journal article Open Access May 24, 2024

Infantile pyknocytosis, a neonatal hemolytic anemia with Heinz bodies: A cohort study

View at Publisher Save 10.1002/pbc.31078
Abstract
Abstract
Infantile pyknocytosis (IP) is a rare, probably misestimated, cause of non‐immune 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‐biological 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 g/dL and 11%, respectively, and strikingly, Heinz bodies were evident in 50% of the newborns, but in 100% after prolonged incubation (4 hours). 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.
Topics

No keywords indexed for this article. Browse by subject →

References
35
[1]
Hegyi T "Neonatal hyperbilirubinemia and the role of unbound bilirubin" J Matern Fetal Neonatal Med (2022) 10.1080/14767058.2021.2021177
[7]
Ackerman BD "Infantile pyknocytosis in Mexican‐American infants" Am J Dis Child (1969)
[10]
Limme B "[Infantile pyknocytosis: a rare form of neonatal hemolytic anemia. 5 case‐studies]" Arch Pediatr (2008)
[23]
Gasser C "[The hemolytic anemia of premature infants with spontaneous Heinz body formation; a new syndrome observed in 14 cases]" Helv Paediatr Acta (1953)
[24]
Kunzer W "[Heinz body formation in erythrocytes of the newborn]" Folia Haematol Int Mag Klin Morphol Blutforsch (1956)
[25]
Infantile Pyknocytosis in a Premature Dichorionic Diamniotic Twin

Mandy E. Spoorenberg, Renate E. Wachters-Hagedoorn, Richard van Wijk et al.

Journal of Pediatric Hematology/Oncology 10.1097/mph.0000000000002004
[26]
Kraus D "Infantile pyknocytosis: a rare form of neonatal anemia" Isr Med Assoc J (2010)
[31]
Benahadi A "Distribution of ABO and Rhesus D blood antigens in Morocco" Internet J Biol Anthropol (2013)
[34]
Büyükyüksel C "[ABO and Rh (D) blood groups in the Turkish population]" Rev Fr Transfus (1973)