Resilience and Coping in a Rare Skeletal Disease Population to Face Coronavirus (COVID-19) Outbreak Distress: a Longitudinal Study (RELOAD)

Exploring Resilience and Coping Strategies of Young Population in Response to Severe Acute Respiratory Syndrome Corona Virus 2 (SARS-CoV-2) Outbreak: a Longitudinal Study in a Cohort of Patients Affected by Rare Skeletal Disorders.

In the COVID-19 outbreak context, people living with rare diseases have been highly troubled with anxiety, loneliness, and depression. The project evaluates resilience and coping strategies to address pandemic impact by discussion in a dedicated focus group using a web-based platform. The goal is to improve, in a sustainable manner, the coping skills and psychological well-being of children, adolescents, and young adults affected by rare skeletal diseases.

Study Overview

• Status: Completed
• Conditions: Osteogenesis Imperfecta; Multiple Osteochondroma; Ollier Disease; Maffucci Syndrome

Detailed Description

March 11th, 2020, the World Health Organization (WHO) declared the SARS-CoV-2 pandemic accordingly with the spread and size of the infection.

The restrictions implemented by the governments have substantially changed people's lifestyles, facilitating the emergence of emotions such as fear, anxiety, and a sense of loneliness.

The age groups most involved by these burdens have been childhood, adolescence, and young adults. These groups have experienced dramatic changes in their daily life in order the relational, educational, and social networks. Nonetheless, these links outline the ideal environment for the development of resilience and the promotion of coping strategies to face traumatic events.

These lifestyle challenges and their consequences are even more evident in the context of the rare diseases since the outbreak containment measures have emphasized the troubles with which people affected by these conditions are daily forced to confront, in terms of access to care and treatments, resulting in increased stress and anxiety.

The current phase represents a sensitive step for the resumption of the activities in charge of the health and social service aimed at the citizens and especially to people with special needs, such as people suffering from rare diseases.

The pandemic experience has promoted new forms of meeting and contact between individuals using web-based platforms, playing a fundamental role in preserving the community and "sociality" dimensions.

In this scenario, the rare diseases could be an attractive model since the pandemic impinges over their bio-psycho-social characteristics at different levels. Resilience and coping strategies adopted by rare diseases patients could configure as an exemplum to face the psychological distress resulting from anti-pandemic measures, suggesting new approaches of taking charge by health service.

This study evaluates resilience and coping strategies to address pandemic impact by discussion in a dedicated focus group using a web-based platform. The goal is to improve, in a sustainable manner, the coping skills and psychological well-being of children, adolescents, and young adults affected by rare skeletal diseases.

• Study Type: Observational
• Enrollment (Actual): 15

Contacts and Locations

Study Locations

• Italy
  • Bologna, Italy, 40139
    • IRCCS Istituto Ortopedico Rizzoli

Participation Criteria

Eligibility Criteria

• Ages Eligible for Study: 8 years to 24 years (Child, Adult)
• Accepts Healthy Volunteers: No
• Genders Eligible for Study: All

• Sampling Method: Non-Probability Sample

Study Population

Patient screening and enrollment will be carried out through:

• Rare Skeletal Disorders out-patient visit
• Rare Skeletal Disorders in-patient visit
• National Patients Organizations

Description

Inclusion Criteria:

Clinical and/or molecular diagnosis of Multiple osteochondromas; Clinical and/or molecular diagnosis of Ollier disease/Maffucci syndrome; Clinical and/or molecular diagnosis of Osteogenesis imperfecta; Consent to participate in the study

Exclusion Criteria:

Subject doesn't match the inclusion criteria

Study Plan

How is the study designed?

What is the study measuring?

Primary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Resilience	Ego-Resiliency Scale (ER89); 14 items; 4-point Likert scale (1=does not apply at all; 2=applies slightly, if at all; 3=applies somewhat; and 4=applies very strong); Score Resiliency Trait: 47-56 Very High, 35-46 High, 23-34 Undetermined, 11-22 Low, 0-10 Very Low	Evaluation at baseline of the study
Resilience	Ego-Resiliency Scale (ER89); 14 items; 4-point Likert scale (1=does not apply at all; 2=applies slightly, if at all; 3=applies somewhat; and 4=applies very strong); Score Resiliency Trait: 47-56 Very High, 35-46 High, 23-34 Undetermined, 11-22 Low, 0-10 Very Low	Change from baseline at 3 month
Resilience	Ego-Resiliency Scale (ER89); 14 items; 4-point Likert scale (1=does not apply at all; 2=applies slightly, if at all; 3=applies somewhat; and 4=applies very strong); Score Resiliency Trait: 47-56 Very High, 35-46 High, 23-34 Undetermined, 11-22 Low, 0-10 Very Low	Change from baseline at 6 month

