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1. Early childhood height-adjusted total kidney volume as a risk marker of kidney survival in ARPKD

Author

Nalcacioglu, Hulya, Dunand, Oliver, Mastrangelo, Antonio, Murer, Luisa, Emma, Francesco, Ruzgiene, Dovile, Taranta-Janusz, Katarzyna, Balasz-Chmielewska, Irena, Miklaszewska, Monika, Stanczyk, Malgorzata, Sikora, Przemyslaw, Kowalewska, Claudia, Szczepanska, Maria, Teixeira, Ana, Rachisan, Andreea, Papachristou, Fotios, Paripović, Dušan, Prikhodina, Larisa, Jilani, Houweyda, Bayazit, Aysun Karabay, Soylu, Alper, Candan, Cengiz, Sever, Lale, Emre, Sevinc, Cicek, Neslihan, Akinci, Nurver, Mir, Sevgi, Poyrazoğlu, Hakan M., Tabel, Yilmaz, Mencarelli, Francesca, Burgmaier, Kathrin, Kilian, Samuel, Arbeiter, Klaus, Atmis, Bahriye, Büscher, Anja, Derichs, Ute, Dursun, Ismail, Duzova, Ali, Eid, Loai Akram, Galiano, Matthias, Gessner, Michaela, Gokce, Ibrahim, Haeffner, Karsten, Hooman, Nakysa, Jankauskiene, Augustina, Körber, Friederike, Longo, Germana, Massella, Laura, Mekahli, Djalila, Miloševski-Lomić, Gordana, Rus, Rina, Shroff, Rukshana, Stabouli, Stella, Weber, Lutz T., Wygoda, Simone, Yilmaz, Alev, Zachwieja, Katarzyna, Zagozdzon, Ilona, Dötsch, Jörg, Schaefer, Franz, Liebau, Max Christoph, Potemkina, Alexandra, Ranguelov, Nadejda, Collard, Laure, De Mul, Aurélie, Feldkoetter, Markus, Seeman, Tomas, Zieg, Jakub, Thumfart, Julia, Grundmann, Franziska, Buchholz, Björn, Pape, Lars, Gross, Oliver, Patzer, Ludwig, Schild, Raphael, Haffner, Dieter, Bernhardt, Wanja, Wuehl, Elke, Henn, Michael, Halbritter, Jan, Klaus, Günter, Lechner, Felix, Lange-Sperandio, Bärbel, Uetz, Barbara, Benz, Marcus, König, Jens, Staude, Hagen, Wurm, Donald, Bald, Martin, Soliman, Neveen A., Ariceta, Gema, Rodriguez, Juan David Gonzalez, de la Cerda Ojeda, Francisco, Harambat, Jerome, Ranchin, Bruno, Fila, Marc, Dossier, Claire, Boyer, Olivia, Marlais, Matko, UCL - (SLuc) Département de pédiatrie, Ruzgienė, Dovilė, Burgmaier, Kathrin, Kilian, Samuel, Arbeiter, Klaus, Atmis, Bahriye, Buescher, Anja, Derichs, Ute, Dursun, Ismail, Duzova, Ali, Eid, Loai Akram, Galiano, Matthias, Gessner, Michaela, Gokce, Ibrahim, Haeffner, Karsten, Hooman, Nakysa, Jankauskiene, Augustina, Koerber, Friederike, Longo, Germana, Massella, Laura, Mekahli, Djalila, Milosevski-Lomic, Gordana, Nalcacioglu, Hulya, Rus, Rina, Shroff, Rukshana, Stabouli, Stella, Weber, Lutz T., Wygoda, Simone, Yilmaz, Alev, Zachwieja, Katarzyna, Zagozdzon, Ilona, Doetsch, Joerg, Schaefer, Franz, and Liebau, Max Christoph

