Vector borne disease (VBD) emergence is a complex and dynamic process. Interactions between multiple disciplines and responsible health and environmental authorities are often needed for an effective early warning, surveillance and control of vectors and the diseases they transmit. To fully appreciate this complexity, integrated knowledge about the human and the vector population is desirable. In the current paper, important parameters and terms of both public health and medical entomology are defined in order to establish a common language that facilitates collaboration between the two disciplines. Special focus is put on the different VBD contexts with respect to the current presence or absence of the disease, the pathogen and the vector in a given location. Depending on the context, whether a VBD is endemic or not, surveillance activities are required to assess disease burden or threat, respectively. Following a decision for action, surveillance activities continue to assess trends. The European Centre for Disease Prevention and Control (ECDC) has a responsibility to identify, assess and communicate current and emerging dangers to human health from infectious diseases. Vector-borne diseases (VBDs) pose a special challenge to ECDC and national public health authorities due to their complex nature. Interactions between multiple disciplines and responsible health and environmental authorities are often needed for an effective early warning, surveillance and control of vectors and the diseases they transmit. Because many aspects of their complicated transmission cycle are under strong influence of environmental conditions (including weather), adequate risk assessment, early detection, prevention and control of endemic and emerging VBDs may demand approaches other than or in addition to those developed for non-VBDs. This was also recognized by ECDC and is their rationale for funding a network of both medical entomologists and public health experts, VBORNET. The network addresses the prerequisites for vector surveillance activities of arthropods of importance to priority diseases in the European Union. This paper aspires to bridge the gap between entomologists and public health professionals and to facilitate the communication between them. This first strategic paper of the VBORNET project aims to pinpoint the relevance of the different surveillance elements for VBDs important for public health in Europe. Because of the current focus of ECDC on tick-borne diseases, emphasis is placed on VBDs transmitted by ticks in Europe, in particular Lyme borreliosis.Conventionally, public health is defined as the branch of medicine concerned with the prevention and control of disease and disability in a population, and the promotion of physical and mental health of the population on the international, national, or intra-national administrative level. Currently, within the 'one health' initiative, however, medicine is considered more a branch of public health, due to the multidisciplinary character of the latter.Medical entomology is the application and study of insect and other arthropod biology to disease transmission or sanitary matters. In addition to academic institutions, medical entomologists are commonly employed by vector control agencies, but only seldom by public and veterinary health institutes or governmental policymaking bodies.Coxiella or Salmonella, the approach for surveillance requires also other strategies related to other transmission routes.In general, a vector-borne disease is one of which the causative agent is transmitted between vertebrate hosts by another organism (vector). Here, we use the definition in which an arthropod is required for the transmission and propagation of the pathogen. This narrow definition for vector-borne diseases is used because this class requires a common strategy for monitoring, surveillance and also control. In contrast, for pathogens that are optionally transmitted by vectors such as From a public health point of view, five different types of VBD situations (contexts) are identified according to a simplified tabulation of the current presence or absence of the disease, the pathogen and the vector in a given location Table . Here, aLeishmania-infected dogs from the Mediterranean countries to the Netherlands is an example of the latter. Locations where both the vector and pathogen are absent fall into the fifth type of VBD contexts. Despite incidental findings of a Hyalomma tick, the vector of Crimean-Congo haemorrhagic fever (CCHF) [In the first VBD context, endemic human cases arise through the transmission of a pathogen to a susceptible human host from a reservoir host by an indigenous vector species. In the second VBD context, both pathogen and the vector are present but no human cases occur due to biotic, climatic, environmental or societal reasons. Such a situation can exist when a pathogen responsible for a zoonotic disease circulates in the animal reservoir and/or vector populations but without causing human disease burden, either because humans are not infected or because human infections go unnoticed. Low level circulation of West Nile virus in mosquitoes and birds in non-epidemic years in Italy and France are other examples ,2. In thr (CCHF) , the sitIn short, all endemic VBDs fall under context 1 and the various non-endemic VBDs fall under one of the remaining four contexts (2-5). To identify, assess, communicate and ultimately control VBDs, monitoring and surveillance tools, appropriate to the context, are needed."Surveillance of health and disease includes ongoing data collection, analysis to convert this data into statistics, interpretation of this analysis to produce information and dissemination of this information to those who can take appropriate action. At ECDC, this process is aimed at providing appropriate, quality and timely information for key stakeholders in Member States and European Commission. This permits them to take action by planning and implementing more effective, evidence-based public health policies and strategies relevant to the prevention and control of communicable disease in crisis situations as well as in the long term" .According to ECDC, Generally, estimations of the current impact of a health problem are made through calculations of the burden of disease and the assessments of the impact of future outbreaks in an area through quantitative microbial risk assessments. While applicable for endemic VBDs context 1, Table , Table 1Essential for the assessments of both the burden and the health threat is the identification of the appropriate indicators defined as measurable factors that allow decision makers to estimate objectively the size of a health problem (disease burden) or severity of a threat. These indicators should be appropriate for monitoring the processes or the effects of intervention aiming at decreasing the disease burden (VBD context 1) or preventing the introduction of disease burden (VBD contexts 2-5). For VBDs, indicators not only relate to occurrence of human disease, but also on the abundance of pathogen and/or vector   are ofterivm.nl; ,10). OthDecision making, whether to intervene or not, needs to follow the priority setting process taking cost-benefit ratio into account. Especially, in a time of grim governmental budget cuts, focusing on interventions that achieve the largest health gain per euro spent is sensible. For some VBDs, taking action even when as yet there is no disease, might be preferable.Once a decision to intervene has been made to decrease the disease burden (or group/category of diseases) or to mitigate a threat, surveillance should be implemented in order to measure the effectiveness of the intervention.Within the VBD surveillance feedback system three main situations can be distinguished:1. The collected information does not directly provide estimations of disease burden or threat. Consequently, they do not trigger decisions whether to take action or not, but are merely carried out to investigate unknowns of a VBDs transmission, ecology and/or epidemiology, e.g. tick densities or proportion infected ticks.2. The information enables the estimation of disease burden or threat. However, based on the provided data, the decision was made that intervention is not opportune. Surveillance activities continue to follow trends in the situation.3. The provided information on disease burden or threat leads to the decision to intervene. Control actions are developed and implemented. Surveillance activities continue in order to measure the effectiveness of the intervention on reducing disease burden or threat. To do so, surveillance activities also assess change in the direct intervention parameters. For example, in a situation where intervention is implemented to lower the disease burden of Lyme borreliosis by controlling ticks, the change in tick numbers is assessed, and not only the change in disease burden.Obviously, decision making is a crucial step in VBD surveillance feedback system.Data collection is either passive or active. In veterinary and public health systems, passive activities involve voluntary or mandatory reporting of cases, either animal or humans, that are ill enough to have consulted a practitioner. Active systems involve 'searching' for evidence of disease through routine, periodic or continuous data collection . Active The following five types of data collection can be distinguished with respect to the parameter that is considered, exemplified by issues concerning VBDs:Pathogen data collection that focuses on pathogen detection and identification of all levels, including human case, reservoir host, and intermediate host, but also within the vector.1. Serological data collection that aims at the detection of exposure to a pathogen by monitoring immunological responses in the blood of animals or humans.2. Clinical data collection refers to the monitoring of a clinical syndrome that has a significant impact on veterinary or public health, which is then used to drive decisions about health policy and health education. In human health this predominantly refers to passive surveillance i.e. making use of data collected from patients for other purposes than surveillance . If additional data are collected from the patient explicitly for public health surveillance, it is called active or intensified surveillance. Parallel collection of data from controls may be part of the design in active surveillance allowing continuous periodical case-control analysis. In that case the distinction with classical epidemiological case-control studies is that data collection is not restricted to a predefined time period.3. Syndromic data collection refers to an active or passive system that uses case definitions that are based entirely on clinical symptoms without any differential or laboratory diagnosis.4. Risk data collection that focuses not on prevalence of pathogens or clinical features in animals or humans, but on detecting risk factors for disease transmission. The collection of data on the presence/absence and abundance of vectors falls in this category. In addition, it can also include behavioural risk factor surveillance [5. eillance , which iThe use of standards or the harmonization of data collection, representation and reporting facilitates comparability of information over time, across different approaches and across countries and regions, and allows integration of collected data sets for more powerful analyses. To be credible, a standard should be developed through open participatory process by an internationally recognized accredited standards development organization that is also capable of long-term maintenance and evolution of standards .Early warning is a system in which reception of certain predefined signals will trigger interventions. For example, upon the introduction of a certain exotic vector species (a list of important invasive vector species should be available), vector control measures will be taken. In endemic situations an early warning is triggered when the frequency of occurrence of a disease crosses a predefined threshold .Preparedness is a system in which scenarios are described and public health authorities are prepared for outbreaks of diseases prior to their arrival  to VBDs should be investigated. Serological surveys can give insight into changes in exposure by estimating the number of seroconversions, possibly both past and recent. Large population based sero-surveys used for other purposes . Taken together such approaches may allow an estimate of the number of symptomatic VBD infections [Gathering data