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1. Costs of pediatric liver transplantation among commercially insured and Medicaid-insured patients with cholestasis in the US.

Author

Miloh, Tamir, Goldstein, Andrea, Howard, Robin, Mogul, Douglas, Marden, Jessica, Anderson, Annika, Gaburo, Katherine, Kirson, Noam, and Rosenthal, Philip

Subjects
United States, Humans, Child, Adolescent, Medicaid, Insurance, Health, Retrospective Studies, Health Care Costs, Liver Transplantation, Cholestasis
Abstract

Children with rare cholestatic liver diseases, such as Alagille syndrome, progressive familial intrahepatic cholestasis, and biliary atresia typically require liver transplantation (LT). The objective of this analysis was to assess the economic burden of LT on these patients. Health care resource utilization and costs associated with pediatric LT were retrospectively assessed using insurance claims data from the US IBM MarketScan Commercial and Medicaid databases collected between October 2015 and December 2019. Inclusion criteria were as follows: ≥1 procedure code for LT,

Published
2023

2. Alanine Aminotransferase and Gamma‐Glutamyl Transpeptidase Predict Histologic Improvement in Pediatric Nonalcoholic Steatohepatitis

Author

Newton, Kimberly P, Lavine, Joel E, Wilson, Laura, Behling, Cynthia, Vos, Miriam B, Molleston, Jean P, Rosenthal, Philip, Miloh, Tamir, Fishbein, Mark H, Jain, Ajay K, Murray, Karen F, Schwimmer, Jeffrey B, and Network, for the Nonalcoholic Steatohepatitis Clinical Research

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Liver Disease, Chronic Liver Disease and Cirrhosis, Pediatric, Digestive Diseases, Hepatitis, Oral and gastrointestinal, Good Health and Well Being, Adolescent, Alanine Transaminase, Aspartate Aminotransferases, Child, Cysteamine, Delayed-Action Preparations, Female, Humans, Liver, Male, Non-alcoholic Fatty Liver Disease, Predictive Value of Tests, Prognosis, Remission Induction, Treatment Outcome, gamma-Glutamyltransferase, Nonalcoholic Steatohepatitis Clinical Research Network, Medical Biochemistry and Metabolomics, Immunology, Gastroenterology & Hepatology, Clinical sciences
Abstract

Background and aimsPredictive, noninvasive tools are needed to monitor key features of nonalcoholic fatty liver disease (NAFLD) in children that relate to improvement in liver histology. The purpose of this study was to evaluate the relationship between liver chemistries and liver histology using data from the CyNCh (Cysteamine Bitartrate Delayed-Release for the Treatment of NAFLD in Children) clinical trial.Approach and resultsThis study included 146 children. Improvement in liver histology, defined as decrease in nonalcoholic fatty liver disease (NAFLD) Activity Score ≥2 points without worsening of fibrosis, occurred in 43 participants (30%). There were 46 participants with borderline zone 1 nonalcoholic steatohepatitis (NASH) at baseline, with resolution in 28% (12 of 46). Multivariate models were constructed using baseline and change in alanine aminotransferase (ALT), aspartate aminotransferase (AST), and gamma-glutamyl transferase (GGT) at 52 weeks, for improvement in (1) liver histology primary outcome, (2) borderline zone 1 NASH, and (3) fibrosis. For improvement in histology, the model (P

