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1. Favorable course of leptospirosis and hantavirus-induced acute tubulointerstitial nephritis under corticosteroid treatment

Author

Matthys, Annelies, Dehoorne, Jo, Dendooven, Amélie, Schelstraete, Petra, and Prytula, Agnieszka

Subjects
Hantavirus infections -- Complications and side effects, Corticosteroids -- Dosage and administration -- Testing, Nephritis -- Causes of -- Demographic aspects -- Drug therapy, Leptospirosis -- Complications and side effects, Health
Abstract

Background We present two children with acute tubulointerstitial nephritis (ATIN) caused by leptospirosis in a 12-year-old boy and hantavirus in a 10-year-old girl. The role of glucocorticoids in the management of ATIN triggered by infectious agents is unclear. Case-diagnosis/treatment Both children were hospitalized with jaundice, elevated serum creatinine, and thrombocytopenia. There was no oliguria or hypertension. Urine analysis revealed tubular proteinuria. Kidney biopsy was performed on one patient and showed tubulointerstitial inflammation with mild mesangial proliferation. Both patients were treated with glucocorticoids in view of deteriorating kidney function with respective serum creatinine values of 5.2 and 4.1 mg/dl. Both children exhibited an excellent clinical and biochemical response to treatment. Neither of the patients required dialysis. Positive serology test results indicated a recent leptospirosis and hantavirus infection. Conclusions Leptospirosis and hantavirus associated ATIN share common clinical and biochemical features. Due to the low incidence in Europe these infectious causes of kidney dysfunction may be overlooked. Glucocorticoids may be considered in the management of ATIN., Author(s): Annelies Matthys [sup.1] [sup.2] , Jo Dehoorne [sup.1] [sup.2] , Amélie Dendooven [sup.3] , Petra Schelstraete [sup.2] [sup.4] , Agnieszka Prytula [sup.1] [sup.2] Author Affiliations: (1) https://ror.org/00xmkp704, grid.410566.0, 0000 [...]

Published
2023
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4. Comparison of SARS-CoV-2 seroconversion in children with chronic diseases with healthy children and adults during the first waves of the COVID-19 pandemic

Author

Hoste, Levi, primary, Prytula, Agnieszka, additional, Dehoorne, Jo, additional, De Bruyne, Ruth, additional, Van Biervliet, Stephanie, additional, De Waele, Kathleen, additional, Maes, Evelyn, additional, Bordon, Victoria, additional, Vanlander, Arnaud, additional, Claes, Karlien, additional, Vande Walle, Johan, additional, Schelstraete, Petra, additional, Van daele, Sabine, additional, and Haerynck, Filomeen, additional

Published
2023
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5. Standardized nailfold capillaroscopy in children with rheumatic diseases : a worldwide study

Author

Melsens, Karin, Cutolo, Maurizio, Schonenberg-Meinema, Dieneke, Foeldvari, Ivan, Leone, Maria C, Mostmans, Yora, Badot, Valérie, Cimaz, Rolando, Dehoorne, Jo, Deschepper, Ellen, Frech, Tracy, Hernandez-Zapata, Johanna, Ingegnoli, Francesca, Khan, Archana, Krasowska, Dorota, Lehmann, Hartwig, Makol, Ashima, Mesa-Navas, Miguel A, Michalska-Jakubus, Malgorzata, Müller-Ladner, Ulf, Nuño-Nuño, Laura, Overbury, Rebecca, Pizzorni, Carmen, Radic, Mislav, Ramadoss, Divya, Ravelli, Angelo, Rosina, Silvia, Udaondo, Clara, van den Berg, Merlijn J, Herrick, Ariane L, Sulli, Alberto, Smith, Vanessa, Deroo, Liselotte, Vanhaecke, Amber, Bergkamp, Sandy, Nassar-Sheikh-Rashid, Amara, Elisa, Patrone, Hermann, Walter, Bica, Francesco, Cattelan, Francesco, Gattinara, Maurizio, Özcan, Zeynep, Murias, Sara, Alcobendas, Rosa, Remesal, Augustin, Pimpale, Pallavi, Khubchandani, Raju, and for the EULAR Study Group on Microcirculation in Rheumatic Diseases, [missing]

Subjects
Rheumatology, children, Medicine and Health Sciences, microcirculation, nailfold capillaroscopy, Pharmacology (medical), juvenile rheumatic and musculoskeletal diseases, scleroderma pattern
Abstract

