Thiamine-responsive acute severe pulmonary hypertension in exclusively breastfeeding infants: a prospective observational study

Arch Dis Child. 2021 Mar;106(3):241-246. doi: 10.1136/archdischild-2019-318777. Epub 2020 Sep 3.

Abstract

Objectives: Severe pulmonary hypertension (PH) causing right heart failure can occur due to thiamine deficiency in exclusively breastfeeding infants. This study describes the clinical profile and management of thiamine-responsive acute pulmonary hypertension.

Methods: A prospective observational study of infants presenting with severe PH without any other significant heart or lung disease. History of symptoms, clinical examination, echocardiography and basic investigations were performed. Dietary patterns of mothers were recorded. Thiamine was administered and serial echocardiography was performed.

Results: A total of 250 infants had severe PH and 231 infants responded to thiamine. The mean age was 3.2±1.2 months. Fast breathing, poor feeding, vomiting and aphonia were the main symptoms. Tachypnoea, tachycardia and hepatomegaly were found on examination. Echocardiogram revealed grossly dilated right heart with severe PH. Intravenous thiamine was administered to all the babies based on clinical suspicion. Clinical improvement with complete resolution of PH was noticed within 24-48 hours. Babies were followed up to a maximum of 60 months with no recurrence of PH. All the mothers consumed polished rice and followed postpartum food restriction.

Conclusion: Thiamine deficiency is still prevalent in selected parts of India. It can cause life-threatening PH in exclusively breastfeeding infants of mothers who are on a restricted diet predominantly consisting of polished rice. It can contribute to infant mortality. Thiamine administration based on clinical suspicion leads to remarkable recovery. High degree of awareness and thiamine supplementation in relevant geographical areas is required to tackle this fatal disease.

Keywords: cardiology; general paediatrics; infant feeding; intensive care; nutrition.

Publication types

  • Observational Study
  • Video-Audio Media

MeSH terms

  • Administration, Intravenous
  • Breast Feeding / statistics & numerical data*
  • Diet Therapy / adverse effects*
  • Dietary Supplements / supply & distribution
  • Echocardiography / methods
  • Female
  • Heart Failure / diagnostic imaging
  • Heart Failure / etiology
  • Humans
  • Hypertension, Pulmonary / diagnostic imaging
  • Hypertension, Pulmonary / drug therapy*
  • Hypertension, Pulmonary / etiology
  • Hypertension, Pulmonary / mortality
  • India / epidemiology
  • Infant
  • Male
  • Mothers
  • Postpartum Period
  • Prevalence
  • Prospective Studies
  • Severity of Illness Index
  • Thiamine / administration & dosage
  • Thiamine / therapeutic use*
  • Thiamine Deficiency / complications
  • Thiamine Deficiency / epidemiology
  • Vitamin B Complex / administration & dosage
  • Vitamin B Complex / therapeutic use*

Substances

  • Vitamin B Complex
  • Thiamine