Continuous Subcutaneous Recombinant Parathyroid Hormone (1–34) Infusion in the Management of Childhood Hypoparathyroidism Associated with Malabsorption

Saraff, Vrinda; Rothenbühler, Anya; Högler, Wolfgang; Linglart, Agnès

doi:10.1159/000479867

Cited by 25 publications

(20 citation statements)

References 16 publications

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“…Clinical trials in adults have shown good efficacy, however long term safety needs to be further studied 30. Continuous subcutaneous infusion of recombinant PTH1–34 has successfully been used in children with hypoparathyroidism who were difficult to manage on conventional therapy31 32; however, its use is not licensed in children and no randomised trials have been performed .…”

Section: Treatment Of Hypocalcaemiamentioning

confidence: 99%

Investigation and management of hypocalcaemia

Nadar

Shaw

2020

Arch Dis Child

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Hypocalcaemia is a common clinical scenario in children with a range of aetiological causes. It will often present with common symptoms but may occasionally be identified in an asymptomatic child. An understanding of the physiological regulation of plasma calcium is important in understanding the potential cause of hypocalcaemia and its appropriate management. The age of presentation will influence the likely differential diagnosis. We have presented a stepwise approach to the investigation of hypocalcaemia dependent on the circulating serum parathyroid hormone level at the time of presentation. The acute and long-term management of the underlying condition is also reviewed.

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Section: Treatment Of Hypocalcaemiamentioning

confidence: 99%

Investigation and management of hypocalcaemia

Nadar

Shaw

2020

Arch Dis Child

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“…Rest of the studies were excluded because of the following reasons: study done in children;[ 10 ] prospective design or unrelated population[ 11 12 13 25 26 27 28 29 30 31 32 33 34 35 36 ]; duplication of data[ 37 38 ]; comparison of different PTH doses[ 39 40 41 ]; evaluation of cost-effectiveness of PTH therapy[ 42 ]; and use of either pump therapy or continuous subcutaneous PTH infusion. [ 5 43 44 45 46 ]…”

Section: R Esultsmentioning

confidence: 99%

“…[ 4 ] Moreover, when hypoparathyroidism is associated with malabsorptive disorders (autoimmune polyendocrine syndrome type 1), oral supplementations with calcium and vitamin D are less effective. [ 5 ]…”

Section: Introductionmentioning

confidence: 99%

Parathyroid hormone replacement versus oral calcium and active vitamin D supplementation in hypoparathyroidism: A meta-analysis

Palui

Das

Roy

et al. 2020

Indian J Endocr Metab

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Objectives: Chronic hypoparathyroidism is treated conventionally with active vitamin D and high doses of calcium. Recombinant human parathyroid hormone (PTH) replacement is an attractive option for treating patients with hypoparathyroidism since it can replace the physiological action of native PTH. The aim of our study was to perform a comprehensive evaluation of the effects of PTH replacement on calcium homeostasis, bone metabolism, and daily requirement of calcium and active vitamin D. Materials and Methods: Randomized controlled trials done in chronic hypoparathyroid patients were included in this meta-analysis. The PTH group included subjects receiving a subcutaneous injection of either PTH (1-84) or PTH (1-34) with oral calcium and/or active vitamin D. The control group included those receiving oral calcium and active vitamin D with/without subcutaneous placebo injection. The primary outcome of this meta-analysis was to compare serum calcium, 24-h urinary calcium, and severe adverse effects among PTH and control groups. Results: In this meta-analysis, we did not find any difference in serum calcium level between PTH and control groups [mean difference (MD) - 0.01; 95% confidence interval (CI) - 0.09, 0.06; P = 0.71]. Although there was a trend towards low 24-h urinary calcium in the PTH group, the difference was not statistically significant (MD - 1.43; 95% CI - 2.89, 0.03; P = 0.06). The incidence of serious adverse events was also similar in both groups (RR 1.35; 95% CI 0.58, 3.16; P = 0.49). Conclusion: Both PTH and active vitamin D therapies are associated with comparable serum and urine calcium levels with a similar incidence of serious adverse events in patients with chronic hypoparathyroidism.

