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333

TRANSACTIONS OF THE ROYAL SOCIETY OF TROPICAL MEDICINE AND HYGIENE.

Vol. 44. No. 3. December, 1950.

THE LATHYRISM SYNDROME.*


BY

C. GOPALAN, M.D., PH.D., Nutrition Research Laboratories, Coonoor, South India.

Lathyrism has been reported as occurring in " epidemic " form in certain parts of India, where lathyrus forms part of the dietary of the population. Thus outbreaks of the disease have been recorded in Central Provinces (BUCHANAN, 1902), Rewa (ACTON, 1922 ; YOUNG, 1927), Gilgit (MCCARRISON, 1926; MACKENZIE, 1927), United Provinces (SCOTT, 1930), Punjab (SHAH, 1939), and Bhopal (SHOURIE, 1945). There has been no report of cases of lathyrism from South India where lathyrus is not consumed. MINCHIN (1940), however, reported a series of cases of " primary lateral sclerosis " clinically indistinguishable from lathyrism, from the Government General Hospital, Madras. The present investigation shows that cases presenting the clinical features of lathyrism occur in a sporadic form even in South India. Since, however, South Indian diets do not contain lathyrus, it would seem that lathyrism syndrome is not solely confined to the lathyrus-eating population. The following report is based on 11 cases investigated by the writer and 50 other cases collected from the records of the Government General Hospital.
INCIDENCE.

Economic status.The disease was met with among the poorest classes. The cases investigated by the writer had family incomes of less than Rs. 50/per mensem. The others were cases from the general wards of the hospital where only poor patients are admitted. Dietary habits.All the patients were subsisting on grossly inadequate diets. The dietary consisted mainly of rice gruel with occasional helping of cheap vegetables ; milk, meat and eggs were rarely, if ever, consumed. There was no evidence that lathyrus seeds had ever formed part of the diet of these patients. Age.The majority of subjects were young adults. The incidence was particularly heavy between the ages of 20 and 40, 41 of the 61 cases belonging to this age period. There were six cases in this series which were below 20 years and only four above 50. The youngest patient was a boy of 6 years and the oldest a man of 59. * I am grateful to Dr. V. N. PATWARDHAN, Director, Nutrition Research Laboratories, Coonoor, and Sir GEORGE MCROBERT for their helpful interest in this work.

334

THE LATHYRISM SYNDROME

C. GOPALAN

335

Sex.There was a striking preponderance of males over females in the series, the proportion of males to females being three to one. Locality.The disease was not apparently confined to or predominant in aiy particular portion of the province. The present series included Tamils and Telugus from different districts.
TADLE

1. 3(>-39 40-49 9
1
1

0-9 M ales Fe males 1 1 ' '

10-19 3 1

20-29 17

60- 59

Total.

i j

'I

15

THE ONSET.

The onset of the disease was variable. In nine cases, the history obtained was that the patient had retired to bed apparently normal, and on waking up found inability to use his lower limbs. In 21 other cases the onset was subacute, the disability progressing to its full-fledged state within 10 to 20 days. In the remaining 19 cases, the disease set in insidiously ; there was gradually increasing- weakness and stiffness of the lower limbs, the process being spread out ever 1 or 2 months. In four cases there was a history of fever of short duration (4 to 7 days) immediately preceding the attack. Several patients in this series had had the disease for many years when they were first seen. There was no history of remissions and exacerbations of the disorder in these subjects. Eleven patients in the series had suffered from paraesthesias of the lower extremities immediately preceding, during and for some time after the onset of the disease. These took the form of tingling, pins and needles, numbness, cramp-like pains and burning sensation in the soles of the feet.
CLINICAL FEATURES.

