…Moreover, another unilateral case was reported, in relation to an ectopic adenoma, by Badhe et al [83]; Seo et al [10] added a case of a teenager who underwent VATS-based removal of an intra-thymic adenoma [10]. Alternatively, an ectopic site within the piriform sinus of a 9-year-old child required a reperformed parathyroidectomy [84]. Overwhelmingly, most recent reports include thoracoscopic resection of ectopic adenomas [11,29,68,69].…”

Section: Surgical Proceduresmentioning

confidence: 99%

“…Table 4. Case reports and series (<10 patients per article) of pediatric PHP (the list starts with the most recent publications) [9][10][11]15,29,[34][35][36][37][38][39][40]44,66,[68][69][70][71][72][73][74]77,78,[83][84][85]; the published cases with specific genetic testing were already introduced in Table 3 [17,53,61,62,64,65,67]. Abbreviations: C = case; CT = computed tomography; PT = parathyroid; PHP = primary hyperparathyroidism; SPECT/CT = single-photon emission computerized tomography/computed tomography; VATS = video-assisted thoracic surgery.…”

Section: Outcomementioning

confidence: 99%

“…We exceptionally included four studies that applied a (maximum) cutoff age of 20-21 (rather than 18), but for which the average and median ages of the entire studied populations were below 18 [5,26,27,52]. Case reports included patients within an age range of 9 to 17 years [9][10][11]15,29,[34][35][36][37][38][39][40]44,66,[68][69][70][71][72][73][74]77,78,[83][84][85]. Overall, no clear female predominance was registered, as is seen in the adult population with PHP [51,52].…”

Section: Trans-pandemic Insights Into Paediatric Phpmentioning

confidence: 99%

See 3 more Smart Citations

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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Section: Trans-pandemic Insights Into Paediatric Phpmentioning

confidence: 99%

Section: Surgical Proceduresmentioning

confidence: 99%

Section: Outcomementioning

confidence: 99%

Section: Trans-pandemic Insights Into Paediatric Phpmentioning

confidence: 99%

See 2 more Smart Citations

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

Carsote,

Stanciu,

Popa

et al. 2023

Biomedicines

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Ondansetron

2023

Reactions Weekly

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Invasive Testing for Preoperative Localization of Parathyroid Tumors

Graf,

Cochran,

Sadowski

et al. 2023

Journal of the Endocrine Society

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Context The identification of parathyroid tumor(s) in patients with persistent/recurrent primary hyperparathyroidism (PHPT) is critical for a successful reoperative surgery. If noninvasive studies (ultrasound, computed tomography, magnetic resonance imaging, sestamibi) fail to conclusively localize the tumor, invasive procedures (arteriography and selective venous sampling) are performed. Objective To describe our experience with invasive studies for parathyroid tumor localization and provide follow-up data on selective arterial hypocalcemic stimulation with central venous sampling, a technique developed at our center. Methods We identified patients who underwent preoperative invasive testing for localization of parathyroid tumor from 1991 to 2020. The result of each invasive localization study [arteriogram, hypocalcemic stimulation and selective venous sampling (SVS)] was categorized as true-positive, false-positive, and false-negative based on histology and biochemical outcome. Results Ninety-four patients with 96 tumor occurrences underwent invasive testing for parathyroid tumor localization. Arteriogram, hypocalcemic stimulation, and SVS accurately localized the tumor in 47 of 94 (50%), 56 of 93 (60%), and 51 of 62 (82%) tumors, respectively. Hypocalcemic stimulation was more likely to correctly localize the tumor when arteriogram showed a blush [37 of 50 (74%) vs 19 of 43 (44%), P = .01]. When both arteriogram and hypocalcemic stimulation yielded concordant positive findings, SVS did not change management in the 18 cases in which all 3 were performed. Twelve patients remained with persistent PHPT; all had recurrent disease with multiple affected glands. Conclusion Hypocalcemic stimulation is a useful adjunct in patients with PHPT who require invasive localization and can obviate the need for SVS. Clinical Trial number: NCT04969926

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Case Report: Nine-year-old with parathyroid adenoma within the piriform sinus

Cited by 4 publications

References 17 publications

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

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

Ondansetron

Invasive Testing for Preoperative Localization of Parathyroid Tumors

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