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Real-time three-dimensional trans-oesophageal echocardiography-guided balloon dilation of pulmonary venous pathway obstruction in a patient with dextro-transposition of the great arteries after atrial switch surgery: a case report

Part of:Interventional Cardiology

Published online by Cambridge University Press:  24 June 2019

Manabu NittaOpen the ORCID record for Manabu Nitta[Opens in a new window],
Teruyasu Sugano and
Kiwamu Iwata
Manabu Nitta*
Affiliation:
Department of Medical Science and Cardiorenal Medicine, Yokohama City University Graduate School of Medicine, Yokohama, Japan
Teruyasu Sugano
Affiliation:
Department of Medical Science and Cardiorenal Medicine, Yokohama City University Graduate School of Medicine, Yokohama, Japan
Kiwamu Iwata
Affiliation:
Department of Medical Science and Cardiorenal Medicine, Yokohama City University Graduate School of Medicine, Yokohama, Japan
*
Author for correspondence: Manabu Nitta, MD, Department of Medical Science and Cardiorenal Medicine, Yokohama City University Graduate School of Medicine, 3-9, Fukuura, Kanazawa, Yokohama, 236-0004, Japan. Tel: +81-45-787-2635; Fax: +81-45-701-3738. E-mail: n0714.got.mail@gmail.com
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Abstract

A percutaneous transcatheter balloon dilation of a pulmonary venous pathway obstruction was successfully performed in a 40-year-old patient after a Mustard procedure. During the procedure, real-time three-dimensional trans-oesophageal echocardiography demonstrated the morphology of the obstruction. Our case highlights the usefulness of real-time three-dimensional trans-oesophageal echocardiography as a guide for transcatheter intervention in the increasing number of adults with CHD.

Keywords

Dextro-transposition of the great arteriesmustard surgerypulmonary venous obstructionreal-time three-dimensional trans-oesophageal echocardiographytranscatheter balloon dilation

Information

Type
Brief Report
Information
Cardiology in the Young , Volume 29 , Issue 7 , July 2019, pp. 983 - 985
Copyright
© Cambridge University Press 2019 

Dextro-transposition of the great arteries is a congenital anomaly with ventriculoarterial discordance. Mustard and colleagues first reported on atrial switch surgery, the so-named “Mustard procedure”, which converts the parallel circulations of dextro-transposition of the great arteries into a circulation in series, thereby correcting cyanosis.Reference Mustard1 Pulmonary venous obstruction is one of the baffle-associated complications after a Mustard procedure, which induces pulmonary congestion and pulmonary hypertension.Reference Champsauer, Sokol and Trusler2, Reference Hagler, Donald and Douglas3 Real-time three-dimensional trans-oesophageal echocardiography (3D-TOE) provides useful information for recognising complex intra-cardiac abnormalities. To the best of our knowledge, this is the first case report that demonstrates the use of real-time 3D-TOE as guidance for the balloon dilation of pulmonary venous pathway obstruction after a Mustard procedure.

Case

The patient was a 40-year-old male who experienced dyspnoea upon exertion. Soon after birth he was diagnosed with dextro-transposition of the great arteries and at the age of 3 months underwent a Mustard procedure. Cardiac catheterisation demonstrated a 14.2 mmHg mean pressure gradient across the pulmonary-venous-obstructed site. A cross-sectional area of the pulmonary venous pathway obstruction was measured as 0.62 cm2 by Gorlin’s equation and a cardiac index of 1.65 L/min/m2 (Fig 1a). In addition, his systemic right ventricular (RV) function was impaired at 38% of ejection fraction. Surgery seemed too invasive, so the patient was offered percutaneous transcatheter balloon dilation of the pulmonary venous pathway obstruction.

Figure 1.