Published
2021

3. Progression of Fatty Liver Disease in Children Receiving Standard of Care Lifestyle Advice

Author

Xanthakos, Stavra A, Lavine, Joel E, Yates, Katherine P, Schwimmer, Jeffrey B, Molleston, Jean P, Rosenthal, Philip, Murray, Karen F, Vos, Miriam B, Jain, Ajay K, Scheimann, Ann O, Miloh, Tamir, Fishbein, Mark, Behling, Cynthia A, Brunt, Elizabeth M, Sanyal, Arun J, Tonascia, James, Abrams, Stephanie, Garner, Donna, Hertel, Paula, Himes, Ryan, Lawson, Alicia, Triggs, Nicole, Bramlage, Kristin, Carr, April, Cecil, Kim, McNeill, Meghan, Mouzaki, Marialena, Trout, Andrew, Xanthakos, Stavra, Bernstein, Kimberlee, DeVore, Stephanie, Kohli, Rohit, Lake, Kathleen, Podberesky, Daniel, Towbin, Alex, Mencin, Ali, Reynoso, Elena, Alazraki, Adina, Cleeton, Rebecca, Cordero, Maria, Hernandez, Albert, Karpen, Saul, Munos, Jessica Cruz, Raviele, Nicholas, Vos, Miriam, Bozic, Molly, Carr, Laura, Cummings, Oscar W, Harlow, Kathryn, Klipsch, Ann, Ragozzino, Emily, Rao, Girish, Kafka, Kimberly, Scheimann, Ann, Fishbein, Mark H, Ito, Joy, Mohammad, Saeed, Whitington, Peter F, Barlow, Sarah, Carpenter, Danielle, Cattoor, Theresa, Derdoy, Jose, Freebersyser, Janet, Jain, Ajay, King, Debra, Lai, Jinping, Siegner, Joan, Stewart, Susan, Torretta, Susan, Wriston, Kristina, Angeles, Jorge, Arin, Jennifer, Behling, Cynthia, Bross, Craig, Carrier, Carissa, Collins, Jennifer, De La Pena, Diana, Durelle, Janis, Huckaby, Mary Catherine, Middleton, Michael S, Newton, Kimberly, Sirlin, Claude, Ugalde-Nicalo, Patricia, Courtier, Jesse, Gill, Ryan, Langlois, Camille, Perito, Emily Rothbaum, Tsai, Patrika, Blondet, Niviann, Cooper, Kara, Murray, Karen, Otto, Randolph, and Yeh, Matthew

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Obesity, Clinical Trials and Supportive Activities, Clinical Research, Digestive Diseases, Liver Disease, Chronic Liver Disease and Cirrhosis, Pediatric, Diabetes, Hepatitis, Oral and gastrointestinal, Adolescent, Age Factors, Alanine Transaminase, Aspartate Aminotransferases, Biomarkers, Biopsy, Blood Glucose, Child, Diabetes Mellitus, Type 2, Disease Progression, Female, Healthy Lifestyle, Humans, Male, Non-alcoholic Fatty Liver Disease, Pediatric Obesity, Prospective Studies, Randomized Controlled Trials as Topic, Risk Assessment, Risk Factors, Risk Reduction Behavior, Severity of Illness Index, Time Factors, Treatment Outcome, ALT, Cirrhosis, Histology, Natural History, NASH Clinical Research Network, Neurosciences, Paediatrics and Reproductive Medicine, Gastroenterology & Hepatology, Clinical sciences, Nutrition and dietetics
Abstract

Background & aimsNonalcoholic fatty liver disease (NAFLD) is the most common pediatric chronic liver disease. Little is known about outcomes in recognized youth.MethodsWe compared paired liver biopsies from 122 of 139 children with NAFLD (74% male; 64% white; 71% Hispanic; mean age, 13 ± 3 years; age range, 8-17 years) who received placebo and standard of care lifestyle advice in 2 double-blind, randomized clinical trials within the nonalcoholic steatohepatitis (NASH) clinical research network from 2005 through 2015. We analyzed histologic changes with respect to baseline and longitudinal change in clinical variables using regression analysis.ResultsAt enrollment, 31% of the children had definite NASH, 34% had borderline zone 1 NASH, 13% had borderline zone 3 NASH, and 21% had fatty liver but not NASH. Over a mean period of 1.6 ± 0.4 years, borderline or definite NASH resolved in 29% of the children, whereas 18% of the children with fatty liver or borderline NASH developed definite NASH. Fibrosis improved in 34% of the children but worsened in 23%. Any progression to definite NASH and/or in fibrosis was associated with adolescent age, and higher waist circumference, levels of alanine or aspartate aminotransferase, total and low-density lipoprotein cholesterol at baseline (

