Manual Therapy (1997) 22),91-97 (©1997 Pearson Professional Lid Original article Upper cervical instability: are clinical tests reliable? E, Cattrysse, R. A. H. M. Swinkels, R. A.B, Oostendorp, W. Duquet Faculty of Medicine and Pharmacology, Free University of Brussels, Brussels; Practice of Physical Therapy and ‘Manual Therapy; Wuustwezel, Belgium; Practice of Physical Therapy and Manual Therapy. Geldrop: National Institute of Allied Health Professionals, Amersfoort: Practice of Physical Therapy and Manual Therapy, Heeswijk- Dinther, the Netherlands; Faculty of Movement and Sports Sciences, Free University of Brussels, Brussel, Belgium SUMMARY. The aim of this preliminary study was to investigate the reliability of a selection of manual tests used for the examination of instability ofthe upper cervical region. Eleven children with Down's syndrome were examined by four independent examiners with different levels of experience in manual therapy. Three tests as described by Van der El (1992) were used: the lateral displace- ment test, the Sharp-Purser test, and the upper cervical flexion test. Scores of tests and retests were statistically analysed by calculating the percentage of agreement, and Brennan and Prediger’s modified kappa, and with the binomial test. The results showed a significant agreement between test and retest for the upper cervical flexion test in three out of four investigators. Agreement between investigators was significant in four out of six combinations between two investigators, and near to significant for this test (Ppp 07) inthe two remaining combinations. The other two investigated tests and the total score showed no tendancy towards a consistent level of signif- icant intra- or interobserver reliability. INTRODUCTION spinal cord or nerve root, and, in addition, there is no development of incapacitating deformity or pain due to A recent review of the literature by Swinkels & structural changes’. Costendorp (1996) showed that a generally accepted _A practical and more useful definition was given by definition of instability does not exist. White & Panjabi. Frymoyer (1991): “Segmental Instability is a loss of (1978) stated: “All physicians use the term ‘stability’, spinal motion segment stiffness, such that force applica- but they may have a variety of different concepts and tion to that motion segment produces greater displace: definitions in mind as they use it’. They defined the ment(s) than would be seen in a normal structure, result- concept as follows: ‘Clinical instability isthe loss of the ing in a painful condition, the potential for progressive ability of the spine under physiological loads to main- deformity, and neurological structure at risk’. In this tain relationship between vertebra in such a way that description a link is made between the mechanical way there is neither damage nor subsequent irritation to the of describing ‘instability’ and the clinical signs. The author also refered to the stabilizing function of muscle action by stating that the amount of muscular control is| ‘Education in Manual Therapy, Brussels, Practice of Physical ‘more important than the degree of (byper)mobility itself Eoveaton anual Thay. Bre, Piss oF Panjabi etal (1994) defined the concept of ‘neutral RUA HLM. Swinkel, Faas o Medicine nd Pharacology. zone" in elation tothe definition of instability. The neu- Pomgrdute Education in ean ey one ie tral zone is the displacement from the neutral position to rater of Pica Thay and Manual They, Goto, the position of zero tension in a sequence of repeated Phamactogy, Potente Even Mena They movements. An in vito study ofthe effect of extemal Srascs Begum, Nom estas Alle Heath Petesionds, fixation on ¢ motion segment showed thatthe neateal iiaselfDinhcr te Nekenands WeDaque, aly of Medicine 2068 more reduced than the "Range of Motion’. The tnd Pharmacology, Pong Eavaion i Manual Trp authors also stated that in instability the neutral zone Fron of Mavens ad Sports Scenes, re Ulver of increases more than the range of motion. ‘Corespondene fo: EC, Gootossuant 28,2990 Waustwezl Currently the most common way of investigating Belgium, instability in the upper cervical region is by X-ray. The a92. Manual Therapy Aalas-Dens Interval (ADD is measured in fe tral position and extension. The ADI is the distance between the most anterior point of the dens of the axis and the back ofthe anterior arch of the atlas. ‘The American Society of Radiology (Swinkels & ostendorp 1996) pointed out that an. ADI>2.5-3mm {abnormal for adults and atlantoaxial instability starts atan ADI24.5-Smm in children. In some groups of patients atlantoaxial