Subjects
Liver Cirrhosis, Male, glomerulus filtration rate, Medizin, cell surface receptor, preschool child, DISEASE, Cohort Studies, Chronic kidney disease, Polycystic kidney disease, Medicine, genetics, Longitudinal Studies, Child, pathophysiology, Ultrasonography, Kidney, Multidisciplinary, longitudinal study, organ size, chronic kidney failure, biological marker, cohort analysis, Prognosis, Autosomal Recessive Polycystic Kidney Disease, PKHD1 protein, human, Multidisciplinary Sciences, medicine.anatomical_structure, female, Quartile, Child, Preschool, Cohort, Disease Progression, Science & Technology - Other Topics, Glomerular Filtration Rate, medicine.medical_specialty, kidney, Adolescent, Science, Urology, Renal function, Kidney Volume, Receptors, Cell Surface, Article, Humans, ddc:610, human, Renal Insufficiency, Chronic, Polycystic Kidney, Autosomal Recessive, Paediatric kidney disease, Science & Technology, business.industry, echography, Infant, medicine.disease, mortality, kidney polycystic disease, disease exacerbation, physiology, business, metabolism, Biomarkers, Kidney disease
Abstract

Autosomal recessive polycystic kidney disease (ARPKD) is characterized by bilateral fibrocystic changes resulting in pronounced kidney enlargement. Impairment of kidney function is highly variable and widely available prognostic markers are urgently needed as a base for clinical decision-making and future clinical trials. In this observational study we analyzed the longitudinal development of sonographic kidney measurements in a cohort of 456 ARPKD patients from the international registry study ARegPKD. We furthermore evaluated correlations of sonomorphometric findings and functional kidney disease with the aim to describe the natural disease course and to identify potential prognostic markers. Kidney pole-to-pole (PTP) length and estimated total kidney volume (eTKV) increase with growth throughout childhood and adolescence despite individual variability. Height-adjusted PTP length decreases over time, but such a trend cannot be seen for height-adjusted eTKV (haeTKV) where we even observed a slight mean linear increase of 4.5 ml/m per year during childhood and adolescence for the overall cohort. Patients with two null PKHD1 variants had larger first documented haeTKV values than children with missense variants (median (IQR) haeTKV 793 (450–1098) ml/m in Null/null, 403 (260–538) ml/m in Null/mis, 230 (169–357) ml/m in Mis/mis). In the overall cohort, estimated glomerular filtration rate decreases with increasing haeTKV (median (IQR) haeTKV 210 (150–267) ml/m in CKD stage 1, 472 (266–880) ml/m in stage 5 without kidney replacement therapy). Strikingly, there is a clear correlation between haeTKV in the first eighteen months of life and kidney survival in childhood and adolescence with ten-year kidney survival rates ranging from 20% in patients of the highest to 94% in the lowest quartile. Early childhood haeTKV may become an easily obtainable prognostic marker of kidney disease in ARPKD, e.g. for the identification of patients for clinical studies. © 2021, The Author(s)., ESPN 2014.2; PKD Foundation, PKDF; Bundesministerium für Bildung und Forschung, BMBF: 01GM1515, 01GM1903; Universität zu Köln, UoC; Marga und Walter Boll-Stiftung; Universitätsklinikum Köln, We thank the German Society for Pediatric Nephrology (GPN), the ESCAPE Network, and the European Society for Paediatric Nephrology (ESPN; Working Groups CAKUT and Inherited Renal Diseases) for their support. ML was supported by grants of the GPN, ESPN (Grant ESPN 2014.2), and the German PKD foundation. KB and ML were supported by the Medical Faculty of the University of Cologne (Koeln Fortune program), and the Marga and Walter Boll-Foundation. FS and ML are supported by the German Federal Ministry of Research and Education (BMBF grant 01GM1515 and 01GM1903). This work was generated within the European Reference Network for Rare Kidney Disorders (ERKNet).

