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1. Incidence and predictors of hepatocellular carcinoma in patients with autoimmune hepatitis

Author

van Gerven, N., van Erpecum, K., Ouden, J den, Brouwer, J., Vrolijk, J., Gevers, T.J., Drenth, J., Guichelaar, M., Bouma, G., Schreuder, T.C.M.A., van der Wouden, E.J., Baak, L.C., Stadhouders, P., Klemt-Kropp, M., Verhagen, M., Bhalla, A., Kuijvenhoven, J., Almasio, P., Alvarez, F., Andrade, R., Arikan, C., Assis, D., Bardou-Jacquet, E., Biewenga, M., Cancado, E., Cazzagon, N., Chazouillères, O., Colloredo, G., Cuarterolo, M., Dalekos, G., Debray, D., Robles-Díaz, M., Dyson, J., Efe, C., Engel, B., Ferri, S., Fontana, R., Gatselis, N., Gerussi, A., Halilbasic, E., Halliday, N., Heneghan, M., Hirschfield, G., van Hoek, B., Hørby Jørgensen, M., Indolfini, G., Iorio, R., Invernizzi, P., Jeong, S., Jones, D., Kelly, D., Kerkar, N., Lacaille, F., Lammert, C., Leggett, B., Lenzi, M., Levy, C., Liberal, R., Lleo, A., Lohse, A., Lopez, S. Ines, de Martin, E., McLin, V., Mieli-Vergani, G., Milkiewicz, P., Mohan, N., Muratori, L., Nebbia, G., van Nieuwkerk, C., Oo, Y., Ortega, A., Páres, A., Pop, T., Pratt, D., Purnak, T., Ranucci, G., Rushbrook, S., Schramm, C., Stättermayer, A., Swain, M., Tanaka, A., Taubert, R., Terrabuio, D., Terziroli, B., Trauner, M., Valentino, P., van den Brand, F., Vergani, D., Villamil, A., Wahlin, S., Ytting, H., Zachou, K., Zeniya, M., Colapietro, Francesca, Maisonneuve, Patrick, Lytvyak, Ellina, Beuers, Ulrich, Verdonk, Robert C., van der Meer, Adriaan J., van Hoek, Bart, Kuiken, Sjoerd D., Brouwer, Johannes T., Muratori, Paolo, Aghemo, Alessio, Carella, Francesco, van den Berg, Ad P., Zachou, Kalliopi, Dalekos, George N., Di Zeo-Sánchez, Daniel E., Robles, Mercedes, Andrade, Raul J., Montano-Loza, Aldo J., van den Brand, Floris F., Slooter, Charlotte D., Macedo, Guilherme, Liberal, Rodrigo, de Boer, Ynto S., and Lleo, Ana

Published
2024
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4. Incidence and predictors of hepatocellular carcinoma in patients with autoimmune hepatitis

Author

Colapietro, Francesca, primary, Maisonneuve, Patrick, additional, Lytvyak, Ellina, additional, Beuers, Ulrich, additional, Verdonk, Robert C., additional, van der Meer, Adriaan J., additional, van Hoek, Bart, additional, Kuiken, Sjoerd D., additional, Brouwer, Johannes T., additional, Muratori, Paolo, additional, Aghemo, Alessio, additional, Carella, Francesco, additional, van den Berg, Ad P., additional, Zachou, Kalliopi, additional, Dalekos, George N., additional, Di Zeo-Sánchez, Daniel E., additional, Robles, Mercedes, additional, Andrade, Raul J., additional, Montano-Loza, Aldo J., additional, van den Brand, Floris F., additional, Slooter, Charlotte D., additional, Macedo, Guilherme, additional, Liberal, Rodrigo, additional, de Boer, Ynto S., additional, Lleo, Ana, additional, van Gerven, N., additional, van Erpecum, K., additional, Ouden, J den, additional, Brouwer, J., additional, Vrolijk, J., additional, Gevers, T.J., additional, Drenth, J., additional, Guichelaar, M., additional, Bouma, G., additional, Schreuder, T.C.M.A., additional, van der Wouden, E.J., additional, Baak, L.C., additional, Stadhouders, P., additional, Klemt-Kropp, M., additional, Verhagen, M., additional, Bhalla, A., additional, Kuijvenhoven, J., additional, Almasio, P., additional, Alvarez, F., additional, Andrade, R., additional, Arikan, C., additional, Assis, D., additional, Bardou-Jacquet, E., additional, Biewenga, M., additional, Cancado, E., additional, Cazzagon, N., additional, Chazouillères, O., additional, Colloredo, G., additional, Cuarterolo, M., additional, Dalekos, G., additional, Debray, D., additional, Robles-Díaz, M., additional, Dyson, J., additional, Efe, C., additional, Engel, B., additional, Ferri, S., additional, Fontana, R., additional, Gatselis, N., additional, Gerussi, A., additional, Halilbasic, E., additional, Halliday, N., additional, Heneghan, M., additional, Hirschfield, G., additional, van Hoek, B., additional, Hørby Jørgensen, M., additional, Indolfini, G., additional, Iorio, R., additional, Invernizzi, P., additional, Jeong, S., additional, Jones, D., additional, Kelly, D., additional, Kerkar, N., additional, Lacaille, F., additional, Lammert, C., additional, Leggett, B., additional, Lenzi, M., additional, Levy, C., additional, Liberal, R., additional, Lleo, A., additional, Lohse, A., additional, Lopez, S. Ines, additional, de Martin, E., additional, McLin, V., additional, Mieli-Vergani, G., additional, Milkiewicz, P., additional, Mohan, N., additional, Muratori, L., additional, Nebbia, G., additional, van Nieuwkerk, C., additional, Oo, Y., additional, Ortega, A., additional, Páres, A., additional, Pop, T., additional, Pratt, D., additional, Purnak, T., additional, Ranucci, G., additional, Rushbrook, S., additional, Schramm, C., additional, Stättermayer, A., additional, Swain, M., additional, Tanaka, A., additional, Taubert, R., additional, Terrabuio, D., additional, Terziroli, B., additional, Trauner, M., additional, Valentino, P., additional, van den Brand, F., additional, Vergani, D., additional, Villamil, A., additional, Wahlin, S., additional, Ytting, H., additional, Zachou, K., additional, and Zeniya, M., additional

