{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,4]],"date-time":"2026-05-04T06:23:02Z","timestamp":1777875782878,"version":"3.51.4"},"reference-count":38,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2021,6,1]],"date-time":"2021-06-01T00:00:00Z","timestamp":1622505600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2021,6,1]],"date-time":"2021-06-01T00:00:00Z","timestamp":1622505600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Thyroid\u00ae"],"published-print":{"date-parts":[[2021,6]]},"abstract":"<jats:sec>\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>The etiology of most cases of congenital hypothyroidism (CHT) due to thyroid dysgenesis (DG) is unknown. If transient environmental factors can impact on thyroid gland development, then clustering of cases in time and\/or space may occur, and this would be more likely in thyroid DG than dyshormonogenesis (DHG).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>\n                      The newborn screening program for CHT in Scotland is linked to a central database that includes case details such as postcode. The etiology of CHT is investigated in many cases of CHT using scintigraphy and\/or ultrasonography. We looked for evidence of a change in CHT incidence with year of birth and according to season of the year. We then undertook space\u2013time clustering analysis (using a method based on\n                      <jats:italic toggle=\"yes\">K<\/jats:italic>\n                      -functions, with nearest neighbor thresholds) of CHT in Scotland between 1979 and 2015. We also looked for evidence of overall changes associated with sex and area-based birth density.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>\n                      Of 531 cases with CHT during the study period, 290 cases had been categorized as DG (\n                      <jats:italic toggle=\"yes\">n<\/jats:italic>\n                      \u2009=\u2009229) or DHG (\n                      <jats:italic toggle=\"yes\">n<\/jats:italic>\n                      \u2009=\u200961) following more detailed investigation. The incidence of CHT increased with year of birth and was in part linked to changing methodology, but there was no seasonality. There was no evidence of overall space\u2013time clustering (\n                      <jats:italic toggle=\"yes\">p<\/jats:italic>\n                      \u2009=\u20090.06), but there was evidence of clustering in babies with DG (\n                      <jats:italic toggle=\"yes\">p<\/jats:italic>\n                      \u2009=\u20090.007). This picture appeared to be most closely linked to underlying thyroid gland hypoplasia rather than thyroid gland agenesis or ectopia. There was significant space\u2013time clustering for both males and females, but clustering was restricted to lesser birth density areas. There was also evidence of clustering for unknown cases (\n                      <jats:italic toggle=\"yes\">p<\/jats:italic>\n                      \u2009&lt;\u20090.001). Clustering of these cases was restricted to females but was present for cases from both greater and lesser birth density areas. There was no evidence of clustering in cases of DHG.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions:<\/jats:title>\n                    <jats:p>These data suggest that an unidentified environmental factor or factors may be involved in the etiology of thyroid DG in Scotland. The variation in CHT incidence observed internationally may reflect environmental as well as genetic factors.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1089\/thy.2020.0005","type":"journal-article","created":{"date-parts":[[2020,11,13]],"date-time":"2020-11-13T00:07:46Z","timestamp":1605226066000},"page":"876-883","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":11,"title":["Congenital Hypothyroidism: Space\u2013Time Clustering of Thyroid Dysgenesis Indicates a Role for Environmental Factors in Disease Etiology"],"prefix":"10.1177","volume":"31","author":[{"given":"Richard J.Q.","family":"McNally","sequence":"first","affiliation":[{"name":"Population Health Sciences Institute, Newcastle University, Sir James Spence Institute, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jeremy H.","family":"Jones","sequence":"additional","affiliation":[{"name":"Royal Hospital for Children, Glasgow, United Kingdom."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mohamad Guftar","family":"Shaikh","sequence":"additional","affiliation":[{"name":"Royal Hospital for Children, Glasgow, United Kingdom."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Malcolm D.C.","family":"Donaldson","sequence":"additional","affiliation":[{"name":"Section of Child Health, Royal Hospital for Children, University of Glasgow School of Medicine, Glasgow, United Kingdom."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Karen","family":"Blakey","sequence":"additional","affiliation":[{"name":"Population Health Sciences Institute, Newcastle University, Sir James Spence Institute, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tim D.","family":"Cheetham","sequence":"additional","affiliation":[{"name":"Department of Paediatric Endocrinology, Royal Victoria Infirmary, Newcastle upon Tyne, United Kingdom."},{"name":"Translational and Clinical Research Institute, Newcastle University, Newcastle upon Tyne, United Kingdom."