Cervicogenic headache. Smith/Robinson approach in bilateral cases

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1 Cervicogenic headache. Smith/Robinson approach in bilateral cases Jürgen Jansen a Ottar Sjaastad b a Department of Neurosurgery, Georg-August Universität, Göttingen, Germany b Department of Neurology, St. Olavs Hospital, University of Trondheim Hospitals, Trondheim, Norway Reprint requests to: Prof. Ottar Sjaastad Gautes gate Trondheim - Norway tora.rui@ntnu.no Accepted for publication: December 28, 2006 Summary The aim was to follow the postoperative fate of cervicogenic headache (CEH) patients with a hard-to-treat, bilateral headache, operated upon by the Smith/Robinson procedure, a stabilization and decompression operation in the cervical spine. CEH is a typically unilateral headache, but in this study, bilateral cases were deliberately selected (n=28). The patients were, otherwise, diagnosed according to the Cervicogenic Headache International Study Group (CHISG) CEH criteria. In most cases, the discs C 4-5, C 5-6 and C 6-7 were affected, and one or two discs were removed by anterior approach; an interbody fusion was carried out. Immediately postoperatively up to 2-3 months there was pain freedom. Secondary deterioration was reported to us in 10 patients, in nine of whom it occurred within three years. The remaining 18 patients were followed up for months; the mean duration of improvement was 22.7 months. Bilateral, Smith/Robinson operated CEH patients seemed to fare as well as unilateral ones. KEY WORDS: cervical spine, cervicogenic headache, headache, Smith/Robinson operation. Introduction The most typical cases of cervicogenic headache (CEH) are the unilateral ones. Accordingly, researchers in the past, before the clinical picture was better outlined, were encouraged to search for unilateral cases. There was a reason for this early purism: it was felt that the bilateral form would be too easily mixed up with tension-type headache (T-TH), a supposedly bilateral headache, and confusion with T-TH could determine the fate of the concept of CEH. It has, however, been reasonably clear since the beginning of the CEH story that there would also exist bilateral cases of CEH: unilaterality on two sides. The reasoning behind this term is the same as that used by Harris (1) in trigeminal neuralgia: the chance that the disease process can be duplicated on the side opposite the affected one must be assumed to be considerable. Indeed, the bilateral cases may even form a rather sizeable group. These patients seem to suffer as much as those with unilateral headache; they should, accordingly, be offered similar treatment possibilities. The pathogenetic mechanisms are assumed to be similar in the uni- and bilateral cases. However, before taking the step of trying to treat this group, by invasive treatment, it should be ascertained, by all available means, that such cases are genuine CEH cases. We have previously (2,3), and also in a recent work (4), reported long-term follow-up results in CEH with unilateral headache, employing a stabilization and decompression operation [Smith/Robinson (5)] in the middle/ low cervical spine. In the early phase of the latter study (4), clearly positive results were obtained in solitary cases. It was then decided to extend the study to include bilateral CEH cases. In the present work, the late results of this operative treatment of severely plagued, long-term CEH sufferers with bilateral headache will be given. Materials and methods Patients This patient series was collected from 1991 onwards, and the study was carried out in a prospective fashion. The material consisted of 28 CEH patients: 20 females and 8 males, with a F/M ratio of 2.5. The mean age at operation was 50 years (range years) and at headache onset, 44 years (range years), giving a mean preoperative period of 6 years (range years). The youngest patient had sustained a compression fracture in the neck. There were three patients with a case history of only 0.5 years, all of them having sustained a neck trauma of the distortion type. The pain was chronic in 26 and intermittent in two patients. Exacerbations were appreciable in 17 of the chronic cases. All were therapy resistant. As regards the pertinent drugs, in particular analgesics, these patients had tried what it is natural to try and seemed to have come to the end of this therapeutic line. Functional Neurology 2006; 21(4):

