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1. Morphological changes and their associations with clinical parameters in children with nephropathic cystinosis and chronic kidney disease prior to kidney replacement therapy over 25 years

Author

Brügelmann, Malina, Müller, Sophia, Bohlen, Alina V., Hohenfellner, Katharina, Büscher, Anja, Kemper, Markus J., Fröde, Kerstin, Kanzelmeyer, Nele, Oh, Jun, Billing, Heiko, Gellermann, Jutta, Müller, Dominik, Weber, Lutz T., Acham-Roschitz, Birgit, Arbeiter, Klaus, Tönshoff, Burkhard, Hagenberg, Martina, Žebec, Mislav S., Haffner, Dieter, and Zivicnjak, Miroslav

Published
2024
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2. Chest configuration in children and adolescents with infantile nephropathic cystinosis compared with other chronic kidney disease entities and its clinical determinants

Author

Müller, Sophia, Kluck, Rika, Jagodzinski, Celina, Brügelmann, Malina, Hohenfellner, Katharina, Büscher, Anja, Kemper, Markus J., Fröde, Kerstin, Oh, Jun, Billing, Heiko, Thumfart, Julia, Weber, Lutz T., Acham-Roschitz, Birgit, Arbeiter, Klaus, Tönshoff, Burkhard, Hagenberg, Martina, Pavičić, Leo, Haffner, Dieter, and Zivicnjak, Miroslav

Published
2023
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3. Heterozygous variants in the DVL2 interaction region of DACT1 cause CAKUT and features of Townes–Brocks syndrome 2

Author

Christians, Anne, Kesdiren, Esra, Hennies, Imke, Hofmann, Alejandro, Trowe, Mark-Oliver, Brand, Frank, Martens, Helge, Gjerstad, Ann Christin, Gucev, Zoran, Zirngibl, Matthias, Geffers, Robert, Seeman, Tomáš, Billing, Heiko, Bjerre, Anna, Tasic, Velibor, Kispert, Andreas, Ure, Benno, Haffner, Dieter, Dingemann, Jens, and Weber, Ruthild G.

Published
2023
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5. Seasonal variation of blood pressure in children

Author

Ziegelasch, Niels, Vogel, Mandy, Siekmeyer, Werner, Billing, Heiko, Dähnert, Ingo, and Kiess, Wieland

Subjects
Seasonal variations (Diseases) -- Research, Pediatric research, Hypertension -- Diagnosis -- Care and treatment -- Environmental aspects, Health
Abstract

Seasonal blood pressure (BP) variation is mostly found between the summer and winter months. Guidelines for diagnosis and treatment of hypertension in children have not considered this variation until recently. This review aims to present an overview of seasonal BP variation in childhood along with potential underlying pathophysiological mechanisms and long-term implications as well as conclusions for future studies. In pediatric cohorts, seven studies investigated seasonal changes in BP. These changes amount to 3.4-5.9 mmHg (or 0.5-1.5 mmHg per - 1 °C difference in environmental temperature) in systolic BP with a peak in fall or winter. Potential mechanisms and mediators of seasonal BP variation include sympathetic activation of the nervous system with an increase of urinary and plasma norepinephrine levels in the winter season. Additionally, the physical activity among children and adolescents was inversely correlated with BP levels. Temperature sensitivity of BP and pediatric BP levels predict future systolic BP and target-organ damage. Therefore, cardiovascular events may even be long-term complications of seasonal BP variation in pediatric hypertensive patients. Overall, these data strongly suggest an important effect of ambient temperature on BP in children. Additional studies in pediatric cohorts are needed to define how best to incorporate such variation into clinical practice., Author(s): Niels Ziegelasch [sup.1] , Mandy Vogel [sup.1] [sup.2] , Werner Siekmeyer [sup.1] , Heiko Billing [sup.1] , Ingo Dähnert [sup.3] , Wieland Kiess [sup.1] [sup.2] [sup.4] Author Affiliations: (1) [...]