The presenting complaint was inability to use the lower limbs. The limbs were reported to be weak and stiff. The disability was so great in some palierts that they were confined to bed. Others managed to get about with the aid of sticks. The patients were markedly ataxic while attempting to stand or walk and the gait was spastic. On examination, the tone of the muscles of the lower limbs was found to be considerably increased and their power very much diminished. The limbs were spastic. There was no fibrillation ; wasting was not observed except in a few cases of long duration where there was a moderate degree of wasting presumably due to disuse. The knee-jerks were exaggerated in all cases and pstellar clonus could be elicited in some cases. The ankle jerks were also exaggerated and ankle clonus was frequently present. The plantar response was extensor on both sides in all but three cases in which it could not be elicited.

, abdominal reflexes were variable ; in 28 cases they were normal in all quadrants, in 12 others they were brisk, in 11 cases they were sluggish, in one SOT the other of the upper quadrants and in nine others they were absent in all 'quadrants. fe The deep reflexes in the upper limbs were normal in 52 cases and unusually '"brisk in nine cases. There was no spasticity of the upper limbs in any case. t There was a striking absence of sensory changes. Touch, temperature, pressure, pain, position, muscle, joint and vibration sensations were all fully retained. Patients did not complain of girdle pain and there was no zone of hyperaesthesia. Sphincteric disturbances were completely absent in 57 cases. Four patients, however, suffered from precipitancy of micturition. The cranial nerves were all normal. There were no mental changes. No local disease of the spine could be detected on clinical examination and X-ray of the spine carried out in 19 cases did not reveal any abnormality. The cerebrospinalfluidwas clear and under normal pressure. There was no evidence of obstruction to the flow of cerebrospinal fluid. Quickenstedt's sign was positive in all cases. Myelography carried out in three cases also revealed no obstruction. The protein, chloride and sugar content of the fluid were within normal range in all cases and cells were either few or absent. Lange's test was negative and so were the Wassermann and Kahn reactions. The blood also gave a negative Wassermann reaction. Haematological examination.In all the 11 cases investigated by the writer there was hypochromic microcytic anaemia of moderate severity. In 12 other cases in which the results of haematological examination were recorded, there was also evidence of moderate anaemia of the microcytic hypochromic type. Sternal marrow in all nine cases in which it was examined showed normoblastic reaction. Fractional gastric analysis conducted in 13 cases showed normal gastric acidity in three cases, hypochlorhydria in five cases and achlorhydria in five cases. In two of the latter, the achlorhydria was histamine-fast. (Sternal marrow was examined in all cases showing achlorhydria and showed normoblastic reaction.) Clinical signs of dietary deficiency were not recorded in the cases collected from the hospital records. In the cases examined by the writer the state of nutrition was not worse than that obtaining among other patients in the general wards of the hospital. None of these cases showed angular stomatitis, glossitis or ocular signs of B complex deficiency. Phrynoderma was not seen in any case. Night blindness was present in one case. All the cases, however, showed conjunctival discoloration and patchy thickening of subconjunctival tissue. Mosaic dermatosis affecting the anterolateral aspects of the legs was present in four cases. Two patients who were past 40 years exhibited cataract.

THE LATHYRISM

SYNDROME

337
(ABLE H.

Reflexes.

Tone. 'A
Upper limbs. Low, litnbi .

roipinal fluid. | Lange's Chemistry. test. Normal ' Sternal marrow. Normal !

Blood. Peripheral W.R. & picture. Kahn. H.C. spine. analysis.

<-J. A.J # *

B.J.

S.J. A.R. P.R. 1 4 *3 ! ! 4 *2

pT

Ext.

Normal ..

Normal Hypo.

Ind. Ext. ! '" i

Ml

III

*
#

4 4

M M 1

II 1 M

! i 1

"

-'' !

Achl. (not histaminefast) Normal Hypo. Normal Achl. (histamine-fast) Hypo.

xnee jerk. A.J. ankle jerk. B.J. biceps jerk. S J . supinator jerk. A.R. abdomen P.R. plantar reflex. present. exaggerated. negative. M. male. F. female. H.C. hypochromic anaemia. Hypo, hypochlorhydria. Achl. achlorhydria. P.M. precipitancy of i DISCUSSION.

