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CASE REPORT
Year : 2016  |  Volume : 17  |  Issue : 3  |  Page : 199-201

Bronze baby: A rare presentation of biliary atresia


1 Department of Neonatology, Lotus Hospitals for Women and Children, Hyderabad, Telangana, India
2 Division of Pediatric Critical Care and Neonatology, Lotus Hospitals for Women and Children, Lakdikapul, Hyderabad, Telangana, India

Date of Web Publication5-Jul-2016

Correspondence Address:
Rahul Mansing Kadam
Department of Neonatology, Lotus Hospitals for Women and Children, 6-2-29, Lakdikapul
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2319-7250.179480

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  Abstract 

Bronze baby syndrome is a dark brown pigmentation of skin occurring sometimes following phototherapy. No specific treatment is required, as the pigmentation spontaneously disappears after the discontinuation of phototherapy. A 12-day-old neonate with dark brown pigmentation of skin was referred as cyanotic heart disease with the mass in the liver for emergency surgical removal. Infant was found to have conjugated hyperbilirubinemia. Workup was performed for the possible causes. Infant was diagnosed to have extra-hepatic biliary atresia. Dark brown pigmentation should not be interpreted as cyanosis. Always rule out the underlying causative liver disease.

Keywords: Bronze baby syndrome, hepatobiliary iminodiacetic acid scan, phototherapy


How to cite this article:
Kadam RM, Prasad V. Bronze baby: A rare presentation of biliary atresia. Indian J Paediatr Dermatol 2016;17:199-201

How to cite this URL:
Kadam RM, Prasad V. Bronze baby: A rare presentation of biliary atresia. Indian J Paediatr Dermatol [serial online] 2016 [cited 2019 Dec 13];17:199-201. Available from: http://www.ijpd.in/text.asp?2016/17/3/199/179480


  Introduction Top


Bronze baby syndrome is the dark grey-brown pigmentation of skin, mucous membrane, and urine following phototherapy. Hepatic dysfunction has to be there for this condition to be present. We report a neonate with dark brown pigmentation of the skin. Lesions were spread over the abdomen, chest, back, and both upper and lower limb after receiving phototherapy for neonatal jaundice.


  Case Report Top


This term 2.6 kg birth weight, appropriate for gestational age, female neonate was born to a 26-year-old primigravida mother by forceps-assisted vaginal delivery. Infant is a product of consanguineous marriage. Antenatal period was uneventful. Baby did not cry immediately after birth and required resuscitation (details not known). Infant required oxygen supplementation for a day in view of transient tachypnea of newborn (TTNB). Infant was noticed to have yellowish discoloration of life on day 2 of life. Infant's serum bilirubin level was 21 mg/dl. Infant was treated with phototherapy and phenobarbitone for 4 days. Infant's serum bilirubin level was still high (total - 20.4 mg/dl, direct - 6.3 mg/dl) and infant was noticed to have dark discoloration of the entire body. Phototherapy was continued for 2 more days, and two-dimensional echocardiogram (2D Echo) was performed with suspicion of cyanotic heart disease (dark pigmentation being interpreted as cyanosis) which revealed tiny ventricular septal defect (VSD) with cystic mass in liver compressing inferior vena cava (IVC). Parents were counseled for the need of emergency surgery to remove the mass.

The infant was referred to our center on day 12 of life with above-mentioned problems. At admission infant's vitals were stable with pre- and post-ductal saturations being normal.

The infant was noticed to have dark brown pigmentation of the entire body [Figure 1]. There was cephalhematoma in the right parietal region. Infant's systemic examination was unremarkable except for a systolic murmur. Infant's 2D Echo revealed tiny mid-muscular VSD and IVC displaced to midline due to cystic mass below diaphragm. Computed tomography scan of the abdomen was performed which revealed presence of a right adrenal hematoma. Liver and gall bladder were normal.
Figure 1: Clinical photograph at the time of admission depicting generalized dark-brown discoloration of entire body

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Infant was diagnosed to have Bronze baby syndrome and was evaluated for a possible cause. Infant was passing dark colored urine and clay colored stools. Infant's serum bilirubin level was 14.8 mg//dl with direct bilirubin being 7.2 mg/dl. Infant's rest of the liver function tests were normal. Infant's metabolic parameters were normal, and blood sugar levels were within euglycemic range. Infant's urine was negative for reducing substances. Infant's HIDA scan was performed after priming with phenobarbitone for 5 days which was suggestive of extrahepatic biliary atresia [Figure 2].
Figure 2: Hepatobiliary iminodiacetic acid scan depicting no tracer activity in intestine with tracer uniformly taken up by liver suggestive of extra-hepatic biliary atresia

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  Discussion Top


It is assumed that abnormal accumulation of photoisomer of bilirubin is the cause of this condition. The second cause postulated is abnormal hepatic function leading to copper- porphyrin complex which is photo destroyed leading to brown pigmentation. The third explanation is an accumulation of biliverdin leading to pigmentation. It is probably due to a reduction in hepatic excretory function of bilirubin photoproducts. The pigment accumulated in the body may be polymerized and forms bilifuscin-like substances following a free radical reaction. It is concluded that the brown pigment is formed via unknown pigment.[1]

It should be differentiated from gray baby syndrome exclusively seen in neonates and very young infants receiving high doses of chloramphenicol. The infant is cyanosed, acidotic, has cold peripheries and has the signs of all of the marked hypotonia, poor feeding, vomiting, loose stools, and a distended abdomen.

No treatment is required for the bronze baby syndrome as the pigmentation slowly disappears after stopping phototherapy. Three cases have been reported; there was one case each of bile duct atresia, galactosemia, and parenteral nutrition with cholestasis. The bronze color disappears spontaneously, and the prognosis is dependent on the causative liver disease for which investigations should be performed routinely.[2]It is essential to identify the underlying liver disease to determine the prognosis of this syndrome.

Declaration of Patient Consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Onishi S, Itoh S, Isobe K, Togari H, Kitoh H, Nishimura Y. Mechanism of development of bronze baby syndrome in neonates treated with phototherapy. Pediatrics 1982;69:273-6.  Back to cited text no. 1
    
2.
Cuvellier JC, Gottrand F, Largillière C, Farriaux JP. The bronze baby syndrome Apropos of 3 cases. Ann Pediatr (Paris) 1990;37:669-71.  Back to cited text no. 2
    


    Figures

  [Figure 1], [Figure 2]



 

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