Archives of disease in childhood. Fetal and neonatal edition · 2014
Severe hypercalcaemia due to subcutaneous fat necrosis: presentation, management and complications.
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Shumer Daniel E, Thaker Vidhu, Taylor George A, Wassner Ari J
Аннотация
OBJECTIVE: Subcutaneous fat necrosis (SCFN) is a rare form of panniculitis in infants that generally occurs following birth trauma, meconium aspiration, or therapeutic cooling. Severe hypercalcaemia occurs in a subset of patients, but data on its presentation, management and outcomes are limited. This report details the clinical course and complications of infants treated for severe hypercalcaemia (peak serum calcium ≥3.0 mmol/L) due to SCFN.
DESIGN: Chart review of all infants with SCFN seen at a single paediatric centre over a 13-year period.
PATIENTS: Seven infants with SCFN developed severe hypercalcaemia, with median peak serum calcium 4.1 mmol/L (range 3.3-5.1).
RESULTS: Severe hypercalcaemia occurred before 6 weeks of age, and was asymptomatic in 3/7 patients (43%). Most patients were treated with intravenous hydration, furosemide, glucocorticoids and low-calcium formula, which restored normocalcaemia in a median of 9 days (range 2-42). Fever developed during treatment in 4/7 infants (57%): two patients had bacterial infections and two had no infectious source identified. Nephrocalcinosis was present in 5/6 patients (83%) who were evaluated by renal ultrasound. Nephrocalcinosis failed to resolve in all cases over a median follow-up of 20 months (range 8-48), but no renal dysfunction was observed. Eosinophilia, which has not been reported previously in SCFN, was present in 6/7 patients (86%).
CONCLUSIONS: In this largest series to date of infants with severe hypercalcaemia due to SCFN, novel findings include the common occurrence of fever and a high incidence of persistent nephrocalcinosis without evidence of adverse renal outcomes.
DESIGN: Chart review of all infants with SCFN seen at a single paediatric centre over a 13-year period.
PATIENTS: Seven infants with SCFN developed severe hypercalcaemia, with median peak serum calcium 4.1 mmol/L (range 3.3-5.1).
RESULTS: Severe hypercalcaemia occurred before 6 weeks of age, and was asymptomatic in 3/7 patients (43%). Most patients were treated with intravenous hydration, furosemide, glucocorticoids and low-calcium formula, which restored normocalcaemia in a median of 9 days (range 2-42). Fever developed during treatment in 4/7 infants (57%): two patients had bacterial infections and two had no infectious source identified. Nephrocalcinosis was present in 5/6 patients (83%) who were evaluated by renal ultrasound. Nephrocalcinosis failed to resolve in all cases over a median follow-up of 20 months (range 8-48), but no renal dysfunction was observed. Eosinophilia, which has not been reported previously in SCFN, was present in 6/7 patients (86%).
CONCLUSIONS: In this largest series to date of infants with severe hypercalcaemia due to SCFN, novel findings include the common occurrence of fever and a high incidence of persistent nephrocalcinosis without evidence of adverse renal outcomes.