for the estimation of the disease burden has been quite successful for several food-borne infections . However program ) could bfections  and careHospital referral and sequelae to infections from VBDs could be obtained from GP sentinel systems as well in addition to data from hospital registration systems. Mortality due to VBD, in principle, can be obtained from mortality monitoring by cause of death.Although it is acknowledged that Lyme borreliosis is increasing in many countries ,20, the Due to the lack of the pathognomonic features of the clinical symptoms with the exception of erythrema migrans, and the very low sensitivity of serological tests, incidence rates needed for estimating the disease burden may be largely underestimated or uncertain. The lack of evidence for Lyme borreliosis such as no tick noticed, no erythrema migrans no positive serology will lead to underdiagnosis and underreporting of the sequelae due to the infection.Borrelia spp. this might even be as low as 50% or lower. Clearly, getting the figures correct is a major challenge warranting a concerted action on an international level.The number of tick bites relates to the entomological risk index and depends on the tick population and contact rate of humans and ticks Figure . ExposurIn the Netherlands, methods measuring proxies of disease burden for Lyme disease are under development. In 2011, all physicians are approached to provide retrospective data and to forward extensive questionnaires to their LB patients in the past 12 months. Patient questionnaires are used to reconstruct the evolution of their disease, at what stage they started seeking medical care, therapy failure, related costs and quality of life.The data collection described above generally applies for assessing disease burden of an endemic pathogen. However, the forecasting, early detection, prevention and control of emerging VBDs assess threat Table . In caseVector sampling programs are essential to determine presence/absence and abundance of vectors for risk surveillance and preparedness, but also for pathogen surveillance at the vector level. For the latter, it is important to be able to distinguish between the different levels of pathogenicity (pathotypes) of particular pathogens, or at least between non-pathogenic and pathogenic species. Since VBDs, by definition, require vectors for their transmission, the absence of vectors put the transmission of VBDs to a halt. As a consequence, eradication or control of the vector population is an important intervention tool for VBDs. To measure the effect of vector control, indicators on the presence/absence and abundance of the vector in a given location are essential. In addition, the longevity of individual vectors is also an important determinant, but difficult to monitor. The control of the major host of the vector or the reservoir host(s) of the pathogen may also be means to control the disease.While data on the size of the study population (and consequently the sampling fraction) are readily available for domestic animals and humans, basic information on vector distribution and abundance does not exist for most countries or locations. The most important epidemiological parameters for vector monitoring are vector diversity (presence/absence of species), spatial and temporal variation in vector abundance and pathogen prevalence in the vector population. These parameters can usually only be gathered through active vector surveillances and pathogen prevalence assessment in the collected vectors. In contrast to biases in passive surveillance, sampling strategies in active surveillance can be largely kept under control. Active surveillance, however, is also more costly and labour intensive than passive surveillance. In specific cases, involving mostly ectoparasites such as ticks, passive surveillance alone gives very useful information and is even preferred . SightinPresence/absence data sets would be ideally derived from databases of local monitoring programs assessing the spatial and temporal abundance patterns of vectors, which may be obtained by systematic methods involving consistent sampling and standardized collection protocols. However, more often data is assembled from research projects addressing other questions than presence/absence, or from control agencies. For arthropod vectors such methods typically produce data expressed per unit such as the number of adult mosquitoes per trap night, or effort such as the number of nymphal ticks per flagging hour or drag-meters. These measures are relative numbers and do not estimate density (number per unit area) or absolute size . Accuracy is important as thresholds of abundance such as counts per night are used to make decisions on the density of the vector-borne health risk or the type of focus and intensity of control. Precision and standardization are important if estimates are compared over time and or space. A sampling program consists of a program design, data collection and statistical analyses. However the data analysis is only as informative as the validity of the samples. Studies specifically designed for the collection of absence/presence data would be highly desirable.Since scientific evidence has established the trend of ongoing climate change related to human activities, the potential for climate-induced change in distribution patterns of arthropod vectors and their associated pathogens has emerged as an area of special concern to human and animal health. Model simulations of potential future climate related change in spatial patterns of distribution or abundance of vectors, distribution or prevalence of vector-borne disease agents in vector populations, or distribution or incidence of vector-borne diseases affecting humans or domestic animals have been presented . HoweverIn theory, vector-borne pathogens can be detected in donor or recipient individuals , and in the vector. For monitoring purposes, detection of pathogens in vectors is often the method of choice as vectors might accumulate different pathogenic species from various different sources of reservoir hosts and the presence of the pathogen in vertebrate host can be sometimes of short duration (e.g. for arboviruses). Pathogen detection in wildlife is very laborious and often not possible because of the protected status of many wild animals that precludes the sampling from target organs as is often required for detection. Companion animals such as dogs and horses have been used (successfully) as sentinel animals for human infection . PathogeOne of the major concerns in conducting monitoring programs to detect pathogens  in vectors is the handling of field-collected mosquitoes and ticks. In case of arbovirus detection, RNA degradation should be prevented, and vectors should be maintained in a cold chain through the various handling procedures or immediately after killing be submerged in RNA preservative buffer to reduce the risk of pathogen inactivation -34.Traditional circumstances in the field-collection might not allow for the maintenance of a cold-chain or the prompt processing of the mosquitoes (fresh kill) upon capture. The use of a RNA preservative might increase RNA stability under adverse conditions but is not recommended when species identification is required . The goaFor some pathogens the infection rate is extremely low, even in highly endemic areas. One option is to analyse a number of specimens  together in one pool. In general, increasing the pool size will decrease the test sensitivity. In high endemic areas a loss in sensitivity is more acceptable than in low endemic and in particular arbovirus-free areas. On the other hand the total number of mosquitoes that need to be analysed  will be considerably higher in low endemic and arbovirus-free areas. The extent of the sensitivity loss with increasing pool sizes will depend on the virus-mosquito species combination and the detection assay used  . Also, iMicroscopic analyses of vector-lysates have been gradually replaced by PCR-based techniques. PCR-based techniques greatly improved sensitivity, specificity, and throughput capacity of the number of samples and number of pathogens to be tested. However, PCR-based technologies have some limitations, which should be taken into account during surveillance activities.Borrelia henselae, Rickettsia typhii and R. prowazeki in questing ticks [PCR-based methods are based on the detection of specific DNA sequences: It does not necessarily mean that viable, infectious microorganisms are detected! PCR may result in the amplification of a wrong target DNA or RNA. For the purpose of early warning, the proportion of false positive test results should be minimized ,46. In tng ticks ,50. BesiSurveillance of vector-borne diseases aims at the accurate and timely measurement of the introduction of vectors and pathogens, the incidence of disease, and the effect of preventive, control and curative actions taken.The surveillance feedback system strongly depends on the actions that will be taken upon the outcomes of the surveillance. For example, the control option for measles and mumps is vaccination. A surveillance system measuring newly reported cases of measles and mumps as the indicator parameter is sufficient. In situations where more intervention measures need to be considered or are taken, the identification of indicators that measure the effect of intervention is more challenging. An intervention, for example, that aims to raise the awareness of the risk of acquiring Lyme borreliosis through tick bites might increase the number of reported tick bites or even erythema migrans, but the number of Lyme patients with chronic or late-stage symptoms would decrease. Moreover, the best indicator for an intervention involving tick control on large mammals would be the number of ticks on large mammals. However, acquiring such data is very costly. An alternative indirect indicator here would be the tick density in the environment. Obviously, in a feedback VBD surveillance system Figure , the parIn case the infection is present at an endemic level  authorities need to know in which situation they find themselves for a given VBD with two main groups: endemic  or threat, surveillance should be conducted in order to measure the effectiveness of the intervention.This paper intended to improve the mutual understanding and enhance communication between the communities of medical entomology and public health. Both worlds need to work together to assess the disease burden or threat of vector-borne diseases and to decide on intervention measures. To this end, we developed a framework for a surveillance feedback system for VBDs, which share many characteristics with other infectious diseases. However, because of the involvement of a vector , additional issues need to be considered, as illustrated by the complementary layers to the disease burden pyramid, together forming the intervention and surveillance pyramid for VBDs.For surveillance purposes, international, national and local  authorities need to know in which situation they find themselves for a given VBD table . We suggBesides data on the presence of diseases, pathogens or vectors, convincing data for true absence is essential. Evidence for the latter is, however, much harder to provide. Furthermore, insights from recent scientific and medical developments should be incorporated into the list in Table The availability of information, however, to assess the context of a certain disease situation should be based on monitoring data, for which availability differs greatly between regions or countries. Pan-European surveillance activity could be instrumental for ECDC and their member states to prioritize their future activities, based on evidence. When deemed necessary, contingency plans, including intervention strategies, can be developed and surveillance activities implemented as integrated part of the VBD surveillance feedback system guiding the Member States. Interactions between multiple disciplines and responsible ministries are essential for an effective and efficient early warning, surveillance and control of vectors and VBDs.We hope to increase the awareness of VBD situations in EU, and to present a first step for decisions on the implementation of more harmonized surveillance activities by EU member and associate states.The authors declare that they have no competing interests.All authors participated in the formation of the manuscript's content. MB coordinated the preparation and writing of the manuscript. All authors contributed to helpful discussions, read and approved the final manuscript.


