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Eur J Cardiothorac Surg 2003;23:119-121
© 2003 Elsevier Science NL

Case report

Intimo-intimal intussusception: a rare clinical form of aortic dissection

^a Service de Chirurgie Cardiaque, Hôpital Sud CHU Amiens, 80054 Amiens Cedex 01, France
^b Service de Cardiologie A, Hôpital Sud CHU Amiens, Amiens, France

Received 5 August 2002; received in revised form 6 September 2002; accepted 1 October 2002.

* Corresponding author. Tel.: +33-3-22-45-59-25; fax: +33-3-22-45-53-31
e-mail: gtouati.hms{at}invivo.edu

	Abstract

Top Abstract 1. Introduction 2. Clinical case 3. Discussion References

 
A 33-year-old hypertensive man presented with epigastric pain radiating to the back. Transoesophageal echocardiography (TOE) revealed an intimal flap on the aortic arch and descending aorta. No intimal flap of the ascending aorta was detected on TOE or CT. The diagnosis was made on opening the ascending aorta: complete circumferential dissection of the ascending aorta flush with the coronary ostia, with no residual intimal flap, and intimo-intimal glove-finger intussusception of the internal channel into the descending thoracic aorta. Aortic intussusception is a very rare form of Type I dissection, and the absence of intimal tear in the ascending aorta can be misleading and delay the diagnosis.

Key Words: Intimo-intimal intussusception • Aortic dissection • Aortic arch

	1. Introduction

Top Abstract 1. Introduction 2. Clinical case 3. Discussion References

 
Dissection of the ascending aorta is a life-threatening disease and therefore constitutes a diagnostic and therapeutic emergency. Visualization of an intimal flap separating the true and false channels on morphological examinations is a pathognomonic sign of aortic dissection confirming the positive diagnosis. In some clinical forms, such as intimo-intimal intussusception, the absence of an intimal flap in the ascending aorta can lead to delayed diagnosis and treatment.

The authors discuss the diagnostic traps and the ways of avoiding them, in the light of a case of intimo-intimal intussusception.

	2. Clinical case

Top Abstract 1. Introduction 2. Clinical case 3. Discussion References

 
Mr C., 33 years old, was treated for hypertension by beta-blockers that he stopped himself in January 2001. The history of the present illness began with a combination of abdominal pain and vomiting and an episode of loss of consciousness. The assessment performed did not reveal any cause for this pain. Mr C. was admitted to our hospital 48 h later for recurrence of epigastric pain radiating to the neck, accompanied by nausea and vomiting.

Upper gastrointestinal endoscopy demonstrated fundic gastritis and bulbo-duodenitis. The symptoms were relieved by IV Omeprazole (MOPRAL^®). On the day after admission, Mr C. reported crushing chest pain radiating to the neck. Examination demonstrated a dissociation of blood pressure with 160/90 on the left and 100/60 on the right, associated with marked reduction of the left femoral pulse. Mr C. was then transferred to the cardiac intensive care unit in our establishment with suspected aortic dissection.

Transthoracic echocardiography demonstrated dilatation of the ascending aorta to 45 mm with minimal central aortic regurgitation, already diagnosed 3 years previously, no pericardial effusion, normal ejection fraction and no visible flap in the ascending aorta.

Thoracic CT demonstrated dissection with a flap arising in the right brachiocephalic trunk and involving all of the thoracic descending aorta as far as the right common iliac artery and confirmed the absence of an intimal flap in the ascending aorta.

The diagnosis was confirmed by transoesophageal echocardiography (TOE), which demonstrated aortic dissection involving the arch and all of the thoracic descending aorta with a perfused false channel. No flap was detected in the ascending aorta, but a haematoma was detected 6 cm from the base of the sigmoid valve. Grade I aortic regurgitation due to annular dilatation was also observed.

The assessment was completed by Doppler examination of the supraaortic vessels, which showed a dissected brachiocephalic trunk with an intermittent flap ascending into the right subclavian artery. The left common carotid artery was also dissected. The left subclavian artery was patent and not dissected.

In the abdomen, the dissection involved the coeliac trunk and superior mesenteric artery, and the left external iliac artery was also dissected.

In this case of aortic dissection of the aortic arch, not affecting the ascending aorta, it was decided to wait for 48 h. The persistence of back pain despite well controlled blood pressure suggested the possibility of ongoing dissection with the possibility of rupture and so the decision was made to operate.

The diagnosis was established intraoperatively. Intraoperative exploration revealed circumferential dissection of the ascending aorta with intussusception of the inner cylinder towards the aortic arch.

Surgical treatment consisted of replacement of the ascending aorta and aortic arch with reimplantation of the supraaortic vessels without circulatory arrest under moderate hypothermia (33–34 °C). Cardiopulmonary bypass was performed between an atrio-caval venous cannula and a femoral arterial cannula. Cerebral protection was ensured by selective perfusion of the brachiocephalic trunk and left common carotid artery. After opening the aortic arch, the descending thoracic aorta was occluded by a Robiscek Pruitt aortic occlusion catheter.

