RESUMEN
BACKGROUND: People with a rare disease commonly experience long delays from the onset of symptoms to diagnosis. Rare diseases are challenging to diagnose because they are clinically heterogeneous, and many present with non-specific symptoms common to many diseases. We aimed to explore the experiences of people with myositis, primary immunodeficiency (PID), and sarcoidosis from symptom onset to diagnosis to identify factors that might impact receipt of a timely diagnosis. METHODS: This was a qualitative study using semi-structured interviews. Our approach was informed by Interpretive Phenomenological Analysis (IPA). We applied the lens of uncertainty management theory to tease out how patients experience, assess, manage and cope with puzzling and complex health-related issues while seeking a diagnosis in the cases of rare diseases. RESULTS: We conducted interviews with 26 people with a rare disease. Ten participants had been diagnosed with a form of myositis, 8 with a primary immunodeficiency, and 8 with sarcoidosis. Time to diagnosis ranged from 6 months to 12 years (myositis), immediate to over 20 years (PID), and 6 months to 15 years (sarcoidosis). We identified four themes that described the experiences of participants with a rare disease as they sought a diagnosis for their condition: (1) normalising and/or misattributing symptoms; (2) particularising by clinicians; (3) asserting patients' self-knowledge; and (4) working together through the diagnosable moment. CONCLUSIONS: Managing medical uncertainty in the time before diagnosis of a rare disease can be complicated by patients discounting their own symptoms and/or clinicians discounting the scale and impact of those symptoms. Persistence on the part of both clinician and patient is necessary to reach a diagnosis of a rare disease. Strategies such as recognising pattern failure and accommodating self-labelling are key to diagnosis.
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Investigación Cualitativa , Enfermedades Raras , Humanos , Enfermedades Raras/diagnóstico , Femenino , Masculino , Adulto , Incertidumbre , Persona de Mediana Edad , Adulto Joven , Miositis/diagnóstico , Anciano , Sarcoidosis/diagnóstico , Adolescente , Diagnóstico TardíoRESUMEN
BACKGROUND: Sarcoidosis is a chronic inflammatory granulomatous disease of unknown cause. Delays in diagnosis can result in disease progression and poorer outcomes for patients. Our aim was to review the current literature to determine the overall diagnostic delay of sarcoidosis, factors associated with diagnostic delay, and the experiences of people with sarcoidosis of diagnostic delay. METHODS: Three databases (PubMed/Medline, Scopus, and ProQuest) and grey literature sources were searched. Random effects inverse variance meta-analysis was used to pool mean diagnostic delay in all types of sarcoidosis subgroup analysis. Diagnostic delay was defined as the time from reported onset of symptoms to diagnosis of sarcoidosis. RESULTS: We identified 374 titles, of which 29 studies were included in the review, with an overall sample of 1531 (694 females, 837 males). The overall mean diagnostic delay in all types of sarcoidosis was 7.93 months (95% CI 1.21 to 14.64 months). Meta-aggregation of factors related to diagnostic delay in the included studies identified three categories: (1) the complex and rare features of sarcoidosis, (2) healthcare factors and (3) patient-centred factors. Meta-aggregation of outcomes reported in case studies revealed that the three most frequent outcomes associated with diagnostic delay were: (1) incorrect diagnosis, (2) incorrect treatment and (3) development of complications/disease progression. There was no significant difference in diagnostic delay between countries with gatekeeper health systems (where consumers are referred from a primary care clinician to specialist care) and countries with non-gatekeeper systems. No qualitative studies examining people's experiences of diagnostic delay were identified. CONCLUSION: The mean diagnostic delay for sarcoidosis is almost 8 months, which has objective consequences for patient management. On the other hand, there is a paucity of evidence about the experience of diagnostic delay in sarcoidosis and factors related to this. Gaining an understanding of people's experiences while seeking a diagnosis of sarcoidosis is vital to gain insight into factors that may contribute to delays, and subsequently inform strategies, tools and training activities aimed at increasing clinician and public awareness about this rare condition. TRIAL REGISTRATION: PROSPERO Registration number: CRD42022307236.