We report the unusual case of a patient with acromegalic arthropathy who presented with severe shoulder arthrosis with marked osteophytosis. This patient is currently pain-free and has good shoulder function 4 years following a Copeland shoulder hemiarthroplasty. Acromegaly is a rare condition of growth hormone oversecretion, but arthropathy is a common feature of the natural history of the disease. This is the first published case report of the outcome of shoulder arthroplasty in a patient with acromegalic arthropathy and demonstrated that a good result can be obtained in this patient. The patient was diagnosed with acromegaly in 1980 and was treated with transsphenoidal surgery and radiotherapy. These treatment modalities were unsuccessful in controlling growth hormone levels. Control was gained many years later by bromocriptine treatment combined with the delayed effect of radiation.A fall in 2005 resulted in an olecranon fracture requiring open reduction and internal fixation, and during followup the patient complained of severe pain and stiffness of the right shoulder. The patient reported an inability to perform any active movements of the shoulder, and passive range of movement was restricted to 40 degrees of elevation, 20 degrees of external rotation, and internal rotation to the coronal midline. X-rays revealed severe arthrosis with extensive osteophytosis Figures  and 2.A right Copeland hemiarthroplasty was performed in 2006, and multiple osteophytes were removed. The procedure was technically challenging, with both a deltopectoral and a Mackenzie anterosuperior approach being required in order to resect the extensive osteophytes.At two weeks postoperatively, an excellent recovery was noted, with a full passive range of movement and active elevation to 90 degrees achieved. By the second postoperative month, the patient was pain-free, active abduction, and elevation of 100 degrees was achieved with good power. At the latest followup, 4 years postoperatively, the patient has a pain-free shoulder and is easily able to perform activities above shoulder height. X-rays show no radiological evidence of loosening or bone resorption Figures  and 4.Acromegaly is a rare disorder  of excess growth hormone secretion by a pituitary adenoma of somatotroph cells. Growth hormone excess has effects on multiple systems; it's most common musculoskeletal effects are gigantism, prognathism, arthropathy, osteopaenia, carpal tunnel syndrome,  hypermobAcromegalic arthropathy most commonly affects knees, hips, and shoulders.  50–60% aTo our knowledge, this case study is the first to present the outcome of a hemiarthroplasty as successful treatment for acromegalic arthropathy of the shoulder. The outcome in this case is particularly interesting as the patient had active acromegaly at the time of surgery, and the acromegalic arthropathy was actively progressing with further osteophytosis.This is the first published case report of the outcome of shoulder arthroplasty in a patient with acromegalic arthropathy and demonstrated that a good result can be achieved in this patient in terms of pain relief and range of movement.


Prune belly syndrome is a rare condition produced by an early mesodermal defect that causes abdominal abnormalities. However, the literature indicates that disturbances related to ectodermal development may also be present. This is the first case report in the literature to suggest that dental abnormalities are part of the broad spectrum of clinical features of prune belly syndrome. Because the syndrome causes many serious medical problems, early diagnosis of abnormalities involving the primary and permanent dentitions are encouraged.The authors report the clinical case of a 4-year-old Caucasian boy with prune belly syndrome. In addition to the triad of abdominal muscle deficiency, abnormalities of the gastrointestinal and urinary tracts, and cryptorchidism, a geminated mandibular right central incisor, agenesis of a mandibular permanent left incisor, and congenitally missing primary teeth  were noted.This original case report about prune belly syndrome highlights the possibility that dental abnormalities are a part of the broad spectrum of clinical features of the syndrome. Therefore, an accurate intra-oral clinical examination and radiographic evaluation are required for patients with this syndrome in order to provide an early diagnosis of abnormalities involving the primary and permanent dentitions. Prune belly syndrome (PBS), described for the first time in the 1800s -4. It isThis syndrome occurs in 1 of 35.000–50.000 live births and most cases (up to 95%) are in males . In anotThe exact etiology of the generalized disturbance is unknown . Accordiet al.[Kabakus et al. found otet al..The hypothesis that there may be a combination of PBS to oral problems has been raised. An association was found between this syndrome and isolated gingival fibromatosis and facial dysmorphism .Other reports have shown that congenital kidney and urinary tract anomalies can cause serious renal function problems in the patient, which occasionally progress to chronic renal failure (CRF) and end-stage renal failure (ESRF) . Thus, tEnamel hypoplasia with generalized hypocalcemic dental lines, demineralization of the trabecular bone of the jaw, loss of the lamina dura, and discontinuity of the mandibular cortical bone were cited as secondary to CRF and hyperparathyroidism . PetechiThis is the first report of a patient with PBS associated with dental anomalies. Given that PBS is a rare disease for which the oral and dental aspects are seldom described in the literature, the goal of the present case report is to contribute to a better understanding of this syndrome.The child was included in the Baby Clinic of Araçatuba Dental School program for preventive dental care at 11 months of age, and this clinical case report is being presented when the child is 4 years old.The child’s mother reported that her child had a PBS during the anamnesis procedure, and he had been undergoing regular medical check-ups since birth because of this finding. The pregnancy was planned by the parents, and the mother received prenatal care. An ultrasound examination performed during the fifth week of gestation showed a gastrointestinal malformation of the fetus.A subsequent morphologic ultrasound examination revealed the following findings: gastrointestinal abnormality, enlarged kidneys, and a low amount of amniotic liquid. However, although the parents were aware that their child would be born with health problems during the gestation period, they did not receive conclusive information regarding the baby’s condition.The baby boy was born prematurely at 7 months by caesarian, with a weight of 1450 kg and height of 41 cm. According to the mother, postnatal examination revealed that the baby had a distended and wrinkled abdomen, resembling a dried prune. The form of the penis was abnormal, with an enlarged diameter and lax excess skin. Despite these characteristics, an immediate diagnosis was not made.Numerous medical examinations were performed during this period, and the results combined with the clinical features resulted in a diagnosis of PBS. The child does not have relatives with any type of syndrome.These features included abdominal musculature deficiency, gastrointestinal and urinary abnormalities, a larger-than-normal bladder, malformed kidneys , and cryptorchidism .Although only 1 kidney was found to function normally on medical examination, the mother stated that the child did not have problems associated with this and denied impairment of renal function. The mother reported episodes of urinary infection requiring antibiotic treatment that were related to excess skin on the penis in the first to third years of life.Moreover, the mother reported that the child did not have cardiac or pulmonary disease, and no anal alteration was found. The alterations mentioned above may result from the syndrome.The characteristics of the inferior and superior limbs, hands, feet, fingers, and toes were normal. Structural abnormalities were only noted in both of the child’s knees, which is a feature consistent with this syndrome, according to the mother.The child underwent plastic surgery of the abdomen and penis and a surgery to position the testes correctly at 2 years of age. The present state of the child’s abdomen is shown in Figure The following oral health status findings were observed at the initial clinical examination and in all follow-up appointments: no dental caries, low plaque indices, no supragingival calculus deposits, and no other periodontal problems. The following dental anomalies were clinically and radiographically noted when the primary dentition was complete: a geminated mandibular right central incisor with 1 root and 1 canal; the absence of a mandibular permanent left incisor Figure  and 3; aThe child’s family received preventive and educational dental guidance and the child is seen by a dentist 4 times a year since his placement in the Baby Clinic of Araçatuba Dental School program. The parents have maintained strict oral health care of the child, which is demonstrated by the aspects cited above regarding the child’s oral health.The monitoring system of the program and the parent’s access to appointments whenever necessary, i.e., not only in cases of dental emergency, allows the dental team to register any manifestation of oral diseases. Oral soft tissue manifestations arising from disorders caused by the syndrome -19 have Although metabolic disorders can affect mineralized structures , an alteWhile the etiology of PBS is unknown, the literature related to this disorder describes it as a mesodermal defect  consistiPBS has a broad spectrum of clinical features with different levels of severity, and the authors highlight the possibility that dental anomalies can be a part of this rare syndrome. The consensus is that treatment should be multidisciplinary. For maintenance of oral health, accurate intra-oral clinical and radiographic evaluations are required for patients with this syndrome in order to provide an early diagnosis of other abnormalities involving the primary and permanent dentitions.Written informed consent was obtained from the parents for publication of this Case report and any accompanying images. A copy of the written consent is available for review by the Series Editor of this journal.The authors declare that they have no competing interests.MDB and COF participated in the clinical dental care of the patient and have continued performing regular clinical and radiographic follow-up. RFC supervised the clinical dental care of the patient. MDB was responsible for the literature search and wrote the paper. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1472-6831/12/56/prepub


