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Congenital heart block associated with Sjögren syndrome: case report

Karwan A Moutasim1, Penelope J Shirlaw, Michael P Escudier

  • 1Cancer Sciences Division, School of Medicine, University of Southampton, Southampton SO16 6YD, UK. k.a.moutasim@soton.ac.uk

International Archives of Medicine
|July 30, 2009
PubMed

Insights

Congenital heart block, a rare pregnancy complication linked to Sjögren Syndrome, can lead to fetal death or require infant pacing. This case highlights a mother with Sjögren Syndrome who had two sons with congenital heart block.

Area of Science:

  • Obstetrics and Gynecology
  • Rheumatology
  • Pediatric Cardiology

Background:

  • Congenital heart block (CHB) is a rare but serious pregnancy complication.
  • Sjögren Syndrome (SS) is an autoimmune disorder associated with CHB.
  • CHB can result in fetal demise or necessitate lifelong pacemaker dependency.

Purpose of the Study:

  • To present a case of primary Sjögren Syndrome in a mother with a history of two offspring diagnosed with congenital heart block.
  • To review the existing literature on the association between Sjögren Syndrome and congenital heart block.
  • To discuss potential mechanisms, maternal outcomes, and preventive strategies for CHB in offspring of mothers with SS.

Main Methods:

  • Case report of a 64-year-old female patient with primary Sjögren Syndrome.
  • Review of medical literature concerning Sjögren Syndrome and congenital heart block.
  • Discussion of proposed pathomechanisms and clinical management.

Main Results:

  • The patient had two sons diagnosed with congenital heart block, both requiring pacemaker implantation.
  • The literature review identified a significant association between maternal Sjögren Syndrome and congenital heart block in offspring.
  • Suggested mechanisms involve transplacental passage of maternal autoantibodies.

Conclusions:

  • Maternal Sjögren Syndrome is a significant risk factor for congenital heart block in offspring.
  • Early detection and monitoring are crucial for managing CHB in affected infants.
  • Further research into preventive strategies and long-term maternal outcomes is warranted.
Abstract

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