Illness Perception and Clinical Treatment Experiences in Patients with M. Maroteaux-Lamy (Mucopolysaccharidosis Type VI) and a Turkish Migration Background in Germany

Introduction

Mucopolysaccharidosis VI (MPS VI) is an inherited lysosomal storage disease caused by a mutation of the gene for arylsulfatase B (ASB). Of the thirty-one patients registered in Germany, almost fifty percent have a Turkish migration background. MPS VI is treated by enzyme replacement therapy (ERT), which is time-consuming and expensive.

Methods

This interdisciplinary study explored the illness perceptions and clinical treatment experiences among ten MPS VI patients with a Turkish migration background in two centers for metabolic diseases (Berlin and Mainz, Germany). The clinical treatment situation was observed and semi-structured interviews were conducted with patients and health care personnel, in addition to participatory observation in four patients'' everyday environments in Berlin. The data from the interviews, patient records, and personal field notes were encoded, cross-related, and analyzed.

Results

Patients'' acknowledgement of the disease and coping strategies are influenced predominantly by the perception of their individual health status and the handling of the disease within their family. Patients'' willingness to cooperate with treatment strategies is further modified by their knowledge of the disease and the relationships with their health care providers. In this analysis, cultural factors turned out to be marginally relevant.

Conclusion

As with other chronic and debilitating diseases, effective treatment strategies have to reach beyond delivering medication. Health care providers need to strengthen the support for patients with a migration background. In this regard, they should respect the patients'' cultural and social background and their personal perception of the disease and the therapy. Yet structural and social aspects (clinical setting, family and educational background) may be more crucial here than “cultural barriers.”     

Clinical and Organizational Issues in the Management of Surviving Breast and Colorectal Cancer Patients: Attitudes and Feelings of Medical Oncologists

Background

The fast growing demand and the shortage of resources are pushing toward more efficient models of survivorship care delivery. The Associazione Italiana di Oncologia Medica (AIOM) established an interdisciplinary working group with the purpose of promoting organizational improvements at the national level. A survey aimed at assessing attitudes and feelings of oncologists was considered preliminary to further initiatives.

Methods

A 25-item questionnaire, sent to the mailing list of the Society, explored the following issues on the practice of breast and colorectal cancer patients'' follow up: 1) organization; 2) clinical features; 3) feelings about the different meanings of follow-up.

Results

Ninety-one oncologists of 160 institutions (57%) answered to the questionnaire. Although follow up is considered a distinct oncological activity in 68%, a fully shared organization between specialists is not common and communications with Primary Care Physicians are not structured in the majority of the cases. Fifty-five and 30% of the oncologists follow breast and colorectal cancer patients indefinitely. In case of discharge a survivorship care plan is delivered in only 9%. The majority of respondents do not hold a role of follow up in mortality reduction.

Conclusions

Although survivorship care represents a significant part of the oncologists'' workload, an “oncology-centered” model is largely adopted and established care pathways are still incomplete. Survivorship care needs to be put at the center of an educational policy and of a widespread organizational effort, directed at improving appropriateness and quality.     

Identifying Factors to Improve Oral Cancer Screening Uptake: A Qualitative Study

Aims

To engage with high risk groups to identify knowledge and awareness of oral cancer signs and symptoms and the factors likely to contribute to improved screening uptake.

Methods

Focus group discussions were undertaken with 18 males; 40+ years of age; smokers and/or drinkers (15+ cigarettes per day and/or 15+ units of alcohol per week), irregular dental attenders living in economically deprived areas of Teesside.

Results

There was a striking reported lack of knowledge and awareness of oral cancer and its signs and symptoms among the participants. When oral/mouth cancer leaflets produced by Cancer Research UK were presented to the participants, they claimed that they would seek help on noticing such a condition. There was a preference to seek help from their general practitioner rather than their dentist due to perceptions that a dentist is ‘inaccessible’ on a physical and psychological level, costly, a ‘tooth specialist’ not a ‘mouth specialist’, and also not able to prescribe medication and make referrals to specialists. Interestingly, none of the 18 participants who were offered a free oral cancer examination at a dental practice took up this offer.

Conclusions

The uptake of oral cancer screening may be improved by increasing knowledge of the existence and signs and symptoms of oral cancer. Other factors that may increase uptake are increased awareness of the role of dentists in diagnosing oral cancer, promotion of oral cancer screening by health professionals during routine health checks, and the use of a “health” screening setting as opposed to a “dental” setting for such checks.     

