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Augustus 1964 S.A. TYDSKR I F VIR GENEESKU 'D E 567ANEURYSM OF THE INTERNAL CAROTID ARTERY PRESENTING AS APERITONSILLAR ABSCESS

L. c. J. VAN RENSBURG, M.B., CH.B., M.MED. (SURG.), F.C.S. (s.A.), F.R.C.S. (ENG.), Senior Lecturer in Surgery,University of Stellenbosch, and Kar l Bremer Hospital, Bellville, Cape

Arterial aneurysms are rare in children and aneurysms ofth e internal carotid artery ar e even rarer . Very few caseshave been reported i n the l it erature and the managementof such cases presents a problem.Case Report

The patient was a Coloured male, aged 5 years (Fig. 2).History (from mother). Three weeks before admission thechild injured his l eft a rm bu t definitely not his neck. A fewdays later a swelling appeared on the left side of the neck.This swelling gradual ly increased in size and the pat ientdeveloped a certain degree of dysphagia. Initially the swellingwas painless bu t for about 3 days before admission he experi enced a degree of pain and also developed a slight temperature.Twenty-four hours before admission the child was admitted toanother hospital, where he was treated with antibiotics and adiagnosis of a peritonsillar abscess was made. The patient wasadmitted to the Karl Bremer Hospital on 13 May 1963. Hepresented with a temperature of loo-4P and a pulse rate oflOO/minute. There was a marked swelling of the left side oft he neck and carotid region. A left fac ia l-nerve palsy wasevidentOn examination of the throat there was a marked peri tonsillar swelling, bu t with no oedema or redness of the fauces;the left tonsil appeared to be displaced inferiorly; there wassome exudate in the pharynx, bu t no other abnormalities wereevident Further general examination proved to be negative.Special investigations. WBe 22,OOO/cu.mm. Differentialcount, polymorphs 82%, Iymphocytes 8%, monocytes 2%, staffcells 5%, myelocytes 2%, metra-myelocytes 1%. Hb 71 G/100 rnl., ESR 128 mm. in 1 hour.The patient was first seen by Dr. P. de Vill iers , regist ra r inthe Ear , Nose and Throat Department, who very wisely inserted a needle into the peritonsillar swelling to find a jet ofbright red blood. Subsequently a tracheostomy was performedfollowed by a bilateral carotid angiogram (Fig. 1).

Angiogram. On the left side there was a large saccularaneurysm of the internal carotid artery, about 2t inches abovethe common carotid bifurcation. Cerebral arter ial fi lling wasfound but only of the middle cerebral artery. The right carotidangiogram was found to be normal; there was good cross-fillingto the left side.After routine pre-operative preparation the patient was takento the operating theatre. Because of the high situation of theaneurysm, it was thought that excision and graft replacementwould be very difficult. Because of the patient's age his cooperation was inadequate and it was therefore decided to do anEEG on the ope ra ting table. Under general anaesthesia theinternal carotid artery could be compressed and occluded toelucidate the EEG changes. I f the changes had been marked,it would have been decided to apply a Crutchfield clamp forthe gradual occlusion of the common carotid artery afterligation of the external carotid artery. Whi le the EEG electrodes were being placed and before anaesthesia was inducedthe aneurysm ruptured, which resulted in a sudden gush ofblood from the patient's nose, tracheostomy tube and mouth.While blood was vigorously sucked out of his pharynx andtrachea there was no option but to expose the common carotidartery without anaesthesia. The artery was then immediatelycompressed digitally and this controlled the bleeding. Thepatient was then anaesthetized.By the time the patient was under anaesthesia it wasnoticed that there were no EEG changes, i.e. after compression lasting fo r at least 5 minutes. Compression was continuedwith no EEG changes and therefore the internal and externalcarotid arter ies were ligated. The aneurysm was not exc isedbecause of its high position and because of the close proximityto several cranial nerves.

