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Malignancy and mortality in pediatric patients with inflammatory bowel disease: a multinational study from the porto
Lissy de Ridder1, Dan Turner, David C Wilson
11Department of Pediatric Gastroenterology, The Erasmus MC Sophia Children's Hospital, Rotterdam, The Netherlands; 2Department of Pediatric Gastroenterology, Shaare Zedek Medical Center, The Hebrew University of Jerusalem, Jerusalem, Israel; 3Department of Pediatric Gastroenterology, Child Life and Health, University of Edinburgh, Edinburgh, United Kingdom; 4Department of Pediatric Gastroenterology, Dr. v. Hauner Children's Hospital, University of Munich Medical Centre, Munich, Germany; 5Department of Pediatric Gastroenterology, Unidad para el Cuidado Integral de la Enfermedad Inflamatoria Intestinal Pediátrica, Sección de Gastroenterología, Hepatología y Nutrición Pediatrica, Hospital Sant Joan de Déu, Barcelona, Spain; 6Department of Pediatric Gastroenterology, Centre for Clinical Research, Västmanland Hospital, Västerås; 7Karolinska Institutet, Stockholm, Sweden; 8Department of Pediatric Gastroenterology, Bristol Royal Hospital for Children, Bristol, United Kingdom; 9Department of Pediatrics, Gastroenterology and Nutrition, Polish-American Children's Hospital, Jagiellonian University Medical College, Cracow, Poland; 10Pediatric Day Care Unit, Department of Pediatrics, Bnai Zion Medical Center, Haifa, Israel; 11First Department of Paediatrics, Athens University, Athens, Greece; 12Department of Pediatric Gastroenterology, Second Faculty of Medicine, Charles University and University Hospital Motol, Prague, Czech Republic; 13Department of Pediatric Gastroenterology, "Iuliu Hatieganu" University of Medicine and Pharmacy, Second Pediatric Clinic, Children's Emergency Hospital, Cluj-Napoca, Romania; 14Pediatric Gastroenterology and Liver Unit, "La Sapienza" University, Rome, Italy; 15Department of Pediatric Gastroenterology, Semmelweis University, Budapest, Hungary; 16Department of Pediatric Gastroenterology, Université Paris Descartes, Sorbonne Paris Cité, APHP, Hôpital Necker Enfants Malades, Paris, France; 17Department of Pediatric Gastroenterology, Refer
Insights
Cancer and mortality are rare but significant concerns in pediatric inflammatory bowel disease (IBD). Infections are the leading cause of death, followed by cancer, especially in patients on multiple immunosuppressants.
Area of Science:
- Pediatric Gastroenterology
- Inflammatory Bowel Disease (IBD) Research
- Pediatric Oncology and Mortality Studies
Background:
- Pediatric inflammatory bowel disease (IBD) severity and intensive treatments may elevate cancer and mortality risks.
- Limited evidence exists on the extent of malignancy and mortality in pediatric IBD patients.
- The Porto Pediatric IBD working group initiated a survey to address this knowledge gap.
Purpose of the Study:
- To conduct a multinational survey on cancer and mortality in pediatric IBD patients.
- To assess the incidence and causes of cancer and death in children diagnosed with IBD.
Main Methods:
- A survey was distributed to pediatric gastroenterologists across 20 European countries and Israel.
- Pediatric gastroenterologists retrospectively reported cancer and/or mortality data for pediatric IBD patients.
- Data collection covered the period from IBD onset through 2006-2011.
Main Results:
- 18 cancers and 31 deaths were identified in 44 pediatric IBD patients (median age 10.0 years).
- Infectious causes accounted for the most deaths (n=14), followed by cancer (n=5) and uncontrolled IBD (n=4).
- Hematopoietic tumors were the most common malignancies (n=11), including hepatosplenic T-cell lymphoma and EBV-associated lymphomas.
Conclusions:
- While rare, cancer and mortality rates in pediatric IBD are not insignificant.
- Mortality is predominantly linked to infections, particularly with multiple immunosuppressive agents, then cancer and disease activity.
- At least 6 lymphomas suggest a potential association with treatment regimens.
Background:
The combination of the severity of pediatric-onset inflammatory bowel disease (IBD) phenotypes and the need for intense medical treatment may increase the risk of malignancy and mortality, but evidence regarding the extent of the problem is scarce. Therefore, the Porto Pediatric IBD working group of ESPGHAN conducted a multinational-based survey of cancer and mortality in pediatric IBD.
Methods:
A survey among pediatric gastroenterologists of 20 European countries and Israel on cancer and/or mortality in the pediatric patient population with IBD was undertaken. One representative from each country repeatedly contacted all pediatric gastroenterologists from each country for reporting retrospectively cancer and/or mortality of pediatric patients with IBD after IBD onset, during 2006-2011.
Results:
We identified 18 cases of cancers and/or 31 deaths in 44 children (26 males) who were diagnosed with IBD (ulcerative colitis, n = 21) at a median age of 10.0 years (inter quartile range, 3.0-14.0). Causes of mortality were infectious (n = 14), cancer (n = 5), uncontrolled disease activity of IBD (n = 4), procedure-related (n = 3), other non-IBD related diseases (n = 3), and unknown (n = 2). The most common malignancies were hematopoietic tumors (n = 11), of which 3 were hepatosplenic T-cell lymphoma and 3 Ebstein-Barr virus-associated lymphomas.
Conclusions:
Cancer and mortality in pediatric IBD are rare, but cumulative rates are not insignificant. Mortality is primarily related to infections, particularly in patients with 2 or more immunosuppressive agents, followed by cancer and uncontrolled disease. At least 6 lymphomas were likely treatment-associated by virtue of their phenotype.
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