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CASE REPORT
Year : 2008  |  Volume : 52  |  Issue : 6  |  Page : 853 Table of Contents     

Cardiac Catheterization in Thoraco-Omphalocardiopagus Twins: A Case Report


1 Associate Professor, Department of Cardiac Anaesthesia, Cardiothoracic Sciences centre, All India Institute of Medical Sciences, New Delhi-110029, India
2 Professor & HOD, Department of Cardiac Anaesthesia, Cardiothoracic Sciences centre, All India Institute of Medical Sciences, New Delhi-110029, India

Date of Acceptance28-Oct-2008
Date of Web Publication19-Mar-2010

Correspondence Address:
Usha Kiran
Department of Cardiac Anaesthesia, Cardiothoracic Sciences centre, All India Institute of Medical Sciences, New Delhi-110029
India
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Source of Support: None, Conflict of Interest: None


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The incidence of conjoined twin is rare and anaesthesia for procedures on conjoined twins is a demanding, exacting and meticulous exercise, whether prior to or during separation.
literature on the anaesthetic management of these cases is sparse. The following case report details the expert and vigilant anaesthetic management leading to successful diagnostic cardiac catheterization. The report emphasizes the importance of synchronous ventilation, teamwork and communication required in cases such as this. This case report also details the difficulties encountered and how to overcome them during the prolonged procedure.

Keywords: Conjoined twins, Cardiac catheterization


How to cite this article:
Choudhury M, Kiran U. Cardiac Catheterization in Thoraco-Omphalocardiopagus Twins: A Case Report. Indian J Anaesth 2008;52:853

How to cite this URL:
Choudhury M, Kiran U. Cardiac Catheterization in Thoraco-Omphalocardiopagus Twins: A Case Report. Indian J Anaesth [serial online] 2008 [cited 2020 Jan 28];52:853. Available from: http://www.ijaweb.org/text.asp?2008/52/6/853/60701


   Introduction Top


The earliest known record of conjoined twins is about the Biddendon girls born in 1100 AD in Kent. [1] Since then there is increasing number of attempts to surgically separate them. But till today in most of the cases their separation is not feasible. However they continue to fascinate the medical personnel and gen­eral public. Recent advances regarding the diagnosis and anaesthetic management of these cases has been introduced in the 1980s. They often have complex cardiovascular anomalies for which a thorough preopera­tive cardiac evaluation is a must, before taking the de­cision regarding their separation. [2],[3] Different anaesthetic techniques have been described by various authors for the management of these cases during their surgical separation. [4],[5],[6] But to our knowledge there is no report regarding the anaesthetic management of these cases during a diagnostic cardiac catheterization procedure which is also not a less challenging one. We describe our experience concerning the management of thoraco-omphalocardiopagus twins during the periods of pro­longed cardiac catheterization.


   Case report Top


A pair of male conjoined twins, twin-1 and twin­II was delivered by emergency caesarean section de­livery at 39 weeks of gestation. Clinically they were diagnosed as thoraco-omphalocardiopagus [Figure 1] .

The combined weight was 4300 grams (both the twins appeared to have equal weight). The APGAR score of twin-I was 3, 8 and that of twin-II was 6, 8 at 1 and 5 minutes respectively. They were nursed in an incubator and in the meanwhile, investigated and prepared for surgical separation. Haematological and biochemical tests were found to be within normal limits [Table 1] and approximately equal in both the twins. Both of them had oxygen saturations of 65-75% on a 4 L.min -1 of flow by oxygen.