2 J. Jansen et al. Several patients had suicidal thoughts, and their situation was considered precarious. This was the main reason why some patients (n=3) were treated surgically early in their case history (i.e. ca. 0.5 year after onset), instead of awaiting the natural development of the disorder. The patients were invariably out of work. A total of 14 patients had radioculopathy, two had radioculopathy and myelopathy, while one had myelopathy only; 11 cases were without radioculopathy/ myelopathy. CEH was invariably a main indication for operation, not the radioculopathy/brachialgia; 11 patients were thus operated upon specifically for CEH. All had bilateral head pain (inclusion criterion). In 27 patients, the pain started in the neck and spread to the front; in one solitary case, the pain was steadily occipital. In addition, the pain spread to the interscapular area in seven cases; to the eye in six; to the left side of the chest in four, and to the shoulder in 23 cases. One patient had additional upper jaw pain, and two had lower jaw pain. Tinnitus was present in two patients. Since we were not dealing with unilateral headache without side-shift (as is the case in ordinary CEH), migraine symptoms emerged as differential diagnostic features: nausea was present in seven and a pulsating quality of the pain was present in two patients. The pain was dull/pressing in general. Pain exacerbations could be provoked by neck movements in 21 cases. In seven cases, there were no definite provocation mechanisms (Table I). The pressure exerted externally was approximately of the same magnitude as used in other contexts, e.g. as in fibromyalgia [i.e., ca. 4 kg (6,7)]. An exceptional patient, a 18-year-old male, had sustained a cervical compression fracture. In this patient, the headache could be provoked, the pain radiating from the neck to the forehead. The range of motion in the neck was clearly restricted (Table I). Retrospondylosis was invariably present (Table I). Osteochondrosis was observed in 25 cases, and MRI consistently showed narrowing of the subarachnoid space (Table I). There was close-to-complete correspondence between x-ray and MRI findings with regard to the variables that could be compared. MRI showed contact between protruded disc and cord in 12 cases. Myelo-CT was carried out in 10 cases. Peridural, anesthetic blockades were carried out in 23 cases, and all showed a positive result; root blockade was done in one case with a positive result (Table I). In four cases, no blockades were performed. One patient refused to undergo this test. In the remaining three cases, other supplementary studies showed such clear disc protrusion or fracture, corresponding to the distribution of the brachialgia, that anesthesia procedures were considered superfluous. Except for the distribution of the headache (bilaterality was a premise), the diagnostic criteria of the Cervicogenic Headache International Study Group (CHISG) (8) were generally fulfilled (Table I). If, in addition to unilaterality, there are positive blockades, and attacks can be provoked mechanically, then the CHISG criteria are fulfilled (8), in other words there are three positive criteria. The lack of unilaterality in the present study creates a particular situation. This shortcoming can presumably be compensated for by bringing into the picture four Table I - Comparison of bilateral CEH and unilateral CEH. Headache Unilateral Bilateral (n. of patients=32) (n. of patients =28) Female/male ratio: Range of motion in the neck, restriction (I) (100%) Provocation of head pain, subjective (II) Provocation of head pain objective (III) } (75%) No definite provocation mechanism Spondylosis, cervical x-ray Osteochondrosis, cervical x-ray Narrowing of subarachnoid space Ipsilateral neck/shoulder pain (IV) (82%) Anesthetic procedures, positive (V) (~100%) The unilateral CEH series is described in Jansen J, Sjaastad O, Acta Neurol Scand 2007 (in press). 1 Test not performed in one patient with compression fracture; 2 Information lacking in one; 3 The exceptional case: cervical spine fracture; 4 Positive in all examined patients. I-V = CEH criteria (8) 206 Functional Neurology 2006; 21(4):

3 Neck surgery, bilateral cervicogenic headache separate, diagnostic factors (actually five, if subjective and objective provocations are considered separately, Table I). In all the cases, pathological changes were observed for at least three variables, and in many of them for four variables. In addition, a spreading of the pain from the neck to the forehead was almost invariably present (96%). This is a symptom strongly favoring a CEH diagnosis, although, so far, it is not included among the CEH criteria. All in all, we reckoned the diagnosis to be substantiated in all the cases. Postoperative improvement was defined as either pain freedom or as a 50-80% improvement, when compared to the preoperative situation. A state of <50% improvement after the initial, complete/almost complete improvement (experienced by all the subjects during the period they wore a collar) was defined as a secondary deterioration. Operative procedure The operative procedure, in principle, followed that of Smith/Robinson (5). With