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CASE REPORT Table of Contents  
Ahead of print publication
Role of multimodality imaging in postsurgical calcified aneurysm of patent ductus arteriosus


1 Rajaie Cardiovascular Medical and Research Center, Echocardiography Research Center, Iran University of Medical Sciences, Tehran, Iran
2 Rajaie Cardiovascular Medical and Research Center, Radiology Research Center, Iran University of Medical Sciences, Tehran, Iran
3 Rajaie Cardiovascular Medical and Research Center, Interventional Research Center, Iran University of Medical Sciences, Tehran, Iran
4 Rajaie Cardiovascular Medical and Research Center, Cardiac Surgery Research Center, Iran University of Medical Sciences, Tehran, Iran

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  Abstract 


A patent ductus arteriosus (PDA) is a common congenital malformation in pediatric patients and accounts for 7%–10% of congenital heart diseases. The PDA is a posttricuspid shunt resulting from an arterial communication between the upper descending aorta and the distal pulmonary artery, is an important part of a normal fetal cardiac anatomy and usually closes spontaneously within 1 week after birth. Closure is indicated in patients with significant related symptoms, history of endarteritis, or dilation of the left atrium and/or the left ventricle (LV). Before the development of percutaneous PDA closure, surgical closure was recommended as the main routine treatment. An occasional occurrence is the recanalization of a surgically ligated ductus arteriosus, which may be associated with the serious complication of aneurysm formation. Herein, we report a rare case of a longstanding neglected PDA aneurysm as a complication of postsurgical PDA closure and discuss the weaknesses and strengths of cardiac imaging in this regard. A 31-year-old man with a history of surgical ligation of the PDA 20 years ago was referred to us for preoperative evaluation before elective noncardiac surgery. Electrocardiography revealed a normal sinus rhythm and an LV volume-overload pattern. Severe LV enlargement and moderate systolic dysfunction associated with a large residual PDA were found on transthoracic echocardiography. Cardiac catheterization and cardiac computed tomography angiography confirmed the presence of a calcified aneurysm of PDA, which was not suitable for percutaneous PDA closure. The patient underwent successful surgical closure without any residue or complications.

Keywords: Calcified aneurysm, endarteritis, heart failure, patent ductus arteriosus


How to cite this URL:
Saed D, Sadeghpour A, Alizadehasl A, Pouraliakbar H, Firouzi A, Hosseini S, Bayati P. Role of multimodality imaging in postsurgical calcified aneurysm of patent ductus arteriosus. Arch Cardiovasc Imaging [Epub ahead of print] [cited 2018 Dec 14]. Available from: http://www.cardiovascimaging.com/preprintarticle.asp?id=238933



  Introduction Top


A patent ductus arteriosus (PDA) is a common congenital malformation in pediatric patients and accounts for 7%–10% of congenital heart diseases.[1] A PDA is a posttricuspid shunt originating from an arterial communication between the upper descending aorta and the distal pulmonary artery. The PDA is an important part of a normal fetal cardiac anatomy and usually closes spontaneously within 1 week after birth. The estimated incidence of PDAs is 56/100,000 live births.[2]

Closure is indicated in any patient with heart failure symptoms or a history of endarteritis. Asymptomatic patients with the dilation of the left atrium or the left ventricle (LV) should also undergo PDA closure.[3] Before the development of percutaneous PDA closure, surgical closure was recommended as the main routine treatment.[4] The recanalization of a surgically ligated ductus arteriosus may be associated with the serious complication of aneurysm formation.[5]

If diagnosis and/or treatment are delayed, complications such as growth failure, heart failure, bacterial endarteritis, and pulmonary hypertension may ensue with a concomitant significant increase in surgical morbidity and/or mortality. Furthermore, delay in appropriate treatment may lead to pulmonary vascular diseases (Eisenmenger syndrome) and render the patient inoperable.[6] Transthoracic echocardiography has some limitations in precisely delineating the anatomy, but secondary effects of the PDA such as enlargement of left heart chambers and increased LV stroke volume could be certainly evaluated. Continuous-wave Doppler echocardiography illustrates continuous flow at the origin of the left pulmonary artery. Transesophageal echocardiography better delineates length and diameter of the PDA in some cases, while using three-dimensional (3D) echocardiography determines some types of PDA more accurately than 2D echocardiography. Although the PDA may be visible on computed tomography (CT) angiography, it requires ionizing radiation and intravenous contrast agents. Occasionally, calcification and/or aneurysm formation may be detected.[7] Although angiography is not usually required for the diagnosis of PDA, it may be needed during per cutaneous interventions or before surgery also to evaluate any associated lesions.[8]


  Case Report Top


A 31-year-old man with a history of surgical ligation of the PDA 20 years earlier was referred to our adult congenital heart disease clinic for the percutaneous closure of his residual PDA. Aside from a history of chronic atypical chest pain and some episodes of dyspnea on exertion, the patient had a history of endarteritis shortly after the surgical PDA closure 20 years ago, resulting in 6-week hospital stay and intravenous antibiotic therapy. Then, he was followed up for a small residual PDA of no clinical significance. On referral to us, physical examination was unremarkable except for a bounding pulse and a loud continuous machinery murmur at the upper left sternal border. Electrocardiography revealed a normal sinus rhythm and an LV volume-overload pattern. Laboratory data, including the sedimentation rate and inflammatory markers, illustrated no abnormal findings. Transthoracic echocardiography demonstrated severe LV enlargement with an ejection fraction of 40%, together with a top-normal right ventricular size and mild right ventricular dysfunction.

