The Jamaican Haemophilia Registry: Describing the burden of disease.

INTRODUCTION: Jamaica has an estimated 200 persons with haemophilia (PWH), who face significant constraints in access to specialized haemophilia care, including access to clotting factor concentrates. AIM: The aim of this paper is to establish the current burden of disease in PWH in Jamaica. METHODS: PWH were enrolled through the University Hospital of the West Indies, Jamaica. The impact of haemophilia was assessed using a comprehensive battery of heath outcome measures that included the following: laboratory, clinical information and validated outcome measures of joint structure and function, activity, and health-related quality of life (HRQoL) to provide a health profile of the Jamaican haemophilia population. RESULTS: In all, 45 PWH were registered (mean age: 29, range: 0.17-69 years), including 13 children (<18 years of age) and 32 adults. In this sample, 41 had haemophilia A (30 severe) and 4 had haemophilia B (3 severe); 10 patients with haemophilia A were inhibitor positive. The results indicate that adults with haemophilia in Jamaica have significant joint damage: mean Haemophilia Joint Health Score (HJHS) = 42.1 (SD = 17.3); moderate activity levels - mean Haemophilia Activities List (HAL) score = 64.8 (SD = 17.8); and low HRQoL scores - mean Haemo-QoL-A score = 62.3 (SD = 19.4). Results for children are also reported but should be interpreted with caution due to the small sample size. CONCLUSIONS: There is a very high burden of disease in PWH in Jamaica. The health profiles reported in this paper are an essential first step in advocating for a multidisciplinary Comprehensive Care Program for assessment and care of PWH in Jamaica.

Describing the quality of life of boys with haemophilia in China: Results of a multicentre study using the CHO-KLAT.

INTRODUCTION: The treatment of haemophilia varies across countries and across regions within some countries. Similar variation has been observed in health-related quality of life (HR-QoL). Relatively little is known about the HR-QoL of boys with haemophilia in China. AIM: The aim of this study was to describe the HR-QoL of boys with haemophilia in China using the Canadian Haemophilia Outcomes-Kids Life Assessment Tool (CHO-KLAT). METHODS: Boys (4-18 years of age) with haemophilia and their parents were enroled in a cross-sectional study. All parents/guardians of study subjects were requested to complete a CHO-KLAT questionnaire during a clinic visit, and report on several other clinical and socioeconomic factors in the past year. Boys who were > 7 years also completed the CHO-KLAT. RESULTS: A total of 269 parents of boys with haemophilia, from 13 hospitals in 12 provinces, were enroled during 2014. The boys ranged from 4.0 to 17.9 years of age; 91% had haemophilia A, most had moderate (52%) or severe (36%) disease, and most were receiving sub-optimal on-demand therapy or low-dose prophylactic therapy. Child self-report CHO-KLAT scores were available for 171 boys ≥7 years of age and ranged from 24.2 to 85.3 with a mean of 57.6 (n = 171). Parent proxy-reported CHO-KLAT scores ranged from 25.0 to 88.7 with a mean of 55.1 (n = 269). CONCLUSION: HR-QoL scores in boys with haemophilia in China were substantially lower than reported from Canadian and European boys with haemophilia. Longer term prospective studies are required to examine the factors impacting the HR-QoL for boys with haemophilia in China.

Health-related quality of life questionnaires in individuals with haemophilia: a systematic review of their measurement properties.

