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Clinical Featuresand Associations of 560 Cases Of Journal ofNeurology, Neurosurgery, and Psychiatry 1990;53:1043-1045 1043 J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.53.12.1043 on 1 December 1990. Downloaded from Clinical features and associations of 560 cases of motor neuron disease Ting-Ming Li, Eva Alberman, Michael Swash Abstract nosis of motor neuron disease from multiple In 560 cases of motor neuron disease, sclerosis, cervical spondylosis with myelo- studied retrospectively from their case- pathy and stroke, diseases with which motor notes in three teaching centres, the age at neuron disease may easily be confused, has onset ranged from 13 to 87 years (mean been studied previously using a discriminant 56 years), and the mean duration of ill- analysis procedure in 362 of 378 cases.' This ness until death was 2-6 years. In the sub- showed that 96% of these cases were correctly group of the disease presenting with classified so that, for these hospitals, there was progressive bulbar palsy presenting good agreement in the diagnostic criteria in after age 59 years, there was a previously use. The medical and occupational back- unrecognised excess of females sufficient grounds of these patients were compared with to equalise the sex ratio of incidence of those of 220 control patients, taken from the the disease in this age group. No poten- case records of the same three hospitals for the tially causative clinical associations years 1981-84. These control patients emerged; no relation was noted between suffered from Parkinson's disease, cervical occupational exposure to leather spondylosis, or multiple sclerosis, and had products, trauma or surgical procedures been used as controls in our previous study of and the disease. There was a trend for the differential diagnosis of motor neuron patients with motor neuron disease to disease from other neurological conditions.' give a history of abstention from alcohol. Sex and age of onset The cause of motor neuron disease is un- Of the 560 patients with motor neuron disease known, but epidemiological studies are 342 (61%) were male, a sex ratio of 1-57:1. important in studying new approaches to this There was a marked male excess in patients inexorably progressive and fatal disease. In all presenting below the age of 60 years, but the such studies it is important to assess the sex distribution in patients presenting after accuracy of diagnosis, 2as well as evaluating this age was nearly equal (fig 1). This change the clinical patterns of the disease so that in sex ratio with increasing age was accounted possible associations that might prove impor- for by the preponderance of women present- tant in assessing new notions of causation can ing with bulbar symptoms in the older age be tested. We have reviewed the clinical data ranges (fig 2). The mean age of onset for men http://jnnp.bmj.com/ on 560 consecutive patients investigated at was 54-6 years (SD 12-5 years) and for women three neurological centres in the United King- 58 years (SD 11 4 years). In the whole group dom and report our observations here. of 560 patients the mean age of onset was 56 years (range 13-87 years), although 71% reported the onset of symptoms between age Methods 46 and 66 years (fig 1). Patients and controls on September 26, 2021 by guest. Protected copyright. The medical records of patients diagnosed Age at death and duration of illness with motor neuron disease at The London The age at death was recorded for only 125 of Hospital, London between 1965-82, at the the 560 cases. In these patients the mean age Radcliffe Infirmary, Oxford between 1976- at death was 57-4 years (SD 12-9 years), and 84, and at the National Hospital for Nervous the mean duration of illness was 2-6 years (SD Diseases, London between 1976-84 were re- 2-4 years). The age at death ranged from 22- viewed. Cases were ascertained from the diag- 88 years. nostic index at each of these hospitals using Departments of the ICD codes 356-3 (1964-69), 348-9 (1970- Symptoms and signs at presentation Epidemiology and 78) and 335-2 (1977-84). In these three cen- The majority of patients (63%) reported mus- Neurology, The Royal tres the diagnosis was established in each case cle weakness as the first symptom, 23% London Hospital, London, United only after full clinical evaluation and inves- reported difficulty in speaking, 16% difficulty Kingdom tigation. The latter included haematological in walking and 11% muscle wasting. The TMLi and liver function tests, and radiological disease started in the upper limbs in 44%, in E Alberman M Swash studies and, in most cases cerebrospinal fluid the lower limbs in 37%, and in the bulbar (CSF) examination and