- Email: [email protected]
Towards improving the reporting quality of clinical case reports in complementary medicine: Assessing and illustrating the need for guideline development
Complementary Therapies in Medicine (2015) 23, 141—148
Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.elsevierhealth.com/journals/ctim
Towards improving the reporting quality of clinical case reports in complementary medicine: Assessing and illustrating the need for guideline development R.A. van Haselen ∗ International Institute for Integrated Medicine, 22 Eden Street, Suite 467, Kingston KT1 1DN, United Kingdom Available online 19 January 2015
KEYWORDS Clinical case reports; Reporting guideline development
∗
Summary Background: Case reports have had a varying level of recognition as a source of evidence throughout the history of medicine. In recent years, there has been a revival of interest in clinical case reports in both conventional and complementary medicine. There is a need to further improve the reporting quality of clinical case reports of different Complementary and Alternative Medicine (CAM) therapies. Objectives: To provide an overview of the different objectives for clinical case reports, identify those that are most relevant for CAM, and to develop a conceptual framework for purpose orientated clinical case reporting guidelines for CAM therapies. To practically illustrate the chosen approach by developing a clinical case reporting guideline for homeopathic cases. Methods: The various objectives of clinical case reports were described by Prof. Milos Jenicek, and the potential relevance of these objectives for CAM were discussed and graded by a mixed panel of experts. A conceptual framework for developing clinical case reporting guidelines for CAM treatments with specific objectives is proposed. The aim is to integrate both ‘generic’ and ‘CAM therapy specific’ quality items. This framework has been practically applied to the development of a reporting guideline for clinical case reports in homoeopathy which will be reported in a second article. Results: An overview is given of the clinical case reporting literature. The conceptual framework for the development of purpose orientated CAM clinical case reporting guidelines is presented. This framework is based on alignment with the recently published ‘generic’ CARE guideline for reporting of clinical case reports, whilst addressing the CAM specific elements at the same time. Conclusions: The scope and importance of clinical case reporting guideline development in CAM is illustrated. A conceptual framework for developing CAM specific clinical case reporting
Tel.: +44 2081449524. E-mail address: [email protected]
http://dx.doi.org/10.1016/j.ctim.2015.01.009 0965-2299/© 2015 Elsevier Ltd. All rights reserved.
142
R.A. van Haselen guidelines was established. It has been implemented using homoeopathy as an illustration, and this will be reported in a separate article. Further improvements in clinical case reporting in CAM will greatly contribute to CAM research and education, as well as to improved patient care. © 2015 Elsevier Ltd. All rights reserved.
Introduction Case reports have had a variable level of recognition as a source of evidence throughout the history of medicine. Whilst case reports have remained consistently important as part of teaching medicine, the value attributed to case reports as a source of evidence has been greatly diminished since the predominant emergence of modern, evidence based medicine in the second half of the 20th century.1 Within this framework several ‘hierarchies’ of evidence have been developed for assessing the quality of evidence, The US Preventive Services Task Force and the Oxford Centre for Evidence-Based Medicine schemes being prominent examples. In each of these, case reports feature low in, or at the bottom of, the hierarchy. The hierarchical approach towards evidence has been criticised as being too one-sidedly focused only on the perspective of establishing causal attribution to a treatment within the context of assessing efficacy. Whilst from this perspective clinical case reports are justifiably graded as weak evidence, from another perspective such as for instance safety assessment, case reports are often the first ‘signals’ of potential adverse drug reactions and sometimes single cases can even contribute significantly to establishing a causal link. In particular in the CAM community, nonhierarchical systems have been propagated, using words and metaphors such as an ‘evidence house’,2 ‘evidence profile’,3 ‘evidence mosaic’,4 or a ‘circular’ approach towards evidence.5 At the core of the above approaches is the notion that the hierarchy of evidence depends on the principal research objective, and as illustrated by the above example, the value of case reports is different in safety assessment as compared to efficacy assessment. Furthermore case reports can provide valuable insights in the development and cure of diseases in the course of time. The latter is also referred to as ‘disease evolution’ and this concept has been developed in various CAM systems such as for instance classical homeopathy6 and bioregulatory medicine.7 These research purposes can be categorised further using dimensions such as ‘hypothesis confirmation’ versus ‘hypothesis generation, ‘ideal world’ versus ‘real world’ and ‘absolute’ versus ‘relative’ effects.8 Within such a framework, case reports are particularly appropriate/strong methods for hypothesis generation in the ‘real world’ setting of daily medicine. The main pioneer who contributed to a re-appraisal of the millennia old art of case observation and case reporting in medicine is Prof. Milos Jenicek who published a book on the topic9 in the late 1990s. In other disciplines, such as the social sciences, further advances and contributions were made in the domain of case study research.10 It should also be noted that in the discipline of psychology, single case experimental designs were already developed in
the 1970s and received increasing attention in psychology,11 conventional medicine, and also complementary medicine12 in the 21st century. However, single case experimental designs are essentially clinical trials and the focus of this paper is on single clinical case reports as observational data. In medicine, it was only in the 21st century that the interest in clinical case reports started to gain further momentum13 and this was accompanied by the launch of several journals dedicated to the publication of high quality clinical cases. The first of these journals, ‘Grand Rounds’, started publishing in 2001 and after this several other journals were launched, including ‘Case Reports in Medicine’ and ‘The Journal of Medical Case Reports’ as the most important general case journals, as well a number of discipline specific cases journals (e.g. Elsevier recently launched a respiratory medicine cases journal). To facilitate easy exchange of information, most operate on an ‘open access basis’ and some journals (e.g. the BMC Cases Database associated with the Journal of Medical Case Reports) and disciplines, e.g. psychotherapy14 have set up associated online clinical case registries/archives. A parallel development in Evidence Based Medicine has been the proliferation of reporting guidelines on different types of research studies, notably clinical trials (CONSORT statement) and Systematic Reviews (PRISMA statement). Apart from these ‘high profile statements’, there are currently a broad range of reporting guidelines available, focusing on a wide range of quantitative and qualitative research designs, as well as specific clinical domains. For an overview of reporting guidelines in general, visit the EQUATOR website (http://www.equator-network.org/home/). Surprisingly, despite this proliferation of reporting guidelines, until recently there was no published general reporting guideline on clinical case reports, and within specific clinical domains, there was only a guideline on reporting cases in clinical behavioural psychology.15 Also, the journal instructions to authors of case reports are limited.16 A positive development in this respect is therefore the publication of the CARE guideline for the publication of clinical case reports.17 The development of this guideline was inspired by the pioneering work of Prof. Jenicek, as well as the expert meeting referred to in this paper. Building on the CARE guidelines, there is a need to further develop a scientific framework how to improve the reliability and value of clinical case reports in different CAM therapies for a variety of purposes. The aim of this paper is to provide an overview of the different objectives for clinical case reports and to identify those which seem most relevant and important to develop further in the context of that particular CAM modality. This approach has been implemented in the domain of homoeopathy, and will be reported in a separate article.