Published
2021
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6. Different approaches to long-term treatment of aHUS due to MCP mutations: a multicenter analysis

Author

Klämbt, Verena, Gimpel, Charlotte, Bald, Martin, Gerken, Christopher, Billing, Heiko, Loos, Sebastian, and Hansen, Matthias

Subjects
Children -- Diseases, Gene mutations -- Health aspects, Hemolytic-uremic syndrome -- Genetic aspects -- Care and treatment -- Diagnosis, Health
Abstract

Background Atypical hemolytic uremic syndrome (aHUS) is a rare, life-threatening microangiopathy, frequently causing kidney failure. Inhibition of the terminal complement complex with eculizumab is the only licensed treatment but mostly requires long-term administration and risks severe side effects. The underlying genetic cause of aHUS is thought to influence the severity of initial and recurring episodes, with milder courses in patients with mutations in membrane cofactor protein (MCP). Methods Twenty pediatric cases of aHUS due to isolated heterozygous MCP mutations were reported from 12 German pediatric nephrology centers to describe initial presentation, timing of relapses, treatment, and kidney outcome. Results The median age of onset was 4.6 years, with a female to male ratio of 1:3. Without eculizumab maintenance therapy, 50% (9/18) of the patients experienced a first relapse after a median period of 3.8 years. Kaplan-Meier analysis showed a relapse-free survival of 93% at 1 year. Four patients received eculizumab long-term treatment, while 3 patients received short courses. We could not show a benefit from complement blockade therapy on long term kidney function, independent of short-term or long-term treatment. To prevent 1 relapse with eculizumab, the theoretical number-needed-to-treat (NNT) was 15 for the first year and 3 for the first 5 years after initial presentation. Conclusion Our study shows that heterozygous MCP mutations cause aHUS with a risk of first relapse of about 10% per year, resulting in large NNTs for prevention of relapses with eculizumab. More studies are needed to define an optimal treatment schedule for patients with MCP mutations to minimize the risks of the disease and treatment., Author(s): Verena Klämbt [sup.1] , Charlotte Gimpel [sup.2] , Martin Bald [sup.3] , Christopher Gerken [sup.4] , Heiko Billing [sup.5] , Sebastian Loos [sup.6] , Matthias Hansen [sup.7] , Jens [...]

Published
2021
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7. A multicenter, randomized, placebo-controlled, double-blind phase 3 trial with open-arm comparison indicates safety and efficacy of nephroprotective therapy with ramipril in children with Alport’s syndrome

Author

Koziolek, Michael, Bramlage, Carsten Paul, Weber, Frauke, Albrecht-Nock, Tanja, Sonntag, Joseph, Frese, Jenny, Kettwig, Matthias, Hilgers, Reinhard, Hansen, Matthias, Wedekin, Mirja, Meyer, Nicole, Klaiber, Susanne, Gessner, Michaela, Liebau, Max, Vogt-Weigeldt, Anne-Kristin, Jungraithmayr, Therese, Ponsel, Sabine, Jacoby, Ulrike, Konrad, Martin, Kranz, Brigitta, Koenig, Jens, Loechtermann, Lisa, Pohl, Michael, Husain, Ralf, Mueller, Katrin, Thumfart, Julia, Schalk, Gesa, Feldkoetter, Markus, Schmidt, Sabine, Sauerstein, Katja, Muschiol, Evelin, Billing, Heiko, Wilkening, Frauke, Gross, Oliver, Tönshoff, Burkhard, Weber, Lutz T., Pape, Lars, Latta, Kay, Fehrenbach, Henry, Lange-Sperandio, Baerbel, Zappel, Hildegard, Hoyer, Peter, Staude, Hagen, König, Sabine, John, Ulrike, Gellermann, Jutta, Hoppe, Bernd, Galiano, Matthias, Hoecker, Britta, Ehren, Rasmus, Lerch, Christian, Kashtan, Clifford E., Harden, Markus, Boeckhaus, Jan, and Friede, Tim

Published
2020
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8. Sex and age as determinants for high blood pressure in pediatric renal transplant recipients: a longitudinal analysis of the CERTAIN Registry

Author

Sugianto, Rizky I., Schmidt, Bernhard M. W., Memaran, Nima, Duzova, Ali, Topaloglu, Rezan, Seeman, Tomas, König, Sabine, Dello Strologo, Luca, Murer, Luisa, Özçakar, Zeynep Birsin, Bald, Martin, Shenoy, Mohan, Buescher, Anja, Hoyer, Peter F., Pohl, Michael, Billing, Heiko, Oh, Jun, Staude, Hagen, Pohl, Martin, Genc, Gurkan, Klaus, Günter, Alparslan, Caner, Grenda, Ryszard, Rubik, Jacek, Krupka, Kai, Tönshoff, Burkhard, Wühl, Elke, and Melk, Anette

