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British Journal of Rheumatology 1996;35:682-685 CASE REPORT UNUSUAL COMPLICATIONS OF CERVICAL SPINE SURGERY FOR CERVICAL MYELOPATHY IN RHEUMATOID ARTHRITIS R. MUNRO, M. DUNCAN, H. A. CAPELL and R. JOHNSTON* Centre for Rheumatic Diseases, Glasgow Royal Infirmary and *Institute of Neurological Sciences, Southern General Hospital, Glasgow SUMMARY Cervical myelopathy is a recognized complication of rheumatoid arthritis and other inflammatory arthropathies. In a significant proportion of patients, surgical stabilization of the cervical spine offers the best opportunity for improvement of symptoms and long-term survival. We report two cases that illustrate some potential complications of cervical spine surgery and which also emphasize the need for vigilance when caring for patients in this group. KEY WORDS: Cervical myelopathy, Surgery. INVOLVEMENT of the cervical spine is common in inflammatory arthropathies. In rheumatoid arthritis, up to 85% of those with moderate to severe disease will have X-ray changes in the cervical spine [1], although only a small proportion of these patients will be symptomatic. Symptoms range from mild discomfort in the occipito-cervical region to severe occipital pain, and from mild intermittent weakness to at worst sudden quadriparesis. Cervical spine involvement is usually defined according to the anatomical site, i.e. either atlanto-axial or subaxial, although both may co-exist in the same patient. Atlanto-axial disease is the most commonly observed type in rheumatoid arthritis and is due to damage to the transverse ligament and/or pannus formation at the synovial lined joint around the odontoid peg. Subluxation may be fixed or reducible and most often involves Cl moving forward on C2. Surgical intervention is indicated if there is pro- gressive neurological deterioration or for intractable cervical/occipital pain. Untreated cervical spine disease with a neurological deficit carries a poor prognosis with up to 50% of patients dying in the first 6 months of follow-up [2, 3]. Surgery to the cervical spine is not without substantial risks; these have been summarized in a recent review by Casey and Crockard [4]. Post-operative morbidity and mortality appear to be related to the degree of pre-existing neurological impairment [3]. We discuss below two patients who underwent cervical spine surgery and who developed unusual post-operative complications, one early, the other late. CASE 1 A 64-yr-old patient with a 17 yr history of seropositivc erosive rheumatoid arthritis was referred for neurosurgical assessment for cervical myelopathy. Her main symptoms Submitted 25 July 1995;revisedversion accepted 2 February 1996. Correspondence to: R. Munro, Centre for Rheumatic Diseases, Glasgow Royal Infirmary, Castle Street, Glasgow. were limb weakness and difficulty in walking. Examination showed brisk reflexes with no objective evidence of muscle weakness. Prior to assessment, she had been reasonably well apart from a coxsackie virus illness 10 weeks previously. She had been treated with second- and third-line agents over the course of her disease. The most recent was chlorambucil which had been stopped during her viral illness, although her white cell count was satisfactory at that time. X-rays of her cervical spine showed subaxial disease with 4 mm stable subluxation of C3 on C4 and 5 mm of subluxation on flexion of C4 on C5. Further assessment included MRI of her cervical spine, which showed severe cord compression (Fig. 1). In view of her symptoms and radiological findings, surgical stabilization was indicated. The procedure involved anterior cervical vertebrectomy with bone graft and plate fixation. At the same procedure, she underwent transoral resection of her odontoid peg with posterior fixation with cement and wires using the Brattstrom technique. The initial post-operative period was unremarkable and she was transferred back to the rheumatology ward on the fourth post-operative day. On day 7 post-operatively, she complained of visual hallucinations and became paranoid. Similar episodes had been noted following previous joint surgery. Over the next 4 days, she had further episodes of confusion, but remained apyrexial and physical examination, including neurological assessment, was unremarkable. On day 16, she complained of unsteadiness on sitting upright and became pyrexial with truncal ataxia, although there were no other new abnormal neurological findings. Laboratory investigations showed the development of a leucocytosis (white cell count 15.3 x 10'/0 and a positive sputum culture which grew scanty Staphylococcus aureus. Other investigations, including a chest X-ray, were un- remarkable. She was commenced on oral flucloxacillin for a presumed chest infection and an urgent head CT scan was performed. This showed an area of low attenuation in the left cerebellar hemisphere with ring enhancement following contrast consistent with an abscess (Fig. 2). She was immediately transferred back to the neurosurgical unit for drainage of the abscess. Although culture of the fluid was negative, she was treated withflucloxacillinfor a total of 8 weeks. The remainder of her recovery was unremarkable and she suffered no further sequelae in relation to the abscess. © 1996 British Society for Rheumatology 682 Downloaded from https://academic.oup.com/rheumatology/article-abstract/35/7/682/1780151 by guest on 21 February 2018

