Small Intestinal Polyp Burden in Pediatric Peutz–Jeghers Syndrome Assessed through Capsule Endoscopy: A Longitudinal Study
Abstract
:1. Introduction
2. Materials and Methods
2.1. Ethical Conduct of Research
2.2. Cohort Accrual
2.3. Individual Data Extraction
2.4. Data Recording
2.5. Statistical Analysis
3. Results
3.1. Cohort Characteristics
3.2. Symptoms at the Time of Study
3.3. Polyp Burden and Distribution
3.4. Polyp Burden and Outcome: Relationship with DAE
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Acknowledgments
Conflicts of Interest
Abbreviations
PJS | Peutz–Jeghers Syndrome |
VCE | Video Capsule Endoscopy |
CMKC | Children’s Mercy Kansas City |
DAE | device-assisted enteroscopy |
DBE | Double Balloon Enteroscopy |
pPJS | pediatric Peutz–Jeghers Syndrome |
MRE | magnetic resonance enterography |
PV | pathogenic variant |
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Maximum Polyp Size (mm) | ||||||
---|---|---|---|---|---|---|
Total Polyp Count | No Visible Polyps | <5 | 6–10 | 11–20 | 21–30 | >30 |
0 | 4 | 0 | 0 | 0 | 0 | 0 |
1–10 | 0 | 9 | 2 | 1 | 1 | 1 |
11–20 | 0 | 3 | 3 | 3 | 1 | 1 |
21–30 | 0 | 5 | 6 | 2 | 1 | 0 |
31–40 | 0 | 0 | 2 | 1 | 0 | 0 |
41–50 | 0 | 1 | 0 | 1 | 0 | 0 |
51–60 | 0 | 0 | 0 | 1 | 0 | 0 |
61–70 | 0 | 0 | 0 | 1 | 0 | 1 |
NA | 0 | 3 | 3 | 1 | 3 | 0 |
Outcome Variable | ||||||
---|---|---|---|---|---|---|
Explanatory Variable | Total Polyps | Max Polyp Size | Max Luminal Occlusion | |||
Beta (95% CI) | p | Beta (95% CI) | p | Beta (95% CI) | p | |
Max polyp size | 0.5 (0, 0.9) | 0.036 | ||||
Max luminal occlusion | 0.5 (0.1, 0.9) | 0.026 | 0.9 (0.8, 1) | <0.001 | ||
Patient age at procedure | 0.3 (−0.2, 0.8) | 0.253 | −0.1 (−0.7, 0.5) | 0.742 | 0 (−0.6, 0.5) | 0.856 |
Female (vs. male) | 0 (−0.4, 0.4) | 0.848 | −0.3 (−0.7, 0.1) | 0.112 | −0.2 (−0.6, 0.3) | 0.433 |
Symptomatic | 0 (−0.2, 0.2) | 0.973 | 0.1 (−0.2, 0.3) | 0.639 | 0 (−0.3, 0.2) | 0.840 |
DBE (vs. no DBE, surgeries excluded) | −0.1 (−0.4, 0.2) | 0.452 | 0.1 (−0.2, 0.4) | 0.560 | 0.1 (−0.1, 0.3) | 0.273 |
Total VCEs for patient | 0 (−0.4, 0.4) | 0.930 | −0.1 (−0.4, 0.3) | 0.610 | 0 (−0.4, 0.3) | 0.812 |
Study duration | 0.4 (0, 0.9) | 0.068 | 0.1 (−0.3, 0.4) | 0.609 | 0.2 (−0.1, 0.4) | 0.298 |
Total Polyps (Categorized) | Max Polyp Size (Categorized) | Max Luminal Occlusion (Categorized) | ||||
---|---|---|---|---|---|---|
B (95% CI) | p-Value | B (95% CI) | p-Value | B (95% CI) | p-Value | |
Female (vs. male) | −0.1 (−1.6, 1.5) | 0.945 | −0.9 (−2, 0.2) | 0.105 | −0.5 (−1.9, 0.9) | 0.472 |
