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1. Evaluating Eosinophilic Colitis as a Unique Disease Using Colonic Molecular Profiles: A Multi-Site Study

Author

Tetsuo Shoda, Margaret H. Collins, Mark Rochman, Ting Wen, Julie M. Caldwell, Lydia E. Mack, Garrett A. Osswald, John A. Besse, Yael Haberman, Seema S. Aceves, Nicoleta C. Arva, Kelley E. Capocelli, Mirna Chehade, Carla M. Davis, Evan S. Dellon, Gary W. Falk, Nirmala Gonsalves, Sandeep K. Gupta, Ikuo Hirano, Paneez Khoury, Amy Klion, Calies Menard-Katcher, John Leung, Vincent A. Mukkada, Philip E. Putnam, Jonathan M. Spergel, Joshua B. Wechsler, Guang-Yu Yang, Glenn T. Furuta, Lee A. Denson, Marc E. Rothenberg, J. Pablo Abonia, Seema Aceves, Samuel Almonte, Rachel Andrews, Sara Anvari, Ashley Arrington, Nicoleta Arva, Fred Atkins, Dominique Bailey, Alexis Berry, Bridget Besl, Scott Bolton, Peter Bonis, Wendy Book, Kimberly Bray, Teresa Brown, Cassandra Burger, Deirdre Burke, Jonathon Cahoon, Kelley Capocelli, Eric Chiou, Margaret Collins, Carla Davis, Evan Dellon, Maureen DeMarshall, Lauren DiTommaso, Ranjan Dohil, Michael Eby, Gary Falk, David Fleischer, Heather Foote, Kelci Foss, Joel Friedlander, Patricia Fulkerson, Glenn Furuta, Debra Geno, Thomas Greuter, Sandeep Gupta, Frank Hamilton, Kirk Harris, Jennifer Harris, Girish Hiremath, Nicole Holland-Thomas, Lea Jacinto, Amir Kagalwalla, Timothy Kaseta, David Katzka, Kaitlin Keeley, Emad Khosh-Hemmat, Eileen King, Kara Kliewer, Jennifer Knowles, Kendra Kocher, Ellyn Kodroff, Jeffrey Krischer, Shay Kyle, Meredith Levy, Chris Liacouras, Denise Mack, Lisa Martin, Ellen Martin, Talaya McCright-Gill, Paul Menard-Katcher, Gabriela Mendoza, Melissa Mingler, Mike Minnicozzi, Amanda Muir, Vincent Mukkada, Cristin Murray-Petzold, Robert Newbury, Quan Nhu, Anthony Olive, Oghenekpaobor (Joel) Oyibo, Allisa Paliana, Zhaoxing Pan, Robbie Pesek, Kathryn Peterson, Heidi Poppendeck, Philip Putnam, Fabian Rivera, Marc Rothenberg, Amanda Rudman Spergel, Kathleen Sable, Alain Schoepfer, Melissa Scott, Rachel Sheridan, Selma Sinanovic, Jonathan Spergel, Mary Jo Strobel, Kiki Sun, Amy Tasco, Crystal Tholen, Katherine Thompson, Tiffany Tomkinson, Daisy Tran, Alexandra Tylicki, Tiina Urv, Mei-Lun Wang, Joshua Wechsler, Barry Wershil, Lisa Wheatley, Leah Wilkey, Angelika Zalewski, and Amy Zicarelli

Subjects
Colitis, Microscopic, Hepatology, Gastritis, Eosinophilia, Gastroenterology, Humans, Colitis, Inflammatory Bowel Diseases, Article, Enteritis
Abstract