Published
2021

2. Treatment and long-term outcome in primary nephrogenic diabetes insipidus

Author

Lopez-Garcia, Sergio C, Downie, Mallory L, Kim, Ji Soo, Boyer, Olivia, Walsh, Stephen B, Nijenhuis, Tom, Papizh, Svetlana, Yadav, Pallavi, Reynolds, Ben C, Decramer, Stéphane, Besouw, Martine, Perelló Carrascosa, Manel, La Scola, Claudio, Trepiccione, Francesco, Ariceta, Gema, Hummel, Aurélie, Dossier, Claire, Sayer, John A, Konrad, Martin, Keijzer-Veen, Mandy G, Awan, Atif, Basu, Biswanath, Chauveau, Dominique, Madariaga, Leire, Koster-Kamphuis, Linda, Furlano, Mónica, Zacchia, Miriam, Marzuillo, Pierluigi, Tse, Yincent, Dursun, Ismail, Pinarbasi, Ayse Seda, Tramma, Despoina, Hoorn, Ewout J, Gokce, Ibrahim, Nicholls, Kathleen, Eid, Loai A, Sartz, Lisa, Riordan, Michael, Hooman, Nakysa, Printza, Nikoleta, Bonny, Olivier, Arango Sancho, Pedro, Schild, Raphael, Sinha, Rajiv, Guarino, Stefano, Martinez Jimenez, Victor, Rodríguez Peña, Lidia, Belge, Hendrica, Devuyst, Olivier, Wlodkowski, Tanja, Emma, Francesco, Levtchenko, Elena, Knoers, Nine V A M, Bichet, Daniel G, Schaefer, Franz, Kleta, Robert, Wasilewska, Anna, Longo, Germana, Espinosa, Laura, Miglinas, Marius, Stroescu, Ramona, Huseynova, Shafa, Stabouli, Stella, Sathyanarayana, Vijaya, Andronesi, Andreea G, Hahn, Deirdre, Sharma, Deepak, Petrosyan, Edita, Frangou, Eleni, Mohebbi, Nilufar, Dinçel, Nida Temizkan, Braconnier, Philippe, Gilbert, Rodney D, Sambo, Adamu, Tasic, Velibor, Henne, Thomas, Bockenhauer, Detlef, UCL - SSS/IREC/NEFR - Pôle de Néphrologie, Lopez-Garcia, Sergio C, Downie, Mallory L, Kim, Ji Soo, Boyer, Olivia, Walsh, Stephen B, Nijenhuis, Tom, Papizh, Svetlana, Yadav, Pallavi, Reynolds, Ben C, Decramer, Stéphane, Besouw, Martine, Perelló Carrascosa, Manel, La Scola, Claudio, Trepiccione, Francesco, Ariceta, Gema, Hummel, Aurélie, Dossier, Claire, Sayer, John A, Konrad, Martin, Keijzer-Veen, Mandy G, Awan, Atif, Basu, Biswanath, Chauveau, Dominique, Madariaga, Leire, Koster-Kamphuis, Linda, Furlano, Mónica, Zacchia, Miriam, Marzuillo, Pierluigi, Tse, Yincent, Dursun, Ismail, Pinarbasi, Ayse Seda, Tramma, Despoina, Hoorn, Ewout J, Gokce, Ibrahim, Nicholls, Kathleen, Eid, Loai A, Sartz, Lisa, Riordan, Michael, Hooman, Nakysa, Printza, Nikoleta, Bonny, Olivier, Arango Sancho, Pedro, Schild, Raphael, Sinha, Rajiv, Guarino, Stefano, Martinez Jimenez, Victor, Rodríguez Peña, Lidia, Belge, Hendrica, Devuyst, Olivier, Wlodkowski, Tanja, Emma, Francesco, Levtchenko, Elena, Knoers, Nine V A M, Bichet, Daniel G, Schaefer, Franz, Kleta, Robert, and Bockenhauer, Detlef

Subjects
Nephrology, Pediatrics, medicine.medical_specialty, endocrine system diseases, 030232 urology & nephrology, 030204 cardiovascular system & hematology, urologic and male genital diseases, 03 medical and health sciences, 0302 clinical medicine, nephrogenic diabetes insipidus, Internal medicine, medicine, AVPR2, Transplantation, business.industry, AQP2, medicine.disease, Nephrogenic diabetes insipidus, Mental health, Obesity, female genital diseases and pregnancy complications, Cohort, flow uropathy, business, Body mass index, chronic kidney disease, Kidney disease
Abstract

BackgroundPrimary nephrogenic diabetes insipidus (NDI) is a rare disorder and little is known about treatment practices and long-term outcome.MethodsPaediatric and adult nephrologists contacted through European professional organizations entered data in an online form.ResultsData were collected on 315 patients (22 countries, male 84%, adults 35%). Mutation testing had been performed in 270 (86%); pathogenic variants were identified in 258 (96%). The median (range) age at diagnosis was 0.6 (0.0–60) years and at last follow-up 14.0 (0.1–70) years. In adults, height was normal with a mean (standard deviation) score of −0.39 (±1.0), yet there was increased prevalence of obesity (body mass index >30 kg/m2; 41% versus 16% European average; P ConclusionThis large NDI cohort shows an overall favourable outcome with normal adult height and only mild to moderate CKD in most. Yet, while full-time employment was similar to the European average, educational achievement was lower, and more than half had urological and/or mental health problems.

