Contribution of genetic factors to high rates of neonatal hyperbilirubinaemia on the Thailand-Myanmar border
Open accessAbstract
Very high unconjugated bilirubin plasma concentrations in neonates (neonatal hyperbilirubinaemia; NH) may cause neurologic damage (kernicterus). Both increased red blood cell turn-over and immaturity of hepatic glucuronidation contribute to neonatal hyperbilirubinaemia. The incidence of NH requiring phototherapy during the first week of life on the Thailand-Myanmar border is high (approximately 25%). On the Thailand-Myanmar border we investigated the contribution of genetic risk factors to high bilirubin levels in the first month of life in 1596 neonates enrolled in a prospective observational birth cohort study. Lower gestational age (<38 weeks), mutations in the genes encoding glucose-6-phosphate dehydrogenase (G6PD) and uridine 5′-diphospho-glucuronosyltransferase (UGT) 1A1 were identified as the main independent risk factors for NH in the first week, and for prolonged jaundice in the first month of life. Population attributable risks (PAR%) were 61.7% for lower gestational age, 22.9% for hemi or homozygous and 9.9% for heterozygous G6PD deficiency respectively, and 6.3% for UGT1A1*6 homozygosity. In neonates with an estimated gestational age ≥ 38 weeks, G6PD mutations contributed PARs of 38.1% and 23.6% for “early” (≤ 48 hours) and “late” (49–168 hours) NH respectively. For late NH, the PAR for UGT1A1*6 homozygosity was 7.7%. Maternal excess weight was also a significant risk factor for “early” NH while maternal mutations on the beta-globin gene, prolonged rupture of membranes, large haematomas and neonatal sepsis were risk factors for “late” NH. For prolonged jaundice during the first month of life, G6PD mutations and UGT1A1*6 mutation, together with lower gestational age at birth and presence of haematoma were significant risk factors. In this population, genetic factors contribute considerably to the high risk of NH. Diagnostic tools to identify G6PD deficiency at birth would facilitate early recognition of high risk cases.
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Integrity signals
Registry
Cites
Statistics
Claims the paper makes
The claims and statistics of this paper haven’t been checked yet.
Run the check — free, no account needed. It reads the open-access text and checks the sources it cites.
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
Newborns from the Karen ethnic group are at increased risk of neonatal hyperbilirubinemia.
“Newborns from the Karen ethnic group are at increased risk of neonatal hyperbilirubinemia (Bancone et al., 2022; Thielemans et al., 2021).”
From Mother, child and adolescent health outcomes in two long-term refugee camp settings at the Thai-Myanmar border 2000–2018: a retrospective analysis (2024) · cites this paper as Bancone et al., 2022
Self-check
Percentages, sums and reported differences re-computed from the paper’s own numbers.
The self-check needs the paper’s full text.
Cites
(2004) · The Lancet · doi:10.1016/s0140-6736(03)15268-3 · Cited by 12,717 · see citing works
Management of Hyperbilirubinemia in the Newborn Infant 35 or More Weeks of Gestation
Hyperbilirubinemia, S. O. (2004) · PEDIATRICS · doi:10.1542/peds.114.1.297 · Cited by 2,835 · see citing works
Glucose-6-phosphate dehydrogenase deficiency
Cappellini, M., & Fiorelli, G. (2008) · The Lancet · doi:10.1016/s0140-6736(08)60073-2 · Cited by 1,590 · see citing works
