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Clinical Characteristics and Long-term Outcomes of Children With Fibrosing Pancreatitis
Isabelle Scheers1,2, Govind B Chavhan3, Rose Chami4
1Division of Gastroenterology, Hepatology and Nutrition, Hospital for Sick Children, University of Toronto, Toronto, Ontario, Canada.
Insights
Pediatric fibrosing pancreatitis (P-FP) shares features with autoimmune pancreatitis (AIP). Long-term outcomes are generally good, but monitoring for exocrine insufficiency and other immune diseases is recommended.
Area of Science:
- Gastroenterology
- Pediatric Gastroenterology
- Immunology
Background:
- Fibrosing pancreatitis (FP) and autoimmune pancreatitis (AIP) share clinical similarities but are not definitively linked.
- Pediatric fibrosing pancreatitis (P-FP) requires further investigation into its characteristics and long-term prognosis.
Purpose of the Study:
- To evaluate AIP criteria in a cohort of pediatric FP patients.
- To investigate the clinical features, management, and long-term outcomes of pediatric FP.
Main Methods:
- Retrospective analysis of 14 pediatric FP patients from Toronto and 42 from literature review.
- Comparison with pediatric AIP (P-AIP) cases.
- Follow-up assessment of Toronto P-FP patients via questionnaire.
Main Results:
- Jaundice and abdominal pain were common presenting symptoms in P-FP.
- Imaging revealed enlarged pancreas head and bile duct narrowing; histopathology showed fibrosis.
- Elevated IgG4 in serum was observed in 25% of patients; long-term complications included exocrine insufficiency and atrophy, but no disease relapse or diabetes.
Conclusions:
- Pediatric FP presents similarly to some P-AIP cases, particularly those with jaundice.
- Long-term outcomes for P-FP are generally favorable, irrespective of intervention.
- Ongoing monitoring is advised due to potential development of exocrine insufficiency and other immune-mediated diseases.
Objectives:
Fibrosing pancreatitis (FP) shares clinical features with autoimmune pancreatitis (AIP), although both entities have not been definitely linked. This study aimed to assess the presence of AIP criteria in an historic FP patient cohort and investigate the clinical features, management, and long-term outcomes of pediatric FP (P-FP).
Methods:
Clinical data of 14 P-FP patients from Toronto and 42 P-FP cases from a literature review were collected and compared to pediatric AIP (P-AIP). Toronto P-FP patients were recontacted to assess their current health status using a brief questionnaire.
Results:
Jaundice and abdominal pain were the symptoms at presentation in 44 of 56 (79%) and 50 of 56 (89%) P-FP patients, respectively. Common findings on cross sectional imaging were an enlarged pancreas head with narrowing of the distal common bile duct (51/54, 94%). Histopathology mainly showed gland fibrosis (39/39, 100%). Three of twelve (25%) P-FP patients had elevated IgG4 in serum. None of the patients were treated with corticosteroids, but some underwent surgical or endoscopic intervention. Toronto patients were followed for a median of 13.6 years (interquartile range: 2.9-22.8). Complications during follow-up included exocrine pancreatic insufficiency (3/14, 21%) and pancreatic gland atrophy (5/13, 38%); but none of the patients had disease relapse or developed diabetes type 3c. Five (5/14, 36%) patients developed other immune-mediated diseases over time.
Conclusions:
Clinical features of patients with P-FP resembled those recently described in a subgroup of P-AIP presenting with jaundice. Long-term outcome of these patients is generally good, with or without invasive interventions. As some patients may develop exocrine pancreatic insufficiency and/or other immune-mediated diseases, ongoing clinical monitoring is recommended.
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