Congenital heart disease (CHD) affects approximately 0.8-1% of all live births, with a significant number of these patients now surviving into adulthood and childbearing age due to advances in medical and surgical management.[1,2] This improved survival has led to an increasing number of women with CHD reaching reproductive age, creating new challenges for obstetric and anaesthetic management.[3] The spectrum of congenital heart disease encompasses a wide range of lesions, from simple defects such as atrial septal defects to complex cyanotic conditions like tetralogy of Fallot, each presenting unique physiological challenges during pregnancy.[4]

Pregnancy in women with CHD presents unique challenges due to the physiological cardiovascular changes that occur during gestation, which can exacerbate pre-existing cardiac conditions.[5] The maternal cardiovascular system undergoes substantial adaptations during pregnancy, including a 30-50% increase in blood volume, 30-40% increase in cardiac output, and a decrease in systemic vascular resistance.[6,7] These changes peak during the third trimester and may cause decompensation in women with underlying cardiac pathology.[8]

Different types of congenital heart lesions pose varying degrees of risk and require distinct management strategies. Left-to-right shunts, such as those created by ruptured sinus of Valsalva aneurysms or uncorrected atrial septal defects, can lead to volume overload and pulmonary hypertension.[9] Right-to-left shunts, exemplified by uncorrected tetralogy of Fallot, result in systemic hypoxemia and pose risks of paradoxical embolism.[10] Complex lesions leading to Eisenmenger syndrome represent the most challenging scenarios, with fixed pulmonary hypertension and significant maternal mortality risk.[11]

Sinus of Valsalva aneurysm (SOVA) is a rare cardiac anomaly, with an incidence of 0.1-3.5% of all congenital heart defects.[12] It occurs due to a defect in the aortic media, resulting in lack of fusion between the aortic media and the annulus fibrosus of the aortic valve.[13] The right coronary sinus is most commonly affected (70-90%), followed by the non-coronary sinus (10-25%), with the left coronary sinus being the least frequently involved (<5%).[14] Unruptured SOVA is often asymptomatic, but rupture typically presents with acute onset of symptoms including palpitations, chest pain, and signs of heart failure due to the sudden left-to-right shunt that develops.[15,16]

Tetralogy of Fallot represents one of the most common cyanotic congenital heart defects, comprising approximately 3-5% of all congenital heart disease cases.[17] The classic tetrad includes ventricular septal defect, right ventricular outflow tract obstruction, right ventricular hypertrophy, and overriding aorta.[18] While most patients undergo corrective surgery in childhood, a small percentage may reach adulthood with uncorrected anatomy, presenting significant challenges during pregnancy due to chronic hypoxemia and the risk of hypercyanotic spells.[19]

Eisenmenger syndrome develops when a large left-to-right shunt leads to irreversible pulmonary vascular disease and eventual shunt reversal.[20] This condition carries one of the highest maternal mortality risks in pregnancy, with reported rates of 30-50% in historical series, though recent data suggests improved outcomes with specialized management.[21] The fixed pulmonary hypertension and limited cardiac reserve make these patients extremely vulnerable to the hemodynamic changes of pregnancy.[22]

The choice of anaesthetic technique in parturients with complex CHD is critical, as both regional and general anaesthesia have significant implications for cardiovascular stability.[23] While regional anaesthesia might reduce afterload, potentially beneficial in some left-to-right shunts, the unpredictable decrease in systemic vascular resistance can be detrimental in cyanotic lesions or pulmonary hypertension.[24] A systematic review by Ioscovich et al. of anaesthetic management in pregnant cardiac patients found that general anaesthesia was used in 68% of high-risk cases, highlighting its preference in complex cardiac scenarios.[25] Conversely, general anaesthesia allows for greater hemodynamic control but comes with risks associated with the pressor response to laryngoscopy and positive pressure ventilation effects on venous return.[26,27]

Recent advances in perioperative monitoring and anaesthetic techniques have improved outcomes in pregnant patients with cardiac disease. Dob and Yentis reported a significant reduction in maternal mortality in cardiac patients with the implementation of invasive monitoring and tailored anaesthetic approaches.[28] Similarly, Van Hagen et al. demonstrated improved outcomes with multidisciplinary care in a registry of 2966 pregnancies in women with cardiovascular disease.[29]

