Table 2.
Main papers reporting patients with PHPT in the pediatric age.
| Author | Year | No. of pediatric patients | Main results |
|---|---|---|---|
| Allo et al. [12] | 1982 | 53 | Adenoma in 64.2% of studied patients; hyperplasia in 30.2%, overall in <18 y.o. (38%) vs. >18 y.o. (18.5%) patients |
| Lawson et al. [13] | 1996 | 11 | All sporadic PHPT. Delayed diagnosis in children; at diagnosis, 91% patients were symptomatic (renal stones 45%, abdominal pain 18%, learning difficulties 18%, musculoskeletal abnormalities 9%, and fatigue 9%). Mean serum Ca++: 3.39 mmol/l at diagnosis |
| Cronin et al. [14] | 1996 | 8 | Pediatric cohort with PHPT due to parathyroid adenoma, presenting mostly with hypercalcemic crisis (50%). Mean serum Ca++: 3.5 mmol/l at diagnosis |
| Loh et al. [15] | 1998 | 22 | Pediatric cohort with PHPT due to parathyroid adenoma, presenting mostly with fatigue (77%) or weakness (64%). Mean serum Ca++: 3.07 mmol/l; mean serum PTH 131 pg/ml at diagnosis |
| Harman et al. [16] | 1999 | 33 | Pediatric cohort with PHPT due to parathyroid adenoma, symptomatic in 94% of cases, mostly renal stones (7/33) and bone disease (9/33). Mean serum Ca++: 3.02 mmol/l at diagnosis; mean adenoma weight 0.96 gr |
| Hsu and Levine [17] | 2002 | 16 | Single adenomas in 11 patients; multiple-gland disease in 2 patients, including 1 with MEN2 |
| Kollars et al. [2] | 2005 | 52 | Symptomatic in 79% of cases; end-organ damage (nephrocalcinosis or lithiasis, acute pancreatitis, or bone involvement) in 44%. Mean serum Ca++: 3.1 mmol/l; mean serum P 1.8 mmol/l; mean serum PTH 76.3 pg/ml at diagnosis |
| Bhadada et al. [18] | 2008 | 14 | Single parathyroid adenoma in 85.7%, 1 patient with four-gland hyperplasia and 1 MEN-1. Main reported symptoms were bone disease, recurrent nephrolithiasis, and pancreatitis. Mean serum Ca++: 2.77 mmol/l; mean P 0.9 mmol/l; mean serum PTH 781 pg/ml at diagnosis |
| Libansky et al. [19] | 2008 | 10 | Pediatric cohort with parathyroid adenoma and 1 ectopic adenoma. Mean serum Ca++: 3.21 mmol/l; mean serum PTH 217.6 pg/ml at diagnosis |
| Mallet E [4] | 2008 | 55 | 31 adenomas and 11 hyperplasias underwent surgery. Medical management (i.v. diphosphonates) in 11 neonates. Mean serum Ca++: 3.64 mmol/l; mean serum P 1.3 mmol/l; mean serum PTH 536 pg/ml at diagnosis |
| Al-shanafey et al. [20] | 2010 | 5 | Surgical management in neonates with severe hyperparathyroidism, all symptomatic with lethargy, poor feeding, and irritability. In all patients, surgical treatment was curative. Mean serum Ca++: 3.84 mmol/l; mean serum PTH 3607 pg/ml at diagnosis |
| George et al. [5] | 2010 | 15 | Single parathyroid adenoma in 100% of patients. Main reported symptoms were bone pain, fractures, proximal myopathy, and renal calculi; 33.3% had postoperative HBS. Mean serum Ca++: 3.35 mmol/l; mean serum P 0.98 mmol/l; mean serum PTH 801 pg/ml at diagnosis; mean adenoma weight 3.84 gr |
| Shah et al. [21] | 2012 | 19 | Pediatric cohort with adenoma, presenting mostly bone pain (68%), weakness (68%), or fractures (52.6%) |
| Li et al. [22] | 2012 | 12 | Parathyroid adenoma in 100% (4/12 ectopic adenoma), presenting mostly urinary and bone tissue impairment. Mean serum Ca++: 3.82 mmol/l; mean serum P 1.18 mmol/l; mean serum PTH 1016 pg/ml at diagnosis |
