A Pilot Study for Evaluation of Hypotonia in Children With Neurofibromatosis Type 1

Soucy, Elizabeth A.; Wessel, Lauren E.; Gao, Feng; Albers, Anne C.; Gutmann, David H.; Dunn, Courtney M.

doi:10.1177/0883073814531823

Cited by 8 publications

(21 citation statements)

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“…In genetic cases it is important to reach the accurate diagnosis so that counseling may be undertaken. The array of diagnoses to consider is increasingly challenging; the severity is variable and is dependent on the underlying causes and thus a careful and thorough approach is necessary [ 4 – 6 ]. The severity and progression of hypotonia vary with each child and their diagnosis.…”

Section: Introductionmentioning

confidence: 99%

“…Authors also indicate that often, despite a thorough workup, a diagnosis cannot be established [ 5 ] reiterating that this should however not compromise management. Moreover, the frequency of hypotonia and potential improvements with therapy [ 9 ] necessitates the need for more accurate and standardized measures [ 6 ]. The future of the floppy infant was cited in literature over the last century [ 10 , 11 ].…”

Section: Introductionmentioning

confidence: 99%

“…Confusion in terminology related to hypotonia was cited in these papers at that stage and continued to be a contentious diagnosis and somewhat erroneous clinical concept over the ensuing years [ 12 ]. Contention over the clinical assessment and use of terms continue to be debated into this century [ 5 , 6 , 13 – 15 ], leaving the clinical assessment of hypotonia open to scrutiny in addition to potential for errors.…”

Section: Introductionmentioning

confidence: 99%

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Evidence-Based Clinical Algorithm for Hypotonia Assessment: To Pardon the Errs

Govender

Joubert

2018

Occupational Therapy International

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Despite the many advances in diagnostics, the clinical assessment of children with hypotonia presents a diagnostic challenge for clinicians due to the current subjectivity of the initial clinical assessment. The aim of this paper is to report on an evidence-based clinical algorithm (EBCA) that was developed for the clinical assessment of hypotonia in children as part of the output of a multiphased study towards assisting clinicians in more accurate assessments. This study formed part of a larger advanced mixed methods design. The preceding phases of the study included a systematic review, a survey amongst clinicians, a consensus process (Delphi technique), and a qualitative critique with multiple focus groups. Samples were drawn from three professional groups (occupational therapists, physiotherapists, and paediatricians). Data were analysed at each stage and merged in the development of the EBCA. The EBCA followed a rigorous process of development and critique. The methods for formulating changes in the revision and development of the EBCA are presented together with a description and presentation of the final algorithm for practice. The overarching concepts that guided the development and refinement of the EBCA are described, taking into consideration knowledge translation, evidence-based practice, and the value of EBCAs in addition to recommendations for stakeholder uptake. The EBCA is envisaged to be useful in practice for clinicians who are faced with the assessment of a child that is suspected as having hypotonia via a systematic process in identifying specific characteristics that are associated with low muscle tone.

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Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Evidence-Based Clinical Algorithm for Hypotonia Assessment: To Pardon the Errs

Govender

Joubert

2018

Occupational Therapy International

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“…In recent years, the clinical assessment of hypotonia has re-emerged as contentious, given the wide range of diagnoses that have hypotonia as an underlying symptom 1,2,3. The initial clinical evaluation is essential in the diagnostic process that ensues and in determining appropriate management4.…”

Section: Introductionmentioning

confidence: 99%

AGREE-II Appraisal of a clinical algorithm for hypotonia assessment

Govender¹

2018

Afr H. Sci.

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ObjectiveThe objective of this study was to systematically appraise the quality of an evidenced-based clinical algorithm for the clinical assessment of hypotonia in children.DesignThe Appraisal of Guidelines for Research and Evaluation (AGREE) II tool with 23 items and six domains was used. The study was located in South Africa. Ten appraisers, who were recruited based on specific selection criteria, completed the assessment.ResultsNine appraisers recommended the EBCA without any modification. Scope and purpose (94%), stakeholder involvement (91%) and editorial independence (99%) were rated the highest with the lower scoring domains being clarity of presentation (85%) and applicability (86%) due to clarity required in areas of resource implications and auditing and monitoring criteria. Inter-rater reliability was strong (ICC 0.7) amongst the appraisers in this study.ConclusionThis is the first independent assessment of the methodological rigour and transparency of a clinical algorithm using the AGREE-II instrument. Determining the quality of the EBCA for practice is essential as this would ultimately aid clinicians towards more accurate clinical assessment of hypotonia which would inevitably impact outcomes and management of the child presenting with this symptom. Whilst the AGREE-II provided initial feedback on the methodological rigour of development, understanding that the AGREE-II instrument evaluates the guideline development process and not the content is also essential in order to consider the next stage which would be to consider clinicians feedback on the clinical utility of this EBCA.

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“…There are currently no standardised assessment tools for children (after infancy and during early childhood) with low muscle tone (Soucy et al . 2015 ). Moreover, the incidence of hypotonia is difficult to ascertain given the fact that it is a symptom of a number of conditions or disorders (Lisi & Cohn 2011 ).…”

Section: Introductionmentioning

confidence: 99%

‘Toning’ up hypotonia assessment: A proposal and critique

Govender

Joubert

2016

AJOD

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BackgroundClinical assessment of hypotonia is challenging due to the subjective nature of the initial clinical evaluation. This poses dilemmas for practitioners in gaining accuracy, given that the presentation of hypotonia can be either a non-threatening or malevolent sign. The research question posed was how clinical assessment can be improved, given the current contentions expressed in the scientific literature.ObjectivesThis paper describes the development and critique of a clinical algorithm to aid the assessment of hypotonia.MethodsAn initial exploratory sequential phase, consisting of a systematic review, a survey amongst clinicians and a Delphi process, assisted in the development of the algorithm, which is presented within the framework of the International Classification of Functioning, Disability and Health. The ensuing critique followed a qualitative emergent–systematic focus group design with a purposive sample of 59 clinicians. Data were analysed using semantical content analysis and are presented thematically with analytical considerations.ResultsThis study culminated in the development of an evidence-based clinical algorithm for practice. The qualitative critique of the algorithm considered aspects such as inadequacies, misconceptions and omissions; strengths; clinical use; resource implications; and recommendations.ConclusionsThe first prototype and critique of a clinical algorithm to assist the clinical assessment of hypotonia in children has been described. Barriers highlighted include aspects related to knowledge gaps of clinicians, issues around user-friendliness and formatting concerns. Strengths identified by the critique included aspects related to the evidence-based nature of the criteria within the algorithm, the suitability of the algorithm in being merged or extending current practice, the potential of the algorithm in aiding more accurate decision-making, the suitability of the algorithm across age groups and the logical flow. These findings provide a starting point towards ascertaining the clinical utility of the algorithm as an essential step towards evidence-based praxis.

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A Pilot Study for Evaluation of Hypotonia in Children With Neurofibromatosis Type 1

Cited by 8 publications

References 10 publications

Evidence-Based Clinical Algorithm for Hypotonia Assessment: To Pardon the Errs

Evidence-Based Clinical Algorithm for Hypotonia Assessment: To Pardon the Errs

AGREE-II Appraisal of a clinical algorithm for hypotonia assessment

‘Toning’ up hypotonia assessment: A proposal and critique

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