instability is described as relatively common. This is especially the case in Down's syndrome. ‘Table 1 shows the frequencies of instability of the upper cervical segments as described in recent litera- ture Itcan be concluded that between 7 and 30 per cent of all individuals with Down's syndrome show allantoaxial instability Recently ‘the Committee on Sports Medicine and Fitness of the American Academy of Paediatrics’ (Risser etal 1995) published a discussion about the cu rent practice of radiological screening of Down's patients before allowing them to participate in sport activities. Most of the Down's patients with instability axe asymptomatic, ie they show radiographic evidence of instability but they do not have any neurological symptoms. Moreover, atlantoaxial instability is not a stable condition and can change in time. A momentary investigation in the way radiological screening is used, ‘cannot give a correct picture ofthe patients status atthe time of participating in spons. In addition, patients who show asymptomatic atlantoaxial instability early in life may become stable at an older age. The survey also showed that symptomatic instability is very rare. Forty- ‘one well desribed case studies were found inthe ltera- ture, The reproducibility ofa diagnosis made on ara graphic examination is very low, and no_study has confirmed a high risk for neurological complications in atlantoaxial instability. Minor neurological symptoms ae potentially more dangerous if the subject partici- pates in sport. The authors conclude that itis not clear i the current strategy of excluding Down's patients who have radiological evidence of atlantoaxial instability from taking part in sportis very efficient. In manual therapy it is stated that therapists should be very careful in treating the upper cervical region ‘Table 1, Froquencies of atlanoaxial instability in Down's syndrome Yearof Autors Origin Publication Frequency ‘Tashler-Manl| 1965 28 Marte-Tishee 1966 20% Semine etal 1978 12% Poeschel etal 1981 55% Poeschel etal 1983 17% Cooke 1984 Cooke 1984 Shafferetal 1984 Shikataet al 1985 ‘Cremers etal 1988 Selkowit, Austalia 192 because of the possibility of atlantoaxial instability Cyriax (1984) and Oostendorp (1988) described differ- ent studies in which the frequencies of neurological complications after manipulative treatment of the upper motion segments are rated between 1:40.00 and 1:10,000.000. To diminish the danger of severe compli- cations in manual therapy from the upper cervical region, therapists should routinely investigate distur- bances in the vertebrobasilar bloodflow (Oostendorp et al 1995) and make a preliminary investigation of the stability ofthe atlantoaxial complex. ‘At present the reliability of the commonly used man- ual tests is not known and in the current literature noth- ing is reported on the subject (Rana et al 1973; Mior et al 1985; Viikara-Juntura 1987; Zachman et al 1989; Capuano-Pucci etal 1991; Vernon etal 1991, Youdas et al 1991; Yeomans et al 1992; Deneyer et al 199: ‘Swinkels & Oostendorp 1996). The only study deserib- ing the predictive value, the sensitivity and the speci- ficity of the Sharp-Purser test , was made by Uitvlught & Indenbaum (1988). These values were calculated by the authors to be 85% , 88% and 95% respectively. ‘Therefore, this preliminary experimental study was ‘set up (0 estimate the intra- and interobserver reliability ‘of manual tests for upper cervical instability. For this purpose the following null-hypotheses were formulated and tested. HO: The relation between the results of the test and the retest of each of three individual selected tests, and of a total score for the examination of atlanto-axial instabil- ity, is not significantly different frem coincidence (P<0.05). HO: The relation between the scores of two or more independent examiners for each of three individual selected tests, and of a total score for the examination of atlanto-axial instability, is not significantly different from coincidence (P $0.05). MATERIALS AND METHODS Patients Eleven children with Down's syndrome who attended a special school in Belgium (Lokeren) were selected for this study. The children were aged between 3 and 14 ‘years (mean age 9 years) and all of them had an X-ray investigation before being accepted at the institute, nor- mally not before the age of three, This means that the time between joining the institute and the moment of testing could have varied between zero and 11 years. ‘The investigations included films of the upper cervi cal spine in flexion, extension and neutral position. Two of the children were diagnosed radiographically