Skip to main content

Advertisement

SpringerLink
Log in

Neurolathyrism: two Ethiopian case reports and review of the literature

  • Review
  • Published:
Journal of Neurology Aims and scope Submit manuscript

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.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Similar content being viewed by others

References

  1. Manmohan S, Gurbachan S (1952) Lathyrism: a neurological and economic problem in India. J Nerv Ment Dis 116:923–932

    Article  Google Scholar 

  2. Singhal GD, Singh LM, Singh KP (1972) Diagnostic considerations in ancient Indian surgery. Singhal, Allahabad, pp 77–78

    Google Scholar 

  3. Littré É (1846) Oeuvres complètes d'Hippocrate. Baillière, Paris, Vol 5; Book 2; Sect 4. p 126

  4. Hendley AG (1903) Lathyrism. Br Med J 2:707–709

    Article  Google Scholar 

  5. Sleeman WH (1844) Rambles and recollections of an Indian official. Hatchard and Sons, London

    Google Scholar 

  6. Haimanot RT, Kidane Y, Wuhib E et al (1990) Lathyrism in rural northwestern Ethiopia: a highly prevalent neurotoxic disorder. Int J Epidemiol 19:664–672

    Article  PubMed  CAS  Google Scholar 

  7. Cheeke PR (1989) Toxicants of plant origin: proteins and amino acids. CRC-Press, Boca Raton, p 170

    Google Scholar 

  8. Goya DF (1863) Los desastres de la Guerra. Plate 51, Madrid

  9. Coni N (2002) A historical survey of medicine in Spain during the Spanish Civil War. J R Soc Med 95(3):147–150

    Article  PubMed  Google Scholar 

  10. Kessler A (1947) Lathyrsmus. Montasschr Psychiatr Neurol 113:345–376

    Article  CAS  Google Scholar 

  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):55–58

    Google Scholar 

  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–53

  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):1113–1126

    Google Scholar 

  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 3:432

    Article  PubMed  CAS  Google Scholar 

  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) 58:112–119

    CAS  Google Scholar 

  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 32:461–466

    Article  Google Scholar 

  17. Ross SM, Roy DN, Spencer PS (1989) β-N-Oxalylamino-l-alanine action of glutamate receptors. J Neurochem 53:710–715

    Article  PubMed  CAS  Google Scholar 

  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):2073–2079

    CAS  Google Scholar 

  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):55–62

    Article  PubMed  CAS  Google Scholar 

  20. Tshala-Katumbay DD, Spencer PS (2007) Toxic disorders of the upper motor neuron system, Chapter 18. Handb Clin Neuro 82:353–372

    Article  Google Scholar 

  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):10287–10296

    PubMed  CAS  Google Scholar 

  22. Ravindranath V (2002) NL: mitochondrial dysfunction in excitotoxicity mediated by l-β-oxalyl aminoalanine. Neurochem Int 40(6):505–509

    Article  PubMed  CAS  Google Scholar 

  23. Ganpathy KT, Dwivedi MP (1961) Studies on clinical epidemiology of lathyrism, Lathyrism Enquiry Reid Unit. Indian Council of Medical Research, Ghandi Memorial Hospital, Rewa

    Google Scholar 

  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):151–156

    Google Scholar 

  25. Drory VE, Rabey MJ, Cohn DF (1992) Electrophysiologic features in patients with chronic neurolathyrism. Acta Neurol Scand 85:401–403

    Article  PubMed  CAS  Google Scholar 

  26. Zaninovic V (2001) Similarities between Tropical Spastic Paraparesis (TSP) and Neurolathyrism. Lathyrus Lathyrism Newsl 2(2):11–14

  27. Acton HW (1922) An investigation into the causation of lathynsm in man. Indian Med Gaz 57:241–247

    Google Scholar 

  28. Dwivedi MP, Prasad VG (1964) An epidemiological study of lathyrism in the district of Rewa, Madhya Pradesh. Indian J Med Res 52:81–114

    PubMed  CAS  Google Scholar 

  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):700–703

    Article  PubMed  Google Scholar 

  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):169–178

    Article  PubMed  Google Scholar 

  31. Gardner AF, Sakiewicz N (1963) A review of neurolathyrism including the Russian and Polish literature. Exp Med Surg 21:164–191

