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Spasmodic torticollis: A psychogenic condition?
Journal of Psychosomatic
Research, Vol. 15, pp. 471 to 477.
SPASMODIC
Persamon Press, 1971.
Printed in Northern Ireland
TORTICOLLIS : A PSYCHOGENIC CONDITION ? J. J. COCKBURN*
torticollis is an uncommon condition. It often occurs as part of a more generalised neurological syndrome such as dystonia musculorum deformans and all such cases have been excluded from this study. The condition is defined as a hyperkinesis of the neck muscles leading to dystonic jerky movements of the head and neck, or to a sustained abnormal posture, or both. Even today different opinions exist about whether some cases are psychogenic and others organic in their aetiology. This distinction was emphasised at the turn of the century, particularly by the French authors Brissaud [I] and Cruchet [2]. It is still discussed in many of the modern textbooks of neurology [3,4] but it has proved to be very difficult clinically to distinguish these groups [5]. Increasingly, amongst neurologists, the view is being taken that most if not all cases have an organic aetiology [6, 71. On the other hand, success for psychotherapy, hypnotherapy or behaviour therapy, has been claimed recently by several authors, but usually the numbers of cases reported have been small [8]. The largest of these studies was by Mary Paterson [9] who reported 21 cases, several of whom responded favourably to psychotherapeutic techniques. Many of these papers lack adequate information about follow up and may represent a psychiatric bias in selection. Patterson and Little [lo] have suggested that some successes attributed to psychotherapy may have occurred in cases originally incorrectly diagnosed as suffering from spasmodic torticollis. Neurotic illness is so common that it might be argued that a condition as rare as spasmodic torticollis is unlikely to be psychogenic in aetiology. Even organically oriented neurologists, such as Podivinsky [I 11, have claimed that spasmodic torticollis occurs in patients with a high percentage of abnormal personality patterns. Herz and Glaser, [7] when following up 43 out of 75 patients seen in 15 yr at the Neurological Institute, New York, concluded that “The personality pattern, as illustrated by the patient, and in some instances further defined by the Rorschach test, revealed abnormal trends in 27 cases”; that is in over 50 per cent of those patients followed up. These claims are not supported by clear data. Autopsy reports of uncomplicated cases of spasmodic torticollis are few and conflicting. Tarlov [12] has recently reviewed the pathological literature, published an autopsy of a case carefully studied at the National Hospital, London, and reported negative findings. METHOD This paper presents some results obtained from part of a wider study of spasmodic torticollis, which will be analysed and reported later. The aim is to examine the hypothesis that spasmodic torticollis is either a psychogenic condition, or occurs in patients with abnormal premorbid personalities. Data from patients were compared with information obtained from a matched hospital control group. To exclude a psychiatric bias, it was decided not to include Bethlem Royal and Maudsley Hospital patients, although the study arose out of seeing patients referred
SPASMODIC
* Consultant
Psychiatrist,
Kingston
and Long Grove Hospital, 471
Surrey, England.
J. J. COCKBURN
472
for psychiatric assessment at this hospital. 55 patients were obtained mainly from non-psychiatric sources. 18 patients were obtained from Maida Vale Hospital, 5 from Guy’s Hospital, 14 from three consultant neurologists and 13 direct from general practitioners, mainly with the help of the College of General Practitioners. Only 5 were referred to me in my capacity as a psychiatrist interested in the condition. 46 patients agreed to be seen and were followed up. Of these 46 patients, 36 had been seen by a neurologist (78 per cent) and 25 had been to a psychiatrist (54 per cent). Many of the patients had been seen by other specialists including surgeons, osteopaths and faith healers. At follow up, patients were neurologically examined, mostly in their homes, and information was obtained about many aspects of the symptomatology and the course of the illness. A small number of patients exhibiting more widespread neurological abnormalities, were excluded. Table 1 shows the distribution of patients at follow up. There were 22 male and 24 females examined, 46 patients in all; a follow up rate of 84 per cent. The mean length of follow up was 12.2 yr the standard error of this mean was 1.3 yrs. TABLE I.-PATIENTS
AT FOLLOW
UP
5 1
Dead Emigrated Unwilling Seen by author Total
4: (84%) 55 (100%)
Mean length of follow up = 12.2yr (S.E. of mean = 1.3 yr).
Figure 1 is a histogram showing the age of onset of spasmodic torticollis. The mean age of onset of 39.7 yr, is similar to the means reported in other large series [5, 7, 10, 13, 141. When the patients were matched with the control group the 5-yr-old boy was excluded. It can be seen that the means of the age of onset of the patients’ torticollis and the mean ages of the controls at interview are very similar. Table 2 shows the social class of the male patients. The figures can be seen to be similar to the General Register Office statistics for males in England, Scotland and Wales in 1966 [15]. At follow up a full history was obtained. An assessment was made of the patient’s premorbid personality, using data such as the length of the patients’ longest job in
II _
IO-
IO
v1 c .? '0 a
5-
-
8 7
5
4’
z z
_ IO 20
30 40
50 60
70
Mean age of onset or interview of FIG. l.-Age of onset of spasmodic torticollis. controls. Patients (S.E. ofmean = 2.2 yr) 39.7 yr; Patients (excluding 5-yr-old) 40.4 yr; Controls 40.3 yr.