BACKGROUND AND AIMS: Colonic eosinophilia, an enigmatic finding often referred to as eosinophilic colitis (EoC), is a poorly understood condition. Whether EoC is a distinct disease or a colonic manifestation of eosinophilic gastrointestinal diseases (EGIDs) or inflammatory bowel disease (IBD) is undetermined. METHODS: Subjects with EoC (n=27) and controls (normal [NL, n=20], Crohn disease [CD, n=14]) were enrolled across sites associated with the Consortium of Eosinophilic Gastrointestinal Disease Researchers (CEGIR). EoC was diagnosed as colonic eosinophilia (ascending ≥100, descending ≥85, sigmoid ≥65 eosinophils/high-power field) with related symptoms. Colon biopsies were subjected to RNA sequencing. Associations between gene expression and histologic features were analyzed with Spearman correlation; operational pathways and cellular constituents were computationally derived. RESULTS: We identified 987 differentially expressed genes (EoC transcriptome) between EoC and NL (>1.5-fold change, P < .05). Colonic eosinophil count correlated with 31% of EoC transcriptome, most notably with CCL11 and CLC (r=0.78 and 0.77, P < .001). Among EoC and other EGIDs, there was minimal transcriptomic overlap; and minimal evidence of a strong allergic type 2 immune response compared with other EGIDs. Decreased cell-cycle and increased apoptosis in EoC compared with NL were identified by functional enrichment analysis and immunostaining using Ki-67 and cleaved caspase-3. Pericryptal circumferential eosinophil collars were associated with the EoC transcriptome (P < .001). EoC transcriptome-based scores were reversible with disease remission and differentiated EoC from IBD, even after controlling for colonic eosinophil levels (P < .0001). CONCLUSION: We established EoC transcriptomic profiles, identified mechanistic pathways, and integrated findings with parallel IBD and EGID data. These findings establish EoC as a distinct disease compared with other EGIDs and IBD, thereby providing a basis for improving diagnosis and treatment.

Published
2022
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2. Loss of Endothelial TSPAN12 Promotes Fibrostenotic Eosinophilic Esophagitis via Endothelial Cell–Fibroblast Crosstalk

Author

Tetsuo Shoda, Ting Wen, Julie M. Caldwell, Netali Ben-Baruch Morgenstern, Garrett A. Osswald, Mark Rochman, Lydia E. Mack, Jennifer M. Felton, J. Pablo Abonia, Nicoleta C. Arva, Dan Atkins, Peter A. Bonis, Kelley E. Capocelli, Margaret H. Collins, Evan S. Dellon, Gary W. Falk, Nirmala Gonsalves, Sandeep K. Gupta, Ikuo Hirano, John Leung, Paul A. Menard-Katcher, Vincent A. Mukkada, Philip E. Putnam, Amanda K. Rudman Spergel, Jonathan M. Spergel, Joshua B. Wechsler, Guang-Yu Yang, Seema S. Aceves, Glenn T. Furuta, Marc E. Rothenberg, Seema Aceves, Samuel Almonte, Rachel Andrews, Ashley Arrington, Nicoleta Arva, Fred Atkins, Dominique Bailey, Alexis Berry, Bridget Besl, Scott Bolton, Peter Bonis, Wendy Book, Kimberly Bray, Teresa Brown, Cassandra Burger, Deirdre Burke, Jonathon Cahoon, Kelley Capocelli, Mirna Chehade, Margaret Collins, Carla Davis, Evan Dellon, Maureen DeMarshall, Lauren DiTommaso, Ranjan Dohil, Michael Eby, Gary Falk, David Fleischer, Heather Foote, Kelci Foss, Joel Friedlander, Patricia Fulkerson, Glenn Furuta, Debra Geno, Thomas Greuter, Sandeep Gupta, Frank Hamilton, Kirk Harris, Jennifer Harris, Girish Hiremath, Nicole Holland-Thomas, Lea Jacinto, Amir Kagalwalla, Timothy Kaseta, David Katzka, Kaitlin Keeley, Emad Khosh-Hemmat, Paneez Khoury, Eileen King, Kara Kliewer, Amy Klion, Jennifer Knowles, Kendra Kocher, Ellyn Kodroff, Jeffrey Krischer, Shay Kyle, Meredith Levy, Chris Liacouras, Denise Mack, Lisa Martin, Ellen Martin, Talaya McCright-Gill, Paul Menard-Katcher, Calies Menard-Katcher, Gabriela Mendoza, Melissa Mingler, Mike Minnicozzi, Amanda Muir, Vincent Mukkada, Cristin MurrayPetzold, Robert Newbury, Quan Nhu, Oghenekpaobor (Joel) Oyibo, Allisa Paliana, Zhaoxing Pan, Robbie Pesek, Kathryn Peterson, Heidi Poppendeck, Philip Putnam, Fabian Rivera, Marc Rothenberg, Amanda Rudman Spergel, Kathleen Sable, Alain Schoepfer, Melissa Scott, Rachel Sheridan, Selma Sinanovic, Jonathan Spergel, MaryJo Strobel, Kiki Sun, Amy Tasco, Crystal Tholen, Katherine Thompson, Tiffany Tomkinson, Daisy Tran, Alexandra Tylicki, Tiina Urv, Mei-Lun Wang, Joshua Wechsler, Barry Wershil, Lisa Wheatley, Leah Wilkey, Angelika Zalewski, and Amy Zicarelli