Published
2020
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9. Correction to: Endurance-oriented training program with children and adolescents on maintenance hemodialysis to enhance dialysis efficacy—DiaSport

Author

Feldkötter, Markus, Thys, Sarah, Adams, Anne, Becker, Ingrid, Büscher, Rainer, Pohl, Martin, Schild, Raphael, Pape, Lars, Schmitt, Claus Peter, Taylan, Christina, Wygoda, Simone, Klaus, Günter, Fehrenbach, Henry, Montoya, Carmen, Konrad, Martin, Billing, Heiko, Schaar, Bettina, and Hoppe, Bernd

Published
2021
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10. Risk Factors for Early Dialysis Dependency in Autosomal Recessive Polycystic Kidney Disease

Author

Ranguelov, Nadejda, Godefroid, Nathalie, Collard, Laure, Lombet, Jacques, Maquet, Julie, Schalk, Gesa, Querfeld, Uwe, Beck, Bodo B., Benzing, Thomas, Buettner, Reinhard, Grundmann, Franziska, Kurschat, Christine, Benz, Kerstin, Tzschoppe, Anja, Buchholz, Björn, Buescher, Rainer, Häffner, Karsten, Pohl, Martin, Gross, Oliver, Krügel, Jenny, Stock, Johanna, Patzer, Ludwig, Oh, Jun, Bernhardt, Wanja, Doyon, Anke, Vinke, Tobias, Sander, Anja, Henn, Michael, Derichs, Ute, Beetz, Rolf, Jeck, Nikola, Lange-Sperandio, Bärbel, Ponsel, Sabine, Kusser, Franziska, Uetz, Barbara, Benz, Marcus, Schmidt, Silke, Huppertz-Kessler, Christina, Kranz, Birgitta, Titieni, Andrea, Wurm, Donald, Leichter, Heinz E., Bald, Martin, Billing, Heiko, Nabhan, Marwa M., Lara, Luis Enrique, Papachristou, Fotios, Emma, Francesco, Cerkauskiene, Rimante, Azukaitis, Karolis, Wasilewska, Anna, Balasz-Chmielewska, Irena, Miklaszewska, Monika, Tkaczyk, Marcin, Sikora, Przemyslaw, Zaniew, Marcin, Niemirska, Ania, Antoniewicz, Jolanta, Lesiak, Justyna, Afonso, Alberto Caldas, Teixeira, Ana, Milosevski-Lomic, Gordana, Paripović, Dusan, Peco-Antic, Amira, Papizh, Svetlana, Bayazit, Aysun Karabay, Anarat, Ali, Soylu, Alper, Kavukcu, Salih, Candan, Cengiz, Caliskan, Salim, Canpolat, Nur, Emre, Sevinc, Alpay, Harika, Akinci, Nurver, Conkar, Secil, Poyrazoglu, Hakan M., Dusunsel, Ruhan, 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, König, Jens, Litwin, Mieczyslaw, Massella, Laura, Mekahli, Djalila, Melek, Engin, Mir, Sevgi, Pape, Lars, Prikhodina, Larisa, Ranchin, Bruno, Schild, Raphael, Seeman, Tomas, Sever, Lale, 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, Wühl, Elke, Wygoda, Simone, Yilmaz, Alev, Zachwieja, Katarzyna, Zagozdzon, Ilona, Zerres, Klaus, Dötsch, Jörg, Schaefer, Franz, and Liebau, Max Christoph

Published
2018
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13. Outcome of renal transplantation in small infants: a match-controlled analysis

Author

Weitz, Marcus, Laube, Guido F., Schmidt, Maria, Krupka, Kai, Murer, Luisa, Müller, Dominik, Hoppe, Bernd, Büscher, Anja, König, Jens, Pohl, Martin, Jungraithmayr, Therese, Thiel, Florian, Billing, Heiko, Grenda, Ryszard, Rubik, Jacek, Kaabak, Michael M., Yalcinkaya, Fatos, Topaloglu, Rezan, Webb, Nicholas, Dello Strologo, Luca, Pape, Lars, Nadalin, Silvio, and Tönshoff, Burkhard

Published
2018
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14. Vaccination titres pre- and post-transplant in paediatric renal transplant recipients and the impact of immunosuppressive therapy