473
Spasmodic torticollis TABLE
2.-SOCIAL
CLASS OF MALE PATIENTS(G.R.O.) ‘A
figures for G.B. (1966)
Social
class
Patients
% patients
I II III IV V NK
1 3 12 4 1 1
4.5 13.6 54.5 18.2 4.5 4.5 99.8
22
4.3 15.0 48.5 20.2 8.7 3.3 100.0
the case of the male patients, previous psychiatric breakdowns, and the presence of neurotic traits in the personality. Where possible corroborative information was obtained from independent sources, and this was available in 30 out of 46 cases. Information was also obtained about events in the year preceding the onset of symptoms. Finally, patients were asked to complete a Maudsley Personality Inventory (M.P.I.) for their personalities as they remembered them before the onset of their torticollis. In several cases a relative was asked to complete a M.P.I. as he remembered the patient before the onset of torticollis, mainly as a check on the reliability of M.P.I. scores. Data were compared with data obtained from hospital controls. These were surgical cases admitted for a condition in which psychological factors were likely to be minimal. Accident cases, “grumbling” appendices, peptic ulcers, ulcerative colitis cases, etc. were excluded. Most of the controls had been admitted for the treatment of acute appendicitis, cholecystitis, varicose veins, hernias, carcinomas etc. Controls were matched for sex, age within 5 yr of age of onset of torticollis, and social class. The boy, whose torticollis began at 5 yr, was not matched. Matching was satisfactory, with the exception of 2 patients of social class IV who were matched with controls from social class III. A miner (class IV) was matched with a garage forecourt attendant (class III) and a female telephonist (class IV) with the wife of a lorry driver (class III). G.R.O. social class is a crude measure, and it is unlikely that the two substitutions affect the results materially. Independent information was available in all but 6 of the controls. Only one matched control refused to cooperate. The difficulties of retrospective studies are well recognised, particularly when one is assessing personality before the development of a rare and disfiguring illness such as spasmodic torticollis. Some retrospective falsification is to be expected and emphasis should therefore be placed on the more concrete data such as work record. A prospective study in a condition as rare as spasmodic torticollis is not possible. RESULTS The small numbers
involved
AND
DISCUSSION
limit statistical
TABLE 3.-MEAN Patients (before illness) = Controls
16.6 (S.E. = 13.4 (S.E.
analysis
LONGEST
of the results.
JOB
yr of mean = 2.7 yr) yr of mean = 1.9 yr)
Not statistically significant.
J. J. COCKBURN
474
The data obtained from patients were often similar to those obtained from the controls. The mean longest job of the 21 male patients before the onset of torticollis was 16.6 yr compared with a mean of 13.4 yr for the control group. TABLE
4.-MAUDSLEY
PERSONALITY
N scores Mean S.D. Torticollis patients Normals (English) Controls
20.1 19.9 19.0
11.1
1I.0 9.7
INVENTORY
E scores Mean S.D. 23.0 24.9 25.0
9.1 9.7
IO.0
Correlations between independent observers’ M.P.I. ratings and patients’ M.P.I. ratings or controls’ M.P.I. ratings. A. Patients (18 indep. observers) Correlation coeff. for N = +0.75 (S.E. of C.C. = 0.10) Correlation coeff. for E = +0.85 (S.E. of CC. = 0.065) B. Controls (35 indep. observers) Correlation coeff. (S.E. of C.C. = Correlation coeff. (SE. of CC. =
for N = +0.78 0.07) for E = +0.55 0.19)
Results obtained from the Maudsley Personality Inventory showed the scores of the patients, the English normal quota sample described in the Manual of the M.P.I. [lo], and the matched controls, to be similar. The use of the M.P.I. in a group of patients with a condition such as spasmodic torticollis presents difficulties, and an expert opinion was obtained when the research plan was formulated. It was decided that the least unsatisfactory method of using the M.P.I. was to request patients to answer the questionnaire as they remembered their personalities before the onset of the illness. The similarity of the means and standard deviations of the patients’ scores to the scores of the normal English standardisation group and those of the It was decided to use the independent ratings, usually control patients is striking. those of a spouse, to check the reliability of these scores. The correlation coefficients can be seen from the table to be high, with the exception of the correlation coefficient for E scores between controls and their independent observers. As the M.P.I. was never designed to obtain ratings on patients by their spouses, many of the questions are subjective and difficult for an observer to rate on behalf of someone else. The higher N scores in women quoted by Eysenck in the M.P.I. Manual were also found in the patients and controls. The male patients had a mean N score of 18.9 (S.E. of mean = 2.4) and females a mean N score of 21.7 (S.E. of mean = 2.3). The male controls had a mean N score of 16.7 (S.E. of mean = 2.2) and the females a mean N of 20.92 (S.E. of mean = 1.8). Eysenck found that women have higher N scores by about one third S.D. Correlations between patients’ N and E scores were assessed. In the male patients the correlation between N and E was -0.39 (S.E. of C.C. = 0.19) and in the female patients the figure was -0.64 (S.E. of C.C. = g-18). In the controls, correlations between N and E were in the males -0.13 (S.E. of C.C. = 0.21) and in the females -0.24 (S.E. of C.C. = 0.19). These figures compare with figures quoted in the Manual of the M.P.I., of a correlation between N and E scores “in the neighbourhood
475
Spasmodic torticollis
of -0.15; occasionally some samples give positive correlations, and others give higher No standard errors of correlation coefficients are mentioned negative correlations”. in the M.P.I. Manual. The frequency of neurotic symptoms in childhood was alike in both groups. TABLE 5.-NEUROTIC
Patients Controls
xz = 7.0076
SYMPTOMSIN CHILDHOOD Symptoms 2
admitted 3 or more
0
1
30 18
5 10
5 11
5 6
45 45
48
15
16
11
90
(N.S.).