The patient's immediate postoperative recovery was normal.There were no neurological changes whatsoever. The followingday the swelling of his face had decreased and within 48 hours

Fig. 1. The aneurysm of the left internal carotid a rtery i s wel l demonstrated. Note its h igh position.it was not iced tha t the lef t facial palsy was improving. Withinanother few days the swelling disappeared completely and sodid the facial-nerve palsy (Figs. 3 and 4).

Fig. 2. Before ope ra tion . Note the left-sided swelling of the neck andface and left facial palsy.Fig. 3 . E ig ht days after opera tion . The swelling has subsided and thereis improvement of the facial palsy.Fig. 4 . Fou rt ee n days after operation. There is min imal swe ll ing andthe facial palsy has recovered.The tracheostomy tube was removed after 1 week and t hepatient was discharged on the 20th postoperative day. He was

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568 S .A . MED ICAL JOURNAL 1 August 196seen a month later and found to be absolutely normal. Itshould be pointed out that no cause for this temperature,raised white blood-cell count and raised ESR was found. Therewas no evidence of subacute bacterial endocarditis, tonsillitisor of a septic focus.Interesting AspectsThere are several interesting aspects to fhis case:1. The unusual s ite of the lesion.2. The exact aetiology of the aneurysm.3. The treatment.Zakrzewski1 quotes Killian who published a review ofthe world's literature concerning aneurysms of the carotidsystem and its branches. There were 3,407 cases. Of these,787 were aneurysms of the cervical carotid artery and173 concerned the extracrania l internal carotid artery,i.e. 5% of the total number. He does not quote the agesof these patients. Bergan and Trippel2 state that aneurysmsof the extracranial carotid arteries are rare, but are beingseen in vascular clinics with greater frequency. Crawford,De Bakey and Cooley3 analyzed 107 cases of peripheralarterial aneurysms which were treated over the preceding5 years . Of these there were only 4 carotid-artery aneu

rysms and these were all due to trauma.DISCUSSION

The aetiology of aneurysms are (a) congenital defects,(b) trauma, (c) infection (syphilitic and mycotic), (d) atherosclerosis and (e) post-stenotic aneurysms.In this patient there was a vague history of trauma to theleft arm. I t may be that this was a traction injury but it isdifficult to see how this could have produced an aneurysmof the internal carotid artery. Secondly, the aneurysm wasfairly high in the neck and trauma at that site is difficultto visualize as the artery would be protected by theascending ramus of the mandible and the mastoid process.

The most likely aetiological factor is probably that ofsepsis and this aneurysm should then be labelled as .amycotic aneurysm. Barker,4 in discussing the pathogenes13of aneurysms states that mycotic aneurysms are caused by:1. In fected emboli from subacute bacterial endocar-ditis (SBE),2. Bacteraemia, and3. Contagious infection.Of such cases 90% are usually due to bacterial endocarditis. The author quotes 11 other authors who documented their mycotic aneurysms. Most o f their patientshad SBE. This patient showed no evidence of subacutebacterial endocarditis. His blood culture was negative. I tis possible that he had an infection in the throat, althoughthis was not confirmed by the mother or by his examinationon admission. There was a slight exudate but no evidenceof a true tonsillitis. On the other hand, the patien t hadpyrexia, a raised white blood-cell count and a raised sedimentation rate. No cause for these abnormali ties wasfound. Whatever th e aetiology, the main factor in thispatient was the tr ea tment , as it was obvious that something had to be done fairly rapidly after the patient'sadmission.

