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Year : 2017  |  Volume : 4  |  Issue : 3  |  Page : 212-213

A case of coombs-positive severe late anemia without hyperbilirubinemia, refractory to blood transfusion, improved with immunoglobulin

1 Department of Paediatrics, Yenepoya Medical College and University, Mangalore, Karnataka, India
2 Department of Anaesthesia, KMC, Mangalore, Karnataka, India

Date of Web Publication13-Jul-2017

Correspondence Address:
Supriya Kushwah
Department of Paediatrics, Yenepoya Medical College and University, Deralakatte, Mangalore, Karnataka
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/cjhr.cjhr_104_16

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Rhesus hemolytic disease of newborn is a well-known disease with early and late complications mainly manifesting as severe hyperbilirubinemia requiring prompt treatment such as exchange transfusion and immunoglobulins. We report a case of Coombs-positive severe late anemia without hyperbilirubinemia which presented with features such as sepsis and failure to gain weight. Baby was refractory to blood transfusion initially, but later on successfully improved with immunoglobulins.

Keywords: Immunoglobulin, late anemia, rhesus hemolytic disease

How to cite this article:
Kushwah S, Kumar A, George N, Mithun H. A case of coombs-positive severe late anemia without hyperbilirubinemia, refractory to blood transfusion, improved with immunoglobulin. CHRISMED J Health Res 2017;4:212-3

How to cite this URL:
Kushwah S, Kumar A, George N, Mithun H. A case of coombs-positive severe late anemia without hyperbilirubinemia, refractory to blood transfusion, improved with immunoglobulin. CHRISMED J Health Res [serial online] 2017 [cited 2023 Mar 28];4:212-3. Available from: https://www.cjhr.org/text.asp?2017/4/3/212/210474

  Introduction Top

Late anemia is a recognized complication of rhesus hemolytic disease of the newborn (HDN), regardless of the initial disease severity.[1] Rhesus isoimmunization is becoming less common nowadays because of the development of anti-D immunoglobulin.[2] In mildly affected baby risk cannot be approximated.

We present a case of Coombs-positive severe late anemia at day-13 without hyperbilirubinemia due to Rhesus isoimmunization which successfully improved by intravenous immunoglobulin (IVIg).

  Case Report Top

A 13-day-old baby born to G4L3B-negative mother by full-term normal vaginal delivery admitted with complaints of paleness of whole body for 3 days and poor feeding for the past 2 days. Antenatal history was insignificant with birth weight of 2.7 kg. At day-2 of life, baby developed mild jaundice with serum total bilirubin/direct bilirubin - 12.7/0.6 mg/dl and received phototherapy for 48 h from outside hospital. Investigations at D-2 were normal with hemoglobin (Hb) - 16.2 g/dl. Mother had not received anti-D in any of the previous pregnancies. A history of phototherapy requirement was present in the previous third baby but no history of exchange transfusion in any baby. No history of any abortion or blood transfusion in mother. At day-10, mother noticed paleness of body with poor feeding that gradually increased in the past 3 days and after that baby was brought to our hospital. At the time of admission, baby was mildly tachypneic, SpO2- 95%, severe pallor was present with other vital signs being stable. Supportive management and oxygen inhalation were started. On admission, investigations were hemoglobin - 3.9 gm/dl, blood group - B positive, reticulocyte counts - 5.2%, corrected reticulocyte count - 1.2%, total bilirubin - 6.7 mg/dl with direct Coombs test being positive. Peripheral smear was suggestive of microcytic hypochromic anemia, but no features suggestive of any hereditary hemolytic anemia. Other tests including sepsis screens were normal. Packed red blood cells (RBCs) were transfused immediately, and after 20 h posttransfusion Hb was 8.8 g/dl. After that packed RBCs were further transfused twice with posttransfusion Hb of 12.5 g/dl. For the next 3 consecutive days, Hb estimation was done, and values obtained were 11.7, 10.8, 9.6 g/dl. In view of Coombs direct positive anemia, IVIg was transfused at 1 g/kg in two divided doses, followed by blood transfusion on the next day. Baby was discharged successfully after 3 days with Hb of 10.7g/dl and started on iron. During follow-up after 7 and 15 days, Hb was consecutively 9.9 and 9.8 g/dl.