This table does not quite reach the 5 per cent level of significance (Degrees of freedom = 3). The similarities at the neurotic end of the spectrum are perhaps more important than the difference between patients and controls denying any childhood neurotic symptoms. If one combines patients admitting one symptom with those denying any symptoms, the numbers become closer (35 patients compared with 28). Only 3 patients and 4 controls admitted having had previous psychiatric treatment, one patient in each group was an in-patient. Marital status at the onset of the illness showed no significant difference between the two groups. There were 20 unmarried patients at the onset of the illness compared with 12 controls. Marriage rates have increased considerably in the past 20 yr particularly in the younger age groups [ 171. The torticollis patients had been seen over the past 7-8 yr, and the mean follow up was about 12 yr, whereas all the controls had been interviewed in the year 1970. Overall marital stability was rated at interview. This was regarded as a crude measure as it is difficult in one interview to make an accurate assessment of the stability of a marriage, and patients and their spouses frequently deny major difficulties which become, on further investigation, only too obvious. 2 patients and 2 controls admitted that their marriage was unhappy. 2 patients and 5 controls had been divorced. The national divorce rate has approximately doubled in the ten years between 1958 and 1968 [17]. Patients and controls were asked about the occurrence of any major psychological trauma in the year preceding the onset of torticollis, or before the controls entered hospital. No significant differences emerged; 13 patients reported major trauma, compared with 10 controls. An effort was made to obtain information about how It was hoped that this might give frequently a patient visited his general practitioner. some index of neuroticism. Controls were asked about the frequency of their visits to their G.P.s in the year before admission to hospital, for any condition excluding that for which they had been admitted. If Table 6 is greatly simplified into those who TABLE 6.-No.
No. of visits Patients Controls
OF VISITS TO G.P. IN YEAR PRECEDING ONSET OF TORTICOLLIS, PRECEDINGCONTROLS’ ADMISSIONTO HOSPITAL 0
1
2
3
4
5
6
7
27 16
2 11
6 6
0 0
1 2
0 1
0 1
0 1
43
13
1203Iil
8 or more
OR IN YEAR
NK
6 6
4 1
45 45
12
s
90
J. J. COCKBLJRN
476
did not visit their G.P.s at all and the remainder, there is a statistically significant difference at the 5 per cent level. This, however, is probably not clinically significant because of the longer time interval involved for the patients (mean follow up period 12-2 yr). This is supported by the fact that 4 patients were unable to remember, compared with one control. Also if one combines “no visits” and “one visit”, the differences practically disappear (patients nil + one visit = 29, controls, 27). TABLE 7.-P~M~RBID
1. Anxiety Patients Controls
:
PERSONALITY
None/mild 21 30
Moderate 21 15
Severe (G.P.) ;
(N.S.)
2. Depression Patients Controls
:
None/mild 33 27
Moderate 9 16
Severe
3. Obsessional Patients Controls
:
None/mild 36 25
Moderate 7 20
Severe (G.P.) 2 (p < 0.05) 0 (x” = 5.09)
4. Suicidal
attempts:
Patients Controls 5. Alcoholism/drugs Patients Controls
None 44 42 None 43 45
One attempt 1 3 A problem 2 0
;
(G.P.)
(N.S.)
More than one or serious ;
(N.S.) Serious
;
(N.S.)
