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4. Outcomes and management in paediatric autoimmune hepatitis presenting as acute liver failure: Individual patient data meta‐analysis.

Author

Fawzy, Aly, Sutton, Harry, Vandriel, Shannon M., Sonnenberg, Mikayla, and Kamath, Binita M.

Subjects
AUTOIMMUNE hepatitis, LIVER failure, PEDIATRICS, LIVER transplantation, DATABASES
Abstract

Background and Aims: Autoimmune hepatitis (AIH) in children presenting in acute liver failure (ALF) can be fatal and often requires liver transplantation (LTx). This individual patient data meta‐analysis (IPD) aims to examine management and outcomes of this population, given the lack of large cohort studies on paediatric AIH first presenting as ALF (AIH‐ALF). Methods: A systematic review was conducted in accordance with the Preferred Reporting Items for Systematic Review and Meta‐Analyses of IPD statement using PubMed and Excerpta Medica dataBASE, and included English studies published between 2000 and 2020. The study included patients under 21 years of age, diagnosed with type 1 or 2 AIH and presenting with ALF. Data extracted included clinical and biochemical characteristics, interventions, and outcomes. Results: Three hundred and thirty eligible patients from 61 studies were identified, with an additional five patients from our institution. The majority were female (66.8%), with a median age of 10. Overall, 59.7% achieved native liver survival (NLS), 35% underwent LTx, and 5% died before LTx. The use of corticosteroids with non‐steroid immunomodulators increased the likelihood of NLS by 2.5‐fold compared to corticosteroids alone. AIH‐1 was associated with 3.3‐fold odds for NLS, compared to AIH‐2. However, on multivariate analysis, only AIH‐1 was identified as an independent predictor for NLS (OR 3.8 [95% CI 1.03–14.2], p =.04). Conclusion: While corticosteroids and non‐steroid immunomodulators treatment may offer enhanced probability of achieving NLS, treatment regimens for AIH‐ALF may need to consider patient‐specific factors, especially AIH type. This highlights the potential for NLS in AIH‐ALF and suggest a need to identify biomarkers which predict the need for combination immunosuppression to avoid LTx. [ABSTRACT FROM AUTHOR]

Published
2024
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6. Event-free survival of maralixibat-treated patients with Alagille syndrome compared to a real-world cohort from GALA.

Author

Hansen, Bettina E., Vandriel, Shannon M., Vig, Pamela, Garner, Will, Mogul, Douglas B., Loomes, Kathleen M., Piccoli, David A., Rand, Elizabeth B., Jankowska, Irena, Czubkowski, Piotr, Gliwicz-Miedzińska, Dorota, Gonzales, Emmanuel M., Jacquemin, Emmanuel, Bouligand, Jérôme, D'Antiga, Lorenzo, Nicastro, Emanuele, Arnell, Henrik, Fischler, Björn, Sokal, Étienne, and Demaret, Tanguy

Published
2024
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8. Serum bile acids are associated with native liver survival in patients with Alagille syndrome: results from the GALA study group

