DOI: http://dx.doi.org/10.18203/2320-6012.ijrms20191496

Gastrointestinal Beriberi as a prodrome of non-alcoholic Wernicke’s encephalopathy: a study of an emerging nutritional deficiency disorder from Kashmir, India

Irfan Ahmad Shah, Yuman Kawoos, Asif Iqbal Shah, Stanzen Rabyang, Henna Naqash, Mohd Rafi Mir

Abstract


Background: Gastrointestinal manifestations of thiamine deficiency have not been well described in literature. Authors aimed to study the symptoms of gastrointestinal beriberi in a cohort of patients of non-alcoholic Wernicke’s encephalopathy and review the relevant literature.

Methods: In a retrospective analysis, case records of 52 patients diagnosed with non-alcoholic Wernicke’s encephalopathy were analyzed for the nature of gastrointestinal symptoms, their duration, severity and associated findings, investigations and response to treatment. The available literature on gastrointestinal symptoms in thiamine deficiency disorders and gastrointestinal beriberi was reviewed.

Results: Gastrointestinal symptoms were found in 46 of the 52 patients. The most common gastrointestinal symptom in our patients was recurrent vomiting in 42 patients. Eight patients had water brash. Ten patients had epigastric pain and 10 patients had anorexia. Based on the nature and severity of symptoms, patients were evaluated for their symptoms using endoscopy, ultrasonography, amylase and lactate levels, and routine laboratory studies and the results were normal in the majority of patients.  Gastrointestinal symptoms settled in all the patients after receiving intravenous thiamine. On reviewing the literature multiple studies were found to have reported prominent gastrointestinal symptoms in patients of Wernickes encephalopathy and other thiamine deficiency related disorders. However, the definition of gastrointestinal beriberi is not clearly stated.

Conclusions: Gastrointestinal symptoms were prominent prodromal manifestations in our cohort of Wernicke’s encephalopathy and have also been amply reported in literature. Presence of gastrointestinal symptoms in individuals predisposed to thiamine deficiency without alternative explanation should be enough to label a patient as gastrointestinal beriberi. The study highlights the importance of recognizing gastrointestinal beriberi as a distinct syndrome that may precede the development of Wernicke’s encephalopathy.


Keywords


Gastrointestinal beriberi, Thiamine deficiency, Wernicke’s encephalopathy

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References


World Health Organization. Factsheet: Thiamine deficiency and its prevention and control in major emergencies, 1999. Accessed at:https://www.who.int/nutrition/publications/emergencies/WHO_NHD_99.13/en/. Accessed on: 10 January 2019.

Cochrane WA, Collins-Williams C, Donohue WL. Superior hemorrhagic polioencephalitis (Wernicke's disease) occurring in an infant-probably due to thiamine deficiency from use of a soya bean product. Pediat. 1961;28(5):771-7.

Donnino M. Gastrointestinal beriberi: a previously unrecognized syndrome. Annals Inter Med. 2004;141(11):898.

Prakash S. Gastrointestinal beriberi: a forme fruste of Wernicke’s encephalopathy?. BMJ Case Reports. 2018.

Gallucci M, Bozzao A, Splendiani A, Masciocchi C, Passariello R. Wernicke encephalopathy: MR findings in five patients. Am J Roentgenol. 1990;155(6):1309-14.

Naidoo DP, Bramdev A, Cooper K. Wernicke's encephalopathy and alcohol-related disease. Postgrad Med J. 1991;67(793):978-81.

Vege ÅS, Sund ST, Lindboe CF. Wernicke's encephalopathy in an autopsy material obtained over a one‐year period. APMIS. 1991;99(7‐12):755-8.

McGready R, Simpson JA, Cho T, Dubowitz L, Changbumrung S, Böhm V, et al. Postpartum thiamine deficiency in a Karen displaced population. Am J Clinic Nutrit. 2001;74(6):808-13.

Caine D, Halliday GM, Kril JJ, Harper CG. Operational criteria for the classification of chronic alcoholics: identification of Wernicke's encephalopathy. J Neurol Neurosurg Psychiat. 1997;62(1):51-60.

Bataller R, Salmeron JM, Munoz JE, et al. Pyloric stenosis complicated by Wernicke-Korsakoff syndrome. Gastroenterol Hepatol. 1997;20:131-3.

Eggspuhler AW, Bauerfeind P, Dorn T, Siegel AM. Wernicke encephalopathy-a severe neurological complication in a clinically inactive Crohn's disease. European Neurol. 2003;50(3):184.

De Wardener HE, Lennox B. Cerebral beriberi (Wernicke's encephalopathy). Review of 52 cases in a Singapore prisoner-of-war hospital. Lancet. 1947;252:11-7.

Antel K, Singh N, Chisholm B, Heckmann JM. Encephalopathy after persistent vomiting: Three cases of non-alcohol-related Wernicke's encephalopathy. SAMJ: South African Med J. 2015;105(6):442-3.

Gascón-Bayarri J, Campdelacreu J, García-Carreira MC, Estela J, Martínez-Yélamos S, Palasí A, et al. Wernicke's encephalopathy in non-alcoholic patients: a series of 8 cases. Neurología. 2011;26(9):540-7.

Hill G, Grimes M, Gaskin G, Turley B. Beriberi in a malnourished woman after an extended period of nausea and vomiting. Annals Long Term Care Clinic Care Aging. 2016;24(4):40-4.

Weidauer S, Nichtweiss M, Lanfermann H, Zanella FE. Wernicke encephalopathy: MR findings and clinical presentation. European Radiol. 2003;13(5):1001-9.