(a) Simultaneous measurement of both PAWP and RV pressure revealed 14.2 mmHg of mean diastolic pressure gradient across the obstructed portion in the pulmonary venous atrium. A cross-sectional area of the obstructed pulmonary venous portion was measured as 0.62 cm2 by Gorlin’s equation. (b) After balloon dilation, the mean pressure gradient across the pulmonary venous obstruction site decreased to 6.6 mmHg and the cross-sectional area enlarged to 1.24 cm2. (c) This angiography shows the right-anterior oblique view, in which contrast medium was injected from the left upper pulmonary vein. A pigtail catheter was passed through the aortic valve, tricuspid valve, and pulmonary venous obstruction site sequentially into the left upper pulmonary vein. (d) A 20-mm VACS balloon was inflated at the obstructed portion in the pulmonary venous atrium. PAWP= pulmonary artery wedge pressure; RV= right ventricular.

The procedure was performed under general anaesthesia with fluoroscopy and a TOE guide. Because of the occlusion of the bilateral femoral veins, a retrograde arterial approach via the right femoral artery was used to access the pulmonary venous atrium. A 10F sheath was inserted and a guide wire passed sequentially through the aortic valve, tricuspid valve, and pulmonary-venous-obstructed portion into the left pulmonary vein (Fig 1c, Movie 1). Real-time 3D-TOE provided a clear image of the pulmonary-venous-obstructed section as a circle-like shape, the right half of which was covered with a membranous tissue (Fig 2a, Movie 2). The membranous tissue seemed very thin and floppy, and therefore could easily be broken and enlarged by balloon dilation. In addition, real-time 3D-TOE clearly demonstrated the spatiality of the membranous tissue, which two-dimensional trans-oesophageal echocardiography (2D-TOE) could not provide. The minimum diameter was 5.7 mm with a reference diameter of 21 mm (Fig 2b); therefore, a 20-mm VACS (Valvuloplasty Balloon Catheters) balloon (OSYPKA AG, Germany) was inflated. After the procedure, the mean pressure gradient decreased to 6.6 mmHg and the cross-sectional area enlarged to 1.24 cm2 (Fig 1b). The patient’s cardiac index improved to 2.10 L/min/m2. Real-time 3D-TOE after the procedure showed an enlarged pulmonary venous atrium in which the membranous tissue was broken (Figs 2c, D, Movie 3).

Figure 2.

(a) Real-time 3D-TOE before the procedure provided an antero-posterior view towards the stenotic portion of the pulmonary venous atrium via the tricuspid valve. It revealed a circle-like shape, the right half of which was covered with a membranous tissue. (b) 2D-TOE before the procedure revealed a 5.7 mm of minimum diameter of the obstructed portion. (c) Real-time 3D-TOE after balloon dilation showed an enlarged pulmonary venous atrium in which the membranous tissue was broken. 3D-TOE= three-dimensional trans-oesophageal echocardiography; 2D-TOE= two-dimensional trans-oesophageal echocardiography.

Discussion

In Japan, the Mustard procedure – an atrial switch surgery for dextro-transposition of the great arteries – was first reported on in 1969 and Jatene surgery, an arterial switch procedure, was initially reported on in 1976. The incidence of pulmonary venous pathway obstruction after a Mustard procedure is reported as being 3–18%.Reference Champsauer, Sokol and Trusler2, Reference Hagler, Donald and Douglas3 In general, it is treated by additional surgery.Reference Cooper, Sullivan and Bull4 However, for some patients considered a high surgical risk, a catheter-based treatment would be indicated.Reference Abdulhamed, Alyousef and Mullins5 A transeptal puncture through the femoral vein is another option for accessing the pulmonary venous atrium.Reference Abdulhamed, Alyousef and Mullins5 Our patient was at high risk for further surgery because of low systemic RV function and occlusion of his bilateral femoral veins would make transeptal puncture difficult.

In previous reports, the use of real-time 3D-TOE for the assessment of dextro-transposition of the great arteries, such as baffle leak or systemic venous obstruction, has been demonstrated.Reference Ahmed, Nekkanti and Nanda6, Reference Cua, Kollins and Roble7 In general, when dealing with baffle obstruction after a Mustard procedure, interventional cardiologists usually use fluoroscopy or intra-cardiac echocardiography not equipped with a three-dimensional function. Compared to the conventional two-dimensional image, our three-dimensional image revealed the characteristics of the stenosis in the pulmonary venous atrium more precisely, which could be easily dilated with balloon.