BACKGROUND: The evaluation of health related quality of life (HRQOL) is essential for a full assessment of the influence of an illness on patients' lives. The aim of this paper is to critically appraise and compare the measurement properties of HRQOL questionnaires studied in haemophilia. METHODS: Bibliographic databases (Embase, Medline, Cinahl and PsycInfo) were searched for articles evaluating measurement properties of HRQOL questionnaires in haemophilia. Articles were excluded that did not report HRQOL measurement properties, or when <50% of the study population had haemophilia. The methodological quality of the selected studies was evaluated using the COSMIN checklist. The measurement properties of the HRQL questionnaires were rated as 'positive', 'indeterminate' or 'negative', accompanied by levels of evidence. RESULTS: The search resulted in 1597 unique hits, of which 22 studies were included. These articles evaluated three questionnaires for children (CHO-KLAT, Haemo-QoL and one unnamed measure) and five for adults (Hemofilia-QoL, Haemophilia Well-Being Index, HAEMO-QoL-A, Haem-A-QoL, and SF-36). The CHO-KLAT was the paediatric measure that showed the strongest measurement properties in high-quality studies. The Haemophilia Well-Being Index and HAEMO-QoL-A performed best among the adult measures. None of the studies reported measurement error and responsiveness. CONCLUSION: Our findings suggest that there is no need for new disease-specific HRQOL questionnaires for haemophilia, but rather that additional research is necessary to document the measurement properties of the currently available questionnaires, specifically focusing on the structural validity, measurement error and responsiveness of these questionnaires.

Comparing the burden of illness of haemophilia between resource-constrained and unconstrained countries: the São Paulo-Toronto Hemophilia Study.

INTRODUCTION: Although the regular replacement of clotting factor concentrates (prophylaxis) has been well established as the standard of care for severe haemophilia, the high cost of factor concentrates has limited access to prophylaxis in countries with under-developed or developing economies. AIMS: We studied the health gap that could be addressed by providing unlimited access to clotting factor concentrates with implementation of long-term prophylaxis initiated from an early age in life. METHODS: We performed a cross-sectional study of a random, representative sample of boys with moderate and severe haemophilia at three haemophilia treatment centres in Sao Paulo, Brazil, and one centre in Toronto, Canada. RESULTS: Canadian subjects were more often treated with prophylaxis, and began treatment at an earlier age. Fewer Canadian subjects had bleeds within the preceding 6 months (19 vs. 34, P = 0.003). Canadian subjects had lower (better) Pettersson radiographic scores (1.5 vs. 6.0, P = 0.0016), lower (better) Hemophilia Joint Health Scores (5.5 vs. 10.5, P = 0.0038), higher (better) Activity Scale for Kids scores (96.6 vs. 92.0, P = 0.033), more time spent in vigorous activity, and higher (better) social participation scores. CONCLUSIONS: Our findings suggest that increasing access to clotting factor concentrates for young boys with severe haemophilia is a global imperative.

A prospective study of health-related quality of life of boys with severe haemophilia A in China: comparing on-demand to prophylaxis treatment.

INTRODUCTION: Treatment for boys with haemophilia in China is rapidly improving; however, comprehensive outcomes have not been examined prospectively. AIM: The aim of this study was to evaluate the effect of short-term full-dose prophylaxis compared to on-demand treatment, on the Health-Related Quality of Life (HR-QoL) of boys with severe haemophilia A (HA) in China. METHODS: Boys with severe HA (FVIII<1%) completed 3 months of on-demand treatment and 3 months of full-dose prophylaxis (25 FVIII IU per kg 3x per week). The primary outcomes were child- and parent-reported Canadian Hemophilia Outcomes - Kids Life Assessment Tool (CHO-KLAT) scores. The number and type of bleeds and Activities Scale for Kids (ASK) scores were also recorded. RESULTS: Analyses included 23 boys between 4 and 15.9 years of age. The number of bleeds decreased by 94% on prophylaxis (P < 0.0001, Wilcoxon Signed-Rank test). The mean child-reported CHO-KLAT scores for boys ≥7 years (n = 20) was 61.4 (±10.9) during on-demand treatment and 61.9 (±11.4) following short-term prophylaxis (P = 0.72, paired t-test). The mean parent-reported CHO-KLAT score during the on-demand phase was 54.4 (±10.5) with an increase of 3.8 points (±8.1; P = 0.04, paired t-test) following prophylaxis. CONCLUSIONS: Child-reported CHO-KLAT scores were lower in boys with severe HA in China than reported in countries with access to full-dose prophylaxis. Boys reported higher HR-QoL scores than their parents. Small improvements in ASK scores were noted following the prophylaxis phase. These changes were only significant in the parent-reported CHO-KLAT scores. Longer term prospective clinical trials are needed in China to determine the impact of prophylaxis on HR-QoL in boys with severe HA.