Subjects
Adult, Male, Pathology, medicine.medical_specialty, Thymic stromal lymphopoietin, Adolescent, Endothelium, Tetraspanins, medicine.drug_class, Proton-pump inhibitor, Young Adult, Esophagus, Fibrosis, Eosinophilic, medicine, Humans, Gene Silencing, RNA, Small Interfering, Child, Eosinophilic esophagitis, Interleukin-13, Hepatology, business.industry, Gastroenterology, Endothelial Cells, Eosinophilic Esophagitis, Fibroblasts, Middle Aged, Eosinophil, medicine.disease, medicine.anatomical_structure, Gene Expression Regulation, Gastrointestinal disease, Child, Preschool, Esophageal Stenosis, Female, business
Abstract

Eosinophilic esophagitis (EoE) can progress to fibrostenosis by unclear mechanisms. Herein, we investigated gene dysregulation in fibrostenotic EoE, its association with clinical parameters and specific pathways, and the functional consequences.Esophageal biopsies from subjects with EoE were collected across 11 Consortium of Eosinophilic Gastrointestinal Disease Researchers sites (n = 311) and 2 independent replication cohorts (n = 83). Inclusion criteria for fibrostenotic EoE were endoscopic rings, stricture, and/or a history of dilation. Endoscopic, histologic, and molecular features were assessed by the EoE Endoscopic Reference Score, EoE Histology Scoring System, EoE Diagnostic Panel, and RNA sequencing. Esophageal endothelial TSPAN12 expression and functional effects on barrier integrity and gene expression were analyzed in vitro.TSPAN12 was the gene most correlated with fibrostenosis (r = -0.40, P.001). TSPAN12 was lower in fibrostenotic EoE and correlated with EoE Endoscopic Reference Score, EoE Diagnostic Panel, and EoE Histology Scoring System (r = 0.34-0.47, P.001). Lower TSPAN12 associated with smaller esophageal diameter (r = 0.44, P = .03), increased lamina propria fibrosis (r = -0.41, P.001), and genes enriched in cell cycle-related pathways. Interleukin (IL)-13 reduced TSPAN12 expression in endothelial cells. Conversely, anti-IL-13 therapy increased TSPAN12 expression. TSPAN12 gene silencing increased endothelial cell permeability and dysregulated genes associated with extracellular matrix pathways. Endothelial cell-fibroblast crosstalk induced extracellular matrix changes relevant to esophageal remodeling.Patients with fibrostenotic EoE express decreased levels of endothelial TSPAN12. We propose that IL-13 decreases TSPAN12, likely contributing to the chronicity of EoE by promoting tissue remodeling through fibroblast-endothelial cell crosstalk.

