Newborn Direct or Conjugated Bilirubin Measurements As a Potential Screen for Biliary Atresia

Harpavat, Sanjiv; Ramraj, Ramya; Finegold, Milton J.; Brandt, Mary L.; Hertel, Paula M.; Fallon, Sara C.; Shepherd, Ross W.; Shneider, Benjamin L.

doi:10.1097/mpg.0000000000001097

Cited by 44 publications

(48 citation statements)

References 19 publications

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“…24 Harpavat et al reported that newborn DB measurements had a high sensitivity and specificity for the screening of BA. 11,12,20,[25][26][27] In our study, using DB of 30 μmol/L as a cutoff to predict BA, the sensitivity was 100%, but the specificity was 52%. The area under the ROC curve of DB, DB/TB, CB MS /TB MS , and CB MS for predicting BA was 0.98, 0.95, 0.73, and 0.57, respectively.…”

Section: Discussionmentioning

confidence: 90%

Can Free Carnitine or Bilirubin in Blood Be Used in Neonatal Screening for Biliary Atresia?

Zheng

Chen

et al. 2019

Eur J Pediatr Surg

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Objective To investigate the efficiency of free carnitine, unconjugated bilirubin (UBIL), bilirubin monoglucuronide (BMG), and bilirubin diglucuronide (BDG) in dry blood spots (DBSs) measured using tandem mass spectrometry (MS/MS) for screening biliary atresia (BA). Materials and Methods All the patients with BA, residing in Shanghai, were collected from four different children's hospitals in Shanghai from January 1, 2015, to June 30, 2017. UBILMS, BMG, BDG, and free carnitine were measured in the DBS samples of 48 patients with BA, 10,008 pediatric patients, and 52,862 newborns using MS/MS. Conjugated bilirubin was measured by MS/MS (CBMS) = BMG + BDG, and total bilirubin was measured by MS/MS (TBMS) = UBILMS + CBMS. Four hundred pediatric patients' direct bilirubin (DB) and total bilirubin (TB), measured by the clinical laboratory and MS/MS, were used as a control. Results The total number of births at the registered permanent residences in Shanghai was 233,000; among them, the occurrence of BA was in 33 patients in 2 years. Therefore, the incidence of BA in Shanghai was 1:7,060. The ratio of DB/TB and CBMS/TBMS of most patients with BA was elevated gradually in the neonatal period and higher than the normal range after 1 month after birth. The area under the receiver operating characteristic curve of DB, DB/TB, CBMS/TBMS, CBMS, and free carnitine for predicting BA was 0.98, 0.95, 0.73, 0.57, and 0.92, respectively. Using the 95% percentile as a cutoff, the sensitivity of DB and free carnitine to predict BA was 100 and 85%, respectively, and the specificity was 52 and 85%, respectively. Conclusion In free carnitine, DB, and CBMS/TBMS tests, blood concentrations are elevated in all infants with BA. However, they may not be elevated while they are newborns. These tests will result in high false negatives or positives. Thus, they should not be used as newborn screening tests for BA due to their lower sensitivity and specificity.

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Section: Discussionmentioning

confidence: 90%

Can Free Carnitine or Bilirubin in Blood Be Used in Neonatal Screening for Biliary Atresia?

Zheng

Chen

et al. 2019

Eur J Pediatr Surg

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“…Identification of cholestatic infants at this point at the latest should leave enough time for completion of a well-organized diagnostic protocol and timely performance of PE before the age of 2 months. General awareness of newborn cholestasis and the practice of routine measurement of serum conjugated bilirubin in all jaundiced newborns older than 2 weeks could be endorsed by active education [27][28][29]. Stool color card screening has improved timing of PE, clearance of jaundice and native liver survival in Taiwan and Japan, where BA incidence is considerably higher than in Europe [30,31].…”

Section: Discussionmentioning

confidence: 99%

Outcomes of biliary atresia in the Nordic countries – a multicenter study of 158 patients during 2005–2016

Pakarinen

Johansen

Svensson

et al. 2018

Journal of Pediatric Surgery

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“…This review summarizes these various theories and focuses on recent studies of pathogenesis. The compelling finding of mild direct hyperbilirubinemia at birth in infants who eventually went on to have BA begs the question as to whether BA is initiated in-utero [16]. …”

Section: Theories Of Pathogenesismentioning

confidence: 99%

Update on investigations pertaining to the pathogenesis of biliary atresia

Kilgore

Mack

2017

Pediatr Surg Int

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Biliary atresia is a devastating biliary disease of neonates that results in liver transplantation for the vast majority. The etiology of biliary atresia is unknown and is likely multifactorial, with components of genetic predisposition, environmental trigger and autoimmunity contributing to disease pathogenesis. This review highlights recent work related to investigations of disease pathogenesis in biliary atresia.

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Newborn Direct or Conjugated Bilirubin Measurements As a Potential Screen for Biliary Atresia

Cited by 44 publications

References 19 publications

Can Free Carnitine or Bilirubin in Blood Be Used in Neonatal Screening for Biliary Atresia?

Can Free Carnitine or Bilirubin in Blood Be Used in Neonatal Screening for Biliary Atresia?

Outcomes of biliary atresia in the Nordic countries – a multicenter study of 158 patients during 2005–2016

Update on investigations pertaining to the pathogenesis of biliary atresia

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