Unraveling the enigma of bangungut: Is sudden unexplained nocturnal death syndrome (SUNDS) in the philippines a disease allelic to the brugada syndrome?

Phillippine Journal of Internal Medicine

Volumn 49, Issue 3, 2011, Pages 169-180

  Gaw, Albert C ^a   Lee, Byron ^a   Gervacio Domingo, Giselle ^b   Antzelevitch, Charles ^c   Divinagracia, Romeo ^d   Jocano Jr , Felipe ^e  

^a UNIVERSITY OF CALIFORNIA   (United States)

^b PHILIPPINE GENERAL HOSPITAL   (Philippines)

^c MASONIC MEDICAL RESEARCH LABORATORY   (United States)

^d UNIVERSITY OF THE EAST   (Philippines)

^e UNIVERSITY OF THE PHILIPPINES   (Philippines)

Author keywords

Bangungut; Brugada syndrome; Pathophysiology; Prevalence; Research agenda; Sunds; The philippine

Indexed keywords

AJMALINE; ALPHA ADRENERGIC RECEPTOR STIMULATING AGENT; BETA ADRENERGIC RECEPTOR BLOCKING AGENT; DIMENHYDRINATE; DISOPYRAMIDE; FLECAINIDE; GLUCOSE; GLYCEROL 3 PHOSPHATE DEHYDROGENASE; INSULIN; PILSICAINIDE; PROCAINAMIDE; PROPAFENONE; SODIUM CHANNEL BLOCKING AGENT; SODIUM CHANNEL NAV1.5; TRICYCLIC ANTIDEPRESSANT AGENT;

AGE DISTRIBUTION; ALCOHOL INTOXICATION; ALLELE; ARTICLE; AUTOPSY; AUTOSOMAL DOMINANT INHERITANCE; BRUGADA SYNDROME; COCAINE DEPENDENCE; DISEASE ASSOCIATION; ECG ABNORMALITY; GENE MUTATION; GENETIC ASSOCIATION; GENETIC SCREENING; HEART BUNDLE BRANCH BLOCK; HEART VENTRICLE ARRHYTHMIA; HEART VENTRICLE FIBRILLATION; HEART VENTRICLE TACHYCARDIA; HEMORRHAGIC PANCREATITIS; HUMAN; PHENOTYPE; SEX DIFFERENCE; ST SEGMENT ELEVATION; SUDDEN DEATH; SUDDEN UNEXPLAINED NOCTURNAL DEATH SYNDROME; SYNDROME; SYSTEMATIC REVIEW;

EID: 82355169000     PISSN: 01199641     EISSN: None     Source Type: Journal    
DOI: None     Document Type: Article

References (79)