as suf- fering from atlantoaxial instability. The diagnosis was based on the criteria of the American Association of Radiologists, by which an ADI of 5 mm or more is indicative of instability (Swinkels & Oostendorp 1996).Unpercerviel instability 93, Al children accepted for this study were mentally and physically capable of undergoing the selected tests in an adequate way. Informed consent was obtained from all parents whose children took part in this survey. ‘The prevalence of upper cervical instability in the experimental group was calculated from the radio- graphs at 18% (Table 2). This is in accordance with the findings of other authors in recent literature (Table 1). Investigators Four manual therapy practitioners with different degrees of experience in manual therapy took part in this study (Table 3) as well as four observers. The observers also assisted the children whilst they were undergoing the tests. Tests ‘Three tests as described by Van der El (1992) were selected; the lateral displacement test in the supine jon, the upper cervical flexion test in the supine position, and the Sharp-Purser test with the patient seated, Standardization of the tests was as described by ‘Van der El. Lateral displacement test Patients position: Supine lying, Investigators position: Standing by the patients head. Fixation: ‘The index ofthe fixating hand is placed atthe ipsilateral side of, the arch ofthe axis, and attempts to maintain its position. Movements: ‘The index finger of the investigating hand is placed ‘Table 2. Description of patients by age and X-ray findings ‘Age in years 4 stable a stable 2 ‘antble it sable le le sale sible stole state tinstable Patient ‘Stablefunstable ‘Table 3. Investigators profile Initaly MS. MS, spender ——_-Fysioher. Man-Ther Experience in years ‘Country oforign MPimle 1982 199013 2RGimle 1986 1998 3 DRifemale 1983 / 2 4 Ghiemale 19937 is ‘Netherlands ‘Netherlands Belgium Netherlands ipsilateraly to the arch of the atlas, the base of the thumb is placed against the lateral side ‘of the occiput, and the thumb against the maxilla; the investigating hand makes @ lateral shifting movement of CO snd C1 on C2. No displacement can be made in the normal situation. ‘The testis primarily carried out between Cl and C2; CO is included ia the movement, because CC-CI blocks may «diminish the degree of ‘observed displacement; lateral displacement should be attempted in both directions. Interpretation: Additional notes: Upper cervical flexion test Patients position: Supine lying Investigators position: Standing by the patients head. Fixation: ‘Thumb and index finger of the fixating hand maintain the position ofthe axis by holding the arch ofthe axi The investigating hand is placed on te occiput ofthe patiens head, while the investigators shoulder isin contact withthe patients forehead; in a very subtle way a ventral flexion movement is made. samentous laxity and neurological disturbances. ‘Movements: Interpretation: Sharp-Purser test Patients position: Sitting; the head is relaxed and ina slightly flexed position, Standing beside the patient. ‘The index finger of the fixating hhand is placed on the spinous process of the axis and ‘maintains its position. ‘The palm of the investigating hhand is placed on the patients forehead and exerts a slight pressure in a dorsal direction, Ligamentous laxity in the atlantoaxial complex and possible neurological disturbances. Investigators position: Fixation: Movements: Interpretation: Scoring All scores for the tests were registered as positive (+), meaning ‘instability is observed", or negative instability is observed!94 Manual Therapy There were no preset criteria for the total score. The {otal score expressed, was the investigators final opin- ion of stability in the patients upper cervical segments, based on the results of the three individual tests. It was scored in the same way as the individual tests (i.e.+/-) Instruments Standard therapy couches were used in this study. In order to make the study double blind, the investi- gators were asked to wear darkened swimming glasses ‘whilst examining the patients during retest. Organisation and standardization of the investigation [No special taining was given tothe investigators but all investigators in this study declared being familiar with the selected tests. There Were no practical problems in executing the tests Four examiners worked simultaneously in four sepa- rate rooms. All examiners examined all patients using the three selected tess. The tests took place before noon. ‘The retests were caried out in the afternoon of the same «ay. The sequence in which the patients were