The immediate postoperative course was uneventful, but the patient subsequently experienced diffuse abdominal pain and diarrhoea on D12, suggesting possible mesenteric ischaemia. This diagnosis was confirmed by duplex scanning. Aortography revealed dissection extending as far as the two iliac arteries with compression of the ostia of the superior mesenteric artery and coeliac trunk by the false channel. The renal arteries were well perfused by the true channel. An infrarenal aorto-bi-iliac bypass graft and implantation of the superior mesenteric artery were therefore performed, together with fenestration of the intimal flap in the infrarenal aorta. The postoperative course was slowly favourable with the return of intestinal transit and resolution of abdominal pain. The follow-up duplex scan of the aorta showed good flow in the superior mesenteric artery. One year later, the patient is asymptomatic.

	3. Discussion

Top Abstract 1. Introduction 2. Clinical case 3. Discussion References

 
Dissection of the ascending aorta is only rarely circumferential. Complete circular dissection of the aorta was reported for the first time by Bostroem [1] in 1887 and by Chiari [2] in 1909 who described "inversion of the internal cylinder".

Intussusception of the internal cylinder in the external aortic cylinder during circumferential dissection can induce obstruction of the aortic lumen or obstruction of the ostia of supraaortic vessels. This complication was described by Hufnagel and Conrad [3] by the name intimo-intimal intussusception. This mechanism is responsible for the clinical features of chest pain accompanied by intermittent neurological disorders and variable symptoms such as loss of consciousness, vertigo, and confusion [4,5]. When intimo-intimal intussusception is situated in the descending thoracic aorta, it may present with clinical features of pseudo-coarctation with asymmetric pulses and asymmetric blood pressure, but with no neurological disorders [6,7].

These clinical manifestations are all the more difficult to interpret in that morphological examinations do not visualize the classical signs of aortic dissection with an intimal flap in the aortic lumen [8].

TOE [9] is considered to be the reference examination for the diagnosis of aortic dissection with a sensitivity of 98%. False-negatives are essentially observed in dissections confined to the junction between the ascending aorta and the aortic arch (blind zone). In the case of intimo-intimal intussusception, the dissection in the aortic arch presents an unusual echocardiographic appearance, with a thick, sinuous flap intussuscepting into the aortic lumen. On multidimensional TOE, sections of the horizontal aorta between 0° and 90° help to refine the diagnosis. TOE does not visualize an intimal flap in the ascending aorta, but may demonstrate the intussusception image in the arch on a 0° transaortic section (Fig. 1) or a circumferential flap situated in the aortic arch on a 63° section (Fig. 2) . The combination of a circumferential flap in the aortic arch and the absence of an intimal flap in the ascending aorta is suggestive of the diagnosis of intimo-intimal intussusception.

View larger version (72K): [in this window] [in a new window]   Fig. 1. Transaortic TOE section in the horizontal aorta at 0° demonstrating an intussusception image.

View larger version (70K): [in this window] [in a new window]   Fig. 2. Transaortic TOE section in the horizontal aorta at 63° demonstrating a sinuous circumferential flap.

	References

Top Abstract 1. Introduction 2. Clinical case 3. Discussion References

 

1. Bostroem E. Das geheilte aneurysma dissecans. Deut Arch Klin Med 1887;42:1.
2. Chiari H. Aneurysma dissecans aortae mit inversion des inneren zylinders. Verh Deut Path Ges 1909;207.
3. Hufnagel C.A., Conrad P.W. Intimo-intimal intussusception in dissecting aneurysms. Am J Surg 1962;103:727.[Medline]
4. Goldberg S.P., Sanders C., Nanda N.C. Aortic dissection with intimal intussusception: diagnosis and management. J Cardiovasc Surg (Torino) 2000;41(4):613-615.[Medline]
5. Neri E., Capannini G., Caron E. The missing flap: consideration about a case of aortic intussusception. J Thorac Cardiovasc Surg 1999;117:829-830.[Free Full Text]
6. Masiello P., Santoro G., Franzesi E., De Lillo L., Fittipaldi O., Di Benedetto G. Aortic pseudo-coarctation: spiral volumetric computed tomography imaging. Ann Thorac Surg 1999;68(4):1421.[Free Full Text]
7. Verdant A., Page A., Blair J.F. Diagnostic and therapeutic observations drawn from the surgical experiences of 108 traumatic ruptures of the descending thoracic aorta. Ann Chir 1998;52(8):813-820.[Medline]
8. Lijoi A., Scarano F., Canale C. Circumferential dissection of the ascending aorta with intimal intussusception. Case report and review of the literature. Tex Heart Inst J 1994;21(2):166-169.[Medline]
9. Banning A.P., Massani N.D., Ikram S., Fraser A.G., Hall R.J. Transoesophageal echocardiography as the sole diagnostic investigation in patients with suspected thoracic aortic dissection. Br Heart J 1994;72(5):461-465.[Abstract/Free Full Text]

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