Validity of the Portuguese CHO-KLAT in Brazil.

INTRODUCTION: It is essential to assess the health-related quality of life outcomes of boys with haemophilia in Brazil. The Canadian Haemophilia Outcomes-Kids Life Assessment Tool (CHO-KLAT) was recently adapted for this population. AIM: To test the construct validity of the Portuguese version of the CHO-KLAT. METHODS: We recruited 50 boys, with moderate [factor VIII (FVIII) level 1-5%] or severe (FVIII level <1%) haemophilia, to participate in a descriptive study to establish a baseline understanding of the current status of boys with haemophilia in Brazil. All boys were required to complete the Brazilian CHO-KLAT and Brazilian Pediatric Quality of Life Inventory (PedsQL) by self-report. We examined the correlation between the CHO-KLAT and PedsQL scores to establish the construct validity of the Brazilian version of the CHO-KLAT. RESULTS: We obtained CHO-KLAT and PedsQL data from 35 boys with severe haemophilia and 15 with moderate haemophilia. They ranged in age from 7.3 to 18.0 years, with a mean of 13.0 years. They reported a mean CHO-KLAT score of 72.3 (range = 44.1-93.9). The mean PedsQL score was 79.9 (range = 45.7-96.7), with physical health (mean of 83.9) being better than psychosocial health (77.8). The Pearson's correlation between CHO-KLAT and PedsQL was 0.47 respectively (P < 0.001). The CHO-KLAT had a moderate and inverse relationship with the degree to which they were bothered by their haemophilia (ρ = -0.53), while the PedsQL had a weaker relationship (ρ = -0.27). CONCLUSION: The results confirm the validity of the Portuguese version of the CHO-KLAT. This measure is now available for clinical trials in boys with haemophilia in Brazil.

Generic and disease-specific quality of life among youth and young men with Hemophilia in Canada.

BACKGROUND: This study was undertaken to explore the longitudinal patterns of health-related quality of life (HRQoL) among youth and young adults with Hemophilia A (HA) over a 3-year period. This report presents the baseline characteristics of the study cohort. METHODS: Males, 14 to 29 years of age, with predominantly severe HA were recruited from six treatment centres in Canada. Subjects completed a comprehensive survey. HRQoL was measured using: the CHO-KLAT2.0 (youth), Haemo-QoL-A (young adults) and the SF-36v2 (all). RESULTS: 13 youth (mean age = 15.7, range = 12.9-17.9 years) and 33 young adults (mean age = 23.6; range = 18.4 -28.7 years) with moderate (7 %) and severe (93 %) HA were enrolled. All were on a prophylactic regimen with antihemophilic factor (Helixate FS®) during the study. The youth had minimal joint damage (mean HJHS = 5.2) compared to young adults (mean HJHS = 13.3). The mean HRQoL scores for youth were: 79.2 (SD = 11.9) for the CHO-KLAT, and 53.0 (5.5) and 52.3 (6.8) for the SF-36 Physical Component Summary (PCS) and Mental Component Summary (MCS) scores respectively. The mean HRQoL scores for young adults were: 85.8 (9.5) for the Haemo-Qol-A, and 50.8 (6.4) and 50.9 (8.8) for PCS and MCS respectively. PCS and MCS scores were comparable to published Canadian norms, however significant differences were found for the domains of Physical Functioning and Bodily Pain. The disease-specific HRQoL scores were weakly correlated with the PCS for youth (CHO-KLAT vs. PCS r = 0.28, p = 0.35); and moderately correlated for the MCS (r = 0.39, p = 0.19). Haemo-QoL-A scores for young adults were strongly correlated with the PCS (r = 0.53, p = 0.001); and weakly correlated with the MCS (r = 0.26, p = 0.13). Joint status as assessed by HJHS was correlated with PCS scores. A history of lifelong prophylaxis resulted in better PCS but worse MCS scores. CONCLUSION: Despite having hemophilia, the youth in this cohort have minimal joint disease and good HRQoL. The young adults demonstrated more joint disease and slightly worse HRQoL in the domains of physical functioning and pain. The data presented here provide new information to inform the selection of Health Related Quality of Life (HRQoL) instruments for use in future clinical trials involving persons with hemophilia. TRIAL REGISTRATION: ClinicalTrials.gov : NCT01034904. Study funded by CSL Behring Canada.

