Strategy for Management of Patients With Hereditary Cancer Syndromes (HCS) in a Rural Environment (LINC)

This study aims to improve cancer prevention and surveillance adherence in patients with Hereditary Cancer Syndromes (HCS), particularly those living in rural areas. The study will evaluate whether enrolling HCS patients in a longitudinal clinical program with individualized care plans and regular follow-up improves adherence to guideline-recommended cancer screening and risk-reduction strategies. Secondary aims include assessing the program's impact on patient distress and perceived care coordination. The study will enroll 200 adults with known pathogenic germline mutations who were previously seen at the UVM Medical Center genetics clinic. Participants will complete surveys at baseline, 12, and 24 months to assess adherence, distress, and care coordination. Findings from this study will inform future efforts to reduce gaps in hereditary cancer care delivery, especially for rural populations.

Study Overview

• Status: Enrolling by invitation
• Conditions: Lynch Syndrome; Hereditary Cancer Syndromes; BRCA1 Hereditary Breast and Ovarian Cancer Syndrome
• Intervention / Treatment: Behavioral: Longitudinal Cancer Genetics Follow-Up Program

Detailed Description

This single-arm, prospective, interventional study will assess the feasibility and impact of a structured longitudinal follow-up program for patients with Hereditary Cancer Syndromes (HCS). Eligible participants will have a known pathogenic germline variant in a cancer risk gene identified by a CLIA-approved lab at least one year prior to enrollment. All participants will be seen at baseline and followed clinically for two years, with additional visits as clinically indicated. Each participant will receive an individualized care plan developed by cancer genetics providers and supported by annual follow-up visits.

The primary objective is to assess whether participation in this program increases adherence to National Comprehensive Cancer Network (NCCN) guideline-recommended cancer screening and prevention strategies (e.g., surveillance imaging, colonoscopy, prophylactic surgery). Secondary objectives include evaluating changes in participant distress and perceived care coordination over time, and examining how rurality (defined using RUCA codes) affects outcomes.

Participants will complete three study instruments at baseline, 12-month, and 24-month follow-up visits:

A gene-specific adherence survey administered by the genetics team,

The Multidimensional Impact of Cancer Risk Assessment (MICRA) questionnaire to assess distress,

The Care Coordination Index (CCI) to evaluate patient-perceived coordination of care.

A retrospective chart review of previously seen HCS patients will also be conducted to estimate baseline adherence rates and support feasibility assessments. Statistical analyses will use generalized probit and linear models to evaluate changes over time and assess effect modification by demographic and clinical variables such as age, sex, gene mutation, and rurality.

This study will generate critical pilot data to inform future controlled studies and improve access to high-quality cancer prevention care, particularly for underserved rural populations.

• Study Type: Interventional
• Enrollment (Estimated): 200
• Phase: Not Applicable

Contacts and Locations

Study Locations

• United States
  • Vermont
    • Burlington, Vermont, United States, 05401
      • University of Vermont Medical Center

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: Adult; Older Adult
• Accepts Healthy Volunteers: No

Description

Inclusion Criteria:

• Patients of all genders must be ≥ 18 years of age.
• Patients must have a known pathogenic germline variant in a cancer risk gene that was identified by a CLIA-approved lab more than one year ago.
• Patients must be able to accurately provide self-report data (i.e., per clinical judgment, cognitive function is intact).
• Patients must be able to complete questionnaires in English.
• Patients must have the ability to provide informed consent.

Exclusion Criteria:

- Patients who tested positive for a germline pathogenic variant associated with cancer risk < 1 year ago are not eligible.

Study Plan

How is the study designed?

Design Details

• Primary Purpose: Health Services Research
• Allocation: N/A
• Interventional Model: Single Group Assignment
• Masking: None (Open Label)

Number of Arms

1

Arms and Interventions

Participant Group / Arm

Intervention / Treatment

Experimental: Longitudinal Cancer Genetics Follow-Up Program; Participants in this arm will be enrolled in a longitudinal cancer genetics follow-up program designed for individuals with known hereditary cancer syndromes (HCS). The intervention includes scheduled clinical visits with a cancer genetics physician and, as needed, a genetic counselor at baseline, 12 months, and 24 months. Participants will receive individualized care plans summarizing surveillance and prevention recommendations. Adherence, distress (MICRA), and care coordination (CCI) will be assessed through surveys administered at each visit.

