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\begin{document}

\title{A rare case on anomalous pulmonary venous drainage in congenital
cardiovascular disease}



\author[1]{Zhuangzhuang Lu}%
\author[1]{Xiao Bai}%
\author[1]{Changcun Fang}%
\author[1]{Guangmin Song}%
\affil[1]{Qilu Hospital of Shandong University}%


\vspace{-1em}



  
  \date{May 27, 2024}


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\begin{abstract}
anomalous pulmonary veins drain into the right side of the left atrium
is an uncommon variety of anomalous pulmonary venous return. Rarely,
anomalous pulmonary venous drainage combined with cor triatriatum and
atrial septal defect. We presented the imaging findings of a male
patient who had anomalous pulmonary venous drainage which has not
previously been described.%
\end{abstract}%



\sloppy


\textbf{A rare case on anomalous pulmonary venous drainage in congenital
cardiovascular disease}

Zhuangzhuang Lu\textsuperscript{1}, MD \textbar{} Xiao
Bai\textsuperscript{1}, PhD \textbar{} Changcun
Fang*\textsuperscript{1}, PhD \textbar{} Guangmin
Song*\textsuperscript{1}, MD, PhD.

\textsuperscript{1}Department of Cardiovascular Surgery, Qilu Hospital
of Shandong University, Jinan, China

*Co-corresponding author: Changcun Fang, PhD. Department of
Cardiovascular Surgery, Qilu Hospital of Shandong University, No. 44
Wenhua West Road, Baotu Spring Street, Lixia District, Jinan City,
250012, Shandong Province,~China. Email: fangchangcun2004@126.com.

Guangmin Song, MD, PhD. Department of Cardiovascular Surgery, Qilu
Hospital of Shandong University, No. 44 Wenhua West Road, Baotu Spring
Street, Lixia District, Jinan City, 250012, Shandong Province,~China.
Email: songgm890@163.com.

\textbf{Abstract:} anomalous pulmonary veins drain into the right side
of the left atrium is an uncommon variety of anomalous pulmonary venous
return. Rarely, anomalous pulmonary venous drainage combined with cor
triatriatum and atrial septal defect. We presented the imaging findings
of a male patient who had anomalous pulmonary venous drainage which has
not previously been described.

KEYWORDS: atrial septal defect, cor triatriatum, anomalous pulmonary
venous drainage

1 \textbar{} INTRODUCTION

This is the first report of rare simultaneous complication of three
cardiac malformations: atrial septal defect, cor triatriatum and
anomalous pulmonary veins drain into the right side of the left atrium.
A single dilated and tortuous pulmonary vein at the right side draining
into the left atrium is extremely rare and has not previously been
described. We successfully operated to repair the atrial septal defect
and correct the cor triatriatum. The study was conducted in accordance
with the Declaration of Helsinki (as revised in 2013). The study was
approved by the Scientific Research Ethics Committee of Qilu Hospital of
Shandong University and individual consent was waived in accordance with
local policies about case reports.

\textbar{} Case report

An 18-year-old male patient presented with a systolic murmur in the
second and third intercostal spaces at the left sternal border during
physical examinations. His transthoracic echocardiogram showed an atrial
septal defect (ASD) with left to right shunt and unclear drainage of
pulmonary veins. Moreover, there was a deformed septum in the left
atrium, which divided the left atrium into two parts (the true left
atrium and an accessory atrium) communicating through a 7mm-diameter
hole. The echocardiogram showed that preoperative pulmonary artery
systolic pressure was about 49 mmHg.

We successfully operated on the patient for closure of the ASD and
correction of the cor triatriatum by removing the deformed septum in the
left atrium. However, the patient was explored with a single tortuous
pulmonary vein draining into the top right corner of the left atrium
during surgery. Nine months after surgery, the patient underwent
computerized tomography angiography (CTA) for follow-up study. Review of
CTA images revealed the presence of a single dilated and tortuous
pulmonary vein at the right side draining into the left atrium. In
addition, a second pulmonary vein at the left side was found to directly
merge into the right-sided pulmonary vein instead of connection with the
left atrium (Figure 1). The transthoracic echocardiogram showed that
pulmonary artery systolic pressure was about 25 mmHg after surgery.

2 \textbar{} DISCUSSION

Both sides of pulmonary veins with a single opening in the left atrium
presented in this case is a rare variety of anomalous pulmonary venous
drainage, which is very common in patients with heterotaxia. But in
patients with normal visceroatrial situs, it usually occurs as an
isolated deformity and barely combined with other complicated congenital
heart diseases such as cor triatriatum\textsuperscript{1}. Previous
studies showed that anomalous drainage of left-sided pulmonary veins is
more common than right-sided pulmonary veins\textsuperscript{2}. To the
best of our knowledge, this is the first report of anomalous pulmonary
venous drainage combined with cor triatriatum and atrial septal defect.
The goal of corrective surgery in patients with anomalous pulmonary
venous drainage, is to eliminate left to right shunt, pulmonary
hypertension and re-establish the structure of the
atrium\textsuperscript{3}. With the pattern of anomalous pulmonary
venous drainage shown in our patient, we decided not to correct the
dilated and tortuous right-sided pulmonary vein because the deformed
vein can normally drain into left atrium and the surgery also can reduce
the pulmonary artery systolic pressure that bring us good clinical
outcomes. The success of this procedure might provide a valuable
reference for surgeons in the future clinical practice. For those
patients with anomalous pulmonary venous drainage into the left atrium
as well as cor triatriatum and atrial septal defects, the pulmonary
artery pressure could be decreased only by repairing the atrial defect
and removing the diaphragm in the left atrium if the patients have
moderate pulmonary hypertension (PAH) before surgery. Furthermore,
pulmonary vein CTA can provide more precise anatomical details to help
confirm the clinical diagnosis.

3\textbar{} CONCLUSION

Both sides of pulmonary veins with a single opening in the left atrium
combined with other complex congenital heart diseases is rarely. The
variety of anomalous pulmonary venous drainage is an extremely rare
abnormality. CTA is very useful to accurately determine the paths of
drainage of pulmonary veins\textsuperscript{3}.

AUTHOR CONTRIBUTIONS

Changcun Fang performed the surgery. Zhuangzhuang Lu and Xiao Bai
prepared the manuscript. Guangmin Song and Changcun Fang revised the
manuscript critically. All authors read and approved the final
manuscript.

FUNDING

This research was supported by the National Natural Science Foundation
of China (81873510) and Youth Foundation of Qilu Hospital of Shandong
University(2019QLQN21).

CONFLICT OF INTERESTS

The authors report no conficts.

ORCID

Zhuangzhuang Lu https://orcid.org/0000-0002-6043-7461

REFERENCES

1.Brown DW, Geva T. Anomalies of the pulmonary veins. In: Allen HD,
Driscol DJ, Shaddy RE, Feltes TF (eds). Moss and Adams' Heart Disease I
Infants, Children, and Adolescents: Including the Fetus and Young Adult.
Vol 2, 9th ed. Lippincott Williams \& Wilkins, Philadelphia, 2016;
881--910.

2.Rajagopal R, Pandey NN, Sinha M, Sharma S. Cardiac type of partial
anomalous pulmonary venous connection. Radiol Cardiothorac Imaging.
2020; 2(1): e190100.

3.Sharma~S, Das~A, Deora~S,~Rajagopal~R. ``Dual'' drainage in cardiac
partial anomalous pulmonary venous return. Journal of Cardiac Surgery.
2022; 37: 1422--1424.

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