{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,20]],"date-time":"2026-05-20T01:26:34Z","timestamp":1779240394143,"version":"3.51.4"},"reference-count":13,"publisher":"BMJ","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Reports"],"accepted":{"date-parts":[[2016,2,24]]},"abstract":"<jats:p>Methemoglobinaemia is a rare cause of cyanosis in newborns. Congenital methemoglobinaemias due to M haemoglobin or deficiency of cytochrome b5 reductase are even rarer. We present a case of congenital methemoglobinaemia presenting at birth in a preterm infant. A baby boy born at 29\u2005weeks and 3\u2005days of gestation had persistent central cyanosis immediately after delivery, not attributable to a respiratory or cardiac pathology. Laboratory methemoglobin levels were not diagnostic. Cytochrome b5 reductase levels were normal and a newborn screen was unable to pick up any abnormal variants of fetal haemoglobin. Genetic testing showed a \u03b3 globin gene mutation resulting in the M haemoglobin, called Hb F-M-Fort Ripley. The baby had no apparent cyanosis at a corrected gestational age of 42\u2005weeks. Although rare, congenital methaemoglobin aemia should be considered in the differential in a preterm with central cyanosis and investigated with genetic testing for \u03b3 globin chain mutations if other laboratory tests are non-conclusive.<\/jats:p>","DOI":"10.1136\/bcr-2016-214381","type":"journal-article","created":{"date-parts":[[2016,3,11]],"date-time":"2016-03-11T14:47:39Z","timestamp":1457707659000},"page":"bcr2016214381","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":7,"title":["Congenital methemoglobinaemia due to Hb F-M-Fort Ripley in a preterm newborn"],"prefix":"10.1136","volume":"2016","author":[{"given":"Satvinder","family":"Ghotra","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Krista","family":"Jangaard","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chantale","family":"Pambrun","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Conrad Vincent","family":"Fernandez","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2016,3,11]]},"reference":[{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.1","doi-asserted-by":"publisher","DOI":"10.1542\/peds.112.2.e158"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.2","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.1995.tb13650.x"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.3","doi-asserted-by":"publisher","DOI":"10.1055\/s-0033-1333756"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.4","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpedsurg.2003.09.045"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.5","doi-asserted-by":"publisher","DOI":"10.1046\/j.1460-9592.2002.00830.x"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.6","doi-asserted-by":"publisher","DOI":"10.1111\/j.1442-200X.2011.03389.x"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.7","doi-asserted-by":"crossref","first-page":"792","DOI":"10.1542\/peds.83.5.792","article-title":"Hemoglobin FM-Fort Ripley: another lesson from the neonate","volume":"83","author":"Glader","year":"1989","journal-title":"Pediatrics"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.8","doi-asserted-by":"crossref","first-page":"734","DOI":"10.1542\/peds.83.5.734","article-title":"Mutant fetal hemoglobin causing cyanosis in a newborn","volume":"83","author":"Priest","year":"1989","journal-title":"Pediatrics"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.9","doi-asserted-by":"publisher","DOI":"10.3109\/03630269209005690"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.10","doi-asserted-by":"publisher","DOI":"10.1002\/humu.1380030310"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.11","doi-asserted-by":"publisher","DOI":"10.1001\/jamapediatrics.2014.3307"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.12","doi-asserted-by":"publisher","DOI":"10.1016\/S0196-0644(99)70167-8"},{"key":"2025042702573179000_2016.mar11_1.bcr2016214381.13","doi-asserted-by":"publisher","DOI":"10.1097\/MPH.0b013e3182667be3"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2016-214381","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,4,27]],"date-time":"2025-04-27T09:57:39Z","timestamp":1745747859000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2016-214381"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2016,3,11]]},"references-count":13,"alternative-id":["10.1136\/bcr-2016-214381"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2016-214381","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2016,3,11]]}}}