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©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
Cohort study to assess geographical variation in cholangiocarcinoma treatment in England
Sophie Jose, Amy Zalin-Miller, Craig Knott, Lizz Paley, Daniela Tataru, Helen Morement, Mireille B Toledano, Shahid A Khan
Sophie Jose, Amy Zalin-Miller, Craig Knott, Health Data Analysis, Health Data Insight CIC, Cambridge CB21 5XE, United Kingdom
Sophie Jose, Amy Zalin-Miller, Craig Knott, Lizz Paley, Daniela Tataru, National Disease Registration Service, National Health Service England, London SE1 8UG, United Kingdom
Helen Morement, Department of Executive, AMMF-The Cholangiocarcinoma Charity, Essex CM24 1QW, United Kingdom
Mireille B Toledano, MRC Centre for Environment and Health, Imperial College London, London SW7 2BX, United Kingdom
Mireille B Toledano, Mohn Centre for Children's Health and Wellbeing, Imperial College London, London SW7 2BX, United Kingdom
Shahid A Khan, Liver Unit, Division of Digestive Diseases, Imperial College London, London SW7 2BX, United Kingdom
Author contributions: Jose S, Knott C, Khan SA, Morement H and Zalin-Miller A designed the study; Paley L and Toledano MB advised on the study design; Jose S, Zalin-Miller A and Knott C had full access to the underlying data in the study; all authors contributed to the interpretation of results; Jose S and Khan SA drafted the manuscript; Knott C, Morement H, Paley L, Tataru D, Toledano MB and Zalin-Miller A reviewed and revised the manuscript; all authors approved the final version of the manuscript had access to the study data and accept responsibility to submit for publication.
Supported by AMMF; National Disease Registration Service, National Health Service England.
Institutional review board statement: The study was reviewed and approved for publication by our Institutional Reviewer.
Informed consent statement: NHS England and has a special legal instruction to collect patient data without needing informed consent. This instruction is granted under section 254 of the Health and Social Care Act 2012.
Conflict-of-interest statement: Dr Jose reports grants from AMMF-The Cholangiocarcinoma Charity, during the conduct of the study.
Data sharing statement: The data that support the findings of this study are available from NHS England. Restrictions apply to the access and use of the data used to undertake this study. A data dictionary is available at
https://digital.nhs.uk/ndrs/data/access-to-data.
STROBE statement: The authors have read the STROBE Statement-checklist of items, and the manuscript was prepared and revised according to the STROBE Statement-checklist of items.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See:
https://creativecommons.org/Licenses/by-nc/4.0/ Corresponding author: Sophie Jose, PhD, Senior Statistician, Health Data Analysis, Health Data Insight CIC, CPC4 Capital Park, Cambridge CB21 5XE, United Kingdom.
sophie.jose@nhs.net
Received: August 7, 2023
Peer-review started: August 7, 2023
First decision: September 1, 2023
Revised: September 22, 2023
Accepted: October 30, 2023
Article in press: October 30, 2023
Published online: December 15, 2023
Processing time: 128 Days and 17.6 Hours
BACKGROUND
Outcomes for cholangiocarcinoma (CCA) are extremely poor owing to the complexities in diagnosing and managing a rare disease with heterogenous sub-types. Beyond curative surgery, which is only an option for a minority of patients diagnosed at an early stage, few systemic therapy options are currently recommended to relieve symptoms and prolong life. Stent insertion to manage disease complications requires highly specialised expertise. Evidence is lacking as to how CCA patients are managed in a real-world setting and whether there is any variation in treatments received by CCA patients.
AIM
To assess geographic variation in treatments received amongst CCA patients in England.
METHODS
Data used in this cohort study were drawn from the National Cancer Registration Dataset (NCRD), Hospital Episode Statistics and the Systemic Anti-Cancer Therapy Dataset. A cohort of 8853 CCA patients diagnosed between 2014-2017 in the National Health Service in England was identified from the NCRD. Potentially curative surgery for all patients and systemic therapy and stent insertion for 7751 individuals who did not receive surgery were identified as three end-points of interest. Linear probability models assessed variation in each of the three treatment modalities according to Cancer Alliance of residence at diagnosis, and for socio-demographic and clinical characteristics at diagnosis.
RESULTS
Of 8853 CCA patients, 1102 (12.4%) received potentially curative surgery. The mean [95% confidence interval (CI)] percentage-point difference from the population average ranged from -3.96 (-6.34 to -1.59)% to 3.77 (0.54 to 6.99)% across Cancer Alliances in England after adjustment for patient sociodemographic and clinical characteristics, showing statistically significant variation. Amongst 7751 who did not receive surgery, 1542 (19.9%) received systemic therapy, with mean [95%CI] percentage-point difference from the population average between -3.84 (-8.04 to 0.35)% to 9.28 (1.76 to 16.80)% across Cancer Alliances after adjustment, again showing the presence of statistically significant variation for some regions. Stent insertion was received by 2156 (27.8%), with mean [95%CI] percentage-point difference from the population average between -10.54 (-12.88 to -8.20)% to 13.64 (9.22 to 18.06)% across Cancer Alliances after adjustment, showing wide and statistically significant variation from the population average. Half of 8853 patients (n = 4468) received no treatment with either surgery, systemic therapy or stent insertion.
CONCLUSION
Substantial regional variation in treatments received by CCA patients was observed in England. Such variation could be due to differences in case-mix, clinical practice or access to specialist expertise.
Core Tip: Outcomes for cholangiocarcinoma (CCA) are extremely poor, with late presentation meaning curative surgery is not an option for many. Systemic therapies to prolong life are limited and stent insertion for disease management is complex. In a national cohort, treatments received (surgery, systemic therapy, stent insertion) by CCA patients across geographic areas were investigated. Half of patients did not receive any of the treatments considered. The proportion that received treatments significantly varied across England. These data provide novel evidence of low and varied treatment rates for CCA patients, warranting further investigation by healthcare providers to try to improve outcomes and reduce inequality.