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Diagnóstico Tardío , Sarcoidosis , Femenino , Humanos , Progresión de la Enfermedad , Investigación Cualitativa , Sarcoidosis/diagnóstico , MasculinoRESUMEN
INTRODUCTION: Sarcoidosis is a rare systemic inflammatory granulomatous disease of unknown cause. It can manifest in any organ. The incidence of sarcoidosis varies across countries, and by ethnicity and gender. Delays in the diagnosis of sarcoidosis can lead to extension of the disease and organ impairment. Diagnosis delay is attributed in part to the lack of a single diagnostic test or unified commonly used diagnostic criteria, and to the diversity of disease manifestations and symptom load. There is a paucity of evidence examining the determinants of diagnostic delay in sarcoidosis and the experiences of people with sarcoidosis related to delayed diagnosis. We aim to systematically review available evidence about diagnostic delay in sarcoidosis to elucidate the factors associated with diagnostic delay for this disease in different contexts and settings, and the consequences for people with sarcoidosis. METHODS AND ANALYSIS: A systematic search of the literature will be conducted using PubMed/Medline, Scopus, and ProQuest databases, and sources of grey literature, up to 25th of May 2022, with no limitations on publication date. We will include all study types (qualitative, quantitative, and mixed methods) except review articles, examining diagnostic delay, incorrect diagnosis, missed diagnosis or slow diagnosis of all types of sarcoidosis across all age groups. We will also examine evidence of patients' experiences associated with diagnostic delay. Only studies in English, German and Indonesian will be included. The outcomes we examine will be diagnostic delay time, patients' experiences, and factors associated with diagnostic delay in sarcoidosis. Two people will independently screen the titles and abstracts of search results, and then the remaining full-text documents against the inclusion criteria. Disagreements will be resolved with a third reviewer until consensus is reached. Selected studies will be appraised using the Mixed Methods Appraisal Tool (MMAT). A meta-analysis and subgroup analyses of quantitative data will be conducted. Meta-aggregation methods will be used to analyse qualitative data. If there is insufficient data for these analyses, a narrative synthesis will be conducted. DISCUSSION: This review will provide systematic and integrated evidence on the diagnostic delay, associated factors, and experiences of diagnosis delay among people with all types of sarcoidosis. This knowledge may shed light on ways to improve diagnosis delays in diagnosis across different subpopulations, and with different disease presentations. ETHICS AND DISSEMINATION: Ethical approval will not be required as no human recruitment or participation will be involved. Findings of the study will be disseminated through publications in peer-reviewed journals, conferences, and symposia. TRIAL REGISTRATION: PROSPERO Registration number: CRD42022307236. URL of the PROSPERO registration: https://www.crd.york.ac.uk/PROSPEROFILES/307236_PROTOCOL_20220127.pdf.
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Diagnóstico Tardío , Sarcoidosis , Humanos , Etnicidad , Incidencia , Metaanálisis como Asunto , Proyectos de Investigación , Revisiones Sistemáticas como AsuntoRESUMEN
BACKGROUND: Recent research revealed diverse content and varying levels of quality in YouTube music therapy videos and prompted questions about viewers' discrimination abilities. OBJECTIVE: This study compares ratings of a YouTube music therapy session video by viewers with different levels of music therapy expertise to determine video elements related to perceptions of representational quality. METHOD: Eighty-one participants included 25 novices (freshmen and sophomores in an introductory music therapy course), 25 pre-interns (seniors and equivalency students who had completed all core Music Therapy courses), 26 professionals (MT-BC or MT-BC eligibility) with a mean of 1.75 years of experience, and an expert panel of 5 MT-BC professionals with a mean of 11 years of experience in special education. After viewing a music therapy special education video that in previous research met basic competency criteria and professional standards of the American Music Therapy Association, participants completed a 16-item questionnaire. RESULTS: Novices' ratings were more positive (less discriminating) compared to experienced viewers' neutral or negative ratings. Statistical analysis (ANOVA) of novice, pre-intern, and professional ratings of all items revealed significant differences p, .05) for specific therapy content and for a global rating of representational quality. Experienced viewers' ratings were similar to the expert panel's ratings. Content analysis of viewers' reasons for their representational quality ratings corroborated ratings of therapy-specific content. CONCLUSIONS: A video that combines and clearly depicts therapy objectives, client improvement, and the effectiveness of music within a therapeutic intervention best represent the music therapy profession in a public social platform like YouTube.