Author

Höcker, Britta, Aguilar, Martin, Schnitzler, Paul, Pape, Lars, Bald, Martin, König, Jens, Marks, Stephen D., Genc, Gurkan, Büscher, Anja, Kemper, Markus J., Billing, Heiko, Pohl, Martin, Dello Strologo, Luca, Webb, Nicholas J. A., Rieger, Susanne, Mankertz, Annette, Krupka, Kai, Bruckner, Thomas, Fichtner, Alexander, and Tönshoff, Burkhard

Published
2018
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15. Kidney/eye syndromes

Author

Salvetti, Anna Paola, additional, Mackensen, Friederike, additional, Schwenger, Vedat, additional, and Billing, Heiko, additional

Published
2018
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16. Clinical courses and complications of young adults with Autosomal Recessive Polycystic Kidney Disease (ARPKD)

Author

Burgmaier, Kathrin, Kilian, Samuel, Bammens, Bert, Benzing, Thomas, Billing, Heiko, Büscher, Anja, Galiano, Matthias, Grundmann, Franziska, Klaus, Günter, Mekahli, Djalila, Michel-Calemard, Laurence, Milosevski-Lomic, Gordana, Ranchin, Bruno, Sauerstein, Katja, Schaefer, Susanne, Shroff, Rukshana, Sterenborg, Rosalie, Verbeeck, Sarah, Weber, Lutz T., Wicher, Dorota, Wühl, Elke, Dötsch, Jörg, Schaefer, Franz, and Liebau, Max C.

Published
2019
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17. Effects of growth hormone treatment on adult height in severely short children with X-linked hypophosphatemic rickets

Author

Meyerhoff, Nadine, Haffner, Dieter, Staude, Hagen, Wühl, Elke, Marx, Michaela, Beetz, Rolf, Querfeld, Uwe, Holder, Martin, Billing, Heiko, Rabl, Wolfgang, Schröder, Carmen, Hiort, Olaf, Brämswig, Jürgen H., Richter-Unruh, Annette, Schnabel, Dirk, Živičnjak, Miroslav, and on Behalf of the Hypophosphatemic Rickets Study Group of the “Deutsche Gesellschaft für Kinderendokrinologie und -diabetologie” and “Gesellschaft für Pädiatrische Nephrologie”

Published
2017
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18. Incomplete vaccination coverage in European children with end-stage kidney disease prior to renal transplantation

Author

Höcker, Britta, Aguilar, Martin, Schnitzler, Paul, Pape, Lars, Dello Strologo, Luca, Webb, Nicholas J. A., Bald, Martin, Genc, Gurkan, Billing, Heiko, König, Jens, Büscher, Anja, Kemper, Markus J., Marks, Stephen D., Pohl, Martin, Wigger, Marianne, Topaloglu, Rezan, Rieger, Susanne, Krupka, Kai, Bruckner, Thomas, Fichtner, Alexander, and Tönshoff, Burkhard

Published
2017
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20. Heterozygous variants in the DVL2 interaction region of DACT1 cause CAKUT and features of Townes–Brocks syndrome 2

Author

Christians, Anne, primary, Kesdiren, Esra, additional, Hennies, Imke, additional, Hofmann, Alejandro, additional, Trowe, Mark-Oliver, additional, Brand, Frank, additional, Martens, Helge, additional, Gjerstad, Ann Christin, additional, Gucev, Zoran, additional, Zirngibl, Matthias, additional, Geffers, Robert, additional, Seeman, Tomáš, additional, Billing, Heiko, additional, Bjerre, Anna, additional, Tasic, Velibor, additional, Kispert, Andreas, additional, Ure, Benno, additional, Haffner, Dieter, additional, Dingemann, Jens, additional, and Weber, Ruthild G., additional

Published
2022
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21. Relationship between age at initiation of cysteamine treatment, adherence with therapy, and glomerular kidney function in infantile nephropathic cystinosis