Published
2022
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3. Advancing patient care through the Consortium of Eosinophilic Gastrointestinal Disease Researchers (CEGIR)

Author

Seema Aceves, Margaret H. Collins, Marc E. Rothenberg, Glenn T. Furuta, Nirmala Gonsalves, J. Pablo Abonia, Samuel Almonte, Rachel Andrews, Ashley Arrington, Nicoleta Arva, Fred Atkins, Dominique Bailey, Alexis Berry, Bridget Besl, Scott Bolton, Peter Bonis, Wendy Book, Kimberly Bray, Teresa Brown, Cassandra Burger, Deirdre Burke, Jonathon Cahoon, Kelley Capocelli, Mirna Chehade, Margaret Collins, Carla Davis, Evan Dellon, Maureen DeMarshall, Lauren DiTommaso, Ranjan Dohil, Michael Eby, Gary Falk, David Fleischer, Heather Foote, Kelci Foss, Joel Friedlander, Patricia Fulkerson, Glenn Furuta, Debra Geno, Thomas Greuter, Sandeep Gupta, Frank Hamilton, Kirk Harris, Jennifer Harris, Ikuo Hirano, Girish Hiremath, Nicole Holland-Thomas, Lea Jacinto, Amir Kagalwalla, Timothy Kaseta, David Katzka, Kaitlin Keeley, Emad Khosh-Hemmat, Paneez Khoury, Eileen King, Kara Kliewer, Amy Klion, Jennifer Knowles, Kendra Kocher, Ellyn Kodroff, Jeffrey Krischer, Shay Kyle, John Leung, Meredith Levy, Chris Liacouras, Denise Mack, Lisa Martin, Ellen Martin, Talaya McCright-Gill, Paul Menard-Katcher, Calies Menard-Katcher, Gabriela Mendoza, Melissa Mingler, Mike Minnicozzi, Amanda Muir, Vincent Mukkada, Cristin MurrayPetzold, Robert Newbury, Quan Nhu, Oghenekpaobor (Joel) Oyibo, Allisa Paliana, Zhaoxing Pan, Robbie Pesek, Kathryn Peterson, Heidi Poppendeck, Philip Putnam, Fabian Rivera, Marc Rothenberg, Amanda Rudman-Spergel, Kathleen Sable, Alain Schoepfer, Melissa Scott, Rachel Sheridan, Selma Sinanovic, Jonathan Spergel, MaryJo Strobel, Kiki Sun, Amy Tasco, Crystal Tholen, Katherine Thompson, Tiffany Tomkinson, Daisy Tran, Alexandra Tylicki, Tiina Urv, Mei-Lun Wang, Joshua Wechsler, Barry Wershil, Lisa Wheatley, Leah Wilkey, Guang-Yu Yang, Angelika Zalewski, and Amy Zicarelli

Subjects
Clinical Trials as Topic, Medical education, Biomedical Research, Eosinophilic gastritis, business.industry, Immunology, Eosinophilic Esophagitis, Patient advocacy, Enteritis, United States, Article, Patient care, Clinical trial, Natural history, National Institutes of Health (U.S.), Multidisciplinary approach, Gastritis, Eosinophilia, Eosinophilic, Immunology and Allergy, Medicine, Patient-reported outcome, business
Abstract

Recent advances in rare disease research are accelerated by the work of consortia that have been supported by the National Institutes of Health. Development of such consortia rely on multidisciplinary relationships and engagement with patient advocacy groups, as well as the National Institutes of Health and industry and academic partners. In this rostrum we present the development of such a process that focuses on eosinophilic gastrointestinal diseases. Principal investigators, patient advocacy groups, research assistants, and trainees work together to perform natural history studies that promote clinical trial readiness tools, conduct clinical trials, train a new generation of investigators, and perform innovative pilot studies.