Patients’premorbid personalities and the personalities of the controlswere assessed, Mild anxiety, depression and mild where possible using corroborative evidence. obsessional features were accepted as normal. Only when the patient had had symptoms severe enough for him to visit his G.P. was he rated “severe”. Similar criteria were used for alcoholic problems. Alcohol was only rated as a problem if it had interfered with work or marriage. No one admitted drug addiction or taking illicit drugs. No significant differences emerged for depressive personality traits, alcoholic Excessive anxiety occurred more problems, suicidal attempts, or excessive anxiety. commonly in the patients, but did not reach the 5 per cent level of significance on a 2 x 2 x2 test using the Yates correction for small numbers. Obsessional features, however, were more commonly admitted in the control group. This statistically significant result requires some comment. It confirms the presence of a considerable amount of mild psychiatric morbidity in the community [18]. Of the 45 controls, 15 suffered excessive anxiety, 18 depression and 20 admitted definite obsessional traits, such as housewives who were unable to leave dishes in the More remarkable is the torticollis sink in order to fulfil an urgent engagement. group, in which 24 patients admitted excessive anxiety in their premorbid personalities This may represent some retrospective but only 9 admitted obsessional traits. falsification, the patient believing that in the “good old days” before his neck became uncontrollable he was able to control everything completely by will-power. Because anxiety usually exacerbates torticollis distant memories of anxiety could be exaggerated. CONCLUSION
The methodology of this study would not necessarily reveal the presence of a small number of psychogenic cases, if such cases exist. If spasmodic torticollis is a hysterical
Spasmodic torticollis
477
one would expect to find either a high incidence of previous psychiatric treatment, instability of job record, frequent precipitating traumatic experiences, or a high N score in the M.P.I. None of these has been demonstrated by this study, and the intractable nature of the illness and electromyographic studies [7] are also against a hysterical aetiology. If spasmodic torticollis is a tic, one should find evidence of frequently occurring obsessional premorbid personalities, and again a high N score in the M.P.I. These have not been demonstrated. It is interesting to speculate that for obsessional personalities to become more common in the patients than in the controls at the 5 per cent level of significance, the number of patients admitting obsessional premorbid traits would have to more than treble; that is, to increase from the 9 patients found in this study, to 31 out of the 45 patients. It is concluded that this study provides no evidence to support the hypothesis that spasmodic torticollis is either a psychogenic condition, or occurs in patients with abnormal premorbid personalities. condition,
Acknow[edgemenfs-I would like to express my gratitude for the most generous help which I have received from many consultants, general practitioners, nursing staff and members of medical executive councils. I have also to thank most sincerely the patients and their relatives for allowing me to subject them to long interviews and examinations, to my two secretaries Mrs. I. Jebson and Mrs. P. Heslop, and also to my wife and family for their encouragement and help. I am indebted to Mr. B. Everitt for statistical advice. I am also indebted to my consultant colleagues for their criticisms and particularly to Dr. R. T. C. Pratt for all his encouragement and interest in this project. Financial assistance was received from Claybury Hospital Management Committee and from a decentralised research grant received from the South West Metropolitan Regional Hospital Board. The South West Metropolitan Regional Hospital Board also provided assistance with computation of results. REFERENCES 1. BRISSAUDE., Lecons sur les maladies Nerveuses. Masson, Paris (1895). 2. CRUCHETJ. R., Traite des Torticolhs Spasmodique. Masson, Paris (1907). 3. BRAIN Lord, Diseases of the Nervous System 6th Edn. Oxford University Press, London (1962). 4. WILSONS. and KINNEARA., Neurology 2nd Edn. Butterworth, London (1955). 5, MEARESR., Features which distinguish groups of spasmodic torticollis. J. Psychosom. Res. 15, 1 (1971). 6. HERZ E. and HOEFER P. F. A., Snasmodic torticollis-I. Phvsioloaic analvsis of involuntarv , motor activity. Arch. Neural. PsyLhiat. 61, 129 (1949). ’ U ’ Clinical evaluation. Arch. Neural. 7. HERZ E. and GLASER G. H., Spasmodic torticollis-II. Psychiat. 61, 227 (1949). Br. Med. J. 1,969 (1940). 8. W&ILESW. H., Treatment of spasmodic torticollis by psychotherapy. Results of osvchotherauv 9. PATERSONMARY T.. Snasmodic torticollis: 12 1, in 21 cases. Lancet ii. 556 (1945). 1 R. M. and LITTLES. C., Spasmodic torticollis J. Nero. Ment. His. 98, 571 (1943). 10. PATTERSON 11. PODIVINSK’~F., Torticollis. In Handbook of Clinical Neurology (Edited by VINKEN P. J. and
BRUYNG. W.), Vol. 6. p. 596. North-Holland, Amsterdam (1968). 12. TARLOVE.. On the nroblem of the natholoev of snasmodic torticollis in man. J. Neurol. Neurosurg. Psychiat.
33, 457 (1970).
’
-’
’
13. SORENSENB. F. and HAMBYW. B., Spasmodic torticollis-results
of 71 surgically treated cases.
Neurology 16, 867 (1966).
14. RYNEARSONE. H. and WOLTMANH. W., Spasmodic torticollis: results of removal of foci of infection and treatment with specific vaccine. Am. J. Med. Sri. 183, 559 (1932). 15. Sample Census of Great Britain 1966. Economic Activity Tables part 3. Table 29. H.M.S.O. (1969). 16. EYSENCK H. J., Manual of the Maudsley Personality Inventory. University of London Press, London (1959). 17. Regisfrar General’s Statistics Review Englandand Wales 1968. Table 02 and Table HI., H.M.S.O. (1970). A Psychiatric Survey. Tavistock Publications, London 18. RYLE A., Neurosis in the ordinaryfamily. (1967).
