Author
Information
Changede P, Gupta S, Chavan N.
(* Assistant Professor, ** Registrar, Additional Professor. Department of Obstetrics and Gynecology, LTMMC & LTMGH, Mumba, India.)
(* Assistant Professor, ** Registrar, Additional Professor. Department of Obstetrics and Gynecology, LTMMC & LTMGH, Mumba, India.)
Abstract
Ebstein's
anomaly (EA) is an uncommon malformation of the tricuspid valve
presenting with a wide array of clinicopathological features.
The incidence of
Ebstein's anomaly in the general population has been estimated as 1
in 210000 or less than 1 % of all congenital heart defects and it has
an equal sex ratio. Ebstein’s anomaly is well tolerated in
pregnancy in the absence
of cardiomegaly, cyanosis and arrhythmias and in patients with mild
cardiac dysfunction and a low Newyork heart assosciation (NYHA)
functional class.
We present one case of Ebstein’s anomaly who underwent cesarean
section under general anesthesia with good fetomaternal outcome.
Introduction
Ebstein’s
anomaly is an uncommon congenital heart disease.
There is dysplastic abnormality of basal tricuspid valve leaflets.
Tricuspid regurgitation occurs due to elongation and downward
displacement of the septal and anterior cusps of the valve. Free
attachments of tricuspid valve is also noted.[1,2]
There is atrialization of proximal part of the right ventricle. The
right ventricle and an enlarged right atrium becomes thin walled and
poorly contractile.[3,4]
Congestive heart failure is the most common cause of death. A
considerable proportion of women with this disease reach childbearing
age
and pose a challenge to the treating obstetrician.
Case
Report
29
years old female was admitted to our tertiary hospital with a
diagnosis of Gravida 7 Para 2 Living 2 Abortion 4, with 38 weeks and
5 days with Ebstein’s anomaly for safe confinement. She had
previous two full term cesarean delivery and four spontaneous
abortions for which check curettage was done. No congenital disease
was detected in her previous offspring’s. Intrapartum and
postpartum period of both pregnancies was uneventful. On admission,
patient was asymptomatic. There was no history of breathlessness,
chest pain, palpitation, syncopal attacks or cyanotic spells. On
verification of past records,
she was diagnosed as a
case of Ebstein’s anomaly with severe tricuspid regurgitation just
before conception, when she had complaints of retrosternal chest
pain, shortness of breath on exertion and generalized weakness. On
examination, she was found to have normal vital parameters with no
signs of cardiac failure. Examination of cardiovascular system
revealed the presence of a pansystolic murmur
with no clicks, best
heard in the tricuspid area. All hematological and radiological
investigations were within normal limits.
Echocardiography
done at term was suggestive of congenital Ebstein’s anomaly with
severe tricuspid regurgitation , mild pulmonary artery hypertension
(38 mm), normal left ventricular systolic function and mild right
ventricular systolic dysfunction . She had a small patent foramen
ovale with right to left shunt. There was apical displacement of
tricuspid leaflets by 23 mm. Anterior tricuspid leaflet (ATL) was
freely mobile sail like with restricted movement of Anterior
tricuspid leaflet tip. Septal tricuspid leaflet (STL) was tethered to
the septum with tip prolapsing into the functional right atrium.
Right ventricular pressure overload pattern was noted, with ejection
fraction of 55 %. Similar echocardiography findings were present
prior to conception. She was taken for an emergency cesarean section
under general anesthesia in view of previous two cesarean sections
with Ebstein’s anomaly with severe tricuspid regurgitation in
labour. Antibiotic prophylaxis was given. A 2.360 kg female child
was delivered with good Apgar score. Her postoperative stay in the
hospital was uneventful. No congenital heart disease was detected in
the baby during follow up. Patient was asymptomatic at follow up
visit of 3 months. Husband was counselled and was willing for
permanent method of sterilization.
Discussion
Ebstein’s
anomaly is a rare congenital heart defect with a prevalence of
0.3–0.5 % characterized primarily by abnormalities of the tricuspid
valve and right ventricle.[5]
In
1866,
Wilhelm
Ebstein has first described this condition but this report was
overlooked and not quoted in the literature till the year 1900.[6]
A considerable proportion of these patients remain asymptomatic and
reach childbearing age. The physiological changes of pregnancy may be
aggravated in patients with Ebstein’s anomaly. Pregnant patients
with Ebstein’s anomaly are usually acyanotic, but those with
interatrial shunting can develop shunt reversal and cyanosis in
pregnancy. Cyanotic patients have with increased rate of premature
deliveries, low birth weight and thromboembolic complications.[7]
Connolly and Warnes
reported
the outcome of 111 pregnancies in 44 women with Ebstein’s anomaly.
In this study 16 patients were cyanotic, 20 had an interatrial
communication (atrial septal defect/ patent foramen ovale). 76% of
pregnancies resulted in live birth, 89% patients delivered vaginally,
11% were delivered by cesarean section. Low mean birth weight was
noted in infants born to cyanotic women as compared to acyanotic
women. (2530 vs. 3140 g, p < 0.001).[8]
42 pregnancies in 12 patients of Ebstein’s anomaly were described
by Donelly et
al.[9]
Pregnancy was well tolerated in most cases, resulting in 36 live
births. Mild dyspnea in third trimester of pregnancy was common, 1
patient had right heart failure and 1 had arrhythmia.
The
vaginal route of delivery is preferred in almost all cases. All
factors leading to congestive heart failure, cyanosis and
arrhythmias,[10]
should
be avoided during labor. In this case,
no
arrhythmias, cyanosis or signs of cardiac failure were observed in
present as well as previous pregnancies and the patient's NYHA
functional remained unchanged.
Conclusion
A
multispeciality team approach including an obstetrician,
neonatologist, physician and a cardiologist is beneficial for good
fetomaternal outcome in patients with Ebstein’s anomaly. Though
pregnancy is well tolerated and fetal outcome is good in most of the
patients, restricting family size and contraceptive advice should be
given to all the patients.
References
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- Dearani JA, Danielson GK. Surgical management of Ebstein’s anomaly in the adult. Semin Thorac Cardiovasc Surg 2005; 17:148-54.
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- Groves ER, Groves JB. Epidural analgesia for labour in a patient with Ebstein’s anomaly. Can J Anaesth 1995; 42 : 77 – 9.
- Attenhofer Jost CH, Connolly HM, et al. Ebstein’s anomaly: review of a multifaceted congenital cardiac condition. Swiss Med Wkly 2005; 135: 269–281.
- Mann RJ, Lie JT. The life story of Wilhelm Ebstein (1836-1912) and his almost overlooked description of a congenital heart disease. Mayo Clin Proc. 1979; 54(3): p197–204.
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- The Task Force on the Management of Cardiovascular Diseases during Pregnancy of the European Society of Cardiology (ESC) et al. ESC Guidelines on the management of cardiovascular diseases during pregnancy. Eur Heart J 2011; 32: 3147–3197.
- Nataloni M, Mocchegiani R. Ebstein’s anomaly and pregnancy: a case report. Ital Heart J 2004; 5: 707–710.
Changede
P, Gupta S, Chavan N. Successful Pregnancy Outcome In A Patient With
Ebstein’s Anomaly. JPGO 2015. Volume 2 Number 9. Available from: http://www.jpgo.org/2015/09/successful-pregnancy-outcome-in-patient.html