Temporal Trends in Acute Adrenal Insufficiency Events in Children With Congenital Adrenal Hyperplasia During 2019-2022.

Autor:	Tseretopoulou X; Developmental Endocrinology Research Group, School of Medicine, Dentistry & Nursing, University of Glasgow, Glasgow G51 4TF, UK.; Office for Rare Conditions, Royal Hospital for Children & Queen Elizabeth University Hospital, Glasgow G51 4TF, UK., Ali SR; Developmental Endocrinology Research Group, School of Medicine, Dentistry & Nursing, University of Glasgow, Glasgow G51 4TF, UK.; Office for Rare Conditions, Royal Hospital for Children & Queen Elizabeth University Hospital, Glasgow G51 4TF, UK., Bryce J; Office for Rare Conditions, Royal Hospital for Children & Queen Elizabeth University Hospital, Glasgow G51 4TF, UK., Amin N; Department of Paediatric Endocrinology, Leeds Teaching Hospitals NHS Trust, Leeds LS1 3EX, UK., Atapattu N; Department of Paediatric Endocrinology, Lady Ridgeway Hospital, Colombo 00800, Sri Lanka., Bachega TASS; Unidade de Endocrinologia do Desenvolvimento, Laboratório de Hormônios e Genética Molecular/LIM42, Disciplina de Endocrinologia, Hospital Das Clinicas, Faculdade De Medicina, Universidade de Sao Paulo, São Paulo, 05508-090, Brazil., Baronio F; Department Hospital of Woman and Child, Pediatric Unit, IRCCS-Azienda Ospedaliero-Universitaria di Bologna, 40138 Bologna, Italy., Ortolano R; Department Hospital of Woman and Child, Pediatric Unit, IRCCS-Azienda Ospedaliero-Universitaria di Bologna, 40138 Bologna, Italy., Birkebaek NH; Department of Paediatrics, Aarhus University Hospital, Aarhus DK-8200, Denmark., Bonfig W; Department of Paediatrics, Technical University München, D-80804 Munich, Germany.; Department of Paediatrics, Klinikum Wels-Grieskirchen, A-4600 Wels, Austria., Cools M; Department of Paediatric Endocrinology, University Hospital Ghent, Ghent University, 9000 Ghent, Belgium., Davies JH; Department of Paediatric Endocrinology, University Hospital Southampton, Southampton SO16 6YD, UK., Thomas T; Department of Paediatric Endocrinology, University Hospital Southampton, Southampton SO16 6YD, UK., de Vries L; The Jesse and Sara Lea Shafer Institute of Endocrinology and Diabetes, Schneider Children's Medical Center of Israel, Petah Tikvah 4920235, Israel.; Faculty of Medical&Health Sciences, Tel Aviv University, Tel Aviv 6997801, Israel., Elsedfy H; Department of Pediatrics, Ain Shams University, 11566 Cairo, Egypt., Amr NH; Department of Pediatrics, Ain Shams University, 11566 Cairo, Egypt., Flueck CE; Pediatric Endocrinology, Diabetology and Metabolism, Department of Pediatrics, Bern University Hospital Inselspital, University of Bern, 3010 Bern, Switzerland.; Pediatric Endocrinology, Diabetology and Metabolism, Department of BioMedical Research, Bern University Hospital Inselspital, University of Bern, 3010 Bern, Switzerland., Globa E; Ukrainian Scientific and Practical Center of Endocrine Surgery, Transplantation of Endocrine Organs and Tissues of MOH of Ukraine, Kyiv 01021, Ukraine., Guran T; Department of Pediatric Endocrinology and Diabetes, Marmara University, 34899 Pendik, Istanbul, Turkey., Yavas-Abali Z; Department of Pediatric Endocrinology and Diabetes, Marmara University, 34899 Pendik, Istanbul, Turkey., Guven A; Department of Paediatric Endocrinology, Baskent University Medical Faculty, Istanbul Hospital, 06790 Istanbul, Turkey., Hannema SE; Department of Pediatric Endocrinology, Amsterdam UMC location Vrije Universiteit Amsterdam, 1007 MB Amsterdam, The Netherlands., Iotova V; Department of Paediatrics, Medical University-Varna, UMHAT 'Sv. Marina,'  9002 Varna, Bulgaria., Konrad D; Department of Endocrinology and Diabetology, University Children's Hospital Zurich, University of Zurich, 8032 Zurich, Switzerland., Lenherr-Taube N; Department of Endocrinology and Diabetology, University Children's Hospital Zurich, University of Zurich, 8032 Zurich, Switzerland., Krone NP; Department of Oncology and Metabolism, University of Sheffield, Sheffield S10 2RX, UK., Leka-Emiri S; Department of Endocrinology-Growth and Development, 'P&A Kyriakou' Children's Hospital, Athens 115 27, Greece., Vlachopapadopoulou E; Department of Endocrinology-Growth and Development, 'P&A Kyriakou' Children's Hospital, Athens 115 27, Greece., Lichiardopol C; Department of Endocrinology, University of Medicine and Pharmacy Craiova, University Emergency Hospital, Craiova 200349, Romania., Marginean O; Department of Paediatrics, University of medicine and Pharmacy 'Victor Babes', Clinical children emergency