Author

Nießl, Christina, primary, Boulesteix, Anne-Laure, additional, Oh, Jun, additional, Palm, Katja, additional, Schlingmann, Peter, additional, Wygoda, Simone, additional, Haffner, Dieter, additional, Wühl, Elke, additional, Tönshoff, Burkhard, additional, Buescher, Anja, additional, Billing, Heiko, additional, Hoppe, Bernd, additional, Zirngibl, Matthias, additional, Kettwig, Matthias, additional, Moeller, Kristina, additional, Acham-Roschitz, Birgit, additional, Arbeiter, Klaus, additional, Bald, Martin, additional, Benz, Marcus, additional, Galiano, Matthias, additional, John-Kroegel, Ulrike, additional, Klaus, Guenter, additional, Marx-Berger, Daniela, additional, Moser, Katja, additional, Mueller, Dirk, additional, Patzer, Ludwig, additional, Pohl, Martin, additional, Seitz, Barbara, additional, Treikauskas, Ulrike, additional, von Vigier, Rodo O., additional, Gahl, William Allen, additional, and Hohenfellner, Katharina, additional

Published
2022
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22. Epidemiology and Morbidity of Epstein-Barr Virus Infection in Pediatric Renal Transplant Recipients: A Multicenter, Prospective Study

Author

Höcker, Britta, Fickenscher, Helmut, Delecluse, Henri-Jacques, Böhm, Stephan, Küsters, Uta, Schnitzler, Paul, Pohl, Martin, John, Ulrike, Kemper, Markus J., Fehrenbach, Henry, Wigger, Marianne, Holder, Martin, Schröder, Monika, Billing, Heiko, Fichtner, Alexander, Feneberg, Reinhard, Sander, Anja, Köpf-Shakib, Sabine, Süsal, Caner, and Tönshoff, Burkhard

Published
2013
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23. Body growth, upper arm fat area, and clinical parameters in children with nephropathic cystinosis compared with other pediatric chronic kidney disease entities

Author

Kluck, Rika, Mueller, Sophia, Jagodzinski, Celina, Hohenfellner, Katharina, Buescher, Anja, Kemper, Markus J., Oh, Jun, Billing, Heiko, Thumfart, Julia, Weber, Lutz T., Acham-Roschitz, Birgit, Arbeiter, Klaus, Toenshoff, Burkhard, Hagenberg, Martina, Kanzelmeyer, Nele, Pavicic, Leo, Haffner, Dieter, Zivicnjak, Miroslav, Kluck, Rika, Mueller, Sophia, Jagodzinski, Celina, Hohenfellner, Katharina, Buescher, Anja, Kemper, Markus J., Oh, Jun, Billing, Heiko, Thumfart, Julia, Weber, Lutz T., Acham-Roschitz, Birgit, Arbeiter, Klaus, Toenshoff, Burkhard, Hagenberg, Martina, Kanzelmeyer, Nele, Pavicic, Leo, Haffner, Dieter, and Zivicnjak, Miroslav

Abstract

Children with infantile nephropathic cystinosis (INC), an inherited lysosomal storage disease resulting in cystine accumulation in all body cells, are prone to progressive chronic kidney disease (CKD), impaired growth and reduced weight gain; however, systematic anthropometric analyses are lacking. In this prospective multicenter study we investigated linear growth, body proportion, body mass index (BMI), upper arm fat area (UFA) and biochemical parameters in 43 pediatric INC patients with CKD stages 1 to 5 and 49 age-matched CKD controls, with 193 annual measurements. INC patients showed more impaired height than CKD controls (-1.8 vs -0.7 z-score; P < .001), despite adequate cysteamine therapy, treatment for Fanconi syndrome and more frequent use of growth hormone. Only the youngest INC patients shared the same body pattern with CKD controls characterized by preferential impairment of leg length and rather preserved trunk length. In late-prepuberty, body pattern changed only in INC patients due to improved leg growth and more impaired trunk length. Mean UFA z-score in INC patients was slightly reduced in early childhood and progressively decreased thereafter reaching -0.8 z-score in adolescence, while CKD controls showed a steady increase in standardized BMI and UFA especially during adolescent age. Menarche in female INC patients was significantly delayed compared to CKD controls. Our data indicate that with age and progression of disease, pediatric INC patients undergo unique changes of body growth and fat stores that are distinct from those with CKD stemming from other causes, suggesting other factors apart from CKD to contribute to this development. Pediatric patients with infantile nephropathic cystinosis display more severe impaired linear growth than other peer CKD patients, despite of cysteamine treatment, supplementation for Fanconi syndrome, and more frequent use of growth hormone, with a distinct change of body proportions and overall lower body fat.