Research, Vol. 15, pp. 471 to 477.
SPASMODIC
Persamon Press, 1971.
Printed in Northern Ireland
TORTICOLLIS : A PSYCHOGENIC CONDITION ? J. J. COCKBURN*
torticollis is an uncommon condition. It often occurs as part of a more generalised neurological syndrome such as dystonia musculorum deformans and all such cases have been excluded from this study. The condition is defined as a hyperkinesis of the neck muscles leading to dystonic jerky movements of the head and neck, or to a sustained abnormal posture, or both. Even today different opinions exist about whether some cases are psychogenic and others organic in their aetiology. This distinction was emphasised at the turn of the century, particularly by the French authors Brissaud [I] and Cruchet [2]. It is still discussed in many of the modern textbooks of neurology [3,4] but it has proved to be very difficult clinically to distinguish these groups [5]. Increasingly, amongst neurologists, the view is being taken that most if not all cases have an organic aetiology [6, 71. On the other hand, success for psychotherapy, hypnotherapy or behaviour therapy, has been claimed recently by several authors, but usually the numbers of cases reported have been small [8]. The largest of these studies was by Mary Paterson [9] who reported 21 cases, several of whom responded favourably to psychotherapeutic techniques. Many of these papers lack adequate information about follow up and may represent a psychiatric bias in selection. Patterson and Little [lo] have suggested that some successes attributed to psychotherapy may have occurred in cases originally incorrectly diagnosed as suffering from spasmodic torticollis. Neurotic illness is so common that it might be argued that a condition as rare as spasmodic torticollis is unlikely to be psychogenic in aetiology. Even organically oriented neurologists, such as Podivinsky [I 11, have claimed that spasmodic torticollis occurs in patients with a high percentage of abnormal personality patterns. Herz and Glaser, [7] when following up 43 out of 75 patients seen in 15 yr at the Neurological Institute, New York, concluded that “The personality pattern, as illustrated by the patient, and in some instances further defined by the Rorschach test, revealed abnormal trends in 27 cases”; that is in over 50 per cent of those patients followed up. These claims are not supported by clear data. Autopsy reports of uncomplicated cases of spasmodic torticollis are few and conflicting. Tarlov [12] has recently reviewed the pathological literature, published an autopsy of a case carefully studied at the National Hospital, London, and reported negative findings. METHOD This paper presents some results obtained from part of a wider study of spasmodic torticollis, which will be analysed and reported later. The aim is to examine the hypothesis that spasmodic torticollis is either a psychogenic condition, or occurs in patients with abnormal premorbid personalities. Data from patients were compared with information obtained from a matched hospital control group. To exclude a psychiatric bias, it was decided not to include Bethlem Royal and Maudsley Hospital patients, although the study arose out of seeing patients referred
SPASMODIC
* Consultant
Psychiatrist,
Kingston
and Long Grove Hospital, 471
Surrey, England.
J. J. COCKBURN
472
for psychiatric assessment at this hospital. 55 patients were obtained mainly from non-psychiatric sources. 18 patients were obtained from Maida Vale Hospital, 5 from Guy’s Hospital, 14 from three consultant neurologists and 13 direct from general practitioners, mainly with the help of the College of General Practitioners. Only 5 were referred to me in my capacity as a psychiatrist interested in the condition. 46 patients agreed to be seen and were followed up. Of these 46 patients, 36 had been seen by a neurologist (78 per cent) and 25 had been to a psychiatrist (54 per cent). Many of the patients had been seen by other specialists including surgeons, osteopaths and faith healers. At follow up, patients were neurologically examined, mostly in their homes, and information was obtained about many aspects of the symptomatology and the course of the illness. A small number of patients exhibiting more widespread neurological abnormalities, were excluded. Table 1 shows the distribution of patients at follow up. There were 22 male and 24 females examined, 46 patients in all; a follow up rate of 84 per cent. The mean length of follow up was 12.2 yr the standard error of this mean was 1.3 yrs. TABLE I.-PATIENTS
AT FOLLOW
UP
5 1
Dead Emigrated Unwilling Seen by author Total
4: (84%) 55 (100%)
Mean length of follow up = 12.2yr (S.E. of mean = 1.3 yr).
Figure 1 is a histogram showing the age of onset of spasmodic torticollis. The mean age of onset of 39.7 yr, is similar to the means reported in other large series [5, 7, 10, 13, 141. When the patients were matched with the control group the 5-yr-old boy was excluded. It can be seen that the means of the age of onset of the patients’ torticollis and the mean ages of the controls at interview are very similar. Table 2 shows the social class of the male patients. The figures can be seen to be similar to the General Register Office statistics for males in England, Scotland and Wales in 1966 [15]. At follow up a full history was obtained. An assessment was made of the patient’s premorbid personality, using data such as the length of the patients’ longest job in
II _
IO-
IO
v1 c .? '0 a
5-
-
8 7
5
4’
z z
_ IO 20
30 40
50 60
70
Mean age of onset or interview of FIG. l.-Age of onset of spasmodic torticollis. controls. Patients (S.E. ofmean = 2.2 yr) 39.7 yr; Patients (excluding 5-yr-old) 40.4 yr; Controls 40.3 yr.
