Case Report
Copyright ©The Author(s) 2017. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Pediatr. Nov 8, 2017; 6(4): 176-179
Published online Nov 8, 2017. doi: 10.5409/wjcp.v6.i4.176
Neonatal pyknocytosis in a preterm dizygotic twin
Alberto Berardi, Eleonora Balestri, Goretta Bonacorsi, Claudio Chiossi, Giovanni Palazzi, Eugenio Spaggiari, Fabrizio Ferrari
Alberto Berardi, Eugenio Spaggiari, Fabrizio Ferrari, Unità Operativa di Terapia Intensiva Neonatale, Dipartimento Integrato Materno-Infantile, Azienda Ospedaliero-Universitaria Policlinico, 41100 Modena, Italy
Eleonora Balestri, Unità Operativa di Terapia Intensiva Neonatale, Azienda Ospedaliera Santa Maria Nuova, 42100 Reggio Emilia, Italy
Goretta Bonacorsi, Unità Operativa di Ematologia, Dipartimento di Scienze Mediche e Chirurgiche, Azienda Ospedaliero-Universitaria Policlinico, 41100 Modena, Italy
Claudio Chiossi, Unità Operativa di Pediatria, Nuovo Ospedale Civile, 41049 Sassuolo, Italy
Giovanni Palazzi, Unità Operativa di Pediatria, Dipartimento Integrato Materno-Infantile, Azienda Ospedaliero-Universitaria Policlinico, 41100 Modena, Italy
Author contributions: Berardi A, Balestri E, Spaggiari E and Ferrari F analyzed the data, designed the report and wrote the paper; Bonacorsi G, Chiossi C and Palazzi G collected the patient’s clinical data and reviewed the manuscript.
Institutional review board statement: All authors approved the final manuscript as submitted. Because this manuscript is a case report, no review board statement was required, but a written informed consent was previously obtained from the parents of the newborn.
Informed consent statement: A written informed consent was previously obtained from the parents of the newborn.
Conflict-of-interest statement: No conflict of interest.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Alberto Berardi, MD, Unità Operativa di Terapia Intensiva Neonatale, Dipartimento Integrato Materno-Infantile, Azienda Ospedaliero-Policlinico Universitario, Via del Pozzo, 41100 Modena, Italy. berardi.alberto@policlinico.mo.it
Telephone: +39-59-4224921 Fax: +39-59-4223770
Received: January 27, 2017
Peer-review started: February 12, 2017
First decision: June 27, 2017
Revised: July 7, 2017
Accepted: September 12, 2017
Article in press: September 12, 2017
Published online: November 8, 2017
Abstract

Infantile pyknocytosis (IP) is a rare, self-limited neonatal haemolytic anaemia that may require multiple blood transfusions. Only a little more than 50 cases have been reported in the medical literature, and the great majority of them concerns term infants. The etiology of IP is not well understood; most likely it results from a transient extra-corpuscular factor, whose nature is unknown, transmitted from mother to child or, alternatively, from a deficiency of an anti-oxidative agent. We report the case of two preterm twins, one of which suffered from IP and developed severe anaemia at age 2 wk, while the other was unaffected. Although no specific agent was identified as the cause of anaemia and IP, we speculate that the transmission of an agent from mother to child was unlikely, as only twin one suffered from IP. Smelly greenish diarrhoea occurred just before the presentation of IP, suggesting that the same agent led to both the diarrhoea and the oxidative injury. Because IP may remain underdiagnosed, it should be considered in cases of early unexplained severe hemolytic anemia.

Keywords: Infantile pyknocytosis, Glucose-6-phosphate dehydrogenase deficiency, Anemia, Oxidative stress, Hemolysis

Core tip: This manuscript describes the first case of infantile pyknocytosis affecting only one of the twins, and contributes to understand the etiology of infantile pyknocytosis.