hospital 'Louis Turcanu' Timisora, Timisora 300011, Romania., Markosyan R; Department of Endocrinology, YSMU, Yerevan 0025, Armenia., Neumann U; Department of Paediatric Endocrinology and Diabetology, Centre for Chronic Sick Children, Institute for Experimental Paediatric Endocrinology, Charité-Universitätsmedizin Berlin, 10117 Berlin, Germany., Niedziela M; Department of Pediatric Endocrinology and Rheumatology, Institute of Pediatrics, Poznan University of Medical Sciences, 61 701 Poznan, Poland., Banaszak-Ziemska M; Department of Pediatric Endocrinology and Rheumatology, Institute of Pediatrics, Poznan University of Medical Sciences, 61 701 Poznan, Poland., Phan-Hug F; Paediatric Endocrinology, EHC-Morges, 1110 Morges, Switzerland.; Paediatric Endocrinology, Hospital Valais-Sion, 1950 Sion, Switzerland., Poyrazoglu S; Istanbul Faculty of Medicine, Department of Paediatrics, Paediatric Endocrinology Unit, Istanbul University, 34093 Çapa, Istanbul, Turkey., Probst-Scheidegger U; Department of Paediatric Endocrinology, Kantonsspital Winterthur, 8401 Zurich, Switzerland., Randell T; Department of Paediatric Endocrinology, Nottingham Children's Hospital, Nottingham NG7 2UH, UK., Russo G; Department of Paediatrics, Endocrine Unit, RCCS Ospedale San Raffaele, 20132 Milan, Italy., Salerno M; Department of Pediatrics, Unit of Immunology, Federico II University, 80131 Naples, Italy., Seneviratne S; Department of Paediatrics, Faculty of Medicine, University of Colombo, Colombo 00700, Sri Lanka., Shnorhavorian M; Department of Urology, University of Washington School of Medicine, Seattle Children's Hospital, Seattle, WA 98105, USA., Thankamony A; Department of Paediatrics,University of Cambridge, Cambridge CB2 2QQ, UK., Tadokoro-Curraro R; Department of Paediatrics,University of Cambridge, Cambridge CB2 2QQ, UK., van den Akker E; Division of Pediatric Endocrinology, Department of Pediatrics, Erasmus MC-Sophia Children's Hospital, 3015 CN Rotterdam, The Netherlands., van Eck J; Division of Pediatric Endocrinology, Department of Pediatrics, Erasmus MC-Sophia Children's Hospital, 3015 CN Rotterdam, The Netherlands., Vieites A; Centro de Investigaciones Endocrinológicas, División de Endocrinología, Hospital de Niños Ricardo Gutiérrez, 1425 Buenos Aires, Argentina., Wasniewska M; Pediatric Unit, Department of Human Pathology of Adulthood and Childhood, University of Messina, 98166 Messina, Italy., Ahmed SF; Developmental Endocrinology Research Group, School of Medicine, Dentistry & Nursing, University of Glasgow, Glasgow G51 4TF, UK.; Office for Rare Conditions, Royal Hospital for Children & Queen Elizabeth University Hospital, Glasgow G51 4TF, UK.
Jazyk:	angličtina
Zdroj:	Journal of the Endocrine Society [J Endocr Soc] 2024 Aug 20; Vol. 8 (10), pp. bvae145. Date of Electronic Publication: 2024 Aug 20 (Print Publication: 2024).
DOI:	10.1210/jendso/bvae145
Abstrakt:	Background: It is unclear whether targeted monitoring of acute adrenal insufficiency (AI) related adverse events (AE) such as sick day episodes (SDEs) and hospitalization rate in congenital adrenal hyperplasia (CAH) is associated with a change in the occurrence of these events. Aim: Study temporal trends of AI related AE in the I-CAH Registry. Methods: In 2022, data on the occurrence of AI-related AE in children aged <18 years with 21-hydroxylase deficiency CAH were compared to data collected in 2019. Results: In 2022, a total of 513 children from 38 centers in 21 countries with a median of 8 children (range 1-58) per center had 2470 visits evaluated over a 3-year period (2019-2022). The median SDE per patient year in 2022 was 0 (0-2.5) compared to 0.3 (0-6) in 2019 ( P = .01). Despite adjustment for age, CAH phenotype and duration of study period, a difference in SDE rate was still apparent between the 2 cohorts. Of the 38 centers in the 2022 cohort, 21 had also participated in 2019 and a reduction in SDE rate was noted in 13 (62%), an increase was noted in 3 (14%), and in 5 (24%) the rate remained the same. Of the 474 SDEs reported in the 2022 cohort, 103 (22%) led to hospitalization compared to 299 of 1099 SDEs (27%) in the 2019 cohort ( P = .02). Conclusion: The I-CAH Registry can be used for targeted monitoring of important clinical benchmarks in CAH. However, changes in reported benchmarks need careful interpretation and longer-term monitoring. (© The Author(s) 2024. Published by Oxford University Press on behalf of the Endocrine Society.)
Databáze:	MEDLINE
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