Objectives: To standardly assess and describe nailfold videocapillaroscopy (NVC) assessment in children and adolescents with juvenile rheumatic and musculoskeletal diseases (jRMD) vs healthy controls (HCs). Material and methods: In consecutive jRMD children and matched HCs from 13 centres worldwide, 16 NVC images per patient were acquired locally and read centrally per international consensus standard evaluation of the EULAR Study Group on Microcirculation in Rheumatic Diseases. A total of 95 patients with JIA, 22 with JDM, 20 with childhood-onset SLE (cSLE), 13 with juvenile SSc (jSSc), 21 with localized scleroderma (lSc), 18 with MCTD and 20 with primary RP (PRP) were included. NVC differences between juvenile subgroups and HCs were calculated through multivariable regression analysis. Results: A total of 6474 images were assessed from 413 subjects (mean age 12.1 years, 70.9% female). The quantitative NVC characteristics were significantly lower or higher in the following subgroups compared with HCs: for density: lower in jSSc, JDM, MCTD, cSLE and lSc; for dilations: higher in jSSc, MCTD and JDM; for abnormal shapes: higher in JDM and MCTD; for haemorrhages: higher in jSSc, MCTD, JDM and cSLE. The qualitative NVC assessment of JIA, lSc and PRP did not differ from HCs, whereas the cSLE and jSSc, MCTD, JDM and cSLE subgroups showed more non-specific and scleroderma patterns, respectively. Conclusions: This analysis resulted from a pioneering registry of NVC in jRMD. The NVC assessment in jRMD differed significantly from HCs. Future prospective follow-up will further elucidate the role of NVC in jRMD.

Published
2023

7. Whole blood tcr vβ21.3 staining as a diagnostic test for multisystem inflammatory syndrome in children : a proof-of-concept study

Author

Hoste, Levi, Willems, Jef, Dhont, Evelyn, Schelstraete, Petra, Dehoorne, Jo, Vandekerckhove, Kristof, Belot, Alexandre, Bader-Meunier, Brigitte, Malcus, Christophe, Tavernier, Simon, and Haerynck, Filomeen

Subjects
Medicine and Health Sciences
Abstract

Background and Aims Multisystem inflammatory syndrome in children (MIS-C) is a rare inflammatory condition, occurring 1-2 months after SARS-CoV-2 infection in children. The clinical features of MIS-C are nonspecific. Since early treatment is of prognostic value, clinicians are faced with the challenge to recognize patients timely. However, lacking a specific test, diagnosing MIS-C remains largely based on clinical expertise. Enrichment of Vβ21.3+ T-cell receptors is a frequent finding in MIS-C. We aimed to assess the value of whole blood (WB) Vβ21.3 staining as a diagnostic test for MIS-C. Methods EDTA blood was obtained in healthy controls (HCs) and patients fulfilling the WHO MIS-C case definition (before immunomodulatory treatment). WB was stained, lysed and fixed within 2h of sampling. By flow cytometry, the relative frequency of Vβ21.3+ T-cells (CD3/CD4/CD8) was assessed as well as activation markers (HLA-DR). Results Four patients and five HCs were included. The Vβ21.3+ T-cell proportions were increased in one case (3.6-5.6% of T-cells), a 12yo girl presenting typical MIS-C and responding rapidly with first-line treatment. Although all other patients fulfilled the MIS-C case definition at inclusion, their further disease course led to alternative diagnoses, including Streptococcus pyogenes sepsis, haemophagocytic lymphohistiocytosis and metabolic cardiomyopathy. These non-MIS-C patients and HCs showed only 1.0-2.6% Vβ21.3+ T-cells. Additionally, in MIS-C 37.7% of Vβ21.3+ lymphocytes were HLA-DR+, while only 0.1-11% in HCs and non-MIS-C patients. Conclusions We provide proof-of-concept that Vβ21.3 staining can be used as a rapid diagnostic test to distinguish MIS-C from other inflammatory diseases. Its sensitivity and specificity should be assessed in larger prospective studies.

Published
2022

12. A human immune dysregulation syndrome characterized by severe hyperinflammation with a homozygous nonsense Roquin-1 mutation