Published
2023
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9. Surgical site infections after simultaneous pancreas kidney and pancreas transplantation in the Swiss Transplant Cohort Study

Author

Schreiber, P.W., primary, Laager, M., additional, Boggian, K., additional, Neofytos, D., additional, van Delden, C., additional, Egli, A., additional, Dickenmann, M., additional, Hirzel, C., additional, Manuel, O., additional, Koller, M., additional, Rossi, S., additional, Schmied, B., additional, Gürke, L., additional, Matter, M., additional, Berney, T., additional, de Rougemont, O., additional, Kuster, S.P., additional, Stampf, S., additional, Mueller, N.J., additional, Amico, P., additional, Aubert, J-D., additional, Banz, V., additional, Beckmann, S., additional, Beldi, G., additional, Berger, C., additional, Berishvili, E., additional, Berzigotti, A., additional, Binet, I., additional, Bochud, P-Y., additional, Branca, S., additional, Bucher, H., additional, Catana, E., additional, Cairoli, A., additional, Chalandon, Y., additional, De Geest, S., additional, De Rougemont, O., additional, De Seigneux, S., additional, Dreifuss, J.L., additional, Duchosal, M., additional, Fehr, T., additional, Ferrari-Lacraz, S., additional, Garzoni, C., additional, Golshayan, D., additional, Goossens, N., additional, Halter, F.H.J., additional, Heim, D., additional, Hess, C., additional, Hillinger, S., additional, Hirsch, H.H., additional, Hirt, P., additional, Hofbauer, G., additional, Huynh-Do, U., additional, Immer, F., additional, Laesser, B., additional, Lamoth, F., additional, Lehmann, R., additional, Leichtle, A., additional, Marti, H.P., additional, Martinelli, M., additional, McLin, V., additional, Mellac, K., additional, Merçay, A., additional, Mettler, K., additional, Müller, A., additional, Müller-Arndt, U., additional, Müllhaupt, B., additional, Nägeli, M., additional, Oldani, G., additional, Pascual, M., additional, Passweg, J., additional, Pazeller, R., additional, Posfay-Barbe, K., additional, Rick, J., additional, Rosselet, A., additional, Rothlin, S., additional, Ruschitzka, F., additional, Schachtner, T., additional, Schanz, U., additional, Schaub, S., additional, Scherrer, A., additional, Schnyder, A., additional, Schuurmans, M., additional, Schwab, S., additional, Sengstag, T., additional, Simonetta, F., additional, Steiger, J., additional, Stirnimann, G., additional, Stürzinger, U., additional, Van Delden, C., additional, Venetz, J-P., additional, Villard, J., additional, Vionnet, J., additional, Wick, M., additional, Wilhelm, M., additional, and Yerly, P., additional

Published
2022
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11. Systematic review of response criteria and endpoints in autoimmune hepatitis by the International Autoimmune Hepatitis Group

