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Pyoderma Gangrenosum in Inflammatory Bowel Disease: A Systematic Review and Meta-Analysis

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Abstract

Background

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.

Aims

To assess the incidence of PG and identify factors associated with PG in IBD patients.

Methods

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.

Results

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.

Conclusions

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.

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Acknowledgments

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.

Funding

This work was supported by the John W. Price and Barbara Thruston Atwood Price Trust and a grant from the Mary K. Oxley Foundation.

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Authors and Affiliations

  1. Price Institute of Surgical Research, The Hiram C. Polk Jr., MD Department of Surgery, University of Louisville, 550 South Jackson Street, Louisville, KY, 40202, USA

    Vanessa States, Stephen O’Brien, Henry L. Roberts, Mason Paas, Kayla Feagins & Susan Galandiuk

  2. Department of Cardiology, University of Louisville, Louisville, KY, USA

    Jayesh P. Rai

  3. Eli Lily and Company, Indianapolis, IN, USA

    Evangeline J. Pierce

  4. Department of Epidemiology, University of Louisville, Louisville, KY, USA

    Richard N. Baumgartner

Authors
  1. Vanessa States
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Contributions

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.

Corresponding author

Correspondence to Susan Galandiuk.

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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.

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Supplementary Figure

 1 PRISMA flow diagram of search process, inclusion, and exclusion criteria for studies investigating PG patient cohorts. (TIFF 11683 kb)

Supplementary Figure

 2 Venn diagram representation of the distribution of PG among total Louisville patient cohort (TIFF 56 kb)

Supplementary material 3 (DOCX 12 kb)

Supplementary material 4 (DOCX 13 kb)

Supplementary material 5 (DOCX 12 kb)

Supplementary material 6 (DOCX 12 kb)

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States, V., O’Brien, S., Rai, J.P. et al. Pyoderma Gangrenosum in Inflammatory Bowel Disease: A Systematic Review and Meta-Analysis. Dig Dis Sci 65, 2675–2685 (2020). https://doi.org/10.1007/s10620-019-05999-4

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