Author

Tavernier, Simon J, Athanasopoulos, Vicki, Verloo, Patrick, Behrens, Gesine, Staal, Jens, Bogaert, Delfien, Naesens, L, de Bruyne, Marieke, van Gassen, Sofie, Parthoens, Eef, Ellyard, Julia, Cappello, Jean, Morris, Lucy, van Gorp, Hanne, van Isterdael, Gert, Saeys, Y, Lamkanfi, Mohamed, Schelstraete, Petra, Dehoorne, Jo, Bordon, Victoria, Van Coster, Rudy, Lambrecht, Bart, Menten, Bjorn, Beyaert, Rudi, Vinuesa, Carola, Heissmeyer, Vigo, Dullaers, Melissa, Haerynck, Filomeen, Tavernier, Simon J, Athanasopoulos, Vicki, Verloo, Patrick, Behrens, Gesine, Staal, Jens, Bogaert, Delfien, Naesens, L, de Bruyne, Marieke, van Gassen, Sofie, Parthoens, Eef, Ellyard, Julia, Cappello, Jean, Morris, Lucy, van Gorp, Hanne, van Isterdael, Gert, Saeys, Y, Lamkanfi, Mohamed, Schelstraete, Petra, Dehoorne, Jo, Bordon, Victoria, Van Coster, Rudy, Lambrecht, Bart, Menten, Bjorn, Beyaert, Rudi, Vinuesa, Carola, Heissmeyer, Vigo, Dullaers, Melissa, and Haerynck, Filomeen

Abstract

Hyperinflammatory syndromes are life-threatening disorders caused by overzealous immune cell activation and cytokine release, often resulting from defects in negative feedback mechanisms. In the quintessential hyperinflammatory syndrome familial hemophagocytic lymphohistiocytosis (HLH), inborn errors of cytotoxicity result in effector cell accumulation, immune dysregulation and, if untreated, tissue damage and death. Here, we describe a human case with a homozygous nonsense R688* RC3H1 mutation suffering from hyperinflammation, presenting as relapsing HLH. RC3H1 encodes Roquin-1, a posttranscriptional repressor of immune-regulatory proteins such as ICOS, OX40 and TNF. Comparing the R688* variant with the murine M199R variant reveals a phenotypic resemblance, both in immune cell activation, hypercytokinemia and disease development. Mechanistically, R688* Roquin-1 fails to localize to P-bodies and interact with the CCR4-NOT deadenylation complex, impeding mRNA decay and dysregulating cytokine production. The results from this unique case suggest that impaired Roquin-1 function provokes hyperinflammation by a failure to quench immune activation.

Published
2019

13. Resilience factors in children with juvenile idiopathic arthritis and their parents : the role of child and parent psychological flexibility

Author

Beeckman, Melanie, Hughes, Sean Joseph, van Ryckeghem, Dimitri, Van Hoecke, Eline, Dehoorne, Jo, Joos, Rik, Goubert, Liesbet, Beeckman, Melanie, Hughes, Sean Joseph, van Ryckeghem, Dimitri, Van Hoecke, Eline, Dehoorne, Jo, Joos, Rik, and Goubert, Liesbet

Abstract

Objective. Chronic pain is central to juvenile idiopathic arthritis (JIA) and is predictive of impaired functioning. Whereas most work has focused on identifying psychosocial risk factors for maladaptive outcomes, we explored the idea that child and parental psychological flexibility (PF) represent resilience factors for adaptive functioning of the child. We also explored differences between general vs pain-specific PF in contributing to child outcomes. Methods. Children with JIA (age eight to 18 years) and (one of) their parents were recruited at the Department of Pediatric Rheumatology at the Ghent University Hospital in Belgium. They completed questionnaires assessing child and parent general and pain-specific PF and child psychosocial and emotional functioning and disability. Results. The final sample consisted of 59 children and 48 parents. Multiple regression analyses revealed that child PF contributed to better psychosocial functioning and less negative affect. Child pain acceptance contributed to better psychosocial functioning, lower levels of disability, and lower negative affect, and also buffered the negative influence of pain intensity on disability. Bootstrap mediation analyses demonstrated that parental (general) PF indirectly contributed to child psychosocial functioning and affect via the child's (general) PF. Parent pain-specific PF was indirectly linked to child psychosocial functioning, disability, and negative affect via child pain acceptance. Conclusions. Our findings indicate that child and parental PF are resilience factors and that pain acceptance buffers the negative impact of pain intensity. Implications for psychosocial interventions that target (pain-specific) PF in children and parents are discussed.