Author

Pape, Simon, primary, Snijders, Romée J.A.L.M., additional, Gevers, Tom J.G., additional, Chazouilleres, Oliver, additional, Dalekos, George N., additional, Hirschfield, Gideon M., additional, Lenzi, Marco, additional, Trauner, Michael, additional, Manns, Michael P., additional, Vierling, John M., additional, Montano-Loza, Aldo J., additional, Lohse, Ansgar W., additional, Schramm, Christoph, additional, Drenth, Joost P.H., additional, Heneghan, Michael A., additional, Almasio, P., additional, Alvarez, F., additional, Andrade, R., additional, Arikan, C., additional, Assis, D., additional, Bardou-Jacquet, E., additional, Biewenga, M., additional, Cancado, E., additional, Cazzagon, N., additional, Chazouillères, O., additional, Colloredo, G., additional, Cuarterolo, M., additional, Dalekos, G., additional, Debray, D., additional, Robles-Díaz, M., additional, Drenth, J., additional, Dyson, J., additional, Efe, C., additional, Engel, B., additional, Ferri, S., additional, Fontana, R., additional, Gatselis, N., additional, Gerussi, A., additional, Halilbasic, E., additional, Halliday, N., additional, Heneghan, M., additional, Hirschfield, G., additional, van Hoek, B., additional, Hørby Jørgensen, M., additional, Indolfini, G., additional, Iorio, R., additional, Jeong, S., additional, Jones, D., additional, Kelly, D., additional, Kerkar, N., additional, Lacaille, F., additional, Lammert, C., additional, Leggett, B., additional, Lenzi, M., additional, Levy, C., additional, Liberal, R., additional, Lleo, A., additional, Lohse, A., additional, Ines Lopez, S., additional, de Martin, E., additional, McLin, V., additional, Mieli-Vergani, G., additional, Milkiewicz, P., additional, Mohan, N., additional, Muratori, L., additional, Nebbia, G., additional, van Nieuwkerk, C., additional, Oo, Y., additional, Ortega, A., additional, Páres, A., additional, Pop, T., additional, Pratt, D., additional, Purnak, T., additional, Ranucci, G., additional, Rushbrook, S., additional, Schramm, C., additional, Stättermayer, A., additional, Swain, M., additional, Tanaka, A., additional, Taubert, R., additional, Terrabuio, D., additional, Terziroli, B., additional, Trauner, M., additional, Valentino, P., additional, van den Brand, F., additional, Villamil, A., additional, Wahlin, S., additional, Ytting, H., additional, Zachou, K., additional, and Zeniya, M., additional

Published
2022
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21. Intracranial hypertension and papilledema in a large cohort of pediatric Alagille syndrome

Author

ROCK, Nathalie, Demaret, T, Stephenne, Xavier, Scheers, Isabelle, Smets, Françoise, McLin, V, Boschi, Antonella, Sokal, Etienne, UCL - SSS/IREC/PEDI - Pôle de Pédiatrie, UCL - (SLuc) Service de gastro-entérologie et hépatologie pédiatrique, and UCL - (SLuc) Service d'ophtalmologie

Subjects
and Child Health, Gastroenterology, Pediatrics, Perinatology
Abstract

Objectives and Study: Ophthalmic abnormalities are amongst the 5 major criteria for diagnosing autosomal dominant Alagille syndrome (AS), of which embryotoxon, pseudo-papilledema (PPO), and pigmentary retinopathy are the most common. Papilledema (PO), with or without intracranial hypertension (ICHT)/pseudotumor cerebri syndrome (PTCS), are rarely described. We here report 9 cases of bilateral PO in an AS cohort, 5 of whom were diagnosed with PTCS that occurred after liver transplantation. Methods: We reviewed ophthalmologic examination data from 85 pediatric patients with clinically (n = 48) and/or genetically (n = 37) proven AS, who were followed in 2 referral hospitals (UCL St Luc Brussels, n = 75; HUG Geneva, n = 10). Patients who received liver transplantation (LT) (n = 40) were compared with pediatric patients (n = 40) with LT for indications other than AS. Results: Sixty-nine patients fulfilled the inclusion criteria; the PO incidence in this cohort was 13% (9/69, 13%). Forty of these patients (40/69, 58%) underwent LT, of whom 2 developed true PO before LT and 6 developed PO after LT (6/40, 15%). One non-LT child (1/28, 3%) had PO, but had a normal neurological examination and rapid resolution. PO resolved spontaneously or remained stable without complications in 4 patients (2 with pre-transplant onset) (4/69, 6%). The remaining 5 patients had a normal neurological examination and cerebral magnetic resonance imaging findings (5/69, 7%). Probable PTCS was diagnosed in 1 patient and definite PCTS was diagnosed in 4 patients. PTCS was treated with steroids alone in 1 patient, and in combination with acetazolamide in 2 patients. In 2 of these 3 patients, ventriculo-peritoneal derivation was ultimately required for severe progressive visual loss. PPO was present in 10 other (10/69, 10%). One patient in the non-AS cohort had PO, yielding an incidence of 2.5% (1/40). Conclusions: True PTCS may be underdiagnosed in AS, but should be considered as a feature of this syndrome. Our findings supports the necessity of close follow-up of ophthalmologic complications before and/or after LT, because of the risk of severe and irreversible loss of vision.