Human UDP-Glucuronosyltransferases: Metabolism, Expression, and Disease
Tukey, R. H., & Strassburg, C. P. (2000) · The Annual Review of Pharmacology and Toxicology · doi:10.1146/annurev.pharmtox.40.1.581 · Cited by 1,525 · see citing works
Bosma, P. J., Chowdhury, J. R., Bakker, C., Gantla, S., Boer, A. M. T., Oostra, B. A., Lindhout, D., Tytgat, G. N. J., Jansen, P. L. M., Elferink, R. P. J. O., et al. (1995) · New England Journal of Medicine · doi:10.1056/nejm199511023331802 · Cited by 1,439 · see citing works
Beutler, E., Gelbart, T., & Demina, A. (1998) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.95.14.8170 · Cited by 854 · see citing works
Neonatal hyperbilirubinaemia: a global perspective
Olusanya, B. O., Kaplan, M., & Hansen, T. W. R. (2018) · The Lancet Child & Adolescent Health · doi:10.1016/s2352-4642(18)30139-1 · Cited by 353 · see citing works
Risk Factors for Severe Hyperbilirubinemia in Neonates
Huang, M., Kua, K.-E., Teng, H., Tang, K.-S., Weng, H.-W., & Huang, C. (2004) · Pediatric Research · doi:10.1203/01.pdr.0000141846.37253.af · Cited by 240 · see citing works
Burden of severe neonatal jaundice: a systematic review and meta-analysis
Slusher, T. M., Zamora, T., Appiah, D., Stanke, J. U., Strand, M. A., Lee, B. W., Richardson, S. B., Keating, E. M., Siddappa, A. M., & Olusanya, B. O. (2017) · BMJ Paediatrics Open · doi:10.1136/bmjpo-2017-000105 · Cited by 239 · see citing works
Glucose-6-Phosphate Dehydrogenase: Update and Analysis of New Mutations around the World
Gómez‐Manzo, S., Marcial‐Quino, J., Vanoye–Carlo, A., Serrano‐Posada, H., Ortega-Cuéllar, D., González‐Valdez, A., Castillo‐Rodríguez, R. A., Hernández‐Ochoa, B., Sierra-Palacios, E. D. C., Rodríguez-Bustamante, E., et al. (2016) · International Journal of Molecular Sciences · doi:10.3390/ijms17122069 · Cited by 222 · see citing works
Olusanya, B. O., Osibanjo, F., & Slusher, T. M. (2015) · PLoS ONE · doi:10.1371/journal.pone.0117229 · Cited by 175 · see citing works
Association of Neonatal Hyperbilirubinemia With Bilirubin UDP-Glucuronosyltransferase Polymorphism
Maruo, Y., Kashiro, N., Satō, H., Doida, Y., & Shimada, M. (1999) · PEDIATRICS · doi:10.1542/peds.103.6.1224 · Cited by 169 · see citing works
Why population attributable fractions can sum to more than one
Rowe, A. K., Powell, K. E., & Flanders, W. D. (2004) · American Journal of Preventive Medicine · doi:10.1016/j.amepre.2003.12.007 · Cited by 126 · see citing works
Hyperbilirubinemia in the Newborn
Lauer, B., & Spector, N. D. (2011) · Pediatrics in Review · doi:10.1542/pir.32-8-341 · Cited by 103 · see citing works
Characterization of G6PD Genotypes and Phenotypes on the Northwestern Thailand-Myanmar Border
Bancone, G., Chu, C. S., Somsakchaicharoen, R., Chowwiwat, N., Parker, D. M., Charunwatthana, P., White, N. J., & Nosten, F. H. (2014) · PLoS ONE · doi:10.1371/journal.pone.0116063 · Cited by 99 · see citing works
Self-citationAkaba, K., Kimura, T., Sasaki, A., Tanabe, S., Wakabayashi, T., Hiroi, M., Yasumura, S., Maki, K., Aikawa, S., & Hayasaka, K. (1999) · Journal of Human Genetics · doi:10.1007/s100380050100 · Cited by 96 · see citing works
UGT1 Promoter Polymorphism Accounts for Increased Neonatal Appearance of Hereditary Spherocytosis
Iolascon, A., Faienza, M. F., Moretti, A., Perrotta, S., & del Giudice, E. M. (1998) · Blood · doi:10.1182/blood.v91.3.1093 · Cited by 82 · see citing works
