{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,4]],"date-time":"2026-08-04T13:56:02Z","timestamp":1785851762692,"version":"3.56.0"},"reference-count":7,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2025,3,23]],"date-time":"2025-03-23T00:00:00Z","timestamp":1742688000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2025,3,23]],"date-time":"2025-03-23T00:00:00Z","timestamp":1742688000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Ital J Pediatr"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>\n                      There are some clinical conditions that can impact the accuracy of spO\n                      <jats:sub>2<\/jats:sub>\n                      measurements, leading to an incorrect diagnosis of hypoxemia. Low oxygen affinity hemoglobinopathy can present with low spO\n                      <jats:sub>2<\/jats:sub>\n                      and discordance with PaO\n                      <jats:sub>2<\/jats:sub>\n                      and general clinical conditions.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case Presentation<\/jats:title>\n                    <jats:p>We report the case of a 14-month-old male, born extremely premature, with severe bronchopulmonary dysplasia (BPD). He required continuous high-flow nasal cannula (HFNC) oxygen therapy and was hospitalized due to a viral respiratory infection causing dyspnea and desaturation. Once the child clinically recovered, all attempts to wean him off oxygen resulted in rapid drops in SpO2. This occurred despite the absence of increased work of breathing, cyanosis, or abnormal PaO2.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Severe BPD and recurrent respiratory issues could have per se justified the persistently low spO2. Incongruence between low spO\n                      <jats:sub>2<\/jats:sub>\n                      values, normal PaO\n                      <jats:sub>2<\/jats:sub>\n                      levels and good clinical condition, once the child was recovered, raised suspicion of low oxygen affinity hemoglobin (Hb) variants. Abnormal Hb peak detected through high-performance liquid chromatography allowed rare diagnosis of Hb Titusville, confirmed by molecular analysis. In conclusion, the case matched a rare low oxygen affinity hemoglobinopathy (Hb Titusville), highlighting its relevance in unexplained hypoxemia. Diagnosis challenges necessitate a systematic approach to prevent misinterpretations.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s13052-025-01900-4","type":"journal-article","created":{"date-parts":[[2025,3,23]],"date-time":"2025-03-23T10:14:32Z","timestamp":1742724872000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["A long-lasting low oxygen saturation hiding Titusville hemoglobin diagnosis in a premature 14-month-old child \u2013 case report"],"prefix":"10.1186","volume":"51","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-1716-1738","authenticated-orcid":false,"given":"Maria Francesca","family":"Patria","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marta","family":"Piotto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cristina","family":"Curcio","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Silvana","family":"Gangi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Youssra","family":"Belhaj","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mara","family":"Lelii","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Barbara","family":"Madini","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alessia","family":"Rocchi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,3,23]]},"reference":[{"key":"1900_CR1","doi-asserted-by":"publisher","first-page":"1017","DOI":"10.1007\/s00134-005-2678-1","volume":"31","author":"R Rodr\u00edguez-Roisin","year":"2005","unstructured":"Rodr\u00edguez-Roisin R, Roca J. Mechanisms of hypoxemia. Intensive Care Med. 2005;31:1017\u201319.","journal-title":"Intensive Care Med"},{"key":"1900_CR2","doi-asserted-by":"publisher","first-page":"321","DOI":"10.1111\/pan.14335","volume":"32","author":"S Bruins","year":"2022","unstructured":"Bruins S, Sommerfield D, Powers N, von Ungern-Sternberg BS. Atelectasis and lung recruitment in pediatric anesthesia: an educational review. Paediatr Anaesth. 2022;32:321\u201329.","journal-title":"Paediatr Anaesth"},{"key":"1900_CR3","doi-asserted-by":"publisher","first-page":"e20182956","DOI":"10.1542\/peds.2018-2956","volume":"143","author":"SB DeMauro","year":"2019","unstructured":"DeMauro SB, Jensen EA, Bann CM, Bell EF, Hibbs AM, Hintz SR, Lorch SA. Home oxygen and 2-Year outcomes of preterm infants with bronchopulmonary dysplasia. Pediatrics. 2019;143:e20182956.","journal-title":"Pediatrics"},{"key":"1900_CR4","doi-asserted-by":"publisher","first-page":"1206","DOI":"10.1002\/ppul.23442","volume":"51","author":"J Yeh","year":"2016","unstructured":"Yeh J, McGrath-Morrow SA, Collaco JM. Oxygen weaning after hospital discharge in children with bronchopulmonary dysplasia. Pediatr Pulmonol. 2016;51:1206\u201311.","journal-title":"Pediatr Pulmonol"},{"key":"1900_CR5","doi-asserted-by":"publisher","first-page":"597","DOI":"10.1002\/ajh.25425","volume":"94","author":"J Yudin","year":"2019","unstructured":"Yudin J, Verhovsek M. How we diagnose and manage altered oxygen affinity hemoglobin variants. Am J Hematol. 2019;94:597\u2013603.","journal-title":"Am J Hematol"},{"key":"1900_CR6","doi-asserted-by":"publisher","first-page":"16920","DOI":"10.1038\/s41598-020-73560-9","volume":"10","author":"B Balcerek","year":"2020","unstructured":"Balcerek B, Steinach M, Lichti J, Maggioni MA, Becker PN, Labes R, Gunga HC, Persson PB, F\u00e4hling M. A broad diversity in oxygen affinity to haemoglobin. Sci Rep. 2020;10:16920.","journal-title":"Sci Rep"},{"key":"1900_CR7","doi-asserted-by":"publisher","first-page":"449","DOI":"10.1111\/jpc.13859","volume":"54","author":"M Mina","year":"2018","unstructured":"Mina M, James R, Gandhi S. Haemoglobin Titusville: a case study and review of the literature. J Paediatr Child Health. 2018;54:449\u201352.","journal-title":"J Paediatr Child Health"}],"container-title":["Italian Journal of Pediatrics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13052-025-01900-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s13052-025-01900-4\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s13052-025-01900-4.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,3,23]],"date-time":"2025-03-23T10:14:35Z","timestamp":1742724875000},"score":1,"resource":{"primary":{"URL":"https:\/\/ijponline.biomedcentral.com\/articles\/10.1186\/s13052-025-01900-4"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3,23]]},"references-count":7,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2025,12]]}},"alternative-id":["1900"],"URL":"https:\/\/doi.org\/10.1186\/s13052-025-01900-4","relation":{"has-preprint":[{"id-type":"doi","id":"10.21203\/rs.3.rs-3823476\/v1","asserted-by":"object"}]},"ISSN":["1824-7288"],"issn-type":[{"value":"1824-7288","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,3,23]]},"assertion":[{"value":"29 January 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"6 February 2025","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"23 March 2025","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written informed consent was obtained from the patient for publication of this case report and accompanying images.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"88"}}