Published
2020

3. Severe neurological outcomes after very early bilateral nephrectomies in patients with autosomal recessive polycystic kidney disease (ARPKD)

Author

Häffner, Karsten, Gross, Oliver, Bernhardt, Wanja, Doyon, Anke, Henn, Michael, Halbritter, Jan, Derichs, Ute, Klaus, Günter, Lange-Sperandio, Bärbel, Uetz, Barbara, Benz, Marcus, Titieni, Andrea, Staude, Hagen, Leichter, Heinz E., Soliman, Neveen A., Lara, Luis Enrique, de la Cerda Ojeda, Francisco, Harambat, Jerome, Ranchin, Bruno, Fila, Marc, Dossier, Claire, Boyer, Olivia, Stabouli, Stella, Hooman, Nakysa, Mencarelli, Francesca, Morello, William, Longo, Germana, Emma, Francesco, Ruzgiene, Dovile, Wasilewska, Anna, Balasz-Chmielewska, Irena, Miklaszewska, Monika, Stanczyk, Malgorzata, Sikora, Przemyslaw, Litwin, Mieczyslaw, Morawiec-Knysak, Aurelia, Teixeira, Ana, Milosevski-Lomic, Gordana, Prikhodina, Larisa, Rus, Rina, Jilani, Houweyda, Melek, Engin, Duzova, Ali, Candan, Cengiz, Sever, Lale, Ranguelov, Nadejda, Yilmaz, Alev, Cicek, Neslihan, Akinci, Nurver, Mir, Sevgi, Dursun, Ismail, Tabel, Yilmaz, Nalcacioglu, Hulya, Shroff, Rukshana, Marlais, Matko, Soylu, Alper, Arbeiter, Klaus, Eid, Loai Akram, Liebau, Max Christoph, Schaefer, Franz, Oh, Jun, Dötsch, Jörg, Zagozdzon, Ilona, Wygoda, Simone, Wurm, Donald, Wühl, Elke, Weber, Lutz Thorsten, Tkaczyk, Marcin, Burgmaier, Kathrin, Ariceta, Gema, Bald, Martin, Buescher, Anja Katrin, Burgmaier, Mathias, Erger, Florian, Gessner, Michaela, GÖKCE, İBRAHİM, König, Jens, Kowalewska, Claudia, Massella, Laura, Mastrangelo, Antonio, Mekahli, Djalila, Pape, Lars, Patzer, Ludwig, Potemkina, Alexandra, Schalk, Gesa, Schild, Raphael, Szczepanska, Maria, Taranta-Janusz, Katarzyna, Collard, Laure, De Mul, Aurélie, Feldkoetter, Markus, Seeman, Tomas, Thumfart, Julia, Grundmann, Franziska, Galiano, Matthias, Buchholz, Björn, Buescher, Rainer, UCL - (SLuc) Service de pédiatrie générale, De Mul, Aurélie, Burgmaier, Kathrin, Ariceta, Gema, Bald, Martin, Buescher, Anja Katrin, Burgmaier, Mathias, Erger, Florian, Gessner, Michaela, Gokce, Ibrahim, Koenig, Jens, Kowalewska, Claudia, Massella, Laura, Mastrangelo, Antonio, Mekahli, Djalila, Pape, Lars, Patzer, Ludwig, Potemkina, Alexandra, Schalk, Gesa, Schild, Raphael, Shroff, Rukshana, Szczepanska, Maria, Taranta-Janusz, Katarzyna, Tkaczyk, Marcin, Weber, Lutz Thorsten, Wuehl, Elke, Wurm, Donald, Wygoda, Simone, Zagozdzon, Ilona, Doetsch, Joerg, Oh, Jun, Schaefer, Franz, Liebau, Max Christoph, and Ege Üniversitesi