The role of the cardiac lymphatic system has been recently appreciated since lymphatic disturbances take part in various heart pathologies. This review presents the current knowledge about normal anatomy and structure of lymphatics and their prenatal development for a better understanding of the proper functioning of this system in relation to coronary circulation. Lymphatics of the heart consist of terminal capillaries of various diameters, capillary plexuses that drain continuously subendocardial, myocardial, and subepicardial areas, and draining (collecting) vessels that lead the lymph out of the heart. There are interspecies differences in the distribution of lymphatic capillaries, especially near the valves, as well as differences in the routes and number of draining vessels. In some species, subendocardial areas contain fewer lymphatic capillaries as compared to subepicardial parts of the heart. In all species there is at least one collector vessel draining lymph from the subepicardial plexuses and running along the anterior interventricular septum under the left auricle and further along the pulmonary trunk outside the heart and terminating in the right venous angle. The second collector assumes a different route in various species. In most mammalian species the collectors run along major branches of coronary arteries, have valves and a discontinuous layer of smooth muscle cells. The existence and importance of cardiac lymphatics has been neglected for many years. Recently more attention has been devoted to this system since it was discovered that besides helping maintain intramyocardial pressure and preventing tissue edema it plays an important role in many heart pathologies, such as atherosclerosis and interstitial fibrosis . LymphatNowadays, due to improved techniques that make it possible to distinguish blood vessels from lymphatics, the lymphatic system is no longer considered as secondary to the blood circulatory system , 10. FurKnowledge of the structure and location of capillary lymphatic system and major draining branches in the heart in various species as well as its relationship with the circulatory system would help in understanding the proper functioning of the heart. Additionally, due to an immense development of modern technologies and the use of transgenic animals in many research tasks, a better understanding of normal cardiac lymphatic vessel embryogenesis is also in progress. Thus, the aim of this paper is to summarize the current knowledge on cardiac lymphatic anatomy in various species and discuss the basic steps of lymphatic vessel development and maturation.myocarditis with the use of Trypanosoma. Due to its strong affinity to lymphatic vessels, this parasite made it easier to study the morphology and confirm the appearance of lymphatic capillaries, for example, in valves or papillary muscles [The techniques for visualization of gross anatomy of lymphatics vary. A useful method to demonstrate location of lymphatics is to inject India-ink to subepicardium, myocardium, or subendocardium, areas of the conduction system and atrioventricular valves, via a needle, glass pipette, or syringe. Additionally, other filling substances have been used by investigators in the past such as radiopaque iodized oil, latex or vital dye (trypan blue), Evans blue, resins, micropulverized barium sulfate, and carbon  combined with polyvinylpyrrolidone , 13–18.  muscles .Nowadays, modern techniques allow lymphatic endothelial cells (LEC) with specific markers to be easily demonstrated in various species on histological, confocal microscope sections and by the use of the “whole mount” immunohistochemical method –25. For μm in diameter and joined larger channels of 60 to 100 μm. Those channels coursed in the superior part of the interventricular septum parallel to the atrioventricular sulcus. The apices of papillary muscles were penetrated by lymphatic plexuses of 30–45 μm with occasional channel vessels of 150 μm. In dog hearts, these lymphatic vessels formed a denser network than in the pig heart. The human subepicardial plexus showed a similar pattern to that of the canine and porcine capillary plexuses [μm in diameter; however, their further courses were not determined [The earliest detailed data on cardiac lymphatics has been described by Patek  who obseplexuses  and courtermined . The ventermined , 28. Atrtermined . The lymtermined . In humatermined , 34.Shimada and colleagues noted that the lymphatic plexus was less extensive in the atria than in the ventricles . In the The capillary lymphatic system has been demonstrated by various techniques to be present in the conduction tissue of mammalian hearts: in the sinoatrial node of man, dog, and rabbit , 34, 35,Adventitial lymphatics have been reported to be rather extensive along the aorta as well as along the coronary arteries in mammalian species. Our work indicated that by the end of the prenatal life an extensive adventitial lymphatic plexus was developed at the roots of the aorta and the pulmonary trunk in mouse hearts . In the The myocardial lymphatic capillaries were less numerous as compared to blood capillaries. The ventricular myocardium of cat, rabbit, and human hearts possessed an average of 1,029 blood capillaries per 1,000 muscle cells  were irregular in shape and diameter, possessed many branches, and exhibited tortuous courses. The caliber of lymphatic capillaries varied, depending on the author, animal species, or technique used, and ranged from 20 to 100 μm  or even μm [μm  or largeThe smaller lymphatic capillaries of the myocardial plexus converged forming larger drainage lymphatic vessels that contained valves. These drainage vessels coursed in the subepicardial space . In the In Macaca monkeys the pattern of cardiac lymphatics has been reported to be comparable to that of man, dog, and pig . Due to The structure and course of precollectors and collecting subepicardial vessels were demonstrated in our lab by injecting mouse hearts with India ink mixed with gelatin and showing their course as dividing branches and merging branches. These vessels vary in size and diameter along their course. The left major collector in murine hearts corresponds to the major cardiac trunk in other mammalian species. It drains the paraconal interventricular sulcus around the left conal vein towards the left side of the pulmonary trunk and upwards to the mediastinum via the nearest lymph node  . The secContrary to human, pig, and dog heart where major lymphatic precollectors/collectors accompany coronary artery branches, in the mouse and rat hearts these vessels accompany branches of cardiac veins (are in a close proximity to major cardiac veins).Patek  and otheCardiac lymphatic flow has been suggested to be propelled by a passive pumping , for exaThe development of cardiac lymphatic vessels has been the subject of a few studies. As development of lymphatics commonly followed blood vessel formation in the body of the embryo, the same sequence was observed in developing organs, such as the heart; that is, the lymphatics occurred later than blood vessels. The species studied were rats, mice, and birds such as quail and chicken –25.+/Lyve-1− strands of cells, clusters, and finally tubules were found to invade the great arteries from the mediastinum of mouse fetuses at 12-13 dpc  have been studied fragmentarily. Only a few papers dealing with injection techniques and with the use of lymphatic markers have been published , 48, 49.2-13 dpc  and Proxectively , 25. Conectively .It has been demonstrated by our group that cells bearing macrophage phenotypes were incorporated to lymphatic vascular walls during prenatal and early postnatal development of cardiac mouse lymphatics . The exaAn immense interest in biology and development of cardiac lymphatic vessels is nowadays justified because they are involved in normal functioning of the cardiovascular system and play a role in various pathologies. Understanding interspecies differences in normal structure and development of lymphatics is important for comparison of those structures in transgenic animals. Animal models with modification of selected genes help pursue a developmental process of lymphatic vessel formation and its regulatory mechanisms. Our recent work on mouse cardiac lymphatics using multiple markers of the lymphatic endothelium points to interspecies differences in capillary plexus location and in the course of major draining vessels. The lymphatics of a normal adult mouse heart, contrary to those in other mammalian species, are restricted to the outer zone of the myocardial wall (as has been previously suggested by Böger and Hort with the use of old techniques) . Two maj


The development, implementation and evaluation of assessments require considerable resources and often cannot be carried out by a single faculty/institution. Therefore some medical faculties have founded cooperation projects which mainly focus on the exchange of multiple choice questions (MCQs).Since these cooperation projects do not entirely support all relevant processes in terms of preparation, implementation and evaluation of assessment, in 2006 the Medical Assessment Alliance (MAA) was founded for mutual support. In addition to MCQs the MAA started to develop innovative assessment formats and facilitate content through a coordinated exchange of experiences. To support cooperation within this network, the web-based Item Management System (IMS) was developed which supports all processes of the assessment workflow as an all-in-one working platform.At present, the Alliance has 28 partner faculties in Europe. More than 2.800 users in 750 working groups are collaborating. Currently 90.000 questions have been stored in the IMS. Since 2007, nearly 4.600 examinations have been successfully conducted.This article describes in detail the unique features of the IMS and contrasts it with the item management systems of other associations. Although medical faculties have considerable freedom in designing their assessments , the devIn English-speaking countries there are consortia which have developed item pools to share assessment content  represents, in contrast to the IDEAL and UMAP consortia, a significant development in using a shared web-based platform, called the Item Management System (IMS). This supports staff in the preparation, implementation, evaluation, and reporting of assessments according to existing guidelines.This article describes the MAA/IMS and critically evaluates experience with it, future challenges, and possible solutions.In 2006 the medical faculty of Heidelberg, the Charité Berlin and the LMU Munich set up the MAA  to facilThere are annual two-day consensus meetings with the leaders of staff responsible for assessments from all partner faculties. These, for example, jointly develop innovative assessment formats, exchange OSCE checklists, and construct guidelines for the review process. Joint decisions are made on awarding external contracts, adjusting processes, and improving the user-friendliness of the system. In 2010 and 2011 the following topics were addressed in Alliance meetings: Review activity, development of a composite internal progress test and selection tests, exchange of OSCE checklists, and the development of encounter cards.Since regular formative feedback promotes competency-based teaching , the AllTo strengthen inter-faculty collaboration, the web-based IMS was implemented. It is an all-in-one platform which supports the work of staff responsible for the preparation, administration, evaluation and reporting of assessments . The cosCurrently 2.800 users are registered with IMS who are working together in 750 groups. In these groups, people are no longer just limited to their own faculty but have found inter-faculty colleagues with whom they design items and examinations and cooperate on other aspects of assessment processes.To date 4.600 examinations composed of a total of 123.000 questions have been successfully administered to more than 800.000 medical students using IMS. 584.000 statistical test parameters  have been calculated and these help in the compilation of future exams. These have been uploaded to the IMS database.The database stores more than 90.000 questions and a number of these can be made accessible to other faculties by the item author. Currently 19% of all questions are available to all users of the Alliance for their exams. Figure Each IMS user receives their own protected user account and can access the IMS based on clearly defined roles and responsibilities with a personalized view. To support collaborative work, colleagues can set up working groups and design exams as a team or conduct quality control processes.In IMS, the complete assessment sequence is laid out as described by the Assessment Committee of the German Medical Association (Gesellschaft für Medizinische Ausbildung GMA) and the guidelines of the Centre of Excellence for Medical Assessments . In the Assessment content (items) can be created and saved in IMS in various formats, including long menu, free text, MCQs, key feature cases, OSCE checklists, and structured oral examinations . EnterinIn addition to classification by subject and sub-subject, every faculty can create their own classification system in the IMS to classify tasks according to their specific learning targets. This allows, for example in the case of a Swiss Faculty, for classification into a Bachelor’s and Master’s system which is differentiated into several course units and modules. The implementation of a classification system linked to the German National Competency-based Learning Targets Catalogue in Medicine (NKLM)  is in prAccording to existing guidelines, all examination questions should be evaluated in a pre-review prior to their use in a live assessment . This