Pamidronate: Treatment for Severe Hypercalcemia in Neonatal Subcutaneous Fat Necrosis

Abstract

Background: Subcutaneous fat necrosis (SCFN) of the newborn is an uncommon disorder that occurs in the first weeks of life after foetal distress. It can be complicated by potentially life-threatening hypercalcemia. Treatments of hypercalcemia have included hydration, furosemide and corticosteroids. Only one report has described the use of intravenous bisphosphonates for this condition. We propose that pamidronate could be the first line therapy for severe hypercalcemia in SCFN. Patients and Results: Four newborns presented between 2001 and 2004 with SCFN complicated by severe hypercalcemia. At diagnosis, ionized calcium levels were higher than 1.4 mmol/l and were associated with high urinary calcium/creatinine ratios and high 1,25-dihydroxyvitamin D levels. Despite treatment with IV fluids, low calcium diet and furosemide, calcium levels remained high. The patients were given 3–4 doses (0.25–0.50 mg/kg/dose) of pamidronate. Urinary calcium/creatinine ratios and calcium levels decreased within 48–96 h. 1,25-dihydroxyvitamin D levels normalized with resolution of the skin lesions. No persistent nephrocalcinosis was observed. Conclusion: Pamidronate is effective, well-tolerated in the short-term and obviates the need for prolonged treatment with furosemide and corticosteroids. To prevent nephrocalcinosis, pamidronate might be considered as first line treatment for severe hypercalcemia in SCFN.

Two Cases of Mistaken Polyuria and Nephrocalcinosis in Infants with Glucose-Galactose Malabsorption: A Possible Role of 1,25(OH)₂D₃

“…Although there are over 40 mutations of SLC5A1 that have been linked to GGM [36], only 2 other patients with nephrocalcinosis and GGM have their mutations described in the literature [7, 8]. Therefore, there are insufficient data for genotype-phenotype associations at this point [18, 36]. …”

“…Initially, a renal tubulopathy or hypervitaminosis D was suspected, and to counteract the hypercalcemia and hypercalciuria, the patient received a total dose of 0.5 mg/kg of intravenous bisphosphonate therapy [18]. After administration of the bisphosphonates, the calcemia and calciuria normalized but high levels of 1,25(OH) 2 D 3 (up to 135 pg/mL; normal: 20–62.5) [14] persisted, despite concomitant low PTH levels.…”

Commentary on &lsquo;Pamidronate: Treatment for Severe Hypercalcemia in Neonatal Subcutaneous Fat Necrosis&rsquo; by Alos N. et al., <i>Hormone Research</i> 2006;65:289&ndash;294

“…Alos et al [1] published a case series regarding the use of pamidronate as firstline treatment for hypercalcemia in subcutaneous fat necrosis. Early use of pamidronate seemed to reduce nephrocalsinosis.…”

“…The skin changes were less extensive with no areas of inflammation but some induration remained. Less than 10 hypercalcemic patients with SCFNA have been treated with intravenous pamidronate and reported in the literature [1,2] . Alos et al [1] suggested that pamidronate could be considered as first-line treatment for severe hypercalcemia in SCFN with the aim of reducing nephrocalcinosis.…”

“…Less than 10 hypercalcemic patients with SCFNA have been treated with intravenous pamidronate and reported in the literature [1,2] . Alos et al [1] suggested that pamidronate could be considered as first-line treatment for severe hypercalcemia in SCFN with the aim of reducing nephrocalcinosis. The patient reported by Aucharaz et al [2] was successfully managed with fluids, diuretics and steroid therapy but developed nephrocalcinosis.…”