Behavioral: Longitudinal Cancer Genetics Follow-Up Program; Participants will be enrolled in a structured, two-year longitudinal follow-up program designed for individuals with known hereditary cancer syndromes (HCS). The program includes baseline, 12-month, and 24-month clinic visits with a cancer genetics physician and, as needed, a genetic counselor. During each visit, participants will receive a personalized care plan outlining guideline-based cancer prevention and surveillance recommendations. Participants will also complete adherence surveys with their provider, and independently complete the MICRA and Care Coordination Index (CCI) surveys to assess distress and care coordination. Visits may be conducted in person or via televideo, based on clinical need and participant preference.

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Adherence to Cancer Prevention and Surveillance Guidelines	Proportion of participants who are adherent to National Comprehensive Cancer Network (NCCN) guideline-recommended cancer risk-reducing strategies-including surveillance imaging, colonoscopy, chemoprevention, and prophylactic surgeries-measured at baseline, 12 months, and 24 months. Adherence will be determined through a gene-specific adherence survey completed during clinic visits and supported by clinical documentation.	Baseline, 12 months, and 24 months

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Change in Participant Distress Level as Measured by the MICRA Questionnaire	This outcome will assess change in participant distress related to hereditary cancer risk using the validated Multidimensional Impact of Cancer Risk Assessment (MICRA) questionnaire. The MICRA provides a total distress score and subscale scores for positive experiences, intrusive thoughts, and distress. Participants complete the MICRA at baseline, 12-month, and 24-month follow-up visits. Scores will be compared over time to evaluate the impact of enrollment in a longitudinal cancer genetics program on emotional distress. Unit of Measure: Mean change in MICRA total distress score.	2 years
Change in Perceived Care Coordination	This outcome will assess change in participants' perceived care coordination over time using the validated Care Coordination Index (CCI). The CCI is a participant-reported measure that evaluates multiple domains of care coordination. Scores range from 0 to 100, with higher scores indicating better perceived care coordination. Participants will complete the CCI at baseline, 12-month, and 24-month follow-up visits. Changes in scores will be analyzed to determine whether enrollment in a formal longitudinal follow-up program improves care integration. Unit of Measure: Mean change in Care Coordination Index (CCI) score Assessment Tool: Care Coordination Index (CCI) - participant-reported questionnaire	2 years

Collaborators and Investigators

• Sponsor: University of Vermont Medical Center

Study record dates

Study Major Dates

• Study Start (Actual): December 29, 2023
• Primary Completion (Estimated): December 1, 2027
• Study Completion (Estimated): December 1, 2027

Study Registration Dates

• First Submitted: May 8, 2025
• First Submitted That Met QC Criteria: January 27, 2026
• First Posted (Actual): February 2, 2026

Study Record Updates

• Last Update Posted (Actual): February 2, 2026
• Last Update Submitted That Met QC Criteria: January 27, 2026
• Last Verified: January 1, 2026

More Information

Terms related to this study

• Keywords: Rural Health; Lynch Syndrome; Hereditary Cancer Syndromes
• Additional Relevant MeSH Terms: Neoplasms by Site; Neoplasms; Genetic Diseases, Inborn; Metabolic Diseases; Intestinal Diseases; Gastrointestinal Neoplasms; Digestive System Neoplasms; Digestive System Diseases; Gastrointestinal Diseases; Colorectal Neoplasms; Intestinal Neoplasms; Colonic Diseases; DNA Repair-Deficiency Disorders; Congenital, Hereditary, and Neonatal Diseases and Abnormalities; Nutritional and Metabolic Diseases; Colorectal Neoplasms, Hereditary Nonpolyposis; Neoplastic Syndromes, Hereditary
• Other Study ID Numbers: STUDY00002507/UVMCC2204

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: NO
• IPD Plan Description: Anonymized patient related data will be incorporated into publications and will be made available to other investigators at reasonable request.

Drug and device information, study documents

• Studies a U.S. FDA-regulated drug product: No
• Studies a U.S. FDA-regulated device product: No