International cross-cultural validation study of the Canadian haemophilia outcomes: kids' life assessment tool.

Health-related quality of life (HRQoL) assessment is recognized as an important outcome in the evaluation of different therapeutic regimens for persons with haemophilia. The Canadian Haemophilia Outcomes-Kids' Life Assessment Tool (CHO-KLAT) is a disease-specific measure of HRQoL for 4 to 18-year-old boys with haemophilia. The purpose of this study was to extend this disease-specific, child-centric, outcome measure for use in international clinical trials. We adapted the North American English CHO-KLAT version for use in five countries: France, Germany, the Netherlands, Spain and the United Kingdom (UK). The process included four stages: (i) translation; (ii) cognitive debriefing; (iii) validity assessment relative to the PedsQL (generic) and the Haemo-QoL (disease-specific) and (iv) assessment of inter and intra-rater reliability. Cognitive debriefing was performed in 57 boys (mean age 11.4 years), validation was performed in 144 boys (mean age 11.0 years) and reliability was assessed for a subgroup of 64 boys (mean age 12.0 years). Parents also participated. The mean scores reported by the boys were high: CHO-KLAT 77.0 (SD = 11.2); PedsQL 83.8 (SD = 11.9) and Haemo-QoL 79.6 (SD = 11.5). Correlations between the CHO-KLAT and PedsQL ranged from 0.63 in Germany to 0.39 in the Netherlands and Spain. Test-retest reliability (concordance) for child self-report was 0.67. Child-parent concordance was slightly lower at 0.57. The CHO-KLAT has been fully culturally adapted and validated for use in five different languages and cultures (in England, the Netherlands, France, Germany and Spain) where treatment is readily available either on demand or as prophylaxis.

Validation of the Chinese version of the Canadian Haemophilia Outcomes-Kids' Life Assessment Tool (the CHO-KLAT).

It is important to assess the health-related quality of life outcomes of boys in China, but there are no tools validated for this purpose. The objective of the study was to assess the validity of the Simplified Chinese version of the CHO-KLAT2.0 . We recruited 60 boys with either haemophilia A (HA) or haemophilia B (HB) and their parents from four regions in China, and assessed the validity of CHO-KLAT compared to the PedsQL. All participants complete the CHO-KLAT a second time 1-2 weeks later to assess reliability. The boys ranged in age from 7 to 18 (mean = 12.4; SD = 3.03) years. The severity distribution was: mild (9), moderate (10) and severe (41). On-demand therapy was received by 26 boys, while 18 received low-dose prophylaxis (HA: 10 IU kg(-1) 2-3 times/week(-1) , and HB: 20 IU kg(-1) 1 time/week(-1) ). The mean CHO-KLAT scores were 63.7 (SD = 10.6) for child-report and 58.3 (SD = 11.4) for parent-report. Validity was supported by a correlation of 0.67 (P < 0.0001) with the PedsQL for child-report and 0.64 (P < 0.0001) for parent-report. The test-retest reliability was 0.88 (95% CI: 0.82-0.94) for child-report, and 0.90 (95% CI: 0.86-0.95) for parent-report. Inter-rater reliability was 0.46 (95% CI: 0.26-0.66). CHO-KLAT scores were 11 points higher among patients who had been on prophylaxis 3 times per week for ≥24 weeks. These results confirm the reliability and validity of the Chinese version of the CHO-KLAT. This measure is suitable for use in prospective clinical trials in boys with haemophilia in China.

Promoting resilience and wellbeing through an outdoor intervention designed for Aboriginal adolescents.

