{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,6]],"date-time":"2026-06-06T23:25:33Z","timestamp":1780788333523,"version":"3.54.1"},"reference-count":35,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T00:00:00Z","timestamp":1716508800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T00:00:00Z","timestamp":1716508800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Blood &amp;amp; Cancer"],"published-print":{"date-parts":[[2024,8]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Infantile pyknocytosis (IP) is a rare, probably misestimated, cause of non\u2010immune neonatal hemolytic anemia evolving in two phases: an initial phase with severe jaundice, followed by a second phase with hemolytic anemia, which may require neonatal intensive care. The diagnosis of IP is based on the transient presence on blood smear of hyperdense, contracted, and\/or spiculated red blood cells (pyknocytes), associated with the spontaneous resolution of clinico\u2010biological features and the exclusion of other causes. If the etiology remains undetermined, some contributing factors, such as oxidative stress, have been proposed. We report the description of 16 patients with IP aiming at clarifying the circumstances associated with the development of this acquired disorder. In the acute phase, the mean hemoglobin nadir and pyknocyte count were 7.8\u00a0g\/dL and 11%, respectively, and strikingly, Heinz bodies were evident in 50% of the newborns, but in 100% after prolonged incubation (4\u00a0hours). A high proportion of Mediterranean or African ancestry was noted in newborns, as well as a significant number of peripartum events, such as respiratory distress. If the etiology of IP is certainly multifactorial, our series reinforces the role of oxidative stress, which may, at least in part, find origin in desaturation episodes in newborns.<\/jats:p>","DOI":"10.1002\/pbc.31078","type":"journal-article","created":{"date-parts":[[2024,5,25]],"date-time":"2024-05-25T01:25:51Z","timestamp":1716600351000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":7,"title":["Infantile pyknocytosis, a neonatal hemolytic anemia with Heinz bodies: A cohort study"],"prefix":"10.1002","volume":"71","author":[{"given":"Marie","family":"Drouilly","sequence":"first","affiliation":[{"name":"Service d'H\u00e9matologie Biologique, CHRU Nancy  Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laur\u00e9lia","family":"Jourdan","sequence":"additional","affiliation":[{"name":"Service de N\u00e9onatologie, CHRU Nancy  Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Delphine","family":"G\u00e9rard","sequence":"additional","affiliation":[{"name":"Service d'H\u00e9matologie Biologique, CHRU Nancy  Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer","family":"Russello","sequence":"additional","affiliation":[{"name":"Service d'H\u00e9matologie Biologique, CHU Montpellier  Montpellier France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9357-4542","authenticated-orcid":false,"given":"Victor","family":"Bob\u00e9e","sequence":"additional","affiliation":[{"name":"Service d'H\u00e9matologie Biologique, CHU Rouen  Rouen France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexia","family":"Audouy","sequence":"additional","affiliation":[{"name":"Service d'H\u00e9matologie Biologique, CHRU Nancy  Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aur\u00e9lie","family":"Phulpin","sequence":"additional","affiliation":[{"name":"Service d'Onco\u2010H\u00e9matologie P\u00e9diatrique, CHRU Nancy  Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6037-6403","authenticated-orcid":false,"given":"Julien","family":"Perrin","sequence":"additional","affiliation":[{"name":"Service d'H\u00e9matologie Biologique, CHRU Nancy  Nancy France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,5,24]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"crossref","DOI":"10.1080\/14767058.2021.2021177","article-title":"Neonatal hyperbilirubinemia and the role of unbound bilirubin","volume":"35","author":"Hegyi T","year":"2022","journal-title":"J Matern Fetal Neonatal Med"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-023-28254-3"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1159\/000357378"},{"key":"e_1_2_11_5_1","doi-asserted-by":"publisher","DOI":"10.1001\/archpedi.1959.02070020229009"},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2141.2006.06033.x"},{"key":"e_1_2_11_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.pedneo.2021.05.015"},{"key":"e_1_2_11_8_1","article-title":"Infantile pyknocytosis in Mexican\u2010American infants","volume":"117","author":"Ackerman BD","year":"1969","journal-title":"Am J Dis Child"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0022-3476(67)80416-5"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.5409\/wjcp.v6.i4.176"},{"key":"e_1_2_11_11_1","article-title":"[Infantile pyknocytosis: a rare form of neonatal hemolytic anemia. 5 case\u2010studies]","volume":"15","author":"Limme B","year":"2008","journal-title":"Arch Pediatr"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21583"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM196511182732109"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2141.1962.tb06519.x"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1111\/trf.14467"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2019004019"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.25358"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.jp.7211852"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.semperi.2008.10.010"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41598-023-27579-3"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2021.08.002"},{"key":"e_1_2_11_22_1","doi-asserted-by":"publisher","DOI":"10.1159\/000491626"},{"key":"e_1_2_11_23_1","doi-asserted-by":"publisher","DOI":"10.1097\/MPH.0000000000001461"},{"key":"e_1_2_11_24_1","article-title":"[The hemolytic anemia of premature infants with spontaneous Heinz body formation; a new syndrome observed in 14 cases]","volume":"8","author":"Gasser C","year":"1953","journal-title":"Helv Paediatr Acta"},{"key":"e_1_2_11_25_1","article-title":"[Heinz body formation in erythrocytes of the newborn]","volume":"73","author":"Kunzer W","year":"1956","journal-title":"Folia Haematol Int Mag Klin Morphol Blutforsch"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1097\/MPH.0000000000002004"},{"key":"e_1_2_11_27_1","article-title":"Infantile pyknocytosis: a rare form of neonatal anemia","volume":"12","author":"Kraus D","year":"2010","journal-title":"Isr Med Assoc J"},{"key":"e_1_2_11_28_1","doi-asserted-by":"publisher","DOI":"10.1002\/ccr3.288"},{"key":"e_1_2_11_29_1","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.30067"},{"key":"e_1_2_11_30_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.anpedi.2023.08.009"},{"key":"e_1_2_11_31_1","doi-asserted-by":"publisher","DOI":"10.1055\/s-0037-1607283"},{"key":"e_1_2_11_32_1","first-page":"1","article-title":"Distribution of ABO and Rhesus D blood antigens in Morocco","volume":"6","author":"Benahadi A","year":"2013","journal-title":"Internet J Biol Anthropol"},{"key":"e_1_2_11_33_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1246-7820(03)00100-9"},{"key":"e_1_2_11_34_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.tracli.2018.08.021"},{"key":"e_1_2_11_35_1","first-page":"403","article-title":"[ABO and Rh (D) blood groups in the Turkish population]","volume":"16","author":"B\u00fcy\u00fcky\u00fcksel C","year":"1973","journal-title":"Rev Fr Transfus"},{"key":"e_1_2_11_36_1","doi-asserted-by":"publisher","DOI":"10.5694\/j.1326-5377.1960.tb68732.x"}],"container-title":["Pediatric Blood &amp; Cancer"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/pbc.31078","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1002\/pbc.31078","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/pbc.31078","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,16]],"date-time":"2026-03-16T06:38:14Z","timestamp":1773643094000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/pbc.31078"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,5,24]]},"references-count":35,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2024,8]]}},"alternative-id":["10.1002\/pbc.31078"],"URL":"https:\/\/doi.org\/10.1002\/pbc.31078","archive":["Portico"],"relation":{},"ISSN":["1545-5009","1545-5017"],"issn-type":[{"value":"1545-5009","type":"print"},{"value":"1545-5017","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,5,24]]},"assertion":[{"value":"2023-11-01","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-05-02","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-05-24","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}],"article-number":"e31078"}}