Two-dimensional and color-flow imaging in the parasternal short-axis and suprasternal views showed a moderately large residual PDA with a continuous turbulent flow between the proximal PDA and the descending aorta. Transesophageal echocardiography was performed to better evaluate the size and shape of the residual PDA: the aortic and pulmonary artery sides were 11 mm and 9 mm, respectively [Figure 1], [Figure 2] and [Video 1], [Video 2]. Furthermore, there was a large echo-free space adjacent to the proximal PDA. Although at echocardiography a calcified aneurysm of PDA was suspected, it was not very well defined. CT angiography of the thoracic aorta with an intravenous contrast agent revealed aneurysmal dilatation of the PDA with wall calcification [Figure 3]; in addition, the total aneurysm length was 31 mm and the internal lumen diameter was 12 mm.
Figure 1: Left panel: Color flow imaging of patent ductus arteriosus in parasternal short-axis view (white arrow). Middle panel: Continuous-wave Doppler study of patent ductus arteriosus. Right panel: Color flow imaging of patent ductus arteriosus at transesophageal echocardiography. Ao: Aorta

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Figure 2: Three-dimensional transesophageal echocardiography image of patent ductus arteriosus en face view (white arrow)

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Figure 3: Cardiac computed tomography angiography, Left panel: Sagittal view showing calcified aneurysm of patent ductus arteriosus Right panel: Three-dimensional volume rendering of patent ductus arteriosus

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Left and right heart catheterization showed a calcified aneurysm of PDA adjacent to the left pulmonary artery and a pulmonary artery pressure of 40/20 mmHg [Video 3] and [Video 4].




In light of the above-mentioned findings and suspicion of a true calcified aneurysm of PDA as opposed to a PDA pseudoaneurysm, our heart team opted for the surgical approach. The patient underwent mid-sternotomy open-heart surgery [Figure 4], whereby the calcified aneurysm of ductus was excised successfully.
Figure 4: Surgical view of calcified aneurysm of patent ductus arteriosus (black arrow)

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Intraoperative transesophageal echocardiography confirmed no residual PDA, and the patient was discharged home a few days later without any surgical complications.


  Discussion Top


Herein, we presented a rare case of the late diagnosis of a residual calcified aneurysm of PDA after its surgical ligation with a history of endarteritis following surgery. In our patient, echocardiography demonstrated the residual PDA with a significant left-to-right shunt; nonetheless, finding a calcified aneurysm of PDA requires sufficient knowledge about postoperative complications and careful evaluations via echocardiography and cardiac CT. Angiography is a useful method for hemodynamic assessment and possible percutaneous closure of the residual aneurysm of PDA. As our patient was not suitable for percutaneous closure and we could not distinguish between a calcified aneurysm of PDA and the pseudoaneurysm formation of the PDA, we candidated him for open-heart surgery.

Nowadays, most PDAs are amenable to percutaneous device closure and transcatheter occlusion has become the treatment of choice for most of these congenital malformations.[8],[9] On the rare occasion of the presence of a very large ductus, the surgical division of the PDA remains the preferred modality. Nevertheless, our patient had already undergone ligation alone rather than total excision, which is inherently associated with more recanalization and aneurysm formation. Rarely, a large window-type PDA may have insufficient length to permit ligation, and the suitable surgical procedure is patch closure with cardiopulmonary bypass.[10] The complete closure rate of surgical ligation (most often accompanied by the division of the ductus) ranges from 94% to 100%. Important surgical complications include bleeding, pneumothorax, and infection.[11]

Another salient point with respect to our patient was the occurrence of endarteritis shortly after the surgical closure of the PDA. The complication was cured with medical therapy alone, but the residual flow in a surgically manipulated ductus – especially when it is associated with an episode of endarteritis – should never be deemed negligible. In other words, our patient had been initially grossly mismanaged. The question, therefore, is what course of action should be taken in this scenario. Obviously, surgical treatment is the first choice, but the intraoperative complication feared the most is injury secondary to bleeding from the aortic or pulmonary stump owing to the presence of heavy calcification.[12],[13]

A PDA aneurysm is a rare lesion with a spontaneous or acquired origin and with an incidence rate of 8%. Ductal aneurysms may develop in the wake of infective endarteritis, surgical closure (as was the case in our patient), or transcatheter coil occlusion.[14]

In 25% of patients, an underlying disorder such as trisomy 21, trisomy 13, Smith–Lemli–Opitz syndrome, type IV Ehlers–Danlos syndrome, or Marfan syndrome is present (which was not the case in our patient).[15] In our patient, surgical intervention successfully resolved this scenario and he was discharged home without any residual ductus flow.