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British Journal of Rheumatology 1996;35:682-685

CASE REPORTUNUSUAL COMPLICATIONS OF CERVICAL SPINE SURGERY FOR

CERVICAL MYELOPATHY IN RHEUMATOID ARTHRITISR. MUNRO, M. DUNCAN, H. A. CAPELL and R. JOHNSTON*

Centre for Rheumatic Diseases, Glasgow Royal Infirmary and *Institute of Neurological Sciences, Southern GeneralHospital, Glasgow

SUMMARYCervical myelopathy is a recognized complication of rheumatoid arthritis and other inflammatory arthropathies. In a significantproportion of patients, surgical stabilization of the cervical spine offers the best opportunity for improvement of symptoms andlong-term survival. We report two cases that illustrate some potential complications of cervical spine surgery and which alsoemphasize the need for vigilance when caring for patients in this group.

KEY WORDS: Cervical myelopathy, Surgery.

INVOLVEMENT of the cervical spine is common ininflammatory arthropathies. In rheumatoid arthritis,up to 85% of those with moderate to severe disease willhave X-ray changes in the cervical spine [1], althoughonly a small proportion of these patients will besymptomatic. Symptoms range from mild discomfortin the occipito-cervical region to severe occipital pain,and from mild intermittent weakness to at worstsudden quadriparesis. Cervical spine involvement isusually defined according to the anatomical site, i.e.either atlanto-axial or subaxial, although both mayco-exist in the same patient. Atlanto-axial disease is themost commonly observed type in rheumatoid arthritisand is due to damage to the transverse ligament and/orpannus formation at the synovial lined joint around theodontoid peg. Subluxation may be fixed or reducibleand most often involves Cl moving forward on C2.

Surgical intervention is indicated if there is pro-gressive neurological deterioration or for intractablecervical/occipital pain. Untreated cervical spine diseasewith a neurological deficit carries a poor prognosis withup to 50% of patients dying in the first 6 months offollow-up [2, 3]. Surgery to the cervical spine is notwithout substantial risks; these have been summarizedin a recent review by Casey and Crockard [4].Post-operative morbidity and mortality appear to berelated to the degree of pre-existing neurologicalimpairment [3]. We discuss below two patients whounderwent cervical spine surgery and who developedunusual post-operative complications, one early, theother late.

CASE 1A 64-yr-old patient with a 17 yr history of seropositivc

erosive rheumatoid arthritis was referred for neurosurgicalassessment for cervical myelopathy. Her main symptoms

Submitted 25 July 1995; revised version accepted 2 February 1996.Correspondence to: R. Munro, Centre for Rheumatic Diseases,

Glasgow Royal Infirmary, Castle Street, Glasgow.

were limb weakness and difficulty in walking. Examinationshowed brisk reflexes with no objective evidence of muscleweakness. Prior to assessment, she had been reasonably wellapart from a coxsackie virus illness 10 weeks previously. Shehad been treated with second- and third-line agents over thecourse of her disease. The most recent was chlorambucilwhich had been stopped during her viral illness, although herwhite cell count was satisfactory at that time. X-rays of hercervical spine showed subaxial disease with 4 mm stablesubluxation of C3 on C4 and 5 mm of subluxation on flexionof C4 on C5.

