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[Peutz-Jeghers syndrome: variability of gastrointestinal expression at pediatric age]
Ana I Lopes1, Juan Gonçalves, Ana M Palha
1Serviço de Pediatria, Hospital de Santa Maria, Lisboa.
Insights
Peutz-Jeghers syndrome (PJS) presents unique challenges in children under 10. Early gastrointestinal lesions and dysplasia highlight the need for tailored pediatric surveillance guidelines.
Area of Science:
- Genetics and rare diseases
- Pediatric gastroenterology
- Oncology
Context:
- Peutz-Jeghers syndrome (PJS) is a rare autosomal dominant disorder.
- Characterized by gastrointestinal polyposis, mucocutaneous pigmentation, and increased cancer risk.
- Pediatric PJS primarily involves intestinal polyp complications, but neoplastic risk exists.
Purpose:
- To highlight the diverse gastrointestinal manifestations of PJS in children under 10.
- To underscore the limitations of conventional diagnostic tools in this age group.
- To emphasize the need for specific surveillance guidelines for young children with PJS.
Summary:
- Presents 5 pediatric cases (under 10 years) of PJS with varied GI presentations.
- Includes a case with colonic polyp dysplasia, indicating early neoplastic potential.
- Conventional radiology and endoscopy have limitations in diagnosing young children.
Impact:
- Suggests that significant PJS lesions can occur before the recommended surveillance age of 10.
- Advocates for the development of specific diagnostic and management guidelines for pediatric PJS.
- Highlights the potential need for advanced diagnostic tools like video-capsule endoscopy in young patients.
Abstract:
Peutz-Jeghers syndrome is a rare autosomal dominant condition, characterized by gastrointestinal polyposis, mucocutaneous pigmentation and high risk of neoplasia in multiple organs. At pediatric age, major clinical impact is related to complications associated to intestinal polyps, but neoplasic risk isn't negligible. Though clinical surveillance is recommended since the age of 10 years, relevant lesions may occur before that age. Conventional radiology and endoscopy have recognized limitations at this age group, and new diagnostic and intervention tools, such as video-capsule and per-operative enteroscopy, are not yet widely used. We present 5 pediatric cases (age under 10 years) with diversity of gastrointestinal expression (including one case with histologic evidence of dysplasia in a large colonic polyp), emphasizing the need of specific guidelines concerning young children.
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