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J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.50.3.349 on 1 March 1987. Downloaded from Journal of Neurology, , and Psychiatry 1987;50:349-353 Short report

Dissecting of the vertebral following cervical manipulation: A case report

JOHN W DUNNE, G NEIL CONACHER,* MAKHAN KHANGURE, CLIVE G HARPERt From the Departments ofNeurology, Neuropathology, Radiology, Royal Perth Hospital, Perth, Australia

SUMMARY Neck manipulation may uncommonly be associated with serious and even fatal vascular complications. Although well recognised, the nature of the vascular has only rarely been directly established by pathological examination. The case is reported of a 43-year-old man who died following neck manipulation, and in whom multiple dissecting aneurysms within both verte- bral arteries were demonstrated radiologically and found at necropsy. Bilateral dissecting aneurysms were found both at the level of atlanto-axial articulation and close to the origins of the vertebral arteries. No predisposition was found, other than early consistent with the patient's age. Protected by copyright.

Alternative medicine, including chiropractic and oste- mainly having been adduced from radiographic opathy, enjoys an ever-increasing popularity. The findings. The nature of the vascular injury has only Australian Health Survey for 1977-1978 estimated rarely been directly established by pathological exam- that in a four-week period just over 2% of Austra- ination,2 3 4 14 thus prompting this report. lians had consulted a chiropractor, naturopath or We describe the case of a 43-year-old man who died osteopath.' However, an increasing number of following neck manipulation and in whom multiple reports indicate that spinal manipulation can be asso- dissecting aneurysms within both vertebral arteries ciated with serious and even fatal vascular2-14 and were demonstrated radiographically and found at non-vascular . Vascular injury related to neck necropsy. manipulation has mainly involved the vertebral and basilar arteries2- 10 13 14 although several reports Case report describe injuries to the internal carotid ."'12 occlusion of the vertebral and A 43-year-old truck driver presented with a three-month his- and tory of intermittent and neck discomfort associ- basilar- - 4 traumatic artery arteries,2 vertebral ated with long-distance driving. He went to a naturopath, http://jnnp.bmj.com/ pseudo-aneurysm,45 haemorrhage from perforation since he had had similar symptoms treated successfully by of the vertebral artery,4 and vertebral artery dis- local manipulation 3 years before. Immediately after a ther- secting aneurysm6 have all been reported, these apeutic twisting of the neck he experienced the sudden onset of vertigo, with transient loss of consciousness followed by persistent vertigo, vomiting, and oscillopsia. There was no previous history of similar episodes, , or other Address for reprint requests: Dr JW Dunne, Mayo Clinic, 200 1st significant illness. Street S.W.. Rochester, MN 55905, USA. Upon examination 6 hours after the onset he was drowsy, but orientated and co-operative. There was no neck stiffness on October 2, 2021 by guest. *Present address: Kingston Penitentiary Treatment Center, or photophobia. A slurring dysarthria, rotatory nystagmus Kingston. Ontario. Canada. in the primary position, and mild bilateral upper limb intention tremor were present. The patient was able to sit +Present address: Department of , University of Sydney, but was unwilling to stand because of vertigo. There were no Sydney NSW 2006. Australia. cranial or neck . Pulse was 70/minute and regular, and sounds were nor- Received 20 December 1985 and in revised form 4 June 1986. 130/70mmHg, Accepted 19 June 1986 mal. Radiographs of the cervical spine showed minimal 349 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.50.3.349 on 1 March 1987. Downloaded from 350 Dunne, Conacher, Khangure, Harper