Improving the reporting quality of clinical case reports in complementary medicine Table 1
Reasons for single case reporting assessed for their relative applicability to CAM treatments.
Reason for single case reporting
Relative applicability
Comment
Clinical situation as valuable experience that cannot be reproduced for ethical reasons
High
Confirmation of something already known
High
Solving a challenging problem in medical ethics
High
Clinical failures, errors, and disasters in diagnosis and treatment
High
Unusual comorbidity (diagnosis, treatment for comorbidity, outcomes)
High
Limited access to cases (economy, ethics, social reasons, patient choices) Establishment of diagnostic criteria for a ‘‘new’’ disease
High
• CAM treatments are widely used as a complementary treatment in cancer patients.21,22 Due to a strong patient preference for the CAM treatment or due to the severity of the disease (e.g. cancer) it is difficult or not possible to conduct clinical trials.23 • Also, often little or no systematically collected previous data on outcomes is available, this provides a clear rationale for using clinical case reports as a preliminary data gathering method. • CAM therapeutic systems often have complex conceptual frameworks for triggering or causing disease that are hard or impossible to reproduce in an experimental setting. For example, adverse disease evolution due to ‘suppression of disease’ by conventional medicines within the conceptual framework of homoeopathy. This will usually take place in a systematic case report review and/or a lot of what is known or purported in CAM would benefit from further corroboration and refinement based on clinical cases. This touches for instance on what can or cannot be done with CAM in the context of: • The limitations and possibilities of conventional medicine. • Patient preference with regard to CAM treatments or refusal of effective conventional treatment. Important lessons can be learnt from unexpected adverse outcomes of CAM treatments, even though given the relatively good safety profile of most CAM therapies, there occurrences are likely to be relatively rare. In CAM one normally looks at these complex interactions within the context of a holistic approach with the aim of individualising treatment. So in CAM this is quite important but in most cases it is implicit in the overall approach. This applies equally to CAM as to conventional medicine. In CAM therapies, often specific diagnostic systems are used: e.g. pulse and tongue diagnosis in Chinese medicine, dysregulation in bioregulatory medicine,7 validation of symptoms identified in homoeopathic ‘provings’ or otherwise as prescribing criteria,24 etc. Unusual cases with dramatic outcomes are often the trigger for case reports in CAM. In classical homoeopathy, the unusual aspect of the case presentation is very important as an individualising feature and as such intrinsic to the therapeutic system. E.g. In homeopathy and bioregulatory medicine there is a particular theoretical framework for disease progression/evolution (either in the direction of better or worse health) which in specific cases could lead to the impression of an unusual natural history, so this would need to be taken into consideration and assessed separately.
Moderate
Unusual presentation of unknown aetiology
Moderate
Unusual natural history and/or clinical course
Moderate
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144 Table 1
R.A. van Haselen (Continued)
Reason for single case reporting
Relative applicability
Comment
Challenging differential diagnosis
Moderate
Unusual and/or unexpected effects of treatment (outcomes), adverse effects, diagnostic results, whether good or bad
Moderate
Diagnostic and therapeutic ‘‘accidents’’ (causes, consequences, remedies)
Moderate
Patient compliance
Moderate
Patient-doctor interaction (as in psychiatry)
Low
Index case(s) of a potential or real outbreak (more to come)
Not applicable
Possibly less relevant in CAM because the system is less focused on drilling down the disease to a relatively narrow diagnostic category. Emphasis tends to be more on devising a whole person orientated treatment strategy. There are however cases in which there is a need for differential diagnosis within the specific diagnostic system utilised to inform the treatment strategy (e.g. pulse diagnosis to inform acupuncture treatment, selection of the correct individualized medicine in homeopathy). Proper documentation is important particularly to assess purported adverse events due to CAM treatment(s). In such cases it should be thoroughly assessed if co-interventions or interactions could be possible alternative explanations. Another possibility, that will need to be differentiated if appropriate, is an unexpectedly strong reaction (e.g. initial aggravation of symptoms as part of a healing response25,26 ) to the CAM treatment. Diagnostic procedures are rarely invasive in CAM, but for some CAM therapeutic procedures accidents could occur, e.g. pneumothorax as a result of incorrectly applied acupuncture. Multiple case reports could help to point out a possible causal relationship. In some CAM therapies, the therapeutic approach can be complex and/or involve a relatively high level of active participation from the patient.27 Here case reports could help with identifying and better understanding the factors that influence compliance. This can be an issue in CAM modalities such as for instance manual therapies28,29 because it is claimed that the therapeutic interaction in its own right contributes to the observed effects. Case reports could help to better understand the role of various dimensions of the patient—doctor interaction from a hypothesis generating perspective. This is the classical epidemiological situation when there is an outbreak of an epidemic disease. Therefore, this does not apply specifically to CAM.