Published
2020

4. International Liver Transplantation Society Global Census: First Look at Pediatric Liver Transplantation Activity Around the World

Author

Rodriguez-Davalos, Manuel I., Lopez-Verdugo, Fidel, Kasahara, Mureo, Muiesan, Paolo, Reddy, Mettu S., Flores-Huidobro Martinez, Angel, Xia, Qiang, Hong, Johnny C., Niemann, Claus U., Seda-Neto, Joao, Miloh, Tamir A., Yi, Nam-Joon, Mazariegos, George V., Ng, Vicky L., Esquivel, Carlos O., Lerut, Jan, Rela, Mohamed, Botha, Jean, Mehta, Naimish N., Bakar Hafeez Bhatti, Abu, Zamora-Valdes, Daniel, Anand, Superina, Riccardo, de Kleine, Ruben, Matamoros, Maria A., Bedregal-Mendoza, Tayana, Steve Bynon, J., Rodriguez-Sancho, Luis C., Langnas, Alan, Dhawan, Anil, Ogura, Yasuhiro, Vianna, Rodrigo, Ilyassova, Bibigul, Tantemsapya, Niramol, Bellemare, Sarah, Ovchinsky, Nadia, Bhalerao, Suryabhan, Balci, Deniz, Dip, Marcelo, Imventarza, Oscar, Mangus, Richard S., Fawcett, Jonathan, Antonini, Teresa M., Shrimal, Anurag, Zhu, Zhi-Jun, Rivera, Jairo, Dayangac, Murat, McCall, John, Utz Melere, Melina, Stormon, Michael, Mizuta, Koichi, Gao, Wei, Hening Rahayatri, Tri, Costaguta, Alejandro, Gautier, Sergei, Kazemi, Kourosh, Martinez-Castillo, Jorge, Apinyachon, Worapot, Rastogi, Mukul, Restrepo, Juan C., Takahito, Yagi, Ravichandra, Srinivasa, Vanin, Anabel, Vieira, Sandra, Luque, Carlos, Gondolesi, Gabriel, Hashimoto, Koji, Yazigi, Nada, Lebel, Sylvie, Mavis, Alisha, Arnon, Ronen, Chavez-Vargas, Ricardo, Elisofon, Scott, Desai, Dev M., Okajima, Hideaki, Chinnakotla, Srinath, Hsu, Evelyn, Reyes, Jorge, Orgoi, Sergelen, Raut, Vikram, Hee Oh, Seak, Valentino, Pamela, Emre, Sukru, Spada, Marco, Leung, Dan, Charco, Ramon, Ericzon, Bo-Goran, Fischer, Ryan, Del Rio-Martin, Juan V., Cillo, Umberto, Soliman, Hossam, Varela-Fascinetto, Gustavo, de Villa, Vanessa, Haberal, Mehmet, Covarrubias, Joshue, Colledan, Michele, de Ville de Goyet, Jean, Desai, Chirag, Sundaram, Shikha, Fronek, Jiri, Nadig, Satish N., Mali, Vidyadhar P., Tandoi, Francesco, Kohli, Rohit, Mohan, Neelam, Goyal, Neerav, Santhosh, Drisya, Debray, Dominique, Durmaz, Ozlem, De Bruyne, Ruth, Kaliciński, Piotr, Hierro, Loreto, Venick, Robert, Perito, Emily, Mozer-Glassberg, Yael, Chen, Chao-Long, Rand, Elizabeth, Nikeghbalian, Saman, Kelly, Deirdre, Boillot, Olivier, and Book, Linda S.