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Fig 1 (a) Subtraction print, lateral and AP, ofright vertebral artery. The traumatised segment (between arrows) shows narrowing ofthe and irregularity ofthe vessel wall, suggesting a . (b) Subtraction print, lateral and AP, ofleft vertebral artery. In addition to the narrowed vessel lumen (curved arrows) there is a pseudo- (open arrow). J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.50.3.349 on 1 March 1987. Downloaded from Dissecting aneurysms of the vertebral arteries following cervical manipulation: a case report 351 osteophytic lipping of the lower cervical vertebrae but no required intubation and ventilation. He developed a flaccid other abnormalities..Cranial CT scan with and without con- quadriplegia and . Initially reflexes trast was normal. Plasma urea and electrolytes, blood sugar were present, however these were lost over the next 4 hours. level and full blood picture were normal. and Neurological examination a further day later confirmed that hepatitis-B serology were negative as was serum anti-nuclear the patient showed the criteria for brainstem and antibody. advanced life support was discontinued. Over the next 24 hours the patient remained drowsy with persistent generalised headache. He developed moderate Pathologicalfindings neck stiffness, conjugate deviation of the eyes to the left, a The necropsy was performed by Dr J Hilton, forensic right horizontal gaze palsy and impairment of upgaze. Hori- pathologist. General findings were of extensive hypostatic zontal jerk nystagmus was noted on left lateral gaze. Tone pneumonic consolidation, congestion and oedema. The and deep tendon reflexes of the right arm and leg were aug- showed minimal atheroma laid down in streaks, but mented, although there was no obvious weakness and plan- other major arteries appeared healthy. The and spine tar responses remained flexor. were referred to the Department of Neuropathology, Royal Occlusion or dissection of the vertebral arteries was sus- Perth Hospital, for detailed studies. pected and was performed. Both common Macroscopic examination of the brain showed generalised carotid and vertebral arteries were catheterised via a femoral swelling with flattening of the gyri. The , superior approach. The carotid studies were normal. The vertebral medulla, and posterior portion of the left cerebellar hemi- arteries both showed abnormalities extending from mid C2 sphere were markedly softened, and there was bilateral ton- to the level ofthe foramen magnum. On the right side (fig Ia) sillar herniation through the foramen magnum. Extensive the involved segment (between arrows) showed narrowing of necrosis involving both brainstem and was the opacified lumen and irregularity of the vessel wall. On confirmed on microscopic examination. Ischaemic neuronal the left (fig lb) there were similar changes, but in addition change was evident in the carotid arterial territories, but there was a pseudo-aneurysm (open arrow). The posterior without frank necrosis. inferior cerebellar arteries (PICAs) were normal as was the The vasculature at the base of the brain was normal with basilar artery. These appearances were consistent with bilat- no macroscopic evidence of atheroma. The carotid arteries eral dissections. were also normal throughout their lengths. The vertebral The patient was treated conservatively. Forty-two hours arteries were dissected out from their origins at the sub- Protected by copyright. after neck manipulation he had a respiratory arrest and clavian arteries by removal of the anterior arches of the for-

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Fig 2 Sec tion ofthe right vertebral artery at the atlanto-axial articulation, stained to show elastic tissue. is seer: within the dissection which lies between the and adventitia. Elastica van Gieson, x 10. 4.=S .;' J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.50.3.349 on 1 March 1987. Downloaded from

