Laparoscopic Splenectomy for Resistant
Immune Thrombocytopenia in Pregnancy- A
Case Report and Review of Literature
Nirmala Chandrasekaran1*, Michelle Sholzberg2, Ori Rotstein3, Howard Berger4 and Michael Geary5
1Department of Maternal and Fetal Medicine, St Michaels Hospital, Toronto
2Department of Hematology St Michaels Hospital, Toronto
3Department of Surgery, St Michaels Hospital, Toronto
4,5Department of Maternal and Fetal Medicine, St Michaels Hospital, Toronto
Nirmala Chandrasekaran, Department of Maternal and Fetal Medicine, St Michaels Hospital, 30 Bond St, Toronto, ON M5B 1W8. E-mail:
Received: January 03, 2018; Accepted: January 24, 2018; Published: January 31, 2018
Chandrasekaran N, Sholzberg M, Ori R, et al. (2018) Laparoscopic Splenectomy for Resistant Immune Thrombocytopenia in Pregnancy- A Case Report and Review of Literature. SOJ Surgery 5(1): 1-4. DOI: http://dx.doi.org/10.15226/2376-4570/5/1/00151
Background: Thrombocytopenias in pregnancy are the second
most common hematological disorder in pregnancy after anemia.
About 7-10 percent of women experience this during pregnancy and
most are due to gestational thrombocytopenia. About 3-4% are due
to autoimmune process and is normally a diagnosis of exclusion. The
management of this condition is mainly medical with splenectomy
reserved only for very resistant cases. There is very limited literature
on safety of laparoscopic splenectomy during pregnancy. We present
such a case and a review of all cases of laparoscopic splenectomy in
Case Report: A primigravid woman presented with resistant
Immune thrombocytopenia during early pregnancy. She had very little
response to all the medical treatments that are offered in pregnancy.
She then had an uncomplicated laparoscopic splenectomy with good
response during pregnancy. There was good maternal and neonatal
Conclusion: Laparoscopic splenectomy can be safely considered
for resistant cases of splenectomy during pregnancy.
Key Words: Immune Thrombocytopenia; Pregnancy;
Laparoscopy; Splenectomy; Safety
Immune Thrombocytopenia (ITP) is an autoimmune
condition that is caused by immune mediated destruction of
platelets. Pathophysiology of ITP is thought to predominantly
involve IgG Antibodies binding to surface antigens Glycoprotein
(GP) II b-III a or GPI b-IX. Platelets with such auto antibodies
bound to the surface will be cleared from circulation by the
reticuloendothelial system with resultant thrombocytopenia
and increased risk of bleeding. Recent evidence shows that
there is suboptimal production of platelets also due to increased
apoptosis of megakaryocytes . It is predominantly a disease
of young adults, with a prevalence of 1/10,000. Hence it is not an
uncommon entity in pregnancy and accounts for approximately
3% of all cases of thrombocytopenia in pregnancy . ITP is a
diagnosis of exclusion and in pregnancy preeclampsia, HELLP,
infectious diseases, gestational thrombocytopenia must be
considered in the differential. The majority of ITP cases identified
during pregnancy are asymptomatic, but some can be associated
with hemorrhagic complications that can be potentially hazardous
for the mother and the fetus, as the antiplatelet IgG antibodies
can cross the placenta and lead to passive neonatal ITP. There is
no correlation between maternal and neonatal platelet count.
The treatment of ITP aims at restoring the platelet count
to 30,000-50,000 x 109 /liter. First line treatment options
include corticosteroids, intravenous immunoglobulin (IVIG)
and intravenous anti-Rh (anti-D) infusions. Many of the second
and third line cytotoxic and immunosuppressive therapies
are not recommended in pregnancy. One surgical second
line management option in pregnancy for resistant ITP is
splenectomy, but this is rarely required and often not considered
due to concerns of surgical risk to mother and baby. The safety of
laparoscopic splenectomy is not reported in the literature.
We report a case of resistant ITP in pregnancy successfully
and safely treated with laparoscopic splenectomy and compare
this with available data on similar cases from the literature.
A 28-year-old G1P0 presented to our Multidisciplinary Clinic
for Women with Bleeding Disorders in Pregnancy at 18 weeks
gestation following the diagnosis of isolated thrombocytopenia.
Her routine prenatal labs in a community hospital identified a
platelet count of 8000 X 109/liter at 14 weeks gestational age. She
was then admitted to hospital where clinical examination was
remarkable solely for scattered diffuse petechiae and oral wet
purpura. A full work up for thrombocytopenia was performed
and no secondary causes of thrombocytopenia were identified.
Therefore, the diagnosis of ITP was made. IVIG at a dose of one
gram per kilogram was administered for two consecutive days.
There was minimal response to the IVIG, the zenith platelet
count response was 26,000 X 109/litre. It was at this point that
she was referred to our clinic for assistance with management.
