Geipel, Martina

Meyberg-Solomayer, Gabriele

Meyer, Sascha

Nourkami-Tutdibi, Nasenien

Takacs, Zoltan

Wiener Medizinische Wochenschrift

English

PubMed

Significant progress in prenatal care has decreased the incidence of rhesus incompatibility, which may result in hemolytic disease of the fetus and newborn (HDFN). This case report describes an unusual presentation of HDFN in a preterm infant delivered by caesarean section with isolated massive abdominal fluid collection as the leading clinical sign in addition to severe anemia. The immediate drainage of ascites provided transient clinical stabilization with improved pulmonary function in the delivery suite. After admission to the neonatal intensive care unit (NICU), HDFN treatment was initiated. This case report shows the importance of adequately trained staff including neonatologists, pediatricians and NICU nurses in the delivery suite to provide neonatal intensive care for HDFN

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Hydrops fetalis with isolated massive ascites in a preterm neonate with rhesus disease

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images in science and medicineWien Med Wochenschrhttps://doi.org/10.1007/s10354-021-00829-7Hydrops fetalis with isolatedmassive ascites in a pretermneonate with rhesus diseaseNasenien Nourkami-Tutdibi · Martina Geipel · Gabriele Meyberg-Solomayer · Zoltan Takacs · Sascha MeyerReceived: 21 December 2020 / Accepted: 10 February 2021© The Author(s) 2021Summary Significant progress in prenatal care hasdecreased the incidence of rhesus incompatibility,which may result in hemolytic disease of the fetusand newborn (HDFN). This case report describes anunusual presentation of HDFN in a preterm infantdelivered by caesarean section with isolated mas-sive abdominal fluid collection as the leading clinicalsign in addition to severe anemia. The immediatedrainage of ascites provided transient clinical stabi-lization with improved pulmonary function in thedelivery suite. After admission to the neonatal inten-sive care unit (NICU), HDFN treatment was initiated.This case report shows the importance of adequatelytrained staff including neonatologists, pediatriciansand NICU nurses in the delivery suite to provideneonatal intensive care for HDFN.Keywords Erythroblastosis fetalis · Hemolyticdisease of newborn · Anemia · Neonatal intensivecare units · Jaundice, neonatalRhesus incompatibility in pregnancy may result inhemolytic disease of the fetus and newborn (HDFN)[1]. Significant progress in prenatal care strategies andimproved prenatal management options have led toa remarkable reduction in perinatal mortality [2, 3].HDFN can present with a variety of clinical features[4].N. Nourkami-Tutdibi, MD () · M. Geipel · S. MeyerHospital for General Pediatrics and Neonatology, SaarlandUniversity Medical Center, 66421 Homburg/Saar, Germanynasenien.nourkami@uks.euG. Meyberg-Solomayer · Z. TakacsHospital of Gynecology, Obstetrics and ReproductiveMedicine, Saarland University Medical Center,Homburg/Saar, GermanyHere, we present a preterm neonate with HDFNborn at a gestational age of 31+ 4 weeks by caesareansection because of hydrops fetalis and non-reassur-ing cardiotocography (CTG) with a weight of 2200g toa GII, PI mother. Due to severe anemia secondary torhesus disease, packed red blood cells were transfusedin utero three times. Immediately after birth, the in-fant demonstrated signs of severe respiratory compro-mise requiring intubation (at 3min of life) followingadministration of surfactant. On physical exam, grossenlargement of the abdominal cavity was noted withmassive fluid collection (Fig. 1a) and hepatomegaly,mandating the immediate drainage (at 10min of life)of 200mL of ascites (Fig. 1b). In parallel, the neonatereceived fluid resuscitation and packed red blood celltransfusion in the delivery suite due to severe anemiawith an initial hemoglobin concentration of 5.4g/dl.After admission to our neonatal intensive care unit(NICU), the neonate required inhaled nitric oxidefor persistent pulmonary hypertension, phototherapyand administration of immunoglobulins (total doseof 2g/kg body weight) for hemolytic rhesus disease(positive Coombs test; admission serum LDH con-centration of 10,000U/L; maximum serum bilirubinconcentration of 210.8µmol/L within the first 12h oflife). As serum bilirubin was below the level for ex-change transfusion after the initial phase, photother-apy was continued, and no exchange transfusion wasnecessary.On ultrasonography and echocardiography, nopleural or pericardial effusions were initially noted.Despite hypertrophic cardiomyopathy, left ventric-ular function was initially good and no soft tissueedema was seen postnatally (Fig. 1). Transfusion ofplatelets, red blood cells and fresh frozen plasmawere required within the first days of life. During thefurther clinical course, cardiac function deterioratedsecondary to worsened hypertrophic cardiomyopathy.K Hydrops fetalis with isolated massive ascites in a preterm neonate with rhesus diseaseimages in science and medicineFig. 