Subjects
Male, Pediatrics, medicine.medical_treatment, Medizin, 030232 urology & nephrology, INFANTS, lcsh:Medicine, Nephrectomy, RECOMMENDATIONS, Cohort Studies, 0302 clinical medicine, Postoperative Complications, Risk Factors, HYPOTENSION, Polycystic Kidney, Medicine, UNILATERAL NEPHRECTOMY, ENCODES, lcsh:Science, Renal Dialysis/statistics & numerical data, PERITONEAL-DIALYSIS, Kidney, ddc:618, Multidisciplinary, Autosomal recessive polycystic kidney disease (ARPKD), Autosomal Recessive Polycystic Kidney Disease, medicine.anatomical_structure, Cohort, ARegPKD consortium, Disease Progression, Female, Cohort study, Nephrectomy/adverse effects, medicine.medical_specialty, Renal function, MATURATION, Article, 03 medical and health sciences, Polycystic kidney disease, Renal Dialysis, 030225 pediatrics, MANAGEMENT, Humans, In patient, Dialysis, Polycystic Kidney, Autosomal Recessive, Nervous System Diseases/epidemiology/etiology, Autosomal Recessive/surgery, Paediatric kidney disease, business.industry, lcsh:R, 1ST YEAR, NEUROPATHY, Infant, Newborn, Infant, Newborn, medicine.disease, Postoperative Complications/epidemiology/etiology, lcsh:Q, Nervous System Diseases, business
Abstract

To test the association between bilateral nephrectomies in patients with autosomal recessive polycystic kidney disease (ARPKD) and long-term clinical outcome and to identify risk factors for severe outcomes, a dataset comprising 504 patients from the international registry study ARegPKD was analyzed for characteristics and complications of patients with very early (? 3 months; VEBNE) and early (4–15 months; EBNE) bilateral nephrectomies. Patients with very early dialysis (VED, onset ? 3 months) without bilateral nephrectomies and patients with total kidney volumes (TKV) comparable to VEBNE infants served as additional control groups. We identified 19 children with VEBNE, 9 with EBNE, 12 with VED and 11 in the TKV control group. VEBNE patients suffered more frequently from severe neurological complications in comparison to all control patients. Very early bilateral nephrectomies and documentation of severe hypotensive episodes were independent risk factors for severe neurological complications. Bilateral nephrectomies within the first 3 months of life are associated with a risk of severe neurological complications later in life. Our data support a very cautious indication of very early bilateral nephrectomies in ARPKD, especially in patients with residual kidney function, and emphasize the importance of avoiding severe hypotensive episodes in this at-risk cohort. © 2020, The Author(s)., Marga und Walter Boll-Stiftung Universität zu Köln, UoC Bundesministerium für Bildung und Forschung, BMBF: 01GM1903, 01GM1515 PKD Foundation, PKDF European Paediatric Neurology Society, EPNS, We thank the German Society for Pediatric Nephrology (GPN) and the ESCAPE Network for their support. ML was supported by grants of the GPN, the European Society for Paediatric Nephrology (ESPN), the German PKD foundation, the Koeln Fortune program, the GEROK program of the Medical Faculty of University of Cologne, and the Marga and Walter Boll-Foundation. FS and ML are supported by the the German Federal Ministry of Research and Education (BMBF grant 01GM1515 and 01GM1903). KB was supported by the Koeln Fortune program of the Medical Faculty of University of Cologne and the Marga and Walter Boll-Foundation. This work was generated within the European Reference Network for Rare Kidney Disorders (ERKNet). Open Access funding provided by Projekt DEAL.

Published
2020

4. Global Variation of Nutritional Status in Children Undergoing Chronic Peritoneal Dialysis: A Longitudinal Study of the International Pediatric Peritoneal Dialysis Network