Published
2019
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14. Pediatric lupus nephritis presenting with terminal renal failure

Author

Besouw, Martine T. P., Walle, Johan G. Vande, Ilias, Mohamad I., Raes, Ann M., Prytula, Agnieszka A., Claeys, Lieve, Dehoorne, Jo L., and Faculteit Medische Wetenschappen/UMCG

Subjects
Acute renal failure, Systemic lupus erythematosus, Terminal renal failure, Lupus nephritis, Differential diagnosis, Dialysis
Abstract

A 12-year-old Congolese girl presented with acute renal failure, edema, hypertension, hemoptysis, hematuria, and proteinuria after a history of throat infection. Renal ultrasound showed kidneys of normal size, with increased echogenicity of the cortical parenchyma and decreased corticomedullary differentiation. Other additional investigations showed pancytopenia with decreased complement (low C3 and C4). Antinuclear antibodies were strongly positive, including anti-double stranded DNA. Renal biopsy confirmed severe grade IV lupus nephritis. She was treated with high-dose steroids, mycophenolate mofetil and hydroxychloroquine, in addition to hemodialysis. After oneweek of intensive treatment, diuresis recovered and dialysis could be stopped after six sessions. We describe an uncommon case of severe lupus nephritis, presenting with terminal renal failure. Since the rarity of this disease presentation, other more common diagnoses have to be considered. Once the diagnosis of lupus nephritis is established, a choice has to be made between the different induction treatment protocols. The patient's ethnic background and other supportive therapies, such as the need for dialysis, can help to make this choice.

Published
2016

27. Factors associated with 1,25-dihydroxyvitamin D3concentrations in liver transplant recipients: a prospective observational longitudinal study

Author

Prytuła, Agnieszka, Walle, Johan, Vlierberghe, Hans, Kaufman, Jean-Marc, Fiers, Tom, Dehoorne, Jo, and Raes, Ann

Abstract

The aim of the study was to identify factors associated with 1,25(OH)2D3concentrations in liver transplant recipients with emphasis on the renal function and catabolism. We also tested the hypothesis that tacrolimus increases 1,25(OH)2D3concentrations. Serum 25(OH)D3, 1,25(OH)2D3, and 24,25(OH)2D3were measured in 41 patients before, at 2 weeks and 3 months after transplantation. Dose-adjusted tacrolimus concentration was used as a surrogate marker of CYP3A4activity. Factors associated with 1,25(OH)2D3were identified using multivariate linear regression analysis. The median 1,25(OH)2D3levels remained stable: 55 versus 46 pg/ml (P= 0.36) despite an increase in 25(OH)D3from 18 ng/ml at baseline to 26 ng/ml (P= 0.03), serum albumin (34 to 41 g/l, P= 0.02), and comparable eGFR at baseline and month 3 (94 and 92 ml/min, respectively, P= 0.15). At 3 months 19 % of patients had 1,25(OH)2D3< 25 pg/ml. Low eGFR and a low dose-adjusted tacrolimus concentration were both independently associated with 1,25(OH)2D3at 3 months. Liver transplant recipients with impaired renal function or a low dose-adjusted tacrolimus concentration suggesting a high CYP3A4are at risk of low 1,25(OH)2D3concentrations. The use of tacrolimus does not lead to an increase in 1,25(OH)2D3concentrations in a clinical setting.

Published
2016
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28. Pharmacokinetics of desmopressin administered as tablet and oral lyophilisate formulation in children with monosymptomatic nocturnal enuresis.

Author

Bruyne, Pauline, Guchtenaere, Ann, Herzeele, Charlotte, Raes, Ann, Dehoorne, Jo, Hoebeke, Piet, Laecke, Erik, and Walle, Johan

Subjects
TREATMENT of enuresis, DESMOPRESSIN, PHARMACOKINETICS, SYMPTOMS, ORAL medication, DIETARY supplements
Abstract

Desmopressin 120 μg oral lyophilisate and 200 μg tablet are considered bioequivalent, based on extrapolation of studies in a limited number of adults and on one dose-finding study of desmopressin oral lyophilisate in children. However, no comparative pharmacokinetic study in children was executed confirming this statement. No data are available on the influence of food intake on the bioavailability of desmopressin tablet in a pediatric setting, although studies in adults have documented that food intake results in a significantly lower desmopressin plasma concentration. In this study, we analyzed plasma concentrations of desmopressin oral lyophilisate and tablet with concomitant food intake. Twenty-three children with monosymptomatic nocturnal enuresis (mean age, 12.7 years) were recruited. Two tests were performed on two separate days in identical conditions with a standardized food and fluid intake. Desmopressin was administered as desmopressin tablet or desmopressin oral lyophilisate immediately after a meal. Desmopressin plasma concentration was measured at 1 h, 2 h, and 6 h postdosing. No significant difference in plasma concentration of 120 μg desmopressin oral lyophilisate and 200 μg tablet was demonstrated, even with concomitant food intake. A significant difference in variability was found, identifying a smaller variance for desmopressin oral lyophilisate plasma concentrations at all time points. This study demonstrates comparable plasma levels for desmopressin oral lyophilisate, despite the lower dose. The dosage for desmopressin oral lyophilisate is more predictable due to the significantly smaller variance. Therefore, desmopressin oral lyophilisate seems more suitable, especially in the younger age group for which time interval between dinner and drug administration is limited. [ABSTRACT FROM AUTHOR]