Published
2020
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4. Longitudinal Outcomes in Young Patients with Alpha-1-Antitrypsin Deficiency with Native Liver Reveal that Neonatal Cholestasis is a Poor Predictor of Future Portal Hypertension

Author

Jeffrey Teckman, Philip Rosenthal, Kieran Hawthorne, Cathie Spino, Lee M. Bass, Karen F. Murray, Nanda Kerkar, John C. Magee, Saul Karpen, James E. Heubi, Jean P. Molleston, Robert H. Squires, Binita M. Kamath, Stephen L. Guthery, Kathleen M. Loomes, Averell H. Sherker, Ronald J. Sokol, Estella Alonso, Lee Bass, Susan Kelly, Mary Riordan, Hector Melin-Aldana, Jorge Bezerra, Kevin Bove, James Heubi, Alexander Miethke, Greg Tiao, Julie Denlinger, Erin Chapman, Ronald Sokol, Amy Feldman, Cara Mack, Michael Narkewicz, Frederick Suchy, Shikha Sundaram, Johan Van Hove, Benigno Garcia, Mikaela Kauma, Kendra Kocher, Matthew Steinbeiss, Mark Lovell, Kathleen Loomes, David Piccoli, Elizabeth Rand, Pierre Russo, Nancy Spinner, Jessi Erlichman, Samantha Stalford, Dina Pakstis, Sakya King, Robert Squires, Rakesh Sindhi, Veena Venkat, Kathy Bukauskas, Patrick McKiernan, Lori Haberstroh, James Squires, Laura Bull, Joanna Curry, Camille Langlois, Grace Kim, Jeffery Teckman, Vikki Kociela, Rosemary Nagy, Shraddha Patel, Jacqueline Cerkoski, Molly Bozic, Girish Subbarao, Ann Klipsch, Cindy Sawyers, Oscar Cummings, Simon Horslen, Karen Murray, Evelyn Hsu, Kara Cooper, Melissa Young, Laura Finn, Binita Kamath, Vicky Ng, Claudia Quammie, Juan Putra, Deepika Sharma, Aishwarya Parmar, Stephen Guthery, Kyle Jensen, Ann Rutherford, Amy Lowichik, Linda Book, Rebecka Meyers, Tyler Hall, Kasper Wang, Sonia Michail, Danny Thomas, Catherine Goodhue, Rohit Kohli, Larry Wang, Nisreen Soufi, Daniel Thomas, Nitika Gupta, Rene Romero, Miriam B. Vos, Rita Tory, John-Paul Berauer, Carlos Abramowsky, Jeanette McFall, Benjamin Shneider, Sanjiv Harpavat, Paula Hertel, Daniel Leung, Mary Tessier, Deborah Schady, Laurel Cavallo, Diego Olvera, Christina Banks, Cynthia Tsai, Richard Thompson, Edward Doo, Jay Hoofnagle, Averell Sherker, Rebecca Torrance, Sherry Hall, John Magee, Robert Merion, and Wen Ye

Subjects
Adult, Male, medicine.medical_specialty, Cirrhosis, Adolescent, medicine.medical_treatment, Cholestasis, Intrahepatic, Liver transplantation, Article, Young Adult, 03 medical and health sciences, Liver disease, 0302 clinical medicine, Cholestasis, alpha 1-Antitrypsin Deficiency, 030225 pediatrics, Internal medicine, Hypertension, Portal, medicine, Humans, Longitudinal Studies, 030212 general & internal medicine, Neonatal cholestasis, Child, Alpha 1-antitrypsin deficiency, business.industry, Infant, Newborn, Infant, medicine.disease, Liver Transplantation, Transplantation, Child, Preschool, Pediatrics, Perinatology and Child Health, Disease Progression, Portal hypertension, Female, business
Abstract

