Ready to Sail: Evaluating Sailing's Feasibility as Ergotherapy

Rare skeletal diseases represent a significant global health burden, affecting millions of people worldwide. These chronic, debilitating conditions necessitate the development of optimized clinical pathways and comprehensive support systems.

Adventure therapy (AT) has emerged as a promising intervention for individuals with chronic conditions, demonstrating improvements in self-esteem, autonomy, and social skills. Sailing, a form of AT, offers opportunities for physical activity, social inclusion, and emotional well-being in individuals with disabilities.

This study aims to assess the feasibility of using sailing as an occupational therapy intervention for patients with rare skeletal diseases. We hypothesize that sailing can enhance physical, cognitive, and social functioning in this patient population.

A pilot program of sailing lessons for patients with rare skeletal diseases will be conducted. Participants will undergo assessments of physical, cognitive, and social skills before and after the intervention.

This study will be the first to explore the use of sailing-based AT for this patient population. The findings will inform the development of larger-scale studies and contribute to the optimization of rehabilitation programs for this patient population.

• 5-day sailing activity (Other): * Sailing activity (4 h/die) * Stretching exercises pre and post-sailing activity * Post-activity debriefing

Primary outcomes

• Health-related Quality of Life [T0: Baseline]: EuroQol 5-Dimension (EQ-5D) instrument offers valuable information about patient-reported health status. The questionnaire consists of two sections-descriptive system and visual analog scale (VAS)-and yields two scores: Index Value (IV), and overall health status (VAS). In the descriptive system section, patients report their level of problems in five dimensions-mobility, self-care, usual activities, pain/discomfort, and anxiety/depression-and rate their health on a 5-level options for adults, and 3-level for adolescents and children. Based on the pattern of their responses, patients generate a health profile ranging from best health (11111 = highest level) to worst health (55555 = lowest level). Each health profile is transformed into an IV, that varies from 0 (absence of life/death) to 1 (perfect health). The VAS measures the patients' overall health status, with a score ranging from 0 ("The worst health you can imagine") to 100 ("The best health you can imagine").
• Health-related Quality of Life [T1: up to 2 weeks]: EuroQol 5-Dimension (EQ-5D) instrument offers valuable information about patient-reported health status. The questionnaire consists of two sections-descriptive system and visual analog scale (VAS)-and yields two scores: Index Value (IV), and overall health status (VAS). In the descriptive system section, patients report their level of problems in five dimensions-mobility, self-care, usual activities, pain/discomfort, and anxiety/depression-and rate their health on a 5-level options for adults, and 3-level for adolescents and children. Based on the pattern of their responses, patients generate a health profile ranging from best health (11111 = highest level) to worst health (55555 = lowest level). Each health profile is transformed into an IV, that varies from 0 (absence of life/death) to 1 (perfect health). The VAS measures the patients' overall health status, with a score ranging from 0 ("The worst health you can imagine") to 100 ("The best health you can imagine").
• Health-related Quality of Life [T2: up to 3 weeks]: EuroQol 5-Dimension (EQ-5D) instrument offers valuable information about patient-reported health status. The questionnaire consists of two sections-descriptive system and visual analog scale (VAS)-and yields two scores: Index Value (IV), and overall health status (VAS). In the descriptive system section, patients report their level of problems in five dimensions-mobility, self-care, usual activities, pain/discomfort, and anxiety/depression-and rate their health on a 5-level options for adults, and 3-level for adolescents and children. Based on the pattern of their responses, patients generate a health profile ranging from best health (11111 = highest level) to worst health (55555 = lowest level). Each health profile is transformed into an IV, that varies from 0 (absence of life/death) to 1 (perfect health). The VAS measures the patients' overall health status, with a score ranging from 0 ("The worst health you can imagine") to 100 ("The best health you can imagine").
• Health-related Quality of Life [T3: up to 3 months]: EuroQol 5-Dimension (EQ-5D) instrument offers valuable information about patient-reported health status. The questionnaire consists of two sections-descriptive system and visual analog scale (VAS)-and yields two scores: Index Value (IV), and overall health status (VAS). In the descriptive system section, patients report their level of problems in five dimensions-mobility, self-care, usual activities, pain/discomfort, and anxiety/depression-and rate their health on a 5-level options for adults, and 3-level for adolescents and children. Based on the pattern of their responses, patients generate a health profile ranging from best health (11111 = highest level) to worst health (55555 = lowest level). Each health profile is transformed into an IV, that varies from 0 (absence of life/death) to 1 (perfect health). The VAS measures the patients' overall health status, with a score ranging from 0 ("The worst health you can imagine") to 100 ("The best health you can imagine").

