Small Intestinal Polyp Burden in Pediatric Peutz–Jeghers Syndrome Assessed through Capsule Endoscopy: A Longitudinal Study

Author:

Stewart Jeremy1ORCID,Fleishman Nathan R.2ORCID,Staggs Vincent S.3ORCID,Thomson Mike4,Stoecklein Nicole5,Lawson Caitlin E.6ORCID,Washburn Michael P.7ORCID,Umar Shahid8ORCID,Attard Thomas M.59ORCID

Affiliation:

1. Division of Pediatric Gastroenterology, University of Texas Southwestern Medical Center, Children’s Medical Center, Dallas, TX 75235, USA

2. Division of Gastroenterology, Levine Children’s Hospital, Charlotte, NC 28203, USA

3. Biostatistics and Epidemiology Core, Division of Health Services and Outcomes Research, Children’s Mercy Hospital, Kansas City, MO 64108, USA

4. Department of Paediatric Gastroenterology, Sheffield Children’s Hospital NHS Foundation Trust, Sheffield University, Sheffield S10 2TH, UK

5. Division of Gastroenterology, Children’s Mercy Hospital, Kansas City, MO 64108, USA

6. Division of Genetics, Children’s Mercy Hospital, Kansas City, MO 64108, USA

7. Department of Cancer Biology, The University of Kansas Medical Center, Kansas City, KS 66160, USA

8. Department of Surgery, The University of Kansas Medical Center, Kansas City, KS 66160, USA

9. University of Missouri–Kansas City School of Medicine, 2464 Charlotte St, Kansas City, MO 64108, USA

Abstract

The management of pediatric Peutz–Jeghers Syndrome (PJS) focuses on the prevention of intussusception complicating small intestinal (SI) polyposis. This hinges on the accurate appraisal of the polyp burden to tailor therapeutic interventions. Video Capsule Endoscopy (VCE) is an established tool to study SI polyps in children, but an in-depth characterization of polyp burden in this population is lacking. Methods: We performed a retrospective longitudinal cross-sectional analysis of VCE studies in pediatric PJS patients at our institution (CMKC) from 2010 to 2020. Demographic, clinical, and VCE findings reported by three reviewers in tandem were accrued. Polyp burden variables were modeled as functions of patient and study characteristics using linear mixed models adjusted for clustering. Results: The cohort included 15 patients. The total small bowel polyp count and largest polyp size clustered under 30 polyps and <20 mm in size. Luminal occlusion correlated closely with the estimated polyp size. Polyp distribution favored proximal (77%) over distal (66%) small bowel involvement. The adjusted largest polyp size was greater in males. Double Balloon Enteroscopy was associated with a decreased polyp burden. Conclusions: The polyp burden in pediatric PJS patients favors the proximal third of the small intestine, with relatively small numbers and a polyp size amenable to resection through enteroscopy. Male gender and older age were related to an increased polyp burden.

Publisher

MDPI AG

Subject

Pediatrics, Perinatology and Child Health

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