Secondary Outcome Measures

Outcome Measure	Measure Description	Time Frame
Anxiety	Hospital Anxiety and Depression Scale (HADS); 14 items: 7 items relate to anxiety and 7 relate to depression; a 4-point (0-3) Likert-scale; Cutoffs for both HADS-anxiety and HADS-depression as >= 8, and for HADS-total >= 15	Evaluation at baseline of the study
Anxiety	Hospital Anxiety and Depression Scale (HADS); 14 items: 7 items relate to anxiety and 7 relate to depression; a 4-point (0-3) Likert-scale; Cutoffs for both HADS-anxiety and HADS-depression as >= 8, and for HADS-total >= 15	Change from baseline at 3 month
Anxiety	Hospital Anxiety and Depression Scale (HADS); 14 items: 7 items relate to anxiety and 7 relate to depression; a 4-point (0-3) Likert-scale; Cutoffs for both HADS-anxiety and HADS-depression as >= 8, and for HADS-total >= 15	Change from baseline at 6 month
Depression	Hospital Anxiety and Depression Scale (HADS); 14 items: 7 items relate to anxiety and 7 relate to depression; a 4-point (0-3) Likert-scale; Cutoffs for both HADS-anxiety and HADS-depression as >= 8, and for HADS-total >= 15	Evaluation at baseline of the study
Depression	Hospital Anxiety and Depression Scale (HADS); 14 items: 7 items relate to anxiety and 7 relate to depression; a 4-point (0-3) Likert-scale; Cutoffs for both HADS-anxiety and HADS-depression as >= 8, and for HADS-total >= 15	Change from baseline at 3 month
Depression	Hospital Anxiety and Depression Scale (HADS); 14 items: 7 items relate to anxiety and 7 relate to depression; a 4-point (0-3) Likert-scale; Cutoffs for both HADS-anxiety and HADS-depression as >= 8, and for HADS-total >= 15	Change from baseline at 6 month
Loneliness	UCLA Loneliness Scale (Version 3); 20-item scale; 4-point rating scale (1=Never; 2=Rarely; 3=Sometimes; and 4=Often); score ranging from 20 to 80.	Evaluation at baseline of the study
Loneliness	UCLA Loneliness Scale (Version 3); UCLA Loneliness Scale (Version 3); 20-item scale; 4-point rating scale (1=Never; 2=Rarely; 3=Sometimes; and 4=Often); score ranging from 20 to 80.	Change from baseline at 3 month
Loneliness	UCLA Loneliness Scale (Version 3); UCLA Loneliness Scale (Version 3); 20-item scale; 4-point rating scale (1=Never; 2=Rarely; 3=Sometimes; and 4=Often); score ranging from 20 to 80.	Change from baseline at 6 month

Collaborators and Investigators

• Sponsor: Istituto Ortopedico Rizzoli
• Investigators: Principal Investigator: Luca Sangiorgi, MD, PhD, IRCCS Istituto Ortopedico Rizzoli

Study record dates

Study Major Dates

• Study Start (Actual): May 12, 2021
• Primary Completion (Actual): December 31, 2022
• Study Completion (Actual): December 31, 2022

Study Registration Dates

• First Submitted: April 12, 2021
• First Submitted That Met QC Criteria: April 12, 2021
• First Posted (Actual): April 14, 2021

Study Record Updates

• Last Update Posted (Actual): March 1, 2023
• Last Update Submitted That Met QC Criteria: February 28, 2023
• Last Verified: February 1, 2023

More Information

Terms related to this study

• Keywords: COVID-19; Rare Diseases; Psychological Distress; e-Health; Focus group; Coping Strategies; Children, Adolescents, Young Adults
• Additional Relevant MeSH Terms: Neoplasms, Connective and Soft Tissue; Neoplasms by Histologic Type; Neoplasms; Genetic Diseases, Inborn; Musculoskeletal Diseases; Connective Tissue Diseases; Bone Diseases; Neoplastic Syndromes, Hereditary; Neoplasms, Bone Tissue; Neoplasms, Connective Tissue; Bone Diseases, Developmental; Osteochondrodysplasias; Collagen Diseases; Exostoses; Hyperostosis; Osteogenesis Imperfecta; Osteochondroma; Exostoses, Multiple Hereditary; Osteochondromatosis; Enchondromatosis
• Other Study ID Numbers: 150/2021/Oss/IOR

Plan for Individual participant data (IPD)

• Plan to Share Individual Participant Data (IPD)?: NO

Drug and device information, study documents

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