Relationship Between Bilirubin UDP-Glucuronosyl Transferase 1A1 Gene and Neonatal Hyperbilirubinemia
Huang, C., Chang, P., Huang, M., Chen, E., Hung, K., & Tsou, K.-I. (2002) · Pediatric Research · doi:10.1203/00006450-200210000-00022 · Cited by 77 · see citing works
Review of bilirubin neurotoxicity I: molecular biology and neuropathology of disease
Riordan, S. M., & Shapiro, S. M. (2019) · Pediatric Research · doi:10.1038/s41390-019-0608-0 · Cited by 75 · see citing works
Studies of prolonged neonatal jaundice in the breast-fed infant
Gartner, L. M., & Arias, I. M. (1966) · The Journal of Pediatrics · doi:10.1016/s0022-3476(66)80422-5 · Cited by 72 · see citing works
Kaplan, M., Hammerman, C., Vreman, H. J., Stevenson, D. K., & Beutler, E. (2001) · The Journal of Pediatrics · doi:10.1067/mpd.2001.115312 · Cited by 67 · see citing works
Bancone, G., Ménard, D., Khim, N., Kim, S., Canier, L., Nguong, C., Phommasone, K., Mayxay, M., Dittrich, S., Vongsouvath, M., et al. (2019) · Malaria Journal · doi:10.1186/s12936-019-2652-y · Cited by 65 · see citing works
Self-citationBancone, G., Gornsawun, G., Chu, C. S., Porn, P., Pal, S., Bansil, P., Domingo, G. J., & Nosten, F. H. (2018) · PLoS ONE · doi:10.1371/journal.pone.0196716 · Cited by 59 · see citing works
Self-citationMaruo, Y., Morioka, Y., Fujito, H., Nakahara, S., Yanagi, T., Matsui, K., Mori, A., Sato, H., Tukey, R. H., & Takeuchi, Y. (2014) · The Journal of Pediatrics · doi:10.1016/j.jpeds.2014.01.060 · Cited by 54 · see citing works
Neonatal Intensive Care in a Karen Refugee Camp: A 4 Year Descriptive Study
Turner, C., Carrara, V., Aye Mya Thein, N., Chit Mo Mo Win, N., Turner, P., Bancone, G., White, N. J., McGready, R., & Nosten, F. (2013) · PLoS ONE · doi:10.1371/journal.pone.0072721 · Cited by 52 · see citing works
Self-citationThielemans, L., Gornsawun, G., Hanboonkunupakarn, B., Paw, M. K., Porn, P., Moo, P. K., Overmeire, B. V., Proux, S., Nosten, F. H., McGready, R. M., et al. (2018) · Wellcome Open Research · doi:10.12688/wellcomeopenres.13373.1 · Cited by 49 · see citing works
Self-citationYamamoto, K., Soeda, Y., Kamisako, T., Hosaka, H., Fukano, M., Satō, H., Fujiyama, Y., Adachi, Y., Satoh, Y., & Bamba, T. (1998) · Journal of Human Genetics · doi:10.1007/s100380050050 · Cited by 48 · see citing works
Steinbrook, E., Min, M. C., Kajeechiwa, L., Wiladphaingern, J., Paw, M. K., Pimanpanarak, M. P. J., Hiranloetthanyakit, W., Min, A. M., Tun, N. W., Gilder, M. E., et al. (2021) · BMC Pregnancy and Childbirth · doi:10.1186/s12884-021-04276-5 · Cited by 47 · see citing works
Self-citationSato, H., Uchida, T., Toyota, K., Kanno, M., Hashimoto, T., Watanabe, M., Nakamura, T., Tamiya, G., Aoki, K., & Hayasaka, K. (2012) · Journal of Human Genetics · doi:10.1038/jhg.2012.116 · Cited by 43 · see citing works
Hyperbilirubinemia in the Newborn
Lauer, B., & Spector, N. D. (2011) · Pediatrics in Review · doi:10.1542/pir.32.8.341 · Cited by 43 · see citing works
The Influence of Infant and Maternal Sickle Cell Disease on Birth Outcome and Neonatal Course
Brown, A. K. (1994) · Archives of Pediatrics and Adolescent Medicine · doi:10.1001/archpedi.1994.02170110042007 · Cited by 31 · see citing works
Neonatal hyperbilirubinaemia in heterozygous giucose‐6‐phosphate dehydrogenase deficient females
Meloni, T., Forteleoni, G., Dore, A., & Cutillo, S. (1983) · British Journal of Haematology · doi:10.1111/j.1365-2141.1983.tb02017.x · Cited by 31 · see citing works