Ind. indifferent. *4 present in all four quadrants. *3 present in three quadrants. 2 present in two quadrants. Ab. abrupt. Su. subacute. Gr. gradual.

The condition described here is thus a pure upper motor neurone lesion confined to the lower extremities. General nervous diseases like subacute combined degeneration, amyotrophic lateral sclerosis, disseminated sclerosis, syringomyelia, and neurosyphilis on the one hand, and local lesions affecting the spinal cord like tumours, tuberculous disease of the spine, etc., on the other, could be easily excluded in these cases. Indeed, the condition does not fit in into any of the clinical entities known at present except that it may be classed jndcr primary lateral sclerosis which, however, is no more than a convenient label. The disease, however, has several features in common with lathyrism and is, in fa"t, clinically indistinguishable from it. Thus, lathyrism is also characterised by spastic paraplegia affecting the lower limbs, normal cerebro-spinal fluid, absence of sensory changes and absence of involvement of other parts of the body. The age and sex incidence observed in these cases is also in
conformity with that reported in lathyrism. Thus BUCHANAN (1902), YOUNG (1926), SHAH (1939), and SHOURIE (1945) all observed that the majority of

cases of lathyrism were in the younger age groups and that males were predominantly affected by the disease. The striking discrepancy, however, was that none of the cases reported here had ever subsisted on lathyrus. The relationship of this syndrome to lathyrism and the role of malnutrition in its development require elucidation. It is noteworthy that all the subjects suffering from the disease belonged to the poorest section of the community

subsisting on grossly inadequate diets. SHOURIE (1946) also found that the victims of lathyrism were the poorest class of people. However, clinical signs of deficiency diseases were lacking and the same was true of lathyrism (SHOURIE, 1946). MELLANBY (1931), from his work on puppies, believed that vitamin A deficiency was mainly responsible for the development of lathyrism and was inclined to class lathyrism as a food deficiency disease. YOUNG (1927) found night blindness common in a village affected by lathyrism, and also observed that the disease did not occur in neighbouring villages where the diet contained as much lathyrus but more vitamin A, fish and meat. SHAH (1939) reported great improvement in his cases of lathyrism when vitamins A and D were given. On the other hand, BASU and others (1936) pointed out that Lathyrus sativus, which formed the staple food in famine conditions, was a poor source of tryptophane. The occurrence of spastic paraplegia in experimental animals fed on different deficient diets has been reported by several workers. MELLANBY (1931) demonstrated lesions in the spinal cord in puppies on a diet deficient in vitamin A. Paralysis of the hind limbs and posterior third of the body has been reported in mice fed on diets deficient in pantothenic acid (MORRIS and LIPPINCOTT, 1941). Spastic paralysis of the hind limbs was observed in rats (BOAS, 1927) and dogs on a biotin deficient diet. Degeneration of the lumbar cord has been reported in rats on a vitamin E deficient diet (RINGSTED, 1935), while WINTROBE and others (1940) demonstrated degeneration of the spinal cord in pigs on a diet deficient in some unknown factor.

338
THE LATIIVRISM SYNDROME

1
The salient fact that emerges from these diverse claims is that the integrity 1 of the spinal cord is dependent on the supply of an adequate diet and that probably more than one essential nutrient is concerned in the process. A convincing proof of this in the human subject is the reported occurrence of spastic paraplegia among malnourished prisoners of war (DENNY-BROWN, 1945 ; SPILLANE, 1947). SPILLANE has remarked on the similarity of these cases of spastic paraplegia to cases of lathyrism. There was obviously no question of lathyrus playing any part in these cases. The one striking feature common to cases reported here, to cases of spastic paraplegia among prisoners of war, and to cases of lathyrism is the background of malnutrition. MELLANBY (1934) postulated " that Lathyrism was lack of some protective food which could normally prevent the detrimental effects of / toxins which are always present in the germ of grain." The toxin may act by binding, an essential nutrient and thus rendering it unavailable to the subject. The occurrence of the lathyrism syndrome among non-lathyrus eating populations suggests that the toxic agent, may not always be the same and sometimes may be other than that contained in the seeds of Vicia sativa or various kinds of lathyrus. Lathyrism may thus be looked upon as a syndrome caused by nutritional deficiency which apparently weakens the resistance of the lower segments of the spinal cord to various toxic agents.
SUMMARY OF CONTENTS.

