Bronze Baby Syndrome

      Keywords

      A male infant born at 37 weeks of gestation and weighing 2935 g was delivered by induced vaginal delivery to a 20-year-old multiparous mother after a pregnancy complicated by Rhesus isoimmunization. His serum total bilirubin was 10.6 mg/dL at 6 hours of age and 14.4 mg/dL at 10 hours of age. The family history was significant for a sibling who was also Coombs positive with hemolytic jaundice requiring several days of phototherapy. The infant was placed under a multiple-bank LED phototherapy device and treated with intravenous fluids and immune globulin to avoid exchange transfusion. Total serum bilirubin levels peaked at 15.5 mg/dL on day 3. From days 3 to 5 of age, progressive bronze-gray discoloration of the skin was noted (Figure). Liver function tests and methemoglobin levels were normal. Direct bilirubin levels were elevated, but remained less than 2.2 mg/dL. Phototherapy was maintained until 8 days of age. He was discharged 12 days after birth with improving jaundice. Follow-up examinations showed resolution of skin discoloration over 3-4 weeks.
      Figure
      FigureA normally-pigmented Caucasian newborn infant experienced progressive but self-limited bronze-gray discoloration following initiation of phototherapy. Skin discoloration peaked on days 5-8. Phototherapy was continued until serum bilirubin levels were reassuring. Bronzing improved over the ensuing weeks and skin color was similar to his siblings at 5 months of age.
      Bronze baby syndrome (BBS) is an unusual neonatal dyschromia that occurs during phototherapy in some neonates with elevated serum levels of conjugated bilirubin. Hyperpigmentation is considered a justification for early discontinuation of phototherapy. However, phototherapy-induced bronzing is a self-limited process, and continuance of phototherapy may be preferable to the risks of exchange transfusion. BBS was first described by Kopelman et al
      • Kopelman A.E.
      • Brown R.S.
      • Odell G.B.
      The “bronze” baby syndrome: a complication of phototherapy.
      in 1972, soon after the emergence of phototherapy as a treatment for neonatal jaundice. Bronze pigmentation can also occur in various abdominal organs and body fluids, but there is controversy over the risks for kernicterus and staining of the central nervous system.
      • Clark C.F.
      • Torii S.
      • Hamamoto Y.
      • Kaito H.
      The “bronze baby” syndrome: postmortem data.
      • Tan K.L.
      • Jacob E.
      The bronze baby syndrome.
      • Bertini G.
      • Dani C.
      • Fonda C.
      • Zorzi C.
      • Rubaltelli F.F.
      Bronze baby syndrome and the risk of kernicterus.
      BBS has been attributed to increased accumulation of bilirubin photoisomers, metabolic precursors or degradation products, or copper-porphyrin conjugates.
      • Purcell S.M.
      • Wians Jr, F.H.
      • Ackerman Jr, N.B.
      • Davis B.M.
      Hyperbiliverdinemia in the bronze baby syndrome.
      • Rubaltelli F.F.
      • Da Riol R.
      • D'Amore E.S.
      • Jori G.
      The bronze baby syndrome: evidence of increased tissue concentration of copper porphyrins.
      The true mechanistic cause remains uncertain.
      • McDonagh A.F.
      Bilirubin, copper-porphyrins, and the bronze-baby syndrome.
      Liver disease should be ruled out in infants who develop BBS, because hepatocellular disease may be present in these patients with conjugated hyperbilirubinemia.
      • Ottinger D.
      Bronze baby syndrome.
      Collectively, these findings suggest that early recognition of BBS can guide further evaluation of hematologic and other disorders, but should not prevent the use of phototherapy for hyperbilirubinemia in infants with elevated levels of conjugated bilirubin.

      References

        • Kopelman A.E.
        • Brown R.S.
        • Odell G.B.
        The “bronze” baby syndrome: a complication of phototherapy.
        J Pediatr. 1972; 81: 466-472
        • Clark C.F.
        • Torii S.
        • Hamamoto Y.
        • Kaito H.
        The “bronze baby” syndrome: postmortem data.
        J Pediatr. 1976; 88: 461-464
        • Tan K.L.
        • Jacob E.
        The bronze baby syndrome.
        Acta Paediatr Scand. 1982; 71: 409-414
        • Bertini G.
        • Dani C.
        • Fonda C.
        • Zorzi C.
        • Rubaltelli F.F.
        Bronze baby syndrome and the risk of kernicterus.
        Acta Paediatr. 2005; 94: 968-971
        • Purcell S.M.
        • Wians Jr, F.H.
        • Ackerman Jr, N.B.
        • Davis B.M.
        Hyperbiliverdinemia in the bronze baby syndrome.
        J Am Acad Dermatol. 1987; 16: 172-177
        • Rubaltelli F.F.
        • Da Riol R.
        • D'Amore E.S.
        • Jori G.
        The bronze baby syndrome: evidence of increased tissue concentration of copper porphyrins.
        Acta Paediatr. 1996; 85: 381-384
        • McDonagh A.F.
        Bilirubin, copper-porphyrins, and the bronze-baby syndrome.
        J Pediatr. 2011; 158: 160-164
        • Ottinger D.
        Bronze baby syndrome.
        Neonatal Netw. 2013; 32: 200-202