Ultrasound abdomen suggested a possible fusion of livers, billiary tract and gut. Due to technical difficul­ties for stabilizing the echocardiographic transducer, a proper imaging plane could not be obtained. However; the transthoracic echocardiography revealed the pres­ence of two separate double chambered atrium, three ventricles and a close connection between the twins heart. Twin-1 was intubated at the 16 th hours of his birth due to the development of acute respiratory in­sufficiency and gradually weaned off from ventilatory support within 24 hour period of intubation. Diagnostic cardiac catheterization was planned after their initial stabilization. Both the twins were found to be conscious, active and had spontaneous respiration at the time they were shifted from the paediatric nursery to the cardiac catheterization laboratory. On arrival in the cardiac cath­eterization laboratory they were found to be tachypnoeic. Vital signs were as follows: Twin-1 had an arterial blood pressure of 69/41 mmHg, a heart rate of 140 beats/minute, a respiratory rate of 60-70 breaths / minute, an oxygen saturation of 65% and was afe­brile. Twin-II had an arterial blood pressure of 55/30 mmHg, a heart rate of 156 beats/minute, a respiratory rate of 70-80 breaths/minute, an oxygen saturation of 70% and was also afebrile.

Continuous monitoring of oxygen saturation (SpO 2 ), blood pressure, electrocardiogram (ECG) was started in both of them. Two sets of ECG electrodes were applied, at the back of each twin. ECG revealed the presence of two 'P' waves overlapping with each other but a single QRS complex. Twin-1 had already a patent intravenous line to which maintenance fluid cham­ber was connected. Another 22 gauge intravenous cath­eter was inserted on twin-II's right hand. Both the twins were allowed to breathe spontaneously with O2: air at the ratio of 50:50 from the system. After preoxygenation for five minute 8 mg of ketamine and 0.6 mg of vecuronium bromide was administered to twin-II. Both of them were found to be paralyzed at the same time. Mask ventilation was started immediately for both of them and intubation was done subsequently followed by artificial ventilation. The twins' position (facing each other, attached chest to chest;[Figure 1] made direct laryn­goscopy extremely akward. As the heads were turned to give more room for direct laryngoscopy, there was possible distortion of the laryngeal anatomy. Twin-I was intubated in single attempt with a 3mm size uncuffed portex endotracheal tube whereas we could able to secure twin-II 's'trachea in the fourth attempt. During the process Twin-II developed one bout of laryn­gospasm which was resolved with mask ventilation and deepening of anaesthesia. Limited space in the cardiac catheterization laboratory forced us to ventilate both the babies from the gas source coming from a single anaesthesia machine. We attached one 'Y' connection at the gas source from the machine end to which two non-rebreathing Jackson-ree's modification of Ayres 'T' piece system were attached [Figure 2]. Both the twins were ventilated manually by one anaesthesiologist.The fresh gas flow was 10 liters/minute. Single dose of mor­phine hydrochloride (1 mg) was used as the sole analgesic. Anaesthesia was maintained with incremental doses of midazolam and vecuronium bromide. Oxy­gen: air was administered at the ratio of 40:60. The heart rate, blood pressure and oxygen saturation of both the babies were found to be equal at all the time and maintained near the base line value through out the pro­cedure. The arterial blood gas (ABG) values of both of them were exactly the same at different time intervals and revealed no abnormalities [Table 2]. Cardiac cath­eterization demonstrated the presence of separate two chambered atrium for both the twins, four ventricles having interconnection between them through septal defects. In addition to these twin-1 had total anoma­lous pulmonary venous connection and coarctation of aorta. A patent ductus arteriosus and pulmonary stenosis were found in twin-II. Fluid replacement was done ac­cording to the blood pressure and central venous pres­sure monitoring.

Inspite of administration of all the drugs and fluid through twin-II's intravenous line, we did not find any circulatory and ABG disparity at any point of time among the twins. The total duration of the procedure was three hours. At the end, they were found to be conscious, started spontaneous breathing though their effort was not adequate. They were shifted to the car­diac intensive care unit (ICU) and put on SIMV mode and gradually weaned off within 5 hours period. On reaching the ICU the ABG was repeated and no de­viation from the base line value was found.