Published
2019

23. Intracranial hypertension and papilloedema in a large cohort of pediatric alagille syndrome

Author

ROCK, Nathalie, Demaret, Tanguy, Stephenne, Xavier, Scheers, Isabelle, Smets, Françoise, McLin, V, Boschi, Antonella, Sokal, Etienne, UCL - SSS/IREC/PEDI - Pôle de Pédiatrie, UCL - (SLuc) Service d'ophtalmologie, and UCL - (SLuc) Service de gastro-entérologie et hépatologie pédiatrique

Subjects
Hepatology, eye diseases
Abstract

Background: Ophthalmic abnormalities are amongst the 5 major criteria for diagnosing autosomal dominant Alagille Syndrome (AS). Embryotoxon, pseudo-papilledema and pigmentary retinopathy being most frequently part of the syndrome. Papilledema with or without intracranial hypertension (ICHT) are rarely described. We report 9 cases of bilateral papilloedema, 4 associated to ICHT at lumbar puncture (LP) , all being diagnosed idiopathic ICHT in children with AS. Methods: We reviewed ophthalmologic examination data from 85 pediatric patients with clinically (n=48) and/or genetically (n=37) proven AS followed in two referring hospitals (UCL St Luc Brussels, n=75; HUG Geneva n=10). Patients were included if data were available before and after liver transplantation (LT). Results: 69 patients fulfilled inclusions criteria. 41 patients (41/69, 59%) of this group had LT of whom 2 developed true papilloedema before LT and 6 after LT (6/41, 14%). One papilloedema, but with normal neurologic exam and a rapid resolution was found in a non-transplanted child (1/28, 3%). Papilloedema resolved spontaneously in 4 patients (2 with pre-transplant onset) (4/69, 6%). In a fifth patient, severe papilloedema without documented ICHT (normal LP) resolved after switching tacrolimus to cyclosporine and adding acetazolamide. In only 4 remaining patients; the ICHT was revealed at LP. In all patients normal neurological exam and cerebral MRI were reported (4/69, 6%). ICHT was treated by steroids alone in one patient, and in the 3 others patients in combination with acetazolamide and a switch from tacrolimus to cyclosporine. Among these 3 patients, ventriculo-peritoneal derivation was ultimately required in 2 for severe progressive visual loss. We detected pseudo-papilloedema in 7 additional children (7/69, 10%); which was persistent and unchanged after LT in n=4. Conclusion: Besides pseudopapilloedema, true papilloedema with or without proven ICHT in a context of AS occurs in probably more patients than expected. A close follow-up of ophthalmologic complications should be implemented for these patients before and/or after LT because the risk of severe and irreversible visual loss.

Published
2018

24. Chronic graft injury and long-term outcome after paediatric liver transplantation

Author

Hartleif, S., Rajanayagam, J., Cousin, V., Debray, D., Demetris, A. J., Evans, H., Fischler, B., Gonzales, E., Gouw, A., Haller, W., Hubscher, S., Lacaille, F., Malenicka, S., McLin, V., Mazariegos, G., Scheenstra, R., Squires, J., Verkade, H., Kelly, D., Sturm, E., Groningen Institute for Organ Transplantation (GIOT), Center for Liver, Digestive and Metabolic Diseases (CLDM), and Lifestyle Medicine (LM)

Published
2017

33. Dyslipidemia and sustained elevations in transaminases from early childhood are common in lysosomal acid lipase deficiency

Author

Deegan, P., primary, Burton, B., additional, Di Rocco, M., additional, Enns, G., additional, Guardmagna, O., additional, Horslen, S., additional, Hovingh, G., additional, Lobritto, S., additional, Malinova, V., additional, McLin, V., additional, Raiman, J., additional, Santra, S., additional, Sharma, R., additional, Sykut-Cegielska, J., additional, Tripuraneni, R., additional, Valayannopoulos, V., additional, Whitley, C., additional, Eckert, S., additional, and Quinn, A., additional

Published
2014
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42. Glomerular and tubular function following orthotopic liver transplantation in children treated with tacrolimus.