Published
2023
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7. Colorectal Dysplasia and Cancer in Pediatric-Onset Ulcerative Colitis Associated With Primary Sclerosing Cholangitis

Author

El-Matary, Wael, Guthery, Stephen L., Amir, Achiya Z., DiGuglielmo, Matthew, Draijer, Laura G., Furuya, Katryn N., Gupta, Nitika, Hochberg, Jessica T., Horslen, Simon, Kerkar, Nanda, Koot, Bart G.P., Laborda, Trevor J., Loomes, Kathleen M., Mack, Cara, Martinez, Mercedes, Miethke, Alexander, Miloh, Tamir, Mogul, Douglas, Mohammed, Saeed, Moroz, Stacy, Ovchinsky, Nadia, Perito, Emily R., Rao, Girish, Ricciuto, Amanda, Sathya, Pushpa, Schwarz, Kathleen B., Shah, Uzma, Singh, Ruchi, Soufi, Nisreen, Valentino, Pamela L., Zizzo, Andréanne, and Deneau, Mark R.

Published
2021
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8. Gastrointestinal Hemorrhage: A Manifestation of the Telomere Biology Disorders

Author

Himes, Ryan W., Chiou, Eric H., Queliza, Karen, Shouval, Dror S., Somech, Raz, Agarwal, Suneet, Jajoo, Kunal, Ziegler, David S., Kratz, Christian P., Huang, James, Lucas, Tiffany L., Myers, Kasiani C., Nelson, Adam S., DiNardo, Courtney D., Alter, Blanche P., Giri, Neelam, Khincha, Payal P., McReynolds, Lisa J., Dufour, Carlo, Pierri, Filomena, Goldman, Frederick D., Sherif, Youmna, Savage, Sharon A., Miloh, Tamir, and Bertuch, Alison A.

Published
2021
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12. Ursodeoxycholic Acid Therapy in Pediatric Primary Sclerosing Cholangitis: Predictors of Gamma Glutamyltransferase Normalization and Favorable Clinical Course

Author

Deneau, Mark, Perito, Emily, Ricciuto, Amanda, Gupta, Nitika, Kamath, Binita M., Palle, Sirish, Vitola, Bernadette, Smolka, Vratislav, Ferrari, Federica, Amir, Achiya Z., Miloh, Tamir, Papadopoulou, Alexandra, Mohan, Parvathi, Mack, Cara, Kolho, Kaija-Leena, Iorio, Raffaele, El-Matary, Wael, Venkat, Veena, Chan, Albert, Saubermann, Lawrence, Valentino, Pamela L., Shah, Uzma, Miethke, Alexander, Lin, Henry, and Jensen, M.K.

Published
2019
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15. Frailty in Children with Liver Disease: A Prospective Multicenter Study

Author

Lurz, Eberhard, Quammie, Claudia, Englesbe, Michael, Alonso, Estella M., Lin, Henry C., Hsu, Evelyn K., Furuya, Katryn N., Gupta, Nitika A., Venkat, Veena L., Daniel, James F., Leonis, Mike A., Miloh, Tamir, Telega, Grzegorz W., Yap, Jason, Menendez, Jerome, Book, Linda S., Himes, Ryan W., Sundaram, Shikha S., Parekh, Rulan, Sonnenday, Chris, Bucuvalas, John, Ng, Vicky L., and Kamath, Binita M.

Published
2018
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22. List of Contributors