339
ANSACTIONS OF THE ROYAL SOCIETY OF .TROPICAL MEDICINE AND HYGIENE.

Vol. 44. No. 3. December, 1950.

NUTRITIONAL ILL HEALTH AMONG MALAY CHILDREN IN THE IPOH DISTRICT OF PERAK, MALAYA* A CASE DESCRIBED OF SEVERE MALNUTRITION WITH ENLARGED LIVER.
BY

A syndrome of spastic paraplegia among poor South Indians subsisting on grossly inadequate diets, not containing lathyrus, has been described. The similarities between this syndrome and lathyrism have been pointed out. The role of malnutrition in the causation of the syndrome has been discussed. REFERENCES.
ACTON, H. W. (1922). Indian vied. Gaz., 57, 241. BASU, K. P., et al. (1936-37). Indian J. med. Res., 24, 1001 and 1027. BOAS, M. A. (1927). Biochem. J., 21, 712. BUCHANAN, \V. J. (1904). Nagpur Civil Administration Report. DENNY-BROWN, H.M.S.O. D. (1945). Report on Neurological Results of Internment. London : MCCAHRISON, R. (1926). Indian 7. med. Res., 14, 379. . (1928). bid., 15, 797. MACKENZIE, L. H. L. (1927). Indian med Gaz., 62, 201. MELLANBY, E. (1931). Brain, 54, 247. . (1934). Nutrition and Disease. London: Oliver and Boyd. MiNCHIN, R. L. H. (1940). Brit. med. J., 1, 253.
MORRIS, H. P. & LIPPINCOTT, S. W. (1941). J. nat. Cancer Inst., 2, 29 and 39.

FLORENCE ADAM THOMSON, Medical Officer, Malaya.

RINCSTED, A. (1935). Biochem. J., 27,788. SHAH, S. R. A. (1939). Indian med. Gas., 74, 385. SHOURIE, L. (1945). Indian J. med. Res., 33, 247. SPILLANE, J. D. (1947). Nutritional Disorders of the Nervous System. Livingstone Publication, p. 187. STOTT, H. (1930). Indian J. med. Res., 18, 51. WINTROBE, M. M., et al. (1940). Johns Hopk. Hosp. Bull., 67, 377. VOUNG, M. T. C. (1927). Indian J. med. Res., 15, 453.

Edinburgh :

Records of a small series (1,703) of Malay children in the Ipoh district of Perak have been collected during the past 9 months. The area covered is about 500 square miles and is mainly rural in character although the nearness of Ipoh, a large town, the capital of the State of Perak (population, 1947 : Chinese, 115,089; Malay, 15,078; Malaysians, 1,354; Indian, 21,867; others, 2,674) has influenced these rural Malays to a great extent. Apart from rice, they rarely grow their own food and much prefer an occasional trip on the bus to town where they buy mainly Chinese grown vegetables, salt fish, sugar and tins of sweetened condensed milk. Meat is rarely eaten, and as nearly all Malays are Mohammedan, pork, the most easily available meat, both domestic and wild, never. Malays are not milk drinkers, and although buffaloes and goats are sometimes kept, the milk in the case of the buffalo is very rarely used, and goats' milk not at all. Careful questioning of mothers both at the town clinic and at the clinics held in the rural areas has revealed these dietetic habits, and as BURGESS (1948) has already noted, diets are grossly deficient in protein, minerals, vitamins and, Published by kind permission of Dr. R. B. MACGREGOR, C.M.G., Director of Medical Services, Federation of Malaya.

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