Further assessment included MRI of her cervical spine,which showed severe cord compression (Fig. 1). In view ofher symptoms and radiological findings, surgical stabilizationwas indicated. The procedure involved anterior cervicalvertebrectomy with bone graft and plate fixation. At the sameprocedure, she underwent transoral resection of her odontoidpeg with posterior fixation with cement and wires using theBrattstrom technique. The initial post-operative period wasunremarkable and she was transferred back to therheumatology ward on the fourth post-operative day. On day7 post-operatively, she complained of visual hallucinationsand became paranoid. Similar episodes had been notedfollowing previous joint surgery. Over the next 4 days, shehad further episodes of confusion, but remained apyrexialand physical examination, including neurological assessment,was unremarkable. On day 16, she complained ofunsteadiness on sitting upright and became pyrexial withtruncal ataxia, although there were no other new abnormalneurological findings.

Laboratory investigations showed the development of aleucocytosis (white cell count 15.3 x 10'/0 and a positivesputum culture which grew scanty Staphylococcus aureus.Other investigations, including a chest X-ray, were un-remarkable. She was commenced on oral flucloxacillin for apresumed chest infection and an urgent head CT scan wasperformed. This showed an area of low attenuation in the leftcerebellar hemisphere with ring enhancement followingcontrast consistent with an abscess (Fig. 2).

She was immediately transferred back to the neurosurgicalunit for drainage of the abscess. Although culture of the fluidwas negative, she was treated with flucloxacillin for a total of8 weeks. The remainder of her recovery was unremarkableand she suffered no further sequelae in relation to the abscess.

© 1996 British Society for Rheumatology682

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MUNRO ET AL:. COMPLICATIONS OF CERVICAL SPINE SURGERY 683

FIG. I.—Case 1. Magnetic resonance image showing cordcompression at C4/5.

CASE 2A 49-yr-old woman with a 40 yr history of juvenile arthritis

of a rheumatoid type presented with intractable occipitalheadaches and electric shock sensations radiating to botharms and legs suggestive of L'Hermitte's sign. At the time ofpresentation, her arthritis was well controlled on auranofin,although previously she had required bilateral kneereplacements and wrist surgery. Neurological examinationrevealed no objective signs of cervical myelopathy, althoughreflexes were brisk bilaterally. X-rays showed moderateatlanto-axial subluxation with 5 mm forward slip of Cl onC2. An MRI scan confirmed subluxation at the Cl/2 levelwith mild cord compression. Cervical spine stabilization wasundertaken by posterior Cl/2 wire fixation and bone graftobtained from the right posterior iliac crest.

In May of 1994, she had a recurrence of her neurologicalsymptoms and cervical spine X-ray showed failure of bonyfusion following her previous surgery. A further posteriorcervical stabilization was performed using Cl/2 transarticularscrew fixation and a further bone graft from the iliac crest.After the second operation, the patient complained ofpersistent pain over the right iliac crest donor site. Severalmonths later, she presented at a routine rheumatology clinicvisit with a history of intermittent abdominal cramps and asoft-tissue swelling over her right iliac crest. A plain pelvicX-ray showed a large defect in her right ileum (Fig. 3) andraised the possibility of bowel herniation through the defect.An ultrasound examination was unhelpful, but CT scanningconfirmed the presence of a significant hernia containingbowel through the iliac defect (Fig. 4). In view of the risk ofstrangulation, it was felt that a surgical repair of the herniawas required. This was performed successfully using a wiremesh to close the defect.

DISCUSSIONThe complications of cerebellar abscess and iliac

crest herniation following cervical spine surgery inRA are unusual, but illustrate a number of well-known problems associated with surgery for cervicalmyelopathy.

Sepsis is a major cause of morbidity and mortalityafter neck surgery, partly due to the debilitated state ofmany patients who require such intervention. Sepsismay be local, but is more commonly at a distal site, e.g.chest or urinary tract. A study by Ranewat et al. [5] of33 patients showed a mortality rate of 27% at 2 yr.Two-thirds of the patients who died did so because ofinfection. Unfortunately, the causative organism incase 1 was never isolated. Haematogenous spread froma chest infection is a strong possibility, particularly inview of the positive sputum culture. Another possiblesource is a contaminant from the patient's skin settlingon the wire at the time of surgery. The mostlikely causative organism in this case would be acoagulase-negative Staphylococcus.

