MARIA FERNANDA ABALEM DE SA CARRICONDO
Projetos de Pesquisa
Unidades Organizacionais
Instituto Central, Hospital das Clínicas, Faculdade de Medicina - Médico
LIM/33 - Laboratório de Oftalmologia, Hospital das Clínicas, Faculdade de Medicina
LIM/33 - Laboratório de Oftalmologia, Hospital das Clínicas, Faculdade de Medicina
7 resultados
Resultados de Busca
Agora exibindo 1 - 7 de 7
- The Michigan Vision-Related Anxiety Questionnaire: A Psychosocial Outcomes Measure for Inherited Retinal Degenerations(2021) LACY, Gabrielle D.; ABALEM, Maria Fernanda; ANDREWS, Chris A.; ABUZAITOUN, Rebhi; POPOVA, Lilia T.; SANTOS, Erin P.; YU, Gina; RAKINE, Hanan Y.; BAIG, Natasha; EHRLICH, Joshua R.; FAHIM, Abigail T.; BRANHAM, Kari H.; SWENOR, Bonnielin K.; LICHTER, Paul R.; DAGNELIE, Gislin; STELMACK, Joan A.; MUSCH, David C.; JAYASUNDERA, K. Thiranpatient-reported outcome measure for screening and monitoring vision-related anxiety in patients with inherited retinal degenerations. center dot DESIGN: Item-response theory and graded response modeling to quantitatively validate questionnaire items generated from qualitative interviews and patient feedback. center dot METHODS: Patients at the Kellogg Eye Center (University of Michigan, Ann Arbor, Michigan, USA) with a clinical diagnosis of an inherited retinal degeneration (n = 128) participated in an interviewer-administered questionnaire. The questionnaire consisted of 166 items, 26 of which pertained to concepts of ""worry""and ""anxiety.""The subset of vision-related anxiety questions was analyzed by a graded response model using the Cai Metropolis-Hastings Robbins-Monro algorithm in the R software mirt package. Item reduction was performed based on item fit, item information, and item discriminability. To assess test-retest variability, 25 participants completed the questionnaire a second time 4 to 16 days later. center dot RESULTS: The final questionnaire consisted of 14 items divided into 2 unidimensional domains: rod function anxiety and cone function anxiety. The questionnaire exhibited convergent validity with the Patient Health Questionnaire for symptoms of depression and anxiety. This vision-related anxiety questionnaire has high marginal reliability (0.81 for rod-function anxiety, 0.83 for cone-function anxiety) and exhibits minimal test-retest
- The Michigan Retinal Degeneration Questionnaire: A Patient-Reported Outcome Instrument for Inherited Retinal Degenerations(2021) LACY, Gabrielle D.; ABALEM, Maria Fernanda; ANDREWS, Chris A.; POPOVA, Lilia T.; SANTOS, Erin P.; YU, Gina; RAKINE, Hanan Y.; BAIG, Natasha; EHRLICH, Joshua R.; FAHIM, Abigail T.; BRANHAM, Kari H.; STELMACK, Joan A.; SWENOR, Bonnielin K.; DAGNELIE, Gislin; MUSCH, David C.; JAYASUNDERA, K. Thirancenter dot PURPOSE: To create a psychometrically validated patient-reported outcome measure for inherited retinal degenerations. center dot DESIGN: Qualitative and quantitative patient-reported outcome (PROs) questionnaire development using item response theory validation. center dot METHODS: One hundred twenty-eight patients with a diagnosis of an inherited retinal degeneration at the Kellogg Eye Center (University of Michigan) were recruited and administered a 166-item questionnaire comprising 7 expert-defined domains. The questionnaire was re-administered 4-16 days later to a subset of 25 participants to assess test-retest variability. Graded response models were fit by Cai & rsquo;s Metropolis-Hastings RobbinsMonro algorithm using the R (version 3.6.3) package mirt. Model data were fit to assess questionnaire dimensionality, to estimate item information, and to score participants. Poorly functioning items were removed, and the model was refit to create the final questionnaire. center dot RESULTS: The psychometrically validated PROs measure was reduced to a 59-item questionnaire measuring 7 unidimesnional domains: central vision, color vision, contrast sensitivity, scotopic function, photopic peripheral vision, mesopic peripheral vision, and photosensitivity. A total of 39 items were removed because of poor factor loading, low item information, poor person ability differentiation, or high item-level interdependence. This novel questionnaire produces a reliable domain score for person ability that does not show significant test-retest variability across repeated administration. center dot CONCLUSIONS: The final PRO questionnaire, known as the Michigan Retinal Degeneration Questionnaire, is psychometrically validated and available for use in the evaluation of patients with inherited retinal degenerations. (Am J Ophthalmol 2021;222:60 & ndash;68.