Author

Abuduxikuer, Kuerbanjiang, primary, Aggarwal, Annu, additional, Amir, Achiya Zvi, additional, Annunziato, Rachel A., additional, Barbosa, Egberto R., additional, Bavdekar, Ashish, additional, Bhatt, Mohit, additional, Blindauer, Claudia A., additional, Borchard, Sabine, additional, C. Bull, Peter, additional, Cançado, Eduardo L.R., additional, Chang, Irene J., additional, Chiappe, Francesca, additional, Cobine, Paul A., additional, Coenen, Iris C.J., additional, Cox, Diane Wilson, additional, Członkowska, Anna, additional, Denk, Helmut, additional, Dhawan, Anil, additional, Dmitriev, Oleg Y., additional, Ferenci, Peter, additional, Frizziero, Luisa, additional, Frydman, Moshe, additional, Gupta, Arnab, additional, Hahn, Si Houn, additional, Harris, Zena Leah, additional, Hermann, Wieland, additional, Houwen, Roderick H.J., additional, Huster, Dominik, additional, Iorio, Raffaele, additional, Ivanova, Irena, additional, Jayakanthan, Samuel, additional, Jung, Sunhee, additional, Kaler, Stephen G., additional, Kerkar, Nanda, additional, Kieffer, Dorothy A., additional, Kirk, Richard, additional, Kyrana, Eirini, additional, Lackner, Carolin, additional, Latorre, Mauricio, additional, Lepori, Maria B., additional, Litwin, Tomasz, additional, Loudianos, Georgios, additional, Lutsenko, Svetlana, additional, Mak, Chloe M., additional, Medici, Valentina, additional, Midena, Edoardo, additional, Miloh, Tamir, additional, Parrozzani, Raffaele, additional, Polishchuk, Roman S., additional, Poujois, Aurélia, additional, Poupon, Joël, additional, Quindipan, Catherine, additional, Ranucci, Giusy, additional, Ray, Kunal, additional, Roberts, Eve A., additional, Rommens, Johanna, additional, Rongioletti, Mauro, additional, Rose, Carl, additional, Rosenthal, Philip, additional, Rupp, Christian, additional, Schiano, Thomas D., additional, Schilsky, Michael L., additional, Shteyer, Eyal, additional, Sintusek, Palittiya, additional, Siotto, Mariacristina, additional, Socha, Piotr, additional, Solioz, Marc, additional, Squitti, Rosanna, additional, Sussman, Norman L., additional, Tanner, Stuart, additional, Torbenson, Vanessa, additional, Troncoso, Rodrigo, additional, Uauy, Ricardo, additional, van de Sluis, Bart, additional, Vest, Katherine E., additional, Vierling, John M., additional, Walshe, John M., additional, Wang, Jian-She, additional, Weiss, Karl H., additional, Woimant, France, additional, Yi, Ling, additional, Zeid, Cynthia Abou, additional, Zhu, Xinyu, additional, Zimbrean, Paula C., additional, and Zischka, Hans, additional

Published
2019
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23. Vedolizumab Therapy in Children With Primary Sclerosing Cholangitis: Data From the Pediatric Primary Sclerosing Cholangitis Consortium

Author

Laborda, Trevor J., Ricciuto, Amanda, Aumar, Madeleine, Carman, Nicholas, DiGuglielmo, Matthew, Draijer, Laura G., Furuya, Katryn N., Gupta, Nitika, Koot, Bart G.P., Loomes, Kathleen M., Lytvyak, Ellina, Martinez, Mercedes, Miloh, Tamir, Montano-Loza, Aldo J., Perito, Emily R., Sathya, Pushpa, Shah, Uzma, Shteyer, Eyal, Singh, Ruchi, Taylor, Amy, Valentino, Pamela L., Vitola, Bernadette, Zerofsky, Melissa, Zizzo, Andréanne, and Deneau, Mark R.

Published
2020
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24. Assessing the Validity of Adult-derived Prognostic Models for Primary Sclerosing Cholangitis Outcomes in Children

Author

Deneau, Mark R., Valentino, Pamela L., Mack, Cara, Alqoaer, Khaled, Amin, Mansi, Amir, Achiya Z., Aumar, Madeleine, Auth, Marcus, Broderick, Annemarie, DiGuglielmo, Matthew, Draijer, Laura G., El-Matary, Wael, Ferrari, Federica, Furuya, Katryn N., Gottrand, Frederic, Gupta, Nitika, Homan, Matjaz, Jensen, M.K., Kamath, Binita M., Kim, Kyung Mo, Kolho, Kaija-Leena, Koot, Bart, Iorio, Raffaele, Martinez, Mercedes, Miloh, Tamir, Mohan, Parvathi, Palle, Sirish, Papadopoulou, Alexandra, Ricciuto, Amanda, Saubermann, Lawrence, Sathya, Pushpa, Shteyer, Eyal, Smolka, Vratislav, Tanaka, Atsushi, Varier, Raghu, Venkat, Veena, Vitola, Bernadette, Woynarowski, Marek, and Guthery, Stephen