the patient placed in the supine position, the Smith/Robinson procedure is performed through an anterior approach to the cervical spine. After sharply dividing the anterior, longitudinal ligament, the contents of the disc space and the dorsal as well as the dorso-lateral disc protrusions are removed. Protrusions of the thickened dorsal ligament are also carefully removed. At variance with the regular Smith/Robinson procedure, dorsal osteophytes, narrowing the epidural space and displacing the spinal dura, are also safely excised with a curette and/or a drill. After decompression of the spinal dura as well as the nerve roots, a bone dowel stemming from the anterior iliac crest is inserted into the intervertebral space to stabilize the intervertebral space. The patients were ordered to wear a stiff collar for two-three months. Ethics committee The local ethics committee stated that no formal application was required, since the method was a well known one and had been practiced for a long time locally. Another indication with a well-known operative technique would, in the same way, not result in any censorship. This study was controlled (see later). Results Intervention and postoperative control The Smith/Robinson operations were carried out at levels C 3-4 to C 6-7. All proved to have disc protrusions. A total of 45 levels were treated in the 28 patients (Table II). Thus, in 17 cases, the operation was carried out at two different levels. The levels at which surgical intervention was carried out were similar in these patients and in unilateral CEH cases (4), and the statistics showed no difference between the distribution of the levels of affection in unilateral and bilateral cases (Table II). All the patients were followed up until the removal of the collar. Whereas control follow ups were scheduled for 3, 5, and 12 months, in reality the patients were controlled at somewhat irregular intervals after removal of the collar, either by consultation or, in the odd case, by telephone. All patients enjoyed pain freedom in the immediate, postoperative period, during which none of them were treated with analgesics. Table III (columns A and B) shows the total number of patients controlled at given times postoperatively. The patients were followed up postoperatively for a varying number of months: range (Table III). Table III (over) should be understood as explained below with reference to the first line (reading across). Table II - Bilateral and unilateral CEH. Comparison of affected cervical levels. Headache Level of pathology: Unilateral Bilateral (n. of patients=32) (n. of patients =28) C C C C C C 7 -Th Total n The unilateral CEH series is described in Jansen J, Sjaastad O, Acta Neurol Scand 2007 (in press). C 5-6 /C 6-7 level operations as a percentage of the total number of operations: unilateral headache: 77%; bilateral headache: 77%. Statistics concerning distribution of cervical levels: P=0.675 (Pearson s Chi-square). Functional Neurology 2006; 21(4):

4 J. Jansen et al. There were 28 patients available for control, as indicated in the leftmost column. All these 28 patients were controlled at some point during the 2-5 months postoperative period (column B). At this regular 2-5 months control, 9 ( pain freedom ) and 2 (50-80% improvement) were doing well; 9+2 make 11. These 11 patients (column A), out of the original 28, were observed, either for the last time during the 2-5 months period and at that time were pain-free/clearly improved (n=8, rightmost column), or suffered a recurrence during the latter part of the 2-5 months interval, i.e. after they had been seen at the regular 2-5 months observation. In this example (first line of the table), there were 3 patients with a 50-70% deterioration; 8 (pain-free/improved, rightmost column) plus 3 (secondary deterioration) make 11. Accordingly, there will be 17 patients (28-11=17; leftmost column, second line) available for control in the follow-up period indicated in column B (6-11 months). Some patients in the secondary deterioration category (second and third columns from the right, Table III) were followed up for a longer time than indicated in column B. These were the re-operated ones (Table IV, rightmost column). The observation time exceeded two years in 10 patients, and six of these patients were known to remain well (Table III). A total of 18 patients fared well (i.e. 64%), with either pain freedom or improvement at the given times (Table III). The figures making this total of 18 are valid only for the time these patients could be followed up (Table III). The mean duration of improvement for the material was 22.7 months, range: months. Upper and lower jaw pains also disappeared after operation. Tinnitus, that was present in two patients preoperatively, continued postoperatively. Secondary deterioration