Shoshin Beriberi and Severe Accidental Hypothermia as Causes of Heart Failure in a 6-Year-Old Child: A Case Report and Brief Review of Literature

Abstract

Severe accidental hypothermia has been demonstrated to affect ventricular systolic and diastolic functions, and rewarming might be responsible of cardiovascular collapse. Until now, there have been only a few reports on severe accidental hypothermia, none of which involved children. Herein, we describe here a rare case of heart failure in a 6-year-old boy admitted to the emergency unit owing to severe hypothermia and malnutrition. After he was warmed up (core temperature of 27.2°C at admission), he developed cardiac arrest, requiring vasoactive amines administration, and veno-arterial extracorporeal membrane oxygenation. Malnutrition and refeeding syndrome might have caused the thiamine deficiency, commonly known as beriberi, which contributed to heart failure as well. He showed remarkable improvement in heart failure symptoms after thiamine supplementation. High-dose supplementation per os (500 mg/day) after reconstitution of an adequate electrolyte balance enabled the patient to recover completely within 2 weeks, even if a mild diastolic cardiac dysfunction persisted longer. In conclusion, we describe an original pediatric case of heart failure due to overlap of severe accidental hypothermia with rewarming, malnutrition, and refeeding syndrome with thiamine deficiency, which are rare independent causes of cardiac dysfunction. The possibility of beriberi as a cause of heart failure and adequate thiamine supplementation should be considered in all high-risk patients, especially those with malnutrition. Refeeding syndrome requires careful management, including gradual electrolyte imbalance correction and administration of a thiamine loading dose to prevent or correct refeeding-induced thiamine deficiency.

Lessons of the month 1: Shoshin beriberi: A case report of fulminant cardiovascular collapse, intractable hyperlactatemia and deteriorating consciousness

“…2 However, there have been multiple reports of isolated cases in recent years; the main contributing factors of which were alcohol excess, use of total parenteral nutrition without vitamin supplementation or chronic malnutrition. 1,2,[8][9][10] Likewise to the case presented here, a profound lactic acidosis was noted upon presentation in the other cases mentioned, with the onset of rapid onset congestive cardiac failure occurring prior to or during hospitalisation. A rising lactate and high vasopressor requirements were other common features.…”

Pediatric thiamine deficiency disorders in high‐income countries between 2000 and 2020: a clinical reappraisal

“…The outcome can be exacerbated by loop diuretics without prompt thiamine administration. 66,113,114,125 One of the major findings was the occurrence of acute respiratory distress in tachycardic infants, expressed as a pulmonary arterial hypertension with hyperlactatemia, which is remarkably reversed by thiamine treatment, as noted earlier. 72,74,111,125,127,131 The popularization of pediatric bedside cardiac Doppler ultrasound in infants and the readily available assay of lactate point-of-care testing have allowed such a refined diagnostic and therapeutic achievement, which is becoming routine practice in HICs.…”

“…Diverse TD expressions are described, with clinical, biological (associated hyperlactatemia), and radiologically specific or atypical signs of TD. The cases cover a broad panel of clinical manifestations: shock, pulmonary arterial hypertension, Shoshin beriberi, type B lactic acidosis (reversible by thiamine administration), altered mental status that can progress to coma, limb weakness, ocular abnormalities, and signs of truncated or the entire triad of WE 23,108–127 C10 : PTD related to a few documented cases of autistic spectrum disorders associated with TD and a case of botulism associated with thiaminase production (2%) 128–132 …”

“…It is described as hyperlactatemia associated with a high‐output cardiac failure, predominantly in the right or both cardiac chambers, and in its edematous form with a pitting edema in the lower limbs. The outcome can be exacerbated by loop diuretics without prompt thiamine administration 66,113,114,125 …”

“…The cases cover a broad panel of clinical manifestations: shock, pulmonary arterial hypertension, Shoshin beriberi, type B lactic acidosis (reversible by thiamine administration), altered mental status that can progress to coma, limb weakness, ocular abnormalities, and signs of truncated or the entire triad of WE. 23,[108][109][110][111][112][113][114][115][116][117][118][119][120][121][122][123][124][125][126][127] C10: PTD related to a few documented cases of autistic spectrum disorders associated with TD and a case of botulism associated with thiaminase production (2%). [128][129][130][131][132] In summary, as opposed to LMICs, many cases of PTD in HICs occur in childhood and adolescence.…”