examined was chosen at random. The patients were accompanied by one of the physiotherapists of the school who put them at their ease. These physiotherapists also had to act as observers for the investigation, and had to write down the scores made by the examiners. Examiners were tested against themselves and against each other, not against X-ray findings because the review of literature indicated that X-ray findings can not be taken asa ‘golden standard’ Statistical analysis ‘The results of test and retest were analysed in a 2x2 table for each of the investigators. Scores on the individ- ual fests were compared with those of each of the other invesigators in the same way. In analysing the results percentage of agreement (PA) and Brennan & Prediger’s modified kappa (k,) were used as coefficients of reliability (Brennan & Prediger 1981). Significance of departure from a chance distribution in the 2x2 table was measured with the binomial test between number of agreeing and the number of non- agreeing scores. The one tailed significance was calcu- lated with 0.50 as expected proportions. RESULTS Intraobserver reliabili ‘Tables 4a-4d give the results of the measurement of the intraobserver reliability. The values of the reliability Table 4 (2) Inraobserver eelibilty forthe lateral dsplacemest est Investigator PA Pow 1 ais 64 03" 2 543 0.09 050 3 636 027 om 4 w3 087 02 (@)Inraobserve et lity forthe upper cervical flexion est Investigator PA a Pi 1 1000) 1.00 <0oi 2 636 “02 027 3 SLs ot 03 4 3 ost 02 (6) nirabserver ibility for the Sharp-Purser test Investigator PA, ky Ps Hl 83 ost 002" 2 2 as. on 3 sis 029 050, 4 3 ost 02" (@)Intaobserver eal forthe otal care Investigator PA ky Pe Ir I 04s oat 2 636 02 02 3 364 on 039, 4 3 067 02" ‘= significant at 5% level ss Significant at 1% level PA = percentage of agreement Ke modified kappa, P= Probability level ofthe binomial test. coefficients PA and k, indicate the level of association between test and retest scores. The probability level (sia) is obtained from the binomial test between the number of agreeing and number of non agrecing test- retest scores. A significant value of Pig indicates significant directional departure from a chance ratio between agreement and non-agreement, hence a signif cant relation between test and retest scores. Kappa-values can be interpretated by the following classification (Feinstein 1987): <0 is ‘poor’, 0 to 0.20 ‘slight’, 0.21-0.40 ‘fair’, 0.41-0.60 ‘moderate’, 0.61-0.80 ‘subtantial” and 0.81-1.00 is ‘almost perfect” agreement. Investigator 3 shows a significant level of intra ‘observer reliability forthe three selected tests but not for the total score. The kappa scores for these significant findings vary between 0.64 and 1.00, and the percentage of agreement varies between 81.8 and 100.0. Investigator 2 shows no significant Pac, levels and intraobserver reliability for investigator 3 is only sigr icant forthe upper cervical flexion test (Pyin =0.03) Investigator 4 obtains a significant py level of 0.02 forall tests (PA =83.3 and k,=0.67). For all these: icant findings kappa levels are ‘substantial to almost perfect‘Tables Sa-Sd give the results of the measurement of the interobserver reliability for each test separately: the values of the reliability coefficients PA and ky, and Ppin obtained from the binomial test between number of agreeing and number of non-agreeing scores. A high value of PA and k, indicates a high level of association between the two observers. A significant value of Pyia indicates a significant positive relation between the scores of the two observers. ‘The lateral displacement test and the total score show no significant level of interobserver reliability for none of the six combinations between two investigators. ‘The Sharp-Purser test is only significant for the iner- observer reliability between investigator 1 and 4 (Pj=0.02). ‘The upper cervical flexion test shows significant interobserver reliability in four out of six combinations. In the other two combinations the significance level equals 0.07, ‘able S(e) Imerobserver liability for the lateral displacement est Investigator PA Pon 12 500 0.00 061 re is2 5.64 0.99 rs 500 0.00. ost 23 86 027 027 2 500 0.0 061 4 ma 4s oul (©) Imerobserver liability forthe upper cervical flexion est Investigator PA 5 Pai re 833 os? 02+ 3 S18 ost 03" ey 750 050 007 2 1000 1.00 oor 2 750, 030 007 a BL ost 03+ ()merobserver eliability forthe Sharp-Puser test Investigator PA ky Pie 12 583 017 039 3 545 0.09 030, ra 333 067 02 23 385 009 050 Da 583 017 039 a na 04s on (@onerobserver elibity for the Teal sore Pr on 050 050. oor 09 050 0.09 030 0.09 07 