Secondary outcomes

• Self-esteem [T0: Baseline]: Rosenberg Self-Esteem Scale: 10-item scales, ranging from 0-30. Scores between 15 and 25 are within normal range; scores below 15 suggest low self-esteem.
• Self-esteem [T1: up to 2 weeks]: Rosenberg Self-Esteem Scale: 10-item scales, ranging from 0-30. Scores between 15 and 25 are within normal range; scores below 15 suggest low self-esteem.
• Self-esteem [T2: up to 3 weeks]: Rosenberg Self-Esteem Scale: 10-item scales, ranging from 0-30. Scores between 15 and 25 are within normal range; scores below 15 suggest low self-esteem.
• Self-esteem [T3: up to 3 months]: Rosenberg Self-Esteem Scale is a 10-item scale that assesses the self-esteem, ranging from 0-30. Scores between 15 and 25 are within normal range; scores below 15 suggest low self-esteem.
• Motor Coordination [T0: Baseline]: Performance Oriented Mobility Assesment (Tinetti Balance Scale) is an administered 16-item task-oriented test that measures gait and balance abilities, primarily to assess fall risk. It uses a 3-point ordinal scale of 0, 1 and 2. Gait is scored over 12 and balance is scored over 16. The total score (Fall Risk) ranges from 0 to 28, with higher scores indicating better balance and gait performance (19 or below=High risk of falls; 20-23=Moderate risk of falls; 24-28=Low risk of falls).
• Motor Coordination [T1: up to 2 weeks]: Performance Oriented Mobility Assesment (Tinetti Balance Scale) is an administered 16-item task-oriented test that measures gait and balance abilities, primarily to assess fall risk. It uses a 3-point ordinal scale of 0, 1 and 2. Gait is scored over 12 and balance is scored over 16. The total score (Fall Risk) ranges from 0 to 28, with higher scores indicating better balance and gait performance (19 or below=High risk of falls; 20-23=Moderate risk of falls; 24-28=Low risk of falls).
• Motor Coordination [T2: up to 3 weeks]: Performance Oriented Mobility Assesment (Tinetti Balance Scale) is an administered 16-item task-oriented test that measures gait and balance abilities, primarily to assess fall risk. It uses a 3-point ordinal scale of 0, 1 and 2. Gait is scored over 12 and balance is scored over 16. The total score (Fall Risk) ranges from 0 to 28, with higher scores indicating better balance and gait performance (19 or below=High risk of falls; 20-23=Moderate risk of falls; 24-28=Low risk of falls).
• Motor Coordination [T3: up to 3 months]: Performance Oriented Mobility Assesment (Tinetti Balance Scale) is an administered 16-item task-oriented test that measures gait and balance abilities, primarily to assess fall risk. It uses a 3-point ordinal scale of 0, 1 and 2. Gait is scored over 12 and balance is scored over 16. The total score (Fall Risk) ranges from 0 to 28, with higher scores indicating better balance and gait performance (19 or below=High risk of falls; 20-23=Moderate risk of falls; 24-28=Low risk of falls).
• Balance [T0: Baseline]: Berg Balance Scale is a 14-item objective measure that assesses static balance and fall risk, item-level scores range from 0-4, determined by ability to perform the assessed activity. The total score ranges from 0 to 56, with higher scores indicating better balance. A score of less than 45 suggests an increased risk of falling.
• Balance [T1: up to 2 weeks]: Berg Balance Scale is a 14-item objective measure that assesses static balance and fall risk, item-level scores range from 0-4, determined by ability to perform the assessed activity. The total score ranges from 0 to 56, with higher scores indicating better balance. A score of less than 45 suggests an increased risk of falling.
• Balance [T2: up to 3 weeks]: Berg Balance Scale is a 14-item objective measure that assesses static balance and fall risk, item-level scores range from 0-4, determined by ability to perform the assessed activity. The total score ranges from 0 to 56, with higher scores indicating better balance. A score of less than 45 suggests an increased risk of falling.
• Balance [T3: up to 3 months]: Berg Balance Scale is a 14-item objective measure that assesses static balance and fall risk, item-level scores range from 0-4, determined by ability to perform the assessed activity. The total score ranges from 0 to 56, with higher scores indicating better balance. A score of less than 45 suggests an increased risk of falling.
• Kinesiophobia [T0: Baseline]: The Tampa Scale for Kinesiophobia (TSK) is a validated self-report questionnaire designed to assess the fear of movement or (re)injury in individuals experiencing pain or musculoskeletal disorders. The TSK consists of 13 items (Italian validated version), each rated on a 4-point Likert scale, ranging from 1 ("Strongly Disagree") to 4 ("Strongly Agree"). The scores are summed to yield a total score ranging from 13 to 52, with higher scores indicating greater levels of kinesiophobia. The TSK includes two subscales: 1.Activity Avoidance, with a score ranging from 6 to 24; 2.Harm Beliefs, with a score ranging from 7 to 28. Higher scores on both the Activity Avoidance and Harm Beliefs subscales indicate greater levels of kinesiophobia, reflecting increased fear of movement due to injury concerns (Activity Avoidance subscale) or harmful beliefs about physical activity (Harm Beliefs subscale).
• Kinesiophobia [T2: up to 3 weeks]: The Tampa Scale for Kinesiophobia (TSK) is a validated self-report questionnaire designed to assess the fear of movement or (re)injury in individuals experiencing pain or musculoskeletal disorders. The TSK consists of 13 items (Italian validated version), each rated on a 4-point Likert scale, ranging from 1 ("Strongly Disagree") to 4 ("Strongly Agree"). The scores are summed to yield a total score ranging from 13 to 52, with higher scores indicating greater levels of kinesiophobia. The TSK includes two subscales: 1.Activity Avoidance, with a score ranging from 6 to 24; 2.Harm Beliefs, with a score ranging from 7 to 28. Higher scores on both the Activity Avoidance and Harm Beliefs subscales indicate greater levels of kinesiophobia, reflecting increased fear of movement due to injury concerns (Activity Avoidance subscale) or harmful beliefs about physical activity (Harm Beliefs subscale).