352 Dunne, Conacher, Khangure, Harper amina transversaria. The right vertebral artery showed areas transverse foramina. Such proximal dissections may of contusion of the adventitia at 7 cm and 10-12cm from its be missed in radiographic examinations because of origin. The arteries were sectioned transversely at 1 cm inter- their position. Perhaps because of this, and the infre- vals. Macroscopically, dissecting aneurysms were seen in the quency of pathological examination of the entire right vertebral artery at 0-5 cm and 9-12 cm from its origin, length of the vertebral arteries, extra-cranial vertebral and in the left vertebral artery at 1-3 cm and 1 1-12 cm. The arterial walls were intact in areas between. Thrombus was artery dissections have only rarely been described."' present within the dissections with occlusion of the original Controversy persists as to what degree are cerebral lumina at the level of the atlanto-axial articulation, artery dissections spontaneous or traumatic. Some 11-12cm from the origins of the arteries (fig. 2). Contrast have related all such cases to trauma, often as min- medium from post-mortem radiographic studies was found imal as occurs with cervical rotation. In others, vari- within the false lumen on the right side 3 cm from the origin, ous conditions predisposing to arterial injuries have establishing the existence of a continuity with the true arte- been cited: the presence of cervical osteophytes com- rial lumen. The basilar arteries and PICAs were normal. pressing the vertebral artery,6 ,6 con- Microscopic examination showed that the dissections genital or acquired defects of the media,'8 syphilis,'9 were located between the media and adventitia, and that there was mild thickening of the intima with focal degener- fibroelastic intimal abnormalities,20 homocyst- ation of the elastica along the entire length of the arteries, inuria,2' and .22 However, consistent with early atheroma. In the left vertebral artery an as in our patient where the only finding was early area of fibrosis was found cm from its origin, and small atheroma consistent with the patient's age, most vessel formation between the tunica media and adventitia patients have been young adults with normal ana- was seen. This was felt to be consistent with a possible old tomy and no demonstrable predisposing cause. small dissecting aneurysm. Clinical manifestations are influenced by the extent Later examination of entire vertebral arteries in a man of and nature of injury and the available collateral circu- 46 with motor neuron disease, a;ad a man of 42 with a per- occur late, often forated berry aneurysm of the middle cerebral artery, lation. The onset of symptoms may showed similar thickening of the intima and degeneration of over 24 hours after the injury. 12 Temporary occlusion the elastica. or minor injury to one or both vertebral arteries may Protected by copyright. lead to transient brainstem ischaemia, which is likely Discussion to be the most common result of the vascular injury. The occurrence of such episodes is a warning sign that The course of the vertebral artery and its relationship has sometimes been ignored in the history of patients to the neighbouring structures renders it particularly who develop more serious complications when vulnerable to mechanical trauma. The vertebral manipulation is repeated.7 '0 artery is relatively fixed in its passage through the Lateral medullary has been the most fre- transverse foramina, and particularly in the region of quent syndrome described,2 -' but sudden the atlanto-axial articulation where rotation and tilt- death, quadriplegia, and the locked-in syndrome have ing of the head and neck largely occurs.48 Angio- also been reported.269 In addition, patients with dis- graphy during head rotation has demonstrated verte- section involving the intradural portion may present bral artery compression at the level of Cl associated with a subarachnoid haemorrhage.23 -26 with vertebro-basilar ischaemic symptoms," and Radiographic findings may be diverse, ranging similar occlusion of one vertebral artery has also been from complete occlusion to irregular arterial narrow- shown in volunteers.16 The majority of ing of a variable extent with or without accom- reported vertebral artery injuries which have followed panying dilatation.4-68 17 24-26 The findings in this http://jnnp.bmj.com/ manipulation, yoga, calisthenics, and even spontane- case suggest that the full length of the vertebral arte- ous head turning have occurred in this area.46 12 ries, including their origins, should be examined. Such activities have usually involved cervical hyper- The optimal treatment for patients with dissecting extension, rotation, or both. aneurysm, traumatic or spontaneous, has not been We confirm that the vertebral arteries are particu- determined. A conservative approach to neu- larly vulnerable at the level of the atlanto-axial articu- rologically stable patients has been recommen- lation, where bilateral dissections between the media ded.4 12 17 25 27 In the case of unilateral injury col- and