  Conclusions Top


We showed that even a longstanding neglected large calcified PDA aneurysm after surgical ligation in conjunction with an episode of endarteritis may be successfully treated surgically in experienced hands without any residue or complications, provided that in addition to full echocardiographic study, comprehensive multimodality imaging of the detailed anatomy and relationship with various adjacent vascular and extravascular structures delineated in properly selected cases, with CT angiography and/or catheterization.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Hwang B, Lee PC, Weng ZC, Fu YC, Hsing HP, Lu JH, et al. Comparison of the one-and-a-half-year results of closure of patent ductus arteriosus by transcatheter coils placement with surgical ligation. Angiology 2000;51:757-63.  Back to cited text no. 1
    
2.
Wang K, Pan X, Tang Q, Pang Y. Catheterization therapy vs surgical closure in pediatric patients with patent ductus arteriosus: A meta-analysis. Clin Cardiol 2014;37:188-94.  Back to cited text no. 2
    
3.
Ross RS, Feder FP, Spencer FC. Aneurysms of the previously ligated patent ductus arteriosus. Circulation 1961;23:350-7.  Back to cited text no. 3
    
4.
Hoffman JI, Christianson R. Congenital heart disease in a cohort of 19,502 births with long-term follow-up. Am J Cardiol 1978;42:641-7.  Back to cited text no. 4
    
5.
Warnes CA, Williams RG, Bashore TM, Child JS, Connolly HM, Dearani JA, et al. ACC/AHA 2008 guidelines For the management of adults with congenital heart disease: A report of the American college of cardiology/American heart association task force on practice guidelines (Writing committee to develop guidelines on the management of adults with congenital heart disease). Developed in collaboration with the American Society of Echocardiography, Heart Rhythm Society, International Society for Adult congenital heart disease, Society for Cardiovascular Angiography and Interventions, and Society of Thoracic Surgeons. J Am Coll Cardiol 2008;52:e143-263.  Back to cited text no. 5
    
6.
Mullins CE. Patent ductus arteriosus. In: Garson A Jr., Bricker JT, McNamara DG, editors. The Science and Practice of Pediatric Cardiology. Boston: Lea & Febiger; 1990. p. 1055-6.  Back to cited text no. 6
    
7.
Schneider DJ, Moore JW. Patent ductus arteriosus. Circulation 2006;114:1873-82.  Back to cited text no. 7
    
8.
Berdjis F, Moore JW. Balloon occlusion delivery technique for closure of patent ductus arteriosus. Am Heart J 1997;133:601-4.  Back to cited text no. 8
    
9.
Taneja K, Gulati M, Jain M, Saxena A, Das B, Rajani M, et al. Ductus arteriosus aneurysm in the adult: Role of computed tomography in diagnosis. Clin Radiol 1997;52:231-4.  Back to cited text no. 9
    
10.
Grünenfelder J, Bartram U, Van Praagh R, Bove KE, Bailey WW, Meyer RA, et al. The large window ductus: A surgical trap. Ann Thorac Surg 1998;65:1790-1.  Back to cited text no. 10
    
11.
Hawkins JA, Minich LL, Tani LY, Sturtevant JE, Orsmond GS, McGough EC, et al. Cost and efficacy of surgical ligation versus transcatheter coil occlusion of patent ductus arteriosus. J Thorac Cardiovasc Surg 1996;112:1634-8.  Back to cited text no. 11
    
12.
Kelly DT. Patent ductus arteriosus in adults. Cardiovasc Clin 1979;10:321-6.  Back to cited text no. 12
    
13.
Morgan JM, Gray HH, Miller GA, Oldershaw PJ. The clinical features, management and outcome of persistence of the arterial duct presenting in adult life. Int J Cardiol 1990;27:193-9.  Back to cited text no. 13
    
14.
Kerwin AJ, Jaffe FA. Postoperative aneurysm of the ductus arteriosus, with fatal rupture of a mycotic aneurysm of a branch of the pulmonary artery. Am J Cardiol 1959;3:397-403.  Back to cited text no. 14
    
15.
Varma PK, Vallath G, Neema PK, Sinha PK, Sivadasanpillai H, Menon MU, et al. Clinical profile of post-operative ductal aneurysm and usefulness of sternotomy and circulatory arrest for its repair. Eur J Cardiothorac Surg 2005;27:416-9.  Back to cited text no. 15
    

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Correspondence Address:
Anita Sadeghpour,
Rajaie Cardiovascular Medical and Research Center, Echocardiography Research Center, Iran University of Medical Sciences, Tehran
Iran
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/ACVI.ACVI_2_18



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  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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    -  Sadeghpour A
    -  Alizadehasl A
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    -  Bayati P


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