caFigure Examinations can easily be assembled by dragging and dropping questions into exams. Apart from tasks created personally by the authors, they also have access to a public pool available to all Alliance members.To further promote efficiency in the organisation of assessments, it is possible to choose various blueprint sub-functions. For example, 1) a table of contents blueprint can be used for targeted exam compilation , 2) an evaluation blueprint can be used to define the distribution of points, and 3) a date blueprint can be used to monitor all examinations of a faculty over a fixed period of time (e.g. for compiling an exam in a timely manner before the exam date).IMS allows the user to create printable exam sheets (in PDF format). The items can be permuted using flexible algorithms to create several versions of an examination which could be sorted by subject, item-type, etc.. In addition, standard interfaces for transferring data to computer-based assessment programs or document readers are integrated.An examination can be conducted on paper, as traditionally the case, computer-supported using machine-readable answer sheets, computer-based in a computer lab, as an oral exam, or as an OSCE. By using IMS interfaces, each partner faculty can use its own established systems or standard Alliance tools, such as the document reading system Klaus®  or the cFollowing the examination, the raw data can be evaluated using IMS. The system has interfaces for importing this data from a document reading system or a computer-based examination system. The system calculates scores for subtests and total test scores based on parameters which can be individually set, such as the maximum number of points of a question or the grading algorithm . IMS also calculates detailed test statistics, including question and exam indicators such as difficulty, accuracy or the index of discrimination .Potentially wrong tasks can be identified through conspicuous statistical test parameters. In such a case, the points awarded in such tasks can be corrected as part of a post-review or tasks can be removed from the overall exam score. Such information is stored in the IMS in order to document the deficiencies and to facilitate revision by the author. Furthermore, comments by teaching staff or test participants can be recorded.Figure Apart from direct support for the aspects listed above, IMS offers further infrastructure assistance to study deaneries such as monitoring upcoming exams and keeping a record of actors used as standardized patients.The IMS also provides support for the development and implementation of standardised objective clinical or oral-practical examinations (e.g. OSCE). The organisation and management of actor-patients is often an additional challenge due to the large number of people involved. The module integrated into IMS for this purpose can be used to manage events, assign roles, and reimburse the actor-patients. In addition to its use in OSCE examinations, this module can also be used to organise the use of actor patients in the teaching of the regular curriculum. This not only frees up considerable resource in the planning of teaching activities but also improve quality assurance.Consensus Statement and Recommendations from the 2010 Ottawa conference which states that “the medical education community still needs to develop a deeper understanding of how technology can underpin and extend assessment practices” [Exchanging assessment experiences and exam tasks created at other faculties form an important basis for improving medical education. The use of a web-based platform to support assessment significantly eases such cooperation. Both the MAA and the IMS assessment platform are in line with the actices” .Within 5 years it has been possible to establish a successful and stable inter-faculty cooperation among 28 faculties. Despite differences in the human and technical resources of the partner faculties, MAA has a high acceptance which, amongst other things, is clearly demonstrated by the speed with which it has grown to include 28 participating faculties and the intensive use of the system. The accession of the medical faculty of Geneva as a French-speaking university and of the faculties in the Netherlands as Dutch-speaking universities is a further signal that the IMS not only offers extra value due to its extensive collection of questions (which for the most part available in German only) but is a complete system with a high degree of functionality and the workflow assistance. This differentiates the IMS from other known assessment associations such as the IDEAL Consortium, which focuses mainly on building a database of exam questions  and UMAPOne of the advantages of the IMS is that authors can decide whether they want to share items with others. Some people choose not to publish their items because they only want them to be used locally. The full benefit of the system will be realized as authors begin to see the quality and efficiency inherent in cross faculty collaboration.The analysis of user experience shows that the further development of the IMS promotes quality development processes at participating faculties. For example, the review activity of IMS participants increased significantly during the past year following changes to the process which included e.g. more flexible ways to perform a review of an item or a better presentation of the review results. To further expand review activity, cooperation in specialised assessment associations  is consistently promoted and supported by the MAA .A technical challenge is the development of powerful search algorithms. At present, more than 17.000 questions are managed in the shared pool and the number is rising steadily. Using additional filters which take statistical test parameters, review results, usage frequencies of the tasks, and MeSH terminology into account , there aCurrently, in spite of the variety of question formats  available in the IMS, the main type used are MCQs (96%). This mirrors results which showed that MCQs are still the predominant format at German medical faculties . In thisThe development of competency-based assessment formats and the creation of the relevant content are increasingly seen as a major challenge internationally. Qualification frameworks like the competencies developed by the Accreditation Council for Graduate Medical Education (ACGME)  or the NIn the past, assessments at individual faculties have focused on summative examinations. It is crucial for steering the students’ learning process to receive feedback on their current knowledge through formative tests. For example, the progress test is a format, growing in popularity, demonstrates growth in knowledge over time . As the The concept of having many partners who contribute to improving quality by providing their special knowledge is one of the main reasons for the success of MAA. Through the consensus meetings and the direct exchange between doctors, psychologists, and IT developers, the system enhances the quality development processes at the faculties (see Table 1). Additionally the participation in the MAA provides economic benefits for each participating faculty because this way of cooperating directly results in savings for every partner.Over a three year period, nearly 450.000€ were needed to develop the first version of the IMS. Following this, the development and maintenance of the IMS is financed by the fees of the participating faculties. The costs for one faculty in the pilot phase is 10 € per student. Thereafter, the cost per year is 30 € per student to a maximum of 25.000 € per year per faculty. This includes organizational and technical support as well as further development of the IMS. Participating faculties benefit economically because they do not have to develop a similar system but only pay for the running costs of the IMS.Through the cooperation of various university partners, the task of creating fair, high quality, competency-based assessments can be shared. Overall, the MAA has fulfilled the expectations in terms of the creation and design of exams. The increase in the number of users and the continuous increase in the number of items suggest that the IMS has been accepted and is used extensively. The IMS has proven itself as a practical and efficient solution for the entire assessment workflow, so that new challenges can be explored and addressed.th 2012.The authors report no conflicts of interest. The authors alone are responsible for the content and writing of this article. Authors guarantee that the manuscript has not been published elsewhere and it has not been submitted simultaneously for publication elsewhere. Data concerning the number of items, examinations, review etc. has been updated on the occasion of resubmission and represents the status by June 30AH specialised in health technology assessment and evaluates the impact of Informatics on Medical Education. He drafted the manuscript and participated in the coordination of the project. KB implemented the first version of the IMS and coordinates the development of the IMS-project. AM provided the acquisition of the data. JJ and JHS are responsible for the medical education program at the Medical hospital and conceived the project. JN and AT participated in the finalization of the manuscript and provided valuable feedback to the IMS and the manuscript. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1472-6920/12/63/prepub


Undoubtedly, the identification of patient suitability for a telerehabilitation assessment should be carried out on a case-by-case basis. However, at present there is minimal discussion of how telerehabilitation systems can accommodate and adapt to various patient factors, which may pose challenges to successful service delivery. The current study examines a subgroup of 10 patients who underwent an online assessment of their swallowing difficulties. Although all assessments were completed successfully; there were certain patient factors, which complicated the delivery of the online assessment session. The paper presents a discussion of the main patient factors observed in this cohort including the presence of speech and/or voice disorders, hearing impairment, dyskinesia, and behavioural and/or emotional issues and examines how the assessment session, the telerehabilitation system, and the staff involved were manipulated to accommodate these patient factors. In order for telerehabilitation systems to be more widely incorporated into routine clinical care, systems need to have the flexibility and design capabilities to adjust and accommodate for patients with varying levels of function and physical and psychological comorbidities. Telerehabilitation services often involve intensive, detail-oriented, and interactive assessments. Hence it is accepted that patients are ideally served by systems and technology designed to optimise high quality visual and audio during a real-time interaction . It is, The use of telerehabilitation to conduct clinical assessments of dysphagia is an area of practice which is currently still in its infancy. Hence the system requirements necessary to optimise capabilities for both the user and the patient during online assessments of swallowing are still being established. Initial work by Lalor et al.  describeBuilding on these lessons learnt, Sharma et al.  publisheWard et al.  subsequeAs part of the development and evaluation of any new telerehabilitation application, there needs to be systematic investigation of the system design elements and their functionality when applied with the desired population or rehabilitation service. Hence it is the aim of the current investigation to examine further the issues which potentially impacted on the service delivery of clinical dysphagia assessment via the telerehabilitation system as used by Ward et al. . Recent Participants were selected from the original cohort of 40 dysphagic participants who participated in the study reported by Ward et al. . Particin = 3) had more than one issue impacting the session. Notes made by the online clinician about the sessions for these 10 participants were subsequently reviewed, and common issues were noted. Specifically, four main participant-related issues were encountered, which were found to influence