Observational Study
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Dec 21, 2019; 25(47): 6857-6865
Published online Dec 21, 2019. doi: 10.3748/wjg.v25.i47.6857
Direct costs of carcinoid syndrome diarrhea among adults in the United States
Arvind Dasari, Vijay N Joish, Raul Perez-Olle, Samyukta Dharba, Kavitha Balaji, Daniel M Halperin
Arvind Dasari, Daniel M Halperin, Department of Gastrointestinal Medical Oncology, The University of Texas MD Anderson Cancer Center, Houston, TX 77030, United States
Vijay N Joish, Raul Perez-Olle, Samyukta Dharba, Kavitha Balaji, Lexicon Pharmaceuticals, Inc., The Woodlands, TX 77381, United States
Author contributions: Dasari A, Joish VN, Perez-Olle R, Dharba S, Balaji K and Halperin DM designed the research; Joish VN and Dharba S analyzed the data; Frimpter J wrote the paper and Dasari A, Joish VN, Perez-Olle R, Dharba S, Balaji K and Halperin DM critically revised all aspects of the manuscript and approved the final version for submission.
Institutional review board statement: We conducted a retrospective cohort study using the IBM® MarketScan® Database, a commercial health insurance claims database for employer-insured beneficiaries in the United States. The database is fully compliant with the Health Insurance Portability and Accountability Act and meets the criteria for a limited-use dataset. Since the patient and provider data included in this analysis were fully de-identified, this study was exempt from the Institutional Review Board review.
Informed consent statement: This study involved analyses of a Health Insurance Portability and Accountability Act-compliant secondary database, the IBM® MarketScan® Database, thus no informed consent was feasible or required.
Conflict-of-interest statement: This study was sponsored by Lexicon Pharmaceuticals, Inc., 8800 Technology Forest Place, The Woodlands, TX 77381, United States. Vijay N Joish, Samkyuta Dharba and Kavitha Balaji are employees of Lexicon Pharmaceuticals, Inc., and Raul Perez-Olle was an employee of Lexicon Pharmaceuticals, Inc. during the conduct of the study and development of the manuscript; they may own common stock or may have been granted stock options or other equity incentive awards. Arvind Dasari and Daniel M Halperin are employees of The University of Texas MD Anderson Cancer Center who received consulting fees from Lexicon Pharmaceuticals, Inc. for their participation and contributions to this study.
Data sharing statement: The statistician for this study, Samyukta Dharba, conducted all statistical analyses using a Health Insurance Portability and Accountability Act-compliant secondary database, the IBM® MarketScan® Database.
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 which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Vijay N Joish, PhD, Executive Director, Epidemiology and Outcomes Research, Lexicon Pharmaceuticals, Inc., 110 Allen Road, Basking Ridge, NJ 07920, United States. vjoish@lexpharma.com
Telephone: +1-908-3604782
Received: June 15, 2019
Peer-review started: June 17, 2019
First decision: August 3, 2019
Revised: November 20, 2019
Accepted: November 23, 2019
Article in press: November 23, 2019
Published online: December 21, 2019
Research background

Carcinoid syndrome (CS) in patients with neuroendocrine tumors has been shown to bear substantial economic costs to patients and healthcare systems; however, the incremental burden of CS diarrhea (CSD) has not been well characterized in the literature. Since patients with CSD are most often of working age, it is important to understand the direct costs of CSD from a population health management perspective. This study aims to provide detail and context related to the direct costs of CSD on insured working age adults in the United States.

Research motivation

Quantifying the economic burden of CSD in this population is important for setting healthcare priorities and allocating healthcare resources in an appropriate and efficient manner. Patients with CSD may be underserved due to a lack of information and insight regarding the burden of CSD overall, and payers need to understand the scope of economic burden of CSD in order to design effective policies and programs. Future research may validate these findings and apply similar methods to additional health system configurations such as integrated delivery networks, single payer systems, and other approaches to population health management found worldwide.

