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Background We developed a paediatric haemodialysis trigger tool (pHTT) for application per haemodialysis (HD) session in children receiving intermittent in-centre HD and systematically monitored adverse events. Methods Single-centre quality improvement study performed over two 8-week cycles. Data collected prospectively using a 'per-dialysis session' pHTT tool including 54 triggers across six domains, adapted from a recently described haemodialysis trigger tool (HTT) for adults. Each trigger was evaluated for level of harm following assessment by two authors. Following a period of training, HD nurses completed the HTT at the end of each dialysis session. Results There were 241 triggers over 182 dialysis sessions, with 139 triggers in 91 HD sessions for 15 children, age range 28-205 months, over an 8-week period (first cycle) and 102 triggers in 91 HD sessions for 13 children, age range 28-205 months, over a further 8-week period (second cycle). After interventions informed by the pHTT, the harm rate per session was significantly reduced from 1.03 (94/91) to 0.32 (29/91), P < 0.001. There was a significant difference between the distribution of triggers by harm category (P < 0.001) and between the proportion of triggers across the various domains of the pHTT (P = 0.004) between the two cycles. No triggers were evaluated as causing permanent harm. Conclusions This pilot study demonstrates potential benefits of a bedside tool to monitor adverse events during haemodialysis in children. Thus, following interventions informed by the pHTT, the harm rate per session was significantly reduced. Under standard patient safety systems, the vast majority of triggers identified by the pHTT would remain unreported and perhaps lead to missed opportunities to improve patient safety. We propose the use of a paediatric HTT as part of standard care by centres providing HD to children in the future. Graphical Abstract A higher resolution version of the Graphical abstract is available as Supplementary information, Author(s): Ramnath Balasubramanian [sup.1] , Rachel Folwell [sup.1] , Arran Wheatley [sup.1] , Heidi Ramsey [sup.1] , Carmen Barton [sup.1] , Christopher J. D. Reid [sup.1] , Manish D. Sinha [...]

Background Valvar abnormalities in children and adults with autosomal dominant polycystic kidney disease (ADPKD) have previously been reported as a frequent occurrence. Mitral valve prolapse (MVP), in particular, has been reported in almost one-third of adult patients and nearly 12% of children with ADPKD. Our objective in this study was to establish the prevalence of valvar abnormalities in a large, contemporary series of children and young people (CYP) with ADPKD. Methods A retrospective, single centre, cross-sectional analysis of the echocardiograms performed on all consecutive children seen in a dedicated paediatric ADPKD clinic. Full anatomical and functional echocardiograms were performed and analysed for valvar abnormalities. Results The echocardiograms of 102 CYP with ADPKD (range 0.25-18 years, mean age 10.3 years, SD ± 5.3 years) were analysed. One (0.98%), 3-year-old boy, had MVP. There was no associated mitral regurgitation. Evaluating variations in normal valvar anatomy, 9 (8.8%) patients, aged 7.1 to 18 years, had minor bowing ± visual elongation of either the anterior or posterior leaflet of the mitral valve, none of which fell within the criteria of true MVP. Three (1.9%) patients, 2 boys and 1 girl aged between 7 and 14 years, had trivial or mild aortic regurgitation. No patients had echocardiographic evidence of tricuspid valve prolapse (TVP). Conclusion In this contemporary cohort of CYP with ADPKD, the incidence of MVP and other valvar lesions is significantly lower than previously reported. Graphical abstract A higher resolution version of the Graphical abstract is available as Supplementary information., Author(s): Alexandra Savis [sup.1] , John M. Simpson [sup.1] , Saleha Kabir [sup.1] , Kelly Peacock [sup.1] , Hayley Beardsley [sup.1] , Manish D. Sinha [sup.2] [sup.3] Author Affiliations: (1) [...]