Profiling dizziness in older primary care patients: an empirical study

Background

The diagnostic approach to dizzy, older patients is not straightforward as many organ systems can be involved and evidence for diagnostic strategies is lacking. A first differentiation in diagnostic subtypes or profiles may guide the diagnostic process of dizziness and can serve as a classification system in future research. In the literature this has been done, but based on pathophysiological reasoning only.

Objective

To establish a classification of diagnostic profiles of dizziness based on empirical data.

Design

Cross-sectional study.

Participants and Setting

417 consecutive patients of 65 years and older presenting with dizziness to 45 primary care physicians in the Netherlands from July 2006 to January 2008.

Methods

We performed tests, including patient history, and physical and additional examination, previously selected by an international expert panel and based on an earlier systematic review. We used the results of these tests in a principal component analysis for exploration, data-reduction and finally differentiation into diagnostic dizziness profiles.

Results

Demographic data and the results of the tests yielded 221 variables, of which 49 contributed to the classification of dizziness into six diagnostic profiles, that may be named as follows: “frailty”, “psychological”, “cardiovascular”, “presyncope”, “non-specific dizziness” and “ENT”. These explained 32% of the variance.

Conclusions

Empirically identified components classify dizziness into six profiles. This classification takes into account the heterogeneity and multicausality of dizziness and may serve as starting point for research on diagnostic strategies and can be a first step in an evidence based diagnostic approach of dizzy older patients.     

Understanding of Statistical Terms Routinely Used in Meta-Analyses: An International Survey among Researchers

Objective

Biomedical literature is increasingly enriched with literature reviews and meta-analyses. We sought to assess the understanding of statistical terms routinely used in such studies, among researchers.

Methods

An online survey posing 4 clinically-oriented multiple-choice questions was conducted in an international sample of randomly selected corresponding authors of articles indexed by PubMed.

Results

A total of 315 unique complete forms were analyzed (participation rate 39.4%), mostly from Europe (48%), North America (31%), and Asia/Pacific (17%). Only 10.5% of the participants answered correctly all 4 “interpretation” questions while 9.2% answered all questions incorrectly. Regarding each question, 51.1%, 71.4%, and 40.6% of the participants correctly interpreted statistical significance of a given odds ratio, risk ratio, and weighted mean difference with 95% confidence intervals respectively, while 43.5% correctly replied that no statistical model can adjust for clinical heterogeneity. Clinicians had more correct answers than non-clinicians (mean score ± standard deviation: 2.27±1.06 versus 1.83±1.14, p<0.001); among clinicians, there was a trend towards a higher score in medical specialists (2.37±1.07 versus 2.04±1.04, p = 0.06) and a lower score in clinical laboratory specialists (1.7±0.95 versus 2.3±1.06, p = 0.08). No association was observed between the respondents'' region or questionnaire completion time and participants'' score.

Conclusion

A considerable proportion of researchers, randomly selected from a diverse international sample of biomedical scientists, misinterpreted statistical terms commonly reported in meta-analyses. Authors could be prompted to explicitly interpret their findings to prevent misunderstandings and readers are encouraged to keep up with basic biostatistics.     

Mapping the knowledge structure of research on patient adherence: knowledge domain visualization based co-word analysis and social network analysis

Background

Patient adherence is an important issue for health service providers and health researchers. However, the knowledge structure of diverse research on treatment adherence is unclear. This study used co-word analysis and social network analysis techniques to analyze research literature on adherence, and to show their knowledge structure and evolution over time.

Methods

Published scientific papers about treatment adherence were retrieved from Web of Science (2000 to May 2011). A total of 2308 relevant articles were included: 788 articles published in 2000–2005 and 1520 articles published in 2006–2011. The keywords of each article were extracted by using the software Biblexcel, and the synonym and isogenous words were merged manually. The frequency of keywords and their co-occurrence frequency were counted. High frequency keywords were selected to yield the co-words matrix. Finally the decomposition maps were used to comb the complex knowledge structures.

Results

Research themes were more general in the first period (2000 to 2005), and more extensive with many more new terms in the second period (2006 to 2011). Research on adherence has covered more and more diseases, populations and methods, but other diseases/conditions are not as hot as HIV/AIDS and have not become specialty themes/sub-directions. Most studies originated from the United States.

Conclusion

The dynamic of this field is mainly divergent, with increasing number of new sub-directions of research. Future research is required to investigate specific directions and converge as well to construct a general paradigm in this field.     

Neglected diseases in the news: a content analysis of recent international media coverage focussing on leishmaniasis and trypanosomiasis

Background

Although the pharmaceutical industry''s “neglect” of neglected tropical diseases (NTDs) has been investigated, no study evaluating media coverage of NTDs has been published. Poor media coverage exacerbates the neglect. This study aimed to investigate, describe, and analyse international media coverage of “neglected diseases” in general and three specific NTDs—African trypanosomiasis, leishmaniasis, and Chagas disease—from 1 January 2003 to 1 June 2007.

