Document Detail


Detection of anticonductive tissue autoantibodies in a patient with chronic intestinal pseudo-obstruction and sick sinus syndrome.
MedLine Citation:
PMID:  24081107     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
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.
Authors:
Giacomo Caio; Umberto Volta; Enrico Cerrato; Paolo Clavenzani; Nicolò Montali; Rosanna Cogliandro; Vincenzo Stanghellini; Pier Giorgio Golzio; Fiorenzo Gaita; Gianrico Farrugia; Roberto De Giorgio
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, N.I.H., Extramural; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  European journal of gastroenterology & hepatology     Volume:  25     ISSN:  1473-5687     ISO Abbreviation:  Eur J Gastroenterol Hepatol     Publication Date:  2013 Nov 
Date Detail:
Created Date:  2013-10-01     Completed Date:  2014-05-13     Revised Date:  2014-07-31    
Medline Journal Info:
Nlm Unique ID:  9000874     Medline TA:  Eur J Gastroenterol Hepatol     Country:  England    
Other Details:
Languages:  eng     Pagination:  1358-63     Citation Subset:  IM    
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MeSH Terms
Descriptor/Qualifier:
Adult
Autoantibodies / blood*
Autoimmune Diseases / immunology*
Glucocorticoids / therapeutic use
Heart Conduction System / immunology*
Humans
Intestinal Pseudo-Obstruction / complications,  immunology*,  pathology
Male
Muscle, Smooth / pathology
Sick Sinus Syndrome / complications,  drug therapy,  immunology*
Grant Support
ID/Acronym/Agency:
DK52766/DK/NIDDK NIH HHS; R01 DK052766/DK/NIDDK NIH HHS
Chemical
Reg. No./Substance:
0/Autoantibodies; 0/Glucocorticoids

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