- Construct Validity of Inherited Retinal Disease-Specific Patient-Reported Outcome Measures(2023) JAYASUNDERA, K. Thiran; ABUZAITOUN, Rebhi O.; POPOVA, Lilia; ABALEM, Maria Fernanda; ANDREWS, Chris A.; LACY, Gabrielle D.; FRESCO, David M.; MUSCH, David C.PURPOSE: To evaluate aspects of construct validity of the Michigan Retinal Degeneration Questionnaire (MRDQ) and the Michigan Vision-related Anxiety Ques-tionnaire (MVAQ). center dot METHODS: Subjects with a clinical diagnosis of an inherited retinal disease (IRD) were recruited prospec-tively and 3 tests were used to assess construct valid-ity: the ability to distinguish different IRD phenotypes; test a priori hypothesis of an association between vision-related anxiety and vision-related disabilities; and corre-late MRDQ and MVAQ with the National Eye Insti-tute Visual Functioning Questionnaire 25 (NEI VFQ-25) and the Impact of Vision Impairment (IVI). One-way analysis of variance (ANOVA) was used to com-pare different phenotypes for mean domain scores for MRDQ/MVAQ. Pearson correlations were performed be-tween; Cone-Function Anxiety and Central Vision con-trolling for better eye visual acuity, Rod-Function Anxi-ety and Scotopic Function controlling for visual field area (III4e and IV4e), and scores of MRDQ/MVAQ, NEI VFQ-25, and IVI. center dot RESULTS: The study sample consisted of 146 pa-tients evenly divided between males and females, and mean age was 50 years. The 1-way ANOVA test was significant for distinguishing IRD phenotypes in 6 do-mains of MRDQ/MVAQ. Cone-Function Anxiety corre-lated with Central Vision controlling for visual acuity, Rod-Function Anxiety correlated with Scotopic Func-tion controlling for visual field area, and all domains in MRDQ/MVAQ had significant correlations with NEI VFQ-25 and IVI composite scores. center dot CONCLUSION: MRDQ and MVAQ domenstrate aspects of construct-validity set forth by the US Food and Drug Administration. The study futher supports the use of both patient-reported outcome measures in IRD clinical trials and natural history studies. (Am J Ophthalmol 2023;248: 116-126. (c) 2022 Elsevier Inc. All rights re-served.)
- Positive feedback loop between vision-related anxiety and self-reported visual difficulty(2023) POPOVA, Lilia T.; ABUZAITOUN, Rebhi O.; FRESCO, David M.; ABALEM, Maria Fernanda; ANDREWS, Chris A.; MUSCH, David C.; EHRLICH, Joshua R.; JAYASUNDERA, K. ThiranBackgroundPatients with Inherited Retinal Diseases typically experience progressive, irreversible vision loss resulting in low vision and blindness. As a result, these patients are at high risk for vision-related disability and psychological distress, including depression and anxiety. Historically, the relationship between self-reported visual difficulty (encompassing metrics of vision-related disability and quality of life, among others) and vision-related anxiety has been regarded as an association and not a causal relationship. As a result, there are limited interventions available that address vision-related anxiety and the psychological and behavioral components of self-reported visual difficulty.Materials and MethodsWe applied the Bradford Hill criteria to evaluate the case for a bidirectional causal relationship between vision-related anxiety and self-reported visual difficulty.ResultsThere is sufficient evidence to satisfy all nine of the Bradford Hill criteria of causality (strength of association, consistency, biological gradient, temporality, experimental evidence, analogy, specificity, plausibility, and coherence) for the relationship between vision-related anxiety and self-reported visual difficulty.ConclusionsThe evidence suggests that there is a direct positive feedback loop-a bidirectional causal relationship-between vision-related anxiety and self-reported visual difficulty. More longitudinal research on the relationship between objectively-measured vision impairment, self-reported visual difficulty, and vision-related psychological distress is needed. Additionally, more investigation of potential interventions for vision-related anxiety and visual difficulty is needed.