Oral prednisone was commenced at one milligram per kilogram
and IVIG was repeated to two further doses one month after the
initial doses. The patient continued to have diffuse petechiae and
small oral blood blisters. There was minimal response to either of
these treatments and the patient was admitted to the hospital for
consideration of additional therapy including a surgical consult
for splenectomy and bone marrow biopsy. The bone marrow
biopsy was consistent with ITP and no other abnormalities
were identified. Additional studies were negative including the
direct antiglobulin test, paroxysmal nocturnal hemoglobinuria
flow cytometry, viral serology, Helicobacter pylori serology,
antiphospholipid antibody tests and autoimmune serology. She
was of blood group B positive. Whilst in the hospital, she was also
treated with high dose anti-D (50 micrograms per kilogram) for
Figure 1: Platelet count according to patient gestational age in weeks
and therapeutic trials
two consecutive days with no response once again. Anti-D was
well tolerated; there was no evidence of maternal hemolysis
nor fetal anemia on middle cerebral artery doppler ultrasound
assessment. The patient refused additional immunosuppressive
therapy including rituximab. Hence, the decision was made to
proceed with a laparoscopic splenectomy at 22 weeks gestational
age after appropriate immunizations. She received 2 adult
pools of platelets prior to the procedure and underwent an
uncomplicated laparoscopic splenectomy. The patient made an
uneventful and rapid recovery. Immediately post operatively, she
showed a good platelet count response (zenith 258,000 X 109/
liter but the platelets fell to 35,000 109/liter) two weeks’ postsplenectomy
at which point she was restarted on prednisone at
30mg daily with good response (Figure 1).
An attempt at tapering the prednisone dose was done
which resulted in a platelet count drop to of 8000 X 109/liter. She
received one more dose of IVIG and the prednisone was increased
to 30mg/ day and this maintained a platelet count above 100,000
X 109/liter. The pregnancy progressed well and she had a preterm
pre-labor rupture of membranes at 35 weeks and progressed to
have a normal spontaneous vaginal delivery after five hours of
a live male infant weighing 2775 grams. The infant’s platelets at
birth were 152,000 X 109/liter and there was no further decline
in his platelet count. The postpartum and the neonatal course for
mother and baby were uneventful and both were discharged on
day 2 postpartum. She went into sustained remission postpartum
and has since had a subsequent pregnancy with no evidence of
relapse or neonatal thrombocytopenia.
Table 1: All cases of antenatal laparoscopic splenectomy reported in
the literature from 2000 to 2016. No adverse events occurred in any of
the neonates born to mothers who had a splenectomy [8,9,10,11,12,13].
First line treatment
Gestation at splenectomy
Anglin et al
K Iwase et al
Griffiths et al
Felbinger et al
Koji Kubota et al
Bernal-Macías et al
Bernal-Macías et al
Thrombocytopenia is one of the most common hematological
disorders in pregnancy, second only to anemia. There is a
physiological fall in platelet count during pregnancy due to
various reasons like hemodilution, increased platelet clearance
by enhanced macrophage activation, and increased platelet
aggregation driven by increased levels of thromboxane A2.
Thrombocytopeniamay be diagnosed for the first time in pregnancy
and the distinction between gestational thrombocytopenia and
ITP can be challenging as it relies purely on phenomenologic
criteria . The current expert-guided recommendation is to
start treatment for ITP in pregnancy when the platelet count
is less than 30,000 to 50,000 x 109/ liter, or sooner when there
is evidence of clinical bleeding or when pregnancy approaches
term due to risk aversion regarding neuraxial anesthesia
and postpartum hemorrhage. The first line treatment for ITP
in pregnancy is oral prednisone and IVIG both of which are
considered safe in pregnancy. Large doses of intravenous anti-D
have been used for treatment outside pregnancy in Rh positive
non-splenectomised patients as it results in preferential splenic
phagocytosis of the antibody coated red blood cells rather than
platelets . The use of anti-D in pregnancy is limited to small
studies but the response rate is similar to that of IVIG . Even
though medications like azathioprine have been used successfully
in non-pregnant populations, its success in pregnancy has yet to
be determined and its use is limited by delayed onset of action
in the order of months Also, rituximab, a chimeric monoclonal
antibody targeting CD20 B-cell surface antigen has been shown
to be of benefit in the treatment of ITP. A recent meta-analysis of
rituximab use in ITP suggested it has a place early in the tailored
strategy of treatment of ITP, especially before splenectomy
.The safety data for rituximab is largely unknown and the
data is extrapolated from women who required therapy for
lymphoma in pregnancy. The thrombopoietin receptor agonists
which are effective therapy for resistant and refractory ITP is
contraindicated in pregnancy.
Splenectomy during pregnancy was once considered
a high-risk procedure with high mortality rate. During early
pregnancy, it is associated with increased risk of fetal loss and
premature labor and during later stages, splenectomy may be
technically challenging due to uterine enlargement. The first
case of splenectomy via laparotomy in pregnancy was reported
in 1996 by Steimer on a primigravid woman who presented at 28
weeks of gestation with life-threatening pulmonary hemorrhage
and severe thrombocytopenia [16,7]. Since then several cases
of open splenectomy have been reported both during the
antepartum period and during cesarean section [9,10,11,12,13].
The safety of laparoscopic surgeries in pregnancy, mainly
for ectopic pregnancies and adnexal masses, has been well
established and also results in improved outcomes such as earlier
return to activity and reduction in narcotic use. Given the state of
surgical advances, efficacy and safety of laparoscopic techniques,
splenectomy can be considered when required for management
of resistant ITP in pregnancy [14,15].
In conclusion, our recent case and the six previously
reported cases of laparoscopic splenectomy during pregnancy
suggest that splenectomy can be safely considered in cases of
resistant ITP in pregnancy in carefully selected cases. One can
make an argument to watch and wait if the patient does not have
any symptoms of bleeding. The bleeding phenotype in our patient
led to the aggressive management. Splenectomy is a reasonable
and safe option for patients who fail to respond to the traditional
first line treatments and when there is no time for azathioprine
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