Methods This paper will provide an overview of the various objectives of clinical case reports and an assessment of the potential relevance for CAM, with particular reference to the example of homoeopathy as a major CAM modality. The various objectives of clinical case reports have been described by Jenicek18 and the potential relevance of these objectives for CAM were discussed by a panel of invited experts which consisted of clinicians, methodologists and journal editors in a meeting in Heidelberg in 2008 (including Prof. Jenicek). The relative applicability of the different objectives in a CAM context was discussed by this panel and graded as high, medium or low as a basis for further prioritisation and discussion.
Further to this, a series of articles on the topic of case based research were published in Complementary Therapies in Medicine in recent years, with a view to further raising the awareness of this topic from a variety of angles.12,19,20 Relevant elements have been integrated in the development of a reporting guideline for clinical case reports in homoeopathy as a practical illustration how a CAM therapy specific reporting guidelines could be developed. The results of this study are reported in a separate article.
Results The review of the objectives of clinical case reports and their relative applicability to CAM are summarised in Table 1.
Improving the reporting quality of clinical case reports in complementary medicine
Figure 1
Venn diagram illustrating the overall concept for clinical case report guideline development in CAM.
Figure 2
Venn diagram illustrating the concept for clinical case report guideline development in acupuncture.
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R.A. van Haselen
Figure 3 Venn diagram illustrating the concept for clinical case report guideline development in homeopathy with the objective to further clinically validate proving symptoms.
Table 1 indicates that the majority of objectives for clinical case reports identified by Jenicek apply to CAM. The three most important clusters of reasons for clinical case reporting in CAM can be conceptually grouped under the headers ‘impossibility/difficulty to conduct clinical trials’, ‘corroboration/refinement of existing knowledge’ and ‘safety’. Considering the complexity of the different objectives for clinical case reports, it is clear that a ‘one size fits all’ approach will not suffice. Whilst the recently published CARE guidelines provide a useful general framework that can be used as a basis, reporting guideline development will need to be further tailored to the specific objectives and particularities of the CAM approach. As a ‘generic’ quality standard for clinical cases, Jenicek9 proposed an ‘architecture of a clinical case report’ which is also reflected in the CARE guideline. Further methodological and practical considerations regarding clinical case reports are given in various publications by Jenicek18,30—34 and others.35,36 One of the first available examples of discipline specific clinical case reporting guidelines is the publication by Virues-Ortega et al. on clinical case reports in behavioural clinical psychology.15 For the further development of clinical case reporting guidelines in CAM, I am proposing a conceptual approach as illustrated in Fig. 1. The figure is set up as a ‘Venn diagram’, which distinguishes ‘generic’ and ‘treatment specific’ elements. The most ‘generic’ level consists of the basic ‘architectural’ elements as formulated by Jenicek and the CARE guidelines. In addition, generic quality aspects particular to
CAM treatments are proposed. This could cover particular aspects related to the holistic, individualised approach common to many CAM approaches. It should be noted however that these ‘generic’ elements are not sharply distinguished due to the lack of a ‘sharp’ definition of CAM.37 The purpose is to help identifying a number of aspects that are common to many CAM treatments for which common interdisciplinary reporting guidelines items could be identified. The CAM treatment specific items are determined by CAM discipline specific characteristics in conjunction with the specific objective(s) of the clinical case report. This conceptual approach is illustrated practically in further detail in for acupuncture in Fig. 2 and for homoeopathy in Fig. 3. Fig. 2 fills in some of the details, in particular it illustrates the concept that for acupuncture specific quality items, one can ‘borrow’ from existing guidelines developed as extensions of the CONSORT guidelines for reporting clinical trials of acupuncture.38 These STRICTA guidelines include a number of items such as the style of acupuncture used and needling details which describe the acupuncture intervention in further detail. These elements are of course also useful as quality items for acupuncture case reports. Fig. 3 provides the most detailed overview of the approach using the clinical validation of ‘proving’ symptoms on the basis of case reports as an example. With regard to the CAM specific ‘generic items’ as an example further details on the duration and nature of the consultation, as well as on the use of patient centred outcome measures such as the ‘Measure Yourself Medical Outcome Profile’ (MYMOP)39 could be provided. Similar to the acupuncture
Improving the reporting quality of clinical case reports in complementary medicine example, for a description of the homoeopathy intervention, use can be made of the CONSORT extension for homoeopathy trials called RedHot.40 Items such as the type of homoeopathy, further details on the homoeopathic consultation and medications as well as the occurrence of initial aggravations following treatment, are useful. The use of other guidelines or therapy specific assessment tools, if available, could also be considered. For instance, for the outcome assessment in chronic cases, use could be made of an assessment tool that evaluates the occurrence of disease evolution in according with homoeopathic principles of cure proposed by Constantin Hering.6,41 In this figure, the clinical case report specific objective is indicated as the ‘clinical validation of proving symptoms’. From a methodological point of view it should be pointed out that two approaches are possible. In the ‘prospective’ approach a number of criteria are ‘pre-formulated’ and only cases that meet these predefined criteria will be selected for detailed clinical case reporting in accordance with the guideline. In the ‘retrospective’ approach, homoeopaths will treat their patients as usual and record these treatments using relevant information technology and then be asked to provide/identify cases in their ‘cases pool’ which fulfil a number of specific criteria. One of these criteria could for instance be the occurrence of a dramatic improvement/cure on a particular remedy, followed by the second criterion of a sufficiently documented patient history, which could be used to compile and write up a case report in line with the guideline requirements. In conclusion, the overall concept involves a ‘modular’ approach in which, depending on the objectives of the clinical case report and the CAM discipline involved, existing guidelines, such as in particular the CARE guideline, are further complemented by additional item which are optimally ‘tailored’ to the specific therapeutic context.