PA™= percentage of agreement moses kappa, ‘Pra probability level ofthe binomial est. Uppercervicalinstabiry 95 In all of the significant findings percentage of agreement varies between 81.8 and 100.0 , and the k varies between 0.64 and 1.00 (ie. ‘substantial 10 almost perfect’). DISCUSSION ‘The prevalence of atlantoaxial instability in the group of patients studied, was very similar to the rates mentioned in the literature. Several authors in different countries mention a prevalence of atlantoaxial instability of between 7% and 30% for patients with Down’s syn- drome (Table 1). In this study the prevalence was 18%. Although the group of patients was small, it was unique because all patients had been radiologically screened previously. In the literature it is mentioned that atlantoaxial instability is nota static concept, but can change in time. ‘This means that the situation of a group of patients at the ‘moment of manual testing is not always the same as it was at the time of X-ray screening. Especially if this screening took place some time before, asin this study. ‘The group of patients in this study consisted of children only. Specific practical problems were related (o this fact. For example, hand placement cannot be the same as in adults because of the size of vertebral segments and reference-points, ‘The lateral displacement test, the upper cervical flex- jon test and the Sharp-Purser test as described by Van der El (1992) were chosen because they are among the few well described tests for the investigation of the upper cervical stability (Swinkels & Oostendorp 1996). ‘The description ofthese tests by Van der El was used as a standard for use in this study. No special training ‘was given to the investigators, but all investigators declared being familiar with the tests. There were no practical problems in executing them. Several investigators in this study, however, men- tioned difficulties in interpreting the tests as described, even for the lateral displacement test: the lateral dis- placement test should be scored on the existence of dis- placement or not; this clear criterion seems to have few practical implications however. The test and retest took place on the same day. This ‘may have a negative influence on the results, as fatigue of the investigations may have been a factor. This could be a factor for the patients as well as for the investi- gators Its possible that a learning process occurred due to the proximity of the tests, which may have influenced the results in a positive way. Some of the examiners clearly felt more sure of their interpretations in retest ing. For this study investigators were selected with differ- cent degrees of experience in physiotherapy and manual therapy. One can, therefore, expect that a higher experi- cence would lead to a higher level of intraobserver relia- bility (Tables 42-40).96. Manual Therapy Only investigators 1 and 4, ie the most and the least experienced, showed significant intraobserver reliabi ity for each of the three selected tests. Investigator 4 (least experienced) also showed significant intraob- server reliability for the total score. Investigator 3 Gust finishing her studies in manual therapy) showed si ‘cant intraobserver reliability for the upper cervical flex- ion test. No significant intraobserver reliability was obtained for investigator 2. Based on these results it cannot be concluded that a higher degree of experience in manual therapy leads to a higher degree of intraobserver reliability for the selected tests. In analysing the raw scores, however, investigator 4 was shown to be the only investigator with a very low number of positive findings. From a total of 88 findings only eight were scored as ‘posi- tive’. These included the total scores which were an expression of the investigators final conclusion on the existence of instability in the patient, based on the findings of the individual tests. Statistically this way of scoring of investigator ¢ leads to a high degree of intraobserver reliability, but it can also be an expres sion of the investigators inability to decide on the existence of instability. In this case the most experi- enced manual therapist (investigator 1) would be the only one with a realistic significant level of intraob- server reliability ‘The results show that the upper cervical flexion test hhas a significant intraobserver reliability for 3 out of 4 investigators (Ppix< 0.05) (Table 4a-d). In 4 out of 6 combinations between two investiga- tors, the upper cervical flexion test shows also a signi cant level of interobserver reliability (Table 5b). In the ‘two other combinations the