the clinicians' ratings of the efficiency of online assessment for each individual. The issues included the presence of (1) speech and/or voice disorders (six participants), (2) hearing impairments (three participants), (3) coexisting movement disorders (dyskinesia) (one participant), and (4) behavioural and or emotional issues (three participants) . Some inHowever, despite the online clinician indicating that the telerehabilitation environment may not have been the most efficient means to assess these particular 10 individuals, a review of each session revealed that all assessments were still completed successfully, and a diagnostic decision was achieved for each individual. Furthermore, the level of agreement between the online and the FTF clinicians ratings for the primary diagnostic decisions relating to (a) safe fluid consistency and (b) safe food consistency revealed 100% exact agreement . Hence i The following sections discuss the four key patient factors, which were found to influence the telerehabilitation session and the strategies used to compensate for and adapt to these issues. A weak and dysphonic voice quality and reduced speech intelligibility of six individuals see  added coIn the study by Hill et al. , which aFor three individuals, the presence of unaided hearing impairment created challenges during the assessment. The hearing impairments caused occasional communication breakdowns between the participants and the O-SP. In all instances it is very probable that the telerehabilitation session would have run more smoothly if the hearing aids were available and in working order for the session. The experience with these three individuals highlights the need to ensure patients are adequately prepared prior to the session and clear instructions given to family/care staff to ensure patients who require hearing aids come adequately prepared for the session. Although the individuals were unaided during the session, it was possible to compensate for this and successfully complete the assessment. The Assistant helped by adjusting the speaker volume at the patient end, and by repeating the instructions of the online clinician directly to the patient. The O-SP also modified instructions to make them as short, simple, and clear as possible for the patient. Although it was not necessary for any of these three participants, preparation of simple written instructions for patients, which could be typed in by the online clinician and electronically posted onto the screen of the computer system at the patient's end, could also be used to help compensate for auditory-perceptual breakdowns.Although the presence of a hearing impairment does not prohibit individuals from participating in telehealth services –12, in tOne patient presented with a diagnosis of Parkinson's disease and associated cervical dyskinesia. Due to her dyskinesia, she was unable to retain a position that maintained her within the usual visual field set for the web camera during assessments. Zoom capabilities were also of limited use. To compensate, the free standing camera, positioned for a wider angle field of view, was used to enable visualization of the head, neck, and upper torso. While not ideal, particularly during food and fluid trials when close focus on the mouth and upper throat is preferable, the wider angle helped compensate for the movement and maintain vision of the participant most of the time. The free standing camera could also be positioned by the Assistant relative to the participant, rather than constantly repositioning. A further issue with this participant was that she displayed signs of fatigue, and short breaks were needed throughout the session. The need for these short breaks lengthened the duration of the assessment session. The findings highlight the need to schedule a longer assessment session for certain individuals with more complex conditions. Behavioural or emotional issues impacted the duration and the flow of three of the assessment sessions. For these individuals, the Assistant was instrumental in facilitating the sessions success. Previous studies have reported that attention deficits can be challenging to manage in an online environment , 13, 14.Patient impulsivity raised challenges during the food/fluid trials. This was particularly an issue when participants took large or multiple sips of fluids during the trials, unheeding instructions from the O-SP. Again, in these cases, intervention by the Assistant helped to control the rate of oral intake and discourage talking until it was clear they had completed the swallow. One participant (Pt 10), who was largely noncommunicative, presented with a flat affect and provided delayed responses, was challenging to assess online. Strategies that helped during the session were to provide instructions slowly and for Assistant to repeat instructions to ensure that the participant attended to the tasks required.For Participant 1, her emotional state impacted the flow of the session. Her acute awareness of her loss of function since surgery meant she frequently became emotional when she was unable to carry out tasks . She frequently required some time to regain composure to continue the assessment session. Reassurances from the O-SP and the physical presence of the Assistant helped to assist.Dysphagia is a symptom caused by a wide range of aetiologies including neurological injury/insult, degenerative disorders, trauma, surgical interventions, and even the natural aging process. Hence there is a high likelihood that patients referred for a dysphagia assessment via telerehabilitation will present with a range of comorbidities as well as altered cognitive and emotional states. The current study demonstrates that in spite of the range of challenges raised by the altered capabilities of these 10 participants, all assessments were completed successfully through modifications of the current equipment and through the help of the Assistant at the patient end. It is acknowledged, however, that this is a small cohort and the systems capabilities to accommodate all possible patient factors cannot be considered to be complete. As new systems are proposed and technology advances, ongoing investigation into how these systems perform and can adjust to compensate for various patient factors will emerge. From such research, it will be possible in the future to more clearly identify the minimum telerehabilitation system requirements needed to assess patients with various comorbidities.


Priority grade assessment according to urgency level of the patients (triage) is considered vital in emergency medicine casualties. Little is known of the experiences of pre-hospital emergency medicine triage performed by General Practitioners (GPs) in the community. In this study we bring such experiences from a Norwegian island community, with special emphasis on over- and undertriage.In the island municipality of Austevoll, Western Norway, where the GPs and the ambulance services both take part in all medical emergency cases, all these cases were recorded during a 2-year period (2005–2007). We compared the triage of the patients at the stage of the telephone reception of the incident, and the subsequent revision of the triage at the first personal examination of the patient.236 emergency medical events were recorded, comprising 240 patients. Of these, 42% were downgraded between the stages , 11% were upgraded  and 47% remained in unchanged priority group. Of the diagnostic groups, acute abdominal cases had the highest probability of being upgraded between stages, while the aggregated diagnostic group of syncopes, seizures, intoxications and traumas had the highest probability of being downgraded. The principal reason for upgrading was lack of necessary information at the stage of call. In a minority of cases the upgrading was due to real patient deterioration between stages.In pre-hospital triage of emergency patients, downgrading happens between notification of events and actual patient examination in a substantial proportion. Upgradings of cases are considerably fewer, but the potential serious implications of upgrading warrants individual scrutiny of such cases. Dispatch guidelines, urgency assessment and clinical triage have long been applied in the health services in order to prioritize among patients with acute serious illness or injury. Such syovertriage must generally be accepted[undertriage represents potential risks for clinical outcome, as emergency situations may develop into life-threatening situations during the pre-hospital course.Pre-hospital triage of emergency patients is necessarily an inexact process and some degree of accepted. This meintuitive triage. Also, in the majority of primary care events, triage is not used to prioritize between patients, but for assessing the level of urgency for a single patient, so that mode and speed of adequate transport can be determined. Accordingly there is a lack of data about reliability and validity of such pre-hospital assessments of adequate level of response. It must be anticipated that intuitive triage will have uncertain accuracy and generate both under-triage and over-triage of patients, with subsequent changes of priority grade along the course.In day to day practice in many pre-hospital situations, however, assessment and triage of patients are performed without use of a formal triage system. Instead, different kinds of health care personnel apply the best appraisal that one is capable of, based on personal competence and experience, and thus assign priority and risk in an informal manner. This is typically the situation in primary care, where general practitioners (GPs) or ambulance personnel on call outs attend an emergency scene, using spontaneous and intuitive synthesizing of the available information and background knowledge. This process may be called In a recent study we analyzed all patients managed by the pre-hospital emergency services in a Norwegian community during a two-year period,7. Here,The study design and main results of the project evaluating emergency medical events in Austevoll municipality over a period of two years 2005–2007) have been published elsewhere,7. Auste have beeAll activity associated with medical emergencies in Austevoll in the period October 2005 through September 2007 was recorded, with an exception of acute psychiatric events and patients giving birth. All emergencies were recorded, irrespective of whether the notification was made by the EMCC (which comprised for a little more than one third of the events) or more local instances. A medical emergency was defined as an event for which the doctor on call, based on the first notification, found the acuteness of the incident serious enough to see the patient without any delay. Information was also recorded for events which the doctor initially assessed as less serious, but for which clinical examination provided information that would have led to a call out, if the first notification had given the doctor this information.For every medical emergency the doctor and ambulance personnel completed a registration form immediately after the event. The form was available electronically and on paper. Doctors were asked 84 questions and ambulance personnel 29 questions about clinical and practical aspects of the event. Upon notification of the event, the GP triaged the situation on the basis of all available information and background knowledge, irrespective of classifications made by the EMCC operator or others. The GP used the same categories as Index[Differences between diagnostic groups were tested for statistical significance using cross tabulation and Pearson’s chi square tests. For analyses of cells containing less than five cases Fisher’s exact test was used. We subsequently adjusted for gender and age applying binary logistic regression analyses. Results were reported as ORs with 95% confidence intervals (CI). Age was recoded into three groups . P <0.05 were considered statistically significant. SPSS version 18.0 was used.The project was defined as a quality assurance project by the Norwegian Social Science Data Services, and thus not reported to the Regional Committee for Medical and Health Research Ethics. It was reported to the Norwegian Data Inspectorate, according to the Norwegian