INTRODUCTION: Aboriginal people in Canada (First Nations, Inuit and Métis) have a lower health status compared to the Canadian population. There is a particular concern about the mental health and wellbeing of First Nations adolescents living on reserves. Interventions following principles of outdoor education and adventure therapy appear to be an appropriate fit for this population. These approaches have proven effective in non-Aboriginal populations, yet there is very little evidence on the efficacy of these types of program for Aboriginal adolescents. The primary purpose of this study was to evaluate the impact of an outdoor adventure leadership experience (OALE) on the resilience and wellbeing of First Nations adolescents from one reserve community. The secondary purposes were to explore whether this impact was sustainable, and whether there were any intervening factors that may have influenced the impact. METHODS: The collaborative research team used a mixed-method design to evaluate the 10-day OALE for adolescents from Wikwemikong Unceded Indian Reserve in northern Ontario, Canada. The main outcome assessed was resilience, measured by self-report, using the 14-Item Resilience Scale (RS-14). Several other exploratory measures assessed other aspects of health and well-being. The questionnaire package was administered at three different time periods: (T1) one day before the OALE; (T2) one month after the OALE; and (T3) one year after the OALE. The Mental Component Score (MCS) of the SF-12v2 was used to confirm any changes in resilience. Open-ended questions were appended to the questionnaire at the 1-year point to identify any intervening factors that may have impacted any changes in resilience and wellbeing. The primary analysis compared mean RS-14 scores at T1 with those at T2. Responses to the open-ended questions were analyzed using content analysis. RESULTS: Over two summers (2009 and 2010), 73 youth 12-18 years of age from Wikwemikong participated in a standardized 10-day OALE program. This represented 15% of the on-reserve population of adolescents in this age range. Survey responses from 59 (80.8%) participants were available for analysis at T1, compared to 47 (64.4%) at T2 and 33 (45.2%) at T3. The mean RS-14 score was 73.65 at baseline, and this improved 3.40 points (p=0.011) between T1 and T2. However, the resilience scores at T3 (1 year post-OALE) had a mean of 74.19, indicating a return back to pre-OALE levels. The mean MCS score at T1 was 48.23 and it improved over the subsequent two time periods. Several intervening factors reported at T3 may have influenced the decrease in resilience scores from T2 to T3. These included changes in family living situation, death in the family, and other life stressors that occurred over the course of the year. CONCLUSIONS: Outcome scores from this study provide a unique glimpse into the self-reported health and wellbeing for adolescents within one First Nations community in Canada. The OALE program was beneficial in promoting resilience for adolescents in Wikwemikong over the short-term. Future studies are necessary to assess whether the OALE (or similar outdoor type interventions) are effective within other communities.

Process and experience of cross-cultural adaptation of a quality of life measure (CHO-KLAT) for boys with haemophilia in Brazil.

Health-related quality of life (HRQoL) is an important outcome from the perspective of boys with haemophilia and their parents. Few studies have captured the HRQoL of boys with haemophilia in developing countries. This article reports on the cross-cultural adaptation of the Canadian Haemophilia Outcomes - Kids Life Assessment Tool (CHO-KLAT) for use in São Paulo, Brazil. The CHO-KLAT(2.0) was translated into Portuguese, and then translated back into English. The original English and back-translation versions were compared by a group of three clinicians, whose first language was Portuguese. The resulting Portuguese version was assessed through a series of cognitive debriefing interviews with children and their parents. This process identified concepts that were not clear and revised items to ensure appropriate understanding through an iterative process. The initial back-translation was not discrepant from the original English version. We made changes to 66% of the CHO-KLAT(2.0) items based on clinical expert review and 26% of the items based on cognitive debriefings. In addition, two new items were added to the final Portuguese version to reflect the local cultural context. The final result had good face validity. This process was found to be extremely valuable in ensuring the items were accurately interpreted by the boys/parents in São Paulo Brazil. The results suggest that professional translators, clinical experts and cognitive debriefing are all required to achieve a culturally appropriate instrument. The Portuguese CHO-KLAT(2.0) is well understood by Sao Paulo boys/parents. The next step will be to test its validity and reliability locally.

Importance of literacy for self-reported health-related quality of life: a study of boys with haemophilia in Brazil.