| Belcher et al. [8] | 2013 | 230 | Literature review of studies regarding PHPT in the youth and adolescents. Single adenomas in 80% of patients, multiple-gland hyperplasia in 16.5% (MGH), double adenomas in 0.9%, and normal parathyroid gland in 2.6%. Of MGH, 50% were MEN I, MEN II, or familial non-MEN. Tc(99m)-sestamibi and ultrasound were 86% (37/43) and 74.5% (70/94) sensitive |
| Burke et al. [27 | 2013 | 19 | Study aiming to enhance the radioguided parathyroidectomy. Adenoma in 74% and hyperplasia in 26%. No complications were noted in the pediatric patients after surgery. Mean serum Ca++: 3.05 mmol/; mean serum PTH 177 pg/ml at diagnosis. Mean serum Ca++: 2.35 mmol/; mean serum PTH 33 pg/ml after the surgery. Mean adenoma weight 0.44 gr |
| Alagaratnam S and Kurzawinski [9] | 2014 | 29 | Pediatric cohort with adenoma presenting mostly gastrointestinal symptoms (41%) and skeletal manifestations (20.7%) |
| Roizen and Levine [6] | 2014 | 268 | A meta-analysis comparing biochemical profiles in the youth and adults: greater hypercalcemia and hypercalciuria in youths at similar concentrations of serum intact PTH. Mean serum Ca++: 3.2 mmol/; mean serum P 0.9 mmol/l; mean serum PTH 331 pg/ml; mean ALP 995 UI/l at diagnosis. Mean gland weight 2 gr |
| Mancilla et al. [23] | 2017 | 16 | Pediatric cohort with parathyroid and thymic (2/16) adenoma, mostly symptomatic (75%). Mean serum Ca++: 3.02 mmol/; mean serum PTH 177.3 pg/ml at diagnosis |
| Lou et al. [11] | 2017 | 40 | Pediatric cohort with different patterns at diagnosis; increasing rate of diagnosis in asymptomatic subjects, higher postoperative complications, and disease recurrence in patients with positive familial history. Mean serum Ca++: 2.91 mmol/; mean serum PTH 152.5 pg/ml at diagnosis |
| Vannucci et al. [24] | 2018 | 22 | Pediatric cohort with MEN-1 mutation presenting PHPT in 50% of cases, mostly asymptomatic (10/11) |
| Saponaro et al. [25] | 2018 | 31 | Young and adult population with PHPT: significantly lower PTH, higher serum in the younger group. Nephrolithiasis, fragility, fracture, and densitometric parameter rates did not differ between groups. Mean serum Ca++: 2.73 mmol/; mean serum P 0.77 mmol/l; mean serum 25-hydroxyvitamin D 19.6 ng/ml, mean serum PTH 111 pg/ml; mean ALP 232 UI/l at diagnosis |
| Wang et al. [7] | 2018 | 59 | Pediatric cohort reporting bone pain as the most common manifestation, high rate of rickets (45.8%) compared to adults (23.7%), and an important correlation to short stature. Hypercalciuria, more frequent in pediatrics, hypophosphatemia, and urolithiasis among adults. Mean serum Ca++: 3.01 mmol/; mean serum PTH 177 pg/ml; mean serum ALP 374 UI/l; mean serum 25-hydroxyvitamin D 12.9 ng/ml at diagnosis |
| Rampp et al. [26] | 2020 | 86 | Pediatric cohort with parathyroid and thymic (22/86) adenoma presenting systemic and neurocognitive symptoms in 64% and nephrolithiasis in 20%. Mean serum Ca++: 2.93 mmol/; mean serum PTH 110 pg/ml at diagnosis. Mean gland weight 0.3 gr. Mean serum Ca++: 2.42 mmol/l after the surgery |
| Jovanovic et al. [3] | 2020 | 14 | Adults and youth comparison; bone disease in the youth (42.9%) and asymptomatic disease in adults (39.3%). Preoperative serum calcium and PTH significantly higher in the youth than in adults. Mean serum Ca++: 3.47 mmol/; mean serum P 0.8 mmol/l; mean serum PTH 572.6 pg/ml at diagnosis. Mean serum Ca++: 2.42 mmol/; mean serum P 0.92 mmol/l; mean serum PTH 22.8 pg/ml; 25-hydroxyvitamin D 39.5 ng/ml after the surgery |