Respiratory muscle training (RMT) in late-onset Pompe disease (LOPD): Effects of training and detraining.

BACKGROUND: Determine the effects of a 12-week respiratory muscle training (RMT) program in late-onset Pompe disease (LOPD). METHODS: We investigated the effects of 12-weeks of RMT followed by 3-months detraining using a single-subject A-B-A experimental design replicated across 8 adults with LOPD. To assess maximal volitional respiratory strength, our primary outcomes were maximum inspiratory pressure (MIP) and maximum expiratory pressure (MEP). Effect sizes for changes in MIP and MEP were determined using Cohen's d statistic. Exploratory outcomes targeted motor function, and peak cough flow (PCF) was measured in the last 5 subjects. RESULTS: From pretest to posttest, all 8 subjects exhibited increases in MIP, and 7 of 8 showed increases in MEP. Effect size data reveal the magnitude of increases in MIP to be large in 4 (d≥1.0) and very large in 4 (d≥2.0), and effect sizes for increases in MEP were large in 1 (d≥1.0) and very large in 6 (d≥2.0). Across participants, pretest to posttest MIP and MEP increased by a mean of 19.6% (sd=9.9) and 16.1% (sd=17.3), respectively. Respiratory strength increases, particularly for the inspiratory muscles, were generally durable to 3-months detraining. CONCLUSIONS: These data suggest our 12-week RMT program results in large to very large increases in inspiratory and expiratory muscle strength in adults with LOPD. Additionally, increases in respiratory strength appeared to be relatively durable following 3-months detraining. Although additional research is needed, RMT appears to offer promise as an adjunctive treatment for respiratory weakness in LOPD.

Letter-by-letter reading: natural recovery and response to treatment.

The present investigation provides a longitudinal study of an individual (RB) with acquired alexia following left posterior cerebral artery stroke. At initial testing, RB exhibited acquired alexia characterized by letter-by-letter (LBL) reading, mild anomic aphasia, and acquired agraphia. Repeated measures of reading accuracy and rate were collected for single words and text over the course of one year, along with probes of naming and spelling abilities. Improvements associated with natural recovery (i.e., without treatment) were documented up to the fourth month post onset, when text reading appeared to be relatively stable. Multiple oral reading (MOR) treatment was initiated at 22 weeks post-stroke, and additional improvements in reading rate and accuracy for text were documented that were greater than those expected on the basis of spontaneous recovery alone. Over the course of one year, reading reaction times for single words improved, and the word-length effect that is the hallmark of LBL reading diminished. RB's response to treatment supports the therapeutic value of MOR treatment to in LBL readers. His residual impairment of reading and spelling one-year post stroke raised the question as to whether further progress was impeded by degraded orthographic knowledge.

A five-phase model for clinical-outcome research.

UNLABELLED: Through a variety of approaches, speech-language pathologists and audiologists have produced strong evidence that treatments are generally potent. However, we have largely ignored the accepted standards for clinical-outcome testing used throughout the broader research community (e.g., by other clinical disciplines, federal regulators, and third-party payers). Several clinical professions recognize a comprehensive model for organizing and scaffolding the many forms of clinical-outcome research. An adaptation of this five-phase model of clinical-outcome research is examined as a means for structuring forms of clinical research throughout audiology and speech-language pathology. Within the organizing structure, relationships become apparent between types and grades of scientific evidence and the processes underpinning evidence-based practice which ultimately lead to decisions on the status of intervention protocols. LEARNING OUTCOMES: Readers will be able to distinguish the phases of clinical-outcome research in a comprehensive model. Readers will be able to identify relationships between the structure of the model and broadly recognized concepts associated with the terms 'efficacy' and 'effectiveness.' Readers will be able to identify indicators of quality for controlled clinical trials.

Reporting point and interval estimates of effect-size for planned contrasts: fixed within effect analyses of variance.