Background This study aimed to investigate the association of acute kidney injury (AKI) with change in estimated glomerular filtration rate (eGFR) in children with advanced chronic kidney disease (CKD). Methods Single centre, retrospective longitudinal study including all prevalent children aged 1-18 years with nondialysis CKD stages 3-5. Variables associated with CKD were analysed for their potential effect on annualised eGFR change ([DELTA]GFR/year) following multiple regression analysis. Composite end-point including 25% reduction in eGFR or progression to kidney replacement therapy was evaluated. Results Of 147 children, 116 had at least 1-year follow-up in a dedicated CKD clinic with mean age 7.3 ± 4.9 years with 91 (78.4%) and 77 (66.4%) with 2- and 3-year follow-up respectively. Mean eGFR at baseline was 29.8 ± 11.9 ml/min/1.73 m.sup.2 with 79 (68%) boys and 82 (71%) with congenital abnormalities of kidneys and urinary tract (CAKUT). Thirty-nine (33.6%) had at least one episode of AKI. Mean [DELTA]GFR/year for all patients was - 1.08 ± 5.64 ml/min/1.73 m.sup.2 but reduced significantly from 2.03 ± 5.82 to - 3.99 ± 5.78 ml/min/1.73 m.sup.2 from youngest to oldest age tertiles (P < 0.001). There was a significant difference in primary kidney disease (PKD) (77% versus 59%, with CAKUT, P = 0.048) but no difference in AKI incidence (37% versus 31%, P = 0.85) between age tertiles. Multiple regression analysis identified age ([beta] = - 0.53, P < 0.001) and AKI ([beta] = - 3.2, P = 0.001) as independent predictors of [DELTA]GFR/year. 48.7% versus 22.1% with and without AKI reached composite end-point (P = 0.01). Conclusions We report AKI in established CKD as a predictor of accelerated kidney disease progression and highlight this as an additional modifiable risk factor to reduce progression of kidney dysfunction. Graphical abstract, Author(s): Nabil Melhem [sup.1] , Pernille Rasmussen [sup.1] , Triona Joyce [sup.1] , Joanna Clothier [sup.1] , Christopher J. D. Reid [sup.1] , Caroline Booth [sup.1] , Manish D. Sinha [...]

Background Catheter-associated right atrial thrombus (CRAT) is a recognised complication of central venous catheter (CVC) use for haemodialysis (HD) patients. Methods This was a single-centre retrospective longitudinal observational study of consecutive children aged 6 months-18 years over a 7-year period receiving in-centre chronic HD. Echocardiograms as per routine cardiac surveillance were performed 6 months or earlier given clinical concerns. Results Sixty-five children, 36 boys (55.4%), median (IQR) age 11.8 (5.3, 14.7) years, received HD for kidney failure with replacement therapy (KFRT). Initial modality was HD in 45 (69.2%), with CVC as initial access in 42 (93.3%) and AVF in 3 (6.7%); in the remaining 20 (30.8%) patients PD was the initial modality before switching to HD. Seven of 65 (10.8%) developed CRAT at median 2 (0.8, 8.4) months from CVC insertion, with one CRAT detected 3 days following insertion. One child had 2 episodes of CRAT and one additionally thrombosed their AVF. No patient had an underlying primary kidney disease associated with a pro-thrombotic state. Those with CRAT were younger, had more frequent CVC change and received dialysis for longer duration compared to those with no CRAT. Six episodes of CRAT (75%) received anticoagulation therapy. Infective complications were observed in 25% and catheter malfunction in 50%. Five CRAT episodes (62.5%) resulted in CVC loss. One patient died after a haemorrhagic complication of anticoagulation and sepsis, and another developed life-threatening superior vena cava obstruction syndrome. Overall mortality 14% (1/7). Conclusions This is the first report of CRAT in a paediatric HD population. There was ~ 11% incidence of CRAT in patients receiving chronic HD detected by surveillance echocardiography. Although frequently asymptomatic, CRAT is associated with serious sequelae. Anticoagulation and surveillance with expert echocardiography remain mainstays of management. Graphical abstract, Author(s): Martin Garcia-Nicoletti [sup.1] , Manish D. Sinha [sup.1] [sup.2] , Alexandra Savis [sup.3] , Shazia Adalat [sup.1] , Narayan Karunanithy [sup.4] [sup.5] , Francis Calder [sup.1] [sup.6] Author Affiliations: [...]

