Advertisement

Journal of Neurology

, Volume 259, Issue 7, pp 1263–1268 | Cite as

Neurolathyrism: two Ethiopian case reports and review of the literature

  • Yohannes W. Woldeamanuel
  • Anhar Hassan
  • Guta Zenebe
Review

Abstract

Neurolathyrism is a toxic myelopathy caused by ingestion of the Lathyrus sativus grasspea. An irreversible acute to subacute spastic paraparesis or quadriparesis ensues. Despite public education, new cases of this preventable disease still occur. Two Ethiopian cases of neurolathyrism are reported to illustrate the disease, followed by a literature review. Two teenage male farmers from the same village developed irreversible spastic myelopathy following L. sativus ingestion. There was no sensory, sphincter or bulbar dysfunction. Likely causative factors identified were increased consumption of L. sativus prior to and following disease onset, heavy physical exertion and male gender, similar to those reported in the literature. Neurolathyrism is an entirely preventable neurotoxic myelopathy with permanent disability accrued. Treatment is symptomatic. Because of personal disability and subsequent socioeconomic effects, this disease warrants further public health measures to prevent occurrence. Education, avoidance of the grasspea and measures to reduce toxin burden are possible methods.

Keywords

Neurolathyrism Spastic paraparesis Spastic quadraparesis Ethiopia Lathyrus sativus Grasspea 

Notes

Acknowledgments

The authors would like to thank Guta Zenebe MD, for assistance with funding the patients’ investigations. The authors would also like to thank Farrah J. Mateen MD, for constructive comments.

Conflict of interest

The authors declare that there are no conflicts of interest.

Supplementary material

415_2011_6306_MOESM1_ESM.jpg (37 kb)
Supplementary material 1 (JPG 37 kb)
415_2011_6306_MOESM2_ESM.jpg (34 kb)
Supplementary material 2 (JPG 34.2 kb)

Supplementary material 3 (MPG 9410 kb)