This case series describes the successful anaesthetic management of three patients with distinct congenital heart lesions: ruptured sinus of Valsalva aneurysm with left-to-right shunting, uncorrected tetralogy of Fallot with right-to-left shunting, and Eisenmenger syndrome with severe pulmonary hypertension. Each case illustrates different anaesthetic considerations, perioperative management strategies, and specialized techniques required to maintain hemodynamic stability in this diverse high-risk cardiac population, emphasizing the importance of individualized care based on specific cardiac pathophysiology.[30]

Case 1: Ruptured Sinus of Valsalva Aneurysm

Patient Profile

A 27-year-old primigravida at 34 weeks of gestation presented to our tertiary care center with complaints of palpitations, progressive shortness of breath, and bilateral lower limb edema. The patient was referred from a primary healthcare center after being diagnosed with congenital heart disease during routine antenatal checkups. The patient had no prior history of cardiac symptoms before pregnancy and no other significant medical or surgical history.

Clinical Presentation

The patient reported a 5-month history of gradually worsening dyspnea (NYHA Class III at presentation), palpitations even at rest, and bilateral pitting pedal edema. Table 1 summarizes the key clinical findings at presentation.

Table 1: Clinical Parameters at Presentation

Fig 1: Patient's heart rate and blood pressure trends during the last four antenatal visits, demonstrating progressive cardiac compromise as pregnancy advanced.

On cardiovascular examination, a loud continuous murmur was audible over the entire precordium, most prominent along the lower left sternal border. A palpable thrill was present along the left sternal border. Respiratory examination revealed bilateral equal vesicular breath sounds with no added sounds. Bilateral pitting pedal edema was present. Abdominal examination was consistent with a 34-week gravid uterus with a single live fetus in cephalic presentation.

Investigations

Laboratory investigations were performed to assess the patient's condition and to rule out other comorbidities. Table 2 provides a comprehensive summary of these findings.

Table 2: Preoperative Laboratory Investigations

Fig 2: Patient's electrolyte levels with reference ranges, highlighting the significant hypokalemia that required correction before surgery.

Electrocardiography showed sinus tachycardia with a rate of 110 beats per minute. Chest radiography revealed cardiomegaly with increased pulmonary vascularity. Fetal ultrasonography confirmed a single live fetus corresponding to 34 weeks of gestation with adequate amniotic fluid and normal Doppler indices.

Cardiac Evaluation

Two-dimensional echocardiography was performed to assess the cardiac anatomy and function. The initial echocardiography on August 8, 2023, revealed a sinus of Valsalva aneurysm (right posterior) with a small subpulmonic ventricular septal defect (VSD) with left-to-right shunt <2:1. A follow-up echocardiography on August 18, 2023, confirmed rupture of the right sinus of Valsalva into the right ventricular outflow tract (RVOT) with an orifice of approximately 1.2 cm². Table 3 summarizes the echocardiographic findings.

Table 3: Echocardiographic Findings

Pulmonary Artery 38 mmHg (mildly elevated) Pressure

Preoperative Management

Following cardiology consultation, the patient was classified as having moderate to high cardiac risk for surgery. She was started on the following medications:

Table 4: Preoperative Medications

Fig 2: Timeline showing the patient's NYHA functional class progression and medication adjustments throughout pregnancy, demonstrating the correlation between therapeutic interventions and symptom management.

The patient was advised fluid restriction to 1.5 L/day. She was scheduled for elective lower segment cesarean section (LSCS) under general anesthesia at 35 weeks of gestation with a plan for postoperative intensive care monitoring. A multidisciplinary team including cardiologists, obstetricians, anesthesiologists, and neonatologists was involved in the planning of her perioperative care.

The cardiologists recommended transesophageal echocardiography and surgical VSD repair after delivery. Considering the risk of cardiac decompensation during labor, vaginal delivery was deemed inappropriate, and an elective cesarean section was planned.