TREATME T OF EXTRACRANIAL CAROTID ANEURYSMSAmbrose Pare, in 1552, published the first account ofoperative ligation of the common carotid artery. Hispat ient developed aphasia and a lef t monoplegia. Watson

and Silverstone5 state that the first authentic and completely reported case was done on 17 October 1803. Flemminga ship's surgeon on board HM.s. Tannant, l iga ted thcommon caro tid artery without mishap for a suicidalaceration of the neck. Astley Cooper performed the fircommon carotid ligation fo r an aneurysm of the arteron 1 November 1805. His patient developed a hemiplegion the 8th postoperative day and d ied on the 21st dayRogers6 reports 19 personal cases of ligation of the common carotid artery for intracranial aneurysms. He statethat it is reasonably safe provided that the tolerance teto the common carotid artery is satisfactory. He quotes'I have no hesitation in stating that in a reasonably fperson provided the tolerance test is satisfactory, itsafe to divide the great vessel in the neck. On the othehand, an experience of delayed hemiplegia in one case ointernal carotid ligation along with the series of complcations and fatalities, in recently published cases of ligatioof this artery in the neck, leads me to assert with equaconfidence that ligation of the internal carotid artery asprimary procedure, is dangerous.'In this paper the author advised that the artery shoul

be divided to prevent embolus formation from bloodclowhich formed in the d is ta l segment. He postulates thathere is an anastomosis between the external carotiarteriej) in the neck across the midline. Therefore, aftetying the common carot id artery there is still some bloodflow to the internal carot id . The main difficulty of carotil igation before modern techniques were developed, waresidual neurological defects.Pilcher and Thuss7 surveyed the literature in detai l anconcluded that 20 - 30% of patients will develop symptomreferable to the brain after carotid-artery ligation. Watso

and Silverstone5 llnalyzed 20 cases where the commocarotid artery was ligated fo r carcinoma of the head anneck. 11 of the 20 patients died from cerebral complcations. They suggest that the frequent variations and abnormalities in the anatomy of the arteries of the neck anbrain explain the variety of cerebral complications occurring after common-carotid ligation.Thompson and Austin8 report 6 cases of carotid aneurysms which they t reated by wrapping with fascia latabecause of these grave neurological complications. Theiresults were good. Lahey and Warren9 thought that ligatioof the common carotid artery for carotid body tumorwas prohibitive. Dandylo treated 88 patients, ligating thinternal carotid artery. Five developed neurological complications and 4 were fatal. .Moore and Bakerl l suggest that it seems likely thadevelopment of an increased collateral circulation to th

brain, stimulated by the presence of an aneurysm and ta greater extent by an ar teriovenous fistula, has permi tteocclusion of the common or internal carotid artery witless risk than under other circumstances. They quote DFournestraux's collected series of statistics where therea striking difference in mortality rate from carotid ligatiounder various circumstances: ligation fo r haemorrhag54%, for tumours 46%, fo r aneurysm 135%, for pulsatinexopthalmos 7%. The authors treated 88 patients witcarcinoma of the head and neck where they had to perform a common carotid or an internal carotid-arterligation. Cerebral complications developed in 45-4%, an