1 a Severe anemia and massive abdominal distensiondue to ascites immediately after birth. b Normalization of skincolor following intubation, transfusion of packed red bloodcells and reduction in abdominal circumference after drainageof ascites in the delivery roomOn day 6 of life, the patient presented with suddenbradycardia and cardiac arrest. A pericardial effusionwas successfully drained, but resuscitation was nev-ertheless unsuccessful. Autopsy revealed septic toxiccardiovascular failure due to necrotizing enterocolitiswith extensive ischemic hemorrhagic of the intestinalmucosa.This case shows an unusual presentation of HDFNin a preterm infant with isolated massive abdominalfluid collection as the leading clinical sign in additionto severe anemia in the delivery suite. The immedi-ate drainage of ascites provided transient clinical sta-bilization with improved pulmonary function in thedelivery suite. To provide adequate neonatal inten-sive care in the delivery suite in patients with HDFN,adequate staffing is of utmost importance; our pa-tient was cared for by two fully trained neonatologists,one pediatrician, and one nurse with full training inneonatology. Because of the unusual presentation inour patient with possible further underlying disease(e.g., genetic, metabolic), genetic studies were per-formed, including exome analysis, which did not re-veal any pathological findings.Author Contribution Nasenien Nourkami-Tutdibi was re-sponsible for neonatal patient care, writing and critical re-view of the manuscript. Martina Geipel was responsible forneonatal patient care and critical review of the manuscript.Gabriele Meyberg-Solomayer was responsible for antenatalcare and critical review of the manuscript. Sascha Meyer wasresponsible for neonatal patient care and writing and criticalreview of the manuscriptFunding Open Access funding enabled and organized byProjekt DEAL.Conflict of interest N. Nourkami-Tutdibi, M. Geipel, G.Mey-berg-Solomayer, Z. Takacs, and S. Meyer declare that theyhave no competing interests.Open Access This article is licensed under a Creative Com-mons Attribution 4.0 International License, which permitsuse, sharing, adaptation, distribution and reproduction inanymedium or format, as long as you give appropriate creditto the original author(s) and the source, provide a link tothe Creative Commons licence, and indicate if changes weremade. The images or other third party material in this articleare included in the article’sCreativeCommons licence, unlessindicated otherwise in a credit line to thematerial. If materialis not included in the article’s Creative Commons licence andyour intended use is not permitted by statutory regulation orexceeds the permitted use, you will need to obtain permis-sion directly from the copyright holder. To view a copy of thislicence, visit http://creativecommons.org/licenses/by/4.0/.References1. SnowiseS,JohnsonA.Immunehydropsfetalis. In:Obstetricimaging: fetal diagnosis and care. 2nd ed. : Elsevier;2017. pp. 526–532.e1. https://doi.org/10.1016/B978-0-323-44548-1.00123-6.2. Smits-Wintjens VEHJ, Walther FJ, Lopriore E. Rhesushaemolytic disease of the newborn: postnatal manage-ment,associatedmorbidityandlong-termoutcome. SeminFetalNeonatalMed. 2008;13:265–71.3. Elalfy MS, Elbarbary NS, Abaza HW. Early intravenousimmunoglobin (two-dose regimen) in themanagement ofsevere Rh hemolytic disease of newborn—a prospectiverandomizedcontrolledtrial. EurJPediatr. 2011;170:461–7.4. HuangHR,TsayPK,ChiangMC,LienR,ChouYH.Prognosticfactors and clinical features in liveborn neonates withhydropsfetalis. AmJPerinatol. 2007;24:33–8.Publisher’sNote SpringerNature remainsneutralwith regardto jurisdictional claims in published maps and institutionalaffiliations.Hydrops fetalis with isolated massive ascites in a preterm neonate with rhesus disease K

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Hydrops fetalis with isolated massive ascites in a preterm neonate with rhesus disease

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