Published
2022

25. Body growth, upper arm fat area, and clinical parameters in children with nephropathic cystinosis compared with other pediatric chronic kidney disease entities

Author

Kluck, Rika, primary, Müller, Sophia, additional, Jagodzinski, Celina, additional, Hohenfellner, Katharina, additional, Büscher, Anja, additional, Kemper, Markus J., additional, Oh, Jun, additional, Billing, Heiko, additional, Thumfart, Julia, additional, Weber, Lutz T., additional, Acham‐Roschitz, Birgit, additional, Arbeiter, Klaus, additional, Tönshoff, Burkhard, additional, Hagenberg, Martina, additional, Kanzelmeyer, Nele, additional, Pavičić, Leo, additional, Haffner, Dieter, additional, and Zivicnjak, Miroslav, additional

Published
2022
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29. Markers of childhood lupus nephritis indicating disease activity

Author

Edelbauer, Monika, Kshirsagar, Sudhir, Riedl, Magdalena, Haffner, Dieter, Billing, Heiko, Tönshoff, Burkhard, Ross, Sophia, Dötsch, Jörg, Amon, Oliver, Fehrenbach, Henry, Steuber, Christian, Beissert, Antje, Hager, Josef, Wechselberger, Gottfried, Weber, Lutz T., and Zimmerhackl, Lothar Bernd

Published
2011
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30. Age-related stature and linear body segments in children with X-linked hypophosphatemic rickets

Author

Živičnjak, Miroslav, Schnabel, Dirk, Billing, Heiko, Staude, Hagen, Filler, Guido, Querfeld, Uwe, Schumacher, Marius, Pyper, Anke, Schröder, Carmen, Brämswig, Jürgen, Haffner, Dieter, and Hypophosphatemic Rickets Study Group of the “Arbeitsgemeinschaft für Pädiatrische Endokrinologie” and “Gesellschaft für Pädiatrische Nephrologie”

Published
2011
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32. Endurance-oriented training program with children and adolescents on maintenance hemodialysis to enhance dialysis efficacy-DiaSport

Author

Feldkotter, Markus, Thys, Sarah, Adams, Anne, Becker, Ingrid, Buescher, Rainer, Pohl, Martin, Schild, Raphael, Pape, Lars, Schmitt, Claus Peter, Taylan, Christina, Wygoda, Simone, Klaus, Guenter, Fehrenbach, Henry, Montoya, Carmen, Konrad, Martin, Billing, Heiko, Schaar, Bettina, Hoppe, Bernd, Feldkotter, Markus, Thys, Sarah, Adams, Anne, Becker, Ingrid, Buescher, Rainer, Pohl, Martin, Schild, Raphael, Pape, Lars, Schmitt, Claus Peter, Taylan, Christina, Wygoda, Simone, Klaus, Guenter, Fehrenbach, Henry, Montoya, Carmen, Konrad, Martin, Billing, Heiko, Schaar, Bettina, and Hoppe, Bernd

Abstract

Objective Pediatric patients spend significant time on maintenance hemodialysis (HD) and traveling. They are often not capable of participating in sports activities. To assess the effects of exercise training during HD on dialysis efficacy in children and adolescents, we set up a multi-center randomized controlled trial (RCT). Methods Patients on HD, age 6 to 18 years, were randomized either to 3x weekly bicycle ergometer training or to no train g during HD for 12 weeks. Change in single-pool KtN (spKt/V) was the primary outcome parameter. Results We randomized 54 patients of whom 45 qualified (23 in the intervention and 22 in the waiting control group, 14.5 +/- 3.01 years, 32 male and 13 female) for the intention-to-treat (ITT) population. Only 26 patients fmished study per-protocol (PP). Training was performed for an average of 11.96 weeks (0.14-13.14) at 2.08 +/- 0.76 times per week and for a weekly mean of 55.52 +/- 27.26 min. Single-pool Kt/V was similar in the intervention compared to the control group (1.70 [0.33] vs. 1.79 [0.55]) at VO and (1.70 [0.36] vs. 1.71 [0.51]) at V1; secondary endpoints also showed no difference in both ITT and PP analysis. No significant adverse events were reported. No bleeding or needle dislocation occurred in 1670 training sessions. Conclusions Intradialytic bicycle training is safe, but does not improve dialysis efficacy and physical fitness. However, the study can be considered underpowered, particularly because of high dropout rates. Future studies need better strategies to increase motivation and compliance and other more effective/intensive exercise measures should be evaluated.

