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Development of the neurotrophic keratopathy questionnaire: qualitative research.

ABSTRACT: BACKGROUND:Neurotrophic keratopathy/keratitis (NK) is a rare disease of the cornea that can lead to anatomical loss of the eye. Little is known about the NK experience from the patients' perspective. The objectives of this study were to examine the symptomatic experience and impacts of NK on patients and assess the overall comprehension, relevance, and content validity of a new questionnaire. METHODS:This was a cross-sectional, qualitative study conducted with NK patients with varying levels of disease severity, recruited from one clinical site. One-on-one interviews using concept elicitation and cognitive interviewing techniques were conducted. RESULTS:Fourteen NK patients participated; 64.3% were female (n?=?9), mean age was 65.7?±?13.3, and 14.3% (n?=?2), 21.4% (n?=?3), and 64.3% (n?=?9) were classified as Mackie stage I, stage II, or stage III, respectively. Participants reported 24 concepts, including: redness (n?=?12, 86%), sensitivity to light (n?=?11, 79%), general discomfort (n?=?9, 64%), dry eye (n?=?9, 64%), reduced visual acuity (n?=?9, 64%), blurred vision (n?=?8, 57%), and eye fatigue (n?=?8, 57%). No new concepts were reported after the 13th interview. The most frequently reported impacts included frustration (n?=?10, 71%), driving impairment (n?=?8, 57%), reading impairment (n?=?7, 50%), difficulty watching television (n?=?7, 50%), and concern with potentially losing their eyesight due to NK (n?=?6, 43%). Participants provided positive feedback on the draft NK Questionnaire (NKQ) and felt that it was comprehensive and relevant to their experience with NK. Additionally, the recall period, instructions, item concepts, and response options were well-understood by participants. Minor revisions were made to the tool for consistency (i.e., the timeframe "in the past 7 days" was added to items 12-14); item 14 was modified to include "how often"; examples were added to item 9. CONCLUSIONS:The results of the concept elicitation portion of the qualitative study support the content validity of the draft NKQ. The clinically significant concepts identified in the literature and raised during concept elicitation are included as items in the questionnaire. Further assessment of the psychometric properties should be conducted in support of this new tool to measure the effect of new treatments on symptoms and impacts associated with NK.

SUBMITTER: Murray LT

PROVIDER: S-EPMC7198680 | biostudies-literature | 2020 May

REPOSITORIES: biostudies-literature

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Development of the neurotrophic keratopathy questionnaire: qualitative research.

Murray Lindsey T LT McCormack Julie J Grobeiu Ioana I Wiklund Ingela I Kimel Miriam M Van Nooten Floortje F

Journal of patient-reported outcomes 20200504 1

<h4>Background</h4>Neurotrophic keratopathy/keratitis (NK) is a rare disease of the cornea that can lead to anatomical loss of the eye. Little is known about the NK experience from the patients' perspective. The objectives of this study were to examine the symptomatic experience and impacts of NK on patients and assess the overall comprehension, relevance, and content validity of a new questionnaire.<h4>Methods</h4>This was a cross-sectional, qualitative study conducted with NK patients with var ...[more]

PMID: 32367451

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Project description:PurposeNeurotrophic keratopathy is a degenerative disease characterized by damage to the corneal nerves leading to corneal hypoesthesia and anaesthesia. The resultant progressive visual deterioration is refractory to existing conventional treatment options. Corneal neurotization is a novel and effective surgical procedure that directly targets the underlying pathology of nerve loss by stimulating new corneal nerve growth. This study reports the outcomes and the pre- and postoperative in vivo confocal microscopy findings of the first published Australian case of indirect, minimally invasive, corneal neurotization using an ipsilateral sural nerve autograft.ObservationsAn 11-year-old boy developed corneal hypoesthesia in the left eye following surgical debulking of a cerebellopontine angle arachnoid cyst. He was diagnosed with Mackie Stage 1 neurotrophic keratopathy. Due to his hypoesthesia, he had developed recurrent microbial keratitis and corneal ulceration secondary to foreign bodies sustained during contact sports. At presentation, he reported photophobia and dry eye symptoms, corrected-distance visual acuity was 6/18, Cochet-Bonnet aesthesiometer demonstrated reduced corneal sensation (5-15mm), Schirmer's I test was 15mm, and in vivo confocal microscopy showed a complete absence of a subepithelial corneal plexus. He underwent indirect, minimally invasive, corneal neurotization using the ipsilateral supratrochlear nerve and a sural nerve autograft. Subjective improvement in corneal sensation was noticed by the patient at 2 months. Objective improvement, measured on Cochet-Bonnet aesthesiometer, was first observed at 6 months with steady stepwise improvement to 20-35mm at 21 months. Importantly, due to the increase in corneal sensation, the patient did not develop any further corneal complications. At 12 months, dry eye symptoms resolved and Schirmer's I test improved to 30mm. At 15 months, corrected-distance visual acuity improved to 6/5 and in vivo confocal microscopy demonstrated evidence of corneal reinnervation with nerves running through the subepithelial space surrounded by healthy and active keratocytes.Conclusions and importanceCorneal neurotization represents an exciting development in the armamentarium for the treatment of neurotrophic keratopathy and can be considered for younger patients with early-stage disease.