Published
2014
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29. Immunoglobulin, glucocorticoid, or combination therapy for multisystem inflammatory syndrome in children: a propensity-weighted cohort study

Author

Channon-Wells, Samuel, Vito, Ortensia, McArdle, Andrew J, Seaby, Eleanor G, Patel, Harsita, Shah, Priyen, Pazukhina, Ekaterina, Wilson, Clare, Broderick, Claire, D'Souza, Giselle, Keren, Ilana, Nijman, Ruud G, Tremoulet, Adriana, Munblit, Daniel, Ulloa-Gutierrez, Rolando, Carter, Michael J, Ramnarayan, Padmanabhan, De, Tisham, Hoggart, Clive, Whittaker, Elizabeth, Herberg, Jethro A, Kaforou, Myrsini, Cunnington, Aubrey J, Blyuss, Oleg, Levin, Michael, Chouli, Mohamed, Hamadouche, Nacera, Ladj, Mohamed Samir, Agrimbau Vázquez, Jorge, Carmona, Rodrigo, Collia, Adrian Gustavo, Ellis, Alejandro, Natta, Diego, Pérez, Laura, Rubiños, Mayra, Veliz, Natalia, Yori, Silvana, Britton, Philip N., Burgner, David P., Carey, Emma, Crawford, Nigel W., Giuliano, Hayley, McMinn, Alissa, Wong, Shirley, Wood, Nicholas, Holter, Wolfgang, Krainz, Matthias, Ulreich, Raphael, Zurl, Christoph, Dehoorne, Jo, Haerynck, Filomeen, Hoste, Levi, Schelstraete, Petra, Vandekerckhove, Kristof, Willems, Jef, Almeida Farias, Camila Giuliana, Almeida, Flávia Jacqueline, Alves Leal, Izabel, Araujo da Silva, André Ricardo, Araujo e Silva, Anna Esther, Barreiro, Sabrina T.A., Bomfim Prado da Silva, Daniella Gregória, Cervi, Maria Celia, dos Santos Naja Cardoso, Mirian Viviane, Henriques Teixeira, Cristiane, Jarovsky, Daniel, Martins Araujo, Julienne, Naaman Berezin, Eitan, Palazzi Sáfadi, Marco Aurélio, Paternina-de la Ossa, Rolando Andres, Souza Vieira, Cristina, Dimitrova, Anna, Ganeva, Margarita, Stefanov, Stefan, Telcharova-Mihaylovska, Albena, Biggs, Catherine M., Lopez, Alison, Scuccimarri, Rosie, Tan, Ryan, Wasserman, Sam, Withington, Davinia, Ampuero, Camila, Aravena, Javiera, Bustos B, Raul, Casanova, Daniel, Cruces, Pablo, Diaz, Franco, García-Salum, Tamara, Godoy, Loreto, Medina, Rafael A., Valenzuela Galaz, Gonzalo, Camacho-Moreno, Germán, Avila-Aguero, María L., Brenes-Chacón, Helena, Camacho-Badilla, Kattia, Ivankovich-Escoto, Gabriela, Naranjo-Zuniga, Gabriela, Soriano-Fallas, Alejandra, Yock-Corrales, Adriana, Amer, Maysa Abbas, Abdelmeguid, Yasmine, Ahmed, Yomna H.H.Z., Badib, Adham, Badreldin, Karim, Elkhashab, Yara, Heshmat, Hassan, Hussein, Amna, Mohamed Hussein, Amna Hussein, Ibrahim, Sandra, Shoman, Walaa, Yakout, Radwa M, Heinonen, Santtu, Angoulvant, François, Belot, Alexandre, Ouldali, Naïm, Beske, Florian, Heep, Axel, Masjosthusmann, Katja, Reiter, Karl, van den Heuvel, Ingeborg, von Both, Ulrich, Agrafiotou, Aikaterini, Antachopoulos, Charalampos, Charisi, Konstantina, Eleftheriou, Irini, Farmaki, Evangelia, Fotis, Lampros, Kafetzis, Dimitrios, Koletsi, Patra, Kourtesi, Katerina, Lampidi, Stavroula, Liakopoulou, Theodota, Maritsi, Despoina, Michailidou, Elisa, Milioudi, Maria, Mparmpounaki, Ioanna, Papadimitriou, Eleni, Papaevangelou, Vassiliki, Roilides, Emmanuel, Tsiatsiou, Olga, Tsolas, Georgios, Tsolia, Maria, Vantsi, Petrina, Banegas Pineda, Linda Yajeira, Borjas