Published
2020

33. Endurance-oriented training program with children and adolescents on maintenance hemodialysis to enhance dialysis efficacy-DiaSport (Jun, 10.1007/s00467-021-05114-8, 2021)

Author

Feldkotter, Markus, Thys, Sarah, Adams, Anne, Becker, Ingrid, Buescher, Rainer, Pohl, Martin, Schild, Raphael, Pape, Lars, Schmitt, Claus Peter, Taylan, Christina, Wygoda, Simone, Klaus, Guenter, Fehrenbach, Henry, Montoya, Carmen, Konrad, Martin, Billing, Heiko, Schaar, Bettina, Hoppe, Bernd, Feldkotter, Markus, Thys, Sarah, Adams, Anne, Becker, Ingrid, Buescher, Rainer, Pohl, Martin, Schild, Raphael, Pape, Lars, Schmitt, Claus Peter, Taylan, Christina, Wygoda, Simone, Klaus, Guenter, Fehrenbach, Henry, Montoya, Carmen, Konrad, Martin, Billing, Heiko, Schaar, Bettina, and Hoppe, Bernd

Abstract

A Correction to this paper has been published

Published
2020

34. Bone and Mineral Metabolism in Children with Nephropathic Cystinosis Compared with other CKD Entities

Author

Ewert, Annika, Leifheit-Nestler, Maren, Hohenfellner, Katharina, Buescher, Anja, Kemper, Markus J., Oh, Jun, Billing, Heiko, Thumfart, Julia, Stangl, Gabriele, Baur, Anja C., Foeller, Michael, Feger, Martina, Weber, Lutz T., Acham-Roschitz, Birgit, Arbeiter, Klaus, Toenshoff, Burkhard, Zivicnjak, Miroslav, Haffner, Dieter, Ewert, Annika, Leifheit-Nestler, Maren, Hohenfellner, Katharina, Buescher, Anja, Kemper, Markus J., Oh, Jun, Billing, Heiko, Thumfart, Julia, Stangl, Gabriele, Baur, Anja C., Foeller, Michael, Feger, Martina, Weber, Lutz T., Acham-Roschitz, Birgit, Arbeiter, Klaus, Toenshoff, Burkhard, Zivicnjak, Miroslav, and Haffner, Dieter

Abstract

Context: Children with nephropathic cystinosis (NC) show persistent hypophosphatemia, due to Fanconi syndrome, as well as mineral and bone disorders related to chronic kidney disease (CKD); however, systematic analyses are lacking. Objective: To compare biochemical parameters of bone and mineral metabolism between children with NC and controls across all stages of CKD. Design: Cross-sectional multicenter study. Setting: Hospital clinics. Patients: Forty-nine children with NC, 80 CKD controls of the same age and CKD stage. Main outcome measures: Fibroblast growth factor 23 (FGF23), soluble Klotho, bone alkaline phosphatase (BAP), tartrate-resistant acid phosphatase 5b (TRAP5b), sclerostin, osteoprotegerin (OPG), biochemical parameters related to mineral metabolism, and skeletal comorbidity. Results: Despite Fanconi syndrome medication, NC patients showed an 11-fold increased risk of short stature, bone deformities, and/or requirement for skeletal surgery compared with CKD controls. This was associated with a higher frequency of risk factors such as hypophosphatemia, hypocalcemia, low parathyroid hormone (PTH), metabolic acidosis, and a specific CKD stage-dependent pattern of bone marker alterations. Pretransplant NC patients in mild to moderate CKD showed a delayed increase or lacked an increase in FGF23 and sclerostin, and increased BAP, TRAP5b, and OPG concentrations compared with CKD controls. Post-transplant, BAP and OPG returned to normal, TRAP5b further increased, whereas FGF23 and PTH were less elevated compared with CKD controls and associated with higher serum phosphate. Conclusions: Patients with NC show more severe skeletal comorbidity associated with distinct CKD stage-dependent alterations of bone metabolism than CKD controls, suggesting impaired mineralization and increased bone resorption, which is only partially normalized after renal transplantation.

