Detection of anticonductive tissue autoantibodies in a patient with chronic intestinal pseudo-obstruction and sick sinus syndrome

Giacomo Caio, Umberto Volta, Enrico Cerrato, Paolo Clavenzani, Nicolò Montali, Rosanna Cogliandro, Vincenzo Stanghellini, Pier Giorgio Golzio, Fiorenzo Gaita, Gianrico Farrugia, Roberto De Giorgio

Research output: Contribution to journalArticle

4 Scopus citations

Abstract

A 26-year-old patient was diagnosed as having chronic intestinal pseudo-obstruction with manometric and histopathologic features suggestive of an intestinal myopathy. Histology was characterized by smooth muscle degeneration without inflammatory or immune cells. The severe gut dysfunction required full parenteral nutritional support. After a few months, the patient developed symptomatic tachy-brady arrhythmia episodes with syncopes. A thorough diagnostic work-up led to a diagnosis of sick sinus syndrome, which was managed by pacemaker implantation and administration of β-blockers. This led to a partial improvement in tachy-brady arrhythmia episodes. Nonetheless, the patient continued to experience sustained supraventricular tachyarrhythmia runs, poorly responsive to increasing β-blocker doses. To investigate the origin of the cardiologic impairment, the patient was tested for anticonductive tissue autoantibodies, which were positive, thus supporting a possible autoimmune origin of the dysrhythmia. Other autoantibodies were negative. On the basis of these findings, the patient was treated with high-dose steroids, which were then tapered. The patient responded to the steroid treatment and did not experience further episodes of syncope and tachyarrhythmias. The severe gut dysfunction remained unchanged. This case highlights an association between severe gut dysfunction and cardiac conductive tissue abnormalities, with autoantibodies to conductive tissue possibly causing the dysrhythmia. The severe gut and heart (likely autoimmune-mediated) dysfunction presented in this case provides a basis to further assess a link between intestinal and cardiac abnormal rhythmicity.

Original languageEnglish (US)
Pages (from-to)1358-1363
Number of pages6
JournalEuropean Journal of Gastroenterology and Hepatology
Volume25
Issue number11
DOIs
StatePublished - Nov 2013

Keywords

  • anticonductive tissue autoantibodies
  • chronic intestinal pseudo-obstruction
  • intestinal myopathy
  • sick sinus syndrome
  • α-smooth muscle actin

ASJC Scopus subject areas

  • Hepatology
  • Gastroenterology

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    Caio, G., Volta, U., Cerrato, E., Clavenzani, P., Montali, N., Cogliandro, R., Stanghellini, V., Golzio, P. G., Gaita, F., Farrugia, G., & De Giorgio, R. (2013). Detection of anticonductive tissue autoantibodies in a patient with chronic intestinal pseudo-obstruction and sick sinus syndrome. European Journal of Gastroenterology and Hepatology, 25(11), 1358-1363. https://doi.org/10.1097/MEG.0b013e3283632dbc