Project description:Neurotrophic Keratopathy (NK) is a neurodegenerative corneal disease that results in diminished corneal sensation. Previous studies have found that Cenegermin 0.002%, a recombinant human nerve growth factor (rhNGF), improves corneal epithelial healing in stage 2 and 3 NK patients. However, rhNGF effect on corneal sensation and nerve regeneration has not been well established. Thus, this study aims to analyze the effect of rhNGF on corneal nerve regeneration using in vivo confocal microscopy (IVCM) and on corneal sensitivity in NK patients. This is a retrospective, longitudinal, case-control study that included patients with NK, treated with rhNGF for at least 4 weeks, with pre- and post-treatment IVCM images available for analysis. Chart reviews were conducted documenting prior medical and surgical history, clinical signs and symptoms, and corneal sensation using Cochet-Bonnet esthesiometry. Corneal nerve parameters were assessed by IVCM. Sex- and age-matched reference controls were selected from a database of healthy subjects for comparison. The study included 25 patients, with 22 (88%) stage 1, two (8%) stage 2, and 1 (4%) stage 3 NK patients, with a median age of 64 years (range: 30-93 years). Total, main, and branch nerve densities [median (range) in mm/mm2] were lower in the NK group pre-treatment [2.3 (0.0-21.1); 1.7 (0.0-13.0); 0.5 (0.0-10.2); respectively] vs. controls [22.3 (14.9-29.0); 10.1 (3.2-15.4); and 12.1 (6.2-18.4), (p < 0.0001 for all), respectively]. Post-treatment nerve densities increased compared to pre-treatment to 5.3 (0.0-19.4, p = 0.0083) for total, 3.5 (0.0-13.2, p = 0.0059) for main, and 2.0 (0.0-10.4, p = 0.0251) for branch nerves, but remained lower than controls (p < 0.0001 for all). Corneal sensation increased from 2.3 ± 1.1 cm pre-treatment to 4.1 ± 1.4 cm post-treatment (p = 0.001). Median best corrected visual acuity significantly increased following rhNGF treatment from 0.4 (0.0-1.6) to 0.12 (-0.1 to 1.6) (p = 0.007). Patients with NK treated with at least 4 weeks of rhNGF, showed a significant increase in corneal nerve densities after treatment. A significant increase in corneal sensation, as well as best corrected visual acuity, was observed following treatment.

Project description:Bridging social capital is defined as the connections between individuals who are dissimilar with respect to socioeconomic and other characteristics. There is an important gap in the literature related to its measurement. We describe the development and validation of a questionnaire to measure bridging social capital. We focused the development of the questionnaire to be suitable for use in Latino immigrant populations in the U.S. The structure of the questionnaire comprised the following: Socialization in the job place (5 items); Membership in community activities (16 items); Participation in community activities (5 items); Contact with similar/different people (7 items); Assistance (17 items); Trust of institutions, corporations and other people(14 items); and Trust of intimate people (3 items). First, we used focus groups (N=17 participants) to establish content validity with an inductive thematic analysis to identify themes and subthemes. Changes were made to the questionnaire based on difficulty, redundancy, length and semantic equivalence. Second, we analyzed the questionnaire's psychometric properties (N=138). We tested internal consistency with Cronbach alpha and construct validity with a Confirmatory Factor Analysis (CFA) for each sub-scale to test theoretical unity; discriminant validity to observe differences between participants from high and low SES backgrounds and different language; and content validity with an independent expert panel. Cronbach alphas ranged from 0.80 (Assistance) to 0.92 (Trust). CFA results indicated that CFI and TLI were higher than 0.90 in almost all the scales, with high factor loadings. The Wilcoxon tests indicated that there were statistically significant mean differences between SES and language groups (p<0.00). The independent expert panel determined that the questionnaire had good content validity. This is the first demonstration of a psychometrically validated questionnaire to measure bridging social capital in an immigrant population in the United States. Our questionnaire may be suitable for further refinement and adaptation to other immigrant groups in different countries.

Project description:Despite the growing interest in understanding the psycho-social impact of rare genetic diseases, few studies examine this concept and even fewer seek to obtain feedback from families who have lived the experience. The aim of this project was to involve families of children living with osteogenesis imperfecta (OI) in the development of a tool to assess the impact of OI on the lives of patients and their families.This project used an integrated knowledge translation approach in which knowledge users (clinicians and people living with OI and their families) were consulted throughout the four steps of development, that is: content mapping, item generation, tool appraisal and pre-testing of the questionnaires. The International Classification of Functioning and Health was used as a framework for content mapping. Based on a scoping review we selected two validated tools to use as a basis for developing the questionnaire. The final parent self-report version measured six domains: experience of diagnosis; use of health services; use of social and psychological support services; expectations about tertiary specialized centers; and socio-demographic information.A total of 27 out of 40 families receiving care at the Shriners Hospital for Children-Canada and invited to participate in the pre-test returned the completed questionnaires. In more than two-thirds of families (69%; n = 18) OI was suspected either at or within the first 3 months after birth. Up to 46% of families consulted between 3 and 5 doctors (46%; n = 12) prior to final diagnosis. The use of services by families varied from 0 to 16 consultations, 0 to 9 exploratory examinations and 1 to 10 types of allied health services. In the 12 months prior to the study, fewer than a quarter of children had been admitted, for treatment, for hospital stays of longer than 8 hours or to an emergency department (24% and 9% respectively). Only 29% of parents received psychological support.This joint development process generated a tool, with good psychometric properties, that provides unique insight into the experiences of patients and families with OI, the psycho-social impact of the illness, and their service needs and expectations.

Dataset Information

Development of the neurotrophic keratopathy questionnaire: qualitative research.

Publications

Development of the neurotrophic keratopathy questionnaire: qualitative research.

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