Genetic polymorphisms in Thai neonates with hyperbilirubinemia
Prachukthum, S., Nuntnarumit, P., Pienvichit, P., Chuansumrit, A., Songdej, D., Kajanachumpol, S., Pakakasama, S., & Hongeng, S. (2009) · Acta Paediatrica · doi:10.1111/j.1651-2227.2009.01275.x · Cited by 29 · see citing works
Association of Neonatal Hyperbilirubinemia with UGT1A1 Gene Polymorphisms: A Meta-Analysis
Yu, Z., Zhu, K., Wang, L., Liu, Y., & Sun, J. (2015) · Medical Science Monitor · doi:10.12659/msm.894043 · Cited by 28 · see citing works
Zhou, J., Yang, C., Zhu, W., Chen, S., Zeng, Y., Wang, J., Zhao, H., Chen, Y., & Lin, F. (2018) · BioMed Research International · doi:10.1155/2018/7803175 · Cited by 28 · see citing works
Bancone, G., Gilder, M. E., Chowwiwat, N., Gornsawun, G., Win, E., Cho, W. W., Moo, E., Min, A. M., Charunwatthana, P., Carrara, V. I., et al. (2017) · Wellcome Open Research · doi:10.12688/wellcomeopenres.12338.2 · Cited by 28 · see citing works
Self-citationWang, J., Yin, J., Xue, M., Lyu, J., & Wan, Y. (2020) · Gene · doi:10.1016/j.gene.2020.144409 · Cited by 23 · see citing works
Kuesap, J., & Na‐Bangchang, K. (2018) · Korean Journal of Parasitology · doi:10.3347/kjp.2018.56.2.167 · Cited by 21 · see citing works
Nguyen, T., Zhao, W., Yang, X., & Zhong, D.-N. (2020) · Pediatric Research · doi:10.1038/s41390-020-0825-6 · Cited by 21 · see citing works
Thielemans, L., Peerawaranun, P., Mukaka, M., Paw, M. K., Wiladphaingern, J., Landier, J., Bancone, G., Proux, S., Elsinga, H., Trip-Hoving, M., et al. (2021) · PLOS ONE · doi:10.1371/journal.pone.0258127 · Cited by 20 · see citing works
Self-citationRodie, M., Barclay, A. R., Harry, C., & Simpson, J. (2010) · Archives of Disease in Childhood · doi:10.1136/adc.2010.199984 · Cited by 17 · see citing works
Steadman, S., Ahmed, I., McGarry, K., & Rasiah, S. V. (2016) · Archives of Disease in Childhood · doi:10.1136/archdischild-2015-309265 · Cited by 15 · see citing works
Carvalho, O. M. C., Augusto, M. C. C., Medeiros, M. Q., Lima, H. M. P., Viana, A. B., Júnior, E. A., & Carvalho, F. H. C. (2018) · The Journal of Maternal-Fetal & Neonatal Medicine · doi:10.1080/14767058.2018.1473367 · Cited by 15 · see citing works
Min, A. M., Saito, M., Simpson, J. A., Kennedy, S., Nosten, F. H., & McGready, R. M. (2021) · PLoS ONE · doi:10.1371/journal.pone.0255902 · Cited by 14 · see citing works
Self-citationThe role of UGT1A1 (c.-3279 T > G) gene polymorphisms in neonatal hyperbilirubinemia susceptibility
Li, Z., Li, S., & Hao, L. (2020) · BMC Medical Genetics · doi:10.1186/s12881-020-01155-2 · Cited by 13 · see citing works
Molou, E., Schulpis, K. H., Thodi, G., Georgiou, V., Dotsikas, Y., Papadopoulos, K., Biti, S., & Loukas, Y. L. (2014) · Scandinavian Journal of Clinical and Laboratory Investigation · doi:10.3109/00365513.2013.879733 · Cited by 12 · see citing works
Thielemans, L., Trip-Hoving, M., Bancone, G., Turner, C. L., Simpson, J. A., Hanboonkunupakarn, B., van Hensbroek, M. B., van Rheenen, P., Paw, M. K., Nosten, F. H., et al. (2017) · BMC Pediatrics · doi:10.1186/s12887-017-0798-8 · Cited by 12 · see citing works
Self-citationRougée, L. R. A., Miyagi, S. J., & Collier, A. C. (2016) · Europe PMC (PubMed Central) · Cited by 12 · see citing works
Shwe, S., Boo, N. Y., Ong, H. K., Chee, S.-C., Maslina, M., Ling, M. M. M., & Ahluwalia, A. K. (2020) · PubMed · Cited by 4 · see citing works
alpha-Thalassaemia and hyperbilirubinaemia in G-6-PD-deficient newborns.
