Clinical Cases in Dysphagia

Margaret Walshe, Maggie-Lee Huckabee, Margaret Walshe, Maggie-Lee Huckabee

• Publisher: Routledge
• Year: 2018
• ISBN:
  9781351615839
• Edition: 1
• Topic: Medicine
• Subtopic: Audiology & Speech Pathology

Dysphagia is a complex condition that can have significant social, developmental and psychological effects. Alongside the physiology and pathophysiology of the condition, dysphagia can rob patients of the most basic pleasures, such as eating and drinking, causing ongoing difficulties for individuals in social situations throughout the lifespan.

As an acknowledged component of evidence-based practice, the humble case report encourages clinical reflection and may be the spark that generates new thinking and motivation for future research. Clinical Cases in Dysphagia provides an opportunity to gain insight into the unique and varied presentation and management of dysphagia across a range of different conditions. With chapters provided by expert clinicians and based on clinical examples 'from the trenches', the reader may gain insights into their own practice patterns, refining their clinical problem solving and valuing the education that is offered to each of us by our patients.

With additional online resources to support the case-based approach, the book emphasizes the importance of multidisciplinary care and reflects everyday clinical practice, making it a must-read for clinicians and students.

1 Case reports in dysphagia

An introduction

Margaret Walshe and Maggie-Lee Huckabee

Introduction

In the perfect world of clinical service delivery, all of our practices for diagnosis and rehabilitation would be fully supported by multiple, high-level, randomized controlled trials with consequent systematic reviews to provide assurance of efficacy. But our clinical practices are far from perfect and our evidence is far from comprehensive. Most descriptions of evidence-based practice thus allow the ‘[integration of] individual clinical expertise with the best available external clinical evidence from systematic research’ (Sackett et al. 1996, p. 71). The valuing of clinical expertise is never intended to negate the power of empirical research, but does honour contribution of even a single clinical observation to spark the motivation for further investigation. Without the initial insights offered by a single patient, our well-supported practices would never have emerged. Therefore, although considered the lowest level of evidence, the humble case report in some respects is the origin of new thinking.

The aim of this chapter is to clarify use of the term ‘case report’, describe various types of case reports and emphasize the importance of using these to advance our clinical knowledge and practice in dysphagia. We provide a proposed template for writing case reports and some advice on publishing these for dissemination.

Terminology

‘Case study’, ‘case report’, ‘grand round’, ‘case series’, ‘clinical case description’ are all used interchangeably in the literature essentially to describe the same thing. In this text we distinguish between case studies and case reports. In case reports, changes in outcomes or responses to intervention cannot be explicitly attributed to an intervention and there are no attempts to control for extraneous factors. Unlike case studies and ‘n of 1’ single study designs, case reports tend not to be planned or controlled but are rather a description of events as they occurred, in order to make a specific point (see Lillie et al. 2011). Case studies and n of 1 trials are discussed further in Chapter 10 by Murray in this text, and the term ‘single case study’ is reserved for research that is consistent with the experimental n of 1 design.

Importance of case reports

Case reports are important. Packer (2017, p. 4) suggests that ‘case reports are all about novelty, serendipity, new ideas, fresh hypotheses and therapeutic surprises. Rather than provide confirmation, they provide inspiration’. Case reports can be considered naturalistic in that they involve the examination or exploration of cases in natural real-life contexts. Thus, they are excellent reflections of patient care, describing unusual presentations or exploring management approaches in real-life contexts while providing insight into facets of intervention frequently missed in larger research studies and clinical trials. They reflect the typical working environment rather than that of a research laboratory. They help generate an in-depth understanding of a specific topic to create knowledge and inform patient management. Case reports are considered important in recognizing and describing new or rare conditions, diseases or manifestation of disorders. There are peer-reviewed journals that are devoted entirely to case reports. Some of these, for example, Respiratory Medicine Case Reports, Case Reports in Neurological Medicine, Case Reports in Otolaryngology, may be of particular relevance to clinicians working in dysphagia.

Case reports serve a number of purposes. They can be used to convey a key message for other clinicians encountering similar individuals. This may be to draw attention to incidents where dysphagia is an initial sign or symptom of a serious underlying condition that is not immediately obvious. One example is a case report by Simmons and Bursaw (2015) describing the unusual presentation of a 61-year-old man with acute-onset dysphagia and progressive neuropathology that were ultimately associated with lymphoma and treated accordingly. Prompt diagnosis of the lymphoma was critical to the patient’s medical outcome. Case reports are also used to describe the presentation and management of dysphagia in rare conditions. Lewis et al. (2015) describe the assessment and management of dysphagia in a person with Eagle’s syndrome, a rare condition associated with abnormal length and positioning of the styloid process.

Case reports can further provide an in-depth exploration of the intricacy and individuality of a particular case in a real-life context from multiple perspectives, helping inform policy development and/or to direct and change professional practice. They may be used to help other clinicians learn from the errors of the authors (Sanei-Moghaddam et al. 2013) or challenge myths in clinical practice.

Case reports have helped us build and expand theoretical models to explain phenomena. One example of this is in the field of aphasia where cognitive neuropsychological models of language impairment were tested and developed based on phenomena exposed through single cases of individuals with impaired function (Ellis et al. 1983).

Finally, case reports assist in academic teaching by contributing to problem-based education for health- and social care professionals. Coyle et al. (2007), for example, use three case reports exemplifying both common and unusual clinical dilemmas in dysphagia to illustrate the process of evidence-based clinical problem-solving.