Methods

Archives of 11 leading international, English-language media were searched. A content analysis was done, coding for media organisation, date, author, type of report, slant, themes, and “frames”. Semi-structured interviews with journalists and key informants were conducted for further insight.

Principal Findings

Only 113 articles in a 53-month time period met the inclusion criteria, with no strong trends or increases in coverage. Overall, the BBC had the highest coverage with 20 results, followed by the Financial Times and Agence France Presse. CNN had the least coverage with one result. The term “neglected diseases” had good media currency and “sleeping sickness” was far more widely used than trypanosomiasis. The disease most covered was leishmaniasis and the least covered was Chagas. Academic researchers were most commonly quoted as a main source, while the World Health Organization (WHO) and pharmaceutical industry were the least quoted. Journalists generally agreed NTDs had not been adequately covered, but said a lack of real news development and the need to cater to domestic audiences were major obstacles for NTD reporting. All journalists said health agencies, particularly WHO, were not communicating adequately about the burden of NTDs.

Conclusions

Public health agencies need to raise priority for NTD advocacy. Innovative strategies, such as reporting grants or creating a network of voices, may be needed.     

Global radiation oncology waybill

Background/aim

Radiation oncology covers many different fields of knowledge and skills. Indeed, this medical specialty links physics, biology, research, and formation as well as surgical and clinical procedures and even rehabilitation and aesthetics. The current socio-economic situation and professional competences affect the development and future or this specialty. The aim of this article was to analyze and highlight the underlying pillars and foundations of radiation oncology, indicating the steps implicated in the future developments or competences of each.

Methods

This study has collected data from the literature and includes highlights from discussions carried out during the XVII Congress of the Spanish Society of Radiation Oncology (SEOR) held in Vigo in June, 2013. Most of the aspects and domains of radiation oncology were analyzed, achieving recommendations for the many skills and knowledge related to physics, biology, research, and formation as well as surgical and clinical procedures and even supportive care and management.

Results

Considering the data from the literature and the discussions of the XVII SEOR Meeting, the “waybill” for the forthcoming years has been described in this article including all the aspects related to the needs of radiation oncology.

Conclusions

Professional competences affect the development and future of this specialty. All the types of radio-modulation are competences of radiation oncologists. On the other hand, the pillars of Radiation Oncology are based on experience and research in every area of Radiation Oncology.     

Evaluation of Construct and Criterion Validity for the ‘Liverpool Osteoarthritis in Dogs’ (LOAD) Clinical Metrology Instrument and Comparison to Two Other Instruments

Objective

To test the ‘Liverpool Osteoarthritis in Dogs’ (LOAD) questionnaire for construct and criterion validity, and to similarly test the Helsinki Chronic Pain Index (HCPI) and the Canine Brief Pain Inventory (CBPI).

Design

Prospective Study.

Animals

222 dogs with osteoarthritis.

Procedure

Osteoarthritis was diagnosed in a cohort of dogs on the basis of clinical history and orthopedic examination. Force-platform analysis was performed and a “symmetry index” for peak vertical force (PVF) was calculated. Owners completed LOAD, CBPI and HCPI instruments. As a test of construct validity, inter-instrument correlations were calculated. As a test of criterion validity, the correlations between instrument scores and PVF symmetry scores were calculated. Additionally, internal consistency of all instruments was calculated and compared to those previously reported. Factor analysis is reported for the first time for LOAD, and is compared to that previously reported for CBPI and HCPI.

Results

Significant moderate correlations were found between all instruments, implying construct validity for all instruments. Significant weak correlations were found between LOAD scores and PVF symmetry index, and between CBPI scores and PVF symmetry index.

Conclusion and Clinical Relevance

LOAD is an owner-completed clinical metrology instrument that can be recommended for the measurement of canine osteoarthritis. It is convenient to use, validated and, as demonstrated here for the first time, has a correlation with force-platform data.     

Measuring Unsafe Abortion-Related Mortality: A Systematic Review of the Existing Methods

Background

The WHO estimates that 13% of maternal mortality is due to unsafe abortion, but challenges with measurement and data quality persist. To our knowledge, no systematic assessment of the validity of studies reporting estimates of abortion-related mortality exists.

