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Ann Thorac Surg 2006;82:2274-2276
© 2006 The Society of Thoracic Surgeons

Case Reports

Intimo-Intimal Intussusception of the Aorta

Pallav J. Shah, MCha, Sanjay S. Singh, MCHa, Salim S. Chaloob, MBChBa, Clayton Lang, MBBSa, James Taylor, FRACPb, James R. Edwards, FRACSa,*

a Department of Cardiac Surgery, Royal Adelaide Hospital, Adelaide, Australia
b Department of Radiology, Royal Adelaide Hospital, Adelaide, Australia

Accepted for publication May 5, 2006.

* Address correspondence to Mr Edwards, Department of Cardiac Surgery, Level 4, East Wing, Royal Adelaide Hospital, North Terrace, Adelaide, SA 5000, Australia. (Email: jedward1{at}mail.rah.sa.gov.au).


   Abstract
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 Abstract
Introduction
 Comment
 References
 
A 50-year-old man presented with acute onset of chest pain. Subsequent transesophageal echocardiography and computed tomography scan showed absence of a flap in the ascending aorta and a clear dissection flap involving the arch and descending aorta. Magnetic resonance imaging showed a tear and a small flap in the right coronary sinus. During surgery, we found a total circumferential intimal tear at the sinotubular junction with intimo-intimal intussusception of the internal channel into the arch. Dissection without intimal flap and aortic intussusception is a rare form of type A dissection, which is difficult to diagnose on routine investigations and can delay treatment.


   Introduction
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 Abstract
 Introduction
Comment
 References
 
The total disruption of the intima with circumferential intimal flap is an unusual presentation. Boestroem reported the first case in 1887 and Chiari in 1909, who described it as an "Inversion of the Internal Cylinder" [1]. This complication was later described by Hufnagel and Conrad [2], who named it intimo-intimal intussusception.

A 50-year-old dentist with history of chronic smoking presented with acute onset of chest pain radiating to the jaw and chest tightness. He had Marfanoid features. The electrocardiogram was inconclusive, and a roentgenogram showed a widened superior mediastinum. He had mild elevation of troponin T and thus was initially treated with aspirin and Clexane (Aventis Pharma Pty Ltd, Lane Cove NSW).

Results of gastroscopy and abdominal ultrasonography were negative. An echocardiogram revealed a grossly dilated aortic root and ascending aorta (5.8 to 6.0 cm), moderate central aortic regurgitation, and an intimal flap with perfused false lumen starting close to the arch but not in ascending aorta. A computed tomography (CT) angiogram (Fig 1) showed no intimal flap in a dilated ascending aorta (5 cm) but did show a dissection involving the arch and extending to the iliac arteries. An ill-defined irregularity was present both on echocardiography and CT scan at the right sinus of Valsalva. We persisted with antihypertensive measures while a magnetic resonance imaging (MRI) scan was organized to further define the anatomy.


Figure 1
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  		Fig 1. Postcontrast axial computed tomography scan demonstrates intussuscepted dissection flap (A) in aortic arch with very narrow residual true lumen (C) and a larger false lumen (B).

 
The MRI (Fig 2) showed aneurysmal dilatation of the aortic root and ascending aorta (5.1 cm in diameter). Both the black blood and cine sequences showed a dissection flap arising from the anterior aspect of the ascending aorta in the region of the aortic root. The cine sequences showed this flap to be relatively immobile, helping to differentiate it from an aortic valve cusp. The flap could not be followed in continuity to the aortic arch. The dissection became readily visible in the aortic arch involving the innominate artery, left common carotid, and left subclavian origins.


Figure 2
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  		Fig 2. "White blood cine" magnetic resonance image demonstrates dilated ascending aorta with abrupt reduction in flow beyond the left subclavian artery due to dissection.

 
Because of the presence of this aortic root abnormality together with a pericardial effusion, we thought surgical intervention was warranted despite the absence of a classic ascending aortic dissection flap and false lumen. On opening the pericardium, we saw a thinned out and bluish hue over the ascending aorta, typical of a classic type A dissection.

The femoral artery and right atrium were cannulated, and cardiopulmonary bypass was commenced. The patient was cooled to 22°C. On opening the ascending aorta (Fig 3) under circulatory arrest, we found total absence of the intimal layer. Looking downstream, we located the missing intimal flap, which had peeled off the ascending aorta thus denuding it. Inversion of the ascending aortic intima was arrested by the innominate artery origin.