adventitia were found. Importantly, the presence lateral circulation may be adequate, and spontaneous on October 2, 2021 by guest. of a communication between the lumen and the origin resolution has been shown radiographically.'7 27 of the dissecting plane of the right vertebral artery Anticoagulant therapy has been advocated in order to was demonstrated, suggesting that the haematoma prevent thrombosis of the true lumen or distal arose from within the vessel lumen rather than from embolisation from a mural thrombus.46 1727 How- the . The pathological findings also indi- ever, this treatment has not been widely accepted cate that the vertebral arteries may be vulnerable at a because of the occurrence of vessel rupture with point close to their origins, before they enter the perivascular4 or subarachnoid haemorrhage,23 - 26 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.50.3.349 on 1 March 1987. Downloaded from Dissecting aneurysms of the vertebral arteries following cervical manipulation: a case report 353 and the possibility of such therapy extending pre- lation. J Trauma 1977;17;3:248-9. existing dissection, or transforming an infarction into 12 Dragon R, Saranchak H, Lakin P, Strauch G. Blunt to the carotid and vertebral arteries. Am J an haemorrhage.28 injuries intracerebral Surg 1981;141:497-500. In those with progressive signs, , including 13 Green D, Joynt RJ. Vascular accidents to the brainstem revascularisation procedures and proximal arterial associated with neck manipulation. JAMA occlusion, has been tried.24-26 However, the exact 1959;170:522-4. indications for surgery and its advantages over con- 14 Schmitt HP. Rupturen und thrombosen der arteria verte- servative treatment have yet to be established. bralis nach gedeckten mechanischen insulten. Schweiz We thank Dr M Sadka for permission to report this Arch Neurol Neurochir Psychiatr 1976;119:363-79. 15 Barton JW, Margolis MT. Rotational obstruction of the case Torre for secretarial assis- and Miss Gail Della vertebral artery at the atlanto-axial joint. Neu- tance. roradiology 1975;9:117-20. 16 Faris AA, Poser CM, Wilmor DW, Agnew CH. Radio- References logic visualisation of the neck vessels in healthy men. Neurology 1963;13:386-96. 1 Cameron RJ. Australian Health Survey 1977-8: Consul- 17 Goldstein SJ. Dissecting hematoma of the cervical verte- tations with Health Professionals. No. 4322.0. Can- bral artery. J Neurosurg 1982;56:451-4. berra: Australian Bureau of Statistics. 1981. 18 Takita K, Shirato H, Akasaka T, Hukazawa H. Dis- 2 Pratt-Thomas HR, Berger KE. Cerebellar and spinal secting aneurysm of the vertebro-basilar artery. No To injuries after chiropractic manipulation. JAMA Shinkei 1979;31:121 1-8. 1947;133:600-3. 19 Scholefield BG. A case of aneurysm of the basilar artery. 3 Ford FR, Clark D. Thrombosis of the basilar artery with Guys Hosp Rep 1924;74:485-7. softenings in the cerebellum and brainstem due to 20 Berkovic SF, Spokes RL, Anderson RM, Bladin PF. manipulation of the neck. Bull Johns Hopkins Hosp Basilar artery dissection. J Neurol Neurosurg 1956;98:37-42. Psychiatry 1983;46: 126-9. 4 Sherman DG, Hart RG, Easton JD. Abrupt change in 21 Grieco AJ. : pathogenetic mechanisms. head position and . Stroke Am J Med Sci 1977;273:120-32. Protected by copyright. 1981;12:2-6. 22 Hegedus KH, Nemeth G. Fibromuscular dysplasia of 5 Simmons KC, Soo YS, Walker G, Harvey P. Trauma to the basilar artery. Arch Neurol 1984;41:4402. the vertebral artery related to neck manipulation. Med 23 Farrell MA, Gilbert JJ, Kaufman JC. Fatal intracranial J Aust 1982;1:187-8. arterial dissection: clinical pathological correlation. J 6 Schellhas KP, Latchaw RE, Wendling LR, Gold LH. Neurol Neurosurg Psychiatry 1985;48: 111-21. Vertebrobasilar injuries following cervical manipu- 24 Yonas H, Agamanolis D, Takaoka Y, White RJ. Dis- lation. JAMA 1980;244: 1450-53. secting intracranial aneurysms. Surg Neurol 7 Smith RA, Estridge MN. Neurologic complications of 1977;8:407-15. head and neck manipulations. JAMA 1962;182; 25 Berger MS, Wilson CB. Intracranial dissecting 5:528-31. aneurysms of the posterior circulation. J Neurosurg 8 Mueller S, Sahs AL: Brainstem dysfunction related to 1984;61:882-94. cervical manipulation. Neurology 1976;26:547-50. 26 Friedman AH, Drake CG. Subarachnoid haemorrhage 9 Horn SW. The "Locked-in" syndrome following chiro- from intracranial dissecting aneurysm. J Neurosurg practic manipulation of the spine. Ann Emerg Med 1984;60:325-34. 1983;12:648-50. 27 Shuster E, Mokri B, Sundt TM, Houser OW, Piepgras 10 Dumas R et Guard 0. Les accidents vasculaires du tronc D. Spontaneous dissection of the internal carotid arte- cerebral survenant a la suite de manipulations cervi- ries (Abstract). Neurology 1985;35(Suppl. 1):212. cales. Ann Med 28 Hochberg FH, Bean C, Fisher CM, Roberson GH. Phys 1979;22;1:62-70. http://jnnp.bmj.com/ 11 Beatty RA. Dissecting haematoma of the internal car- Stroke in a 15-year-old girl secondary to terminal otid artery following chiropractic cervical manipu- carotid dissection. Neurology 1975;25:725-9. on October 2, 2021 by guest.