Author

Schaefer, Franz, Benner, Laura, Borzych-Duzalka, Dagmara, Zaritsky, Joshua, Xu, Hong, Rees, Lesley, Antonio, Zenaida L., Serdaroglu, Erkin, Hooman, Nakysa, Patel, Hiren, Sever, Lale, Vondrak, Karel, Flynn, Joseph, Rebori, Anabella, Wong, William, Holtta, Tuula, Yildirim, Zeynep Yuruk, Ranchin, Bruno, Grenda, Ryszard, Testa, Sara, Drozdz, Dorota, Szabo, Attila J., Eid, Loai, Basu, Biswanath, Vitkevic, Renate, Wong, Cynthia, Pottoore, Stephen J., Mueller, Dominik, Dusunsel, Ruhan, Celedon, Claudia Gonzalez, Fila, Marc, Sartz, Lisa, Sander, Anja, Warady, Bradley A., Adragna, M., Coccia, P. A., Suarez, A., Valles, P. G., Salim, R., Alconcher, L., Arbeiter, K., van Hoeck, K., Koch, V, Feber, J., Harvey, E., White, C., Valenzuela, M., Villagra, J., Cano, F., Contreras, M. A., Vogel, A., Zambrano, P., Hevia, P., Chiu, M. C., Ding, Jie, Vanegas, J. J., Higuita, L. M., Roussey, G., Ulinski, T., Krid, S., Fischbach, M., Harambat, J., Samaille, Ch, Buescher, R., Oh, J., Pape, L., John, U., Klaus, G., Billing, H., Stafanidis, C., Papachristou, F., Bagga, A., Kanitkar, M., Sinha, R., Sethi, S., Verrinam, E., Vidal, E., Leozappa, G., Landau, D., Paik, K. H., Bilal, A., Sahpazova, E., Lim, Y. N., Barbosa, L. Sanchez, Groothoff, J. W., Konijenberg, Y., Silva, Y., Al Ryami, M., Munarriz, R. Loza, Leszepanska, B., Szczepanska, M., Brumariu, O., Kari, J., Kruscic, D., Yap, H. K., Ariceta, G., Aguirre, M., Santos, F., Niwhiska-Faryna, B., Bayazit, A., Bakkaloglu, C. A. S., Bakkaloglu, S., Bilge, I, Yavascan, O., Mir, S., Simkova, Eva, Christian, M., Greenbaum, L., Neu, A., Askenazi, D., Al-Akash, A., Swartz, S., Brophy, P., Rheault, M., Pradhan, M., Pediatric surgery, Amsterdam Reproduction & Development (AR&D), Çukurova Üniversitesi, Büscher (R.), Rainer (Beitragende*r), Paediatric Nephrology, AGEM - Inborn errors of metabolism, APH - Methodology, APH - Quality of Care, ARD - Amsterdam Reproduction and Development, Ege Üniversitesi, Int Pediat Peritoneal Dial Network, Children's Hospital, Clinicum, and HUS Children and Adolescents

Subjects
Male, 0301 basic medicine, Pediatric Obesity, Longitudinal study, Pediatrics, medicine.medical_treatment, Medizin, lcsh:Medicine, Overweight, DISEASE, 0302 clinical medicine, Risk Factors, Prevalence, Longitudinal Studies, Registries, Child, lcsh:Science, RISK, 2. Zero hunger, OUTCOMES, Multidisciplinary, 3. Good health, Europe, OBESITY, Child, Preschool, GROWTH, Female, medicine.symptom, Underweight, Peritoneal Dialysis, Engineering sciences. Technology, medicine.medical_specialty, Asia, Adolescent, Nutritional Status, Article, Peritoneal dialysis, 03 medical and health sciences, Enteral Nutrition, Thinness, medicine, Humans, Dialysis, business.industry, MORTALITY, lcsh:R, Infant, nutritional and metabolic diseases, medicine.disease, Obesity, BODY-MASS INDEX, 030104 developmental biology, 3121 General medicine, internal medicine and other clinical medicine, Kidney Failure, Chronic, lcsh:Q, Americas, business, Body mass index, 030217 neurology & neurosurgery, Kidney disease
Abstract

