Pamidronate as first-line treatment of hypercalcemia in neonatal subcutaneous fat necrosis: A case series.

Hypercalcemia Nephrocalcinosis Pamidronate

Journal

Paediatrics & child health
ISSN: 1205-7088
Titre abrégé: Paediatr Child Health
Pays: England
ID NLM: 9815960

Informations de publication

Date de publication:
Feb 2021
Historique:
received: 01 05 2019
accepted: 12 09 2019
entrez: 5 2 2021
pubmed: 24 12 2019
medline: 24 12 2019
Statut: epublish

Résumé

Subcutaneous fat necrosis (SCFN) can be complicated by severe hypercalcemia, which is frequently asymptomatic. Nephrocalcinosis is associated with hypercalcemia and, in other clinical settings, has been linked to furosemide and glucocorticoid use. First-line bisphosphonate therapy treating hypercalcemia in neonatal SCFN is not well described. To describe the biochemical changes and risk of nephrocalcinosis in infants with hypercalcemia, secondary to neonatal SCFN, treated with initial pamidronate. A retrospective chart review of five infants treated with initial pamidronate and without furosemide or glucocorticoids. Data were collected on the following: timing of presentation, therapeutic response, and presence of nephrocalcinosis. Hypercalcemia resolved after 2.8±1.7 days; this is compared to 7.6±2.8 days from previously reported cases utilising alternative therapies (P=0.012). There were no episodes of rebound hypercalcemia or hypocalcemia. Nephrocalcinosis was present in four of five cases. When including published cases, age at diagnosis was associated with presenting serum calcium (P=0.003) and nephrocalcinosis was associated with higher serum calcium (P=0.014) and time from SCFN to hypercalcemia diagnosis (P=0.002). This retrospective case series demonstrates that first-line pamidronate treatment was effective and safe in the resolution of hypercalcemia. Nephrocalcinosis was observed, despite the avoidance of furosemide and glucocorticoid therapy, and associated with greater disease severity and duration of hypercalcemia.

Sections du résumé

BACKGROUND BACKGROUND
Subcutaneous fat necrosis (SCFN) can be complicated by severe hypercalcemia, which is frequently asymptomatic. Nephrocalcinosis is associated with hypercalcemia and, in other clinical settings, has been linked to furosemide and glucocorticoid use. First-line bisphosphonate therapy treating hypercalcemia in neonatal SCFN is not well described.
OBJECTIVES OBJECTIVE
To describe the biochemical changes and risk of nephrocalcinosis in infants with hypercalcemia, secondary to neonatal SCFN, treated with initial pamidronate.
METHODS METHODS
A retrospective chart review of five infants treated with initial pamidronate and without furosemide or glucocorticoids. Data were collected on the following: timing of presentation, therapeutic response, and presence of nephrocalcinosis.
RESULTS RESULTS
Hypercalcemia resolved after 2.8±1.7 days; this is compared to 7.6±2.8 days from previously reported cases utilising alternative therapies (P=0.012). There were no episodes of rebound hypercalcemia or hypocalcemia. Nephrocalcinosis was present in four of five cases. When including published cases, age at diagnosis was associated with presenting serum calcium (P=0.003) and nephrocalcinosis was associated with higher serum calcium (P=0.014) and time from SCFN to hypercalcemia diagnosis (P=0.002).
CONCLUSIONS CONCLUSIONS
This retrospective case series demonstrates that first-line pamidronate treatment was effective and safe in the resolution of hypercalcemia. Nephrocalcinosis was observed, despite the avoidance of furosemide and glucocorticoid therapy, and associated with greater disease severity and duration of hypercalcemia.

Identifiants

pubmed: 33542779
doi: 10.1093/pch/pxz141
pii: pxz141
pmc: PMC7850270
doi:

Types de publication

Journal Article

Langues

eng

Pagination

e52-e56

Informations de copyright

© Crown copyright 2019.

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Auteurs

Alexander D Chesover (AD)

Division of Endocrinology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario.

Jennifer Harrington (J)

Division of Endocrinology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario.

Farid H Mahmud (FH)

Division of Endocrinology, The Hospital for Sick Children, University of Toronto, Toronto, Ontario.

Classifications MeSH