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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] μg/mL vs 0.7 [0.3, 1.6] μg/mL, P = 0.0002). The frequency of elevated serum GM-CSF Ab concentration ≥1.6 μg/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
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]