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Familial Association of Granulocyte-Macrophage Colony-Stimulating Factor Autoantibodies in Inflammatory Bowel Disease.


ABSTRACT: OBJECTIVES:Elevated granulocyte-macrophage colony-stimulating factor auto-antibodies (GM-CSF Ab) are associated with increased intestinal permeability and stricturing behavior in Crohn disease (CD). We tested for familial association of serum GM-CSF Ab level in CD and ulcerative colitis (UC) families. METHODS:Serum GM-CSF Ab concentration was determined in 230 pediatric CD probands and 404 of their unaffected parents and siblings, and 45 UC probands and 71 of their unaffected parents and siblings. A linear mixed effects model was used to test for familial association. The intra-class correlation coefficient (ICC) was used to determine the degree of association of the serum GM-CSF Ab level within families in comparison with the degree of association among families. RESULTS:The median (IQR) serum GM-CSF Ab concentration was higher in CD probands than in UC probands (1.5 [0.5,5.4]??g/mL vs 0.7 [0.3, 1.6]??g/mL, P?=?0.0002). The frequency of elevated serum GM-CSF Ab concentration ?1.6??g/mL was increased in unaffected siblings of CD probands with elevated GM-CSF Ab, compared with unaffected siblings of CD probands without elevated GM-CSF Ab (33% vs 13%, respectively, P?=?0.04). A similar result was observed within UC families. In families of CD patients, the mean (95th CI) ICC was equal to 0.153 (0.036, 0.275), P?=?0.001, whereas in families of UC patients, the mean (95th CI) ICC was equal to 0.27 (0.24, 0.31), P?=?0.047. CONCLUSIONS:These data confirmed familial association of serum GM-CSF Ab levels. This could be accounted for by either genetic or environmental factors shared within the family.

SUBMITTER: Wright SS 

PROVIDER: S-EPMC5916026 | biostudies-literature | 2018 May

REPOSITORIES: biostudies-literature

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Familial Association of Granulocyte-Macrophage Colony-Stimulating Factor Autoantibodies in Inflammatory Bowel Disease.

Wright Sandra S SS   Trauernicht Anna A   Bonkowski Erin E   McCall Courtney A CA   Maier Elizabeth A EA   Bezold Ramona R   Lake Kathleen K   Chalk Claudia C   Trapnell Bruce C BC   Kim Mi-Ok MO   Kugathasan Subra S   Denson Lee A LA  

Journal of pediatric gastroenterology and nutrition 20180501 5


<h4>Objectives</h4>Elevated granulocyte-macrophage colony-stimulating factor auto-antibodies (GM-CSF Ab) are associated with increased intestinal permeability and stricturing behavior in Crohn disease (CD). We tested for familial association of serum GM-CSF Ab level in CD and ulcerative colitis (UC) families.<h4>Methods</h4>Serum GM-CSF Ab concentration was determined in 230 pediatric CD probands and 404 of their unaffected parents and siblings, and 45 UC probands and 71 of their unaffected pare  ...[more]

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