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Journal of Medicinal and Chemical Sciences 6 (2023) 2679-2685

Journal homepage: http://www.jmchemsci.com/

Case Report

Hemolytic Anemia in Pregnancy: A Rare and Challenging Case in


Indonesia
Miftah Hasanah , Yetti Hernaningsih*

Department of Clinical Pathology, Faculty of Medicine, Airlangga University, Dr. Soetomo General Academic
Hospital, Surabaya, Indonesia

ARTICLE INFO ABSTRACT


Article history Autoimmune hemolytic anemia (AIHA) is an infrequent etiology of anemia
during pregnancy, and its occurrence in the postpartum period is even
Receive: 2023-05-24
more uncommon. This study presents the case of a 27-year-old woman
Received in revised: 2023-06-21 who exhibited severe anemia and thrombocytopenia six weeks after
Accepted: 2023-06-23 delivery. Hemolysis was confirmed upon evaluation, and a diagnosis of
Manuscript ID: JMCS-2305-2081 AIHA was established based on a positive direct antiglobulin test (DAT)
Checked for Plagiarism: Yes and the presence of autoantibodies targeting red blood cells (RBCs).
Language Editor: Treatment with steroids resulted in an improvement in the patient's
hemoglobin levels. The demographic characteristics observed in our case
Dr. Fatima Ramezani
align with prior research, which has documented mild anemia during
Editor who approved publication: pregnancy, lower gestational age, and decreased fetal birth weight in
Dr. Ehab AlShamaileh primary AIHA cases. Furthermore, thrombocytopenia can be manifested in
AIHA during pregnancy, necessitating long-term monitoring of such
DOI:10.26655/JMCHEMSCI.2023.11.12 patients. The AIHA rarity as a causative factor for anemia during
pregnancy, particularly in the postpartum period, underscores the
KEYWORDS significance of including AIHA in the differential diagnosis for severe
Case report anemia. Moreover, extended follow-up care is essential for managing these
Anemia patients. Employing a multidisciplinary approach to prenatal care
facilitates the identification and management of rare complications, such
Thrombocytopenia
as AIHA.
Pregnancy
Hematology
AIHA
Maternal medicine

GRAPHICALABSTRACT
Hernaningsih Y., and Hasanah M. / J. Med. Chem. Sci. 2023, 6(11) 2679-2685