Background There are limited data regarding vitamin and trace element blood concentrations and supplementation needs in children with non-dialysis stages 3-5 of chronic kidney disease (CKD). Methods Retrospective cross-sectional review for nutritional blood concentrations measured over a recent 2-year period. In our CKD clinics, nutritional bloods including copper, zinc, selenium and vitamin A, vitamin E, active vitamin B.sub.12 and folate are monitored annually. Vitamin D status is monitored every 6-12 months. Results We reviewed 112 children (70 boys) with median (IQ1, IQ3) age 8.97 (4.24, 13.80) years. Estimated median (IQ1, IQ3) GFR (mL/min/1.73 m.sup.2) was 28 (21, 37). Vitamin A, active vitamin B.sub.12 and vitamin E concentrations were within normal range in 19%, 23% and 67% respectively, with all others being above normal range. Vitamin D blood concentrations were within desired range for 85% (15% had low levels) and folate blood concentrations were within normal range in 92%, with the remainder above or below target. For trace elements, 60%, 85% and 87% achieved normal ranges for zinc, selenium and copper respectively. Deficiencies were seen for zinc (35%), copper (7%), folate (3%) and selenium (1%), whilst 5%, 6% and 14% had zinc, copper and selenium levels above normal ranges. Conclusions Several vitamin and trace element blood concentrations were outside normal reference ranges. Monitoring vitamin D and zinc blood concentrations is indicated due to the percentages with low levels in this group. Targeted vitamin and trace element supplementation should be considered where indicated rather than commencing multivitamin and/or mineral supplementation., Author(s): Triona Joyce [sup.1] , Pernille Rasmussen [sup.2] , Nabil Melhem [sup.2] , Joanna Clothier [sup.2] , Caroline Booth [sup.2] , Manish D Sinha [sup.2] [sup.3] Author Affiliations: (1) grid.420545.2, [...]

Background There is growing recognition of hypertension in a significant proportion of children with ADPKD. In this study, we assessed blood pressure and cardiovascular status in children with ADPKD. Methods A prospective two-centre observational study of children (< 18 years) with ADPKD was compared against age- and BMI-matched healthy controls. Children underwent peripheral BP (pBP) measured using an aneroid sphygmomanometer and auscultation, 24-h ambulatory BP monitoring (ABPM), non-invasive central BP (cBP) measurement, carotid-femoral pulse wave velocity (PWVcf) measured using applanation tonometry and measurement of indexed left ventricular mass (LVMI) using echocardiography. This study received independent ethical approval. Results Forty-seven children with ADPKD and 49 healthy controls were recruited (median age 11 years vs. 12 years). Children with ADPKD had significantly higher systolic pBP (mean 112 ± 13.5 mmHg vs. 104 ± 11 mmHg, p < 0.001), higher systolic cBP (mean 97 ± 12.8 mmHg vs. 87 ± 9.8 mmHg, p < 0.001) and lower pulse pressure amplification ratio (1.59 ± 0.2 vs. 1.67 ± 0.1, p = 0.04) compared to healthy children. Thirty-five percent of children with ADPKD showed a lack of appropriate nocturnal dipping on 24-h ABPM. There was no difference in PWVcf between children with ADPKD and healthy children (mean 5.74 ± 1 m/s vs. 5.57 ± 0.9 m/s, p = 0.46). Those with ADPKD had a significantly higher LVMI (mean 30.4 ± 6.6 g/m.sup.2.7 vs. 26.2 ± 6.2 g/m.sup.2.7, p = 0.01). Conclusions These data highlight the high prevalence of hypertension in children with ADPKD, also demonstrating early cardiovascular dysfunction with increased LVMI and reduced PP amplification despite preserved PWVcf, when compared with healthy peers. These early cardiovascular abnormalities are likely to be amenable to antihypertensive therapy, reinforcing the need for routine screening of children with ADPKD., Author(s): Matko Marlais [sup.1] [sup.2] , Sreedevi Rajalingam [sup.2] , Haotian Gu [sup.2] , Alexandra Savis [sup.3] , Manish D Sinha [sup.2] [sup.4] , Paul JD Winyard [sup.1] Author Affiliations: [...]