Discussion/conclusions This paper outlined areas where there is a clear rationale for the need for guideline development for clinical case reports in some of the key CAM modalities. In addition, a methodological and process framework for CAM specific guideline development was identified, which builds on generic clinical case reporting elements, whilst at the same time addressing the CAM specific elements as appropriate. This will create more clarity and transparency in the reporting of clinical cases, and enable the reader to form her/his own opinion. A limitation of the chosen approach is that it only addresses the areas in CAM where there is a clear need and justification for further standardisation of processes. Another trend, also present as streams of thought in for instance psychotherapy, goes into the direction of emphasising the unique qualitative aspects of the patient—doctor interaction. In these types of approaches, sometimes also referred to as ‘narrative based medicine’, the use of standardised guidelines and templates would clearly be counterproductive: it would be perceived as ‘killing off’, or taking away from, the unique nature of the patient—doctor encounter. This paper does not fully assess and discuss the level of differentiation in the scope and need for reporting guidelines. It is therefore important to obtain views from relevant experts in the field, if, and when, the use
147
of reporting guidelines could be counterproductive and/or inappropriate. A further limitation is that the grading of the relative applicability of clinical case reporting objectives to CAM based on a single expert meeting was not extensive, nor objective. Therefore it mainly serves as a basis for further discussion and development. A more extensive assessment of relevance should also take into consideration some further perspectives (e.g. ‘evidence based’ versus ‘narrative based’) and domains of application (e.g. teaching versus practice versus research) as additional dimensions. The need for reporting guidelines is increasingly recognised in medicine and the publication of the CARE guideline is an important milestone that adds to the current momentum. Further improvements in clinical case reporting in CAM can greatly contribute to CAM research and education, as well as to improved clinical care for patients.
Conflict of interest The author previously worked as an employee for Heel, which provided financial support for the expert meeting and project.
Acknowledgements I would like to acknowledge the invaluable input of those who attended the expert meeting in Heidelberg, i.e. (in alphabetical order): Ivo Bianchi (Italy), Milos Jenicek (Canada), Menachem Oberbaum (Israel), Oluwadamilola Olaku (USA), David Riley (USA), Alta Smit (Germany) and Peter Smith (Germany). Heel for providing financial support for the organisation of the expert meeting and project. Cees Baas and Jean Pierre Jansen for their feedback on drafts of the manuscript.
References 1. Sackett DL, Rosenberg WMC, Gray JAM, Haynes RB, Richardson WS. Evidence based medicine: what it is and what it isn’t. BMJ 1996;312(7023):71—2. 2. Jonas WB. Building an evidence house: challenges and solutions to research in complementary and alternative medicine. Forsch Komplementarmed Klass Naturheilkd 2005;12(3):159—67. 3. Reilly D, Taylor MA. Part II: Framework and approach: II. 2 The evidence profile. Complement Ther Med 1993;1(Suppl. 1 (0)):11—2. 4. van Haselen R. Medical study formats: an overview. J Biomed Ther 2010;4(2):26—7. 5. Walach H, Falkenberg T, Fønnebø V, Lewith G, Jonas WB. Circular instead of hierarchical: methodological principles for the evaluation of complex interventions. BMC Med Res Methodol 2006:6. 6. Saine A. Hering’s law: law, rule or dogma?; 2014 (last accessed) http://wwwhomeopathyca/articles det12shtml 7. Smit A, O’Byrne A, Van Brandt B, Bianchi I, Kuestermann K. Introduction to bioregulatory medicine. Stuttgart, New York: Thieme; 2009. 8. van Haselen R. Purpose-orientated clinical research. J Biomed Ther 2011;5(1):26—7.