Author

Murillo Perez, Carla Fiorella, primary, Vandriel, Shannon M., additional, Wang, Jian-She, additional, Liting, Li, additional, She, Huiyu, additional, Jankowska, Irena, additional, Czubkowski, Piotr, additional, Gliwicz, Dorota, additional, Gonzalès, Emmanuel, additional, Jacquemin, Emmanuel, additional, Bouligand, Jérôme, additional, D’Antiga, Lorenzo, additional, Nicastro, Emanuele, additional, Fischler, Björn, additional, Arnell, Henrik, additional, Siew, Susan, additional, Stormon, Michael, additional, Loomes, Kathleen M., additional, Piccoli, David A., additional, Rand, Elizabeth B., additional, Squires, James E., additional, Karpen, Saul J., additional, Romero, Rene, additional, Kasahara, Mureo, additional, Önal, Zerrin, additional, Sokal, Etienne, additional, Demaret, Tanguy, additional, Wiecek, Sabina, additional, Lacaille, Florence, additional, Debray, Dominique, additional, Hardikar, Winita, additional, Shankar, Sahana, additional, Valentino, Pamela, additional, Sundaram, Shikha, additional, Chaidez, Alexander, additional, Ebel, Noelle, additional, Feinstein, Jeffrey, additional, Mozar-Glazberg, Yael, additional, Lin, Henry, additional, Rock, Nathalie, additional, Verkade, Henkjan J., additional, Jensen, M.K., additional, Jaramillo, Catalina, additional, Kim, Kyungmo, additional, Oh, Seak Hee, additional, Brecelj, Jernej, additional, Alam, Seema, additional, Indolfi, Giuseppe, additional, Blondet, Niviann, additional, Fawaz, Rima, additional, Nastasio, Silvia, additional, Calvo, Pier Luigi, additional, Nebbia, Gabriella, additional, Arıkan, Cigdem, additional, Larson-Nath, Catherine, additional, Zizzo, Andréanne N., additional, Sandahl, Thomas Damgaard, additional, Tzivinikos, Christos, additional, El-Koofy, Nehal, additional, Elmonem, Mohamed, additional, Desai, Dev, additional, Karnsakul, Wikrom, additional, Karthikeyan, Palaniswamy, additional, Bulut, Pinar, additional, Kerkar, Nanda, additional, Wolters, Victorien, additional, Roberts, Amin J, additional, Evans, Helen, additional, Sanchez, Maria Camila, additional, Cavalieri, Maria Lorena, additional, Kelly, Deirdre, additional, Lee, Way Seah, additional, Hajinicolaou, Christina, additional, Lertudomphonwanit, Chatmanee, additional, Fischer, Ryan, additional, Bernabeu, Jesús Quintero, additional, Quiros-Tejeira, Ruben E., additional, Melere, Melina, additional, Carvalho, Elisa, additional, Eshun, John, additional, Zellos, Aglaia, additional, Dezsőfi, Antal, additional, Pinto, Raquel Borges, additional, Schwarz, Kathleen, additional, Rogalidou, Maria, additional, Garcia, Jennifer, additional, Tamara, María Legarda, additional, Beretta, Marisa, additional, Mujawar, Quais, additional, Santos-Silva, Ermelinda, additional, Busoms, Cristina Molera, additional, Lurz, Eberhard, additional, Gonçalves, Cristina, additional, Jimenez-Rivera, Carolina, additional, Bañales, Jesús M., additional, Shah, Uzma, additional, Thompson, Richard, additional, Hansen, Bettina, additional, and Kamath, Binita M., additional

Published
2023
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9. Reply

Author

Vandriel, Shannon M., primary and Kamath, Binita M., additional

Published
2023
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10. HIV viral suppression results in higher antibody responses in HIV-positive women vaccinated with the quadrivalent human papillomavirus vaccine

Author

Alimenti, Ariane, Dobson, Simon, Hankins, Catherine, Hill, Janet, Krajden, Mel, McAlpine, Jessica, Pick, Neora, Singer, Joel, Stone, Sarah, Summers, Marcie, Tan, Darrell, Vicol, Laura, van Schalkwyk, Julie, Money, Deborah M., Moses, Erin, Blitz, Sandra, Vandriel, Shannon M., Lipsky, Nancy, Walmsley, Sharon L., Loutfy, Mona, Trottier, Sylvie, Smaill, Fiona, Yudin, Mark H., Klein, Marina, Harris, Marianne, Cohen, Jeffrey, Wobeser, Wendy, Bitnun, Ari, Lapointe, Normand, Samson, Lindy, Brophy, Jason, Karatzios, Christos, Ogilvie, Gina, Coutlée, François, and Raboud, Janet

Published
2016
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12. THU-291 - Serum bile acids are associated with native liver survival in patients with Alagille syndrome: results from the GALA study group