Objectives We investigated vitamin B6 blood concentrations in children on long-term dialysis at our centre. Methods Retrospective cross-sectional review of vitamin B6 blood concentrations in children on maintenance dialysis [peritoneal dialysis (PD), intermittent haemodialysis (IHD)]. Results We reviewed 28 children (16 boys), 15 IHD and 13 PD with median (interquartile range, IQR) age of 9.4 (2.4, 14.3) years. The median (IQR) vitamin B6 concentration was 223.4 (74.2, 392.8) nmol/L measured a median (IQR) of 9 (4, 16.5) months following commencement of dialysis. None of the children had vitamin B6 deficiency. Vitamin B6 concentrations were raised in 17 (61%), eight of these received a supplement. Nineteen (68%) received vitamin B6 and/or a supplement containing vitamin B6 whilst 11 (39%) received an enteral feed and a supplement. In those with normal vitamin B6 concentrations who were not receiving an enteral feed or an oral nutritional supplement (n = 6), all achieved normal concentrations without need for vitamin B6 supplementation. There were no differences between those on PD versus IHD (269.2 nmol/L vs. 130 nmol/L, P = 0.65). Conclusions We report no children with vitamin B6 deficiency although > 50% had elevated vitamin B6 concentrations. We suggest if dietary assessment of vitamin B6 intake indicates insufficient intake, measurement of blood concentrations will help confirm if supplementation is required. Routine vitamin B6 supplementation and monitoring is currently not indicated in children on chronic dialysis., Author(s): Triona Joyce [sup.1] , Frances Court Brown [sup.1] , Shazia Adalat [sup.2] , Christopher J. D. Reid [sup.2] , Manish D. Sinha [sup.2] [sup.3] Author Affiliations: (Aff1) grid.420545.2, Department [...]

Background There are few recent data regarding blood micronutrient concentrations and supplementation in children on maintenance dialysis. We investigated micronutrient concentrations following dialysis commencement. Methods Retrospective review, including all children on maintenance dialysis (peritoneal dialysis, PD; intermittent haemodialysis, IHD), for nutritional blood concentrations measured over the first 12 months. Patients received pyridoxine and Dialyvit® daily with planned 3-monthly micronutrient concentration monitoring including selenium, manganese, copper, zinc, folate and vitamins A, D, B12 and E. Results We reviewed 47 children (24 girls) including 19 PD and 28 IHD, median age (IQR) 11.4 (2.8,14.4) years. 33 were white, 5 Asian, 5 black and 4 of other ethnic origins. Vitamin A, B12 and E concentrations were within range in 6%, 20% and 13% respectively, with all others above normal range. Serum folate and vitamin D concentrations were within the desired range of 55%, with the rest above or below target. For trace elements, 37%, 60%, 65% and 89% achieved normal ranges for zinc, manganese, copper and selenium respectively. Deficiencies were seen for zinc (43%), copper (28%), folate (6%) and selenium (4%), whereas 7%, 7%, 20% and 40% had copper, selenium, zinc and manganese levels above normal ranges. Despite standard pyridoxine supplementation, only 6 children were monitored during the study period. Conclusions Concentrations of several trace elements and vitamins were outside reference ranges. Response to systematic monitoring and targeted supplementation should be evaluated in future studies. Paediatric dialysis centres should consider undertaking routine nutritional bloods monitoring, particularly for vitamin D, zinc and copper., Author(s): Triona Joyce [sup.1] , Frances Court Brown [sup.1] , Dean Wallace [sup.2] , Christopher J. D. Reid [sup.2] , Manish D. Sinha [sup.2] Author Affiliations: (1) Department of Nutrition [...]