473
Spasmodic torticollis TABLE
2.-SOCIAL
CLASS OF MALE PATIENTS(G.R.O.) ‘A
figures for G.B. (1966)
Social
class
Patients
% patients
I II III IV V NK
1 3 12 4 1 1
4.5 13.6 54.5 18.2 4.5 4.5 99.8
22
4.3 15.0 48.5 20.2 8.7 3.3 100.0
the case of the male patients, previous psychiatric breakdowns, and the presence of neurotic traits in the personality. Where possible corroborative information was obtained from independent sources, and this was available in 30 out of 46 cases. Information was also obtained about events in the year preceding the onset of symptoms. Finally, patients were asked to complete a Maudsley Personality Inventory (M.P.I.) for their personalities as they remembered them before the onset of their torticollis. In several cases a relative was asked to complete a M.P.I. as he remembered the patient before the onset of torticollis, mainly as a check on the reliability of M.P.I. scores. Data were compared with data obtained from hospital controls. These were surgical cases admitted for a condition in which psychological factors were likely to be minimal. Accident cases, “grumbling” appendices, peptic ulcers, ulcerative colitis cases, etc. were excluded. Most of the controls had been admitted for the treatment of acute appendicitis, cholecystitis, varicose veins, hernias, carcinomas etc. Controls were matched for sex, age within 5 yr of age of onset of torticollis, and social class. The boy, whose torticollis began at 5 yr, was not matched. Matching was satisfactory, with the exception of 2 patients of social class IV who were matched with controls from social class III. A miner (class IV) was matched with a garage forecourt attendant (class III) and a female telephonist (class IV) with the wife of a lorry driver (class III). G.R.O. social class is a crude measure, and it is unlikely that the two substitutions affect the results materially. Independent information was available in all but 6 of the controls. Only one matched control refused to cooperate. The difficulties of retrospective studies are well recognised, particularly when one is assessing personality before the development of a rare and disfiguring illness such as spasmodic torticollis. Some retrospective falsification is to be expected and emphasis should therefore be placed on the more concrete data such as work record. A prospective study in a condition as rare as spasmodic torticollis is not possible. RESULTS The small numbers
involved
AND
DISCUSSION
limit statistical
TABLE 3.-MEAN Patients (before illness) = Controls
16.6 (S.E. = 13.4 (S.E.
analysis
LONGEST
of the results.
JOB
yr of mean = 2.7 yr) yr of mean = 1.9 yr)
Not statistically significant.
J. J. COCKBURN
474
The data obtained from patients were often similar to those obtained from the controls. The mean longest job of the 21 male patients before the onset of torticollis was 16.6 yr compared with a mean of 13.4 yr for the control group. TABLE
4.-MAUDSLEY
PERSONALITY
N scores Mean S.D. Torticollis patients Normals (English) Controls
20.1 19.9 19.0
11.1
1I.0 9.7
INVENTORY
E scores Mean S.D. 23.0 24.9 25.0
9.1 9.7
IO.0
Correlations between independent observers’ M.P.I. ratings and patients’ M.P.I. ratings or controls’ M.P.I. ratings. A. Patients (18 indep. observers) Correlation coeff. for N = +0.75 (S.E. of C.C. = 0.10) Correlation coeff. for E = +0.85 (S.E. of CC. = 0.065) B. Controls (35 indep. observers) Correlation coeff. (S.E. of C.C. = Correlation coeff. (SE. of CC. =
for N = +0.78 0.07) for E = +0.55 0.19)
Results obtained from the Maudsley Personality Inventory showed the scores of the patients, the English normal quota sample described in the Manual of the M.P.I. [lo], and the matched controls, to be similar. The use of the M.P.I. in a group of patients with a condition such as spasmodic torticollis presents difficulties, and an expert opinion was obtained when the research plan was formulated. It was decided that the least unsatisfactory method of using the M.P.I. was to request patients to answer the questionnaire as they remembered their personalities before the onset of the illness. The similarity of the means and standard deviations of the patients’ scores to the scores of the normal English standardisation group and those of the It was decided to use the independent ratings, usually control patients is striking. those of a spouse, to check the reliability of these scores. The correlation coefficients can be seen from the table to be high, with the exception of the correlation coefficient for E scores between controls and their independent observers. As the M.P.I. was never designed to obtain ratings on patients by their spouses, many of the questions are subjective and difficult for an observer to rate on behalf of someone else. The higher N scores in women quoted by Eysenck in the M.P.I. Manual were also found in the patients and controls. The male patients had a mean N score of 18.9 (S.E. of mean = 2.4) and females a mean N score of 21.7 (S.E. of mean = 2.3). The male controls had a mean N score of 16.7 (S.E. of mean = 2.2) and the females a mean N of 20.92 (S.E. of mean = 1.8). Eysenck found that women have higher N scores by about one third S.D. Correlations between patients’ N and E scores were assessed. In the male patients the correlation between N and E was -0.39 (S.E. of C.C. = 0.19) and in the female patients the figure was -0.64 (S.E. of C.C. = g-18). In the controls, correlations between N and E were in the males -0.13 (S.E. of C.C. = 0.21) and in the females -0.24 (S.E. of C.C. = 0.19). These figures compare with figures quoted in the Manual of the M.P.I., of a correlation between N and E scores “in the neighbourhood
475
Spasmodic torticollis
of -0.15; occasionally some samples give positive correlations, and others give higher No standard errors of correlation coefficients are mentioned negative correlations”. in the M.P.I. Manual. The frequency of neurotic symptoms in childhood was alike in both groups. TABLE 5.-NEUROTIC
Patients Controls
xz = 7.0076
SYMPTOMSIN CHILDHOOD Symptoms 2
admitted 3 or more
0
1
30 18
5 10
5 11
5 6
45 45
48
15
16
11
90
(N.S.).