The second of our cases illustrates two problems:non-union and complications at the donor site for thebone graft. Zoma et al. [6] reported a series of 32 RApatients undergoing cervical spine stabilization, ofwhich 25% of patients required more than oneprocedure. In general, repeat surgery was requiredbecause of poor union or stability following the firstoperation. More recent studies suggest that surgeryshould be performed early before the development ofbasilar imagination or sub-axial instability, as patientswith isolated atlanto-axial disease appear to have amuch lower incidence of recurrent post-operative

FIG. 2.—Case 1. Computerized tomography of the brain showing aleft cerebellar abscess.

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684 BRITISH JOURNAL OF RHEUMATOLOGY VOL. 35 NO. 7

FIG. 3.—Case 2. Plain pelvic radiograph with a defect of the right iliac crest.

FIG. 4.—Case 2. Computerized tomography of the pelvis showing a hernia containing bowel through the defect in the right iliac crest..

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MUNRO ET AL.: COMPLICATIONS OF CERVICAL SPINE SURGERY 685

instability [7, 8]. Unfortunately, in case 2, despite earlysurgical intervention, the symptoms recurred due toincomplete bony fusion following resorption of thebone graft.

Complications at the bone graft donor site aremore unusual. There have been a number of reportsof fracture at the iliac bone graft harvest [9] site,although there have been no reports of iliac bonedefects leading to bowel heraiation. The harvestedbone for grafting is taken from the outer cortical areawith some medullary bone. It is possible, in our case,that a breach was made in the inner cortical bone withthe defect subsequently enlarging due to boneresorption.

In view of the obvious problems with surgery forcervical myelopathy, it is fortunate that only a smallpercentage of the rheumatoid population withinflammatory joint disease will require surgery.Many of these patients are frail and have a pooroutlook if managed conservatively. Thus, early surgicalintervention offers a significant chance of sustainedimprovement in terms of symptoms and overallsurvival. Nevertheless, constant vigilance is essentialwhen follow-up of such patients is undertaken.

ACKNOWLEDGEMENTS

We would like to acknowledge the assistance ofMiss Ruth McKee, consultant surgeon in relation tocase 2, Dr Alastair Forrester, consultant radiologist

for radiographic studies, and Miss Ann Tierney whotyped the manuscript.

REFERENCES1. Bland JH. Rheumatoid arthritis of the cervical spine.

J Rheumatol 1974;1:319-41.2. Mark JS, Sharp J. Rheumatoid cervical myelopathy.

Q J Med 1981;199:307-19.3. Boden SD, Dodge LD, Bohlman HH, Rechtine GR.

Rheumatoid arthritis of the cervical spine. A long termanalysis with predictors of paralysis and recovery. J BoneJoint Surg 1993;75A:1282-97.

4. Casey ATH, Crockard A. In the rheumatoid patient:surgery to the cervical spine. Br J Rheumatol1995;34:1078-86.

5. Ranewat CS, O'Leary P, PeUicci P, Tsairis P, MarchiselloP, Dorr L. Cervical spine fusion in rheumatoid arthritis.J Bone Joint Surg 1979;61A:1001-10.

6. Zoma A, Sturrock RD, Fisher WD, Freeman P,Hamblen DL. Surgical stabilisation of the rheumatoidcervical spine. / Bone Joint Surg 1987;69B:8-12.

7. Stirrat AN, Fyfe IS. Surgery of the rheumatoid cervicalspine. Correlation of pathology and prognosis. ClinOrthop 1993393:135-43.

8. Agarwal AK, Peppelman WC, Kraus DR et at.Recurrence of cervical spine instability in rheumatoidarthritis following previous fusion: can disease pro-gression be prevented by early surgery? J Rheumatol1992;19:1364-70.

9. Hu RW, Bohlman HH. Fracture at the iliac bone graftsite after fusion of the spine. Clin Orthop 1994;309:208-13.

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