We describe a PCR-confirmed case of Plasmodium knowlesi infection with a high parasitemia level and clinical signs of severe malaria in a migrant worker from Malaysian Borneo in the Netherlands. Investigations showed that commercially available rapid antigen tests for detection of human Plasmodium infections can detect P. knowlesi infections in humans.

BackgroundMucopolysaccharidosis type II (MPSII) patients frequently suffer from dyspnoea caused by restrictive airway disease due to skeletal abnormalities as well as glycosaminoglycans (GAG) accumulation at different levels of the airway, including the trachea. In this study we describe the extent of the tracheal and bronchial narrowing, the changes in airway diameter during respiration and the effects of these obstructions on respiratory function in adult MPSII patients.MethodsFive adult MPSII patients (mean age 40 years) were included. Pulmonary function tests and in- and expiratory chest CT scans were obtained. Cross-sectional areas of trachea and main bronchi were measured at end-inspiration and -expiration and percentage collapse was calculated.ResultsThere was diffuse narrowing of the entire intra-thoracic trachea and main bronchi and severe expiratory collapse of the trachea in all patients. At 1 cm above the aortic arch the median % collapse of the trachea was 68 (range 60 to 77 %), at the level of the aortic arch 64 (range 21–93 %), for the main bronchi this was 58 (range 26–66 %) on the left and 44 (range 9–76 %) on the right side. The pulmonary function tests showed that this airway collapse results in obstructive airway disease in all patients, which was severe (forced expiratory volume <50 % of predicted) in four out of five patients.ConclusionIn adult MPS II patients, central airways diameters are strikingly reduced and upon expiration there is extensive collapse of the trachea and main bronchi. This central airways obstruction explains the severe respiratory symptoms in MPSII patients.Electronic supplementary materialThe online version of this article (doi:10.1186/s13023-016-0425-z) contains supplementary material, which is available to authorized users.

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activity, using an intervention of known efficacy (Infliximab) as external standard. Methods Until now, 12 RA patients (ACR criteria) have been included (1 male), having high or moderate disease activity (DAS28 >3.2); 3 10 patients already had a 6-week follow-up. The intervention consisted of treatment with Infliximab and Methotrexate, which is likely to induce a relatively large improvement after 6 weeks. The RADAI was self-administered 1 week before the first infusion with Infliximab (T0). At the first infusion (T1), the patient filled in the RADAI again and the physician assessed the DAS28.After 6 weeks (T2), the RADAI and the DAS28 were again assessed. For analysis of reliability (T0 and T1) the ICC3,1 and the Limits-of Agreement [4] were used. Responsiveness was studied by judging the change in RADAI at T2 in relation to the Limits-of-Agreement and the DAS28 response criteria. 3 Results The RADAI scores at T0 and T1 were mean (sd) 5.0 (1.7) and 4.8 (1.9). The mean (sd) difference was -0.2 (0.8), (paired t-test, p = 0.41). The ICC3,1 was 0.89 (p < 0.05). The Upper Limit-of-Agreement was at 1.6 and the Lower at -2.0. According to the DAS28 response criteria, 3 patients were classified as having no response at T2, 3 had a moderate and 4 a good response. The change in RADAI did not exceed the Lower Limit-of-Agreement for 4 of the 7 responders. The responders had a median (range) change in RADAI of -2.1 (-0.9 to -8.0), signed rank test, p = 0.02). Conclusion The data collection is ongoing till N = 20. The first results on reliability and responsiveness are promising. However, if the Limits-of-Agreement turn out to be much wider than -1 and +1, the reliability of the RADAI should be improved.

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