WOS: 000461762600003, PubMed ID: 30894599, While children approaching end-stage kidney disease (ESKD) are considered at risk of uremic anorexia and underweight they are also exposed to the global obesity epidemic. We sought to investigate the variation of nutritional status in children undergoing chronic peritoneal dialysis (CPD) around the globe. The distribution and course of body mass index (BMI) standard deviation score over time was examined prospectively in 1001 children and adolescents from 35 countries starting CPD who were followed in the International Pediatric PD Network (IPPN) Registry. The overall prevalence of underweight, and overweight/obesity at start of CPD was 8.9% and 19.7%, respectively. Underweight was most prevalent in South and Southeast Asia (20%), Central Europe (16.7%) and Turkey (15.2%), whereas overweight and obesity were most common in the Middle East (40%) and the US (33%). BMI SDS at PD initiation was associated positively with current eGFR and gastrostomy feeding prior to PD start. Over the course of PD BMI SDS tended to increase on CPD in underweight and normal weight children, whereas it decreased in initially overweight patients. In infancy, mortality risk was amplified by obesity, whereas in older children mortality was markedly increased in association with underweight. Both underweight and overweight are prevalent in pediatric ESKD, with the prevalence varying across the globe. Late dialysis start is associated with underweight, while enteral feeding can lead to obesity. Nutritional abnormalities tend to attenuate with time on dialysis. Mortality risk appears increased with obesity in infants and with underweight in older children., International Society for Peritoneal Dialysis; Baxter Health Care; Fresenius Medical Care, The authors gratefully acknowledge the support by the International Society for Peritoneal Dialysis, Baxter Health Care, and Fresenius Medical Care. We also appreciate the continued dedicated support of the IPPN by the medical and nursing staff in all collaborating centers.

Published
2019

5. Risk Factors for Early Dialysis Dependency in Autosomal Recessive Polycystic Kidney Disease

Author

Kathrin Burgmaier, Kevin Kunzmann, Gema Ariceta, Carsten Bergmann, Anja Katrin Buescher, Mathias Burgmaier, Ismail Dursun, Ali Duzova, Loai Eid, Florian Erger, Markus Feldkoetter, Matthias Galiano, Michaela Geßner, Heike Goebel, Ibrahim Gokce, Dieter Haffner, Nakysa Hooman, Bernd Hoppe, Augustina Jankauskiene, Guenter Klaus, Jens König, Mieczyslaw Litwin, Laura Massella, Djalila Mekahli, Engin Melek, Sevgi Mir, Lars Pape, Larisa Prikhodina, Bruno Ranchin, Raphael Schild, Tomas Seeman, Lale Sever, Rukshana Shroff, Neveen A. Soliman, Stella Stabouli, Malgorzata Stanczyk, Yilmaz Tabel, Katarzyna Taranta-Janusz, Sara Testa, Julia Thumfart, Rezan Topaloglu, Lutz Thorsten Weber, Dorota Wicher, Elke Wühl, Simone Wygoda, Alev Yilmaz, Katarzyna Zachwieja, Ilona Zagozdzon, Klaus Zerres, Jörg Dötsch, Franz Schaefer, Max Christoph Liebau, Nadejda Ranguelov, Nathalie Godefroid, Laure Collard, Jacques Lombet, Julie Maquet, Gesa Schalk, Uwe Querfeld, Bodo B. Beck, Thomas Benzing, Reinhard Buettner, Franziska Grundmann, Christine Kurschat, Kerstin Benz, Anja Tzschoppe, Björn Buchholz, Rainer Buescher, Karsten Häffner, Martin Pohl, Oliver Gross, Jenny Krügel, Johanna Stock, Ludwig Patzer, Jun Oh, Wanja Bernhardt, Anke Doyon, Tobias Vinke, Anja Sander, Michael Henn, Ute Derichs, Rolf Beetz, Nikola Jeck, Bärbel Lange-Sperandio, Sabine Ponsel, Franziska Kusser, Barbara Uetz, Marcus Benz, Silke Schmidt, Christina Huppertz-Kessler, Birgitta Kranz, Andrea Titieni, Donald Wurm, Heinz E. Leichter, Martin Bald, Heiko Billing, Marwa M. Nabhan, Luis Enrique Lara, Fotios Papachristou, Francesco Emma, Rimante Cerkauskiene, Karolis Azukaitis, Anna Wasilewska, Irena Balasz-Chmielewska, Monika Miklaszewska, Marcin Tkaczyk, Przemyslaw Sikora, Marcin Zaniew, Ania Niemirska, Jolanta Antoniewicz, Justyna Lesiak, Alberto Caldas Afonso, Ana Teixeira, Gordana Milosevski-Lomic, Dusan Paripović, Amira Peco-Antic, Svetlana Papizh, Aysun Karabay Bayazit, Ali Anarat, Alper Soylu, Salih Kavukcu, Cengiz Candan, Salim Caliskan, Nur Canpolat, Sevinc Emre, Harika Alpay, Nurver Akinci, Secil Conkar, Hakan M. Poyrazoglu, Ruhan Dusunsel, Çukurova Üniversitesi, Burgmaier, Kathrin, Kunzmann, Kevin, Ariceta, Gema, Bergmann, Carsten, Buescher, Anja Katrin, Burgmaier, Mathias, Dursun, Ismail, Duzova, Ali, Eid, Loai, Erger, Florian, Feldkoetter, Markus, Galiano, Matthias, Gessner, Michaela, Goebel, Heike, Gokce, Ibrahim, Haffner, Dieter, Hooman, Nakysa, Hoppe, Bernd, Jankauskiene, Augustina, Klaus, Guenter, Koenig, Jens, Litwin, Mieczyslaw, Massella, Laura, Mekahli, Djalila, Melek, Engin, Mir, Sevgi, Pape, Lars, Prikhodina, Larisa, Ranchin, Bruno, Schild, Raphael, Seeman, Tomas, Sever, Late, Shroff, Rukshana, Soliman, Neveen A., Stabouli, Stella, Stanczyk, Malgorzata, Tabel, Yilmaz, Taranta-Janusz, Katarzyna, Testa, Sara, Thumfart, Julia, Topaloglu, Rezan, Weber, Lutz Thorsten, Wicher, Dorota, Wuehl, Elke, Wygoda, Simone, Yilmaz, Alev, Zachwieja, Katarzyna, Zagozdzon, Ilona, Zerres, Klaus, Doetsch, Joerg, Schaefer, Franz, and Liebau, Max Christoph