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“…Winer et al previously demonstrated that synthetic human parathyroid hormone (PTH 1‐34) given by subcutaneous injection or pump effectively maintained serum calcium in the normal range with reduced levels of urine calcium excretion compared to conventional therapy 7‐9 . For many patients with severe hypoparathyroidism, replacement PTH therapy reduced the need for acute emergency IV calcium infusions and improved quality of life 10‐12 . Recombinant human PTH 1‐34 (rhPTH; FORTEO ® ) was approved as a treatment for osteoporosis in 2002 and is used worldwide as an off‐label hormonal replacement therapy in patients with hypoparathyroidism who are refractory to conventional therapy.…”

Section: Introductionmentioning

confidence: 99%

“…Recombinant human PTH 1‐34 (rhPTH; FORTEO ® ) was approved as a treatment for osteoporosis in 2002 and is used worldwide as an off‐label hormonal replacement therapy in patients with hypoparathyroidism who are refractory to conventional therapy. Studies have demonstrated that rhPTH 1‐34 reduced urine calcium excretion and adverse events, and provides stable calcium levels in the normal range 10‐14 . Studies with recombinant human PTH 1‐84 demonstrated its efficacy when added to conventional therapy 15,16 .…”

Section: Introductionmentioning

confidence: 99%

Impact of periprocedural subcutaneous parathyroid hormone on control of hypocalcaemia in APS‐1/APECED patients undergoing invasive procedures

Winer

Schmitt

Ferré

et al. 2020

Clinical Endocrinology

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Context The monogenic disorder autoimmune polyendocrine syndrome type 1 (APS‐1) or autoimmune polyendocrinopathy‐candidiasis‐ectodermal dystrophy (APECED) manifests frequently with hypoparathyroidism, which requires treatment with oral supplementation with calcium and active vitamin D analogs. The majority of APS‐1/APECED patients also suffer from intestinal malabsorption, which complicates the management of hypoparathyroidism and may lead to refractory severe hypocalcaemia. In such situations, reliance on intravenous calcium carries a high risk of nephrocalcinosis and renal damage. Methods Here, we report our experience of periprocedural subcutaneous administration of recombinant human parathyroid hormone (rhPTH 1‐34) in APS‐1/APECED patients. Serum calcium was measured up to five times within the 36‐hour period starting the evening before the scheduled procedure and ending the morning following the procedure. Results Twenty‐seven APS‐1/APECED patients with hypoparathyroidism (aged 4‐67 years) underwent 31 invasive gastrointestinal and/or pulmonary procedures. The patients received an average rhPTH1‐34 dose of 9.6 ± 1.4 µg by subcutaneous injection. 92% of the adults and 54% of children in our cohort had evidence of nephrocalcinosis. Mean calcium levels remained stable and ranged from 2.06 to 2.17 mmol/L with minimal fluctuation. None of our patients experienced periprocedural adverse events connected with hypocalcaemia. Conclusion rhPTH 1‐34 is an alternative to conventional therapy in patients with APS‐1/APECED and hypoparathyroidism undergoing invasive procedures. Subcutaneous PTH1‐34 given directly before and after procedures resulted in well‐controlled serum calcium levels maintained in the low‐normal range and avoided the need for intravenous calcium which may contribute to renal calcifications and tubular damage.

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Continuous Subcutaneous Recombinant Parathyroid Hormone (1–34) Infusion in the Management of Childhood Hypoparathyroidism Associated with Malabsorption

Cited by 25 publications

References 16 publications

Investigation and management of hypocalcaemia

Investigation and management of hypocalcaemia

Parathyroid hormone replacement versus oral calcium and active vitamin D supplementation in hypoparathyroidism: A meta-analysis

Impact of periprocedural subcutaneous parathyroid hormone on control of hypocalcaemia in APS‐1/APECED patients undergoing invasive procedures

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