Continuous Subcutaneous Recombinant Parathyroid Hormone (1-34) Infusion in the management of childhood Hypoparathyroidism associated with Malabsorption

Vrinda Saraff, Anya Rothenbuhler, Wolfgang Högler, Agnes Linglart

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10 Citations (Scopus)
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Abstract

Background/ Aims: Hypoparathyroidism associated with malabsorption can be particularly challenging to manage due to limited and erratic intestinal absorption of calcium and vitamin D analogues, resulting in episodes of hypo- or hypercalcaemia. We evaluated the role of continuous subcutaneous recombinant parathyroid (rhPTH 1-34) hormone infusion (CSPI) in children with hypoparathyroidism associated with intestinal malabsorption resistant to conventional therapy.
Method: Four patients (8 - 13 years), with symptomatic hypocalcaemia resistant to conventional therapy were started on CSPI (follow up 3-8 years), in two paediatric endocrinology units in Europe.
Results: Serum calcium normalised within 48 hours of commencing treatment in all 4 patients. An average rhPTH 1-34 dose of 0.4 µg/kg/day resulted in a substantial reduction in symptomatic hypocalcaemia and hypo/ hypercalcaemia-related hospital admissions. An increased alkaline phosphatase activity was noted in the first six months on CSPI, indicating increase in bone turnover. In 2 patients with elevated urinary calcium excretion pre CSPI, this normalised in the first year on treatment. No significant side effects were noticed in the short or long term, with patient-reported preference of CSPI over conventional treatment.
Conclusion: CSPI is a promising and effective treatment option for managing hypocalcaemia and hyperphosphatemia in children with hypoparathyroidism associated with intestinal malabsorption.
Original languageEnglish
JournalHormone Research in Paediatrics
Early online date19 Sep 2017
DOIs
Publication statusE-pub ahead of print - 19 Sep 2017

Keywords

  • Hypoparathyroidism
  • malabsorption
  • recombinant parathyroid hormone
  • teriparatide
  • continuous subcutaneous infusion

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