References

  1. 1.
    Manmohan S, Gurbachan S (1952) Lathyrism: a neurological and economic problem in India. J Nerv Ment Dis 116:923–932CrossRefGoogle Scholar
  2. 2.
    Singhal GD, Singh LM, Singh KP (1972) Diagnostic considerations in ancient Indian surgery. Singhal, Allahabad, pp 77–78Google Scholar
  3. 3.
    Littré É (1846) Oeuvres complètes d'Hippocrate. Baillière, Paris, Vol 5; Book 2; Sect 4. p 126Google Scholar
  4. 4.
    Hendley AG (1903) Lathyrism. Br Med J 2:707–709CrossRefGoogle Scholar
  5. 5.
    Sleeman WH (1844) Rambles and recollections of an Indian official. Hatchard and Sons, LondonGoogle Scholar
  6. 6.
    Haimanot RT, Kidane Y, Wuhib E et al (1990) Lathyrism in rural northwestern Ethiopia: a highly prevalent neurotoxic disorder. Int J Epidemiol 19:664–672PubMedCrossRefGoogle Scholar
  7. 7.
    Cheeke PR (1989) Toxicants of plant origin: proteins and amino acids. CRC-Press, Boca Raton, p 170Google Scholar
  8. 8.
    Goya DF (1863) Los desastres de la Guerra. Plate 51, MadridGoogle Scholar
  9. 9.
    Coni N (2002) A historical survey of medicine in Spain during the Spanish Civil War. J R Soc Med 95(3):147–150PubMedCrossRefGoogle Scholar
  10. 10.
    Kessler A (1947) Lathyrsmus. Montasschr Psychiatr Neurol 113:345–376CrossRefGoogle Scholar
  11. 11.
    Moneim AM, Van Dorrestein B, Baum M et al (2001) Role of ICARDA in improving the nutritional quality and yield potential of grasspea (Lathyrus sativus L.), for subsistence farmers in dry areas. Lathyrus Lathyrism Newsl 2(2):55–58Google Scholar
  12. 12.
    Lambein F, Haque A (1993) Lathyrus sativus and human Lathyrism, Progress and prospects. In: 2nd International Colloquium on Lathyrism in Bangladesh, Dhaka, pp l–53Google Scholar
  13. 13.
    Haimanot RT, Abegaz B, Wuhib E, Kassina A, Kidane Y, Kebede N, Alemu T, Spencer P (1993) Pattern of Lathyrus sativus (grass pea) consumption and beta-N-oxalyl-α-β-diaminoproprionic acid (β-ODAP) content of food samples in the lathyrism endemic region of northwest Ethiopia. Nutr Res 13(10):1113–1126Google Scholar
  14. 14.
    Rao SLN, Adiga PR, Sharma PS (1964) The isolation & characterization of beta-N-alpha, beta-diaminopropionic acid: a neurotoxin from the seeds of Lathyrus sativus. Biochemistry 3:432PubMedCrossRefGoogle Scholar
  15. 15.
    Narayan SB, Bhatt DP, Bhatt PN, Mehta AR (1989) Detection of beta oxalyl diamino propionic acid in callus cultures of Lathyrus-Sativus. Curr Sci (Bangalore) 58:112–119Google Scholar
  16. 16.
    Lambien F, Haque R, Khan JK, Kebede N, Kuo YH (1994) From soil to brain: zinc deficiency increases the neurotoxicity of Lathyrus sativus and may affect the susceptibility for the motor neurone disease NL. Toxicon 32:461–466CrossRefGoogle Scholar
  17. 17.
    Ross SM, Roy DN, Spencer PS (1989) β-N-Oxalylamino-l-alanine action of glutamate receptors. J Neurochem 53:710–715PubMedCrossRefGoogle Scholar
  18. 18.
    Bridges RJ, Stevens DR, Kahle JS, Nunn PB, Kadri M, Cotman CW (1989) Structure-function studies on N-oxalyl-diamino-dicarboxylic acids and excitatory amino acid receptors: evidence that β-l-ODAP is a selective non-NMDA agonist. J Neuroscience 9(6):2073–2079Google Scholar
  19. 19.
    Willis CL, Meldrum BS, Nunn PB, Anderton BH, Leigh PN (1993) Neuronal damage induced by beta-N-oxalylamino-l-alanine, in the rat hippocampus, can be prevented by a non-NMDA antagonist, 2,3-dihydroxy-6-nitro-7-sulfamoyl-benzo(F)quinoxaline. Brain Res 627(1):55–62PubMedCrossRefGoogle Scholar
  20. 20.
    Tshala-Katumbay DD, Spencer PS (2007) Toxic disorders of the upper motor neuron system, Chapter 18. Handb Clin Neuro 82:353–372CrossRefGoogle Scholar
  21. 21.
    Sriram K, Shankar SK, Boyd MR, Ravindranath V (1998) Thiol oxidation and loss of mitochondrial complex I precede excitatory amino acid-mediated neurodegeneration. J Neurosci 18(24):10287–10296PubMedGoogle Scholar
  22. 22.
    Ravindranath V (2002) NL: mitochondrial dysfunction in excitotoxicity mediated by l-β-oxalyl aminoalanine. Neurochem Int 40(6):505–509PubMedCrossRefGoogle Scholar
  23. 23.
    Ganpathy KT, Dwivedi MP (1961) Studies on clinical epidemiology of lathyrism, Lathyrism Enquiry Reid Unit. Indian Council of Medical Research, Ghandi Memorial Hospital, RewaGoogle Scholar
  24. 24.
    Cohn DF, Streifler M (1981) Human neurolathyrism, a follow-up study of 200 patients. Part I: clinical investigation. Schweiz Arch Neurol Neurochir Psychiatr 128(1):151–156Google Scholar
  25. 25.
    Drory VE, Rabey MJ, Cohn DF (1992) Electrophysiologic features in patients with chronic neurolathyrism. Acta Neurol Scand 85:401–403PubMedCrossRefGoogle Scholar
  26. 26.
    Zaninovic V (2001) Similarities between Tropical Spastic Paraparesis (TSP) and Neurolathyrism. Lathyrus Lathyrism Newsl 2(2):11–14Google Scholar
  27. 27.
    Acton HW (1922) An investigation into the causation of lathynsm in man. Indian Med Gaz 57:241–247Google Scholar
  28. 28.