Published
2020

46. Different approaches to long-term treatment of aHUS due to MCP mutations: a multicenter analysis

Author

Klämbt, Verena, primary, Gimpel, Charlotte, additional, Bald, Martin, additional, Gerken, Christopher, additional, Billing, Heiko, additional, Loos, Sebastian, additional, Hansen, Matthias, additional, König, Jens, additional, Vinke, Tobias, additional, Montoya, Carmen, additional, Lange-Sperandio, Bärbel, additional, Kirschstein, Martin, additional, Hennies, Imke, additional, Pohl, Martin, additional, and Häffner, Karsten, additional

Published
2020
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47. A multicenter, randomized, placebo-controlled, double-blind phase 3 trial with open-arm comparison indicates safety and efficacy of nephroprotective therapy with ramipril in children with Alport’s syndrome

Author

Gross, Oliver, primary, Tönshoff, Burkhard, additional, Weber, Lutz T., additional, Pape, Lars, additional, Latta, Kay, additional, Fehrenbach, Henry, additional, Lange-Sperandio, Baerbel, additional, Zappel, Hildegard, additional, Hoyer, Peter, additional, Staude, Hagen, additional, König, Sabine, additional, John, Ulrike, additional, Gellermann, Jutta, additional, Hoppe, Bernd, additional, Galiano, Matthias, additional, Hoecker, Britta, additional, Ehren, Rasmus, additional, Lerch, Christian, additional, Kashtan, Clifford E., additional, Harden, Markus, additional, Boeckhaus, Jan, additional, Friede, Tim, additional, Koziolek, Michael, additional, Bramlage, Carsten Paul, additional, Weber, Frauke, additional, Albrecht-Nock, Tanja, additional, Sonntag, Joseph, additional, Frese, Jenny, additional, Kettwig, Matthias, additional, Hilgers, Reinhard, additional, Hansen, Matthias, additional, Wedekin, Mirja, additional, Meyer, Nicole, additional, Klaiber, Susanne, additional, Gessner, Michaela, additional, Liebau, Max, additional, Vogt-Weigeldt, Anne-Kristin, additional, Jungraithmayr, Therese, additional, Ponsel, Sabine, additional, Jacoby, Ulrike, additional, Konrad, Martin, additional, Kranz, Brigitta, additional, Koenig, Jens, additional, Loechtermann, Lisa, additional, Pohl, Michael, additional, Husain, Ralf, additional, Mueller, Katrin, additional, Thumfart, Julia, additional, Schalk, Gesa, additional, Feldkoetter, Markus, additional, Schmidt, Sabine, additional, Sauerstein, Katja, additional, Muschiol, Evelin, additional, Billing, Heiko, additional, and Wilkening, Frauke, additional

Published
2020
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48. Bone and Mineral Metabolism in Children with Nephropathic Cystinosis Compared with other CKD Entities

Author

Ewert, Annika, primary, Leifheit-Nestler, Maren, additional, Hohenfellner, Katharina, additional, Büscher, Anja, additional, Kemper, Markus J, additional, Oh, Jun, additional, Billing, Heiko, additional, Thumfart, Julia, additional, Stangl, Gabriele, additional, Baur, Anja C, additional, Föller, Michael, additional, Feger, Martina, additional, Weber, Lutz T, additional, Acham-Roschitz, Birgit, additional, Arbeiter, Klaus, additional, Tönshoff, Burkhard, additional, Zivicnjak, Miroslav, additional, and Haffner, Dieter, additional

Published
2020
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50. Sex and age as determinants for high blood pressure in pediatric renal transplant recipients: a longitudinal analysis of the CERTAIN Registry

Author

Sugianto, Rizky I., primary, Schmidt, Bernhard M. W., additional, Memaran, Nima, additional, Duzova, Ali, additional, Topaloglu, Rezan, additional, Seeman, Tomas, additional, König, Sabine, additional, Dello Strologo, Luca, additional, Murer, Luisa, additional, Özçakar, Zeynep Birsin, additional, Bald, Martin, additional, Shenoy, Mohan, additional, Buescher, Anja, additional, Hoyer, Peter F., additional, Pohl, Michael, additional, Billing, Heiko, additional, Oh, Jun, additional, Staude, Hagen, additional, Pohl, Martin, additional, Genc, Gurkan, additional, Klaus, Günter, additional, Alparslan, Caner, additional, Grenda, Ryszard, additional, Rubik, Jacek, additional, Krupka, Kai, additional, Tönshoff, Burkhard, additional, Wühl, Elke, additional, and Melk, Anette, additional

Published
2019
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