• Kinesiophobia [T3: up to 3 months]: The Tampa Scale for Kinesiophobia (TSK) is a validated self-report questionnaire designed to assess the fear of movement or (re)injury in individuals experiencing pain or musculoskeletal disorders. The TSK consists of 13 items (Italian validated version), each rated on a 4-point Likert scale, ranging from 1 ("Strongly Disagree") to 4 ("Strongly Agree"). The scores are summed to yield a total score ranging from 13 to 52, with higher scores indicating greater levels of kinesiophobia. The TSK includes two subscales: 1.Activity Avoidance, with a score ranging from 6 to 24; 2.Harm Beliefs, with a score ranging from 7 to 28. Higher scores on both the Activity Avoidance and Harm Beliefs subscales indicate greater levels of kinesiophobia, reflecting increased fear of movement due to injury concerns (Activity Avoidance subscale) or harmful beliefs about physical activity (Harm Beliefs subscale).
• Well-being and Mental Health [T0: Baseline]: Clinical Outcomes in Routine Evaluation-Outcome Measure (CORE-OM) is a self-report questionnaire designed to assess psychological distress and monitor treatment response. It consists of 34 items evaluating emotional state and perceived well-being over the past week, rated on a 5-point Likert scale from 0 ("Not at all") to 4 ("Most or all of the time"). The total score is calculated as the mean of all item scores and can be reported in two ways: 1.Raw score: The mean of all item scores (range: 0-4). 2.Standardized score: Obtained by multiplying the raw score by 10, yielding a scale from 0 to 40. The data presented in the results refer to the raw score. Score Interpretation: 0.00-0.99: No clinical distress or minimal risk. 1.00-1.99: Mild distress, potentially significant. 2.00-2.99: Moderate distress, generally indicative of the need for intervention. 3.00-4.00: Severe distress, highly indicative of the need for immediate clinical intervention.
• Well-being and Mental Health [T2: up to 3 weeks]: Clinical Outcomes in Routine Evaluation-Outcome Measure (CORE-OM) is a self-report questionnaire designed to assess psychological distress and monitor treatment response. It consists of 34 items evaluating emotional state and perceived well-being over the past week, rated on a 5-point Likert scale from 0 ("Not at all") to 4 ("Most or all of the time"). The total score is calculated as the mean of all item scores and can be reported in two ways: 1.Raw score: The mean of all item scores (range: 0-4). 2.Standardized score: Obtained by multiplying the raw score by 10, yielding a scale from 0 to 40. The data presented in the results refer to the raw score. Score Interpretation: 0.00-0.99: No clinical distress or minimal risk. 1.00-1.99: Mild distress, potentially significant. 2.00-2.99: Moderate distress, generally indicative of the need for intervention. 3.00-4.00: Severe distress, highly indicative of the need for immediate clinical intervention.
• Well-being and Mental Health [T3: up to 3 months]: Clinical Outcomes in Routine Evaluation-Outcome Measure (CORE-OM) is a self-report questionnaire designed to assess psychological distress and monitor treatment response. It consists of 34 items evaluating emotional state and perceived well-being over the past week, rated on a 5-point Likert scale from 0 ("Not at all") to 4 ("Most or all of the time"). The total score is calculated as the mean of all item scores and can be reported in two ways: 1.Raw score: The mean of all item scores (range: 0-4). 2.Standardized score: Obtained by multiplying the raw score by 10, yielding a scale from 0 to 40. The data presented in the results refer to the raw score. Score Interpretation: 0.00-0.99: No clinical distress or minimal risk. 1.00-1.99: Mild distress, potentially significant. 2.00-2.99: Moderate distress, generally indicative of the need for intervention. 3.00-4.00: Severe distress, highly indicative of the need for immediate clinical intervention.
• Physical Functioning and Psychological Well-being [T0: Baseline]: Pediatric Outcomes Data Collection (PODCI) evaluates functional health status through an 83-item questionnaire, and uses domain-specific scales. Each scale generates a score ranging from 0 to 100, with higher scores indicating better functioning or less impairment.
• Physical Functioning and Psychological Well-being [T1: up to 2 weeks]: Pediatric Outcomes Data Collection (PODCI) evaluates functional health status through an 83-item questionnaire, and uses domain-specific scales. Each scale generates a score ranging from 0 to 100, with higher scores indicating better functioning or less impairment.
• Physical Functioning and Psychological Well-being [T2: up to 3 weeks]: Pediatric Outcomes Data Collection (PODCI) evaluates functional health status through an 83-item questionnaire, and uses domain-specific scales. Each scale generates a score ranging from 0 to 100, with higher scores indicating better functioning or less impairment.
• Physical Functioning and Psychological Well-being [T3: up to 3 months]: Pediatric Outcomes Data Collection (PODCI) evaluates functional health status through an 83-item questionnaire, and uses domain-specific scales. Each scale generates a score ranging from 0 to 100, with higher scores indicating better functioning or less impairment.