1. Vatta M, Dumaine R, Varghese G, Richard TA, Shimizu W, Aihara N, Nademanee K, Brugada R, Brugada J, Veerakul G, Li H, Bowles NE, Brugada P, Antzelevitch C, Towbin JA. Genetic and biophysical basis of sudden unexplained nocturnal death syndrome (SUNDS), a disease allelic to Brugada syndrome. Hum Mol Genet 2002;11(3):337-345.
2. Mendoza-Guazon P. Algunas notas sobre bangungut. Revista Filipina Med Y Fam 1917;8:437-442.
3. Aponte GE. The enigma of "bangungut". Ann Intern Med 1960 Jun;52:1258-63.
4. Majoska AV. Sudden death in Filipino men; an unexplained syndrome. Hawaii Med J 1948 Jul- Aug;7(6):469-73.
5. Munger RG, Booton EA. Bangungut in Manila: sudden and unexplained death in sleep of adult Filipinos. Int J Epidemiol 1998 Aug;27(4):677-84.
6. Gervacio-Domingo G, Punzalan FE, Amarillo ML, Dans A. Sudden unexplained death during sleep occurred commonly in the general population in the Philippines: a sub-study of the National Nutrition and Health Survey. J Clin Epidemiol 2007 Jun; 60(6):567-71. Epub 2007 Feb 5.
7. US Centers for Disease Control. Sudden, unexpected, nocturnal deaths among Southeast Asian refugees. MMWR 1981:30:581-584, 589.
8. Tatsanavivat P, Chiravatkul A, Klungboonkrong V, Chaisiri S, Jarerntanyaruk L, Munger RG, Saowakontha S. Sudden and unexplained deaths in sleep (Laitai) of young men in rural northeastern Thailand. Int J Epidemiol 1992;21:904-10.
9. Gotoh K. A histopathological study on the conduction system of the so-called "pokkuri disease" (sudden unexpected cardiac death of unknown origin in Japan). Jpn Circ J 1976;40:753-68.
10. Tungsanga K, Sriboonlue P. Sudden unexplained death syndrome in north-east Thailand. Int J Epidemiol 1993;22:81-87.
11. Tatsanavivat P, Chiravatkul A, Klungboonkrong V, Chaisiri S, Jarerntanyaruk L, Munger RG, Saowakontha S. Sudden and unexplained deaths in sleep (LaiTai) of young men in rural northeastern Thailand. Int J Epidemiol 1992;21:904-910.
12. Vatta M, Dumaine R, Varghese G, Richard TA, Shimizu W, Aihara N, Nademanee K, Brugada R, Brugada J, Veerakul G, Li H, Bowles NE, Brugada P, Antzelevitch C, Towbin JA. Genetic and biophysical basis of sudden unexplained nocturnal death syndrome (SUNDS), a disease allelic to Brugada syndrome. Hum Mol Genet 2002;11(3):337-345.
13. Mendoza-Guazon P. Algunas notas sobre bangungut. Revista Filipina Med Y Fam 1917;8:437-442.
14. Sta. Cruz JZ. The pathology of "Bangungut." J Phil Med Assoc 1951;27:476-81.
15. Aponte GE. The enigma of "bangungut". Ann Intern Med 1960 Jun;52:1258-63.
16. Majoska AV. Sudden death in Filipino men: an unexplained syndrome. Hawaii Med J 1948 Jul-Aug; 7(6):469-73.
17. Sta. Cruz JZ. The pathology of "Bangungut." J Phil Med Assoc 1951:27:476-81.
18. Majoska AV. Sudden death in Filipino men; an unexplained syndrome. Hawaii Med J 1948 Jul-Aug; 7(6):469-73.
19. Aponte GE. The enigma of "bangungut". Ann Intern Med 1960 Jun;52:1258-63.
20. Park H, Weinstein SR. Sudden unexpected nocturnal death syndrome in the Mariana Islands. Am J Forensic Med and Pathol 1990; 11(3):205-207.
21. Munger RG, Booton EA. Bangungut in Manila: sudden and unexplained death in sleep of adult Filipinos. Int J Epidemiol 1998 Aug; 27(4):677-84.
22. Gervacio-Domingo G, Daez ML, Gloria VI, Lim C, Punzalan FE. Development of a questionnaire to measure the incidence of sudden unexplained death syndrome (Bangungut) in the general population. Phil JC 2005;33:33-37.
