Skip to main content

Tipp

Weitere Artikel dieser Ausgabe durch Wischen aufrufen

Erschienen in: Wiener Medizinische Wochenschrift 13-14/2022

Open Access 18.03.2021 | images in science and medicine

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

verfasst von: MD Nasenien Nourkami-Tutdibi, Martina Geipel, Gabriele Meyberg-Solomayer, Zoltan Takacs, Sascha Meyer

Erschienen in: Wiener Medizinische Wochenschrift | Ausgabe 13-14/2022

download
DOWNLOAD
share
TEILEN
print
DRUCKEN
insite
SUCHEN

Summary

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.
Hinweise

Publisher’s Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rhesus incompatibility in pregnancy may result in hemolytic disease of the fetus and newborn (HDFN) [1]. Significant progress in prenatal care strategies and improved prenatal management options have led to a remarkable reduction in perinatal mortality [2, 3]. HDFN can present with a variety of clinical features [4].
Here, we present a preterm neonate with HDFN born at a gestational age of 31 + 4 weeks by caesarean section because of hydrops fetalis and non-reassuring cardiotocography (CTG) with a weight of 2200 g to a GII, PI mother. Due to severe anemia secondary to rhesus disease, packed red blood cells were transfused in utero three times. Immediately after birth, the infant demonstrated signs of severe respiratory compromise requiring intubation (at 3 min of life) following administration of surfactant. On physical exam, gross enlargement of the abdominal cavity was noted with massive fluid collection (Fig. 1a) and hepatomegaly, mandating the immediate drainage (at 10 min of life) of 200 mL of ascites (Fig. 1b). In parallel, the neonate received fluid resuscitation and packed red blood cell transfusion in the delivery suite due to severe anemia with an initial hemoglobin concentration of 5.4 g/dl.
After admission to our neonatal intensive care unit (NICU), the neonate required inhaled nitric oxide for persistent pulmonary hypertension, phototherapy and administration of immunoglobulins (total dose of 2 g/kg body weight) for hemolytic rhesus disease (positive Coombs test; admission serum LDH concentration of 10,000 U/L; maximum serum bilirubin concentration of 210.8 µmol/L within the first 12 h of life). As serum bilirubin was below the level for exchange transfusion after the initial phase, phototherapy was continued, and no exchange transfusion was necessary.
On ultrasonography and echocardiography, no pleural or pericardial effusions were initially noted. Despite hypertrophic cardiomyopathy, left ventricular function was initially good and no soft tissue edema was seen postnatally (Fig. 1). Transfusion of platelets, red blood cells and fresh frozen plasma were required within the first days of life. During the further clinical course, cardiac function deteriorated secondary to worsened hypertrophic cardiomyopathy. On day 6 of life, the patient presented with sudden bradycardia and cardiac arrest. A pericardial effusion was successfully drained, but resuscitation was nevertheless unsuccessful. Autopsy revealed septic toxic cardiovascular failure due to necrotizing enterocolitis with extensive ischemic hemorrhagic of the intestinal mucosa.
This case shows an unusual presentation of HDFN in a preterm infant with isolated massive abdominal fluid collection as the leading clinical sign in addition to severe anemia in the delivery suite. The immediate drainage of ascites provided transient clinical stabilization with improved pulmonary function in the delivery suite. To provide adequate neonatal intensive care in the delivery suite in patients with HDFN, adequate staffing is of utmost importance; our patient was cared for by two fully trained neonatologists, one pediatrician, and one nurse with full training in neonatology. Because of the unusual presentation in our patient with possible further underlying disease (e.g., genetic, metabolic), genetic studies were performed, including exome analysis, which did not reveal any pathological findings.

Conflict of interest

N. Nourkami-Tutdibi, M. Geipel, G. Meyberg-Solomayer, Z. Takacs, and S. Meyer declare that they have no competing interests.
Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://​creativecommons.​org/​licenses/​by/​4.​0/​.

Publisher’s Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
download
DOWNLOAD
share
TEILEN
print
DRUCKEN
Literatur
2.
Zurück zum Zitat Smits-Wintjens VEHJ, Walther FJ, Lopriore E. Rhesus haemolytic disease of the newborn: postnatal management, associated morbidity and long-term outcome. Semin Fetal Neonatal Med. 2008;13:265–71. CrossRef Smits-Wintjens VEHJ, Walther FJ, Lopriore E. Rhesus haemolytic disease of the newborn: postnatal management, associated morbidity and long-term outcome. Semin Fetal Neonatal Med. 2008;13:265–71. CrossRef
3.
Zurück zum Zitat Elalfy MS, Elbarbary NS, Abaza HW. Early intravenous immunoglobin (two-dose regimen) in the management of severe Rh hemolytic disease of newborn—a prospective randomized controlled trial. Eur J Pediatr. 2011;170:461–7. CrossRef Elalfy MS, Elbarbary NS, Abaza HW. Early intravenous immunoglobin (two-dose regimen) in the management of severe Rh hemolytic disease of newborn—a prospective randomized controlled trial. Eur J Pediatr. 2011;170:461–7. CrossRef
4.
Zurück zum Zitat Huang HR, Tsay PK, Chiang MC, Lien R, Chou YH. Prognostic factors and clinical features in liveborn neonates with hydrops fetalis. Am J Perinatol. 2007;24:33–8. CrossRef Huang HR, Tsay PK, Chiang MC, Lien R, Chou YH. Prognostic factors and clinical features in liveborn neonates with hydrops fetalis. Am J Perinatol. 2007;24:33–8. CrossRef
Metadaten
Titel
Hydrops fetalis with isolated massive ascites in a preterm neonate with rhesus disease
verfasst von
MD Nasenien Nourkami-Tutdibi
Martina Geipel
Gabriele Meyberg-Solomayer
Zoltan Takacs
Sascha Meyer
Publikationsdatum
18.03.2021
Verlag
Springer Vienna
Erschienen in
Wiener Medizinische Wochenschrift / Ausgabe 13-14/2022
Print ISSN: 0043-5341
Elektronische ISSN: 1563-258X
DOI
https://doi.org/10.1007/s10354-021-00829-7

Weitere Artikel der Ausgabe 13-14/2022

Wiener Medizinische Wochenschrift 13-14/2022 Zur Ausgabe