Other outcomes

• Information on the Functioning and Disability of an Individual [T0]: Functional Independence Measure (FIM) is a validated clinician-administered tool designed to assess functional independence in individuals with neurological or musculoskeletal conditions. It evaluates the level of assistance required for daily activities across six domains: self-care, sphincter control, mobility, locomotion, communication, and social cognition. The FIM consists of 18 items, each rated on a 7-point ordinal scale (1 = total assistance, 7 = complete independence), with total scores ranging from 18 to 126. Higher scores indicate greater functional independence.
• Information on the Functioning and Disability of an Individual [T3]: Functional Independence Measure (FIM) is a validated clinician-administered tool designed to assess functional independence in individuals with neurological or musculoskeletal conditions. It evaluates the level of assistance required for daily activities across six domains: self-care, sphincter control, mobility, locomotion, communication, and social cognition. The FIM consists of 18 items, each rated on a 7-point ordinal scale (1 = total assistance, 7 = complete independence), with total scores ranging from 18 to 126. Higher scores indicate greater functional independence.
• Kinesiophobia [T1]: The Tampa Scale for Kinesiophobia (TSK) is a validated self-report questionnaire designed to assess the fear of movement or (re)injury in individuals experiencing pain or musculoskeletal disorders. The TSK consists of 13 items (Italian validated version), each rated on a 4-point Likert scale, ranging from 1 ("Strongly Disagree") to 4 ("Strongly Agree"). The scores are summed to yield a total score ranging from 13 to 52, with higher scores indicating greater levels of kinesiophobia. The TSK includes two subscales: 1.Activity Avoidance, with a score ranging from 6 to 24; 2.Harm Beliefs, with a score ranging from 7 to 28. Higher scores on both the Activity Avoidance and Harm Beliefs subscales indicate greater levels of kinesiophobia, reflecting increased fear of movement due to injury concerns (Activity Avoidance subscale) or harmful beliefs about physical activity (Harm Beliefs subscale).
• Well-being and Mental Health [T1]: Clinical Outcomes in Routine Evaluation-Outcome Measure (CORE-OM) is a self-report measure of psychological distress designed to be administered during a course of treatment to determine treatment response. It compries 34 questions about how the participants have been feeling over the last week, using a 5-point Likert scale ranging from 0 ("Not at all") to 4 ("Most or all of the time"). The total score is calculated as the mean of all item scores (ranging from 0 to 4) and can be reported either as a raw score or standardized (e.g., multiplying the mean by 10 to obtain a score on a 0-40 scale). Score Interpretation: 0-0.99: No clinical distress or minimal risk. 1.00-1.99: Mild distress, potentially significant. 2.00-2.99: Moderate distress, generally indicative of a need for intervention. 3.00-4.00: Severe distress, highly indicative of immediate clinical intervention.