Age (years) | 0.2 (0, 0.3) | 0.044 | −0.1 (−0.2, 0) | 0.064 | −0.1 (−0.2, 0) | 0.113 |
Duration (hours) | 0.3 (0.1, 0.5) | 0.001 | −0.1 (−0.2, 0.1) | 0.413 | 0 (−0.2, 0.1) | 0.913 |
Total VCEs | 0.1 (−0.3, 0.5) | 0.761 | 0.1 (−0.1, 0.4) | 0.273 | 0.1 (−0.2, 0.4) | 0.393 |
Procedure | ||||||
DBE | −1.0 (−2.5, 0.6) | 0.222 | −1.1 (−2.7, 0.4) | 0.117 | 0 (−1, 1) | 0.955 |
Surgery | 0.2 (−1, 1.4) | 0.699 | −1.1 (−2.5, 0.4) | 0.128 | 1.3 (−0.9, 3.5) | 0.196 |
Neither | Referent | Referent | Referent |
Tertile 1 | Tertile 2 | Tertile 3 | ||||
---|---|---|---|---|---|---|
B (95% CI) | p-Value | B (95% CI) | p-Value | B (95% CI) | p-Value | |
Female (vs. male) | −0.1 (−0.9, 0.6) | 0.704 | 0.1 (−1.3, 1.6) | 0.855 | −0.1 (−0.7, 0.6) | 0.832 |
Age (years) | 0 (−0.1, 0.1) | 0.528 | 0.1 (0, 0.2) | 0.070 | 0.1 (0, 0.1) | 0.040 |
Duration (hours) | 0 (−0.2, 0.1) | 0.818 | 0.1 (−0.1, 0.3) | 0.149 | 0.1 (−0.1, 0.2) | 0.284 |
Total VCEs | 0 (−0.2, 0.2) | 0.885 | 0 (−0.3, 0.4) | 0.826 | 0 (−0.2, 0.1) | 0.888 |
Procedure | ||||||
DBE | 0.9 (−0.3, 2.1) | 0.102 | −0.3 (−1.9, 1.3) | 0.657 | −0.1 (−1.8, 1.6) | 0.880 |
Surgery | 0.5 (−0.3, 1.3) | 0.198 | −0.4 (−1.5, 0.8) | 0.434 | −0.1 (−1.5, 1.2) | 0.817 |
Neither | Referent | Referent |
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Stewart, J.; Fleishman, N.R.; Staggs, V.S.; Thomson, M.; Stoecklein, N.; Lawson, C.E.; Washburn, M.P.; Umar, S.; Attard, T.M. Small Intestinal Polyp Burden in Pediatric Peutz–Jeghers Syndrome Assessed through Capsule Endoscopy: A Longitudinal Study. Children 2023, 10, 1680. https://doi.org/10.3390/children10101680
Stewart J, Fleishman NR, Staggs VS, Thomson M, Stoecklein N, Lawson CE, Washburn MP, Umar S, Attard TM. Small Intestinal Polyp Burden in Pediatric Peutz–Jeghers Syndrome Assessed through Capsule Endoscopy: A Longitudinal Study. Children. 2023; 10(10):1680. https://doi.org/10.3390/children10101680
Chicago/Turabian StyleStewart, Jeremy, Nathan R. Fleishman, Vincent S. Staggs, Mike Thomson, Nicole Stoecklein, Caitlin E. Lawson, Michael P. Washburn, Shahid Umar, and Thomas M. Attard. 2023. "Small Intestinal Polyp Burden in Pediatric Peutz–Jeghers Syndrome Assessed through Capsule Endoscopy: A Longitudinal Study" Children 10, no. 10: 1680. https://doi.org/10.3390/children10101680
APA StyleStewart, J., Fleishman, N. R., Staggs, V. S., Thomson, M., Stoecklein, N., Lawson, C. E., Washburn, M. P., Umar, S., & Attard, T. M. (2023). Small Intestinal Polyp Burden in Pediatric Peutz–Jeghers Syndrome Assessed through Capsule Endoscopy: A Longitudinal Study. Children, 10(10), 1680. https://doi.org/10.3390/children10101680