Shoshin Beriberi Complicating Severe Pulmonary Hypertension: A Case Report

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J Cardiol 2007 Jun; 49 6 : 361 365 1 Shoshin Beriberi Complicating Severe Pulmonary Hypertension: A Case Report Yuko Masaki Masaaki Masamichi Takakazu Jun-ichi Tetsuya Toshinari Osamu Fusako Shinsuke Seiki GOTO, MD AWATA, MD UEMATSU, MD, FJCC YANO, MD MOROZUMI, MD KOTANI, MD WATANABE, MD ONISHI, MD IIDA, MD SERA, MD NANTO, MD, FJCC NAGATA, MD, FJCC Abstract A 44-year-old male was admitted to our hospital for dyspnea, associated with severe pulmonary hypertension. The patient fell into a shock state on the next day. Hemodynamic measurements revealed high output heart failure with low peripheral vascular resistance. We suspected shoshin beriberi, a fulminant form of cardiac beriberi, by ruling out other common causes of pulmonary hypertension. The rapid recovery after intravenous thiamine administration and the patient s history of improper diet strongly supported the diagnosis. The present case of shoshin beriberi complicating severe pulmonary hypertension shows that history taking is important in elucidating the etiology and selecting the correct treatment. J Cardiol 2007 Jun ; 49 6 : 361 365 Key Words Complications shoshin beriberihypertension, pulmonary Heart failure high-outputdrug therapy thiamine 1 1 44 : : 660 8511 3 1 69 Cardiovascular Division, Kansai Rosai Hospital, Hyogo Address for correspondence: GOTO Y, MD, Cardiovascular Division, Kansai Rosai Hospital, Inabaso 3 1 69, Amagasaki, Hyogo 660 8511; E-mail : yugotou-ham@umin.ac.jp Manuscript received December 15, 2006; revised March 1, 2007 ; accepted March 8, 2007 361

362 :20 30 : 1 1 : 2006 3 3 25 4 10 : 175cm90 kg113/60mmhg 96/min 22 /min36.3 2 Erb Levine : ; WBC 7 100/ l RBC 465 10 4 / l Hb 15.6 g/dl Ht 46.1% Plt 25.9 10 4 / l ; T-Bil 2.5 mg/dl AST 27 IU/l ALT 20 IU/l ALP 188 IU/l-GTP 21 U/l LDH 319 IU/l TP 7.6 mg/dl Alb 4.3mg/dl TC 170 mg/dl TG 67mg/dl CPK 21 IU/l Na 136 meql/l Cl 96mEq/l K 3.7mEq/l BUN 35.7mg/dl Cr 1.85mg/dl CRP 0.4 mg/dl BS 98mg/dl HbA 1c 5.2% Lactate 5.0 meq/l TSH 1.70 U/ml FT3 2.07 pg/ml FT4 1.22 ng/dl O 2 4 l PaO 2 74.6 mmhg PCO 2 27.5mmHg ph 7.46 B.E. 3.2 mmol/l HCO 3 19.0mmol/l Sat 94.9%; PT 17.1 sec APTT 240.0 sec TAT 6.9 g/l D 4.96 g/ml FDP 7.8 g/ml BUN/Cr : 1 4 T 5 6 TFig. 1 Fig. 1 Electrocardiogram on admission Electrocardiogram showed low voltage of limb leads and non-specific ST-T abnormalities. X : 61% computed tomography : CT : :M 76% Fig. 2 Teichholz 3.4 l/min/m 2 70mmHg : FDP D TAT CT X CT J Cardiol 2007 Jun; 49 6 : 361 365

363 Fig. 2 Echocardiograms before and after thiamine administration Echocardiograms from the parasternal short-axis approach on admission left and 14 days after admission right. Enlarged right ventricle and interventricular septum shift to left ventricle were observed on admission left, whereas the right ventricle showed tendency to recovery and interventricular septum shift was normalized after thiamine administration right. RV right ventricle; LV left ventricle. 1 2 5 l/min PaO 2 60mmHg 70 mmhg Swan-Ganz 6 g/kg/min 84/55 mmhg 70/38mmHg16 mmhg 6.5 l/min/m 2 492 dyn sec mm 2 /cm 5 271 dyn sec mm 2 /cm 5 F 200 mg 3 Fig. 3 Swan-Ganz 3.7 l/min/m 2 1,643 dyn sec mm 2 /cm 5 672 dyn sec mm 2 /cm 5 3 14 34mmHg Fig. 2 1 4 T 5 6 T 28. 17 ng/ml 20 50 ng/ml 17 47 ng/ml 28 187ng/ml J Cardiol 2007 Jun; 49 6 : 361 365

364 Fig. 3 Time course of blood pressure, pulmonary arterial pressure and serum thiamine levels before and after intravenous administration of fulsultiamine Open arrow indicates intravenous fulsultiamine administration. Swan- Ganz 2 4 3 Wakabayashi 4 Wernicke 2 5 6 Okura 5 J Cardiol 2007 Jun; 49 6 : 361 365

365 1 7 44 1 J Cardiol 2007 Jun; 49 6 : 361 365 1 Phua KH, Goh LG, Koh K, Ong CN, Tan TC, Wong ML, Lee HP: Thiamine deficiency and sudden deaths : Lessons from the past. Lancet 1990 ; 335: 1471 1472 2 Akbarian M, Yankopoulos NA, Abelmann WH : Hemodynamic studies in beriberi heart disease. Am J Med 1966 : 41 : 197 212 3 Seligmann H, Halkin H, Raushfleisch S, Kaufmann N, Motro M, Vered Z, Ezra D : Thiamine deficiency in patients with congestive heart failure receiving long-term furosemide therapy : A pilot study. Am J Med 1991 ; 91 : 151 155 4 Wakabayashi A, Matsumura T, Hirose K, Kawai C : Beriberi heart disease. Kokyu To Junkan 1977 ; 25 : 781 790 in Japanese 5 Okura H, Takatsu Y: High-output heart failure as a cause of pulmonary hypertension. Intern Med 1994 ; 33 : 363 365 6 Khowsathit P, Pongpanich B, Layangool T: Cardiac beriberi : Report of a case with an echocardiographic study. Jpn Heart J 1990; 31 :265 269 7 Kawai C, Wakabayashi A, Matsumura Tadashi, Yui Y : Reappearance of beriberi heart disease in Japan : A study of 23 cases. Am J Med 1980 ; 69: 383 386 J Cardiol 2007 Jun; 49 6 : 361 365