Types of case reports

There are many different types of case report and these may vary from short notes or a brief commentary to a more detailed patient description. Case reports can be categorized as follows:

Instrumental case: here the individual described is acting as in ‘instrument’ and used to illustrate a particular point, perhaps to a target audience or to provide a focal point for discussion on a specific management approach. One example is description by Joshi et al. (2008) of a 46-year-old woman with dermatomyositis and dysphagia. The clinical case report is used to discuss the association between dysphagia and dermatomyositis, and the potential treatment options available. A further example is a case report by Garcia-Carretero et al. (2016) describing a case where dysphagia was the only sign of a brainstem stroke.

Collective case: this involves studying more than one case simultaneously or sequentially in an attempt to provide increased support for a particular argument or theory. One example may be Thompson-Henry and Braddock’s (1995) use of five case reports to highlight the failure of the modified Evans blue dye test to detect aspiration in patients with tracheostomy. A more recent example is a description of a series of patients presenting with a previously undescribed abnormal pharyngeal motility pattern by Huckabee et al. (2014). This report highlights the importance of extending diagnostic modalities and provides speculative discussion of the aetiology of this type of dysphagic presentation.

Evaluative case: this is used to illustrate how well some specific approach is working. This could, for example, be a team approach to management or an intervention approach that has worked effectively. Perhaps this approach has involved a change to some facet of a more traditional management approach. Case reports are useful to demonstrate the result of this change, which may be positive, negative or neutral.

Explanatory cases: these aim to provide a depth of understanding on a particular issue providing potential explanations of perhaps some paradoxical finding. For example, a person with Parkinson’s disease comes to the clinic with no report of difficulty swallowing but has aspirated on a routine barium swallow performed to investigate oesophageal dysmotility. This person has no respiratory signs of aspiration. She later aspirates on videofluoroscopy but is not symptomatic for aspiration. The ensuing case report might propose a theoretical framework to explain this absence of respiratory symptoms of aspiration in the patient. This explanatory case report may argue that the videofluoroscopy is not representative of her swallow function, that she is very physically active and walks for an hour each day with excellent baseline expiratory function. These explanatory case reports provide an excellent starting point for further well-designed prospective research studies.

Reporting clinical cases

The reader is directed here to a useful text by Packer et al. (2017) on writing case reports. It is recommended that authors consider first why they are writing the case report. Is this an exploration of some specific issue in dysphagia, an evaluation of a specific management approach or an attempt to explain some clinical phenomenon? Having identified a suitable journal or ‘home’ for this case report, the next task is to amalgamate the data and write the report. Many journals also require a patient consent form if the person in the case report is readily identifiable. This must be obtained in advance of submitting the report for publication.

Gagnier et al. (2013, p. 223) suggest that ‘case reports written without guidance from reporting standards are insufficiently rigorous to guide clinical practice or inform clinical study design’. There are reporting standards for most types of studies. Templates for these are published on the Equator Network (see www.equator-network.org). Case reports are no different. Some journals, such as the British Medical Journal, have specific guidelines on writing case reports. These must obviously be followed when submitting manuscripts to these journals. Many journals also require a patient consent form but all attempts to protect the person’s identity must be made with no identifying information in the case report.

Most journals adhere to the CAse REport (CARE) reporting guidelines. These were developed to improve the quality of case reports so that they can be of greater use to clinicians, patients and researchers (see Gagnier et al. 2013). An adapted version of the template is provided in Table 1.1.

Table 1.1 CARE guidelines (adapted checklist for case reports in dysphagia)

Conclusion

Finally, it would be short-sighted not to recognize some limitations of case reports. Patients are typically studied in an uncontrolled environment and there are many confounding variables that can affect outcomes and case presentation. Authors should therefore not attempt to generalize findings to other similar populations but rather provide direction for further exploration if required.

In conclusion, case reports are important sources of information for clinical practice and future research. The case reports in this textbook are predominantly instrumental and evaluative case reports, supplemented with online resources to achieve a greater depth of insight into the management of the person with dysphagia. Each case report conveys a specific message to the reader and we encourage readers to use these as a means of reflection on current and future dysphagia practice.

References

Coyle JL, Easterling C, Lefton-Greif M & Mackay L 2007, ‘Evidence-based to reality-based dysphagia practice: three case studies’, The ASHA Leader, 12(14), 10–32.

Ellis AE, Miller D & Sin G 1983, ‘Wernicke’s aphasia and normal language processing: A case study in cognitive neuropsychology’, Cognition, 15(1–3) 111–144.

Gagnier JJ, Kienle G, Altman DG, Moher D, Sox H, Riley D & the CARE Group 2013, ‘The CARE Guidelines: Consensus-based Clinical Case Reporting Guideline Development’, Global Advances in Health and Medicine, 2(5), 38–43.

Garcia-Carretero R, Bruguera M, Rebollo-Aparicio N & Rodeles-Melero J 2016, ‘Dysphagia and aspiration as the only manifestations of a stroke’, BMJ Case Reports, published online 11 February,.

Huckabee ML, Lamvik K & Jones R 2014, ‘Pharyngeal mis-sequencing in dysphagia: characteristics, rehabilitative response, and etiological speculation,’ Journal of Neurological Science, 15(343), 153–158.

Joshi D, Mahmood R, Williams P & Kitchen P 2008, ‘Dysphagia secondary to dermatomyositis treated successfully with intravenous immunoglobulin: a case report’. International Archives of Medicine, 1, 12.

Lewis V, Hoffman Ruddy B, Lehman J, Silverman E & Spector B 2015, ‘Management of dysphagia post operatively in a case of Eagle’s syndrome’, Case Reports in Otolaryngology, Article ID 305736.

Lillie EO, Patay B, Diamant J, Issell B, Topol EJ & Schork NJ 2011, ‘The n-of-1 clinical trial: the ultimate strate...