Research objectives

We aimed to quantify the incremental economic burden of CSD compared with patients who had CS but no CSD. The differentiation of CSD within the broader scope of CS costs is important for resource allocation and policy decisions, and has not been well studied. This objective allows population health managers to more clearly examine the additive costs that are specific to CSD in this patient population, where such discrimination of costs was not previously possible. Future research may build upon these insights and expand them to include indirect costs such as work productivity burden and other important factors.

Research methods

We conducted a retrospective study of CS patients with and without CSD as identified in the IBM® MarketScan® Database between 2014–2016, including the Medicare Supplemental Coordination of Benefits database. Patients had to have at least 1 medical claim for CS and continuous health plan enrollment for at least 12 mo prior to their first CS diagnosis, and for at least 30 d after. We excluded patients with documented claims for acromegaly, and those who participated in a clinical trial during the study period. Measures of healthcare resource use and costs were compared between patients with and without CSD, including Emergency Department (ED) visits, hospital admissions and length of stay, physician office visits, outpatient services, and prescription claims, using univariate and multivariate analyses to evaluate associations of CSD with healthcare resource use and costs, controlling for baseline characteristics. The methods applied in this analysis allowed us to distinguish the direct costs among patients with CS “only” from their peers had CS and CSD. This approach allowed us to characterize the additive, or incremental healthcare-related costs of CSD in the context of a patient population that was as similar as possible to those with CSD.

Research results

Our study identified 4043 patients with CS to be included in the analysis, 1352 with CSD and 2691 with CS only. Baseline demographic and clinical characteristics were similar between groups except that patients with CSD were older, had more comorbidities, and received more somatostatin analog therapy at baseline. Overall, patients with CSD required more healthcare resources and incurred higher costs than their peers with CS only. In particular, patients with CSD had more hospitalizations (44% vs 25%) and Emergency Department visits (55% vs 31%). When adjusted for baseline demographic and clinical characteristics, patients with CSD had higher mean healthcare resource use across all components of care, including hospital admissions (0.8 vs 0.4) and the mean length of stays (5.3 d vs 2.3 d), physician office visits (16.3 vs 12.0), Emergency Department visits (1.3 vs 0.6), outpatient services (34.2 vs 23.1), and prescription claims (41.5 vs 30.5). The total adjusted annual healthcare costs per patient were 50% higher (+ $23865) among those with CSD, driven by outpatient services (+ 56%), prescriptions (+ 48%), ED visits (+ 26%), physician office visits (+ 21%), and hospital admissions (+ 11%). These findings provide quantifiable differences in direct costs between patients with CS “only” and their peers who also have CSD. The increased costs observed across all avenues of care are indicative of the increased burden of CSD on patients and the healthcare resources needed to provide adequate care for this disruptive and damaging condition. Further research may validate these findings and investigate similar incremental costs in other healthcare settings.

Research conclusions

This study demonstrated that the costs of managing CSD are greater than those related to CS alone among insured working age adults in the United States, allowing population health managers to more intimately understand the incremental economic burden of CSD and to develop policies and programs accordingly. The methods applied in this study may be replicated or adopted to other data sources and healthcare settings to continue to characterize the additive costs of CSD in this predominantly working age population. This study provides a clear illustration of costs from the perspective of the employers and insurers, which is essential to effective policy and practice for this relatively young, active patient population. Timely identification and appropriate management of CSD may not only alleviate the clinical and humanistic burden of CSD to these patients, but may also reduce the economic burden of CSD to payers and population health managers.

Research perspectives

Patients with neuroendocrine tumors and CS require substantial healthcare resources to manage this condition, which are greatest among those who also have CSD. Supporting the timely identification and management of CSD should be a priority for population health managers, as this condition has been shown to negatively impact patients, employers, and the healthcare system. Future research may validate and extend these findings, and investigate indirect costs such as the impact of CS and CSD on quality of life, work productivity, and caregivers.