Objectives To identify predictors of portal hypertension, liver transplantation, and death in North American youth with alpha-1-antitrypsin (AAT) deficiency, and compare with patients with AAT deficiency elsewhere. Study design The Childhood Liver Disease Research Network Longitudinal Observational Study of Genetic Causes of Intrahepatic Cholestasis is a prospective, cohort study of pediatric cholestatic liver diseases, including AAT deficiency, enrolling PIZZ and PISZ subjects 0-25 years of age seen since November 2007 at 17 tertiary care centers in the US and Canada. Data from standard-of-care baseline and annual follow-up visits were recorded from medical records, history, physical examination, and laboratory studies. Participants with portal hypertension were identified based on data collected. Results We enrolled 350 participants (60% male) with a native liver; 278 (79%) entered the cohort without portal hypertension and 18 developed portal hypertension during follow-up. Thirty participants required liver transplantation; 2 patients died during 1077 person-years of follow-up. There was no difference in participants with or without preceding neonatal cholestasis progressing to transplantation or death during the study (12% vs 7%; P = .09), or in experiencing portal hypertension (28% vs 21%; P = .16); the hazard ratio for neonatal cholestasis leading to portal hypertension was P = .04. Development of portal hypertension was associated with a reduced height Z-score. Conclusions Portal hypertension in youth with AAT deficiency impacts growth measures. Progression to liver transplantation is slow and death is rare, but the risk of complications and severe liver disease progression persists throughout childhood. A history of neonatal cholestasis is a weak predictor of severe disease.

Published
2020
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5. Mo1187 DILATION REDUCES POSITIVE CORRELATION BETWEEN SYMPTOMS AND HISTOLOGIC FINDINGS IN ADULTS WITH EOSINOPHILIC ESOPHAGITIS

Author

Qin Sun, J.P. Abonia, Ekaterina Safroneeva, Michael D. Eby, Jonathan M. Spergel, Eileen King, Evan S. Dellon, Dan Atkins, Glenn T. Furuta, Jessi Lipscomb, Guang Yu Yang, Gary W. Falk, Margaret H. Collins, Vincent A. Mukkada, Seema S. Aceves, Nirmala Gonsalves, Kelley E. Capocelli, Marc E. Rothenberg, Sandeep K. Gupta, Zhaoxing Pan, Nicoleta C. Arva, Kendra Kocher, Paul Menard-Katcher, Peter A Bonis, John Leung, Alain M. Schoepfer, Ikuo Hirano, and Barry K. Wershil

Subjects
medicine.medical_specialty, Hepatology, business.industry, Internal medicine, Gastroenterology, medicine, Dilation (morphology), business, Positive correlation, Eosinophilic esophagitis, medicine.disease
Published
2020
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6. Fascia Iliaca Compartment Nerve Block Versus Systemic Pain Control for Acute Femur Fractures in the Pediatric Emergency Department

Author

Kendra Kocher, Sara J. Deakyne, Joseph Wathen, Tara L. Neubrand, and Kelley Roswell

Subjects
Male, Pediatric emergency, medicine.medical_specialty, Adolescent, medicine.medical_treatment, Pain, Young Adult, medicine, Humans, Pain Management, Femur, Fascia iliaca, Fascia, Young adult, Child, Adverse effect, Retrospective Studies, Analgesics, business.industry, Infant, Nerve Block, Retrospective cohort study, General Medicine, Surgery, Child, Preschool, Anesthesia, Injections, Intravenous, Pediatrics, Perinatology and Child Health, Emergency Medicine, Nerve block, Population study, Female, Emergency Service, Hospital, business, Femoral Fractures
Abstract

To compare management of acute femur fractures in children who received a fascia iliaca compartment nerve block (FICNB) to those who received systemic intravenously administered analgesics in the pediatric emergency department. The comparison evaluated frequency of use, effectiveness, and associated adverse event profiles.Study population was derived from a retrospective chart review of pediatric patients sustaining acute femur fractures between 2005 and 2009. Cases (received FICNB) were compared with controls (only systemic analgesia) in terms of effectiveness and adverse event. Outcomes included total doses of systemic medications received and comparison of preintervention and postintervention pain scores.Two hundred fifty-nine charts were reviewed: 158 who received FICNB versus 101 who did not. The median dose of systemic medications was 1 dose lower in the FICNB group compared with the systemic medications group. This remained significant after controlling for age and preintervention pain scores (P = 0.02). Median postintervention pain scores in the FICNB group were 1.5 points lower than those in the systemic medications group. This remained significant while controlling for preintervention pain scores and age (P0.01). There was no difference in the total adverse events between the FICNB and the control group in either the unadjusted or adjusted analyses (P = 0.08). The FICNB group had 2 seizure episodes, one of which had associated subarachnoid hemorrhage. No patient in either group experienced bradycardia, arrhythmia, visual disturbance, abnormal hearing, mouth numbness, motor tremors, pain or bleeding at injection site, or prolonged nerve block.We report on the largest number of FICNBs administered in a pediatric emergency department for acute femur fractures. Effectiveness, as measured by pain scores and total doses of systemic analgesia, was improved in the FICNB group versus the control. There was no difference in adverse events between the groups.