Secondary deterioration appeared from two months onwards. A total of 10 patients in this category (9+1 cases) are known to us (Table III). The deterioration was in the range % (Tables III and IV) when we were first contacted because of it. Six of these patients were treated, either by laminoplasty (n=2); epidural blockades (n=3); nerve root blockades, C 2 (n=2); gangliectomy, C 2 (n=1), or facet joint blockades (n=1) upon pain recurrence (Table IV). One patient was treated in various ways, but rejected a final Smith/Robinson operation. He was finally excluded from further evaluation. The final improvement in this group treated for recurrences was 66% (those treated with blockades and/or operations); the best mean result was obtained in the re-operated patients (n=3): 73% (Table IV). The mean improvement just prior to further therapy was 39% (i.e. deterioration : 61%, Table IV). A deterioration of 100% was observed in only one solitary person; after a later laminectomy, the final improvement amounted to 50%. In recent months, we have, by all the available means, tried to trace these patients, but, for all practical purposes, they are lost to follow up. Thus, the information herein is all that can be reproduced. Control series By sheer luck, it was possible to construct a control series of ten CEH patients. In brief, all these patients were later to be included in a series of unilateral CEH patients to be operated upon with the Smith/Robinson method (5). A detailed description of this series will be presented in that context (4). Prior to the final operation, they Table III - Late postoperative results. Total no., A) Final B) Follow-up Pain Improvement Secondary Continued pain available for observation period freedom (n. of deterioration freedom/improvement control at (n. of (months) 1 (n. of patients) 2 (n. of patients) 3 at indicated given times patients) patients) 2 observation time (n. of patients) 50-80% 50-70% % Total A) Total number of controlled patients at the times indicated in column B; 1 Final observation time for those who were not categorized among the secondary deterioration group at this time; 2 At the indicated time, see B); 3 During the later part of the time interval indicated, see B). 4 This figure denotes the total number of patients (or 64%) who have remained well/relatively well, throughout the time they have been controlled (column B). However, the control period may have lasted anything between 2 and 100 months. The figure, 18, does not indicate that all were pain-free at months. 208 Functional Neurology 2006; 21(4):

5 Neck surgery, bilateral cervicogenic headache had been operated upon (cervical spine, n=8; lumbar spine, n=2) during the period of CEH, and in some cases with the Smith/Robinson procedure (n=6) but generally not with CEH as the main indication. We consider each of these operations to have been carried out at the non-optimal level (for CEH, that is). In some cases, the operations were for coexisting tumors. Each patient was his/her own control. This series has the properties of a proper control series. The operative procedures were invariably of no avail for the CEH. Complications In one patient, there was a transitory recurrent nerve paresis. In another patient, there was a rather painful condition after removal of bone from the iliac crest; however, it needed no particular intervention. Discussion This work has taken a long time to come to maturation: first, the operative method had to be adapted; to begin with the Cloward method (9), then the Smith/Robinson method (5), and ultimately modifications of the latter method to make it more suitable for dealing with the specific pathology of CEH. The operative technique finally adopted was a routine procedure in this setting by the late 1970s. The present study was, therefore, far from experimental in character. Epidural blocks were introduced at the beginning of the 1990s. Since the late 1980s, the Göttingen neuroscience setting has been familiar with the clinical picture of CEH. The present work follows in the wake of these essential stages and is the result of the diagnostic/operative activity of the neurosurgeon (JJ) and the collaboration of the neurologist (OS). These two authors enjoy a close and long-lasting working relationship. Unilaterality of pain is desirable in scientific work on CEH. It has, however, long been realized that there may be unilaterality on two sides (8): the likelihood that a patient with unilateral CEH will develop a bilateral headache is much higher than that of a given control individual developing CEH de novo. Although the basis of our study has been broadened by allowing bilateral cases of CEH, making such allowance Table IV - Patients with secondary deterioration. New headache Further therapy: after original operation: Therapeutic Laminoplasty