Meloni, T., Corti, R., Costa, S., Mele, G., & França, V. G. (1980) · Archives of Disease in Childhood · doi:10.1136/adc.55.6.482 · Cited by 4 · see citing works
Flagged first, then by citations
1 of the 51 references couldn’t be matched to a known paper.
Cited by
Sorted by citations
Bancone, G., Gilder, M. E., Win, E., Gornsawun, G., Penpitchaporn, P., Moo, P. K., Archasuksan, L., Wai, N. S., Win, S., Aung, K. K., et al. (2022) · BMJ Open · doi:10.1136/bmjopen-2022-066529 · Cited by 18 · see citing works
Self-citationA metabolite-based resistance mechanism against malaria
Figueiredo, A. L., Rastogi, S., Ramos, S., Nogueira, F., de Villiers, K. A., Sousa, A. G. G., Votborg-Novél, L., von Wedel, C., Tober‐Lau, P., Jentho, E., et al. (2025) · Science · doi:10.1126/science.adq6741 · Cited by 13 · see citing works
Consolidated Newborn Bloodspot Screening Efforts in Developing Countries in the Asia Pacific—2024
Therrell, B. L., Padilla, C. D., Abadingo, M. E., Adhikari, S. P., Aung, T., Aye, T. T., Dey, S. K., Faizi, M., Ganbaatar, E., Giang, T. T., et al. (2024) · International Journal of Neonatal Screening · doi:10.3390/ijns11010002 · Cited by 9 · see citing works
Huang, D., Gu, X., Li, W., Mi, H., Zeng, H., Zhuang, G., Li, S., Shi, C., Wei, T., Zhou, W., et al. (2025) · Journal of genetics and genomics/Journal of Genetics and Genomics · doi:10.1016/j.jgg.2025.09.002 · Cited by 5 · see citing works
Hashmi, A., Darakamon, M. C., Aung, K. K., Mu, M., Misa, P., Jittamala, P., Chu, C., Phyo, A. P., Turner, C., Nosten, F., et al. (2023) · Frontiers in Public Health · doi:10.3389/fpubh.2023.1144642 · Cited by 4 · see citing works
Self-citationHung, Y., Chang, P.-F., & Huang, C. (2024) · Pediatrics & Neonatology · doi:10.1016/j.pedneo.2024.02.003 · Cited by 4 · see citing works
Benner, M. T., Mohr, O., Kaloy, W., Sansoenboon, A., Moungsookjarean, A., Kaiser, P., Carrara, V. I., & McGready, R. (2024) · Primary Health Care Research & Development · doi:10.1017/s146342362400015x · Cited by 3 · see citing works
Self-citationVongbhavit, K., Rearkyai, S., Janyoungsak, P., & Panburana, J. (2025) · Neonatology · doi:10.1159/000547630 · Cited by 3 · see citing works
Bancone, G., Gilder, M. E., Win, E., Gornsawun, G., Moo, P. K., Archasuksan, L., Wai, N. S., Win, S., Hanboonkunupakarn, B., Nosten, F. H., et al. (2024) · BMJ Paediatrics Open · doi:10.1136/bmjpo-2024-002754 · Cited by 3 · see citing works
Self-citationBancone, G., Gilder, M. E., Win, E., Gornsawun, G., Penpitchaporn, P., Moo, P. K., Archasuksan, L., Wai, N. S., Win, S., Aung, K. K., et al. (2022) · medRxiv · doi:10.1101/2022.07.03.22277173 · Cited by 3 · see citing works
Self-citationSaito, M., Gilder, M. E., Carrara, V. I., Archusuksan, L., Eh, H., Eh, N., Ner, M., Win, A. K., Niwetphongprai, L., Koesukwiwat, U., et al. (2025) · Clinical Infectious Diseases · doi:10.1093/cid/ciaf552 · Cited by 2 · see citing works