UNLABELLED: The purpose of this tutorial is threefold: (a) review the state of statistical science regarding effect-sizes, (b) illustrate the importance of effect-sizes for interpreting findings in all forms of research and particularly for results of clinical-outcome research, and (c) demonstrate just how easily a criterion on reporting effect-sizes in research manuscripts can be accomplished. The presentation centers on within-effect analyses of variance including the one-way design for testing pre-post hypotheses and the two-way parallel-groups design for making direct comparisons of competing treatment protocols (e.g., experimental treatment versus control). The presentation is supported with worked examples and a web site containing templates for software applications. EDUCATIONAL OBJECTIVES: The reader will be able to: (1) explain the rationale for the increased use of estimates of effect-size in reporting results in published research manuscripts; (2) describe what an effect-size is (generally considered) and provide a rationale for its importance; (3) distinguish among the many forms of effect-size and apply their features to the most appropriate choices under specific research circumstances; and (4) appropriately report and interpret effect-sizes.

Ethical principles associated with the publication of research in ASHA's scholarly journals: importance and adequacy of coverage.

PURPOSE: The purpose of this 2-part study was to determine the importance of specific topics relating to publication ethics and adequacy of the American Speech-Language-Hearing Association's (ASHA's) policies regarding these topics. METHOD: A 56-item Web-based survey was sent to (a) ASHA journal editors, associate editors, and members of the Publications Board (Group 1); (b) authors, reviewers, and members of ASHA's Board of Ethics (Group 2); and (c) a random sample of the ASHA membership, characterized as journal readers (Group 3). The survey contained 4 questions related to ethical principles associated with the publication of research: (a) In regard to scientific integrity in research publications in general, how important is the issue of [topic]? (b) Should ASHA publication policies address this issue? (c) Do ASHA policies address this issue? (d) If yes, how adequately do ASHA policies address this issue? A second study evaluated the contents of ASHA's publication policy documents in regard to their coverage of the survey topics. RESULTS: Results indicated many of the topics deemed most important by all groups were included in ASHA's publication policy documents; other topics, although included, were not adequately addressed. CONCLUSIONS: ASHA needs a single, unifying publication policy document, and increased education of all groups in the realm of ethics in the publication process is indicated.

Responsible conduct of research in communication sciences and disorders: faculty and student perceptions.

PURPOSE: Two Web-based surveys (Surveys I and II) were used to assess perceptions of faculty and students in Communication Sciences and Disorders (CSD) regarding the responsible conduct of research (RCR). METHOD: Survey questions addressed 9 RCR domains thought important to the responsible conduct of research: (a) human subjects protections; (b) research involving animals; (c) publication practices and responsible authorship; (d) mentor/trainee responsibilities; (e) collaborative science; (f) peer review; (g) data acquisition, management, sharing, and ownership; (h) conflicts of interest; and (i) research misconduct. Respondents rated each of 37 topics for importance and for sufficiency of instructional coverage. RESULTS: Respondents to Survey I were 137 faculty members from 68 (26%) of the 261 graduate programs in CSD. By comparison, 237 students from 39 (15%) programs responded to Survey II. Data about the importance and sufficiency of coverage of each of the 37 items were transformed into z scores to reveal relative ratings among the 37 topics. Data presentations were grouped for topics in each of the 9 RCR domains. Ratings indicated the relatively high importance assigned among the 37 topics by CSD faculty and students. Sufficiency of coverage of those same topics received lower ratings. CONCLUSIONS: The results of these surveys support the notion that students in CSD perceive that they are receiving information about RCR. The data pertaining to sufficiency of coverage provide a basis for improving instruction in this important aspect of research education.

Evaluating single-subject treatment research: lessons learned from the aphasia literature.

The mandate for evidence-based practice has prompted careful consideration of the weight of the scientific evidence regarding the therapeutic value of various clinical treatments. In the field of aphasia, a large number of single-subject research studies have been conducted, providing clinical outcome data that are potentially useful for clinicians and researchers; however, it has been difficult to discern the relative potency of these treatments in a standardized manner. In this paper we describe an approach to quantify treatment outcomes for single-subject research studies using effect sizes. These values provide a means to compare treatment outcomes within and between individuals, as well as to compare the relative strength of various treatments. Effect sizes also can be aggregated in order to conduct meta-analyses of specific treatment approaches. Consideration is given to optimizing research designs and providing adequate data so that the value of treatment research is maximized.