title: Hemolytic disease of the fetus and newborn with late-onset anemia due to anti-M: a case report and review of the Japanese literature.
abstract: Hemolytic disease of the fetus and newborn (HDFN) attributed to M/N-incompatibility varies from asymptomatic to lethally hydropic. Case reports are rare, and the clinical significance of anti-M is not completely understood. A challenging case of HDFN due to anti-M prompted an investigation of the Japanese literature, in order to characterize the clinical spectrum of M/N-incompatibility pregnancies in Japan and report results to English-language readers. Japanese reports of HDFN attributed to M/N incompatibility were compiled. Abstracted data include maternal antibody titers at delivery, fetal direct antiglobulin test, hemoglobin, total bilirubin, reticulocyte count at birth, and therapeutic interventions. We investigated characteristics of HDFN due to M/N-incompatible pregnancies in Japan after encountering a case of severe HDFN along with late-onset anemia in an infant born to a woman carrying IgG anti-M with a titer of 1. In total, thirty-three babies with HDFN due to anti-M and one due to anti-N have been reported in Japan since 1975. The median maternal antibody titer was 64 at delivery and was 16 or less in 10 of 34 women (29%). Five of 34 babies (15%) were stillborn or died as neonates. Twenty-one of 29 survivors (72%) had severe hemolytic anemia and/or hydrops fetalis. The reticulocyte count of neonates with anemia stayed below the reference interval. Sixteen (55%) developed late-onset anemia and 14 (48%) were transfused with M-negative RBCs. Significant positive correlation (P < .05) between the hemoglobin value and the reticulocyte count within 4 days of birth was obtained in 16 babies with anti-M HDFN. In the Japanese population, 21 of 34 cases of M/N-incompatible HDFN (72%) have manifested as severe hemolytic anemia and/or hydrops fetalis. Low reticulocyte count in neonates with late-onset anemia is consistent with suppressed erythropoiesis due to anti-M.
authors.affiliation: Department of Blood Transfusion and Transplantation Immunology, Fukushima Medical University, Fukushima, Japan. Electronic address: hyasuda@fmu.ac.jp.
authors.forename: Hiroyasu
authors.initials: H
authors.lastname: Yasuda
authors.affiliation: Department of Blood Transfusion and Transplantation Immunology, Fukushima Medical University, Fukushima, Japan.
authors.forename: Hitoshi
authors.initials: H
authors.lastname: Ohto
authors.affiliation: Department of Blood Transfusion and Transplantation Immunology, Fukushima Medical University, Fukushima, Japan; Radiation Medical Science Center, Fukushima Medical University, Fukushima, Japan.
authors.forename: Kenneth E
authors.initials: KE
authors.lastname: Nollet
authors.affiliation: Department of Blood Transfusion and Transplantation Immunology, Fukushima Medical University, Fukushima, Japan.
authors.forename: Kinuyo
authors.initials: K
authors.lastname: Kawabata
authors.affiliation: Department of Blood Transfusion and Transplantation Immunology, Fukushima Medical University, Fukushima, Japan.
authors.forename: Shunnichi
authors.initials: S
authors.lastname: Saito
authors.affiliation: Division of Central Laboratories, Gifu Prefectural Tajimi Hospital, Tajimi Japan.
authors.forename: Yoshihito
authors.initials: Y
authors.lastname: Yagi
authors.affiliation: Department of Pediatrics, Gifu Prefectural Tajimi Hospital, Tajimi Japan.
authors.forename: Yutaka
authors.initials: Y
authors.lastname: Negishi
authors.affiliation: Department of Pediatrics, Gifu Prefectural Tajimi Hospital, Tajimi Japan.
authors.forename: Atsushi
authors.initials: A
authors.lastname: Ishida
mesh_terms: D000305:Adrenal Cortex Hormones / Q000627:therapeutic use; D017668:Age of Onset; D001787:Blood Group Incompatibility / Q000097:blood* / Q000453:epidemiology / Q000276:immunology; D001803:Blood Transfusion; D003298:Coombs Test; D004899:Erythroblastosis, Fetal / Q000097:blood / Q000453:epidemiology / Q000209:etiology* / Q000276:immunology; D005260:Female; D006454:Hemoglobins / Q000032:analysis; D006801:Humans; D015160:Hydrops Fetalis / Q000097:blood / Q000453:epidemiology / Q000209:etiology / Q000276:immunology; D007074:Immunoglobulin G / Q000097:blood / Q000276:immunology*; D007231:Infant, Newborn; D007564:Japan / Q000453:epidemiology; D008951:MNSs Blood-Group System / Q000235:genetics / Q000276:immunology*; D008297:Male; D011247:Pregnancy; D011248:Pregnancy Complications / Q000097:blood* / Q000453:epidemiology / Q000276:immunology; D015995:Prevalence; D012010:Red-Cell Aplasia, Pure / Q000097:blood / Q000188:drug therapy / Q000209:etiology* / Q000276:immunology; D017701:Reticulocyte Count; D050497:Stillbirth / Q000453:epidemiology; D055815:Young Adult
chemical_list: D000305:Adrenal Cortex Hormones; D006454:Hemoglobins; D007074:Immunoglobulin G; D008951:MNSs Blood-Group System