148 9. Jenicek M. Clinical case reporting in evidence-based medicine. 2nd ed. Arnold; 2001. 10. Gerring J. Case study research. Principles and practices. Cambridge University Press; 2007. 11. Barlow DH, Nock MK, Hersen M. Single case experimental designs; strategies for studying behavior change. 3rd ed. Boston: Pearson Education Inc.; 2009. 12. Teut M, Linde K. Scientific case research in complementary and alternative medicine — a review. Complement Ther Med 2013;21(4):388—95. 13. Vandenbroucke JP. In defense of case reports and case series. Ann Intern Med 2001;134(4):330—4. 14. Desmet M. Psychoanalytic single cases published in isa ranked journals: the construction of an online archive. Psychother Psychosom 2013;82:120—1. 15. Virués-Ortega J, Moreno-Rodríguez R. Guidelines for clinical case reports in behavioral clinical psychology. Int J Clin Health Psychol 2008;8(3):765—77. 16. Sorinola O, Olufowobi O, Coomarasamy A, Khan KS. Instructions to authors for case reporting are limited: a review of a core journal list. BMC Med Educ 2004;4:1—5. 17. Gagnier JJ, Kienle G, Altman DG, Moher D, Sox H, Riley D. The CARE guidelines: consensus-based clinical case report guideline development. J Clin Epidemiol 2014;67(1):46—51. 18. Jenicek M. Clinical case reports: sources of boredom or valuable pieces of evidence? Natl Med J India 2001;14(4):193—4. 19. Relton C, Viksveen P, Kessler U. The making cases count intiative. Complement Ther Med 2014. 20. Rutten L. The importance of case histories for accepting and improving homeopathy. Complement Ther Med 2013;21(6):565—70. 21. Chang KH, Brodie R, Choong MA, Sweeney KJ, Kerin MJ. Complementary and alternative medicine use in oncology: a questionnaire survey of patients and health care professionals. BMC Cancer 2011:11. 22. Surette S, Vanderjagt L, Vohra S. Surveys of complementary and alternative medicine usage: a scoping study of the paediatric literature. Complement Ther Med 2013;21(Suppl. 1):S48—53. 23. Ziegler R. Mistletoe preparation iscador: are there methodological concerns with respect to controlled clinical trials? Evid Based Complement Alternat Med 2009;6(1):19—30. 24. Gadd B. In search of the reliable repertory. Homeopathy 2009;98(1):60—4. 25. Stub T, Alraek T, Salamonsen A. The Red flag! risk assessment among medical homeopaths in Norway: a qualitative study. BMC Complement Altern Med 2012;12(1):150. 26. Stub T, Salamonsen A, Alraek T. Is it possible to distinguish homeopathic aggravation from adverse effects? A qualitative study. Forsch Komplementarmed 2011;19(1):13—9. 27. Bishop FL, Yardley L, Lewith GT. Treatment appraisals and beliefs predict adherence to complementary therapies: a
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Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.elsevierhealth.com/journals/ctim
Towards improving the reporting quality of clinical case reports in complementary medicine: Assessing and illustrating the need for guideline development R.A. van Haselen ∗ International Institute for Integrated Medicine, 22 Eden Street, Suite 467, Kingston KT1 1DN, United Kingdom Available online 19 January 2015
KEYWORDS Clinical case reports; Reporting guideline development
∗
Summary Background: Case reports have had a varying level of recognition as a source of evidence throughout the history of medicine. In recent years, there has been a revival of interest in clinical case reports in both conventional and complementary medicine. There is a need to further improve the reporting quality of clinical case reports of different Complementary and Alternative Medicine (CAM) therapies. Objectives: To provide an overview of the different objectives for clinical case reports, identify those that are most relevant for CAM, and to develop a conceptual framework for purpose orientated clinical case reporting guidelines for CAM therapies. To practically illustrate the chosen approach by developing a clinical case reporting guideline for homeopathic cases. Methods: The various objectives of clinical case reports were described by Prof. Milos Jenicek, and the potential relevance of these objectives for CAM were discussed and graded by a mixed panel of experts. A conceptual framework for developing clinical case reporting guidelines for CAM treatments with specific objectives is proposed. The aim is to integrate both ‘generic’ and ‘CAM therapy specific’ quality items. This framework has been practically applied to the development of a reporting guideline for clinical case reports in homoeopathy which will be reported in a second article. Results: An overview is given of the clinical case reporting literature. The conceptual framework for the development of purpose orientated CAM clinical case reporting guidelines is presented. This framework is based on alignment with the recently published ‘generic’ CARE guideline for reporting of clinical case reports, whilst addressing the CAM specific elements at the same time. Conclusions: The scope and importance of clinical case reporting guideline development in CAM is illustrated. A conceptual framework for developing CAM specific clinical case reporting
Tel.: +44 2081449524. E-mail address: [email protected]
http://dx.doi.org/10.1016/j.ctim.2015.01.009 0965-2299/© 2015 Elsevier Ltd. All rights reserved.
142
R.A. van Haselen guidelines was established. It has been implemented using homoeopathy as an illustration, and this will be reported in a separate article. Further improvements in clinical case reporting in CAM will greatly contribute to CAM research and education, as well as to improved patient care. © 2015 Elsevier Ltd. All rights reserved.