  Discussion Top

Maternal isoimmunization against the D antigen was the most common cause of HDN, with approximately 93% of cases attributable to anti-D antibodies,[3] but after the introduction of prophylaxis, anti-D was found to account for only 50% of the cases of HDN. A number of studies have shown anti-C, anti-E, anti-Jkb to be the most common rhesus antibodies other than anti-D implicated in HDN but the severity of the disease is usually the greatest with anti-D.[4],[5] Many patients in developing countries do not receive Rh prophylaxis due to inadequate antenatal care or inability to afford anti-D immunoglobulin, so this disease is more common in developing countries. “Late” anemia is a rare complication in infants with HDN, and it does not necessarily reflect the severity of the early neonatal course of the disease. Infants with initially mild hemolysis may show a marked increase in the rate of erythrocyte destruction after the 1st week of life, probably reflecting improving reticuloendothelial function.[6] Due to a parallel maturation of hepatic function with an increasing capacity for bilirubin conjugation and excretion, this increased rate of hemolysis is not always accompanied by a significant rise in serum bilirubin. Although phototherapy has reduced the frequency of exchange transfusion in infants with HDN,[5] the incidence of late anemia is higher and occurs earlier in infants with Rhesus hemolytic disease who do not undergo exchange transfusion. Few studies have shown that hemolysis due to passively acquired antibodies may continue for 6–10 weeks in infants with HDN and after that spontaneous recovery may also occur.[7] Newer therapies and management of the more severely affected cases in tertiary centers have improved the early outcome.[8]

After the introduction of IVIg the need for exchange transfusion has reduced. The current recommendations of the American Academy of Pediatrics and the National Advisory Committee on Blood and Blood Products of Canada and Canadian Blood Services advice the use of IVIg for the treatment of HDN with established jaundice.[9] IVIg has emerged as an important component of treatment in isoimmune hemolytic jaundice. Recommended dose of IVIg is 0.5–1.0 g/kg in neonates with isoimmune hemolytic anemia.

Severe hemolytic anemia without hyperbilirubinemia due to Rh incompatibility is not common so no studies have been done previously regarding the dose and duration of IVIg.

  Conclusion Top

All infants with HDN due to Rhesus isoimmunization especially Coombs positive must be considered to be at risk for late anemia. The adequate follow-up, including repeat Hb measurements, should be done for these babies. Parents should be counseled about the importance of seeking prompt medical attention for the symptoms of anemia in affected infants. IVIg, blood transfusion along with supportive management are the treatment options. Further large studies are required to propose the use and exact dose of immunoglobulins in severe late anemia without hyperbilirubinemia in hemolytic disease of newborn.

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Conflicts of interest

There are no conflicts of interest.

  References Top

Smits-Wintjens VE, 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.  Back to cited text no. 1
Gupte SC, Kulkarni SS. Incidence of Rh immunization between 1981 and 1992. Natl Med J India 1994;7:65-6.  Back to cited text no. 2
Giblett ER. Blood group antibodies causing hemolytic disease of the newborn. Clin Obstet Gynecol 1964;10:1044-55.  Back to cited text no. 3
Velasco Rodríguez D, Pérez-Segura G, Jiménez-Ubieto A, Rodríguez MA, Montejano L. Hemolytic disease of the newborn due to anti-jkb: Case report and review of the literature. Indian J Hematol Blood Transfus 2014;30:135-8.  Back to cited text no. 4
Gottvall T, Hildén JO, Selbing A. Evaluation of standard parameters to predict exchange transfusions in the erythroblastotic newborn. Acta Obstet Gynecol Scand 1994;73:300-6.  Back to cited text no. 5
Palek J, Jarolim P. Clinical expression and laboratory detection of red blood cell membrane protein mutations. Semin Hematol 1993;30:249-83.  Back to cited text no. 6
Moncharmont P, Juron-Dupraz F, Rigal D, Vignal M, Meyer F. Haemolytic disease of two newborns in a rhesus anti-e alloimmunized woman. Review of literature. Haematologia (Budap) 1990;23:97-100.  Back to cited text no. 7
Mukhopadhyay K, Murki S, Narang A, Dutta S. Intravenous immunoglobulins in rhesus hemolytic disease. Indian J Pediatr 2003;70:697-9.  Back to cited text no. 8
Anderson D, Ali K, Blanchette V, Brouwers M, Couban S, Radmoor P, et al. Guidelines on the use of intravenous immune globulin for hematologic conditions. Transfus Med Rev 2007;21 2 Suppl 1:S9-56.  Back to cited text no. 9


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