    Dwivedi MP, Prasad VG (1964) An epidemiological study of lathyrism in the district of Rewa, Madhya Pradesh. Indian J Med Res 52:81–114PubMedGoogle Scholar
  29. 29.
    Getahun H, Lambien F, Van der Stuyft P (2002) ABO blood groups, grass pea preparation, and neurolathyrism in Ethiopia. Trans R Soc Trop Med Hyg 96(6):700–703PubMedCrossRefGoogle Scholar
  30. 30.
    Getahun H, Lambein F, Vanhoorne M, Van der Stuyft P (2005) Neurolathyrism risk depends on type of grass pea preparation and on mixing with cereals and antioxidant. Trop Med Int Health 10(2):169–178PubMedCrossRefGoogle Scholar
  31. 31.
    Gardner AF, Sakiewicz N (1963) A review of neurolathyrism including the Russian and Polish literature. Exp Med Surg 21:164–191PubMedGoogle Scholar
  32. 32.
    Gopalan C (1950) The lathyrism syndrome. Trans R Soc Trop Med Hyg 44(3):333–338PubMedCrossRefGoogle Scholar
  33. 33.
    Fikre A, Lambien F, Gheysen G (2006) A life-saving food plant producing more neurotoxin under environmental stress. Commun Agric Appl Biol Sci 71(1):79–82PubMedGoogle Scholar
  34. 34.
    Ludolph AR, Gimenez-Roldan S, Hugon J, Dumas JL, Spencer PS (1990) Electrophysiological evaluation of human lathyrism––results in Bangladesh and Spain. Lathyrus Lathyrism Newsl 2:9Google Scholar
  35. 35.
    Cantani A (1873) Latirismo (Lathyrismus) illustrato da tre casi clinici. Morgagni 15:745Google Scholar
  36. 36.
    Proust A (1883) Du lathyrisme medullaire spasmodique. Bull Acad Med 12:839Google Scholar
  37. 37.
    Filimonoff I (1926) Zur pathologisch-anatomischen Charak- teristik des Lathyrismus. Ztschr Ges Neurol Psychiat 105:76–92CrossRefGoogle Scholar
  38. 38.
    Buzzard E, Greenfield JG (1921) Pathology of the nervous system. Constable, LondonGoogle Scholar
  39. 39.
    Puig JS, DeVinals RR (1943) Aportacion a la patologia del latirismo. Rev Clin Esp 8:107Google Scholar
  40. 40.
    Sachdev S, Sachdev JC, Pun D, Devider P (1969) Morphological study in a case of lathyrism. J Indian Med Assoc 52:320PubMedGoogle Scholar
  41. 41.
    Striefler M, Cohn DF, Hirano A, Schujman E (1977) The central nervous system in a case of neurolathyrism. Neurology 27(12):1176–1178PubMedCrossRefGoogle Scholar
  42. 42.
    Hirano A, Llena JF, Streifler M, Cohn DF (1976) Anterior horn changes in a case of neurolathyrism. Acta Neuropathol (Berl) 35:277–283Google Scholar
  43. 43.
    Cohn DF, Streifler M (1981) Human neurolathyrism, a follow-up study. Part II: special investigations. Schweiz Arch Neurol Neurochir Psychiatr 128(1):157–163PubMedGoogle Scholar
  44. 44.
    Leite AC, Mendonca GA, Serpa MJ, Nascimento OJ, Araujo AQ (2003) Neurological manifestations in HTLV-Iinfected blood donors. J Neurol Sci 214:49–56PubMedCrossRefGoogle Scholar
  45. 45.
    Silva MT, Mattos P, Alfano A, Araujo AQ (2003) Neuropsychological assessment in HTLV-1 infection: a comparative study among TSP/HAM, asymptomatic carriers, and healthy controls. J Neurol Neurosurg Psychiatry 74:1085–1089PubMedCrossRefGoogle Scholar
  46. 46.
    Boissé L, Gill MJ, Power C (2008) HIV infection of the central nervous system: clinical features and neuropathogenesis. Neurol Clin 26(3):799–819PubMedCrossRefGoogle Scholar
  47. 47.
    Adish AA, Esrey SA, Gyorkos TW, Jean-Baptiste J, Rojhani A (1999) Effect of consumption of food cooked in iron pots on iron status and growth of young children: a randomised trial. Lancet 353:712–716PubMedCrossRefGoogle Scholar
  48. 48.
    Chu YF, Sun J, Wu XZ, Liu RH (2002) Antioxidant and anti-proliferative activities of common vegetables. J Agric Food Chem 50:6910–6916PubMedCrossRefGoogle Scholar
  49. 49.
    Kuo YH, Bau HM, Quemener B, Khan JK, Lambein F (1995) Solid state fermentation of Lathyrus sativus seeds using Aspergillus oryzae and Rhizopus oligosprus sp T-3 to eliminate the neurotoxin β-ODAP without loss of nutritional value. J Sci Food Agric 69:81–89CrossRefGoogle Scholar
  50. 50.
    Akalu G, Johansson G, Nair BM (1998) Effect of processing on the content of β-N-oxalyl-α, β-diaminopropionic acid (β-ODAP) in grass pea (Lathyrus sativus) seeds and flour as determined by flow injection analysis. Food Chem 62:233–237CrossRefGoogle Scholar
  51. 51.
    Santhaand M, Mehta SL (2008) Handbook of New technologies for Genetic Improvement of legumes. Kirti PB (ed) CRC Press, pp 177–185Google Scholar
  52. 52.
    Fikre A, Van Moorhem M, Ahmed S, Lambein F, Gheysen G (2011) Studies on neurolathyrism in Ethiopia: dietary habits, perception of risks and prevention. Food Chem Toxicol 49(3):678–684PubMedCrossRefGoogle Scholar

Copyright information

© Springer-Verlag 2011

Authors and Affiliations

  • Yohannes W. Woldeamanuel
    • 1
    • 2
  • Anhar Hassan
    • 3
  • Guta Zenebe
    • 1
  1. 1.Department of NeurologyAddis Ababa UniversityAddis AbabaEthiopia
  2. 2.The Saul R. Korey Department of NeurologyAlbert Einstein College of MedicineBronxUSA
  3. 3.Center for Movement Disorder and NeurorestorationUniversity of FloridaGainesvilleUSA

Personalised recommendations