Clinical Characteristics, Associated Malignancies and Management of Primary Sclerosing Cholangitis in Inflammatory Bowel Disease Patients: A Multicentre Retrospective Cohort Study

Author:

Guerra Ivan1,Bujanda Luis23,Castro Jesús4,Merino Olga5,Tosca Joan6,Camps Blau7,Gutiérrez Ana38,Gordillo Ábalos Jordi9,de Castro Luisa10,Iborra Marisa311,Carbajo Ana Y12,Taxonera Carlos13,Rodríguez-Lago Iago14,Mesonero Francisco15,de Francisco Ruth16,Gómez-Gómez Gonzalo J17,Chaparro María318,Tardillo Carlos A19,Rivero Montserrat20,Algaba Alicia1,Martín Arranz Eduardo21,Cañete Fiorella322,Vicente Raquel23,Sicilia Beatriz24,Antolín Beatriz25,Prieto Vanessa26,Márquez Lucía27,Benítez José M28,Camo Patricia29,Piqueras Marta30,Gargallo Carla J31,Hinojosa Esther32,Huguet José M33,Pérez Calle José L34,Van Domselaar Manuel35,Rodriguez Cristina36,Calvet Xavier337,Muñoz-Villafranca Carmen38,García-Sepulcre Mariana F39,Munoz-Garrido Patricia23,Fernández-Clotet Agnès4,Gómez Irwin Laura5,Hernández Sherly6,Guardiola Jordi740,Sempere Laura8,González Muñoza Carlos9,Hernández Vicent10,Beltrán Belén311,Barrio Jesús12,Alba Cristina13,Moraleja Irene14,López-Sanromán Antonio15,Riestra Sabino16,Martínez Montiel Pilar17,Garre Ana318,Arranz Laura19,García María José20ORCID,Martín Arranz María Dolores21,Corsino Pilar23,Arias Lara24,Fernández-Salazar Luis25,Fernández-Pordomingo Alejandra26,Andreu Montserrat27,Iglesias Eva28,Ber Yolanda29,Mena Raquel30,Arroyo Villarino María Teresa31,Mora María32,Ruiz Lucía33,López-Serrano Pilar34,Blazquez Isabel35,Villoria Albert336,Fernández María37,Bermejo Fernando1,Banales Jesus M2341,Domènech Eugeni322,Gisbert Javier P318,

Affiliation:

1. Hospital Universitario de Fuenlabrada and Instituto de Investigación del Hospital Universitario La Paz, IdiPaz, Madrid, Spain

2. Hospital Donostia/Instituto Biodonostia, Universidad del País Vasco UPV/EHU, Donostia-San Sebastián, Spain

3. Centro de Investigación Biomédica en Red de Enfermedades Hepáticas y Digestivas (CIBERehd), Spain

4. Hospital Clinic de Barcelona, Barcelona, Spain

5. Hospital Universitario de Cruces, Barakaldo, Vizcaya, Spain

6. Hospital Universitario Clínico de Valencia, Department of Medicine, University of Valencia, Valencia, Spain

7. Hospital Universitari de Bellvitge, Institut d’Investigació Biomèdica de Bellvitge (IDIBELL), l’Hospitalet de Llobregat, Spain

8. Hospital General Universitario de Alicante, Alicante, Spain

9. Hospital de la Santa Creu i Sant Pau, Barcelona, Spain

10. Complexo Hospitalario Universitario de Vigo, Instituto de Investigación Biomédica. Estructura Organizativa de Xestión Integrada de Vigo, Vigo, Spain

11. Hospital Universitario y Politécnico de La Fe, Valencia, Spain

12. Hospital Universitario Rio Hortega, Valladolid

13. Hospital Clínico San Carlos and Instituto de Investigación del Hospital Clínico San Carlos (IdISSC), Madrid, Spain

14. Hospital de Galdakao, Galdakao, Vizcaya, Spain

15. Hospital Universitario Ramón y Cajal, Madrid, Spain

16. Hospital Universitario Central De Asturias, Instituto de Investigación Sanitaria del Principado de Asturias (ISPA), Oviedo, Spain