Hiffler L, Rakotoambinina B, Lafferty N, Martinez Garcia D. Thiamine deficiency in tropical pediatrics: new insights into a neglected but vital metabolic challenge. Frontiers Nutrit. 2016;3:16.

Merkin-Zaborsky H, Ifergane G, Frisher S, Valdman S, Herishanu Y, Wirguin I. Thiamine-responsive acute neurological disorders in nonalcoholic patients. Europ Neurol. 2001;45(1):34-7.

Halavaara J, Brander A, Lyytinen J, Setälä K, Kallela M. Wernicke's encephalopathy: is diffusion-weighted MRI useful?. Neuroradiol. 2003;45(8):519-23.

White ML, Zhang Y, Andrew LG, Hadley WL. MR imaging with diffusion-weighted imaging in acute and chronic Wernicke encephalopathy. Am J Neuroradiol. 2005;26(9):2306-10.

Tanphaichitr V, Thiamin. In: Shils ME, Olson JA, Shike M, Ross AC, eds. Modern Nutrition in Health and Disease, 9th Ed. Baltimore MD: Williams and Wilkins.1999:381-89.

Williams R. Observations on induced thiamine deficiency in man. Arch Intern Med. 1940;66:785-99.

Ho Park S, Kim M, Na DL, Jeon BS. Magnetic resonance reflects the pathological evolution of Wernicke encephalopathy. J Neuroimaging. 2001;11(4):406-11.

Ogershok PR, Rahman A, Brick J, Nestor S. Wernicke encephalopathy in nonalcoholic patients. Am J Med Sci. 2002 ;323(2):107-11.

Chen KT, Twu SJ, Chiou ST, Pan WH, Chang HJ, Serdula MK. Outbreak of beriberi among illegal mainland Chinese immigrants at a detention center in Taiwan. Public Health Reports.2003;118(1):59.

Fattal-Valevski A, Kesler A, Sela BA, Nitzan-Kaluski D, Rotstein M, Mesterman R, et al. Outbreak of life-threatening thiamine deficiency in infants in Israel caused by a defective soy-based formula. Pediat. 2005;115(2):e233-8.

Zhong C, Jin L, Fei G. MR imaging of non-alcoholic Wernicke encephalopathy: a follow-up study. Am J Neuroradiol. 2005;26(9):2301-5.

Sun GH, Yang YS, Liu QS, Cheng LF, Huang XS. Pancreatic encephalopathy and Wernicke encephalopathy in association with acute pancreatitis: a clinical study. World J Gastroenterol 2006;12:4224-7.

Kirbas D, Sutlas N, Kuscu DY, Karagoz N, Tecer O, Altun U. The impact of prolonged hunger strike: clinical and laboratory aspects of twenty-five hunger strikers. Nervous Necrop. 2008;61(9-10):317-24.

Zuccoli G, Santa Cruz D, Bertolini M, Rovira A, Gallucci M, Carollo C, et al. MR imaging findings in 56 patients with Wernicke encephalopathy: non alcoholics may differ from alcoholics. Am J Neuroradiol. 2009;30(1):171-6.

Francini-Pesenti F, Brocadello F, Manara R, Santelli L, Laroni A, Caregaro L. Wernicke's syndrome during parenteral feeding: not an unusual complication. Nutrit. 2009;25(2):142-6.

Dabar G, Harmouche C, Habr B, Riachi M, Jaber B. Shoshin beriberi in critically-ill patients: case series. Nutrit J. 2015;14(1):51.

Qureshi UA, Sami A, Altaf U, Ahmad K, Iqbal J, Wani NA, et al. Thiamine responsive acute life-threatening metabolic acidosis in exclusively breast-fed infants. Nutrit. 2016;32(2):213-6.

Duca J, Lum C. Rare presentation of thiamine deficiency as gastrointestinal syndrome. Hawai'i J Med Public Health. 2014;73:46.

Galvin R, Bråthen G, Ivashynka A, Hillbom M, Tanasescu R, Leone MA. EFNS guidelines for diagnosis, therapy and prevention of Wernicke encephalopathy. European J Neurol. 2010;17(12):1408-18.

Blass JP, Gibson GE. Abnormality of a thiamine-requiring enzyme in patients with Wernicke-Korsakoff syndrome. New Eng J Med. 1977;297(25):1367-70.

Thomson AD, Marshall EJ. The natural history and pathophysiology of Wernicke's encephalopathy and Korsakoff's psychosis. Alcohol Alcohol. 2005;41(2):151-8.

Chaves LC, Faintuch J, Kahwage S, de Assis Alencar F. A cluster of polyneuropathy and Wernicke-Korsakoff syndrome in a bariatric unit. Obes Surg. 2002;12(3):328-4.

Madl C, Kranz A, Liebisch B, Traindl O, Lenz K, Druml W. Lactic acidosis in thiamine deficiency. Clinic Nutrit. 1993;12(2):108-11.

Amrein K, Ribitsch W, Otto R, Worm HC, Stauber RE. Severe lactic acidosis reversed by thiamine within 24 hours. Critical Care. 2011;15(6):457.

Moskowitz A, Graver A, Giberson T, Berg K, Liu X, Uber A, et al. The relationship between lactate and thiamine levels in patients with diabetic ketoacidosis. J Critic Care. 2014;29(1):182-e5.

Donnino MW, Carney E, Cocchi MN, Barbash I, Chase M, Joyce N, et al. Thiamine deficiency in critically ill patients with sepsis. J Critic Care. 2010;25(4):576-81.