Introduction unremarkable, characterized by regular antenatal


care visits and the utilization of prenatal
In 2018, the prevalence of anemia among
supplements without undergoing any laboratory
Indonesian women of reproductive age exceeded
examinations. Upon arrival at the emergency
one million cases, with immune disorders and
department, the patient was diagnosed with
iron deficiency among the leading global causes
severe anemia (Hb 3.8 g/dL) and
[1, 2].
thrombocytopenia (platelet count 87,000/µL).
Within East Java, the incidence of anemia in
The patient possessed blood type O Rh positive.
pregnant women is reported to reach 37.1%,
Notably, her blood pressure was 89/52 mmHg,
with 10% of all pregnant women receiving
pulse rate was 140 beats per minute, and
antenatal care in Surabaya presenting with
respiratory rate was 35 breaths per minute.
anemia [3, 4].
Immediate resuscitation was performed,
Autoimmune hemolytic anemia (AIHA) during
necessitating an emergency blood transfusion.
pregnancy is an uncommon condition, occurring
However, blood products were unavailable at the
at a rate of 1-3 cases per 100,000 per year, and
previous hospital. Due to the risk of hemodilution
may be manifested during both gestation and the
and persistent hypoxia, the use of crystalloids
postpartum period. The majority of cases are
was avoided. The crossmatch results revealed
severe and necessitate treatment with steroids
incompatibility, with +1 major, +3 minor, and +3
and transfusions [5-7].
in the auto control. Subsequently, the patient
AIHA is characterized by the destruction of red
received two units of packed red blood cells
blood cells as a result of autoantibodies and can
(PRC), with the major crossmatch positivity
be classified into warm, cold, or mixed types
exhibiting a weaker reaction than the auto
based on the Direct Antiglobulin Test (DAT) [5].
control. Blood transfusion was limited to a
Approximately 5% of all AIHA cases during
maximum of two units per day until the patient's
pregnancy are present in the postpartum period
hemoglobin levels reached 10 g/dL, accompanied
[5, 8].
by intensive monitoring.
This report presents a case of AIHA diagnosed
The etiology of the patient’s severe anemia was
seven days after cesarean delivery, resulting in
investigated through a comprehensive
severe anemia compounded by delivery-related
examination. Physical assessment revealed
complications yet exhibiting favorable clinical
anemic conjunctiva and the presence of a
outcomes.
palpable mass in the abdomen, encompassing
Case presentation both sides of the cesarean incision, without
alterations in abdominal circumference. No
Case Presentation A 27-year-old primiparous
organomegaly was detected. Abdominal
woman, gravida 3, para 3, was admitted to the
ultrasound unveiled a suspected subfascial
emergency department with complaints of
hematoma measuring 9.4 × 2.41 cm. No free fluid
asthenia and abdominal pain seven days
was observed within the hepatorenal,
following cesarean delivery. The decision for
splenorenal, Douglas cavity, or paracolic gutters.
emergency delivery was prompted by premature
Gynecologic examination exhibited no discernible
membrane rupture and fetal distress. The patient
abnormalities. Laboratory findings, as listed in
was at a gestational age of 32 weeks at the time of
Table 1, indicated severe anemia,
delivery. Preceding the procedure, the patient's
thrombocytopenia, reticulocytosis,
hemoglobin level was measured at 10.8 g/dL, and
hyperbilirubinemia, elevated LDH levels, positive
her platelet count was 10,000/µL. The neonates
ANA test, and normal C3 and C4 levels. No
exhibited a birth weight of 1,900 grams and
abnormalities were identified in PPT and APTT
APGAR scores of 7-8. The patient remained
coagulation tests. Urine examination
hospitalized for three days post-delivery and was
demonstrated hematuria (+3 erythrocytes) and
subsequently discharged in a favorable clinical
trace proteinuria. The direct agglutination test
condition. Her obstetric history was
(DAT) yielded a positive result.
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Hernaningsih Y., and Hasanah M. / J. Med. Chem. Sci. 2023, 6(11) 2679-2685
Table 1: Laboratory Results of the Patient
Parameter Result Reference range
Hb (g/dL) 3.3 11.0-14.7
Platelet (x10 /uL)
3 87 150-450
RET% 2.89 0.8-2.21
LDH (U/L) 562 100-190
Direct Bilirubin (mg/dL) 0.9 < 0.2
Total Bilirubin (mg/dL) 2 0.2-1.0
ANA Test (AU/mL) >400 ≤40
C3 (mg/dL) 74 50-120
C4 (mg/dL) 19.4 17.4-52.2
***A blood smear evaluation, illustrated in Figure 1, exhibited normochromic normocytic anisopoikilocytosis
(microcytes, ovalocytes, and spherocytes), polychromatophilic cells (+), normoblasts (+), and agglutination (+),
indicative of hemolytic anemia.

Figure 1: Blood Smear Evaluation of the Patient, Indicating Spherocyte (Blue Arrow) and Agglutination (Red
Arrow)