Introduction Case reports have had a variable level of recognition as a source of evidence throughout the history of medicine. Whilst case reports have remained consistently important as part of teaching medicine, the value attributed to case reports as a source of evidence has been greatly diminished since the predominant emergence of modern, evidence based medicine in the second half of the 20th century.1 Within this framework several ‘hierarchies’ of evidence have been developed for assessing the quality of evidence, The US Preventive Services Task Force and the Oxford Centre for Evidence-Based Medicine schemes being prominent examples. In each of these, case reports feature low in, or at the bottom of, the hierarchy. The hierarchical approach towards evidence has been criticised as being too one-sidedly focused only on the perspective of establishing causal attribution to a treatment within the context of assessing efficacy. Whilst from this perspective clinical case reports are justifiably graded as weak evidence, from another perspective such as for instance safety assessment, case reports are often the first ‘signals’ of potential adverse drug reactions and sometimes single cases can even contribute significantly to establishing a causal link. In particular in the CAM community, nonhierarchical systems have been propagated, using words and metaphors such as an ‘evidence house’,2 ‘evidence profile’,3 ‘evidence mosaic’,4 or a ‘circular’ approach towards evidence.5 At the core of the above approaches is the notion that the hierarchy of evidence depends on the principal research objective, and as illustrated by the above example, the value of case reports is different in safety assessment as compared to efficacy assessment. Furthermore case reports can provide valuable insights in the development and cure of diseases in the course of time. The latter is also referred to as ‘disease evolution’ and this concept has been developed in various CAM systems such as for instance classical homeopathy6 and bioregulatory medicine.7 These research purposes can be categorised further using dimensions such as ‘hypothesis confirmation’ versus ‘hypothesis generation, ‘ideal world’ versus ‘real world’ and ‘absolute’ versus ‘relative’ effects.8 Within such a framework, case reports are particularly appropriate/strong methods for hypothesis generation in the ‘real world’ setting of daily medicine. The main pioneer who contributed to a re-appraisal of the millennia old art of case observation and case reporting in medicine is Prof. Milos Jenicek who published a book on the topic9 in the late 1990s. In other disciplines, such as the social sciences, further advances and contributions were made in the domain of case study research.10 It should also be noted that in the discipline of psychology, single case experimental designs were already developed in
the 1970s and received increasing attention in psychology,11 conventional medicine, and also complementary medicine12 in the 21st century. However, single case experimental designs are essentially clinical trials and the focus of this paper is on single clinical case reports as observational data. In medicine, it was only in the 21st century that the interest in clinical case reports started to gain further momentum13 and this was accompanied by the launch of several journals dedicated to the publication of high quality clinical cases. The first of these journals, ‘Grand Rounds’, started publishing in 2001 and after this several other journals were launched, including ‘Case Reports in Medicine’ and ‘The Journal of Medical Case Reports’ as the most important general case journals, as well a number of discipline specific cases journals (e.g. Elsevier recently launched a respiratory medicine cases journal). To facilitate easy exchange of information, most operate on an ‘open access basis’ and some journals (e.g. the BMC Cases Database associated with the Journal of Medical Case Reports) and disciplines, e.g. psychotherapy14 have set up associated online clinical case registries/archives. A parallel development in Evidence Based Medicine has been the proliferation of reporting guidelines on different types of research studies, notably clinical trials (CONSORT statement) and Systematic Reviews (PRISMA statement). Apart from these ‘high profile statements’, there are currently a broad range of reporting guidelines available, focusing on a wide range of quantitative and qualitative research designs, as well as specific clinical domains. For an overview of reporting guidelines in general, visit the EQUATOR website (http://www.equator-network.org/home/). Surprisingly, despite this proliferation of reporting guidelines, until recently there was no published general reporting guideline on clinical case reports, and within specific clinical domains, there was only a guideline on reporting cases in clinical behavioural psychology.15 Also, the journal instructions to authors of case reports are limited.16 A positive development in this respect is therefore the publication of the CARE guideline for the publication of clinical case reports.17 The development of this guideline was inspired by the pioneering work of Prof. Jenicek, as well as the expert meeting referred to in this paper. Building on the CARE guidelines, there is a need to further develop a scientific framework how to improve the reliability and value of clinical case reports in different CAM therapies for a variety of purposes. The aim of this paper is to provide an overview of the different objectives for clinical case reports and to identify those which seem most relevant and important to develop further in the context of that particular CAM modality. This approach has been implemented in the domain of homoeopathy, and will be reported in a separate article.
Improving the reporting quality of clinical case reports in complementary medicine Table 1
Reasons for single case reporting assessed for their relative applicability to CAM treatments.
Reason for single case reporting
Relative applicability
Comment
Clinical situation as valuable experience that cannot be reproduced for ethical reasons
High
Confirmation of something already known
High
Solving a challenging problem in medical ethics
High
Clinical failures, errors, and disasters in diagnosis and treatment
High
Unusual comorbidity (diagnosis, treatment for comorbidity, outcomes)
High
Limited access to cases (economy, ethics, social reasons, patient choices) Establishment of diagnostic criteria for a ‘‘new’’ disease
High
• CAM treatments are widely used as a complementary treatment in cancer patients.21,22 Due to a strong patient preference for the CAM treatment or due to the severity of the disease (e.g. cancer) it is difficult or not possible to conduct clinical trials.23 • Also, often little or no systematically collected previous data on outcomes is available, this provides a clear rationale for using clinical case reports as a preliminary data gathering method. • CAM therapeutic systems often have complex conceptual frameworks for triggering or causing disease that are hard or impossible to reproduce in an experimental setting. For example, adverse disease evolution due to ‘suppression of disease’ by conventional medicines within the conceptual framework of homoeopathy. This will usually take place in a systematic case report review and/or a lot of what is known or purported in CAM would benefit from further corroboration and refinement based on clinical cases. This touches for instance on what can or cannot be done with CAM in the context of: • The limitations and possibilities of conventional medicine. • Patient preference with regard to CAM treatments or refusal of effective conventional treatment. Important lessons can be learnt from unexpected adverse outcomes of CAM treatments, even though given the relatively good safety profile of most CAM therapies, there occurrences are likely to be relatively rare. In CAM one normally looks at these complex interactions within the context of a holistic approach with the aim of individualising treatment. So in CAM this is quite important but in most cases it is implicit in the overall approach. This applies equally to CAM as to conventional medicine. In CAM therapies, often specific diagnostic systems are used: e.g. pulse and tongue diagnosis in Chinese medicine, dysregulation in bioregulatory medicine,7 validation of symptoms identified in homoeopathic ‘provings’ or otherwise as prescribing criteria,24 etc. Unusual cases with dramatic outcomes are often the trigger for case reports in CAM. In classical homoeopathy, the unusual aspect of the case presentation is very important as an individualising feature and as such intrinsic to the therapeutic system. E.g. In homeopathy and bioregulatory medicine there is a particular theoretical framework for disease progression/evolution (either in the direction of better or worse health) which in specific cases could lead to the impression of an unusual natural history, so this would need to be taken into consideration and assessed separately.
