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Subcutaneous fat necrosis, hypercalcemia, and prostaglandin E
H Sharata1, D C Postellon, K Hashimoto
1Department of Medicine, University of Wisconsin-Madison 53705, USA.
Insights
Subcutaneous fat necroses (SCFN) in infants are linked to elevated prostaglandin E (PGE) levels. This association was observed with both endogenous and exogenous PGE, suggesting a crucial role for this mediator.
Area of Science:
- Neonatology
- Dermatology
- Endocrinology
Background:
- Subcutaneous fat necrosis (SCFN) is a rare condition affecting newborns, characterized by indurated plaques.
- Endocrine and metabolic derangements can be associated with SCFN, but the underlying mechanisms are not fully understood.
Observation:
- Two neonates presented with SCFN, one with endogenous hypercalcemia and elevated prostaglandin E2 (PGE2) excretion, and another treated with exogenous prostaglandin E1 (PGE1) for congenital heart disease.
- Skin biopsies confirmed panniculitis with necrotic adipocytes and granulomatous infiltrate in both cases.
- The first patient's SCFN and hypercalcemia resolved with systemic steroids and diuretics.
- The second patient's SCFN resolved upon discontinuation of PGE1 infusion, with no new lesions forming.
Findings:
- These cases highlight a significant association between elevated prostaglandin E (PGE) levels and the development of SCFN.
- Endogenous PGE2 elevation was observed in a patient with SCFN and hypercalcemia.
- Exogenous PGE1 administration in a neonate with critical heart disease also preceded the onset of SCFN.
Implications:
- Elevated PGE levels, whether endogenous or exogenous, may play a causal role in the pathogenesis of SCFN.
- This finding suggests that monitoring PGE levels or considering PGE-lowering strategies might be beneficial in managing SCFN.
- The study is the first to report SCFN associated with PGE1 administration, opening new avenues for research into SCFN etiology and treatment.
Abstract:
We present two patients with subcutaneous fat necroses (SCFN) in whom endocrinologic studies revealed an association with elevated prostaglandin E (PGE) levels. A boy born after prolonged labor complicated by meconium aspiration developed erythematous, indurated plaques over the back, arms, buttocks, and cheeks at 4 days of age. A biopsy specimen of involved skin showed panniculitis with foci of necrotic adipocytes containing radially arranged, needle-shaped clefts and a granulomatous infiltrate in the septae. Laboratory studies revealed hypercalcemia of 13.6 mg/dl (normal 8.8-10.1 mg/dl), elevated 1.25-1.25(OH)2D3, and increased urinary excretion of PGE2. The child was hospitalized and treated with systemic steroids and diuretics, with resolution of SCFN and hypercalcemia. The second patient was a girl born with cyanotic heart disease. A diagnosis of Ebstein anomaly was made, and intravenous PGE1 was started to keep patent the ductus arteriosus. Four days later erythematous, indurated plaques were noted on the knee, back, and anterior chest. A skin biopsy specimen revealed SCFN. There was no associated laboratory abnormality. On discontinuing PGE1, no new lesions formed and the existing panniculitis resolved. These two cases demonstrate the association between SCFN and elevated PGE levels (endogenous in patient 1, exogenous in patient 2). No previous reports of SCFN after the administration of PGE1 have appeared in the literature.