Psychosocial outcomes are important in the perspective of boys with haemophilia. However, health-related quality of life (HRQoL) is based on self-report, and assumes adequate literacy. Yet, literacy is rarely assessed prior to data collection. This study sought to identify criteria that might indicate the level of literacy of children being recruited for clinical trials and to develop a simple method to prescreen those whose literacy was uncertain. We developed a brief screening tool in the form of two stories, at a grade 3 reading level, followed by comprehension questions. We applied the screening test to a sample of haemophilic boys between the ages of 7 and 13 years to assess their literacy. The data were analysed to determine the best criteria to use in identifying the ability to independently self-report for HRQoL studies. Twenty-four Brazilian boys (7.9-12.8) completed the testing. The results showed that 17 (70.8%) were literate (were able to both read and comprehend), and could complete a questionnaire without assistance. All boys over 11.0 years of age were sufficiently literate. Grade level was not found to be a helpful criterion. We recommend that all children under the age of 11.0 years be prescreened before providing self-reported HRQoL data. Those with limited literacy should be provided assistance to ensure comprehension of the questions. This is important to ensure high-quality data on HRQoL for future clinical trials.

Why should we care about quality of life in persons with haemophilia?

The very high cost of haemophilia care, including the increase in use of factor prophylaxis in both children and adults requires that funders of clotting factor concentrates require objective measures of health, such as joint status and quality of life (QOL). Many clinical trials, especially those for licensing of new products, are including QOL instruments in their protocols to evaluate the patients' perspective of wellbeing before and during therapy. This article gives a perspective on QOL the importance of QOL measurement in the field of haemophilia and its impact on patient outcome.

Cross-cultural validation of the CHO-KLAT and HAEMO-QoL-A in Canadian French.

Multi-site studies are necessary in the field of haemophilia to ensure adequate sample sizes. Quality of life (QoL) instruments need to be harmonized across languages and cultures to facilitate their inclusion. The purpose of this study was to adapt the Canadian Haemophilia Outcomes - Kids Life Assessment Tool (CHO-KLAT(©)) and HAEMO-QoL-A(©) to French for Canada. The CHO-KLAT and the HAEMO-QoL-A are haemophilia-specific measures of QoL for boys and men respectively. Both measures originated in English, were translated into Canadian French by clinicians with expertise in haemophilia care, back-translated by expert translators and harmonized by a multi-disciplinary team. The harmonized versions were evaluated through a cognitive debriefing process with 6 boys with haemophilia, their parents and 10 men with haemophilia. The final versions were validated in a sample of 19 boys with haemophilia, 19 parents, and 22 men with haemophilia along with a generic QoL scale: the PedsQL for children; and the SF-36 for adults. The translation and cognitive debriefing processes resulted in a preliminary version that maintained the intent of the original questions. The validation study estimated the mean score for the child-reported CHO-KLAT at 71.9 (SD 10.4), and the adult-reported HAEMO-QoL-A at 79.1 (SD 21.3). The CHO-KLAT correlated 0.64 with the PedsQL and the HAEMO-QoL-A correlated 0.78 with the SF-36 physical component summary score. The French-Canadian version of the CHO-KLAT and HAEMO-QoL-A are valid. These measures are available for use in multi-site haemophilia trials and clinical practices to capture QoL data from French Canadians.

Children's and parents' beliefs regarding the value of walking: rehabilitation implications for children with cerebral palsy.

BACKGROUND: Walking for children with cerebral palsy (CP) has physiological and functional benefits, but also holds symbolic significance that largely remains unexplored. The aims of this pilot study were to describe beliefs about the value of walking held by children with CP and their parents, and to examine how these beliefs inform rehabilitation choices and perceptions of 'success'. METHODS: A critical qualitative design was employed. Six parents and six children with CP (Gross Motor Function Classification System III or IV, aged 9 to 18 years) each participated in a private interview. Analyses examined the relationship between dominant social beliefs regarding walking and participants' accounts. RESULTS: Parents' accounts revealed that all adopted a stance of doing something/trying anything as part of being a 'good parent' and maintaining hope. Tapering of walking interventions contributed to feelings of guilt and doubt. Children primarily viewed walking as exercise rather than functional. Their accounts also demonstrated how they internalized negative attitudes towards disability and judged themselves accordingly. CONCLUSIONS: The results of this pilot study provide provisional evidence regarding how dominant social values regarding walking and disability are taken up by parents and children. They suggest that rehabilitation programmes need to consider how they may unintentionally reinforce potentially harmful choices, and how best to engage families in discussions of their evolving values and treatment priorities. Further research is needed with a larger sample.