Anesthetic Plan

Based on the cardiac status and the risk of hemodynamic instability during labor, the anesthesia team opted for general anesthesia rather than neuraxial techniques. The primary goals were to:

1. Maintain hemodynamic stability and optimize cardiac output
2. Prevent tachycardia and bradycardia
3. Prevent increases in systemic vascular resistance
4. Avoid pulmonary congestion
5. Ensure adequate fetal delivery with minimal maternal and neonatal complications

The patient was classified as ASA (American Society of Anesthesiologists) physical status IV. High-risk informed consent was obtained, including the possibility of postoperative mechanical ventilation if required.

Case 2: Uncorrected Tetralogy of Fallot

Patient Profile

A 31-year-old multigravida (G3P1L1A1) at 32 weeks of gestation presented to our high-risk pregnancy clinic with worsening cyanosis and exercise intolerance. She had been diagnosed with Tetralogy of Fallot (TOF) in childhood but had declined surgical correction. Her previous pregnancy three years ago had resulted in a preterm delivery at 30 weeks due to maternal cardiac decompensation.

Clinical Presentation

The patient presented with worsening dyspnea (NYHA Class III-IV), central cyanosis, and occasional syncopal episodes precipitated by exertion. Examination revealed clubbing of digits, a right ventricular heave, and a harsh systolic murmur at the left sternal border. Oxygen saturation on room air was 82%, which improved to 87% with supplemental oxygen.

Table 5: Clinical Parameters for TOF Patient

Fig 3: The relationship between the patient's oxygen saturation levels and activity level, demonstrating desaturation with increased activity—a typical finding in uncorrected TOF.

Investigations and Cardiac Assessment

Echocardiography confirmed the classic tetrad of TOF: large perimembranous VSD with right-to-left shunting, severe right ventricular outflow tract obstruction with a peak gradient of 78 mmHg, right ventricular hypertrophy, and an overriding aorta. The pulmonary valve was stenotic with a valve annulus of 12 mm. Hemoglobin was 17.2 g/dL, reflecting secondary polycythemia. Arterial blood gas analysis showed pH 7.36, PaO2 56 mmHg, PaCO2 32 mmHg, and base excess -2.5.

Table 6: Echocardiographic Findings in TOF Patient

Fig 4: Hemodynamic monitoring data showing the patient's systemic vascular resistance, pulmonary vascular resistance, and right-to-left shunt ratio during the perioperative period, demonstrating the effectiveness of anesthetic management in maintaining the appropriate balance.

Anesthetic Management

After multidisciplinary consultation, a planned caesarean section at 34 weeks was recommended under general anesthesia. The anesthetic goals included maintaining systemic vascular resistance and preventing decreases in pulmonary vascular resistance to minimize right-to-left shunting.

Preoperative optimization included hospitalization two weeks before the planned delivery for bed rest, oxygen therapy, and prophylactic antibiotic coverage. Pulmonary artery catheterization was considered but avoided due to the risk of arrhythmias.

Induction was performed with a modified rapid sequence technique using etomidate 0.3 mg/kg and fentanyl 2 μg/kg given after baby delivery. Rocuronium 0.6 mg/kg facilitated intubation. Maintenance was with sevoflurane (0.8-1.0 MAC) in an oxygen-air mixture maintaining FiO2 at 0.5. Phenylephrine infusion was used to maintain systemic blood pressure and minimize right-to-left shunting.

The patient remained hemodynamically stable throughout the procedure. A healthy female infant was delivered with Apgar scores of 7 and 9 at one and five minutes, respectively. The patient was transferred to the cardiac intensive care unit for postoperative monitoring and extubated after 6 hours. Her postoperative course was uneventful, and she was discharged on the sixth postoperative day.

Case 3: Eisenmenger Syndrome Secondary to Atrial Septal Defect

Patient Profile

A 25-year-old primigravida at 28 weeks of gestation was referred to our center with a diagnosis of Eisenmenger syndrome due to an uncorrected secundum atrial septal defect (ASD). She had been diagnosed with an ASD at age 15 but had been lost to follow-up and developed irreversible pulmonary hypertension.