Physical Final Follow up blockades 1 (LP); training (PT); improvement after further After follow Deterioration Probably (EP) (F) GC2 Analgesics (%) invasive up for: (%) originating (RC2) (AD) therapy (months) from segment: (months) Ref. 120 (n=4) C 3 -C 7 LP PT C 4-5 >C 6-7 Ref. PT, AD (n=1) C 6-7 >C 3-4,C 4-5 LP PT, AD (n=2) C 3-4 instability EP,F,RC2 PT (n=3) 150 C 3-4 ; C 4-5 EP PT, AD >50 C 2, venous RC2 GC compression C 4-5 ; C 6-7 EP AD (n=1) Total Mean: ~ Mean: Epidural (EP), facet joint (F), or C2, anesthetic blockades, with cortisone; repeated injections; 2 This patient demanded a pension postoperatively and was not followed further; 3 >: the segmental abnormality was most marked at the one indicated level; 4 This patient had a complicated postoperative history, partly showing poor results, partly showing 100% improvement; in the end Smith/Robinson operation was rejected; for that reason the patient was excluded from further calculations; 5 After blockades (n=2): 55%; after blockades and/or operations (n=5): 66%; after operation (n=3): 73%. Other abbreviations: Ref.=refused further therapy, due to good quality of life ; LP=laminoplasty; GC2=ganglionectomy, C2. Functional Neurology 2006; 21(4):

6 J. Jansen et al. for bilaterality is actually not a dramatic step. From a nosologic point of view, it is a logical step, which, in the wake of the early purism, has been taken into consideration since As demonstrated in Table I, the clinical profile of the bilateral cases, for most variables, strikingly resembled that of the unilateral cases, except for the laterality. There is, however, a gap between the two as regards the objective provocation of exacerbations, and a somewhat smaller gap as regards shoulder/arm pain. In the foreseeable future, the criterion of unilaterality of headache should still be adhered to, to guarantee the authenticity of the cases. Naturally, the comparison of uni- and bilateral cases can be done subsequently. The results from the present study do not seem to differ appreciably from those in the parallel study, limited to unilateral cases of CEH (4). Also recurrences seem to be of similar magnitude. The striking similarity of the patterns of affected cervical levels in the uni- and bilateral cases (the levels C 5-6 and C 6-7 being heavily represented in both groups, Table II) is in marked contrast to the prevailing view that CEH mainly is a disorder within the GON/C 2 area. If this were the case, then there would probably be two maxima in the distribution profile with a trough between them. It is not even certain that the pathogenesis would prove to be exactly the same in putative upper and lower CEH models. The following should, nevertheless, be emphasized: the two parallel studies comprised 60 patients, collected over years, which corresponds to an annual contribution of ca. 5-6 patients. This study, accordingly, does not suggest that this group is a large one. We have a strong suspicion, however, that the presented series accounts for only a small fraction of the total prevalence of CEH cases. The complication rate was acceptably low in both studies; the complications that did occur were minor ones and mostly had to do with the bone removal from the iliac crest. The results obtained were fairly good. A longer observation time would have been preferable. In wellselected, elderly, severely plagued CEH patients, with protracted suffering, this operation can be a useful alternative, and also for those with bilateral headache. Because of the uncertainties linked to the bilateral form and in particular the putative confusion with T-TH, researchers are still urged to categorize uni- and bilateral forms of CEH separately. References 1. Harris W. Bilateral trigeminal tic. Ann Surg 1939;103: Jansen J. Surgical treatment of non-responsive cervicogenic headache. Clin Exper Rheumatol 2000;18(Suppl 19); S Fredriksen TA, Stolt-Nielsen A, Skaanes KO, Sjaastad O. Headache and lower cervical spine: long-term, postoperative follow-up after decompressive neck surgery. Funct Neurol 2003;18: Jansen J, Sjaastad O. Cervicogenic headache. Long-term prognosis after neck surgery. Acta Neurol Scand 2007 (in press) 5. Smith G, Robinson R. The treatment of certain cervicalspine disorders by anterior removal of the intervertebral disc and interbody fusion. J Bone Joint Surg 1958;40-A McCain G. The clinical features of the fibromyalgia syndrome. In: Værøy H, Merskey H eds Progress in Fibromyalgia and Myofascial pain. Amsterdam; Elsevier 1993: Merskey H, Bogduk N. Classification of Chronic Pain. 2nd edition. Seattle; IASP Press Sjaastad O, Fredriksen TA, Pfaffenrath V. Cervicogenic headache: diagnostic criteria. Headache 1998;38: Cloward R. The anterior approach for removal of ruptured cervical discs. J Neurosurg 1958;15: Functional Neurology 2006; 21(4):