Measuring patient-reported outcomes in haemophilia clinical research.

Patient-reported outcome (PRO) measures have been used to assess quality of life and health state preferences from the patient's perspective. However, they have not been fully utilized in haemophilia clinical practice and research. A series of meetings were convened to review and document the state of the art in PROs relevant to haemophilia. Experts developed a process for selection of measures and identified published measures of health-related quality of life (HRQoL) relevant to patients with haemophilia. These were synthesized and reviewed. Patient preference measures were also identified and reviewed. Although the majority of measures were developed for and validated in adults, several measures were identified for use in paediatric populations. This paper recommends an approach to the selection of PROs for application in haemophilia clinical research and practice and identifies several potential measures relevant for application in haemophilia clinical research and practice.

Previous radiographic experience of children referred for dental extractions under general anaesthesia in the U.K.

OBJECTIVE: To determine what proportion of children undergo radiographic assessment prior to referral to a dental hospital for extractions under general anaesthesia. BASIC RESEARCH DESIGN: This prospective survey was conducted over a 6-month period. A data sheet was used to record the following information: patient's age; referrer's name and place of work (general dental practice or community dental service); teeth to be extracted (primary dentition and/or permanent dentition) and reported previous radiographic examination. Patients were excluded from the study if, following a clinical examination, radiographs were not actually deemed necessary for diagnosis and treatment planning purposes. Clinical setting A paediatric dentistry clinic within a dental hospital in the North of England. Participants 161 patients with a mean age of six years (SD = 2.2, range = 3-14 years) who were referred to the dental hospital for extractions under general anaesthesia. RESULTS: Overall, 12.4% of children had reportedly undergone a previous radiographic assessment prior to hospital referral. A significantly greater proportion of children referred for permanent tooth extractions had been subject to radiographic examination compared to children referred for primary tooth extractions (46.2% as compared to 6.3%; P = 0.001 chi-squared test). Furthermore, patients referred from the community dental service were significantly more likely to have had previous dental radiographs than children referred from general dental practice (36.9% compared to 9.3%; P = 0.003 chi-squared test). CONCLUSIONS: Radiographs do not appear to be routinely employed for caries diagnosis and treatment planning in young children within general dental practice in the U.K.

Dried blood spots for the diagnosis and quantitation of HIV-1: stability studies and evaluation of sensitivity and specificity for the diagnosis of infant HIV-1 infection in Thailand.

Molecular methods for HIV-1 infection using dried blood-spot (DBS) for HIV-1 CRF01_AE subtypes have not been fully optimized. In this study assays for HIV-1 diagnosis or quantitation were evaluated using infant DBS from Thailand. Paired DBS and whole blood samples from 56 HIV-1 CRF01_AE or B'-infected infants were tested for infant diagnosis using modified Amplicor DNA PCR and NucliSens RNA NASBA and an in-house real-time PCR assay. The Amplicor Monitor viral load (VL) assay, with modifications for DBS, was also evaluated. DBS VL were hematocrit corrected. Stability studies were done on DBS stored at -70 degrees C to 37 degrees C for up to 1 year. The DBS diagnostic assays were 96-100% sensitive and 100% specific for HIV-1 diagnosis. DBS HIV-1 VL were highly correlated with plasma VL when corrected using the actual or an assumed hematocrit factor (r(c)=0.88 or 0.93, respectively). HIV-1 DNA in DBS appeared to be more stable than RNA and could be detected after up to 9 months at most temperatures. DBS VL could be consistently determined when stored frozen. These results show that DBS can be used accurately instead of whole blood for the diagnosis of HIV-1 infection and VL quantitation, particularly if samples are appropriately stored.