Study Design

To be included in this study, articles had to meet the following criteria: (1) published between September 1^st, 2000-December 1^st, 2011; (2) utilized data from a country where abortion is “considered unsafe”; (3) specified and enumerated causes of maternal death including “abortion”; (4) enumerated ≥100 maternal deaths; (5) a quantitative research study; (6) published in a peer-reviewed journal.

Results

7,438 articles were initially identified. Thirty-six studies were ultimately included. Overall, studies rated “Very Good” found the highest estimates of abortion related mortality (median 16%, range 1–27.4%). Studies rated “Very Poor” found the lowest overall proportion of abortion related deaths (median: 2%, range 1.3–9.4%).

Conclusions

Improvements in the quality of data collection would facilitate better understanding global abortion-related mortality. Until improved data exist, better reporting of study procedures and standardization of the definition of abortion and abortion-related mortality should be encouraged.     

Cost-Effectiveness of a Community Pharmacist-Led Sleep Apnea Screening Program – A Markov Model

Background

Despite the high prevalence and major public health ramifications, obstructive sleep apnea syndrome (OSAS) remains underdiagnosed. In many developed countries, because community pharmacists (CP) are easily accessible, they have been developing additional clinical services that integrate the services of and collaborate with other healthcare providers (general practitioners (GPs), nurses, etc.). Alternative strategies for primary care screening programs for OSAS involving the CP are discussed.

Objective

To estimate the quality of life, costs, and cost-effectiveness of three screening strategies among patients who are at risk of having moderate to severe OSAS in primary care.

Design

Markov decision model.

Data Sources

Published data.

Target Population

Hypothetical cohort of 50-year-old male patients with symptoms highly evocative of OSAS.

Time Horizon

The 5 years after initial evaluation for OSAS.

Perspective

Societal.

Interventions

Screening strategy with CP (CP-GP collaboration), screening strategy without CP (GP alone) and no screening.

Outcomes measures

Quality of life, survival and costs for each screening strategy.

Results of base-case analysis

Under almost all modeled conditions, the involvement of CPs in OSAS screening was cost effective. The maximal incremental cost for “screening strategy with CP” was about 455€ per QALY gained.

Results of sensitivity analysis

Our results were robust but primarily sensitive to the treatment costs by continuous positive airway pressure, and the costs of untreated OSAS. The probabilistic sensitivity analysis showed that the “screening strategy with CP” was dominant in 80% of cases. It was more effective and less costly in 47% of cases, and within the cost-effective range (maximum incremental cost effectiveness ratio at €6186.67/QALY) in 33% of cases.

Conclusions

CP involvement in OSAS screening is a cost-effective strategy. This proposal is consistent with the trend in Europe and the United States to extend the practices and responsibilities of the pharmacist in primary care.     

Canada's Neglected Tropical Disease Research Network: Who's in the Core—Who's on the Periphery?

Background

This study designed and applied accessible yet systematic methods to generate baseline information about the patterns and structure of Canada''s neglected tropical disease (NTD) research network; a network that, until recently, was formed and functioned on the periphery of strategic Canadian research funding.

Methodology

Multiple methods were used to conduct this study, including: (1) a systematic bibliometric procedure to capture archival NTD publications and co-authorship data; (2) a country-level “core-periphery” network analysis to measure and map the structure of Canada''s NTD co-authorship network including its size, density, cliques, and centralization; and (3) a statistical analysis to test the correlation between the position of countries in Canada''s NTD network (“k-core measure”) and the quantity and quality of research produced.

Principal Findings

Over the past sixty years (1950–2010), Canadian researchers have contributed to 1,079 NTD publications, specializing in Leishmania, African sleeping sickness, and leprosy. Of this work, 70% of all first authors and co-authors (n = 4,145) have been Canadian. Since the 1990s, however, a network of international co-authorship activity has been emerging, with representation of researchers from 62 different countries; largely researchers from OECD countries (e.g. United States and United Kingdom) and some non-OECD countries (e.g. Brazil and Iran). Canada has a core-periphery NTD international research structure, with a densely connected group of OECD countries and some African nations, such as Uganda and Kenya. Sitting predominantly on the periphery of this research network is a cluster of 16 non-OECD nations that fall within the lowest GDP percentile of the network.

Conclusion/Significance

The publication specialties, composition, and position of NTD researchers within Canada''s NTD country network provide evidence that while Canadian researchers currently remain the overall gatekeepers of the NTD research they generate; there is opportunity to leverage existing research collaborations and help advance regions and NTD areas that are currently under-developed.     