Author

Murillo Perez, Carla Fiorella, Vandriel, Shannon M., Wang, Jian-She, Liting, Li, She, Huiyu, Jankowska, Irena, Czubkowski, Piotr, Gliwicz, Dorota, Gonzalès, Emmanuel, Jacquemin, Emmanuel, Bouligand, Jérôme, D’Antiga, Lorenzo, Nicastro, Emanuele, Fischler, Björn, Arnell, Henrik, Siew, Susan, Stormon, Michael, Loomes, Kathleen M., Piccoli, David A., Rand, Elizabeth B., Squires, James E., Karpen, Saul J., Romero, Rene, Kasahara, Mureo, Önal, Zerrin, Sokal, Etienne, Demaret, Tanguy, Wiecek, Sabina, Lacaille, Florence, Debray, Dominique, Hardikar, Winita, Shankar, Sahana, Valentino, Pamela, Sundaram, Shikha, Chaidez, Alexander, Ebel, Noelle, Feinstein, Jeffrey, Mozar-Glazberg, Yael, Lin, Henry, Rock, Nathalie, Verkade, Henkjan J., Jensen, M.K., Jaramillo, Catalina, Kim, Kyungmo, Oh, Seak Hee, Brecelj, Jernej, Alam, Seema, Indolfi, Giuseppe, Blondet, Niviann, Fawaz, Rima, Nastasio, Silvia, Calvo, Pier Luigi, Nebbia, Gabriella, Arıkan, Cigdem, Larson-Nath, Catherine, Zizzo, Andréanne N., Sandahl, Thomas Damgaard, Tzivinikos, Christos, El-Koofy, Nehal, Elmonem, Mohamed, Desai, Dev, Karnsakul, Wikrom, Karthikeyan, Palaniswamy, Bulut, Pinar, Kerkar, Nanda, Wolters, Victorien, Roberts, Amin J, Evans, Helen, Sanchez, Maria Camila, Cavalieri, Maria Lorena, Kelly, Deirdre, Lee, Way Seah, Hajinicolaou, Christina, Lertudomphonwanit, Chatmanee, Fischer, Ryan, Bernabeu, Jesús Quintero, Quiros-Tejeira, Ruben E., Melere, Melina, Carvalho, Elisa, Eshun, John, Zellos, Aglaia, Dezsőfi, Antal, Pinto, Raquel Borges, Schwarz, Kathleen, Rogalidou, Maria, Garcia, Jennifer, Tamara, María Legarda, Beretta, Marisa, Mujawar, Quais, Santos-Silva, Ermelinda, Busoms, Cristina Molera, Lurz, Eberhard, Gonçalves, Cristina, Jimenez-Rivera, Carolina, Bañales, Jesús M., Shah, Uzma, Thompson, Richard, Hansen, Bettina, and Kamath, Binita M.

Published
2023
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13. Discovery of thyrocyte heterogeneity reveals an essential role of Notch signaling in thyroid function and mammalian homeostasis

Author

Mosteiro, Lluc, primary, Thao Nguyen, Thi Thu, additional, Hankeova, Simona, additional, Reichelt, Mike, additional, Vandriel, Shannon M., additional, Lai, Zijuan, additional, Choudhury, Feroza K., additional, Sangaraju, Dewakar, additional, Kamath, Binita M., additional, Scherl, Alexis, additional, Piskol, Robert, additional, and Siebel, Christian W., additional

Published
2022
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15. Natural history of liver disease in a large international cohort of children with Alagille syndrome: Results from the GALA study.

Author

Vandriel, Shannon M., Li, Li‐Ting, She, Huiyu, Wang, Jian‐She, Gilbert, Melissa A., Jankowska, Irena, Czubkowski, Piotr, Gliwicz‐Miedzińska, Dorota, Gonzales, Emmanuel M., Jacquemin, Emmanuel, Bouligand, Jérôme, Spinner, Nancy B., Loomes, Kathleen M., Piccoli, David A., D'Antiga, Lorenzo, Nicastro, Emanuele, Sokal, Étienne, Demaret, Tanguy, Ebel, Noelle H., and Feinstein, Jeffrey A.

Published
2023
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16. Application of real-world evidence analytics: A 6-year event-free survival analysis in alagille syndrome of the gala clinical research database and maralixibat treated patients

Author

Hansen, Bettina E., Vandriel, Shannon M., Vig, Pamela, Garner, Will, Li, Li-Ting, She, Huiyu, Wang, Jian-She, Gilbert, Melissa A., Jankowska, Irena, Czubkowski, Piotr, Gliwicz-Miedzinska, Dorota, Gonzales, Emmanuel M., Jacquemin, Emmanuel, Bouligand, Jerome, Spinner, Nancy, Loomes, Kathleen M., Piccoli, David A., D'Antiga, Lorenzo, Nicastro, Emanuele, Sokal, Etienne, Demaret, Tanguy, Ebel, Noelle, Feinstein, Jeffrey A., Fawaz, Rima, Nastasio, Silvia, Lacaille, Florence, Debray, Dominique, Arnell, Henrik, Fischler, Bjorn, Siew, Susan, Stormon, Michael, Karpen, Saul J., Romero, Rene, Kim, Kyung Mo, Baek, Woo Yim, Hardikar, Winita, Shankar, Sahana, Roberts, Amin J., Evans, Helen M., Jensen, M. Kyle, Kavan, Marianne, Sundaram, Shikha S., Chaidez, Alexander, Karthikeyan, Palaniswamy, Davison, Suzanne, Sanchez, Maria Camila, Cavalieri, Maria Lorena, Verkade, Henkjan J., Lee, Way Seah, Squires, James E., and Center for Liver, Digestive and Metabolic Diseases (CLDM)

Published
2021

18. Clinical features and natural history of 1154 Alagille syndrome patients: results from the international multicenter GALA study group