Moderate
Unusual presentation of unknown aetiology
Moderate
Unusual natural history and/or clinical course
Moderate
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144 Table 1
R.A. van Haselen (Continued)
Reason for single case reporting
Relative applicability
Comment
Challenging differential diagnosis
Moderate
Unusual and/or unexpected effects of treatment (outcomes), adverse effects, diagnostic results, whether good or bad
Moderate
Diagnostic and therapeutic ‘‘accidents’’ (causes, consequences, remedies)
Moderate
Patient compliance
Moderate
Patient-doctor interaction (as in psychiatry)
Low
Index case(s) of a potential or real outbreak (more to come)
Not applicable
Possibly less relevant in CAM because the system is less focused on drilling down the disease to a relatively narrow diagnostic category. Emphasis tends to be more on devising a whole person orientated treatment strategy. There are however cases in which there is a need for differential diagnosis within the specific diagnostic system utilised to inform the treatment strategy (e.g. pulse diagnosis to inform acupuncture treatment, selection of the correct individualized medicine in homeopathy). Proper documentation is important particularly to assess purported adverse events due to CAM treatment(s). In such cases it should be thoroughly assessed if co-interventions or interactions could be possible alternative explanations. Another possibility, that will need to be differentiated if appropriate, is an unexpectedly strong reaction (e.g. initial aggravation of symptoms as part of a healing response25,26 ) to the CAM treatment. Diagnostic procedures are rarely invasive in CAM, but for some CAM therapeutic procedures accidents could occur, e.g. pneumothorax as a result of incorrectly applied acupuncture. Multiple case reports could help to point out a possible causal relationship. In some CAM therapies, the therapeutic approach can be complex and/or involve a relatively high level of active participation from the patient.27 Here case reports could help with identifying and better understanding the factors that influence compliance. This can be an issue in CAM modalities such as for instance manual therapies28,29 because it is claimed that the therapeutic interaction in its own right contributes to the observed effects. Case reports could help to better understand the role of various dimensions of the patient—doctor interaction from a hypothesis generating perspective. This is the classical epidemiological situation when there is an outbreak of an epidemic disease. Therefore, this does not apply specifically to CAM.
Methods This paper will provide an overview of the various objectives of clinical case reports and an assessment of the potential relevance for CAM, with particular reference to the example of homoeopathy as a major CAM modality. The various objectives of clinical case reports have been described by Jenicek18 and the potential relevance of these objectives for CAM were discussed by a panel of invited experts which consisted of clinicians, methodologists and journal editors in a meeting in Heidelberg in 2008 (including Prof. Jenicek). The relative applicability of the different objectives in a CAM context was discussed by this panel and graded as high, medium or low as a basis for further prioritisation and discussion.
Further to this, a series of articles on the topic of case based research were published in Complementary Therapies in Medicine in recent years, with a view to further raising the awareness of this topic from a variety of angles.12,19,20 Relevant elements have been integrated in the development of a reporting guideline for clinical case reports in homoeopathy as a practical illustration how a CAM therapy specific reporting guidelines could be developed. The results of this study are reported in a separate article.
Results The review of the objectives of clinical case reports and their relative applicability to CAM are summarised in Table 1.
Improving the reporting quality of clinical case reports in complementary medicine
Figure 1
Venn diagram illustrating the overall concept for clinical case report guideline development in CAM.
Figure 2
Venn diagram illustrating the concept for clinical case report guideline development in acupuncture.
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R.A. van Haselen
Figure 3 Venn diagram illustrating the concept for clinical case report guideline development in homeopathy with the objective to further clinically validate proving symptoms.