Author

Anastaze Stelle, K., Belli, D. C., Parvex, P., Girardin, E., Giroud, A., Wildhaber, B., and McLin, V. A.

Subjects
GLOMERULAR filtration rate, LIVER transplantation, TACROLIMUS, PEDIATRIC therapy, ANTIHYPERTENSIVE agents
Abstract

Anastaze Stelle K, Belli DC, Parvex P, Girardin E, Giroud A, Wildhaber B, McLin VA. Glomerular and tubular function following orthotopic liver transplantation in children treated with tacrolimus. Abstract: The aim of this study was to analyze the impact of TAC on medium term (three-yr follow-up) renal function in pediatric liver transplant (OLT) recipients. Glomerular and tubular indices were retrospectively analyzed in 24 consecutive OLT pediatric recipients on TAC. CrCl increased significantly each month post-OLT (p = 0.003), with a trend toward significance between pre-OLT and 36 months (p = 0.17). There was no correlation between CrCl and TAC troughs (p = 0.783). Sixteen percent of patients had CrCl <60 mL/min/1.73 m2 pre-OLT vs. none at 36 months post-OLT. TRP values were normal throughout the study. UPr/Cr decreased insignificantly over time and correlated significantly with TAC trough levels (p = 0.031). UCa/Cr values normalized by the third-month post-OLT, decreasing significantly over the time (p = 0.000) but did not correlate with TAC troughs. At three months post-OLT, 65.2% of patients needed antihypertensive therapy, and no patients needed more than one antihypertensive treatment after one yr. Despite nephrotoxic side effects in the early postoperative phase, this study shows that 65.5% patients had a normal renal function by three yr post-OLT. Tubular indices correlated with TAC trough levels. [ABSTRACT FROM AUTHOR]

Published
2012
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47. Liver Transplantation for Pediatric Portopulmonary Hypertension.

Author

Wacker, J., Joye, R., McLin, V., Wildhaber, B., Toso, C., Genecand, L., Lador, F., and Beghetti, M.

Subjects
*LIVER transplantation, *PULMONARY arterial hypertension, *PORTAL hypertension, *LUNG transplantation, *HYPERTENSION, PORTAL vein diseases
Abstract

Portopulmonary hypertension (PoPH) is a serious complication occuring in 1 to 5% of children with portal hypertension and in 11% of children with congenital portosystemic shunt. Outcome of pediatric PoPH remains poor. Liver transplant (LT) is indicated in adults with PoPH fulfilling hemodynamic criteria, but data in children are lacking. We retrospectively reviewed all children that underwent LT for PoPH at our center, and described their characteristics and outcome. Among the 217 pediatric LT performed at our center, 4 children underwent LT for PoPH (Table 1). Three had severe pulmonary arterial hypertension (PAH) before therapy initiation. There was no mortality at a mean follow-up of 5 years. Two patients improved their haemodynamics following LT, while two patients worsened. Patient 1 has significant developmental delay and poor medical adherence, rendering the course of her post LT hemodynamics difficult to interpret. Patient 2 had initial post-LT improvement of her PAH, allowing for epoprostenol wean. Her PAH then progressively worsened, and she underwent lung transplant 7 years after LT. Patient 4 developed portal hypertension after closure of her porto-systemic shunt. After a very complicated post-LT course with sepsis and severe PAH requiring ECMO, her hemodynamics have dramatically improved, allowing epoprostenol wean. Liver transplantation may be considered in selected cases of pediatric PoPH, after optimization of hemodynamics with aggressive treatment using PAH-specific therapies. Hemodynamic criteria for LT need to be better defined, but pre-LT right ventricular function seems to be a critical factor. Post-LT outcome based on our small series is encouraging, and deserves further research. Although some patients may worsen their hemodynamics a few years after LT, postponing the need for lung transplantation may result in better outcome than upfront combined lung and liver transplantation considering the survival prospect following lung transplantation for PoPH. [ABSTRACT FROM AUTHOR]

Published
2023
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48. Treatment and monitoring of children with chronic hepatitis C in the Pre-DAA era: A European survey of 38 paediatric specialists