Conclusion

Real-time 3D-TOE is useful for evaluating the morphology and character of pulmonary venous pathway obstruction and guiding transcatheter intervention in patients with dextro-transposition of the great arteries after atrial switch surgery.

Supplementary Material

To view supplementary material for this article, please visit https://doi.org/10.1017/S1047951119001045.

Author ORCIDs

Manabu Nitta, 0000-0003-2834-1248

Acknowledgements

None.

Financial Support

This report received no specific grant from any funding agency, commercial or not-for-profit sectors.

Conflict of Interest

None.

Ethical Standards

The authors assert that all procedures contributing to this work comply with the ethical standards of the relevant national guidelineson human experimentation (please name) and with the Helsinki Declaration of 1975, as revised in 2008, and has been approved by the institutional committees (Yokohama City University Graduate School of Medicine).

References

Mustard, WT. Successful two-stage correction of transposition of great vessels. Surgery 1964; 55: 469–472.Google Scholar
Champsauer, GL, Sokol, DM, Trusler, GA, et al. Repair of transposition of the great arteries in 123 pediatric patients. Early and long-term results. Circulation 1973; 47: 1032–1041.CrossRefGoogle Scholar
Hagler, DJ, Donald, GR, Douglas, DM, et al. Clinical, angiographic and hemodynamic assessment of late results after Mustard operation. Circulation 1978; 57: 1214–1220.CrossRefGoogle ScholarPubMed
Cooper, SG, Sullivan, ID, Bull, C, et al. Balloon dilation of pulmonary venous pathway obstruction after Mustard repair for transposition of the great arteries. J Am Coll Cardiol 1989; 14: 194–198.CrossRefGoogle ScholarPubMed
Abdulhamed, JM, Alyousef, SA, Mullins, C, et al. Endovascular stent placement for pulmonary venous obstruction after Mustard operation for transposition of great arteries. Heart 1996; 75: 210–212.CrossRefGoogle Scholar
Ahmed, S, Nekkanti, R, Nanda, NC, et al. Three-dimensional transesophageal echocardiographic demonstration of intraatrial baffle obstruction. Echocardiography 2003; 20: 683–686.CrossRefGoogle ScholarPubMed
Cua, CL, Kollins, K, Roble, S, et al. Three-dimensional image of b baffle leak in a patient with a Mustard operation. Echocardiography 2014; 31: E315–E316.CrossRefGoogle Scholar
Figure 0

Figure 1.(a) Simultaneous measurement of both PAWP and RV pressure revealed 14.2 mmHg of mean diastolic pressure gradient across the obstructed portion in the pulmonary venous atrium. A cross-sectional area of the obstructed pulmonary venous portion was measured as 0.62 cm2 by Gorlin’s equation. (b) After balloon dilation, the mean pressure gradient across the pulmonary venous obstruction site decreased to 6.6 mmHg and the cross-sectional area enlarged to 1.24 cm2. (c) This angiography shows the right-anterior oblique view, in which contrast medium was injected from the left upper pulmonary vein. A pigtail catheter was passed through the aortic valve, tricuspid valve, and pulmonary venous obstruction site sequentially into the left upper pulmonary vein. (d) A 20-mm VACS balloon was inflated at the obstructed portion in the pulmonary venous atrium. PAWP= pulmonary artery wedge pressure; RV= right ventricular.

Figure 1

Figure 2.(a) Real-time 3D-TOE before the procedure provided an antero-posterior view towards the stenotic portion of the pulmonary venous atrium via the tricuspid valve. It revealed a circle-like shape, the right half of which was covered with a membranous tissue. (b) 2D-TOE before the procedure revealed a 5.7 mm of minimum diameter of the obstructed portion. (c) Real-time 3D-TOE after balloon dilation showed an enlarged pulmonary venous atrium in which the membranous tissue was broken. 3D-TOE= three-dimensional trans-oesophageal echocardiography; 2D-TOE= two-dimensional trans-oesophageal echocardiography.

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