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1 Augustus 1964 S .A . T Y DS KR I F V IR G EN EE SK UN DE 569306% died. In a later series the r at e of complication wasmuch lower. They found no significant decreased rate ofcomplication with age; there was no significant differenceaccording to the site of l igation, e.g. where the commoncarotid artery alone was ligated, the complication rate was41%. Where the common and internal carotid or internalcarotid alone was ligated, the complication rate was 47%.They found tha t hypotension was the one common factorthat influenced the percentage of patients who developedcerebral complications. The prevention of hypotension hassignificantly decreased the number of complications in theirseries. Matas12 reported 53 occlusions of the commoncarotid artery with 2 deaths. He believed that his goodresults were due to gradual compression of the vessel.Brackett13 analyzed 65 cases where the internal carotidartery was ligated fo r intracranial aneurysms. 21 of thesepat ients had cerebral complications and there were 6deaths. He found that the important f ac to r was t he siteof the aneurysm. Where the aneurysm was in the supraclinoid position, the percentage of cerebral complicationswas high, whereas in patients with arteriovenous fistulaethe risk was very low. Age was not an import ant factor.Black and German,14 however, on a review of 35 cases ofcommon carotid-artery ligation fo r intracranial aneurysms,had very good re sults with no significant cerebral complications.With the advancement of vascular surgery in recentyears , these extracrarual carotid-artery aneurysms havebecome less of a problem as fa r as treatment is concerned.There are many reports of successful treatment where theaneurysm was excised with reconstruct ion of the artery,using hypothermia and general anaesthesia. Rootman andBradford!5 used hypothermia an d a temporary internalby-pass in a patient with an internal carotid-arteryaneurysm. They anastomosed the external carotid arteryto the distal segment o f the interna l carotid. The patientrecovered fully.Nabseth and Deterling16 report 5 patients with mycoticaneurysms which were treated in several different ways.They state that although there ar e several ways of treatingmycotic aneurysms, they feel that the primary objectiveshould be to remove the aneurysms, as ant ibiotics do notconsistently control the infection. Where the aneurysm issituated in a non-cri tical area, s imple l igat ion plus excisionshould be adequate. In a critical area excision plus arterialreconstruction should be done. I f an end-to-end anastomosis is possible, the result was found to be very satisfactory.Occasionally a prosthesis has to be used and the authorsfeel that a plasti c prosthesis is bet te r than a homograft ,and a vein graft even better still.

The aetiology of the aneurysm in th is pat ient is e ithertrauma or infection. I f one considers the high white bloodcell count and th e raised ESR, a mycotic aneurysm isfavoured. Ideally, this patient should have had the aneurysm excised with reconstruction of the artery. Unfortunately, or maybe fortunately, this could not be donebecause of the patient's dramatic change on the operatingtable and all that could be done was a proximal l igat ionwith an apparent good result. I t is fel t that the excision ofthe aneurysm and reconstruction would have been difficultbecause the aneurysm was so high. An interesting featurewas the seventh-nerve palsy which one feels is due to compression of the nerve after i ts exi t from the stylomastoidforamen. This palsy was due to neuropraxia as thenerve recovered gradually and the patient had completefac ia l movements 3 weeks aft er the operation. I t may beargued that one could ligate the internal carotid artery withimpunity in a child, but there is no authoritative evidenceof this in the literature. I t is obvious that one should nottake unnecessary risks and with sound modern arter ialsurgical techniques this would be irresponsible.

SUMMARYA 5-year-old child with a left internal carotid-arteryaneurysm presented with the clinical signs of a peritonsillar abscess. Fortunately a pre-opera tive diagnosis wasmade by angiography. The patient was treated by internalcarotid-artery ligation as an emergency measure when theaneurysm ruptured on the operating table before inductionof the anaesthesia. The pat ient recovered fully with noresidual neurological signs, although there was a left facialnerve palsy on admission.I wish to thank Dr. C. J. du Toit, Head of the Ear, Nose andThroat Department, Karl Bremer Hospital, for referring thiscase. I am indebted to Dr. J. M. M. Basson of the Departmentof Radiology, Karl Bremer Hospital for the angiograms andalso to Mr. R. Ellis for the photographs.

REFERENCES1. Zakrzewski, A. (1963): J. Laryng., 57, 342. .2 . Bergan, J. J. and Trippel, H. (1963): Surg. Chn. N. Amer., 43, 277.3. Crawford, S. E., De Bakey, M. E. and Coo!ey, D. A. (1959): Arch.Surg. , 78, 226 .4. Barker, W. F. (1954): Ann. Surg. , 139, 84. .5 . WalSon, W. L. and Silverstone, S. M. (1939): IbId., 109, 1.6. Roger s, L. (1949): Lancet, I, 949.7 . P il cher C. and Thuss , C . (1934): Arch. Surg., 29, 1024.8. T h o m p ~ o n , J. E. and Aus ti n, D . J. (1957): Ibid., 74, 80.9. Lahey, F. H. and Warren, 1