- Patient-reported outcome measures in inherited retinal degeneration gene therapy trials(2020) LACY, Gabrielle D.; ABALEM, Maria Fernanda; MUSCH, David C.; JAYASUNDERA, Kanishka T.Patient-reported outcome (PRO) measures have the potential to uniquely capture patient experience and serve as an outcome measure in inherited retinal degeneration (IRD) gene therapy trials. An IRD-specific patient-reported outcome measure may yield valuable information that has not been obtained from inherited retinal dystrophy gene therapy trials published to-date. Existing PRO measures have inherent limitations for use in IRD gene therapy trials. Developing an applicable patient-reported outcome measure for such trials needs to incorporate patient input from the target population, demonstrate sound psychometric properties, and be made in accordance with U.S. Food and Drug Administration (FDA) guidelines. This review will discuss the currently available PRO instruments, their limitations for IRD therapeutic trials, and suggestions for future PRO development in IRD populations. The PRO instruments highlighted were identified in PubMed search of English-language journals and previously published review articles.
- Content generation for patient-reported outcome measures for retinal degeneration therapeutic trials(2020) LACY, Gabrielle D.; ABALEM, Maria Fernanda; POPOVA, Lilia T.; SANTOS, Erin P.; YU, Gina; RAKINE, Hanan Y.; ROSENTHAL, Julie M.; EHRLICH, Joshua R.; MUSCH, David C.; JAYASUNDERA, K. ThiranPurpose Generate content for a patient-reported outcome (PRO) measure for use in future clinical trials for inherited retinal degenerations. Methods Patients at the University of Michigan Kellogg Eye Center with a clinical diagnosis of inherited retinal degeneration with varying phenotypes were recruited for interviews. First, in-depth interviews were performed to solicit a wide range of patient experiences pertaining to visual function. Coders qualitatively analyzed the transcripts from these interviews using Atlas.ti software (Version 8.1.3 (522)) to draft questionnaire items. Next, the questionnaire was tested and refined based on participant feedback in cognitive interviews and administrator feedback in the pilot survey administration (pilot interviews). Results A total of 55 participants with a clinical diagnosis of inherited retinal degeneration were interviewed throughout the three study phases: in-depth interviews (n = 26), cognitive interviews (n = 16), and pilot interviews (n = 13). Coded items were analyzed for frequency of occurrence and related themes, then organized into common domains. Within each domain, PRO items were drafted to address the functional limitations or adaptations experienced by patients. Conclusions Items for a PRO measure have been drafted and evaluated for interpretability in the target inherited retinal degeneration patient population. Content validity for the items was established through a process of in-depth interviews, cognitive interviews, and pilot interviews.
- Diurnal variations of foveoschisis by optical coherence tomography in patients with RS1 X-linked juvenile retinoschisis(2018) ABALEM, Maria Fernanda; MUSCH, David C.; BIRCH, David G.; PENNESI, Mark E.; HECKENLIVELY, John R.; JAYASUNDERA, ThiranBackground: To evaluate diurnal variations in macular schisis cavities in patients with X-linked juvenile retinoschisis (XLRS) with pathogenic variants in the RS1 gene using spectral-domain optical coherence tomography (SD-OCT). Methods: Three consecutive patients with a clinical diagnosis of XLRS and pathogenic variants in the RS1, treated with carbonic anhydrase inhibitors (CAIs). Observational procedures: SD-OCT scans of the macula were acquired at 9 a.m., 1 p.m., and 4 p.m. within 24 h. Results: All patients demonstrated increased measures of central foveal thickness in the morning with gradual decrease through the day (9-43%). Major changes were observed between 9 a.m. and 1 p.m. in the central foveal thickness. Conclusion: The central foveal thickness varies during daytime hours in patients with XLRS. This finding may explain the inconsistent and heterogeneous responses to treatment with CAIs and necessitate standardization of measurement times in treatment trials for XLRS as well as in the routine ophthalmic evaluation of these patients.