Case of the Week # 258

P. Budziszewska, MD*; D.Kuka, MD*; K. Sodowski, MD*, P.Jeanty, MD**; M. Hernanz-Schulman, MD***

December 11, 2009 - January 7, 2009

*    Department of Obstetrics and Gynecology, Medical University of Silesia, Ruda Slaska, Poland.
**   Inner Vision Women's Ultrasound, Nashville, Tennessee USA.
***  Diagnostic Imaging, Vanderbilt Children"s Hospital

The following images show a case of a cardiovascular anomaly which was diagnosed at 24 weeks of gestation. Diagnosis was confirmed postnatally.

Images 1-9: 24 weeks.

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Video

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Answer


The current case of the week showed a case of Double aortic arch.

Definition

A double aortic arch is an abnormal formation of the aorta which represents a persistence of both right and left fourth aortic arches that form a complete vascular ring around the trachea and esophagus.

Case report

A 32-old woman G2 P2 was referred to our office at 24 weeks of gestation for a routine examination. On transverse view of the high mediastinum, the aortic arch was seen to extend abnormally to the right chest. This prompted a careful review of the anatomy of the great vessels. A vascular ring formed by the aortic arch and a second vessel, initially though to be the pulmonary artery was found. However, the left-sided branch of the ring was not the pulmonary artery that was visible more to the left, but a left aortic arch. Thus the fetus presented with a double aortic arch with a right dominant arch that encircled the trachea. In a coronal view of the trachea and bronchi, cross sections of the aortic arches were seen on both sides of the trachea. Ipsilateral arches gave rise to the left and right common carotid artery and left and right subclavian artery. The 4-chamber view was normal. Color flow Doppler image showed the same anatomy as shown with gray-scale imaging. There was a left ductus arteriosus. On the coronal section posteriorly at the level of the descending aorta we saw both aortic arches converging in the descending aorta. Follow-up fetal echocardiography was performed every 4 to 6 weeks during the remainder of the pregnancy. A fullterm neonate was born at 40 weeks’ gestation with a birth weight of 3.5 kg and with Apgar scores of 8/9 at 1st/5th minute, respectively. The diagnosis was confirmed on postnatal echocardiography and CT. Stridor developed after an upper respiratory tract infection at 3 months of age. He underwent surgery at 5 months of age.

Images 1,2: Image 1 shows a normal 4-chamber view,  aorta is descending on the right; Image 2 shows right vetricular outflow tract.

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Image 3: Image shows common pulmonary artery (blue), aorta (red) with trachea (pink) in the middle.

 

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Image 4: Image 4 shows dominant right aortic arch (red) and pulmonary artery (blue) encircling the trachea (pink).

 

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Image 5: Image 5 shows pulmonary artery (blue) and aorta branching into dominant right aortic arch and left aortic arch (red), trachea is behind (pink). 

 

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Images 6,7: Image 6 shows double aortic arch in gray scale imaging; Image 7 shows Color Doppler flow within the double aortic arch (blue) and pulmonary artery (red).

 

 

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Image 8: Image shows dominant right aortic arch with only 2 vessels arising from the arch. 

 

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Image 9: Transverse section of the abdomen; right descending aorta (red).

 

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Video 1: Video show both aortic arches which encircle trachea. 

Images 10, 11: Image 10, 3D CT reconstruction demonstrate the dominant right arch and the smaller left arch in front. An apparent interruption of the left arch at it’s connection to the descending aorta, is an artifact; Image 11, high thoracic axial section demonstrates the larger right aorta and thinner left aorta as they separate in front of the trachea.


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Image 12, 13: Image 12, the black silk tie is around the ligamentum arteriosum, the upper blue vessel-loop is around the anterior (left ) arch which is attached to the aortic diverticulum ( of Kommeral) and then heads cephalad as the left subclavian artery. The lower blue loop is around the aortic diverticulum. Image 13, shows the left aortic arch after resection of the diverticulum.

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Image 14: Schematic picture of the double aortic arch.

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Introduction   A double aortic arch is one the most common types of vascular ring [1]. The hypothetical double aortic arch system was suggested by Dr Jesse E.Edwards in 1964 (image 14) [2,3]. In this model the descending aorta is located posterior to the tracheoesophageal pedicle and connected bilaterally to the ascending aorta by lateral aortic arches. Subclavian arteries and common carotid arteries arise from both right and left arches and ductus arterisosus on each sides connects the arch to its ipsilateral pulmonary artery [4]. The right arch is larger and higher in approximately 75% of cases. Rarely, one arch is atretic. In the majority of cases the left ductus is patent. The descending aorta is usually deviated to the side of the patent ductus [5]. A double aortic arch is a complete vascular ring that encircles both the trachea and the esophagus [6, 7]. Aortic arch anomalies may lead to respiratory distress in the neonate or the development of milder symptoms and signs of tracheal or esophageal compression later in life, or they may remain clinically silent [8].