23. Gervacio-Domingo G, Punzalan FE, Amarillo ML, Dans A. Sudden unexplained death during sleep occurred commonly in the general population in the Philippines: a sub study of the National Nutrition and Health Survey. J Clin Epidemiol 2007 Jun; 60(6):567-71. Epub 2007 Feb 5.
24. Gervacio-Domingo G, Isidro J, Tirona J, Gabriel E, David G, Amarillo ML, Morales D, Dans A. The Brugada type 1 electrocardiographic pattern is common among Filipinos. J of Clin Epidemiol 2008;61:1067-1072.
25. Gervacio-Domingo G, Jocson G, Dans A. Frequency of cardiac events at four years among initially asymptomatic Filipinos with Brugada Type 1 electrocardiographic pattern. Am J Cardiol 2011 Mar;107(5):714-716.
26. Brugada P, Brugada J. Right bundle branch block, persistent ST segment elevation and sudden cardiac death: a distinct clinical and electrocardiographic syndrome: a multicenter report. J Am Coll Cardiol 1992;20:1391-6.
27. Antzelevitch C. Cellular basis and mechanism underlying normal and abnormal myocardial repolarization and arrhythmogenesis. Ann Med 2004:36 Suppl 1:5-14.
28. Wilde A, Antzelevitch C, Borggrefe M, Brugada J, Brugada R, Brugada P, Corrado D, Hauer RN, Kass RS, Nademanee K, Priori SG, Towbin JA. Proposed diagnostic criteria for the Brugada syndrome. Circulation 2002;106:2514-2519.
29. Antzelevitch C, Brugada P, Borgreffe M, Brugada J, Brugada R, Corrado D, Gussak I, LeMarec H, Nademanee K, Perez Riera AR, Shimizu W, Schulze-Bahr E, Tan H, Wilde A. Brugada syndrome: Report of the second consensus conference. Circulation 2005;111:659-670.
30. Hirata K, Takagi Y, Nakada M, Kyushima M, Asato H. Beat-to-beat variation of the ST segment in a patient with right bundle branch block, persistent ST segment elevation, and ventricular fibrillation: a case report. Angiology 1998;49:87-90.
31. Brugada P, Brugada J. Right bundle branch block, persistent ST segment elevation and sudden cardiac death: a distinct clinical and electrocardiographic syndrome: a multicenter report. J Am Coll Cardiol 1992;20:1391-6.
32. Brugada R, Brugada J, Antzelevitch C, Kirsch GF, Potenza D, Towbin JA, Brugada P. Sodium channel blockers identify risk for sudden death in patients with ST-segment elevation and right bundle branch block but structurally normal hearts. Circulation 2000;101:510-5.
33. Babaliaros VC, Hurst JW. Tricyclic antidepressants and the Brugada syndrome: an example of Brugada waves appearing after the administration of desipramine. Clin Cardiol 2002;25:395-8.
34. Brugada P, Brugada J, Brugada R. Arrhythmias induction by antiarrhythmic drugs. Pacing Clin Electrophysiol 2000;23:291-2.
35. Antzelevitch C. Cellular basis and mechanism underlying normal and abnormal myocardial repolarization and arrhythmogenesis. Ann Med 2004;36 Suppl 1:5-14.
36. Chen Q, Kirsch GE, Zhang D, Brugada R, Brugada J, Brugada P, Potenza D, Moya A, Borggrefe M, Breithardt G, Ortiz-Lopez R, Wang Z, Antzelevitch C, O'Brien RE, Schulze-Bahr E, Keating MT, Towbin JA, Wang Q. Genetic basis and molecular mechanisms for idiopathic ventricular fibrillation. Nature 1998;392:293-296.