Self-citationCohort Profile: the SMRU Refugee and Migrant Pregnancy Study in Western Thailand and Eastern Myanmar
McGready, R., J White, N., Nosten, F. H., & Collaborators, S. R. A. M. P. S. (2026) · Wellcome Open Research · doi:10.12688/wellcomeopenres.25582.2 · Cited by 2 · see citing works
Self-citationAwareness and Management of Neonatal Hyperbilirubinemia in Rural Omdurman City Outskirts, SudanPreprint
Mohammed, M. A., Suliman, H. M., Osman, B. I., & Abdoon, I. H. (2024) · SSRN Electronic Journal · doi:10.2139/ssrn.4791186
Use of the “STANDARD G6PDTM” quantitative point-of-care test in neonates and infants
Gornsawun, G., Moo, E., Htoo, K., Chalermvisutkul, S., Gilder, M. E., Moo, P. K., Archusuksan, L., Prins, T. J., Hanboonkunupakarn, B., McGready, R., et al. (2026) · PLoS ONE · doi:10.1371/journal.pone.0346837
Self-citation
- Authors
- Bancone, G., Gornsawun, G., Peerawaranun, P., Penpitchaporn, P., Paw, M. K., Poe, D. D., Win, D., Cicelia, N., Mukaka, M., Archasuksan, L., Thielemans, L., Nosten, F., White, N. J., McGready, R., & Carrara, V. I.
- Affiliations
- Mahidol University (TH)
- Université Libre de Bruxelles (BE)
- University of Geneva (CH)
- Corresponding author
- Bancone, Germana
- ORCID
- Venue
- PLOS Global Public Health
- Publisher
- Public Library of Science (PLoS)
- Published
- 2022-06-17
- Volume
- 2
- Issue
- 6
- Pages
- e0000475
- ISSN
- 2767-3375 (ISSN-L)
- PMC
- PMC10021142
- OpenAlex
- W4283030835
- Cited by
- 14
- References
- 51
- Citations by year
- 2026: 2 · 2025: 4 · 2024: 5 · 2023: 1 · 2022: 2
- FWCI
- 3.29
- Type
- journal article
- Language
- en
- Open access
- gold
- doi.org
- Licence
- Locations
- PLOS Global Public Health (publishedVersion, cc-by)
- PubMed Central (publishedVersion, cc-by)
- PubMed (publishedVersion)
- DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion)
- Archive ouverte UNIGE (University of Geneva) (submittedVersion, other-oa)
- Greater South Information System
- Funders
- Wellcome Trust (106698)
- The Belgian Kids Fund for Pediatric Research
- Topics
- Health Sciences › Medicine › Pediatrics, Perinatology and Child Health › Neonatal Health and Biochemistry
- Health Sciences › Medicine › Surgery › Pediatric Hepatobiliary Diseases and Treatments
- Life Sciences › Biochemistry, Genetics and Molecular Biology › Clinical Biochemistry › Metabolism and Genetic Disorders
- Keywords
- neonatal hyperbilirubinaemia, kernicterus, glucose-6-phosphate dehydrogenase deficiency, UGT1A1 mutations, glucose-6-phosphate dehydrogenase, UGT1A1, gestational age, prolonged jaundice, population attributable risk, genetic risk factors, hematoma, Thai-Myanmar border
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
Are you an author, or do you see a problem with this page? Request a review