Following an incompatible result in the initial In addition, the patient was administered
crossmatch test, the patient underwent screening intravenous methylprednisolone at a dose of 125
for irregular antibodies using three Hemascreen mg three times daily for the initial five days. On
panels. The results revealed positive antigens for the sixth day, with the patient's hemoglobin level
anti-k, anti-Kpb, and anti-Lub. Based on these measuring 11.1 g/dL, laparotomy surgery was
findings, the provisional diagnosis for the patient successfully performed. Although the patient's
was autoimmune hemolytic anemia (AIHA) with hemoglobin level slightly decreased to 10.6 g/dL
suspected subfascial hematoma associated with after the surgery, no complications were
Systemic Lupus Erythematosus (SLE). encountered. Regular monitoring was conducted,
A laparotomy surgery was scheduled for the and on the seventh day of treatment, the patient
patient to address the bleeding from the reported no complaints. The steroid dosage was
subfascial hematoma. Preceding the surgery, the gradually tapered, starting with 125 mg of
patient's hemodynamic status was stabilized, and methylprednisolone intravenously once daily for
she received blood transfusion therapy consisting three days, followed by a further reduction to
of two units of Packed Red Cells per day until her 62.5 mg of methylprednisolone intravenously for
hemoglobin level reached 10 g/dL, totaling 12 two days.
units of PRC. The patient exhibited remarkable clinical
improvement and was discharged from the
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hospital with a hemoglobin level of 12.1 g/dL. to red blood cells, which can lead to their
Subsequently, the steroid treatment was destruction. Most cases of AIHA are DAT positive.
transitioned to an oral form, commencing with 16 However, 5-10% of AIHA cases may be DAT
mg of methylprednisolone. negative [13, 14].
Autoantibodies to RBCs rarely develop in
Results and Discussion pregnant women, with anti-c and anti-K
The demographic characteristics of this case align antibodies being particularly associated with
with findings from previous studies [5, 9]. significant hemolysis compared to other
The patient is 27 years old, which is the median antibodies.
age for AIHA during pregnancy. The patient's age In this patient, the autoantibodies identified were
of 27 corresponds to the median age for AIHA anti-k, anti-Kpb, and anti-Lub. These antibodies
during pregnancy. may have arisen due to micro-transfusions
Furthermore, mild anemia during pregnancy and during pregnancy, leading to autoimmunization
lower gestational age were observed, with the against RBCs. Nonetheless, these autoantibodies
termination of pregnancy typically occurring pose no harm to the pregnancy [15].
around the 32nd week [10, 11]. Notably, primary In patients with AIHA, the presence of
AIHA was associated with fetal low birth weight autoantibodies can mask the presence of
in most cases. alloantibodies. Knowledge of the patient's
AIHA is defined as a hemoglobin level below 10 phenotype can guide RBC transfusion strategies
g/dL along with a positive Direct Antiglobulin [16].
Test (DAT) [9]. Thrombocytopenia is a common finding in AIHA
In this patient, evidence of AIHA was supported during pregnancy, and it is often accompanied by
by a markedly low hemoglobin level, immune thrombocytopenia (ITP) in 0.8-3.7% of
reticulocytosis, elevated bilirubin, and LDH levels, cases, a condition known as Evans Syndrome [17,
positive DAT, and the presence of spherocytes in 18].
the blood smear evaluation. In this patient, the thrombocytopenia could
The presence of polychromasia indicates potentially be attributed to gestational
reticulocytosis, while the occurrence of thrombocytopenia, as the platelet count
spherocytes is frequently observed in hemolytic increased during the treatment course, as
processes. Severe cases may also exhibit depicted in Figure 2. However, long-term follow-
normoblasts and agglutination, as seen in this up is recommended to monitor the platelet count
case [12]. and ensure the absence of thrombocytopenia
The DAT, also known as the Coombs test, detects recurrence.
the presence of IgG and complement (C3) bound

Figure 2: Hemoglobin level and platelet count

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Hernaningsih Y., and Hasanah M. / J. Med. Chem. Sci. 2023, 6(11) 2679-2685

Warm AIHA, mediated by IgG, accounts for Disclosure Statement


approximately 80-90% of cases, while cold AIHA,
No potential conflict of interest was reported by
mediated by IgM, is more common in children.
the authors.
Each type of AIHA can be further classified based
on the presence of underlying diseases [12]. Funding
A previous report by Maroto et al. demonstrated
This research did not receive any specific grant
that warm, IgG-mediated AIHA exhibited normal
from funding agencies in the public, commercial,
clinical findings and did not result in adverse
or not-for-profit sectors.
perinatal outcomes [19, 20].
In this patient, Warm AIHA secondary to SLE
Authors' Contributions
remains a possibility. Although the ANA test was
positive, the C3 and C4 levels remained within All authors contributed to data analysis, drafting,
the normal range. SLE can also manifest during and revising of the paper and agreed to be
pregnancy, increasing the risk of pregnancy- responsible for all the aspects of this work.
related complications [21, 22].
However, primary AIHA due to the pregnancy Orcid
itself, which is exceedingly rare, remains the Yetti Hernaningsih
primary etiology of AIHA in this patient, as there https://orcid.org/0000-0001-8773-8267
was no history of other illnesses before or during Miftah Hasanah
the pregnancy. https://orcid.org/0000-0002-9791-847X
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HOW TO CITE THIS ARTICLE
Yetti Hernaningsih, Miftah Hasanah. Hemolytic Anemia in Pregnancy: A Rare and Challenging Case in Indonesia. J. Med.
Chem. Sci., 2023, 6(11) 2679-2685.
DOI: https://doi.org/10.26655/JMCHEMSCI.2023.11.12
URL: https://www.jmchemsci.com/article_174366.html

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