37. Kapplinger JD, Tester DJ, Alders M, Benito B, Berthet M, Brugada J, Brugada P, Fressart V, Guerchicoff A, Harris-Kerr C, Kamakura S, Kyndt F, Koopmann TT, Miyamoto M, Pfeiffer R, Pollevick GD, Probst V, Zumhagen S, Vatta M, Towbin JA, Shimizu W, Schulze-Bahr E, Antzelevitch C, Salisbury BA, Guicheney P, Wilde AAM, Brugada R, Schott JJ, Ackerman MJ. An international compendium of mutations in the SCN5A encoded cardiac sodium channel in patients referred for Brugada syndrome genetic testing. Heart Rhythm 2010;7:33-46.
38. Antzelevitch C, Dumaine R. Electrical heterogeneity in the heart: physiological, pharmacological and clinical implications In: Page E, Fozzard HA, Solaro RJ, Eds: Handbook of Physiology: The Heart. New York: Oxford University Press; 2002.
39. Antzelevitch C. Cellular basis and mechanism underlying normal and abnormal myocardial repolarization and arrhythmogenesis. Ann Med 2004;36 Suppl 1:5-14.
40. Chen Q, Kirsch GE, Zhang D, Brugada R, Brugada J, Brugada P, Potenza D, Moya A, Borggrefe M, Breithardt G, Ortiz-Lopez R, Wang Z, Antzelevitch C, O'Brien RE, Schulze-Bahr E, Keating MT, Towbin JA, Wang Q. Genetic basis and molecular mechanisms for idiopathic ventricular fibrillation. Nature 1998;392:293-296.
41. Kapplinger JD, Tester DJ, Alders M, Benito B, Berthet M, Brugada J, Brugada P, Fressart V, Guerchicoff A, Harris-Kerr C, Kamakura S, Kyndt F, Koopmann TT, Miyamoto M, Pfeiffer R, Pollevick GD, Probst V, Zumhagen S, Vatta M, Towbin JA, Shimizu W, Schulze-Bahr E, Antzelevitch C, Salisbury BA, Guicheney P, Wilde AAM, Brugada R, Schott JJ, Ackerman MJ. An international compendium of mutations in the SCN5A encoded cardiac sodium channel in patients referred for Brugada syndrome genetic testing. Heart Rhythm 2010;7:33-46.
42. Antzelevitch C, Pollevick GD, Cordeiro JM, Casis O, Sanguinetti MC, Aizawa Y, Guerchicoff A, Pfeiffer R, Oliva A, Wollnik B, Gelber P, Bonaros EP, Jr., Burashnikov E, Wu Y, Sargent JD, Schickel S, Oberheiden R, Bhatia A, Hsu LF, Haissaguerre M, Schimpf R, Borggrefe M, Wolpert C. Loss-of-function mutations in the cardiac calcium channel underlie a new clinical entity characterized by ST-segment elevation, short QT intervals, and sudden cardiac death. Circulation 2007;115:442-449.
43. Antzelevitch C, Pollevick GD, Cordeiro JM, Casis O, Sanguinetti MC, Aizawa Y, Guerchicoff A, Pfeiffer R, Oliva A, Wollnik B, Gelber P, Bonaros EP, Jr., Burashnikov E, Wu Y, Sargent JD, Schickel S, Oberheiden R, Bhatia A, Hsu LF, Haissaguerre M, Schimpf R, Borggrefe M, Wolpert C. Loss-of-function mutations in the cardiac calcium channel underlie a new clinical entity characterized by ST-segment elevation, short QT intervals, and sudden cardiac death. Circulation 2007;115:442-449.
44. Van Norstrand DW, Valdivia CR, Tester DJ, Ueda K, London B, Makielski JC, Ackerman MJ. Molecular and functional characterization of novel glycerol-3-phosphate dehydrogenase 1 like gene (GPD1-L) mutations in sudden infant death syndrome. Circulation 2007;116:2253-2259.
45. Watanabe H, Koopmann TT, Le Scouarnec S., Yang T, Ingram CR, Schott JJ, Demolombe S, Probst V, Anselme F, Escande D, Wiesfeld AC, Pfeufer A, Kaab S, Wichmann HE, Hasdemir C, Aizawa Y, Wilde AA, Roden DM, Bezzina CR. Sodium channel b1 subunit mutations associated with Brugada syndrome and cardiac conduction disease in humans. J Clin Invest 2008;118:2260-2268.