electrophysiological muscles in 19%. The latter presentation was Correspondence to: Dr Swash, The Royal studies. especially a feature of older women with London Hospital, London In some, muscle biopsy and necropsy motor neuron disease (fig 2). On examination, El1 BB, United Kingdom were The of these muscle weakness was noted in the upper limbs Received 8 February 1990. studies available. specificity Accepted 5 April 1990 diagnostic procedures in establishing the diag- in 74% of the patients, and in the lower limbs 1 044 Li, Alberman, Swash J Neurol Neurosurg Psychiatry: first published as 10.1136/jnnp.53.12.1043 on 1 December 1990. Downloaded from Tobacco and alcohol usage Past and present smoking habits in the patients with motor neuron disease showed no E Male differences from the controls, although associations with age and male sex were a) * Female evident. There was an overall association 0 between motor neuron disease and past or 0 present abstention from alcohol that was z Il sig- nificant after adjustment for age and sex (p = 0 0055). Thirty six per cent of male and 59% of female patients with motor neuron disease had never drunk alcohol, compared with 25% and 44% respectively of the con- 30-34 35-39 40-44 45-49 70-74 trols. Age at onset Figure 1 Sex and age distribution of 560 cases of motor neuron disease at presentation. Occupational history Data on occupation was not recorded in 22 of the 560 patients with motor neuron disease, in 60%. Muscle wasting was found in the and in 21 of the 220 control cases. One hun- upper limbs in 66%, and in the legs in 40% of dred and twenty nine (43%) of the 218 women in the motor neuron disease group, and 43 patients. Fasciculation was recorded in the in the control upper limbs in 65%, in the lower limbs in (20%) of the 114 women group were classified as "housewives", slightly more 50%, and in the tongue in 37%. The plantar from responses were extensor in of patients. than would have been predicted the 47% cent of the Dysarthria was recorded in 50%, and dys- overall sex ratio. Eighteen per phagia in 410%. Sensory abnormalities were motor neuron disease patients and 12% of the noted in only 2-5% of the cases, and these had controls were in clerical occupations, and 13% other explanations. and 10% respectively were in administrative and executive occupations. There was no Previous medical history difference in the proportions of people engaged in work with leather, leather There was a history of a surgical operation, No other ranging from tonsillectomy to hysterectomy, products, printing or book-binding. in 39% of patients with motor neuron disease. significant differences between the occupa- A crude comparison of the patients with tions of patients and controls were noted. motor neuron disease and the controls showed a similar rate for surgical procedures, al- though the mean age of the control subjects Discussion was less than that of the patients with motor features in neuron disease. Analysis of the prevalence of The presenting clinical patients with motor neuron disease have been exten- surgical operations in different age groups Kurland'4 noted that the sex showed no sively studied.3'3 significant trends or differences ratio was about 1 5:1, reflecting the prepon- from the control patients. Only 0 9% of derance of men in most al- http://jnnp.bmj.com/ patients with motor neuron disease, and 0-5% reported series, though ratios as high as 2 5:15 or even 6 2:1 in of controls reported a history of poliomyelitis. where a different clinical form of the There were no differences in the incidence of India other disease seems to exist,'5 have been noted. In medical conditions between the patients sex ratio was with motor neuron disease and the controls the present series the overall 1 57:1, but for patients older than 60 years it apart from Parkinson's disease, cervical spon- that the dylosis and multiple sclerosis, the diagnostic was equal, an observation suggesting have been on September 26, 2021 by guest. Protected copyright. criteria by which these control patients were sex ratios in previous reports may selected. influenced by the age ranges of the patients forming the data bases of these studies. In the earlier reports the mean age at onset was in the sixth decade, as in our series of patients, in 40T all except the cases reported from India.15 In 35 all these reported cases there was an approx- 30 Male imately equal frequency of origin of the dis- Female In ease in the arms or legs, usually with a smaller a) 25 co proportion beginning in the bulbar mus- 0 20 culature. 0 z 15+ In our cases the disease started in the upper limbs in 44%, in the lower limbs in 37%, and 1 0-- in the bulbar muscles in 19%.
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