Aguilar, Karla Leversia, Cantillano Quintero, Edwin Mauricio, Ip, Patrick, Kwan, Mike Yat Wah, Kwok, Janette, Lau, Yu Lung, To, Kelvin, Wong, Joshua Sung Chih, David, Mate, Farkas, David, Kalcakosz, Szofia, Szekeres, Klaudia, Zsigmond, Borbala, Aslam, Nadeem, Luder, Anthony, Andreozzi, Laura, Bianco, Francesco, Bucciarelli, Valentina, Buonsenso, Danilo, Cimaz, Rolando, De Luca, Maia, Dellepiane, Rosa Maria, Fabi, Marianna, Filice, Emanuele, Lanari, Marcello, Lo Vecchio, Andrea, Mastrolia, Maria Vincenza, Mauro, Angela, Mazza, Angelo, Papa, Mario Virgilio, Romani, Lorenza, Scarano, Sara Maria, Simonini, Gabriele, Tipo, Vincenzo, Verdoni, Lucio, Macharia, Anne-Marie, Musiime, Grace, Reel, Bhupi, Wangai, Frederick, Pace, David, Torpiano, Paul, Anaya-Enriquez, Nancy, Carreon-Guerrero, Juan Manuel, Chacon-Cruz, Enrique, Cheung López, Mariana, Faugier Fuentes, Enrique, Fonseca Flores, Marisol, García-Domínguez, Miguel, Giron Vargas, Ana Luisa, Lopez-Delgado, Ivan, Lopez Hernández, Liliana, Menchaca Aguayo, Hector F., Montaño-Duron, Jesus Gilberto, Pérez-Gaxiola, Giordano, Ramos Tiñini, Pamela, Tostado-Morales, Edgardo, Valadez, Julio, Inchley, Christopher, Klevberg, Sjur, Knudsen, Per Kristian, Måseide, Per Helge, Carrera, Jose Manuel, Castaño, Elizabeth, Daza Timana, Carlos Alberto, De Leon, Tirza, Estripeaut, Dora, Levy, Jacqueline, Norero, Ximena, Record, Javier, Rojas-Bonilla, Magda, Wong, Mayra, Iramain, Ricardo, Hernandez, Roger, Huamán, Gian, Munaico, Manuel, Peralta, Carlos, Seminario, Diego, Zapata Yarlequé, Elmer Hans, Gadzinska, Justyna, Ludwikowska, Kamila, Mandziuk, Joanna, Okarska-Napierała, Magdalena, Alacheva, Zalina A., Alexeeva, Ekaterina, Ananin, Petr V., Antsupova, Margarita, Bakradze, Maya D., Berbenyuk, Anna, Bobkova, Polina, Borzakova, Svetlana, Chashchina, Irina L., El-Taravi, Yasmin, Fisenko, Andrey P., Gautier, Marina S., Glazyrina, Anastasia, Gorlenko, Cyrill, Grosheva, Mariia, Kiselev, Herman, Kondrikova, Elena, Korobyants, Evgeniya, Korsunskiy, Anatoliy A., Kovygina, Karina, Krasnaya, Ekaterina, Kurbanova, Seda, Kurdup, Maria K., Mamutova, Anna V., Mazankova, Lyudmila, Mitushin, Ilya L., Nargizyan, Anzhelika, Orlova, Yanina O., Osmanov, Ismail M., Polyakova, Anastasia S., Pushkareva, Anna, Romanova, Olga, Samitova, Elmira, Shvedova, Anastasia, Sologub, Anna, Iakovleva, Ekaterina, Tepaev, Rustem F., Tkacheva, Anna A., Yegiyan, Margarita, Yusupova, Valeriya, Zholobova, Elena, Grasa, Carlos Daniel, Epalza, Cristina, Lopez Segura, Nuria, Martinon-Torres, Federico, Melendo, Susana, Mendez-Echevarria, Ana, Mesa Guzmán, Juan Miguel, Palacios Argueta, Jorge Roberto, Rivero-Calle, Irene, Rivière, Jacques, Rodríguez-González, Moisés, Rojo, Pablo, Sanchez Manubens, Judith, Soler-Palacin, Pere, Soriano-Arandes, Antoni, Tagarro, Alfredo, Villaverde, Serena, Altman, Maria, Brodin, Petter, Horne, AnnaCarin, Palmblad, Karin, Brotschi, Barbara, Meyer Sauteur, Patrick, Pachlopnik Schmid, Jana, Prader, Seraina, Relly, Christa, Schlapbach, Luregn J., Seiler, Michelle, Strasser, Sophie, Trück, Johannes, Weber, Kathrin, Wütz, Daniela, Hamdan, Alaa, Melhem, Ibrahim, Moussa, Ahmed, Dunk, Joke, Ketharanathan, Naomi, Vermont, Clementien, Akyüz