A literature review showed an association between double aortic arch and congenital heart diseases in approximately 20% of cases most often tetralogy of Fallot, transposition of great vessels, ventricular septal defects. Rarely there can be atresia of the segment of the aortic arch, which can be difficult to differentiate from other aortic arch anomalies associated with chromosomal abnormalities such as microdeletion of chromosome 22q11 [9, 10]. The condition affect males and females fetuses equally [11].

Pathogenesis

The occurance of this anomaly is probably related to persistence of both embryonic aortic arches [9, 11]. The aortic arches are usually symmetrical, the right arch being larger and higher in approximately 75% of cases. As a rule, the descending aorta is contralateral to the side of the dominant arch. In the majority of cases the left ductus is patent [5].

Ultrasound findings

The vascular ring diseases can be recognized in 3 vessels. The suspicion of a double aortic arch is raised by detecting the U-sign which is formed by the combination of both aortic arches and the left ductus arteriosus (images 2,3). In a plane cranial to the U-sign, the aortic root bifurcates in the two arches directly in front of the trachea as a Lambda [12]. The 4-chamber view appears normal, but the descending aorta is deviated medially. In the 3 vessels view the ascending aorta and aortic arch are pointing to the right, whereas the left arch points to the left, and the trachea is seen between. The bifurcation of the aortic arch can be recognized in the upper thorax. The pulmonary trunk bifurcate into left and right pulmonary arteries and gives rise to the ductus arteriosus on the left side of the trachea [12]. The diagnosis cannot be achieved in a longitudinal visualization of the arch, since an arch with a common carotid artery and subclavian artery is visualized both in a left and in a right parasagittal plane to the trachea [12].  A sweep downward in the upper thorax will then demonstrate how the bifurcated aortic arches fuse together to form the neutral descending aorta [12].  A longitudinal coronal plane can demonstrate how both arches merge together to form the descending aorta. However, one has to be careful with this coronal plane, because it is also found in a right arch and a left ductus arteriosus (in the U-sign). The esophagus and trachea are entrapped in this vascular ring [12].

Differential diagnosis

  • Right aortic arch and left ductus which forms a ring (the U-sign).
  • Large azygos arch in case of interrupted inferior vena cava (left isomerism). Following the right arch into the upper thorax and seeking the presence of the brachiocephalic arteries can help in the differentiation [12].

Dicussion

The 4-chamber view is used in the fetal sonographic screening examination for detection of congenital heart disease. The limitations of the 4-chamber view is, that we could not detect anomalies of the outflow tract and great arteries. Integration of the 3-vessel view, an axial view of the fetal upper mediastinum has been proposed in sequential segmental analysis to improve the antenatal detection of congenital heart disease. The location of the aortic arch in relation to the trachea can be easily visualized because the fetal airway is normally filled with fluid, allowing sonographic visualization [1, 9, 12]. In summary, the 3-vessel and trachea view in the upper mediastinum is useful in detecting abnormalities of the aortic arch [9, 2].This report describes the sonographic prenatal diagnosis of double aortic arch , showing the importance of 3 vessels view examination by detecting the anomalies of aortic arch.

Answer of the Nickolay Veropotvelyan, MD. We wanted to show this answer because we find it very accurate and it nicely shows the analytical process of thinking and finding the correct answer!

As usual I use the systematic consequal segmental approach for the fetal heart evaluation.

Image 1:  4-chamber view at diastola is almost normal

-         Position & cardiac axis and heart size are normal

-         normal size relationship of the left and right sides

-         chambers are without any asymmetry and visible septal detects

-         atrial septum primum (I) is present

-         opened MV & TV valves are well recognized

-         opened FO

-         atrio-ventricular connection is concordant

-         normal connection of the pulmonary veins to the LA

-         no cardiac wall hypertrophy (endocardrium echogenecity is also - normal)

-         descending Ao in abnormal  position – is ring-sided  relative to spine

Image 2-4:  3V+T view– are abnormal

-         3 vessels are located sequentially with normal vessels size decrease

-         Crossing of great arteries is present, so TGA is excluded.

And the main point – we can see vascular right arising from Aorta around the trachea and esophagus.

The ring is shaped as the  figure “9” or “6” in horizontal position. It is the most significant echosign.