This table does not quite reach the 5 per cent level of significance (Degrees of freedom = 3). The similarities at the neurotic end of the spectrum are perhaps more important than the difference between patients and controls denying any childhood neurotic symptoms. If one combines patients admitting one symptom with those denying any symptoms, the numbers become closer (35 patients compared with 28). Only 3 patients and 4 controls admitted having had previous psychiatric treatment, one patient in each group was an in-patient. Marital status at the onset of the illness showed no significant difference between the two groups. There were 20 unmarried patients at the onset of the illness compared with 12 controls. Marriage rates have increased considerably in the past 20 yr particularly in the younger age groups [ 171. The torticollis patients had been seen over the past 7-8 yr, and the mean follow up was about 12 yr, whereas all the controls had been interviewed in the year 1970. Overall marital stability was rated at interview. This was regarded as a crude measure as it is difficult in one interview to make an accurate assessment of the stability of a marriage, and patients and their spouses frequently deny major difficulties which become, on further investigation, only too obvious. 2 patients and 2 controls admitted that their marriage was unhappy. 2 patients and 5 controls had been divorced. The national divorce rate has approximately doubled in the ten years between 1958 and 1968 [17]. Patients and controls were asked about the occurrence of any major psychological trauma in the year preceding the onset of torticollis, or before the controls entered hospital. No significant differences emerged; 13 patients reported major trauma, compared with 10 controls. An effort was made to obtain information about how It was hoped that this might give frequently a patient visited his general practitioner. some index of neuroticism. Controls were asked about the frequency of their visits to their G.P.s in the year before admission to hospital, for any condition excluding that for which they had been admitted. If Table 6 is greatly simplified into those who TABLE 6.-No.
No. of visits Patients Controls
OF VISITS TO G.P. IN YEAR PRECEDING ONSET OF TORTICOLLIS, PRECEDINGCONTROLS’ ADMISSIONTO HOSPITAL 0
1
2
3
4
5
6
7
27 16
2 11
6 6
0 0
1 2
0 1
0 1
0 1
43
13
1203Iil
8 or more
OR IN YEAR
NK
6 6
4 1
45 45
12
s
90
J. J. COCKBLJRN
476
did not visit their G.P.s at all and the remainder, there is a statistically significant difference at the 5 per cent level. This, however, is probably not clinically significant because of the longer time interval involved for the patients (mean follow up period 12-2 yr). This is supported by the fact that 4 patients were unable to remember, compared with one control. Also if one combines “no visits” and “one visit”, the differences practically disappear (patients nil + one visit = 29, controls, 27). TABLE 7.-P~M~RBID
1. Anxiety Patients Controls
:
PERSONALITY
None/mild 21 30
Moderate 21 15
Severe (G.P.) ;
(N.S.)
2. Depression Patients Controls
:
None/mild 33 27
Moderate 9 16
Severe
3. Obsessional Patients Controls
:
None/mild 36 25
Moderate 7 20
Severe (G.P.) 2 (p < 0.05) 0 (x” = 5.09)
4. Suicidal
attempts:
Patients Controls 5. Alcoholism/drugs Patients Controls
None 44 42 None 43 45
One attempt 1 3 A problem 2 0
;
(G.P.)
(N.S.)
More than one or serious ;
(N.S.) Serious
;
(N.S.)