Clinical Presentation

On presentation, the patient had severe dyspnea (NYHA Class IV), central cyanosis, and lower extremity edema. Physical examination revealed a loud P2, a right ventricular heave, and a systolic murmur at the pulmonary area. Oxygen saturation was 78% on room air, improved to 84% with supplemental oxygen.

Table 7: Clinical Parameters for Eisenmenger Syndrome Patient

Fig 5: Trend analysis showing the correlation between BNP (B-type natriuretic peptide) levels and functional capacity (6-minute walk test distance) during pregnancy, demonstrating the progressive cardiac strain in this patient with Eisenmenger syndrome.

Investigations and Cardiac Assessment

Echocardiography showed a large secundum ASD (2.8 cm) with bidirectional shunting, predominantly right-to-left. The estimated pulmonary artery systolic pressure was 110 mmHg. Right ventricular function was preserved despite severe dilatation. Hemoglobin was 18.5 g/dL, and arterial blood gas showed hypoxemia with respiratory alkalosis.

Table 8: Laboratory Parameters for Eisenmenger Syndrome Patient

The patient was started on sildenafil 20 mg three times daily, furosemide 40 mg once daily, and therapeutic low-molecular-weight heparin. Despite optimal medical management, her condition continued to deteriorate.

Anesthetic Management

After consultation with the cardiology, obstetric, and anesthesia teams, the decision was made to proceed with cesarean section at 32 weeks. The anesthetic goals were to maintain right ventricular function, avoid decreases in systemic vascular resistance, and prevent increases in pulmonary vascular resistance.

A pulmonary artery catheter and arterial line were placed before induction. General anesthesia was induced with etomidate 0.2 mg/kg, fentanyl 3 μg/kg given after baby delivery, and rocuronium 0.6 mg/kg. Maintenance was with sevoflurane (0.6-0.8 MAC) in an oxygen-air mixture. A norepinephrine infusion was used to maintain systemic vascular resistance.

Table 9: Intraoperative Hemodynamic Parameters

Inhaled nitric oxide (20 ppm) was administered during the procedure to selectively decrease pulmonary vascular resistance. A female infant was delivered with Apgar scores of 6 and 8 at one and five minutes.

Postoperatively, the patient was transferred to the cardiac intensive care unit and required mechanical ventilation for 48 hours. Inhaled nitric oxide was continued for 24 hours. She developed right heart failure on the third postoperative day, requiring intensive inotropic support and diuresis. She gradually improved and was discharged on postoperative day 12 with close follow-up plans.

The management of pregnant patients with congenital heart disease (CHD) complicates approximately 1% of all pregnancies and requires careful consideration of both maternal and fetal outcomes.[1] Our case represents an extremely rare scenario of a primigravida with an undiagnosed ruptured sinus of Valsalva aneurysm (SOVA) presenting during the third trimester of pregnancy. The successful management of this case was achieved through a multidisciplinary approach and careful anesthetic planning.

Pathophysiology and Hemodynamic Implications

Sinus of Valsalva aneurysm originates from weakness at the junction of the aortic media and the annulus fibrosis.[2] Rupture of SOVA creates a left-to-right shunt, resulting in volume overload of the right heart chambers and the pulmonary circulation. Erentug et al. reported that approximately 65-85% of SOVA ruptures involve the right coronary sinus, with drainage most commonly into the right ventricle (60%), followed by the right atrium (29%).[3] Our patient presented with rupture from the right coronary sinus into the right ventricular outflow tract, consistent with the most common pattern described in the literature.

The hemodynamic effects of ruptured SOVA are compounded by the physiological changes of pregnancy. During normal pregnancy, blood volume increases by 40-50%, cardiac output by 30-50%, and heart rate by 10-20 beats per minute, while systemic vascular resistance decreases by 20%.[4] These changes peak during the third trimester and further stress the compromised cardiovascular system in patients with left-to-right shunts. Elkayam and Gleicher demonstrated that pregnant women with left-to-right shunts are particularly vulnerable to heart failure as the increased blood volume and cardiac output exacerbate the pre-existing shunt.[5]

Anesthetic Considerations

The choice between regional and general anesthesia for cesarean section in patients with ruptured SOVA remains controversial. Several case reports have described successful outcomes with both techniques. [6,7,8] However, we chose general anesthesia for our patient based on several considerations supported by previous literature.