Author

Indolfi, Giuseppe, Bailey, Heather, Serranti, Daniele, Giaquinto, Carlo, Thorne, Claire, Jahnel, Jörg, Sokal, Etienne, Lamireau, Thierry, Lacaille, Florence, Debray, Dominique, Girard, Muriel, Feiterna‐Sperling, Cornelia, Wirth, Stefan, Vassiliki, Papaevangelou, Dezsofi, Antal, Guidi, Roberto, Verucchi, Gabriella, D'Antiga, Lorenzo, Nicastro, Emanuele, Maggiore, Giuseppe, Trapani, Sandra, Ricci, Silvia, Resti, Massimo, Giacomet, Vania, Benincaso, Anna Rita, Nebbia, Gabriella, Iorio, Raffaele, Cananzi, Mara, Riva, Silvia, Bossi, Grazia, Dodi, Icilio, Nobili, Valerio, Comparcola, Donatella, Garazzino, Silvia, Calvo, Pier Luigi, Pokorska‐Śpiewak, Maria, Pawlowska, Malgorzata, Gonçalves, Cristina, Gonçalves, Isabel, Tudor, Ana Maria, Julian, Antoni Noguera, Hierro, Loreto, Ramos, Jose T., Fischler, Björn, McLin, Valérie, Kansu, Turkey Aydan, Brown, Maxine, Kelly, Deirdre, Davison, Suzanne, Turkova, Anna, Bamford, Alasdair, UCL - SSS/IREC/PEDI - Pôle de Pédiatrie, UCL - (SLuc) Service de gastro-entérologie et hépatologie pédiatrique, Indolfi, G., Bailey, H., Serranti, D., Giaquinto, C., Thorne, C., Sokal, E., Debray, D., Girard, M., Feiterna-Sperling, C., Wirth, S., Guidi, R., Verucchi, G., D'Antiga, L., Nicastro, E., Maggiore, G., Trapani, S., Ricci, S., Resti, M., Giacomet, V., Benincaso, A. R., Nebbia, G., Iorio, R., Cananzi, M., Riva, S., Bossi, G., Dodi, I., Nobili, V., Comparcola, D., Garazzino, S., Calvo, P. L., Pokorska-Spiewak, M., Pawlowska, M., Goncalves, C., Goncalves, I., Bals, M., Tudor, A. M., Noguera-Julian, A., Ramos, J. T., Fischler, B., Mclin, V., Brown, M., Kelly, D., Davison, S., Turkova, A., Bamford, A., Indolfi G., Bailey H., Serranti D., Giaquinto C., Thorne C., Sokal E., Debray D., Girard M., Feiterna-Sperling C., Wirth S., Guidi R., Verucchi G., D'Antiga L., Nicastro E., Maggiore G., Trapani S., Ricci S., Resti M., Giacomet V., Benincaso A.R., Nebbia G., Iorio R., Cananzi M., Riva S., Bossi G., Dodi I., Nobili V., Comparcola D., Garazzino S., Calvo P.L., Pokorska-Spiewak M., Pawlowska M., Goncalves C., Goncalves I., Bals M., Tudor A.M., Noguera-Julian A., Ramos J.T., Fischler B., McLin V., Brown M., Kelly D., Davison S., Turkova A., and Bamford A.

Subjects
Male, Pediatrics, Cirrhosis, Epidemiology, medicine.medical_treatment, Hepacivirus, Liver transplantation, medicine.disease_cause, Direct-acting antivirals, Liver disease, 0302 clinical medicine, 030212 general & internal medicine, Europe, direct-acting antivirals, epidemiology, treatment, vertical transmission, Child, education.field_of_study, treatment, Age Factors, Europe, Infectious Diseases, Child, Preschool, Vertical transmission, Female, 030211 gastroenterology & hepatology, epidemiology, medicine.medical_specialty, Adolescent, Genotype, Attitude of Health Personnel, Hepatitis C virus, Population, Socio-culturale, Antiviral Agents, 03 medical and health sciences, Chronic hepatitis, Hcv genotype 1, Virology, medicine, Humans, Pediatricians, education, direct-acting antivirals, direct-acting antiviral, Hepatology, business.industry, Infant, Newborn, Infant, Hepatitis C, Chronic, medicine.disease, Treatment, Cross-Sectional Studies, Health Care Surveys, vertical transmission, business
Abstract

