Dataset Information

Upper Gastrointestinal Screening of Polyp Load in Children With Familial Adenomatous Polyposis: Is It Required?

ABSTRACT:

Objectives and study

Patients with familial adenomatous polyposis (FAP) have a propensity to form not only large bowel polyps but also upper gastrointestinal (GI) polyps with malignant potential. International guidance suggests that upper GI screening need not begin until patients are at least into their twenties. It is our experience that patients develop upper GI polyps long before this point which have the potential for malignant change.

Methods

A prospective record of all upper GI endoscopies in children (aged 9 to 17) with FAP was kept across a 12-year period by 1 surgeon in our center. For each scope performed, we recorded the location, histology, and treatment of upper GI polyps.

Results

Twent-eight patients aged 9 to 17 underwent a total of 48 esophagogastroduodenoscopies across a 12-year period. Thirty-eight esophagogastroduodenoscopies (79%) identified at least 1 gastric or duodenal polyp in 22 (79%) patients; 10 (36%) patients had gastric adenomas. Eight (29%) patients showed very high numbers of polyps. All 21 patients who had duodenal polyps had adenomas. None had yet developed malignancy, but 1 patient required extensive polyp excision and one is awaiting endoscopic mucosal resection.

Conclusion

Our results demonstrate that young people with FAP are at the risk of developing upper GI polyps long before current guidance suggests screening the upper GI tract. We advocate for screening of the upper GI tract to start along with colonoscopy and happen at the same sitting for pediatric and young adult patients with FAP.

SUBMITTER: Middleton T

PROVIDER: S-EPMC10754608 | biostudies-literature | 2023 Feb

REPOSITORIES: biostudies-literature

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Publications

Upper Gastrointestinal Screening of Polyp Load in Children With Familial Adenomatous Polyposis: Is It Required?

Middleton Thomas T Sugarman Ian I

JPGN reports 20221202 1

<h4>Objectives and study</h4>Patients with familial adenomatous polyposis (FAP) have a propensity to form not only large bowel polyps but also upper gastrointestinal (GI) polyps with malignant potential. International guidance suggests that upper GI screening need not begin until patients are at least into their twenties. It is our experience that patients develop upper GI polyps long before this point which have the potential for malignant change.<h4>Methods</h4>A prospective record of all uppe ...[more]

PMID: 38293318

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Project description:ImportancePatients with familial adenomatous polyposis (FAP) are at markedly increased risk for duodenal adenomas and cancer. Combination sulindac and erlotinib was previously shown to reduce duodenal polyp burden but was associated with a relatively high adverse event (AE) rate.ObjectiveTo evaluate if a once weekly dosing schedule for erlotinib intervention improves the AE profile, while still providing efficacy with respect to reduced polyp burden, in participants with FAP.Design, setting and participantsSingle-arm trial, enrolling 46 participants with FAP, conducted from October 2017 to September 2019 in eight academic cancer centres.ExposuresParticipants self-administered 350 mg of erlotinib by mouth, one time per week for 6 months.Main outcomes and measuresDuodenal polyp burden (sum of polyp diameters) was assessed in the proximal duodenum by esophagogastroduodenoscopy performed at baseline and 6 months, with mean per cent change defined as the primary efficacy outcome of interest. Rate of grade 2-3 AEs was evaluated as a co-primary outcome. Secondary outcomes included changes in total duodenal polyp count, along with changes in lower gastrointestinal (GI) polyp burden and count (for participants examined by optional lower endoscopy).ResultsForty-six participants (mean age, 44.1 years (range, 18-68); women, 22 (48%)) were enrolled; 42 participants completed 6 months of intervention and were included in the per-protocol analysis. Duodenal polyp burden was significantly reduced after 6 months of weekly erlotinib intervention, with a mean per cent change of -29.6% (95% CI, -39.6% to -19.7%; p<0.0001). Similar results were observed in subgroup analyses defined by participants with advanced duodenal polyposis (Spigelman 3) at baseline (mean, -27%; 95% CI, -38.7% to -15.2%; p<0.0001). Post-intervention Spigelman stage was downstaged in 12% of the participants. Lower GI polyp number was also decreased after 6 months of intervention (median, -30.8%; IQR, -47.4% to 0.0%; p=0.0256). Grade 2 or 3 AEs were reported in 71.7% of subjects, with only two experiencing grade 3 toxicity at least possibly related to intervention.ConclusionIn this single-arm, multi-centre trial of participants with FAP, erlotinib one time per week resulted in markedly lower duodenal polyp burden, and modestly reduced lower GI polyp burden, after 6 months of intervention. While AEs were still reported by nearly three-quarters of all participants, these events were generally lower grade and well-tolerated. These findings support further investigation of erlotinib as an effective, acceptable cancer preventive agent for FAP-associated GI polyposis.Trial registration numberNCT02961374.

Dataset Information

Upper Gastrointestinal Screening of Polyp Load in Children With Familial Adenomatous Polyposis: Is It Required?

Objectives and study

Methods

Results

Conclusion

Publications

Upper Gastrointestinal Screening of Polyp Load in Children With Familial Adenomatous Polyposis: Is It Required?

Similar Datasets

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