Health Registry Act.A total of 236 emergency medical events were recorded, involving 240 patients (mean and median age 51 years (SD 28), 107 (45%) female). The majority of the events (84%) were caused by acute illness and the rest by injuries. GPs’ assessment of priority grade upon notification of emergencies yielded 79 red responses, 142 yellow, and 17 green, while two patients were not classified.101 of the 240 patients (42%) underwent a severity downgrading when seen by the doctor , 26 (11%) were upgraded , while the priority grade was not changed in 111 (47%)  had been seen by a doctor for serious comorbidity and/or the same diagnosis or a precursor of it, in the near time span before the emergency incident. Language problems concerning the communication with the caller were not found to be a problem in any case.An upgrading to the highest priority grade means that the GP unexpectedly found the patient in a state of possible danger for life when arriving at scene or after initial clinical assessment. Table In this two-year observational study of 240 medical emergency patients in an island community of Norway we found that severity priority grade was downgraded from call reception to after patient examination in 42%, representing a large volume of overtriage. Upgradings were much less frequent (11%), although half of these cases were to red (acute) priority grade, thus representing undertriage and a potential for harm. In about one out of four the upgrading was due to deterioration of the clinical condition, and could not have been initially detected. Upgrading was frequent in acute abdomen, while downgrading was particular frequent in the aggregated diagnostic group of syncope, seizures, intoxication and trauma.The strength of our study is that we present population based data from a comprehensive and consecutive collection of all medical emergencies in a Norwegian community. The study also enabled us to compare GPs’ triage of all the patients at two stages, so that we could evaluate magnitudes and characteristics of priority changes (overtriage and undertriage) in a real-world patient setting. We have not found similar studies from a primary care emergency medicine setting.GPs involvement in community emergency service varies, and the involvement in Austevoll may be greater than in the average municipality in Norway. In spite of this, we maintain that our findings are valid and generalizable, because participating GPs differed in experience and competence.Weaknesses of our study include that we do not have any information of what was the final diagnosis and seriousness of the patients who were admitted to hospital. Further, a formal triage system was not applied, as the triage was done as intuitive triage. However, all the GPs used the categories from Norwegian index for medical emergency assistance, which tDowngrading of syncope, seizures, intoxication and trauma cases may be explained by the often experienced dramatic messages that are conveyed in such cases in the initial communication process, while most such cases turn out to be less serious when encountered and examined. The same sequence of reduced perception of seriousness between stages makes upgrading significantly low-frequent for this combined diagnostic group. Cardiac and cerebrovascular disorders had a small, but statistically significant, reduced probability of being downgraded. This might be because the GPs at the moment of patient examination did not feel confident to rule out serious conditions like myocardial infarction or stroke.The detected undertriage of acute abdomen cases was a bit unexpected, and may be a finding of importance for GPs on call in out-of-hours services. Especially, an initial call of an acute abdomen case together with a suspicious history or symptoms for gastrointestinal bleeding should warrant high attention.. When patients readmit with possible alarming symptoms after a recent evaluation, this might be an indication of serious acute disease.We found frequent upgradings in cases where the patient recently had an encounter with a GPConcerning the upgradings to red response, the examination of the qualitative data shows that in most cases we found no significant deterioration in the time course between initial call and examination. However, in most cases the real priority grade was not possible to assess until clinical assessment, examination and e.g. ECG was performed, and the GP could summarize the situation into a final conclusion. In some cases, however, it would have been possible for the GP to comprehend the real priority grade from the moment of call reception, if she or he had penetrated the available anamnestic data in a better way, or had had the possibility of obtaining better information during initial assessment by telephone. Based on our findings there is thus a potential for reducing both undertriage and overtriage, although we are aware that a reasonable portion of over-triage must be accepted as we have shown that a doctor’s competence and skills at scene often are necessary to analyze the clinical situation accurately,9.We were especially interested in any learning messages from cases where the patient unexpectedly was found in a possible life-threatening situation. Indications of gastrointestinal bleeding deserve closer attention from the first doctor. The combination of dyspnoea and other risk factors also warrants high priority. The same is true when information is unclear as to what has happened with injured patients. A post-hoc audit and discussion of such cases among doctors and ambulance personnel may be useful.In general, there are few studies that evaluate the effectiveness and reliability of triage systems for pre-hospital emergency services. The Index used as intuitive triage by GPs). We have shown that both undertriage and overtriage occur in such situations, but it is neither possible to assess or specify the magnitude or implications of these inaccuracies nor can we compare the informal and formal systems.It is thus not possible, based on scientific evidence, to claim that any formal triage system would perform better than available clinical appraisal in a primary care emergency scene (In conclusion, in this comprehensive analysis of all medical emergency cases of a Norwegian rural community during two years, we found that acute abdomen cases had a significantly higher probability of being upgraded in priority, while the combined diagnose group of syncope, seizures, intoxication and trauma had a higher probability of being downgraded. Upgrading of cases are considerably fewer than downgradings, but the potential serious implications of upgrading warrants individual scrutiny of such cases.None of the authors have anything to declare.SR has conceived the study, written the draft and revisions of the manuscript, performed the statistical analyses and approved the final version of the manuscript. EM and SH have contributed substantially to its content and several revisions. All authors take the responsibility for the paper as whole. All authors read and approved the final manuscript.


Shoulder complaints are commonly seen in general practice and physiotherapy practice. The only complaints for which general practitioners (GPs) refer more patients to the physiotherapist are back and neck pain. However, a substantial group have persistent symptoms. The first goal of this study is to document current health care use and the treatment process for patients with shoulder syndromes in both general practice and physiotherapy practice. The second goal is to detect whether there are differences between patients with shoulder syndromes who are treated by their GP, those who are treated by both GP and physiotherapist and those who access physiotherapy directly.Observational study using data from the Netherlands Information Network of General Practice and the National Information Service for Allied Health Care. These registration networks collect healthcare-related information on patient contacts including diagnoses, prescriptions, referrals, treatment and evaluation on an ongoing basis.Many patients develop symptoms gradually and 35% of patients with shoulder syndromes waited more than three months before visiting a physiotherapist. In 64% of all patients, treatment goals are fully reached at the end of physiotherapy treatment. In general practice, around one third of the patients return after the referral for physiotherapy. Patients with shoulder syndromes who are referred for physiotherapy have more consultations with their GP and are prescribed less medication than patients without a referral. Often, this referral is made at the first consultation. In physiotherapy practice, referred patients differ from self-referrals. Self-referrals are younger, they more often have recurrent complaints and their complaints are more often related to sports and leisure activities.There is a fairly large group of patients with persistent symptoms. Early referral by a GP is not advised under current guidelines. However, in many patients, symptoms develop gradually and a wait-and-see policy means more valuable time may pass before physiotherapy intervention takes place. Meanwhile a long duration of complaints is a predictor for poor outcome. Therefore, future research into early referral is required. As physiotherapists, we should develop a way of educating patients to avoid lengthy waiting periods before seeking help. To prevent high costs, physiotherapists could consider a classification of pain and limitations and wait-and-see policy as used by GPs. With early detection, a once-off consultation might be sufficient. Shoulder complaints are the most common complaints of the extremities in an average physiotherapy practice; 9.8% of all patients present with this type of problem . Low bacShoulder complaints can be roughly divided into problems with the glenohumeral joint , shoulder instability, acromioclavicular or sternoclavicular complaints, cervical or cervicothoracic dysfunction and problems with structures in the subacromial space. It has been estimated that approximately 44-80% of all shoulder complaints originate from these anomalies of structures in the subacromial space 10]. Thi. Thi10].As referred to above, there is debate on the best treatment methods for patients with shoulder syndromes. Dorrestijn et al.  and KromUsing data from registration networks, the present study describes patient characteristics and the treatment process for patients treated by a representative group of GPs or physiotherapists who were unaware of the specific purpose of this study. In the Netherlands, patients can access physiotherapy professionals directly  and it is known that use, treatment and outcome may differ depending on the mode of access . HoweverTo describe patient characteristics and the process of care for patients with shoulder syndromes, data were used from the Netherlands Information Network of General Practice (LINH)  and the According to Dutch legislation entitled ‘Regulations on medical research involving human subjects’, ethical approval is required for medical research in which persons are subjected to treatment or are required to behave in a certain manner. As this was not the case for the present study, ethical approval was not necessary. Nevertheless, the Dutch Data Protection Authority was notified of the research. In addition, pursuant to the Personal Data Protection Act, data were collected anonymously, patients were informed about the research by posters and leaflets in practice waiting rooms and patients had the opportunity to refuse participation. The research was carried out in accordance with the Helsinki Declaration.From the LINH database, all patients with shoulder syndromes, ICPC L92  who visIn LINH, for every patient, a care episode was defined as the time between the first and last visit for L92 in 2008. Care episodes were constructed on the basis of EPICON, which is an algorithm that groups ICPC-coded contact records from electronic medical records in general practice into care episodes. This algorithm calculates care episodes for each year separately . Prescriptions were registered in accordance with the Anatomical Therapeutic Chemical (ATC) classification system. Because not all prescriptions were linked to a diagnosis, a list