Condition-Specific Growth Charts for Children With Alagille Syndrome.
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Huysentruyt Koen, Vandriel Shannon M, Roelants Mathieu, Piccoli David A, Loomes Kathleen M, Rand Elizabeth B, Ebel Noelle H, Feinstein Jeffrey A, Jankowska Irena, Czubkowski Piotr, Gliwicz-Miedzinska Dorota, Gonzales Emmanuel M, Jacquemin Emmanuel, Bouligand Jérôme, Karpen Saul J, Romero Rene, Lin Henry C, Fischler Björn, Arnell Henrik, Li Li-Ting, Wang Jian-She, Fawaz Rima, Nastasio Silvia, Kim Kyung Mo, Oh Seak Hee, D'Antiga Lorenzo, Nicastro Emanuele, Fischer Ryan T, Siew Susan M, Stormon Michael, Lertudomphonwanit Chatmanee, Hardikar Winita, Shankar Sahana, Squires James E, Sundaram Shikha S, Larson-Nath Catherine, Kelly Deirdre A, Hartley Jane, Bulut Pinar, Jensen M Kyle, Jaramillo Catalina, Roberts Amin J, Evans Helen M, Sokal Étienne M, Demaret Tanguy, Verkade Henkjan J, Thompson Richard J, Hansen Bettina E, Cole Tim J, Kamath Binita M, Global Alagille Alliance (GALA) Study Group, Debray Dominique, Lacaille Florence, Brecelj Jernej, El-Koofy Nehal M, Elmonem Mohamed A, Lee Way Seah, Sanchez Maria Camila, Cavalieri Maria Lorena, Hajinicolaou Christina, Schwarz Kathleen B, Carvalho Elisa, Rock Nathalie, Karnsakul Wikrom, Quiros-Tejeira Ruben E, Alam Seema, Nebbia Gabriella, Mozer-Glassberg Yael, Valentino Pamela L, Santos-Silva Ermelinda, Önal Zerrin, Dezsofi-Gottl Antal, Melere Melina, Legarda Tamara María, Eshun John, Zellos Aglaia, Indolfi Giuseppe, Rogalidou Maria, Blondet Niviann, Luigi Calvo Pier, Beretta Marisa, Zizzo Andréanne N, Arikan Cigdem, Kasahara Mureo, Kerkar Nanda, Aqul Amal A, Wolters Victorien M, Borges Pinto Raquel, Quintero Bernabeu Jesus, Garcia Jennifer, Wiecek Sabina, Tzivinikos Christos, Mujawar Quais, Jimenez-Rivera Carolina, Molera Busoms Cristina, Gonçalves Cristina, Bujanda Luis
Аннотация
OBJECTIVE: To develop condition-specific growth charts for ALGS using modern statistical approaches.
DESIGN, SETTING, AND PARTICIPANTS: This case series used data from the international, multicenter Global Alagille Alliance (GALA) study accrued between May 14, 2018, and March 20, 2023. Children born at full term between January 1, 1997, and August 31, 2019, with a clinically and/or genetically confirmed ALGS diagnosis and their native liver were included. Data from children with a known history of prematurity were excluded for the development of the growth charts. Data were analyzed from March 25, 2023, to December 30, 2024.
EXPOSURE: Growth of children with Alagille syndrome.
MAIN OUTCOMES AND MEASURES: Generalized additive models for location scale and shape were fitted to generate percentile plots for weight and height relative to age and superimposed on US Centers for Disease Control and Prevention (CDC) growth charts to illustrate differences in growth patterns compared with children with typical development.
RESULTS: Data from 1204 children with ALGS in overlapping cohorts (median [IQR] gestational age, 38 [37-39] weeks) were analyzed (1204 in the weight cohort; 695 boys [57.7%]; 9855 weight observations; 995 with neonatal cholestasis [82.6%]; 306 receiving a liver transplant [25.4%]; 98 deaths [8.1%] and 1106 in the height cohort, 635 boys [57.4%]; 8464 height observations; 906 with neonatal cholestasis [81.9%]; 287 receiving a liver transplant [25.9%]; 86 deaths [7.8%]) were included for the modeling of the weight-for-age and height-for-age charts, respectively. The median birth weight was 2.8 kg (IQR, 2.5-3.0 kg) for boys and 2.6 kg (IQR, 2.4-2.9 kg) for girls. The median birth length was 48.0 cm (IQR, 46.0-50.0 cm) for boys and 47.0 cm (IQR, 45.0-49.0 cm) for girls. The weight-for-age and height-for-age growth charts for boys and girls with AGLS differed significantly from CDC growth charts. The estimated height at age 18 years corresponded to the 50th percentile was 171.5 cm for boys and 156.5 cm for girls on the condition-specific charts vs 176 cm and 163 cm, respectively, on the CDC growth charts.
CONCLUSIONS AND RELEVANCE: These findings suggest that condition-specific growth charts for ALGS may provide a crucial tool for clinicians to evaluate growth and aid in decision-making around listing children for liver transplant.