Figure 3
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  		Fig 3. Operative photograph shows total circumferential intimal tear with intimo-intimal intussusception of the internal channel into the arch.

 
At surgery, the whole of the intima of the ascending aorta was retracted back and tailored to the new distal aortic orifice at the level of the arch. The new reconstituted distal aortic orifice constituted four layers: outer Teflon (Dupont, Wilmington, DE) felt, adventitial layer, restored intimal layer, and inner Teflon felt layer reinforcement to contain the rest of the dissection. The entire ascending aorta from the sinotubular junction to the beginning of the arch was replaced with the Dacron (Dupont) graft. The distal end of the arch was sutured first under circulatory arrest and then cardiopulmonary bypass was recommenced. Proximally, an aortic valve sparing operation was performed with resuspension of the valve to the graft at its commissures. The postbypass transesophageal echocardiography (TEE) showed trivial aortic incompetence.

The patient had a smooth postoperative course, with no neurologic complications. Postoperative echocardiography at discharge showed a residual flap in the distal arch with communication between true and false lumen. A CT scan at discharge showed a persistent flap with intramural hematoma in the arch extending downwards. We plan is to follow-up every 6 months with a CT scan to watch for progression of aneurysmal dilation.


   Comment
Top
 Abstract
 Introduction
 Comment
References
 
In dissection of the ascending aorta, the intimal flap is usually transverse and rarely exceeds more then half of the circumference of the aorta. Intussusception of the internal cylinder into the external cylinder can induce antegrade obstruction of the aortic lumen and arch vessels, leading to intermittent neurologic disorders and obstruction of the descending aorta and resulting in features suggestive of pseudocoarctation with asymmetric pulses and blood pressure [1]. Retrograde intussusception into the left ventricle, partly occluding the coronary vessels, may cause myocardial ischemia. It may also cause detachment of the aortic commissures, leading to prolapse of leaflets and acute aortic insufficiency [3].

TEE still remains the most sensitive diagnostic tool. The combination of a thick sinuous circumferential flap in the aortic arch with absence of an intimal flap in the ascending aorta is suggestive of antegrade intimo-intimal intussusception into the arch [1]. The back-and-forth movement of the cylinder-shaped intima as it prolapses into the left ventricle and thrusts into the aorta during diastole and systole, respectively, is suggestive of retrograde intussusception into the left ventricular outflow tract [3].

CT findings in a patient with intimo-intimal intussusception have been described by Nelsen and colleagues [4]. An enlarged aortic root, mediastinal hematoma, the presence of curvilinear lucencies in the aortic root, the confusing absence of an intimal flap in the midascending aorta, and again, the presence of linear lucencies in the aortic arch, are consistent with intussuscepted ascending aortic intima [4]. MRI and Doppler ultrasonography of the supra aortic vessels can further aid in the diagnosis.

Commonly accepted treatment of this kind of type A dissection is reduction of the intussusception by returning the internal cylinder into the ascending aorta with a brief period of circulatory arrest. Then, depending upon the extent of the intimal tear, replacement of the ascending aorta only or with arch and reimplantation of the head vessels may be required [1].

To date, only 20 cases of circumferential tear of the intimal layer have been reported. The unusual clinical presentations and lack of demonstration by common diagnostic tools may lead to false-negative diagnosis and delay in treatment of the type A dissection.


   References
Top
 Abstract
 Introduction
 Comment
 References
 

  1. Touati G, Carmi D, Trojette F, Jarry G. Intimo-intimal intussusception: a rare clinical form of aortic dissection Eur J Cardiothorac Surg 2003;23:119-121.[Abstract/Free Full Text]
  2. Hufnagel CA, Conrad PW. Intimo-intimal intussusception in dissecting aneurysms Am J Surg 1962;103:727-731.[Medline]
  3. Touati G, Carmi D, Trojette F, Jarry G. Reply to Yavuz et al Eur J Cardiothorac Surg 2003;23:851.[Medline]
  4. Nelsen KM, Spizarny DL, Kastan DJ. Intimointimal intussusception in aortic dissection: CT diagnosis AJR Am J Roentgenol 1994;162:813-814.[Free Full Text]



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This Article
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Sanjay S. Singh
James Taylor
James R. Edwards
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