The burden of paediatric HCV infection across Europe is unknown, as are current policies regarding monitoring and treatment. This collaborative study aimed to collect aggregate data to characterise the population of ≤18-year olds with HCV infection in specialist follow up in a twelve-month period (2016) across the PENTAHep European consortium, and investigate current policies around monitoring and treatment. A cross-sectional, web-based survey was distributed in April 2017 to 50 paediatricians in 19 European countries, covering patients' profile, and monitoring and treatment practices. Responses were received from 38/50 clinicians collectively caring for 663 children with chronic HCV infection of whom three-quarters were aged ≥6 years and 90% vertically-infected. HCV genotype 1 was the most common (n 380; 57.3%), followed by genotype 3, 4 and 2. Seventeen children (3%) with chronic HCV infection were diagnosed with cirrhosis and 6 were reported to have received liver transplantation for HCV-related liver disease. The majority (n 425; 64.1%) of the European children with HCV infection remained treatment-naive in 2016. Age affected clinicians' attitudes towards treatment; 94% reported being willing to use direct-acting antivirals, if available, in adolescents (aged ≥11 years), 78% in children aged 6-10 and 42% in those 3 to 5 years of age (Pearson correlation coefficient -0.98; p 0.0001). This survey provides the largest characterisation of the population of children in clinical follow-up for chronic HCV infection in Europe, alongside important contextual information on their management and treatment. Discussion is needed around strategies and criteria for use of direct-acting antivirals in these children. This article is protected by copyright. All rights reserved

Published
2019

49. Clinical prediction model for prognosis in kidney transplant recipients (KIDMO): study protocol

Author

Schwab, Simon, Sidler, Daniel, Haidar, Fadi, Kuhn, Christian, Schaub, Stefan, Koller, Michael, Mellac, Katell, Stürzinger, Ueli, Tischhauser, Bruno, Binet, Isabelle, Golshayan, Déla, Müller, Thomas, Elmer, Andreas, Franscini, Nicola, Krügel, Nathalie, Fehr, Thomas, Immer, Franz, Swisstransplant Kidney Working Group (STAN), Swiss Transplant Cohort Study, Amico, P., Folie, P., Gannagé, M., Matter, M., Nilsson, J., Peloso, A., de Rougemont, O., Schnyder, A., Spartà, G., Storni, F., Villard, J., Wirth-Müller, U., Wolff, T., Aubert, J.D., Banz, V., Beckmann, S., Beldi, G., Berger, C., Berishvili, E., Berzigotti, A., Bochud, P.Y., Branca, S., Bucher, H., Catana, E., Cairoli, A., Chalandon, Y., De Geest, S., De Seigneux, S., Dickenmann, M., Dreifuss, J.L., Duchosal, M., Ferrari-Lacraz, S., Garzoni, C., Goossens, N., Halter, J., Heim, D., Hess, C., Hillinger, S., Hirsch, H.H., Hirt, P., Hoessly, L., Hofbauer, G., Huynh-Do, U., Laesser, B., Lamoth, F., Lehmann, R., Leichtle, A., Manuel, O., Marti, H.P., Martinelli, M., McLin, V., Merçay, A., Mettler, K., Mueller, N.J., Müller-Arndt, U., Müllhaupt, B., Nägeli, M., Oldani, G., Pascual, M., Passweg, J., Pazeller, R., Posfay-Barbe, K., Rick, J., Rosselet, A., Rossi, S., Rothlin, S., Ruschitzka, F., Schachtner, T., Scherrer, A., Schuurmans, M., Sengstag, T., Simonetta, F., Stampf, S., Steiger, J., Stirnimann, G., Van Delden, C., Venetz, J.P., Vionnet, J., Wick, M., Wilhelm, M., and Yerly, P.

Subjects
Microbiology (medical), Immunology, Immunology and Allergy, 610 Medicine & health, Estimated glomerular filtration rate, Graft survival, Kidney transplantation, Patient-reported health status, Prediction model, Prognosis, Prognostic model, Quality of life, Risk calculator, Risk score, eGFR, 610 Medizin und Gesundheit
Abstract

Background Many potential prognostic factors for predicting kidney transplantation outcomes have been identified. However, in Switzerland, no widely accepted prognostic model or risk score for transplantation outcomes is being routinely used in clinical practice yet. We aim to develop three prediction models for the prognosis of graft survival, quality of life, and graft function following transplantation in Switzerland. Methods The clinical kidney prediction models (KIDMO) are developed with data from a national multi-center cohort study (Swiss Transplant Cohort Study; STCS) and the Swiss Organ Allocation System (SOAS). The primary outcome is the kidney graft survival (with death of recipient as competing risk); the secondary outcomes are the quality of life (patient-reported health status) at 12 months and estimated glomerular filtration rate (eGFR) slope. Organ donor, transplantation, and recipient-related clinical information will be used as predictors at the time of organ allocation. We will use a Fine & Gray subdistribution model and linear mixed-effects models for the primary and the two secondary outcomes, respectively. Model optimism, calibration, discrimination, and heterogeneity between transplant centres will be assessed using bootstrapping, internal-external cross-validation, and methods from meta-analysis. Discussion Thorough evaluation of the existing risk scores for the kidney graft survival or patient-reported outcomes has been lacking in the Swiss transplant setting. In order to be useful in clinical practice, a prognostic score needs to be valid, reliable, clinically relevant, and preferably integrated into the decision-making process to improve long-term patient outcomes and support informed decisions for clinicians and their patients. The state-of-the-art methodology by taking into account competing risks and variable selection using expert knowledge is applied to data from a nationwide prospective multi-center cohort study. Ideally, healthcare providers together with patients can predetermine the risk they are willing to accept from a deceased-donor kidney, with graft survival, quality of life, and graft function estimates available for their consideration. Study registration Open Science Framework ID: z6mvj