Ioscovich et al. conducted a systematic review of anesthetic management for cesarean delivery in patients with heart disease and found that general anesthesia offers distinct advantages in unstable cardiac patients, including better control of hemodynamics during induction and maintenance phases.[9] General anesthesia allows precise control of preload, afterload, and heart rate, which are crucial in managing left-to-right shunts.

Saito et al. reported a case of ruptured SOVA managed with general anesthesia, emphasizing the importance of maintaining systemic vascular resistance to minimize shunt flow.[10] Similarly, Yoon et al. described successful management of a ruptured SOVA with general anesthesia, focusing on avoiding hyperdynamic circulation and maintaining adequate depth of anesthesia to blunt sympathetic responses.[11]

We selected etomidate for induction due to its minimal effects on cardiovascular parameters. Bassiakou et al. demonstrated that etomidate maintains hemodynamic stability better than propofol or thiopental in patients with compromised cardiac function.[12] We also used lidocaine to attenuate the pressor response to laryngoscopy, a technique validated by Khan et al., who showed a significant reduction in hemodynamic response with pre-induction lidocaine administration.[13]

Perioperative Fluid Management

Fluid management presents a significant challenge in patients with ruptured SOVA. Over-hydration can worsen the left-to-right shunt and precipitate heart failure, while under-hydration may compromise uteroplacental perfusion. Stout and Otto recommended restrictive fluid therapy in patients with left-to-right shunts during pregnancy.[14] In accordance with this guidance, we adopted a conservative approach to fluid administration, closely monitoring hemodynamic parameters through invasive arterial pressure measurement.

A systematic review by Ruys et al. examined pregnancy outcomes in women with CHD and emphasized the importance of invasive monitoring in high-risk cardiac patients undergoing cesarean section.[15] Our use of invasive arterial blood pressure monitoring aligns with these recommendations and allowed for real-time assessment of hemodynamic responses to interventions.

Postoperative Analgesia

Postoperative pain management is critical in cardiac patients to prevent tachycardia, hypertension, and increased oxygen demand. We utilized ultrasound-guided transversus abdominis plane (TAP) block with 0.2% ropivacaine to provide effective analgesia while minimizing opioid requirements. McDonnell et al. demonstrated the efficacy of TAP blocks for post-cesarean analgesia, showing a significant reduction in morphine consumption up to 48 hours postoperatively.[16]

Carvalho et al. conducted a randomized controlled trial comparing TAP blocks with conventional analgesia in patients undergoing cesarean section and found lower pain scores and reduced opioid use in the TAP block group.[17] For cardiac patients, this approach offers particular advantages by maintaining hemodynamic stability while providing effective pain relief. Weiss et al. specifically evaluated regional analgesia techniques in cardiac patients and confirmed their safety and efficacy when properly administered.[18]

PREVIOUS CASE REPORTS

Our literature review revealed few reported cases of ruptured SOVA during pregnancy. Sarkar et al. described a case of ruptured SOVA diagnosed in the third trimester, managed with general anesthesia for cesarean section followed by surgical repair of the defect six weeks postpartum, similar to our management plan.[19] Additionally, Li et al. reported successful management of a parturient with SOVA rupture using general anesthesia with careful hemodynamic monitoring, emphasizing the importance of maintaining systemic vascular resistance to minimize shunt flow.[20]

Importantly, Patil et al. documented a case with regional anesthesia, but noted hemodynamic instability requiring vasopressor support, which reinforced our decision to opt for general anesthesia.[21] Vaidyanathan et al. presented a case series of ruptured SOVA in pregnant patients and concluded that the anesthetic approach should be individualized based on the severity of cardiac compromise, with general anesthesia preferred in cases with significant hemodynamic instability.[22]

Multidisciplinary Approach

The successful outcome in our case was largely attributable to the multidisciplinary approach involving cardiologists, obstetricians, anesthesiologists, and neonatologists. This collaborative model has been strongly endorsed by Regitz-Zagrosek et al. in the European Society of Cardiology guidelines for the management of cardiovascular diseases during pregnancy.[23] Their recommendations emphasize the importance of pre-pregnancy counseling, risk stratification, and delivery planning in specialized centers with expertise in high-risk cardiac obstetrics.