Published
2020
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29. Fulminant Hepatic Failure

Author

Miloh, Tamir, Suchy, Fredrick J., Elzouki, Abdelaziz Y., editor, Harfi, Harb A., editor, Nazer, Hisham M., editor, Stapleton, F. Bruder, editor, Oh, William, editor, and Whitley, Richard J., editor

Published
2012
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30. Predicting Liver Allograft Discard: The Discard Risk Index

Author

Rana, Abbas, Sigireddi, Rohini R., Halazun, Karim J., Kothare, Aishwarya, Wu, Meng-Fen, Liu, Hao, Kueht, Michael L., Vierling, John M., Sussman, Norman L., Mindikoglu, Ayse L., Miloh, Tamir, Galvan, N. Thao N., Cotton, Ronald T., O’Mahony, Christine A., and Goss, John A.

Published
2018
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39. Recurrence of Primary Sclerosing Cholangitis After Liver Transplant in Children: An International Observational Study

Author

Martinez, Mercedes, primary, Perito, Emily R., additional, Valentino, Pamela, additional, Mack, Cara L, additional, Aumar, Madeleine, additional, Broderick, Annemarie, additional, Draijer, Laura G., additional, Fagundes, Eleonora D.T., additional, Furuya, Katryn N., additional, Gupta, Nitika, additional, Horslen, Simon, additional, Jonas, Maureen M., additional, Kamath, Binita M., additional, Kerkar, Nanda, additional, Kim, Kyung Mo, additional, Kolho, Kaija‐Leena, additional, Koot, Bart G.P., additional, Laborda, Trevor J., additional, Lee, Christine K., additional, Loomes, Kathleen M., additional, Miloh, Tamir, additional, Mogul, Douglas, additional, Mohammed, Saeed, additional, Ovchinsky, Nadia, additional, Rao, Girish, additional, Ricciuto, Amanda, additional, Rodrigues Ferreira, Alexandre, additional, Schwarz, Kathleen B., additional, Smolka, Vratislav, additional, Tanaka, Atsushi, additional, Tessier, Mary E.M., additional, Venkat, Venna L., additional, Vitola, Bernadette E., additional, Woynarowski, Marek, additional, Zerofsky, Melissa, additional, and Deneau, Mark R., additional

Published
2021
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42. 422 ORAL VANCOMYCIN THERAPY IS ASSOCIATED WITH IBD CLINICAL REMISSION IN PEDIATRIC PSC-IBD

Author

Ricciuto, Amanda, primary, Liu, Kuan, additional, Amir, Achiya Z., additional, Aumar, Madeleine, additional, Broderick, Annemarie, additional, Draijer, Laura, additional, El-Matary, Wael, additional, Fagundes, Eleonora D., additional, Ferrari, Federica, additional, Furuya, Katryn, additional, Gupta, Nitika, additional, Horslen, Simon P., additional, Iorio, Raffaele, additional, Jonas, Maureen M., additional, Kamath, Binita M., additional, Kerkar, Nanda, additional, Kim, Kyung Mo, additional, Kolho, Kaija-Leena, additional, Koot, Bart, additional, Laborda, Trevor, additional, Lee, Christine, additional, Loomes, Kathleen, additional, Mack, Cara L., additional, Martinez, Mercedes, additional, Miloh, Tamir, additional, Mogul, Douglas, additional, Mohammad, Saeed, additional, Ovchinsky, Nadia, additional, Palle, Sirish, additional, Perito, Emily, additional, Ferreira, Alexandre Rodrigues, additional, Schwarz, Kathleen B., additional, Rao, Girish S., additional, Tanaka, Atsushi, additional, Valentino, Pamela L., additional, Venkat, Veena, additional, Vitola, Bernadette E., additional, Vratislav, Smolka, additional, Woynarowski, Marek, additional, Zerofsky, Melissa, additional, and Deneau, Mark R., additional

Published
2021
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45. North American Biliary Stricture Management Strategies in Children After Liver Transplantation: A Multicenter Analysis From the Society of Pediatric Liver Transplantation (SPLIT) Registry.