Author

Vandriel, Shannon M., primary, Liting, Li, additional, She, Huiyu, additional, Wang, Jian-She, additional, Czubkowski, Piotr, additional, Jankowska, Irena, additional, Gliwicz, Dorota, additional, Piccoli, David A., additional, Loomes, Kathleen, additional, Spinner, Nancy, additional, Sokal, Etienne, additional, Demaret, Tanguy, additional, D’Antiga, Lorenzo, additional, Nicastro, Emanuele, additional, Fawaz, Rima, additional, Nastasio, Silvia, additional, Siew, Susan, additional, Stormon, Michael, additional, Arnell, Henrik, additional, Fischler, Björn, additional, Kim, Kyungmo, additional, Baek, Woo Yim, additional, Karpen, Saul, additional, Romero, Rene, additional, Hardikar, Winita, additional, Shankar, Sahana, additional, Roberts, Amin J, additional, Evans, Helen, additional, Sanchez, Maria Camila, additional, Cavalieri, Maria Lorena, additional, Karthikeyan, Palaniswamy, additional, Davison, Suzanne, additional, Jensen, M.K., additional, Kavan, Marianne, additional, Verkade, Henkjan, additional, Lee, Way Seah, additional, Squires, James E., additional, Lertudomphonwanit, Chatmanee, additional, Fischer, Ryan, additional, Lin, Henry, additional, Karnsakul, Wikrom, additional, Kelly, Deirdre, additional, Quiros-Tejeira, Ruben E., additional, Targa, Cristina, additional, Carvalho, Elisa, additional, Alam, Seema, additional, Banales, Jesus M., additional, Indolfi, Giuseppe, additional, Larson-Nath, Catherine, additional, Bulut, Pinar, additional, Calvo, Pier Luigi, additional, Valentino, Pamela, additional, Arıkan, Cigdem, additional, Schwarz, Kathleen, additional, Kanavaki, Ino, additional, Dezsőfi, Antal, additional, Borges-Pinto, Raquel, additional, Wiecek, Sabina, additional, Mujawar, Quais, additional, Brecelj, Jernej, additional, Kerkar, Nanda, additional, Santos-Silva, Ermelinda, additional, Jimenez-Rivera, Carolina, additional, Waisbourd-Zinman, Orith, additional, Nebbia, Gabriella, additional, Gonzales, Emmanuel, additional, Thompson, Richard, additional, Hansen, Bettina, additional, and Kamath, Binita M., additional

Published
2020
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20. HIV viral suppression results in higher antibody responses in HIV-positive women vaccinated with the quadrivalent human papillomavirus vaccine

Author

Money, Deborah M., primary, Moses, Erin, additional, Blitz, Sandra, additional, Vandriel, Shannon M., additional, Lipsky, Nancy, additional, Walmsley, Sharon L., additional, Loutfy, Mona, additional, Trottier, Sylvie, additional, Smaill, Fiona, additional, Yudin, Mark H., additional, Klein, Marina, additional, Harris, Marianne, additional, Cohen, Jeffrey, additional, Wobeser, Wendy, additional, Bitnun, Ari, additional, Lapointe, Normand, additional, Samson, Lindy, additional, Brophy, Jason, additional, Karatzios, Christos, additional, Ogilvie, Gina, additional, Coutlée, François, additional, Raboud, Janet, additional, Alimenti, Ariane, additional, Dobson, Simon, additional, Hankins, Catherine, additional, Hill, Janet, additional, Krajden, Mel, additional, McAlpine, Jessica, additional, Pick, Neora, additional, Singer, Joel, additional, Stone, Sarah, additional, Summers, Marcie, additional, Tan, Darrell, additional, Vicol, Laura, additional, and van Schalkwyk, Julie, additional

Published
2016
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21. FRI312 - Clinical features and natural history of 1154 Alagille syndrome patients: results from the international multicenter GALA study group.

Author

Vandriel, Shannon M., Liting, Li, She, Huiyu, Wang, Jian-She, Czubkowski, Piotr, Jankowska, Irena, Gliwicz, Dorota, Piccoli, David A., Loomes, Kathleen, Spinner, Nancy, Sokal, Etienne, Demaret, Tanguy, D'Antiga, Lorenzo, Nicastro, Emanuele, Fawaz, Rima, Nastasio, Silvia, Siew, Susan, Stormon, Michael, Arnell, Henrik, and Fischler, Björn

Subjects
*SYNDROMES, *PATIENTS, *LIVER diseases
Published
2020
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