Özkan, Esra, Cetin, Benhur Sirvan, Erdeniz, Emine Hafize, Şahin, Irfan Oğuz, Borisova, Galina, Boyarchuk, Oksana, Boychenko, Lidiya, Boyko, Yaryna, Diudenko, Nadiia, Dyvonyak, Olha, Kasiyan, Olexandr, Katerynych, Kostiantyn, Kostyuchenko, Larysa, Mamenko, Marina, Melnyk, Kateryna, Miagka, Nelia, Nazarenko, Liliya, Nezgoda, Iryna, Rykova, Stanislava, Svyst, Olga, Teslenko, Maria, Trykosh, Mykola, Vasilenko, Nataliya, Volokha, Alla, Adams, Charlotte, Akomolafe, Toju, Al-Abadi, Eslam, Alders, Nele, Alifieraki, Styliani, Ansumanu, Hareef, Aston, Emily, Avram, Paula, Bamford, Alasdair, Banks, Millie, Basu Roy, Robin, Beattie, Thomas, Boleti, Olga, Bracken, Abbey, Broad, Jonathan, Cai, James, Carrol, Enitan D., Carter, Michael, Chandran, Anchit, Charlesworth, James, Chawla, Jaya, Cooper, Hannah, Cooray, Samantha, Davies, Patrick, Davis, Francesca, Drysdale, Simon B., Dzora, Ella, Emonts, Marieke, Evans, Ceri, Fidler, Katy, Foster, Caroline, Gong, Chen, Gongrun, Berin, Gonzalez, Carmen, Gorgun, Berin, Grandjean, Louis, Grant, Karlie, Guo, Jonathan, Hacohen, Yael, Hall, Jack, Hamid, Hytham K.S., Hassell, Jane, Hesketh, Christine, Hewlett, Jessica, Hnieno, Ahmad, Holt-Davis, Hannah, Hossain, Aleena, Hu, Shiying, Hudson, Lee D., Jheeta, Sharon, Johnson, Mae, Johnson, Sarah, Jyothish, Deepthi, Kampmann, Beate, Kavirayani, Akhila, Kelly, Deborah, Kirubakaran, Arangan, Kucera, Filip, Langer, Daniel, Lawson, George, Lees, Emily A, Lenihan, Rebecca, Lillie, Jon, Longbottom, Katherine, Lyall, Hermione, Mackdermott, Niamh, Maltby, Sarah, Mclelland, Thomas, McMahon, Anne-Marie, Miller, Danielle, Miranda, Mariana, Mirza, Luwaiza, Morrison, Zoe, Moshal, Karyn, Muller, Jennifer, Musuka, Phoebe, Myttaraki, Evangelia, Nadel, Simon, Nair, Sreedevi, Nuttall, Luke, Oremakinde, Oyinkansola, Osaghae, Daniella, Osman, Fatima, Ostrzewska, Anna, Paccagnella, Davide, Panthula, Mrinalini, Papachatzi, Eleni, Papadopoulou, Charalampia, Patel, Fahim, Payne, Helen, Penner, Justin, Polandi, Shervin, Prendergast, Andrew J., Ranasinghe, Lasith, Ravichandran, Muthukumaran, Rhys-Evans, Sophie, Riordan, Andrew, Rodrigues, Charlene M.C., Roe, Lauren, Romaine, Sam, Schobi, Nina, Seddon, James, Shingadia, Delane, Sikdar, Oishi, Srivastava, Anand, Struik, Siske, Sun, Thomas, Tan, Rachel Wei, Taylor, Alice, Taylor, Amanda, Taylor, Andrew, Tran, Steven, Tsagkaris, Stavros, Tudor-Williams, Gareth, van den Berg, Sarah, van der Velden, Fabian, Ventilacion, Lyn, Wellman, Paul A., Withers Green, Joseph, Yanney, Michael P., Yeung, Shunmay, Badheka, Aditya, Badran, Sarah, Bailey, Dwight M., Burch, Anna Kathryn, Burns, Jane C., Cichon, Catherine, Cirks, Blake, Dallman, Michael D., Delany, Dennis R., Fairchok, Mary, Friedman, Samantha, Geracht, Jennifer, Langs-Barlow, Allison, Mann, Kelly, Padhye, Amruta, Quade, Alexis, Ramirez, Kacy Alyne, Rockett, John, Sayed, Imran Ali, Santos, Roberto P., Shahin, Amr A., Umaru, Samuel, Widener, Rebecca, Mujuru, Hilda Angela, Kandawasvika, Gwendoline, and Best Available Treatment Study (BATS) consortium, on behalf of the