   Discussion Top


Conjoined twins are said to be the result of an incomplete division of embryo between the thirteenth and fifteenth day of fertilization. [7] Thoracopagus twins having cardiovascular anomalies account for 75% of the general cases. [8] A list of authors described the best possible anaesthetic management of these cases during their surgical separation. However, there are no guide­lines for the management of these cases during a pro­longed cardiac catheterization; which is an important diagnostic mode. This procedure itself is not free of any risk. Major complications included death, myo­cardial infarction and cerebrovascular complication fol­lowed by cardiac perforation, arrhythmia, local vascu­lar problem, vasovagal reaction and allergy due to con­trast media. All of these complications are more pro­nounced in infants, complex cardiac defects, valvular heart disease and left ventricular dysfunction. [9] Again each case of conjoined twins have its unique differ­ences, hence their clinical behavior during the anaes­thetic management may vary from case to case. Seda­tion without the induction of general anaesthesia has been successfully performed in children for diagnostic cardiac catheterization studies. [10] However general ana­esthesia is often required in high risk patients. [11] Ac­cordingly, we chose to provide general anaesthesia to those babies with their airway secured with endotra­cheal tubes.

Administration of anaesthetic agents to one twin led to sedation and paralysis of the other twin which we never expected and not described before. Due to the presence of vigilant team we could able to over­come this problem. Though before the procedure both the twins were haemodynamically stable and oxygen­ating satisfactorily, still we were in doubt regarding their behavior with anaesthesia induction. Finally, we were unsure how the twins would respond to positive pres­sure ventilation changes and did not know whether ven­tilation synchrony was warranted. Review of earlier research revealed that anaesthesia for cardiac catheter­ization procedure should provide rapid induction and emergence along with a reliable sedation state. [12] Both intravenous and inhaled anaesthetic agent along with a short acting muscle relaxant may fulfill these require­ments. [12] In addition to the demands of cardiac cath­eterization laboratory environment, we needed to con­sider the complex cardiac physiology of the twins.

While studying the effect of different anaesthetic agents in children having complex cardiac anatomy, Rivenes et al found that sevoflurane and isoflurane main­tain cardiac output, but both the agents decreased car­diac contractility. [13] Williams et al stated that in pres­ence of pulmonary hypertension continuous infusion of ketamine anaesthesia for diagnostic cardiac catheter­ization did not increase the pulmonary vascular resis­tance in children with severe pulmonary hypertension. [14] Propofol- ketamine combination has been reported to be a reasonably good combination in catheterization suite in spontaneously breathing children. [15] However it has been reported to cause a decrease in heart rate. [15] Opioids, as a result of their haemodynamic stability have been the mainstay of pae­diatric cardiac anaesthesia for the last few decades. [16]

Except the general problems faced by a paediat­ric cardiac anaesthesiologist the major concern in these twins with a shared heart is a complete blood exchange every minute. Due to this reason drugs administered to one infant may have unpredictable effect on other. [5],[10],[11] We agree with the previous authors but differ from them in one aspect that in spite of administration of all the drugs to one of them throughout the period, we did not find any effect due to drug overdose. Their clinical behavior remained the same during the whole course.

Because both the twins required high respiratory rates, it was necessary to hand ventilate both the twins throughout the procedure. Another reason to prefer manual ventilation over mechanical ventilation was for a better appreciation of subtle changes in lung compli­ance and airway resistance. The ventilation was man­aged by single anaesthesiologist to maintain synchrony, believing that this would decrease the likelihood of un­toward shunting. The major problems which we faced in the cardiac catheterization laboratory were over­crowding, cardiovascular monitoring, difficulty in main­tenance of airway and vascular cannulation due to the relative position of the twins. To conserve space, we used only one anaesthesia machine and single anaes­thesia team (two anaesthesiologists).

In summary, the basic principles of optimal an­aesthetic management during cardiac catheterization of thoraco-omphalocardiopagus twins (eg. airway, pres­ervation of body temperature, cardiovascular stability) are the same as their management during the surgical separation. Above all to these, addition and titration of drug doses, choice of anaesthetic agents, ventilation pattern too carries a major role for the accurate diag­nosis and interpretation of the catheterization data.