    PubMed  CAS  Google Scholar 

  32. Gopalan C (1950) The lathyrism syndrome. Trans R Soc Trop Med Hyg 44(3):333–338

    Article  PubMed  CAS  Google Scholar 

  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):79–82

    PubMed  CAS  Google Scholar 

  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 2:9

    Google Scholar 

  35. Cantani A (1873) Latirismo (Lathyrismus) illustrato da tre casi clinici. Morgagni 15:745

    Google Scholar 

  36. Proust A (1883) Du lathyrisme medullaire spasmodique. Bull Acad Med 12:839

    Google Scholar 

  37. Filimonoff I (1926) Zur pathologisch-anatomischen Charak- teristik des Lathyrismus. Ztschr Ges Neurol Psychiat 105:76–92

    Article  Google Scholar 

  38. Buzzard E, Greenfield JG (1921) Pathology of the nervous system. Constable, London

    Google Scholar 

  39. Puig JS, DeVinals RR (1943) Aportacion a la patologia del latirismo. Rev Clin Esp 8:107

    Google Scholar 

  40. Sachdev S, Sachdev JC, Pun D, Devider P (1969) Morphological study in a case of lathyrism. J Indian Med Assoc 52:320

    PubMed  CAS  Google Scholar 

  41. Striefler M, Cohn DF, Hirano A, Schujman E (1977) The central nervous system in a case of neurolathyrism. Neurology 27(12):1176–1178

    Article  PubMed  CAS  Google Scholar 

  42. Hirano A, Llena JF, Streifler M, Cohn DF (1976) Anterior horn changes in a case of neurolathyrism. Acta Neuropathol (Berl) 35:277–283

    CAS  Google Scholar 

  43. Cohn DF, Streifler M (1981) Human neurolathyrism, a follow-up study. Part II: special investigations. Schweiz Arch Neurol Neurochir Psychiatr 128(1):157–163

    PubMed  CAS  Google Scholar 

  44. Leite AC, Mendonca GA, Serpa MJ, Nascimento OJ, Araujo AQ (2003) Neurological manifestations in HTLV-Iinfected blood donors. J Neurol Sci 214:49–56

    Article  PubMed  Google Scholar 

  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 74:1085–1089

    Article  PubMed  CAS  Google Scholar 

  46. Boissé L, Gill MJ, Power C (2008) HIV infection of the central nervous system: clinical features and neuropathogenesis. Neurol Clin 26(3):799–819

    Article  PubMed  Google Scholar 

  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 353:712–716

    Article  PubMed  CAS  Google Scholar 

  48. Chu YF, Sun J, Wu XZ, Liu RH (2002) Antioxidant and anti-proliferative activities of common vegetables. J Agric Food Chem 50:6910–6916

    Article  PubMed  CAS  Google Scholar 

  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 69:81–89

    Article  CAS  Google Scholar 

  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 62:233–237

    Article  CAS  Google Scholar 

  51. Santhaand M, Mehta SL (2008) Handbook of New technologies for Genetic Improvement of legumes. Kirti PB (ed) CRC Press, pp 177–185

  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):678–684

    Article  PubMed  CAS  Google Scholar 

Download references

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.

Author information

Authors and Affiliations

  1. Department of Neurology, Addis Ababa University, P. O. Box 1171, Addis Ababa, Ethiopia

    Yohannes W. Woldeamanuel & Guta Zenebe

  2. The Saul R. Korey Department of Neurology, Albert Einstein College of Medicine, 1410 Pelham Parkway South, Kennedy Center Room 303, Bronx, NY, 10461, USA

    Yohannes W. Woldeamanuel

  3. Center for Movement Disorder and Neurorestoration, University of Florida, 3450 Hull Rd, Gainesville, FL, 32607, USA

    Anhar Hassan

Authors
  1. Yohannes W. Woldeamanuel
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Anhar Hassan
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Guta Zenebe
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Yohannes W. Woldeamanuel.

Electronic supplementary material

Below is the link to the electronic supplementary material.

Supplementary material 1 (JPG 37 kb)

Supplementary material 2 (JPG 34.2 kb)

Supplementary material 3 (MPG 9410 kb)

Rights and permissions

Reprints and permissions

About this article

Cite this article

Woldeamanuel, Y.W., Hassan, A. & Zenebe, G. Neurolathyrism: two Ethiopian case reports and review of the literature. J Neurol 259, 1263–1268 (2012). https://doi.org/10.1007/s00415-011-6306-4

Download citation

  • Received:

  • Revised:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00415-011-6306-4

Keywords

Search

Navigation