Author

Valentino, Pamela L., Wang, Tianhao, Shabanova, Veronika, Ng, Vicky Lee, Bucuvalas, John C., Feldman, Amy G., Gonzalez‐Peralta, Regino P., Gupta, Nitika Arora, Miloh, Tamir A., Mohammad, Saeed, Pace, Erika, Sundaram, Shikha S., Yazigi, Nada A., and Soltys, Kyle

Published
2022
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46. Oral Vancomycin, Ursodeoxycholic Acid, or No Therapy for Pediatric Primary Sclerosing Cholangitis: A Matched Analysis

Author

Deneau, Mark R., primary, Mack, Cara, additional, Mogul, Douglas, additional, Perito, Emily R., additional, Valentino, Pamela L., additional, Amir, Achiya Z., additional, DiGuglielmo, Matthew, additional, Draijer, Laura G., additional, El‐Matary, Wael, additional, Furuya, Katryn N., additional, Gupta, Nitika, additional, Hochberg, Jessica T., additional, Horslen, Simon, additional, Jensen, M. Kyle, additional, Jonas, Maureen M., additional, Kerkar, Nanda, additional, Koot, Bart G.P., additional, Laborda, Trevor J., additional, Lee, Christine K., additional, Loomes, Kathleen M., additional, Martinez, Mercedes, additional, Miethke, Alexander, additional, Miloh, Tamir, additional, Mohammad, Saeed, additional, Ovchinsky, Nadia, additional, Rao, Girish, additional, Ricciuto, Amanda, additional, Sathya, Pushpa, additional, Schwarz, Kathleen B., additional, Shah, Uzma, additional, Singh, Ruchi, additional, Vitola, Bernadette, additional, Zizzo, Andréanne, additional, and Guthery, Stephen L., additional

Published
2021
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47. Severe Acute Respiratory Syndrome Coronavirus-2 Infection in Children With Liver Transplant and Native Liver Disease: An International Observational Registry Study

Author

Kehar, Mohit, primary, Ebel, Noelle H., additional, Ng, Vicky L., additional, Baquero, Jairo Eduardo Rivera, additional, Leung, Daniel H., additional, Slowik, Voytek, additional, Ovchinsky, Nadia, additional, Shah, Amit A., additional, Arnon, Ronen, additional, Miloh, Tamir, additional, Gupta, Nitika, additional, Mohammad, Saeed, additional, Kogan-Liberman, Debora, additional, Squires, James E., additional, Sanchez, Maria Camila, additional, Hildreth, Amber, additional, Book, Linda, additional, Chu, Christopher, additional, Alrabadi, Leina, additional, Azzam, Ruba, additional, Chepuri, Bhavika, additional, Elisofon, Scott, additional, Falik, Rachel, additional, Gallagher, Lisa, additional, Kader, Howard, additional, Mogul, Douglas, additional, Mujawar, Quais, additional, Namjoshi, Shweta S., additional, Valentino, Pamela L., additional, Vitola, Bernadette, additional, Waheed, Nadia, additional, Zheng, Ming-Hua, additional, Lobritto, Steven, additional, and Martinez, Mercedes, additional

Published
2021
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49. [Beta]-klotho and FGF-15/19 inhibit the apical sodium-dependent bile acid transporter in enterocytes and cholangiocytes

Author

Sinha, Jyoti, Chen, Frank, Miloh, Tamir, Burns, Robert C., Yu, Zhisheng, and Shneider, Benjamin L.