The right part of the ring is bigger than the left one.

Image 5: 5-chamber view – LV outlet with the ascending Ao

Image 6: the  same scan in CFM Doppler mode – and we can see the beginning of the small left Ao Arch branch.

Descending Ao is right-sided relative to spine

Image 7: “Pulmonary view” – RV outlet with arising PA root – is normal

So, (looking on images 5-7) the bulbo- ventricular and ventriculo- arterial connections are concordant

Image 8: Sagittal scan – Longitudinal view of the Ao Arch.

Unfortunately I can’t see the exact number of the vessels arising to the head and upper limbs. We can definitely observe only 1 vessel.

Image 9: axial scan of fetal  upper abdominal plan

-         abnormal central  position of descending Ao;

-         vena cava inferior in normal right-sided position, ventral and lateral to the Aorta

-         stomach is absent, I thank it is  due to eosophagal  obstruction by the double Ao Arch ring – effect like in case of esophageal atresia. (Unfortunately in these pictures we can’t see a quantity of Amniotic fluid) Polyhydranion  - is may be expected.

-         Looking at the video (with normal  Rhythm and heart contractility) my answer #1:

-         It is right sided double Aortic arch with the dominant right branch and right sided descending aorta.

P.S.

Differential diagnose with the Aberrant right subclavian artery/ARSA/ or a.Lusoria.

(RAo Arch with the ALSA – has another “U” – shaped–sign-picture with the  ring due  to Kommerell’s divertirulum.

Besides it is possible to have here vascular ring with the narrower vessel branching from the left of Ascending Ao. (ARSA originate from the 4-th regressed Ao Arch, connected with  delay dorsal Ao segmett, which are connect with the right 7 intrasegmental artery). The key to the correct Prenatal diagnose is the Ao Arch longitudinal scan evaluation, which allows not  a 3,-but  a 4 arising wessels from the Ao Arch. (Especcialy useful 4D+STIC).

But these anomalies have a different clinic and prognosis. For DAA it is more typical to have  more severe obstructive trachea symptoms  and dysphagia. The final  postnatal diagnose give by CT and MRT, because neonatal echocardiography is useless. As usual, infants with DAA need for cardiosurgery treatment.

With respect, Nickolay.

References

1. Achiron R, Rotstein Z, Heggesh J, Bronshtein M, Zimand S, Lipitz S, Yagel S. Anomalies of the fetal aortic arch: a novel sonographic approach to in-utero diagnosis. Ultrasound Obstet Gynecol. 2002 Dec;20(6):553-7.

2. Edwards JE. ‘‘Vascular rings’’ related to anomalies of the aortic arches. Mod Concepts Cardiovasc Dis 1948; 17: 19–20.

3. Edwards JE. Malformation of the aortic arch system manifested as ‘‘vascular rings’’. Lab Invest 1953; 2: 56–75. 4. Edwards JE. Vascular rings and slings. In Fetal, Neonatal, and Infant Cardiac Disease, Moller JH, Neal WA (eds). Norwalk, CT: Appleton & Lange: 1990; 745–754.

5. Moes CAF. Vascular rings and related conditions. In Congenital Heart Disease, Freedom RM, Mawson JB, Yoo SJ, Benson LN (eds). Textbook of Angiocardiograph. Armonk, NY: Futura Publishing Co., 1997; 947–983.

7. Bronshtein M, Lober A, Berant M, Auslander R, Zimmer EZ. Sonographic diagnosis of fetal vascular rings in early pregnancy. Am J Cardiol 1998; 81:101–103.

8. Patel CR, Lane JR, Spector ML, Smith PC. Fetal echocardiographic diagnosis of vascular rings. J Ultrasound Med. 2006 Feb;25(2):251-7.

9. Yoo SJ, Min JY, Lee YH, Roman K, Jaeggi E, Smallhorn J. Fetal sonographic diagnosis of aortic arch anomalies. Ultrasound Obstet Gynecol. 2003 Nov;22(5):535-46.

10. Park SC, Zuberbuhler JR. Vascular ring and pulmonary sling. In Pediatric Cardiology, Anderson RH, Macartney FJ, Shinebourne EA, Tynan M (eds). Edinburgh, UK: Churchill Livingstone: 1987; 1123–1136.

11. Hornberger LK. Aortic arch anomalies. In Textbook of Fetal Cardiology, Allan L, Hornberger L, Sharland G (eds). London, UK: Greenwich Medical Media:2000; 305–321.

12. Jeanty P, Chaoui R, Pilu R, Romero R. Fetal echocardiography: Part II: the anomalies. TheFetus.net. 2006.

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