Pyoderma Gangrenosum in Inflammatory Bowel Disease: A Systematic Review and Meta-Analysis
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Pyoderma gangrenosum (PG) is an uncommon but severe extra-intestinal manifestation (EIM) of inflammatory bowel disease (IBD). The incidence and risk factors for PG are disputed.
To assess the incidence of PG and identify factors associated with PG in IBD patients.
A search of electronic databases (Ovid and PubMed) was conducted between 1966 and 2019. Studies that calculated the incidence of PG in IBD patient cohorts were included. Patient demographics, IBD subtype, and EIM presence were recorded. A review of our institutional database of 1057 IBD patients was conducted. A multivariate regression model and meta-analysis were conducted to identify risk factors for PG. A random effects model was used to combine the data of included studies.
Fourteen studies were included in addition to 1057 IBD patients and 26 PG cases from the Louisville cohort. In total, there were 379 cases of PG in the cumulative cohort of 61,695 IBD patients. The PG incidence in individual studies ranged from 0.4 to 2.6%. In the institutional cohort, ocular EIMs and a permanent stoma were significant risk factors for PG. In the meta-analysis, PG was associated with female gender (RR = 1.328, 95% CI 1.161–1.520), Crohn’s disease (RR = 1.193, 95% CI 1.001–1.422), erythema nodosum (RR = 9.281, 95% CI 6.081–14.164), and ocular EIM (RR = 4.55, 95% CI 3.04–6.81). There was study heterogeneity when assessing IBD subtype, ocular, and joint EIMs.
There are conflicting data on the incidence and risk factors for PG. This meta-analysis confirms an association between PG and female gender, Crohn’s disease, erythema nodosum, and ocular EIM that have been described in smaller studies.
KeywordsPyoderma gangrenosum Inflammatory bowel disease Systematic review Meta-analysis Extra-intestinal manifestations
The statistical methods for this paper were reviewed by Dr. Shesh Rai, Wendell Cherry Chair in Clinical Trial Research, Professor of Biostatistics, University of Louisville, Louisville, Kentucky, USA.
VS, SG helped in original study design; VS, SOB, HR, MP, KF helped in data collection ; JR, EP, RB, KF helped in data analysis; VS, SOB, SG helped in data interpretation; VS, SOB, HR, JR, KF helped in drafting of article; VS, SOB, MP helped in figures; SOB, MP, EP, KF, RB SG helped in revision of manuscript. All authors had access to all the data and approved final draft of manuscript.
This work was supported by the John W. Price and Barbara Thruston Atwood Price Trust and a grant from the Mary K. Oxley Foundation.
Compliance with Ethical Standards
Conflict of interest
EP is an employee and shareholder in Eli Lily & Company, Indianapolis, Indiana, USA. Eli Lily & Company had no role in this manuscript. This work was completed while she was a graduate student at University of Louisville. There are no other conflicts of interests.
- 1.Kappelman MD, Rifas-Shiman SL, Kleinman K, et al. The prevalence and geographic distribution of Crohn’s disease and ulcerative colitis in the United States. Clin Gastroenterol Hepatol Off Clin Pract J Am Gastroenterol Assoc. 2007;5:1424–1429.Google Scholar
- 6.Christodoulou DK, Katsanos KH, Kitsanou M, Stergiopoulou C, Hatzis J, Tsianos EV. Frequency of extraintestinal manifestations in patients with inflammatory bowel disease in Northwest Greece and review of the literature. Dig Liver Dis Off J Ital Soc Gastroenterol Ital Assoc Study Liver. 2002;34:781–786.Google Scholar
- 12.Xia FD, Liu K, Lockwood S, et al. Risk of developing pyoderma gangrenosum after procedures in patients with a known history of pyoderma gangrenosum—a retrospective analysis. J Am Acad Dermatol. 2018;78:e311.Google Scholar
- 16.U.S. Department of Health and Human Services, Centers for Disease Control and Prevention. Behavioral Risk Factor Surveillance System Survey Questionnaire. Accessed February 24, 2018; 2018.Google Scholar
- 19.Rothman KJ, Greenland S, Lash TL. Modern Epidemiology. Philadelphia: Lippincott Williams & Wilkins; 2008.Google Scholar
- 37.McElnea E, Stephenson K, Fulcher T. Pyoderma gangrenosum affecting the eye, orbit, and adnexa. A review. Orbit (Amsterdam, Netherlands).. 2018;37:26–31.Google Scholar
- 40.Menachem Y, Gotsman I. Clinical manifestations of pyoderma gangrenosum associated with inflammatory bowel disease. Israel Med Assoc J IMAJ. 2004;6:88–90.Google Scholar