Patients’premorbid personalities and the personalities of the controlswere assessed, Mild anxiety, depression and mild where possible using corroborative evidence. obsessional features were accepted as normal. Only when the patient had had symptoms severe enough for him to visit his G.P. was he rated “severe”. Similar criteria were used for alcoholic problems. Alcohol was only rated as a problem if it had interfered with work or marriage. No one admitted drug addiction or taking illicit drugs. No significant differences emerged for depressive personality traits, alcoholic Excessive anxiety occurred more problems, suicidal attempts, or excessive anxiety. commonly in the patients, but did not reach the 5 per cent level of significance on a 2 x 2 x2 test using the Yates correction for small numbers. Obsessional features, however, were more commonly admitted in the control group. This statistically significant result requires some comment. It confirms the presence of a considerable amount of mild psychiatric morbidity in the community [18]. Of the 45 controls, 15 suffered excessive anxiety, 18 depression and 20 admitted definite obsessional traits, such as housewives who were unable to leave dishes in the More remarkable is the torticollis sink in order to fulfil an urgent engagement. group, in which 24 patients admitted excessive anxiety in their premorbid personalities This may represent some retrospective but only 9 admitted obsessional traits. falsification, the patient believing that in the “good old days” before his neck became uncontrollable he was able to control everything completely by will-power. Because anxiety usually exacerbates torticollis distant memories of anxiety could be exaggerated. CONCLUSION
The methodology of this study would not necessarily reveal the presence of a small number of psychogenic cases, if such cases exist. If spasmodic torticollis is a hysterical
Spasmodic torticollis
477
one would expect to find either a high incidence of previous psychiatric treatment, instability of job record, frequent precipitating traumatic experiences, or a high N score in the M.P.I. None of these has been demonstrated by this study, and the intractable nature of the illness and electromyographic studies [7] are also against a hysterical aetiology. If spasmodic torticollis is a tic, one should find evidence of frequently occurring obsessional premorbid personalities, and again a high N score in the M.P.I. These have not been demonstrated. It is interesting to speculate that for obsessional personalities to become more common in the patients than in the controls at the 5 per cent level of significance, the number of patients admitting obsessional premorbid traits would have to more than treble; that is, to increase from the 9 patients found in this study, to 31 out of the 45 patients. It is concluded that this study provides no evidence to support the hypothesis that spasmodic torticollis is either a psychogenic condition, or occurs in patients with abnormal premorbid personalities. condition,
Acknow[edgemenfs-I would like to express my gratitude for the most generous help which I have received from many consultants, general practitioners, nursing staff and members of medical executive councils. I have also to thank most sincerely the patients and their relatives for allowing me to subject them to long interviews and examinations, to my two secretaries Mrs. I. Jebson and Mrs. P. Heslop, and also to my wife and family for their encouragement and help. I am indebted to Mr. B. Everitt for statistical advice. I am also indebted to my consultant colleagues for their criticisms and particularly to Dr. R. T. C. Pratt for all his encouragement and interest in this project. Financial assistance was received from Claybury Hospital Management Committee and from a decentralised research grant received from the South West Metropolitan Regional Hospital Board. The South West Metropolitan Regional Hospital Board also provided assistance with computation of results. REFERENCES 1. BRISSAUDE., Lecons sur les maladies Nerveuses. Masson, Paris (1895). 2. CRUCHETJ. R., Traite des Torticolhs Spasmodique. Masson, Paris (1907). 3. BRAIN Lord, Diseases of the Nervous System 6th Edn. Oxford University Press, London (1962). 4. WILSONS. and KINNEARA., Neurology 2nd Edn. Butterworth, London (1955). 5, MEARESR., Features which distinguish groups of spasmodic torticollis. J. Psychosom. Res. 15, 1 (1971). 6. HERZ E. and HOEFER P. F. A., Snasmodic torticollis-I. Phvsioloaic analvsis of involuntarv , motor activity. Arch. Neural. PsyLhiat. 61, 129 (1949). ’ U ’ Clinical evaluation. Arch. Neural. 7. HERZ E. and GLASER G. H., Spasmodic torticollis-II. Psychiat. 61, 227 (1949). Br. Med. J. 1,969 (1940). 8. W&ILESW. H., Treatment of spasmodic torticollis by psychotherapy. Results of osvchotherauv 9. PATERSONMARY T.. Snasmodic torticollis: 12 1, in 21 cases. Lancet ii. 556 (1945). 1 R. M. and LITTLES. C., Spasmodic torticollis J. Nero. Ment. His. 98, 571 (1943). 10. PATTERSON 11. PODIVINSK’~F., Torticollis. In Handbook of Clinical Neurology (Edited by VINKEN P. J. and
BRUYNG. W.), Vol. 6. p. 596. North-Holland, Amsterdam (1968). 12. TARLOVE.. On the nroblem of the natholoev of snasmodic torticollis in man. J. Neurol. Neurosurg. Psychiat.
33, 457 (1970).
’
-’
’
13. SORENSENB. F. and HAMBYW. B., Spasmodic torticollis-results
of 71 surgically treated cases.
Neurology 16, 867 (1966).
14. RYNEARSONE. H. and WOLTMANH. W., Spasmodic torticollis: results of removal of foci of infection and treatment with specific vaccine. Am. J. Med. Sri. 183, 559 (1932). 15. Sample Census of Great Britain 1966. Economic Activity Tables part 3. Table 29. H.M.S.O. (1969). 16. EYSENCK H. J., Manual of the Maudsley Personality Inventory. University of London Press, London (1959). 17. Regisfrar General’s Statistics Review Englandand Wales 1968. Table 02 and Table HI., H.M.S.O. (1970). A Psychiatric Survey. Tavistock Publications, London 18. RYLE A., Neurosis in the ordinaryfamily. (1967).