46. Delpón E, Cordeiro JM, Núñez L, Thomsen PEB, Guerchicoff A, Pollevick GD, Wu Y, Kanters JK, Larsen CT, Burashnikov A, Christiansen M, Antzelevitch C. Functional effects of KCNE3 mutation and its role in the development of Brugada syndrome. Circ Arrhythm Electrophysiol 2008;1:209-218.
47. Hu D, Barajas-Martinez H, Burashnikov E, Springer M, Wu Y, Varro A, Pfeiffer R, Koopmann TT, Cordeiro JM, Guerchicoff A, Pollevick GD, Antzelevitch C. A mutation in the b3 subunit of the cardiac sodium channel associated with Brugada ECG phenotype. Circ Cardiovasc Genet 2009;2:270-278.
48. Yan GX, Antzelevitch C. Cellular basis for the electrocardiographic J wave. Circulation 1996;93:372-379.
49. Yan GX, Antzelevitch C. Cellular basis for the Brugada Syndrome and other mechanisms of arrhythmogenesis associated with ST segment elevation. Circulation 1999;100:1660-1666.
50. Fish JM, Antzelevitch C. Role of Sodium and Calcium Channel Block in Unmasking the Brugada Syndrome. Heart Rhythm 2004;1:210-217.
51. Fish JM, Antzelevitch C. Cellular and Ionic Basis for the Sex-related Difference in the Manifestation of the Brugada Syndrome and Progressive Conduction Disease Phenotypes. J Electrocardiol 2003;36:173-179.
52. Antzelevitch C. Brugada syndrome. PACE 2006;29:1130-1159.
53. Postema PG, van Dessel PFHM, Kors JA, Linnenbank AC, van Harpen G, Ritsema van Eck HJ, van Geloven N, De Bakker JMT, Wilde AAM, Tan HT. Local depolarization abnormalities are the dominant pathophysiologic mechanism for type 1 electrocardiogram in Brugada syndrome: a study of electrocardiograms, vectorcardiograms, and body surface potential maps during ajmaline provocation. J Am Coll Cardiol 2010;55:789-797.
54. Postema PG, van Dessel PF, Kors JA, Linnenbank AC, van Herpen G, Ritsema van Eck HJ, van Geloven N, de Bakker JM, Wilde AA, Tan HL. Local depolarization abnormalities are the dominant pathophysiologic mechanism for type 1 electrocardiogram in Brugada syndrome: a study of electrocardiograms, vectorcardiograms, and body surface potential maps during ajmaline provocation. J Am Coll Cardiol 2010; 55:789-97.
55. Wilde AAM, Postema PG, Di Diego JM, Viskin S, Morita H, Fish J M, Antzelevitch C. The pathophysiological mechanism underlying Brugada syndrome: depolarization versus repolarization. J Mol Cell Cardiol 2010; In Press: 56.
56. Wilde AA, Antzelevitch C, Borggrefe M, Brugada J, Brugada R, Brugada P, Corrado D, Hauer RN, Kass RS, Nademanee K, Priori SG, Towbin JA. Proposed diagnostic criteria for the Brugada syndrome: consensus report. Circulation 2002; 106:2514-9
57. Fish JM, Antzelevitch C. Cellular and ionic basis for the sex-related difference in the manifestation of the Brugada syndrome and progressive conduction disease phenotype. J of Electrophysiol 2003;36:173-179.
58. Di Diego JM, Cordeiro JM, Goodrow RJ, Fish JM, Zygmunt AC, Pérez GJ, Scornik FS, Antzelevitch C. Ionic and cellular basis of the predominance of the Brugada syndrome phenotype in males. Circulation 2001;106:2004-2011.
59. Babaee Bigi, Aslani A. Significance of cardiac autonomic neuropathy in risk stratification of Brugada syndrome. Eurospace 2008;10:821-824.
60. Ikeda T, Abe A, Yusu S, Nakamura K, Ishiguro H, Mera H, Yotsukura M, Yoshino H. The full stomach test as a novel diagnostic technique for identifying patients at risk of Brugada syndrome. J Cardiov Electrophysiol 2006;17:602-7.