Subjects
Rheumatology, Immunology, Medicine and Health Sciences, Immunology and Allergy
Abstract

Background Multisystem inflammatory syndrome in children (MIS-C), a hyperinflammatory condition associated with SARS-CoV-2 infection, has emerged as a serious illness in children worldwide. Immunoglobulin or glucocorticoids, or both, are currently recommended treatments. Methods The Best Available Treatment Study evaluated immunomodulatory treatments for MIS-C in an international observational cohort. Analysis of the first 614 patients was previously reported. In this propensity-weighted cohort study, clinical and outcome data from children with suspected or proven MIS-C were collected onto a web-based Research Electronic Data Capture database. After excluding neonates and incomplete or duplicate records, inverse probability weighting was used to compare primary treatments with intravenous immunoglobulin, intravenous immunoglobulin plus glucocorticoids, or glucocorticoids alone, using intravenous immunoglobulin as the reference treatment. Primary outcomes were a composite of inotropic or ventilator support from the second day after treatment initiation, or death, and time to improvement on an ordinal clinical severity scale. Secondary outcomes included treatment escalation, clinical deterioration, fever, and coronary artery aneurysm occurrence and resolution. This study is registered with the ISRCTN registry, ISRCTN69546370. Findings We enrolled 2101 children (aged 0 months to 19 years) with clinically diagnosed MIS-C from 39 countries between June 14, 2020, and April 25, 2022, and, following exclusions, 2009 patients were included for analysis (median age 8·0 years [IQR 4·2–11·4], 1191 [59·3%] male and 818 [40·7%] female, and 825 [41·1%] White). 680 (33·8%) patients received primary treatment with intravenous immunoglobulin, 698 (34·7%) with intravenous immunoglobulin plus glucocorticoids, 487 (24·2%) with glucocorticoids alone; 59 (2·9%) patients received other combinations, including biologicals, and 85 (4·2%) patients received no immunomodulators. There were no significant differences between treatments for primary outcomes for the 1586 patients with complete baseline and outcome data that were considered for primary analysis. Adjusted odds ratios for ventilation, inotropic support, or death were 1·09 (95% CI 0·75–1·58; corrected p value=1·00) for intravenous immunoglobulin plus glucocorticoids and 0·93 (0·58–1·47; corrected p value=1·00) for glucocorticoids alone, versus intravenous immunoglobulin alone. Adjusted average hazard ratios for time to improvement were 1·04 (95% CI 0·91–1·20; corrected p value=1·00) for intravenous immunoglobulin plus glucocorticoids, and 0·84 (0·70–1·00; corrected p value=0·22) for glucocorticoids alone, versus intravenous immunoglobulin alone. Treatment escalation was less frequent for intravenous immunoglobulin plus glucocorticoids (OR 0·15 [95% CI 0·11–0·20]; p

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30. Acute renal failure after vomiting in a teenager

Author

Sevasti Karamaria, Ann Raes, Dehoorne, Jo, Agnieszka Prytula, and Vande Walle

Subjects
AKI, ERKNET, Medicine and Health Sciences
Abstract

We present the case of a 12 year old boy who presented to the emergency department because of vomiting and abdominal pain. Initial workup revealed risen creatinine (1,7mg/dl), hyponatriemia (131mmol/l), hypokaliemia (3,7mmol/l), hypochloremia (77mmol/l) and hyperphosphatemia (10,2mg/dl). Furthermore we observed increased corticomedullary differentiation echographically. Due to the atypical presentaion we decided to perform a renal biopsy which was suggestive for the presence of oxalate crystals in the lumina of some tubules. However a 24hour urine collection was normal for oxalate excretion and genetic tests were negative for the most common hyperoxaluria mutations (AGXT, GRHPR). Still the GFR was only partially recovered in the upcoming weeks. The patient was regurarly followed up at our outpatient clinic. We noted failure to thrive, several episodes of vomiting and dehydration paired with transient elevated creatinine. The boy was therefore diagnosed with secondary hyperoxaluria due to dehydration.

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