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Internet , Musicoterapia/métodos , Satisfacción Personal , Grabación en Video/métodos , Adulto , Análisis de Varianza , Femenino , Humanos , Masculino , Persona de Mediana Edad , Opinión Pública , Refuerzo en Psicología , Autoeficacia , Estrés Psicológico/terapia , Adulto JovenRESUMEN
The purpose of this study was to conduct a descriptive analysis of music therapy-related videos on YouTube. Preliminary searches using the keywords music therapy, music therapy session, and "music therapy session" resulted in listings of 5000, 767, and 59 videos respectively. The narrowed down listing of 59 videos was divided between two investigators and reviewed in order to determine their relationship to actual music therapy practice. A total of 32 videos were determined to be depictions of music therapy sessions. These videos were analyzed using a 16-item investigator-created rubric that examined both video specific information and therapy specific information. Results of the analysis indicated that audio and visual quality was adequate, while narrative descriptions and identification information were ineffective in the majority of the videos. The top 5 videos (based on the highest number of viewings in the sample) were selected for further analysis in order to investigate demonstration of the Professional Level of Practice Competencies set forth in the American Music Therapy Association (AMTA) Professional Competencies (AMTA, 2008). Four of the five videos met basic competency criteria, with the quality of the fifth video precluding evaluation of content. Of particular interest is the fact that none of the videos included credentialing information. Results of this study suggest the need to consider ways to ensure accurate dissemination of music therapy-related information in the YouTube environment, ethical standards when posting music therapy session videos, and the possibility of creating AMTA standards for posting music therapy related video.
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Internet , Musicoterapia/métodos , Música , Grabación de Videodisco/normas , Humanos , Satisfacción Personal , Estrés Psicológico/prevención & controlRESUMEN
This study was designed to develop a method for collecting music therapy majors' hypothesized self-attributions of comfort and skill in diverse clinical scenarios and relating their attributions to recall and perceptions of similar personal and clinical experience. Fifty eight music therapy majors, from freshmen to graduate (board-certified) students, watched 10 brief video excerpts. After each excerpt participants moved 2 Continuous Response Digital Interface (CRDI) dials, one labeled "comfort" and the other labeled "skill," to indicate their feelings about themselves as therapist with the client(s) in the excerpt. At the end of viewing all excerpts, participants completed an open-ended total recall paper/pencil form requesting estimation of personal and clinical experience with any population represented in the excerpts. Participants recalled more personal experience compared to clinical experience. A strong positive correlation was found between personal and clinical experience estimates. A strong positive correlation was also found between comfort and skill scores. Personal experience estimates were not related to self-attributions of comfort or skill. Clinical excerpts prompted neutral self-attributions of comfort and skill with comfort being slightly higher than skill self-attributions. Clinical experience estimates, although not related to self-attributions of comfort; were positively related to self-attributions of skill. Exploratory comparisons with small groups within the total sample suggest a highly positive impact of specific music therapy clinical experience, particularly the post-coursework internship, on skill self-attributions. Results are discussed within the context of using the assessment to investigate the predictive value of self-attributes as clinical indicators of persistence for recruitment and retention of potentially successful music therapy students and young professionals.
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Conocimientos, Actitudes y Práctica en Salud , Control Interno-Externo , Recuerdo Mental , Musicoterapia/educación , Música , Estudiantes del Área de la Salud/estadística & datos numéricos , Grabación de Cinta de Video , Adulto , Percepción Auditiva , Femenino , Humanos , Masculino , Musicoterapia/métodos , Autoimagen , Adulto JovenRESUMEN
An online video assignment was developed to facilitate transition from the orientation session to the first contact with hospital patients for music therapy majors and other students enrolled in an Arts in Medicine service learning course (AIMS). All students (N = 84) completed a 2 hour hospital orientation session. After the orientation session the experimental group (n = 42) completed an online video assignment before volunteering at the hospital The control group (n = 42) began volunteering after the orientation session without completing the video assignment. Analysis indicates the majority of both groups initiated their first session independently without assistance from other AIMS volunteers, an experienced AIM volunteer, an AIM assistant, or hospital staff member. The majority of both groups also engaged at least one patient during their first visit at the hospital. Content analysis of "first contact" weekly reports, however, indicated experimental group students wrote longer reports and included more positive comments, particularly about patients, compared to control group students. Volunteers in the experimental group also began their contacts as scheduled on the course calendar compared to later starting dates of control group volunteers.