probability level (Py 0.07) is not significant, but close to the pre-set level of P<0.05. The kappa-score of 0.50 attains a ‘moderate’ level in these two cases ‘These results indicate an acceptable degree of intra- ‘and interobserver reliability for the upper cervical flex- ion test. ‘The lateral displacement test and the Sharp-Purser test have significant intraobserver reliability for 2 out ‘of 4 investigators; and only the Sharp-Purser test has significant interobserver reliability between investiga- tors | and 4 (ie, the most and least experienced). Due to these results, the two tests cannot be regarded as having an acceptable level of intra- and interobserver reliability The total score only gives one significant intraob- server reliability level for investigator 4, and shows no interobserver reliability. It can, therefore, not be accepted as a good reproducible methods forthe invest gation of upper cervical instability in this study. Although the Sharp-Purser test is described in literature as @ good test because of a high degree of predictive value, sensitivity and specificity (Uitvlught & Indenbaum 1988), the results of this study show no significant reproducibility in the investigated group. ‘The question arises whether predictive value, sensitivity and specificity are valuable parameters when reliability is not sufficient, It might, however, be interesting to investigate the predictive value, the sensitivity and the specificity of the upper cervical flexion test in further studies. Tt seems that there is a great need for standardized criteria for interpreting these tests. These are not avail- able in the current descriptions of the selected tests. (One could also consider other criteria such as provo- cation of pain, paresthesia or other discomfort. In this group of patients these criteria could not be used as the children were not capable of adequately reporting such symptoms. ‘The sample used in this present study was rather small, This may have played a negative role inthe statis tical treatment, reducing the chance of finding signifi- cant relationships, if present. Further investigations in other groups of patients ‘may confirm the findings of this study or modify them. ‘Adding other criteria may have great influence on the intra- and interobserver reliability of the tests investi- ‘gated in this study, In the meantime it is advisable to be very cautious when interpreting these tests which rely on the amount of displacement and the end-feel, as a screening for upper cervical instability. CONCLUSION Based on the results of this study it can be concluded that the upper cervical flexion test described by Van der Ef (1992), shows an acceptable degree of intra- and interobserver reliability in this study on a group of 11 children with Down's syndrome. In three out of four investigators the intraobserver reliability was significant and in four out of six combinations between two examiners the interobserver reliability ‘was significant. This gives a fait indication that the null-hypotheses of this study for the upper cervical flexion tests should be rejected. The null hypotheses ‘must, however, be maintained for the lateral displace- ‘ment test, the Sharp-Purser test and for a total score based on the conclusions of the three selected tests: the relation between the results of the test and the retest of the above mentioned tests for the examina- tion of the atlanto-axial instability, and of the total score is not significantly different from coincidence at 5% level (HO/intraobserver reliability). Moreover the relation between the scores of four independent examiners on these same tests for the examination of the atlanto-axial instability, and of the total score, is not significantly different from coincidence at a 5% evel (HO’interobserver reliability). The results show no tendency towards a consistent level of significant intra. and interobserver reliability for these tests in a Down's syndrome population.Upper cevial instability 97 Acknowledgements ‘The authors wish to thank the head, staff and the children of the “Windekind Istute(Lokeren Belgium), and the investigators and observers who took part in his study for their eo-cperation, References Brennan RL. Preiger DJ 1981 Coefficient kappa: some uses, ‘misuses alternatives. Educational and Psychological ‘Measurements 41: 657-699 CCapuano-Pucei D Rhealt W, Aaukai Bracke M, Day R, Pastick ‘M 1991 Intatester and ntertester liability of the cervical range ‘of motion device. archives of Physical and Medical Rehabilitation 724-338-340 Cooke RE 1984 Atlanaria instabii syndrome. 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