Subjects
Membrane proteins -- Health aspects, Biological sciences
Abstract

[beta]-Klotho, a newly described membrane protein, regulates bile acid synthesis. Fibroblast growth factor-15 (FGF-15) and FGF receptor-4 (FGFR4) knockout mice share a similar phenotype with [beta]-Klotho-deficient mice. FGF-15 secretion by the intestine regulates hepatic bile acid biosynthesis. The effects of [beta]-Klotho and FGF-15 on the ileal apical sodium bile transporter (ASBT) are unknown. [beta]-Klotho siRNA treatment of the mouse colon cancer cell line, CT-26, and the human intrahepatic biliary epithelial cells (HIBEC) resulted in upregulation of endogenous ASBT expression that was associated with reduced expression of the farnesoid X receptor (FXR) and the short heterodimer partner (SHP). Silencing [beta]-Klotho activated the ASBT promoter in CT-26, Mz-ChA-1 (human cholangiocarcinoma), and HIBEC cells. Site-directed mutagenesis of liver receptor homolog-1 (mouse) or retinoic acid receptor/retinoid X receptor (RARfRXR) (human) cis-elements attenuated the basal activity of the ASBT promoter and abrogated its response to [beta]-Klotho silencing, siSHP, siFXR, or dominant-negative FXR treatment also eliminated the [beta]-Klotho response. FGF-15 secretion into cell culture media by CT-26 cells was diminished after siFGF-15 or si[beta]-Klotho treatment and enhanced by chenodeoxycholic acid. Exogenous FGF-19 repressed ASBT protein expression in mouse ileum, gallbladder, and in HIBEC and repressed ASBT promoter activity in Caco-2, HIBEC, and Mz-ChA-1 cells. Promoter repression was dependent on the expression of FGFR4. These results indicate that both [beta]-Klotho and FGF-15/19 repress ASBT in enterocytes and cholangiocytes. These novel signaling pathways need to be considered in analyzing bile acid homeostasis. ileum; liver; regulation; gallbladder

Published
2008

50. The Sclerosing Cholangitis Outcomes in Pediatrics (SCOPE) Index: A Prognostic Tool for Children

Author

Deneau, Mark R., primary, Mack, Cara, additional, Perito, Emily R., additional, Ricciuto, Amanda, additional, Valentino, Pamela L., additional, Amin, Mansi, additional, Amir, Achiya Z., additional, Aumar, Madeleine, additional, Auth, Marcus, additional, Broderick, Annemarie, additional, DiGuglielmo, Matthew, additional, Draijer, Laura G., additional, Tavares Fagundes, Eleonora Druve, additional, El‐Matary, Wael, additional, Ferrari, Federica, additional, Furuya, Katryn N., additional, Gupta, Nitika, additional, Hochberg, Jessica T., additional, Homan, Matjaz, additional, Horslen, Simon, additional, Iorio, Raffaele, additional, Jensen, M. Kyle, additional, Jonas, Maureen M., additional, Kamath, Binita M., additional, Kerkar, Nanda, additional, Kim, Kyung Mo, additional, Kolho, Kaija‐Leena, additional, Koot, Bart G.P., additional, Laborda, Trevor J., additional, Lee, Christine K., additional, Loomes, Kathleen M., additional, Martinez, Mercedes, additional, Miethke, Alexander, additional, Miloh, Tamir, additional, Mogul, Douglas, additional, Mohammad, Saeed, additional, Mohan, Parvathi, additional, Moroz, Stacy, additional, Ovchinsky, Nadia, additional, Palle, Sirish, additional, Papadopoulou, Alexandra, additional, Rao, Girish, additional, Rodrigues Ferreira, Alexandre, additional, Sathya, Pushpa, additional, Schwarz, Kathleen B., additional, Shah, Uzma, additional, Shteyer, Eyal, additional, Singh, Ruchi, additional, Smolka, Vratislav, additional, Soufi, Nisreen, additional, Tanaka, Atsushi, additional, Varier, Raghu, additional, Vitola, Bernadette, additional, Woynarowski, Marek, additional, Zerofsky, Melissa, additional, Zizzo, Andréanne, additional, and Guthery, Stephen L., additional

Published
2020
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