Beyond a Dichotomous View of the Concepts of ‘Sex’ and ‘Gender’ Focus Group Discussions among Gender Researchers at a Medical Faculty

Introduction

The concepts of ‘sex’ and ‘gender’ are both of vital importance in medicine and health sciences. However, the meaning of these concepts has seldom been discussed in the medical literature. The aim of this study was to explore what the concepts of ‘sex’ and ‘gender’ meant for gender researchers based in a medical faculty.

Methods

Sixteen researchers took part in focus group discussions. The analysis was performed in several steps. The participating researchers read the text and discussed ideas for analysis in national and international workshops. The data were analysed using qualitative content analysis. The authors performed independent preliminary analyses, which were further developed and intensively discussed between the authors.

Results

The analysis of meanings of the concepts of ‘sex’ and ‘gender’ for gender researchers based in a medical faculty resulted in three categories; “Sex as more than biology”, with the subcategories ‘sex’ is not simply biological, ‘sex’ as classification, and ‘sex’ as fluid and changeable; ”Gender as a multiplicity of power-related constructions”, with the subcategories: ‘gender’ as constructions, ‘gender’ power dimensions, and ‘gender’ as doing femininities and masculinities; “Sex and gender as interwoven”, with the subcategories: ‘sex’ and ‘gender’ as inseparable and embodying ‘sex’ and ‘gender’.

Conclusions

Gender researchers within medicine pointed out the importance of looking beyond a dichotomous view of the concepts of ‘sex’ and ‘gender’. The perception of the concepts was that ‘sex’ and ‘gender’ were intertwined. Further research is needed to explore how ‘sex’ and ‘gender’ interact.     

Quilt Plots: A Simple Tool for the Visualisation of Large Epidemiological Data

Background

Graphical representation of data is one of the most easily comprehended forms of explanation. The current study describes a simple visualization tool which may allow greater understanding of medical and epidemiological data.

Method

We propose a simple tool for visualization of data, known as a “quilt plot”, that provides an alternative to presenting large volumes of data as frequency tables. Data from the Australian Needle and Syringe Program survey are used to illustrate “quilt plots”.

Conclusion

Visualization of large volumes of data using “quilt plots” enhances interpretation of medical and epidemiological data. Such intuitive presentations are particularly useful for the rapid assessment of problems in the data which cannot be readily identified by manual review. We recommend that, where possible, “quilt plots” be used along with traditional quantitative assessments of the data as an explanatory data analysis tool.     

Shared Decision Making in Patients with Stable Coronary Artery Disease: PCI Choice

Background

Percutaneous coronary intervention (PCI) and optimal medical therapy (OMT) are comparable, alternative therapies for many patients with stable angina; however, patients may have misconceptions regarding the impact of PCI on risk of death and myocardial infarction (MI) in stable coronary artery disease (CAD).

Methods and Results

We designed and developed a patient-centered decision aid (PCI Choice) to promote shared decision making for patients with stable CAD. The estimated benefits and risks of PCI+OMT as compared to OMT were displayed in a decision aid using pictographs with natural frequencies and text. We engaged patients, clinicians, health service researchers, and designers with over 20 successive iterations of the decision aid, which were field tested during real-world clinical encounters involving clinicians and patients. The decision aid is intended to facilitate knowledge transfer, deliberation based on patient values and preferences, and shared decision making.

Conclusions

We describe the methods and outcomes of the design and development of a decision aid (PCI Choice) to promote shared decision making between clinicians and patients regarding the choice of PCI+OMT vs. OMT for treatment of stable CAD. We will evaluate the impact of PCI Choice on patient knowledge, decisional conflict, participation in decision-making, and treatment choice in an upcoming randomized trial.     

Diagnostics in mycosis fungoides and Sezary syndrome

Aim

The aim of this paper was to present diagnostic methods helping in the recognition of mycosis fungoides (MF) and Sezary syndrome (SS).

Background

Mycosis fungoides is the most common form of primary cutaneous T-cell lymphomas. It is characterized by a distinctive long-term course and malignant T-cell proliferation. MF diagnosis is not easy, mainly due to the atypical clinical presentation of the disease at an early stage.

Materials and methods

Low specific changes, which can be observed at the histopathological examination. Initially, the skin lesions may resemble psoriasis, atopic dermatitis or chronic eczema. Patients are qualified according to the available, and generally accepted WHO-EORTC classification, based on a combination of clinical and histopathological markers. From a clinical point of view, it is also important to carry out the qualification according to the TNMB assessment, which allows to specify the stage of the disease, and is helpful in the monitoring of the course of disease and therapeutic effects.

Results

In this paper we try to present currently available diagnostic methods.

Conclusion

Diagnosis of MF and SS still causes many problems due to less characteristic changes in the early stage of disease and requires wide interdisciplinary knowledge.