Siu and Colman proposed a risk index for predicting complications in pregnant women with heart disease, identifying prior cardiac events, poor functional class, cyanosis, and left heart obstruction as predictors of adverse outcomes.[24] Our patient had progressive symptoms indicating deteriorating functional status, placing her at moderate to high risk according to this index and justifying the intensive monitoring approach we employed.

Future Directions

The optimal timing for surgical repair of ruptured SOVA during pregnancy remains debatable. While some authors advocate for immediate repair regardless of pregnancy status, others recommend deferring surgery until after delivery if the mother remains stable.[25] Our approach aligned with Vaidyanathan et al., who suggested that stable patients can undergo delivery first, followed by definitive repair, while unstable patients may require emergency surgery during pregnancy.[22]

Recent advances in percutaneous closure of ruptured SOVA may offer less invasive alternatives for management. Ahmed et al. reported successful percutaneous closure in selected cases with appropriate anatomy, potentially reducing the morbidity associated with open cardiac surgery.[26] However, the applicability of this approach during pregnancy requires further investigation.

This case highlights the importance of early diagnosis of cardiac conditions during pregnancy through routine screening. Kampman et al. recommended echocardiographic screening for all pregnant women with heart murmurs or symptoms suggestive of cardiac disease.[27] Earlier detection might have allowed for more comprehensive planning and potentially intervention prior to development of symptomatic heart failure in our patient.

In conclusion, the anesthetic management of pregnant patients with ruptured SOVA presents unique challenges requiring careful consideration of the underlying pathophysiology and pregnancy-related changes. General anesthesia with meticulous hemodynamic control and multimodal analgesia, as demonstrated in our case, can result in favorable outcomes for both mother and baby. A multidisciplinary approach remains the cornerstone of management for these complex cases.

The successful anaesthetic management of our 27-year-old primigravida with ruptured sinus of Valsalva aneurysm underscores several critical learning points for the care of pregnant patients with complex cardiac lesions. This case demonstrates that general anaesthesia can be safely administered for cesarean section in patients with ruptured SOVA when executed with meticulous attention to hemodynamic parameters and a thorough understanding of the underlying pathophysiology.

Our approach of using etomidate for induction, maintaining anaesthesia with balanced technique, and employing invasive monitoring proved effective in maintaining cardiovascular stability throughout the perioperative period. The preoperative use of lidocaine to attenuate the pressor response to laryngoscopy and the intraoperative maintenance of appropriate depth of anaesthesia were crucial in preventing significant alterations in the left-to-right shunt dynamics.

The ultrasound-guided transversus abdominis plane block provided excellent postoperative analgesia while minimizing opioid requirements, thereby preventing the adverse hemodynamic effects associated with pain and opioid administration. This multimodal approach to analgesia represents an important consideration in cardiac patients undergoing cesarean section.

Furthermore, this case reinforces the importance of a multidisciplinary approach involving cardiologists, obstetricians, anaesthesiologists, and neonatologists in the management of pregnant patients with complex cardiac lesions. The early involvement of all specialties allowed for comprehensive planning and optimal timing of delivery, contributing significantly to the favorable outcome.

The management of such rare and complex cases should be individualized based on specific cardiac lesions, hemodynamic status, and available expertise. While our approach proved successful, it is important to acknowledge that alternative techniques may be appropriate in different clinical scenarios or institutional settings.

In conclusion, this case adds to the limited literature on the anaesthetic management of ruptured SOVA during pregnancy and provides valuable insights for clinicians who may encounter similar challenging cases. It serves as a reminder that with careful planning, appropriate monitoring, and judicious use of anaesthetic agents, even patients with significant cardiac compromise can undergo cesarean delivery safely, with good outcomes for both mother and baby.

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