 
   References Top

1.Towey RM, Kisia AKL, Jacobacci S, Muoki M.Anaesthesia for the separation of conjoined twins. Anaesthesia 1979; 34: 184-92.  Back to cited text no. 1      
2.Brown DL, Greenberg DJ. Esophageal echocardiography: A simplified method. Anesthesiol­ogy 1983; 59: 482-3.  Back to cited text no. 2      
3.Patel R, Fox K, Dawson J, Taylor JFN, Graham GR. Car­diovascular anomalies in thoracopagus twins and the importance of pre-operative cardiac evaluation. Br Heart J 1977; 39: 1254-5.  Back to cited text no. 3      
4.Suan C,Ojeda R,Garcia-Perla JL,Cerro J,Romero D,Gilaberg J,Gonzaleza,et al.Paediatr Anaesth1998;8:255-7.  Back to cited text no. 4      
5.Chen TL,Lin CJ,Lai HS,Chen WJ,Chao CC,Liu CC.Anaesthesia management for conjoined twins with complex cardiac anomalies.Can J Anaesth 1996;43:1161-­7.  Back to cited text no. 5  [PUBMED]    
6.Furman EB, Roman DG, Hairabet J, Yokoyama M, Larmon K. Management of anaesthesia for surgical separation of new-born conjoined twins. Anesthesiology 1970; 34: 94-101.  Back to cited text no. 6      
7.Scammon RE. Fetal malformation. Pediatrics, Vol 6 In Abt 1A Ed. Philadelphia, WB Saunders 1925, pp 654.  Back to cited text no. 7      
8.Marin-Padilla M, Chin AJ, Marin Padilla TM. Cardio­vascular Abnormalities in thoracopagus twins. Teratol­ogy 1981;23:101-13.  Back to cited text no. 8      
9.Mc Crindle BW, Nykanen D, Freedom RM, Benson LN. Complications associated with pediatric cardiac cath­eterization. J Am Coll Cardiol 1998;32:1433-1440.  Back to cited text no. 9      
10.Mester R,Easley RB,Brady KM,Chilson K,Tobias JD.Monitored anaesthesia care with a combination of ketamine and dexmedetomidine during cardiac catheter­ization.Am JTher 2003;15:24-30.  Back to cited text no. 10      
11.Bernath MA,Sekarski N.Management of paediatric pa­tients undergoing diagnostic and invasive cardiology procedures. Curr Opin Anaesthesiol 2001; 14:441-446.  Back to cited text no. 11  [PUBMED]  [FULLTEXT]  
12.Tosun Z,Akin A,Guler G,Esmaoglu A,Boyaci A.Dexmedetomidine-ketamine and propofol-ketamine combination in spontaneously breathing patients un­dergoing cardiac catheterization. J Cardiothoracic Vasc Anesth 2006; 20:515-9.  Back to cited text no. 12      
13.Rivenes SM,Lewin MB,Stayer SA, et al. Cardiovascular effects of sevoflurane,isoflurane and halothane and fen­tanyl-midazolam in children with congenital heart disease;an ecocardiographic study of myocardiac con­tractility and hemodynamics.Anesthesiology 2001;94:223-9.  Back to cited text no. 13  [PUBMED]  [FULLTEXT]  
14.William GD,Philip BM,Chu LF,Boltz GB,et al. Ketamine does not increase pulmonary resistance in children with pulmonary hypertension undergoing sevoflurane ana­esthesia and spontaneous ventilation. Anesth Analg 2007; 105:1578-84.  Back to cited text no. 14      
15.Akin A,Esmaoglu A,Guler G,Demircioglu E,Narin N,Boyaci A.Propofol and propfol-ketamine in pediatric patients undergoing cardiac catheterization. Pediatr Cardiol 2005; 26:553-7.  Back to cited text no. 15      
16.Newland MC,Leuschen P,Sarafian LB,et al. Fentanyl intermittent bolus technique for anesthesia in infants and children undergoing cardiac surgery. J Cardiothorac Anesth 1989; 3:407-10.  Back to cited text no. 16  [PUBMED]    


    Figures

  [Figure 1], [Figure 2]
 
 
    Tables

  [Table 1], [Table 2]



 

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