Published
2014
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7. Alignment of parent- and child-reported outcomes and histology in eosinophilic esophagitis across multiple CEGIR sites

Author

Vincent A. Mukkada, Paul Menard-Katcher, Alicia Hurnton, Gary W. Falk, Guang Yu Yang, Wendy Book, Heather Foote, Deirdre Burke, Katherine Thompson, Eileen King, Mary Jo Strobel, Jonathan Kuhl, Ikuo Hirano, Barry K. Wershil, Denise Mack, Jeanie Bailey, Ellyn Kodroff, Allison Dubner, Melissa K. Mingler, Cathy Reidy, Kelley E. Capocelli, Susan Moist, Zhaoxing Pan, John Leung, Amanda B. Muir, Heidi Poppendeck, Maureen DeMarschall, Glenn T. Furuta, Jessi Lipscomb, Megan Lewis, John A. Besse, Melissa Scott, Margaret H. Collins, Dan Atkins, Amanda K. Rudman-Spergel, Sarah McGee, Qin Sun, Michael D. Eby, Evan S. Dellon, Marc E. Rothenberg, Sandeep K. Gupta, Kathleen Sable, Shaobo Guan, Joshua B. Wechsler, Seema S. Aceves, Nirmala Gonsalves, Christina L Carpenter, Kendra Kocher, Shay Kyle, Jeffrey P. Krischer, J. Pablo Abonia, Jonathan M. Spergel, Jacquelyn Covington, Peter A Bonis, Ashley Arrington, Ranjan Dohil, and Philip E. Putnam

Subjects
Parents, Male, Allergy, pediatric QOL EoE module, Disease, Eosinophil, eosinophilic esophagitis, 0302 clinical medicine, Quality of life, Interquartile range, Surveys and Questionnaires, Eosinophilic, Immunology and Allergy, Child, pediatric eosinophilic esophagitis symptom score, eosinophilic oesophagitis, Pediatric, patient-reported outcomes, Child, Preschool, 030220 oncology & carcinogenesis, Female, 030211 gastroenterology & hepatology, Patient-reported outcome, medicine.medical_specialty, Adolescent, Clinical Trials and Supportive Activities, Immunology, Food Allergies, Consortium of Eosinophilic Gastrointestinal Disease Researchers, Article, 03 medical and health sciences, Clinical Research, version 2, Internal medicine, medicine, Humans, Patient Reported Outcome Measures, Preschool, Eosinophilic esophagitis, business.industry, Eosinophilic Esophagitis, medicine.disease, Clinical trial, quality of life, Quality of Life, GERD, symptoms, Self Report, Digestive Diseases, business
Abstract

BACKGROUND: Patient-reported outcome (PRO) metrics for eosinophilic esophagitis (EoE) have been developed and validated but not used in a multicenter pediatric population or systematically aligned with histology. OBJECTIVE: We sought to understand 1) the potential of care-giver report to predict patient self-reported symptoms and 2) the correlation of PRO domains with histology. METHODS: Subjects with EoE (n=310) and their parents participating in the Consortium of Gastrointestinal Eosinophilic Disease Researchers (CEGIR) observational clinical trial were queried for baseline patient symptoms and quality of life (QOL) using the Pediatric Eosinophilic Esophagitis Symptom Score version 2 (PEESSv2.0), Pediatric QOL EoE module (PedsQL-EoE) and biopsies were analyzed using the EoE histology scoring system (EoEHSS). RESULTS: PEESSv2.0 parent and child report aligned across all domains (r=0.68-0.73, p

Published
2018
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