17. Hospital 12 de Octubre, Madrid, Spain

18. Hospital Universitario de La Princesa, Instituto de Investigación Sanitaria Princesa (IIS-IP), Universidad Autónoma de Madrid, Madrid, Spain

19. Hospital Universitario Nuestra Señora Candelaria, Santa Cruz De Tenerife, Spain

20. Hospital Universitario Marqués de Valdecilla and IDIVAL, Santander, Spain

21. Hospital Universitario La Paz and Instituto de Investigación del Hospital Universitario La Paz, IdiPaz, Madrid

22. Hospital Universitari Germans Trias i Pujol, Badalona, Spain

23. Hospital Universitario Miguel Servet, Zaragoza, Spain

24. Hospital Universitario de Burgos, Burgos, Spain

25. Hospital Clínico Universitario de Valladolid, Valladolid, Spain

26. Hospital Universitario de Salamanca, Salamanca, Spain

27. Parc de Salut Mar, Barcelona, Spain

28. Hospital Universitario Reina Sofía and Instituto Maimónides de Investigación Biomédica de Córdoba (IMIBIC), Córdoba, Spain

29. Hospital General San Jorge, Huesca, Spain

30. Consorci Sanitari Terrassa, Terrassa, Spain

31. Hospital Clínico Universitario “Lozano Blesa” and Aragón Health Research Institute (IIS Aragón), Zaragoza, Spain

32. Hospital de Manises, Valencia, Spain

33. Hospital General Universitario de Valencia, Valencia, Spain

34. Hospital Universitario Fundación Alcorcón, Madrid, Spain

35. Hospital Universitario de Torrejón and Universidad Francisco de Vitoria, Madrid, Spain

36. Complejo hospitalario de Navarra, Pamplona, Spain

37. Corporació Sanitària Universitària Parc Taulí, Universitat Autònoma de Barcelona, Sabadell, Spain

38. Hospital Universitario de Basurto, Bilbao, Spain

39. Hospital General Universitario Elche, Alicante, Spain

40. Universitat de Barcelona, Spain

41. Ikerbasque (Basque Foundation for Sciencies), Bilbao, Spain

Abstract

Abstract Background and Aims Primary sclerosing cholangitis [PSC] is usually associated with inflammatory bowel disease [IBD]. An increased risk of malignancies, mainly colorectal cancer [CRC] and cholangiocarcinoma [CCA], has been reported in PSC-IBD patients. Our aim was to determine the clinical characteristics and management of PSC in IBD patients, and the factors associated with malignancies. Methods PSC-IBD patients were identified from the Spanish ENEIDA registry of GETECCU. Additional data were collected using the AEG-REDCap electronic data capture tool. Results In total, 277 PSC-IBD patients were included, with an incidence rate of 61 PSC cases per 100 000 IBD patient-years, 69.7% men, 67.5% ulcerative colitis and mean age at PSC diagnosis of 40 ± 16 years. Most patients [85.2%] were treated with ursodeoxycholic acid. Liver transplantation was required in 35 patients [12.6%] after 79 months (interquartile range [IQR] 50–139). It was more common in intra- and extrahepatic PSC compared with small-duct PSC (16.3% vs 3.3%; odds ratio [OR] 5.7: 95% confidence interval [CI] = 1.7–19.3). The incidence rate of CRC since PSC diagnosis was 3.3 cases per 1000 patient-years [95% CI = 1.9–5.6]. Having symptoms of PSC at PSC diagnosis was the only factor related to an increased risk of CRC after IBD diagnosis [hazard ratio= 3.3: 95% CI = 1.1–9.9]. CCA was detected in seven patients [2.5%] with intra- and extrahepatic PSC, with median age of 42 years [IQR 39–53], and presented a lower life expectancy compared with patients without CCA and patients with or without CRC. Conclusions PSC-IBD patients with symptoms of PSC at PSC diagnosis have an increased risk of CRC. CCA was only diagnosed in patients with intra- and extrahepatic PSC and was associated with poor survival.

Publisher

Oxford University Press (OUP)

Subject

Gastroenterology,General Medicine

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