Published
2023

50. Donation type and the effect of pre-transplant donor specific antibodies – Data from the Swiss Transplant Cohort Study

Author

de Rougemont, Olivier, Deng, Yun, Frischknecht, Lukas, Wehmeier, Caroline, Villard, Jean, Ferrari-Lacraz, Sylvie, Golshayan, Déla, Gannagé, Monique, Binet, Isabelle, Wirthmüller, Urs, Sidler, Daniel, Schachtner, Thomas, Schaub, Stefan, Nilsson, Jakob, Swiss Transplant Cohort Study, Amico, P., Axel, A., Aubert, J.D., Banz, V., Sonja, B., Beldi, G., Berger, C., Berishvili, E., Binet, I., Bochud, P.Y., Branca, S., Bucher, H., Carrel, T., Catana, E., Chalandon, Y., De Geest, S., De Rougemont, O., Dickenmann, M., Dreifuss, J.L., Duchosal, M., Fehr, T., Ferrari-Lacraz, S., Franscini, N., Garzoni, C., Soccal, P.G., Gaudet, C., Golshayan, D., Goossens, N., Hadaya, K., Halter, J., Heim, D., Hess, C., Hillinger, S., Hirsch, H., Hirt, P., Hofbauer, G., Huynh-Do, U., Immer, F., Koller, M., Laager, M., Laesser, B., Lehmann, R., Leichtle, A., Lovis, C., Manuel, O., Marti, H.P., Martin, P.Y., Martinelli, M., McLin, V., Mellac, K., Mercay, A., Mettler, K., Mueller, N., Müller, A., Müller, T., Müller-Arndt, U., Müllhaupt, B., Nägeli, M., Oldani, G., Pascual, M., Posfay-Barbe, K., Rick, J., Rosselet, A., Rossi, S., Rothlin, S., Ruschitzka, F., Schanz, U., Schaub, S., Schnyder, A., Schuurmans, M., Sengstag, T., Simonetta, F., Staufer, K., Stampf, S., Steiger, J., Stirniman, G., Stürzinger, U., Van Delden, C., Venetz, J.P., Villard, J., Vionnet, J., Wick, M., Wilhlem, M., and Yerly, P.

Subjects
Humans, Antibodies, Blood Grouping and Crossmatching, Cohort Studies, Living Donors, Switzerland, ABMR, DBD, DCD, donor specific antibodies, graft loss, kidney transplantation, living donation, virtual cross-match, Immunology, Immunology and Allergy, 610 Medicine & health, 610 Medizin und Gesundheit
Abstract

IntroductionThe type of donation may affect how susceptible a donor kidney is to injury from pre-existing alloimmunity. Many centers are, therefore, reluctant to perform donor specific antibody (DSA) positive transplantations in the setting of donation after circulatory death (DCD). There are, however, no large studies comparing the impact of pre-transplant DSA stratified on donation type in a cohort with a complete virtual cross-match and long-term follow-up of transplant outcome.MethodsWe investigated the effect of pre-transplant DSA on the risk of rejection, graft loss, and the rate of eGFR decline in 1282 donation after brain death (DBD) transplants and compared it to 130 (DCD) and 803 living donor (LD) transplants.ResultsThere was a significant worse outcome associated with pre-transplant DSA in all of the studied donation types. DSA directed against Class II HLA antigens as well as a high cumulative mean fluorescent intensity (MFI) of the detected DSA showed the strongest association with worse transplant outcome. We could not detect a significant additive negative effect of DSA in DCD transplantations in our cohort. Conversely, DSA positive DCD transplants appeared to have a slightly better outcome, possibly in part due to the lower mean fluorescent intensity (MFI) of the pre-transplant DSA. Indeed when DCD transplants were compared to DBD transplants with similar MFI (DiscussionOur results suggest that the negative impact of pre-transplant DSA on graft outcome could be similar between all donation types. This suggests that immunological risk assessment could be performed in a similar way regardless of the type of donor kidney transplantation.

Published
2023

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