was made of the most common prescriptions based on a group of patients with a known diagnosis of L92. For each of these prescriptions, the number of times they were prescribed during a shoulder-syndrome care episode was determined. Interventions were registered using CTG codes, which are standardised codes set by the Dutch healthcare authority for health care claims to health insurers. For the most common codes with a diagnosis of L92, the number of times they occurred during a care episode of shoulder syndrome was determined. Referrals were also registered and for the most common referrals with a diagnosis of L92, the number of times they occurred during a care episode of shoulder syndromes was established. Based on the information in the referral module, the total group of patients was split in two: patients referred for physiotherapy during the care episode versus patients who were not referred for physiotherapy.In LIPZ, a series of consecutive treatment sessions for shoulder syndromes was considered to be a care episode. For each care episode, the gender and age of the patient was registered. Also recorded were the duration of the complaint at intake, whether it was a recurrent complaint (when the complaint appeared after a symptom-free period of at least four weeks and at most two years) and the treatment goal(s). At the end of the treatment, therapists registered a maximum of three interventions that were applied in at least 50% of the treatment sessions. Besides these features of the treatment plan, the outcome was also registered . Based on the mode of access, the total group of patients was split in two: patients referred by their GP versus patients who accessed physiotherapy directly.Descriptive statistics were calculated for all variables using Stata 11. Chi-square tests (a = 0.05) were used to test differences in categorical data between patients with shoulder syndromes with and without a referral; two-sample t-tests were used for continuous data.Incidence of shoulder syndromes in general practice in 2008 was 8.5 patients per 1000 patients, or 38% of all shoulder complaints. Prevalence was 14.2/1000 patients/year, or 42% of all shoulder complaints. GPs treated 82% (n = 1983) of patients themselves and referred 18% (n = 445) to one or more other clinicians, mainly to a physiotherapist  or a medical specialist  . In two-thirds of the referred patients (n = 199), the referral for physiotherapy was given during the first GP consultation without further treatment by the GP, seven percent (n = 20) were referred within two weeks and a further seven percent (n = 22) within one month. In general practice, there was no difference in terms of age or gender between patients who were referred for physiotherapy and those who were not  of all patients presented with shoulder syndromes, accounting for 27% (n = 1182) of all shoulder complaints. Of these, 76% (n = 895) were referred by a GP, 12% n = 139) by a medical specialist and 12% (n = 148) accessed the service directly. Self-referrals differed from referred patients; they were younger and more often male  waited more than three months before visiting a physiotherapist, 45% (n = 469) had already had physiotherapy previously, severity of the complaint (between 0 and 10) was rated 7 and in 75% (n = 224) of the patients the symptoms had developed gradually. Common combinations of interventions were exercises aimed at functions and mobilisation or massage. At the end of treatment, the results did not differ between referred patients and self-referrals: in 64% (n = 668) of all patients with shoulder syndromes the treatment goals were fully reached.The present study sought to determine the characteristics of the patient population and the types of treatment for patients with shoulder syndromes in both general practice and in physiotherapy practice and secondly, whether there are differences between patients who are treated by their GP, those who are referred for physiotherapy and those who access physiotherapy directly. The results show that there are differences between these populations both in terms of the characteristics of the patient and the treatment they receive.Eight out of ten patients with shoulder syndromes that visit a GP are treated solely by the GP and not referred to another clinician. In these patients, treatment was aimed at reducing pain and inflammation. The number of patients referred for physiotherapy in the present study was comparable to that of Kuijpers et al. . Most pashoulder syndromes make less use of direct access; only 13% came through direct access compared with 22-44% of the entire patient population attending the physiotherapist from 2006 to 2010. It is known that self-referral decreases with age. The average age of patients with shoulder syndromes was 56 and therefore, the number of self-referrals can be expected to be lower in comparison with the general patient population in physiotherapy practice. Pain severity might also explain the limited number of self-referrals among patients with shoulder syndromes. Pain is common in shoulder syndromes and the average score on the numeric rating scale for pain severity was seven for both referred patients and self-referrals. Kennedy et al. found a comparable level of pain severity in patients with soft tissue disorders [In previous research, it was demonstrated that patients with shoulder complaints make as much use of direct access as the general patient population when attending the physiotherapist . Howeverisorders . Given tisorders . In threisorders . It seemisorders . HoweverWith regard to the physiotherapy treatment itself, the results of the present study show that in patients with shoulder syndromes, exercises aimed at functions, mobilisation and massage are the main types of intervention, which is partly in line with what is known about the treatment of shoulder injuries. Literature reviews by Green et al.  and KromPhysiotherapy treatment results in a positive outcome in 64% of patients with shoulder syndromes, regardless of the mode of access. In the general patient population in physiotherapy practice, 68% fully reach the treatment goals . Of the Registration networks cover a large number of patients, providing a rich source of data. However, there are some limitations to this method of data collection. In LIPZ, information is collected on all diagnoses. This means detailed information specific to shoulder syndromes is not available; e.g. the existence of neck or back problems or repetitive or provocative movements in work or sport. Furthermore, diagnoses are based on referral letters, which can be ambiguous or imprecise. For example, terms such as ‘shoulder complaints’ are used, without giving further information. The procedure for diagnosing specific shoulder disorders is further complicated by a lack of consensus on the diagnostic criteria. Where diagnosis is difficult, complaints may be described as general shoulder complaints in the first instance, perhaps more so by less experienced clinicians. In this study, these general shoulder complaints are not included as shoulder syndromes in order to prevent heterogeneity as much as possible. Therefore, the results are based on a more homogeneous group of patients, but this may have led to an underestimation of the number of people attending the physiotherapist with shoulder syndromes. To measure the outcome of physiotherapy treatment, an indication of the extent to which the treatment goals were met is registered in LIPZ by the physiotherapist. This is a subjective outcome measure. In 2010, an indication of symptom severity at the beginning of the care episode and at the end was introduced. When patients do not come back, this information, which has to be obtained from the patient, remains unknown. As a result, this outcome measure is only known for a subgroup of patients, which is insufficient for a reliable investigation. Therefore, physiotherapists give an indication of the result, so that an outcome measure is known for every patient. In the present study, referred patients and self-referrals achieved the treatment goals to the same extent. Since the outcome is measured in the same subjective manner, it is not expected that the results would be different. Nevertheless, ideally, patient-reported outcome measures should also be studied.In LINH, a diagnosis was not registered for every consultation. Prescriptions, referrals and interventions are calculated for the total care episode of shoulder syndromes and might therefore have been overestimated. However, this calculation only concerned a selection of frequently used prescriptions, referrals and interventions which prevents the inclusion of those actually relating to a diagnosis other than shoulder syndromes.Finally, data is based on two different patient populations. The physiotherapy database is much smaller and, therefore, a longer time period was selected. However, there were no policy changes in the area or indications that the group of patients consulting their GP changed over the period of the study. Nevertheless, it would be interesting to investigate the care process in a multidisciplinary network incorporating the activities of various health care professionals.In summary, there are differences in general practice between patients who are referred for physiotherapy and those who are not. Patients who are referred are prescribed less medication and are often referred at the first consultation with their GP. This goes against current guideline for GPs and could result in unnecessary or higher costs. On the other hand, possibly due to the gradual onset of complaints and a wait-and-see policy, for many patients, it takes quite a while before they see a physiotherapist, even though it is suggested that a long duration of complaints could be a predictor for poorer outcomes. When a restricted range of motion is the main problem, it is arguable that patients receive less medication but a quick referral to a physiotherapist. Future research into the long term cost-effectiveness of an early referral could demonstrate whether this leads to better outcomes and should therefore be the preferred treatment.As clinicians, we should also develop a way of educating patients about shoulder syndromes to prevent them waiting too long before they seek help. However, this can only be cost-effective when the profession sets clear guidelines on indications for physiotherapy, especially since there is debate on the value of clinical diagnostic tests. The classification of pain and functional limitations and adoption of the wait and see policy as used by GPs could be an example or starting point. Perhaps with early detection, a once-off consultation in which advice is given will be sufficient, especially when pain is severe. The consequences of such initiatives for the prognosis of the individual patient as well as cost-effectiveness should be investigated first.GP: General Practitioner; LINH: Netherlands Information Network of General Practice; LIPZ: National Information Service for Allied Health Care; ICPC: International Classification of Primary Care; EPICON: An application to group ICPC-coded diagnoses from electronic medical records in general practice into episodes of care; ATC: Anatomical Therapeutic Chemical; CTG: Codes for claiming health care services to health insurers; PT: Physiotherapy Practice; NSAID: Non Steroidal Anti Inflammatory Drugs; NRS: Numeric Rating Scale; SD: Standard DeviationThe authors declare that they have no competing interests.MK contributed to conception and design, acquisition of data, analysis and interpretation of data and was involved in drafting the manuscript. IS contributed to conception and design, acquisition of data, analysis and interpretation of data and was involved in revising the manuscript critically. CD contributed to acquisition of data, analysis and interpretation of data and was involved in revising the manuscript critically. DB contributed to analysis and interpretation of data and was involved in revising it critically. CV contributed to conception and design, analysis and interpretation of data and was involved in revising the manuscript critically. All authors read and approved the final manuscript.The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-2474/14/128/prepub