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Conocimientos, Actitudes y Práctica en Salud , Musicoterapia/métodos , Música , Autoeficacia , Estudiantes del Área de la Salud/estadística & datos numéricos , Grabación de Cinta de Video , Voluntarios/educación , Adulto , Curriculum , Femenino , Humanos , Masculino , Competencia Profesional , Relaciones Profesional-Paciente , Estudiantes del Área de la Salud/psicología , Voluntarios/psicología , Voluntarios/estadística & datos numéricos , Adulto JovenRESUMEN
This article updates previous content analyses of the Journal of Music Therapy (JMT) and presents information regarding the behavioral research approach to music therapy as reflected by published studies. JMT articles from 1964 through 1999 were examined to determine if the methodology included a behavioral research design (e.g., reversal, multiple baseline). Case studies not meeting the design criteria were excluded from the sample as were experimental group studies using a behavioral observation method or behavioral technique within a traditional statistical design. Experimental studies, however, in which behavioral research designs were included in treatment conditions and assigned to different groups were included. Articles meeting the behavioral research design criteria were analyzed to determine the type of design, the behavioral observation method and reliability report, the client population, and the music application. Of the 607 articles published in JMT, 96 or 15.8% included a behavioral research design. The lowest percentage occurred in the earliest volumes of JMT during the latter part of the 60s. An increase in behavioral research articles occurred during the 70s and 80s and continued throughout the 90s. Other findings include the following: (a) the most prevalent design was the reversal (ABA) with many variations; (b) the predominant observation methods were frequency counts and interval recording with an increase in observation reliability reports in the 80s and 90s; (c) studies published during the 80s and 90s included a wide variety of client populations compared to the 60s and 70s predominant applications with mental retardation and emotional disturbances; (d) the contingent application of music was highest during the 70s and 80s and virtually nonexistent during the 60s and 90s when other treatment conditions employed music as noncontingent background stimulus, an activity for structuring responses, and a cue for maintaining nonmusic responses. The increase in articles through the decades coupled with the increased diversity of populations suggests that behavioral research designs are flexible and applicable in music therapy practice today as much as they were during the initial phase during the 70s and 80s.
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Musicoterapia/métodos , Musicoterapia/normas , Humanos , Publicaciones Periódicas como Asunto , Pruebas Psicológicas/normas , Proyectos de Investigación/normasRESUMEN
Twelve older adults with cognitive impairments who were participants in weekly community-based group music therapy sessions, 6 older adults in an Alzheimer's caregivers' group, and 6 college student volunteers listened to a 3.5 minute prepared audiotape of instrumental excerpts of patriotic selections. The tape consisted of 7 excerpts ranging from 18 s to 34 s in duration. Each music excerpt was followed by a 7-9 s period of silence, a "wait" excerpt. Listeners were instructed to move a Continuous Response Digital Interface (CRDI) to the name of the music excerpt depicted on the CRDI overlay when they heard a music excerpt. Likewise, they were instructed to move the dial to the word "WAIT" when there was no music. They were also instructed to maintain the dial position for the duration of each music or silence excerpt. Statistical analysis indicated no significant differences between the caregivers' and the college students' group means for total dial changes, correct and incorrect recognitions, correct and incorrect responses to silence excerpts, and reaction times. The mean scores of these 2 groups were combined and compared with the mean scores of the group of elderly adults with cognitive impairments. The mean total dial changes were significantly lower for the listeners with cognitive impairments, resulting in significant differences in all of the other response categories except incorrect recognitions. In addition, their mean absence of response to silence excerpts was significantly higher than their mean absence of responding to music excerpts. Their mean reaction time was significantly slower than the comparison group's reaction time. To evaluate training effects, 10 of the original 12 music therapy participants repeated the listening task with assistance from the therapist (treatment) immediately following the first listening (baseline). A week later the order was reversed for the 2 listening trials. Statistical and graphic analysis of responses between first and second baseline responses indicate significant improvement in responses to silence and music excerpts over the 2 sessions. Applications of the findings to music listening interventions for maintaining attention, eliciting social interaction between clients or caregivers and their patients, and evaluating this population's affective responses to music are discussed.