61. Wichter T, Matheja P, Eckardt L, Kies P, Schäfers K, Schulze-Bahr E, Haverkamp W, Borggrefe M, Schober O, Breithardt G, Schäfers M. Cardiac autonomic dysfunction in Brugada syndrome. Circulation 2002;105:702-706.
62. Kies P, Wichter T, Schafers M, Paul M, Schäfers KP, Eckardt L, Stegger L, Schulze-Bahr E, Rimoldi O, Breithardt G, Schober O, Camici PG. Abnormal myocardial presynaptic norepinephrine recycling in patients with Brugada syndrome. Circulation 2004;110:3017-3022.
63. Mizumaki K, Fujiki A, Nishida K, Iwamoto J, Sakamoto T, Sakabe M, Tsuneda T, Sugao M, Inoue H. Postprandial augmentation of bradycardia-dependent ST elevation in patients with Brugada syndrome. J Cardiov Electrophysiol 2007;18(8):839-844.
64. Tester DJ, Ackerman MJ. The role of molecular autopsy in unexplained sudden cardiac death. Curr Opin Cardiol 2006 May;21(3):166-72.
65. Schott JJ, Alshinawi C, Kyndt F, Probst V, Hoorntje TM, Hulsbeek M, Wilde AA, Escande D, Mannens MM, Le Marec H. Cardiac conduction defects associate with mutations in SCN5A. Nat Gent 1999;23:20-21.
66. Tan HL, Bink-Boelkens MT, Bezzina CR, Viswanathan PC, Beaufort-Krol GC, van Tintelen PJ, van den Berg MP, Wilde AA, Balser JR. A sodium-channel mutation causes isolated cardiac conduction disease. Nature 2001;409:1043-1047.
67. Schwartz PJ, Priori SG, Dumaine R, Napolitano C, Antzelevitch C, Stramba-Badiale M, Richard TA, Berti MR, Bloise R. A molecular link between the sudden infant death syndrome and the long-QT syndrome. N Engl J Med 2000;343:262-267.
68. Melles RB, Katz B. Night terrors and sudden unexplained nocturnal death. Med Hypothesis 1988;26:149-154.
69. Binik TM. Psychological predictors of sudden death: a review and critique. Soc Sci Med 1985;20:667-680.
70. Taggart P, Critchley H, Lambiase PD. Heart-brain interactions in cardiac arrhythmia. Heart 2011,97,698-708.
71. Samuels MA. "Voodoo" death visited: the modern lessons of neurocardiology. Cleve Clin J Med 2007;74:Suppl1:S8-S16.
72. Cannon WB. "Voodoo" death. American Anthropologist 1942;44:169-181.
73. Tan ML. Revisiting Usog, Pasma, Kulam. Quezon City:The University of the Philippines Press, 2008.
74. Jocano FL. Folk Medicine in a Philippine Municipality. Metro Manila, Philippines: PUNLAD Research House, Inc., 2003
75. Wilde AA, Antzelevitch C, Borggrefe M, Brugada J, Brugada R, Brugada P, Corrado D, Hauer RN, Kass RS, Nademanee K, Priori SG, Towbin JA. Proposed diagnostic criteria for the Brugada syndrome: consensus report. Circulation 2002;106:2514-9.
76. Wilde AA, Antzelevitch C, Borggrefe M, Brugada J, Brugada R, Brugada P, Corrado D, Hauer RN, Kass RS, Nademanee K, Priori SG, Towbin JA. Proposed diagnostic criteria for the Brugada syndrome: consensus report. Circulation 2002;106:2514-9.
77. Antzelevitch C, Brugada P, Borgreffe M, Brugada J, Brugada R, Corrado D, Gussak I, LeMarec H, Nademanee K, Perez Riera AR, Shimizu W, Schulze-Bahr E, Tan H, Wilde A. Brugada syndrome: Report of the second consensus conference. Circulation 2005;111:659-670.
78. Morita H, Zipes DP, Morita ST, Wu J. Temperature modulation of ventricular arrhythmogenecity in canine tissue model of Brugada syndrome. Heart Rhythm 2007;4:188-197.
79. Sicouri S, Antzelevitch C. Sudden cardiac death secondary to antidepressant and antipsychotic drugs. Expert Opin Drug Saf 2008 March;7(2):181-194.