Table 1 indicates that the majority of objectives for clinical case reports identified by Jenicek apply to CAM. The three most important clusters of reasons for clinical case reporting in CAM can be conceptually grouped under the headers ‘impossibility/difficulty to conduct clinical trials’, ‘corroboration/refinement of existing knowledge’ and ‘safety’. Considering the complexity of the different objectives for clinical case reports, it is clear that a ‘one size fits all’ approach will not suffice. Whilst the recently published CARE guidelines provide a useful general framework that can be used as a basis, reporting guideline development will need to be further tailored to the specific objectives and particularities of the CAM approach. As a ‘generic’ quality standard for clinical cases, Jenicek9 proposed an ‘architecture of a clinical case report’ which is also reflected in the CARE guideline. Further methodological and practical considerations regarding clinical case reports are given in various publications by Jenicek18,30—34 and others.35,36 One of the first available examples of discipline specific clinical case reporting guidelines is the publication by Virues-Ortega et al. on clinical case reports in behavioural clinical psychology.15 For the further development of clinical case reporting guidelines in CAM, I am proposing a conceptual approach as illustrated in Fig. 1. The figure is set up as a ‘Venn diagram’, which distinguishes ‘generic’ and ‘treatment specific’ elements. The most ‘generic’ level consists of the basic ‘architectural’ elements as formulated by Jenicek and the CARE guidelines. In addition, generic quality aspects particular to
CAM treatments are proposed. This could cover particular aspects related to the holistic, individualised approach common to many CAM approaches. It should be noted however that these ‘generic’ elements are not sharply distinguished due to the lack of a ‘sharp’ definition of CAM.37 The purpose is to help identifying a number of aspects that are common to many CAM treatments for which common interdisciplinary reporting guidelines items could be identified. The CAM treatment specific items are determined by CAM discipline specific characteristics in conjunction with the specific objective(s) of the clinical case report. This conceptual approach is illustrated practically in further detail in for acupuncture in Fig. 2 and for homoeopathy in Fig. 3. Fig. 2 fills in some of the details, in particular it illustrates the concept that for acupuncture specific quality items, one can ‘borrow’ from existing guidelines developed as extensions of the CONSORT guidelines for reporting clinical trials of acupuncture.38 These STRICTA guidelines include a number of items such as the style of acupuncture used and needling details which describe the acupuncture intervention in further detail. These elements are of course also useful as quality items for acupuncture case reports. Fig. 3 provides the most detailed overview of the approach using the clinical validation of ‘proving’ symptoms on the basis of case reports as an example. With regard to the CAM specific ‘generic items’ as an example further details on the duration and nature of the consultation, as well as on the use of patient centred outcome measures such as the ‘Measure Yourself Medical Outcome Profile’ (MYMOP)39 could be provided. Similar to the acupuncture
Improving the reporting quality of clinical case reports in complementary medicine example, for a description of the homoeopathy intervention, use can be made of the CONSORT extension for homoeopathy trials called RedHot.40 Items such as the type of homoeopathy, further details on the homoeopathic consultation and medications as well as the occurrence of initial aggravations following treatment, are useful. The use of other guidelines or therapy specific assessment tools, if available, could also be considered. For instance, for the outcome assessment in chronic cases, use could be made of an assessment tool that evaluates the occurrence of disease evolution in according with homoeopathic principles of cure proposed by Constantin Hering.6,41 In this figure, the clinical case report specific objective is indicated as the ‘clinical validation of proving symptoms’. From a methodological point of view it should be pointed out that two approaches are possible. In the ‘prospective’ approach a number of criteria are ‘pre-formulated’ and only cases that meet these predefined criteria will be selected for detailed clinical case reporting in accordance with the guideline. In the ‘retrospective’ approach, homoeopaths will treat their patients as usual and record these treatments using relevant information technology and then be asked to provide/identify cases in their ‘cases pool’ which fulfil a number of specific criteria. One of these criteria could for instance be the occurrence of a dramatic improvement/cure on a particular remedy, followed by the second criterion of a sufficiently documented patient history, which could be used to compile and write up a case report in line with the guideline requirements. In conclusion, the overall concept involves a ‘modular’ approach in which, depending on the objectives of the clinical case report and the CAM discipline involved, existing guidelines, such as in particular the CARE guideline, are further complemented by additional item which are optimally ‘tailored’ to the specific therapeutic context.
Discussion/conclusions This paper outlined areas where there is a clear rationale for the need for guideline development for clinical case reports in some of the key CAM modalities. In addition, a methodological and process framework for CAM specific guideline development was identified, which builds on generic clinical case reporting elements, whilst at the same time addressing the CAM specific elements as appropriate. This will create more clarity and transparency in the reporting of clinical cases, and enable the reader to form her/his own opinion. A limitation of the chosen approach is that it only addresses the areas in CAM where there is a clear need and justification for further standardisation of processes. Another trend, also present as streams of thought in for instance psychotherapy, goes into the direction of emphasising the unique qualitative aspects of the patient—doctor interaction. In these types of approaches, sometimes also referred to as ‘narrative based medicine’, the use of standardised guidelines and templates would clearly be counterproductive: it would be perceived as ‘killing off’, or taking away from, the unique nature of the patient—doctor encounter. This paper does not fully assess and discuss the level of differentiation in the scope and need for reporting guidelines. It is therefore important to obtain views from relevant experts in the field, if, and when, the use
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of reporting guidelines could be counterproductive and/or inappropriate. A further limitation is that the grading of the relative applicability of clinical case reporting objectives to CAM based on a single expert meeting was not extensive, nor objective. Therefore it mainly serves as a basis for further discussion and development. A more extensive assessment of relevance should also take into consideration some further perspectives (e.g. ‘evidence based’ versus ‘narrative based’) and domains of application (e.g. teaching versus practice versus research) as additional dimensions. The need for reporting guidelines is increasingly recognised in medicine and the publication of the CARE guideline is an important milestone that adds to the current momentum. Further improvements in clinical case reporting in CAM can greatly contribute to CAM research and education, as well as to improved clinical care for patients.
Conflict of interest The author previously worked as an employee for Heel, which provided financial support for the expert meeting and project.
Acknowledgements I would like to acknowledge the invaluable input of those who attended the expert meeting in Heidelberg, i.e. (in alphabetical order): Ivo Bianchi (Italy), Milos Jenicek (Canada), Menachem Oberbaum (Israel), Oluwadamilola Olaku (USA), David Riley (USA), Alta Smit (Germany) and Peter Smith (Germany). Heel for providing financial support for the organisation of the expert meeting and project. Cees Baas and Jean Pierre Jansen for their feedback on drafts of the manuscript.
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