Sex: All, ages: 12 Years to any

Inclusion Criteria:

* Individuals of any gender
* Diagnosis of a rare skeletal disease, confirmed clinically and/or molecularly
* Age 12 years or older
* No history of surgery within six months prior to study enrollment.

Exclusion Criteria:

* Individuals undergoing diagnostic evaluation for a rare skeletal disease
* Participants younger than 12 years of age.
* Individuals who underwent surgery within the preceding 6 months.
* Participants with fractures or musculoskeletal injuries sustained within the past year.
* Individuals who were unable to provide written informed consent.

Participant flow

Outcome results

• Health-related Quality of Life (Primary), Median, score on a scale: Patients With Rare Skeletal Disorders 0.91 (0.65 to 1)
• Health-related Quality of Life (Primary), Median, score on a scale: Patients With Rare Skeletal Disorders 0.91 (0.75 to 1)
• Health-related Quality of Life (Primary), Median, score on a scale: Patients With Rare Skeletal Disorders 0.90 (0.75 to 1)
• Health-related Quality of Life (Primary), Median, score on a scale: Patients With Rare Skeletal Disorders 0.82 (0.72 to 0.95)
• Self-esteem (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 22 (9 to 26)
• Self-esteem (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 21 (11 to 23)
• Self-esteem (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 22 (10 to 25)
• Self-esteem (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 21 (11 to 30)
• Motor Coordination (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 16 (15 to 16)
• Motor Coordination (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 12 (11 to 12)
• Motor Coordination (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 12 (12 to 12)
• Motor Coordination (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 12 (10 to 12)
• Balance (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 56 (56 to 56)
• Balance (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 56 (56 to 56)
• Balance (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 56 (56 to 56)
• Balance (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 56 (56 to 56)
• Kinesiophobia (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 10 (6 to 17)
• Kinesiophobia (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 11 (7 to 16)
• Kinesiophobia (Secondary), Median, score on a scale: Patients With Rare Skeletal Disorders 10.50 (7 to 17)
• Well-being and Mental Health (Secondary), Median, units on a scale: Patients With Rare Skeletal Disorders 0.85 (0.10 to 2.70)
• 10 more outcome measures not shown.

Adverse events

Time frame: 4 months

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• Results publication: Boarini M, Banchelli F, Fittipaldi S, Scognamiglio D, Farella GM, Platano D, Rogati G, Di Sipio E, Villa G, Berti L, Leardini A, Sartini S, Scopinaro A, Sangiorgi L. Feasibility and safety of sailing based rehabilitation for rare skeletal disorders using wearable sensors and patient reported outcomes. Sci Rep. 2025 Oct 23;15(1):37153. doi: 10.1038/s41598-025-22231-8. PMID 41131085

• Study Protocol and Statistical Analysis Plan (PDF, 2024-03-22)

• Luca Sangiorgi, MD, PhD, Principal investigator, IRCCS Istituto Ortopedico Rizzoli