Subjects
Male, 0301 basic medicine, Time Factors, medicine.medical_treatment, ARPKD, Medizin, 030232 urology & nephrology, PROTEIN, Oligohydramnios, Pediatrics, PKHD1 MUTATIONS, 0302 clinical medicine, Pregnancy, Risk Factors, Prospective Studies, ENCODES, GENOTYPE-PHENOTYPE CORRELATIONS, Obstetrics, Hazard ratio, Autosomal Recessive Polycystic Kidney Disease, CLINICAL-EXPERIENCE, Female, Apgar score, Life Sciences & Biomedicine, renal replacement therapy, medicine.medical_specialty, GENETICS, PKHD1, Risk Assessment, Ultrasonography, Prenatal, 03 medical and health sciences, Renal Dialysis, medicine, Humans, Renal replacement therapy, Dialysis, Polycystic Kidney, Autosomal Recessive, Retrospective Studies, Science & Technology, business.industry, Infant, Newborn, Infant, Retrospective cohort study, medicine.disease, 030104 developmental biology, ciliopathy, Pediatrics, Perinatology and Child Health, business, oligohydramnios, Follow-Up Studies
Abstract

OBJECTIVE: To identify prenatal, perinatal, and postnatal risk factors for dialysis within the first year of life in children with autosomal recessive polycystic kidney disease (ARPKD) as a basis for parental counseling after prenatal and perinatal diagnosis. STUDY DESIGN: A dataset comprising 385 patients from the ARegPKD international registry study was analyzed for potential risk markers for dialysis during the first year of life. RESULTS: Thirty-six out of 385 children (9.4%) commenced dialysis in the first year of life. According to multivariable Cox regression analysis, the presence of oligohydramnios or anhydramnios, prenatal kidney enlargement, a low Apgar score, and the need for postnatal breathing support were independently associated with an increased hazard ratio for requiring dialysis within the first year of life. The increased risk associated with Apgar score and perinatal assisted breathing was time-dependent and vanished after 5 and 8 months of life, respectively. The predicted probabilities for early dialysis varied from 1.5% (95% CI, 0.5%-4.1%) for patients with ARPKD with no prenatal sonographic abnormalities to 32.3% (95% CI, 22.2%-44.5%) in cases of documented oligohydramnios or anhydramnios, renal cysts, and enlarged kidneys. CONCLUSIONS: This study, which identified risk factors associated with onset of dialysis in ARPKD in the first year of life, may be helpful in prenatal parental counseling in cases of suspected ARPKD. ispartof: JOURNAL OF PEDIATRICS vol:199 pages:22-+ ispartof: location:United States status: published

Published
2018

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