}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"179","published-online":{"date-parts":[[2021,6,8]]},"reference":[{"key":"e_1_3_5_2_2","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-06-0709"},{"key":"e_1_3_5_3_2","doi-asserted-by":"publisher","DOI":"10.1159\/000363156"},{"key":"e_1_3_5_4_2","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2016-1879"},{"key":"e_1_3_5_5_2","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-17-0107"},{"key":"e_1_3_5_6_2","doi-asserted-by":"publisher","DOI":"10.1542\/peds.2018-1199"},{"key":"e_1_3_5_7_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2265.2009.03568.x"},{"key":"e_1_3_5_8_2","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2012-3174"},{"key":"e_1_3_5_9_2","doi-asserted-by":"publisher","DOI":"10.1007\/s10654-011-9571-8"},{"key":"e_1_3_5_10_2","doi-asserted-by":"publisher","DOI":"10.1136\/adc.76.5.411"},{"key":"e_1_3_5_11_2","doi-asserted-by":"publisher","DOI":"10.1136\/adc.2005.088427"},{"key":"e_1_3_5_12_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2013.11.057"},{"key":"e_1_3_5_13_2","doi-asserted-by":"publisher","DOI":"10.1136\/adc.2006.096776"},{"key":"e_1_3_5_14_2","first-page":"263","article-title":"European Society for Paediatric Endocrinology consensus guidelines on screening, diagnosis, and management of congenital hypothyroidism","volume":"19","author":"Leger J","year":"2014","unstructured":"Leger J, , Olivieri A, , Donaldson M, , Torresani T, , Krude H, , van Vliet G, , Polak M, , Butler G; ESPE-PES-SLEP-JSPE-APEG-APPES-ISPAE; Congenital Hypothyroidism Consensus Conference Group. 2014. European Society for Paediatric Endocrinology consensus guidelines on screening, diagnosis, and management of congenital hypothyroidism. J Clin Endocrinol Metab, 19:263\u2013284.","journal-title":"J Clin Endocrinol Metab"},{"key":"e_1_3_5_15_2","doi-asserted-by":"publisher","DOI":"10.1136\/fn.87.3.F209"},{"key":"e_1_3_5_16_2","doi-asserted-by":"publisher","DOI":"10.1089\/thy.2017.0502"},{"key":"e_1_3_5_17_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2141.2004.05166.x"},{"key":"e_1_3_5_18_2","doi-asserted-by":"publisher","DOI":"10.2307\/2985220"},{"key":"e_1_3_5_19_2","first-page":"1571","article-title":"Space-time clustering patterns in childhood leukaemia support a role for infection","volume":"82","author":"Birch JM","year":"2000","unstructured":"Birch JM, , Alexander FE, , Blair V, , Eden OB, , Taylor GM, , McNally RJQ. 2000. Space-time clustering patterns in childhood leukaemia support a role for infection. Br J Cancer, 82:1571\u20131576.","journal-title":"Br J Cancer"},{"key":"e_1_3_5_20_2","doi-asserted-by":"publisher","DOI":"10.1002\/(SICI)1097-0258(19960930)15:18<1935::AID-SIM406>3.0.CO;2-I"},{"key":"e_1_3_5_21_2","doi-asserted-by":"publisher","DOI":"10.1177\/096228029500400203"},{"key":"e_1_3_5_22_2","unstructured":"Kulldorff M 2018 Information Management Services Inc. SatScan version 9.6. Software for the spatial and space-time scan statistics. Available at www.satscan.org (accessed December11 2020)."},{"key":"e_1_3_5_23_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pmed.0020059"},{"key":"e_1_3_5_24_2","doi-asserted-by":"publisher","DOI":"10.1111\/1467-985X.00186"},{"key":"e_1_3_5_25_2","doi-asserted-by":"crossref","unstructured":"Peters C Schoenmakers N. 2019. The thyroid gland. In: Dattani MT Brook CGD (eds) Brook's Clinical Paediatric Endocrinology. Seventh edition. Wiley New York NY pp 289\u2013334.","DOI":"10.1002\/9781119152712.ch8"},{"key":"e_1_3_5_26_2","doi-asserted-by":"publisher","DOI":"10.1002\/ijc.21726"},{"key":"e_1_3_5_27_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1999.tb01084.x"},{"key":"e_1_3_5_28_2","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2007-0527"},{"key":"e_1_3_5_29_2","doi-asserted-by":"publisher","DOI":"10.1089\/thy.2010.0205"},{"key":"e_1_3_5_30_2","doi-asserted-by":"publisher","DOI":"10.1111\/cen.12474"},{"key":"e_1_3_5_31_2","doi-asserted-by":"publisher","DOI":"10.1210\/en.2018-00542"},{"key":"e_1_3_5_32_2","doi-asserted-by":"publisher","DOI":"10.1080\/00039890409603437"},{"key":"e_1_3_5_33_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.chemosphere.2007.01.010"},{"key":"e_1_3_5_34_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.envres.2016.12.019"},{"key":"e_1_3_5_35_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0176927"},{"key":"e_1_3_5_36_2","doi-asserted-by":"publisher","DOI":"10.1016\/j.envres.2013.10.001"},{"key":"e_1_3_5_37_2","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pmed.0050161"},{"key":"e_1_3_5_38_2","doi-asserted-by":"publisher","DOI":"10.1093\/hmg\/ddx145"},{"key":"e_1_3_5_39_2","doi-asserted-by":"publisher","DOI":"10.1111\/j.0006-341X.1999.00544.x"}],"container-title":["Thyroid\u00ae"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1089\/thy.2020.0005","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1089\/thy.2020.0005","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1089\/thy.2020.0005","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T22:07:11Z","timestamp":1777586831000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1089\/thy.2020.0005"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,6]]},"references-count":38,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2021,6]]}},"alternative-id":["10.1089\/thy.2020.0005"],"URL":"https:\/\/doi.org\/10.1089\/thy.2020.0005","relation":{},"ISSN":["1050-7256","1557-9077"],"issn-type":[{"value":"1050-7256","type":"print"},{"value":"1557-9077","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,6]]}}}