What is the optimal treatment for children with primary hyperparathyroidism?

Durkin, Emily T.; Nichol, Peter F.; Lund, Dennis P.; Chen, Herbert; Sippel, Rebecca S.

doi:10.1016/j.jpedsurg.2010.02.074

Cited by 37 publications

(42 citation statements)

References 18 publications

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“…Patients with normal calcium levels over the long-term are said to be cured. The outcomes of pediatric patients after parathyroidectomy for HPT are similar to adults, with 96-100% cured in reported series (2)(3)(4).…”

Section: Discussionmentioning

confidence: 76%

“…In adults, primary HPT is usually the result of a single parathyroid adenoma (80-85%) or hyperplasia of all four glands (10-15%) (1). In children and young adults with primary HPT, 60-92% have a single adenoma and 0-40% have four-gland hyperplasia (2)(3)(4). In both adults and children with primary HPT four gland hyperplasia is more common in patients with hereditary conditions such as Multiple Endocrine Neoplasia Type 1 (MEN1) (1,5) Patients with hyperparathyroidism can have a wide range of symptoms, or they can be asymptomatic (see Table 1) (2,4).…”

Section: Hyperparathyroidismmentioning

confidence: 99%

“…The history can often predict whether or not a patient has an adenoma or hyperplasia. For example, in a small series of 25 patients with primary HPT who were younger than 18 years and had no family history of parathyroid disease all had a single adenoma (3). The additional importance of localization is apparent from another series that found that 10% of patients had ectopic glands, including intrathymic and intrathyroidal adenomas (2).…”

Section: Parathyroidectomymentioning

confidence: 99%

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Parathyroid Surgery in Children

Burke

Chen

Gosain

2017

Endocrine Surgery in Children

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Section: Discussionmentioning

confidence: 76%

Section: Hyperparathyroidismmentioning

confidence: 99%

Section: Parathyroidectomymentioning

confidence: 99%

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Parathyroid Surgery in Children

Burke

Chen

Gosain

2017

Endocrine Surgery in Children

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“…[6][7][8]10,[13][14][15][16][17][18] Remarkably, PHPT is clinically symptomatic in most young patients, who typically present with signs or symptoms of hypercalcemia, skeletal complications, and/or nephrolithiasis. Few if any young patients with PHPT are discovered incidentally to have asymptomatic hypercalcemia.…”

Section: Introductionmentioning

confidence: 99%

“…6 However, over the past 30 years several large pediatric case series were published that increased the number of reported cases of PHPT in this age group to more than 300. [7][8][9][10][11][12][13][14][15][16][17][18] The largest of the primary studies in this area comprises 55 patients, of whom 11 were infants with PHPT and 44 were young children or adolescents with PHPT. 13 The largest study focusing solely on childhood and adolescent PHPT includes 52 patients.…”

Section: Introductionmentioning

confidence: 99%

Primary Hyperparathyroidism in Children and Adolescents

Roizen

Levine

2015

The Parathyroids

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Primary hyperparathyroidism (PHPT) is a common endocrine disorder in adults in whom the typical presentation is incidentally discovered as asymptomatic hypercalcemia. PHPT is much less common in children and adolescents, but has greater morbidity in this age group, as most young patients with PHPT will have symptomatic hypercalcemia or complications such as kidney stones, abdominal pain, and skeletal fragility. An important feature of PHPT in younger patients is the relatively high prevalence of germline inactivating mutations of the CASR gene, which encodes the calcium-sensing receptor. Biallelic CASR mutations cause neonatal severe hyperparathyroidism, a life-threatening condition that presents within days of life with marked hypercalcemia, respiratory distress, failure to thrive, and skeletal demineralization. By contrast, more common heterozygous CASR mutations are generally associated with a benign variant of PHPT termed familial hypocalciuric hypercalcemia. Appropriate management of PHPT in children and adolescents requires distinction between familial hypocalciuric hypercalcemia, which generally requires no specific treatment, and other forms of PHPT that are best treated by parathyroidectomy.

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A retrospective database analysis of pediatric parathyroidectomies from the United Kingdom registry of endocrine and Thyroid Surgeons

Embury‐Young,

Brennan,

Jackson

et al. 2024

World j. surg.

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IntroductionThe United Kingdom Registry of Endocrine and Thyroid Surgery (UKRETS) holds the largest database of pediatric parathyroidectomy cases globally. There are currently no quoted acceptable cure or complication rates in the literature.MethodsThis retrospective database analysis evaluates the efficacy and safety of targeted parathyroidectomy (tPTx) and bilateral neck exploration (BNE) in first‐time parathyroidectomy for pediatric primary hyperparathyroidism (PHPT) through analysis of the UKRETS database (1995–2022). Pre‐, intra‐ and postoperative outcomes were assessed and analyzed.Results168 cases underwent parathyroidectomy; 25 (15%) familial and 143 (85%) sporadic PHPT. 69% were female with a mean age of 10 years (Range 0–17). BNE was the most common operative approach (61%; n = 103/168). The most frequently used imaging modality was US (80%; n = 135/168). Mean number of glands excised in familial cases was three compared to one gland in sporadic cases (p < 0.05). Familial cases had a significantly higher rate of postoperative hypocalcemia (32% vs. 9%, p < 0.05) and all were BNE. Cure rate was 96.9% (n = 127/131), with differences in cure rates that did not reach statistical significance (sporadic 98.2% vs. familial 90.5%, p = 0.06). Preoperative localization (image‐positive or negative) made no difference to cure rates in either familial (90% vs. 91%, p = 0.94) or sporadic (97.5% vs. 100%, p = 0.4) cases.ConclusionsThis analysis demonstrates that first‐time pediatric parathyroidectomy for PHPT is safe and effective. Familial cases have a higher rate of postoperative hypocalcemia; therefore, parents should be informed of this when consented. Targeted parathyroidectomy is safe and effective in both sporadic and familial cases, as long as there is positive preoperative imaging.

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What is the optimal treatment for children with primary hyperparathyroidism?

Cited by 37 publications

References 18 publications

Parathyroid Surgery in Children

Parathyroid Surgery in Children

Primary Hyperparathyroidism in Children and Adolescents

A retrospective database analysis of pediatric parathyroidectomies from the United Kingdom registry of endocrine and Thyroid Surgeons

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