Case Report

“Primary Hyperparathyroidism (PHPT) in Children: Two Case Reports and Review of the Literature”

Table 2

Main papers reporting patients with PHPT in the pediatric age.

AuthorYearNo. of pediatric patientsMain results

Allo et al. [12]198253Adenoma 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]199611All 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]19968Pediatric 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]199822Pediatric 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]199933Pediatric 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]200216Single adenomas in 11 patients; multiple-gland disease in 2 patients, including 1 with MEN2
Kollars et al. [2]200552Symptomatic 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]200814Single 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]200810Pediatric 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]20085531 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]20105Surgical 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]201015Single 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]201219Pediatric cohort with adenoma, presenting mostly bone pain (68%), weakness (68%), or fractures (52.6%)
Li et al. [22]201212Parathyroid 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]2013230Literature 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. [27201319Study 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]201429Pediatric cohort with adenoma presenting mostly gastrointestinal symptoms (41%) and skeletal manifestations (20.7%)
Roizen and Levine [6]2014268A 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]201716Pediatric 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]201740Pediatric 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]201822Pediatric cohort with MEN-1 mutation presenting PHPT in 50% of cases, mostly asymptomatic (10/11)
Saponaro et al. [25]201831Young 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]201859Pediatric 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]202086Pediatric 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]202014Adults 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