학술논문
Antinuclear antibody-associated autoimmune cytopenia in childhood is a risk factor for systemic lupus erythematosus.
Document Type
Academic Journal
Author
Granel J; Paediatric Clinical Immunology, Pellegrin Hospital, CIC1401, INSERM CICP, Bordeaux University Hospital, Bordeaux, France.; Centre de Référence National des Cytopénies Auto-immunes de l'Enfant, Bordeaux, France.; Fernandes H; Paediatric Clinical Immunology, Pellegrin Hospital, CIC1401, INSERM CICP, Bordeaux University Hospital, Bordeaux, France.; Bader-Meunier B; Paediatric Haematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, Assistance Publique-Hôpitaux de Paris, Paris, France.; Guth A; Paediatric Department, Pontarlier Hospital, Pontarlier, France.; Richer O; Paediatric Clinical Immunology, Pellegrin Hospital, CIC1401, INSERM CICP, Bordeaux University Hospital, Bordeaux, France.; Pillet P; Paediatric Clinical Immunology, Pellegrin Hospital, CIC1401, INSERM CICP, Bordeaux University Hospital, Bordeaux, France.; Leverger G; Centre de Référence National des Cytopénies Auto-immunes de l'Enfant, Bordeaux, France.; Paediatric Oncology Immunology Haematology Unit, Armand-Trousseau University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.; Ducassou S; Paediatric Clinical Immunology, Pellegrin Hospital, CIC1401, INSERM CICP, Bordeaux University Hospital, Bordeaux, France.; Centre de Référence National des Cytopénies Auto-immunes de l'Enfant, Bordeaux, France.; Fahd M; Centre de Référence National des Cytopénies Auto-immunes de l'Enfant, Bordeaux, France.; Paediatric Haematology and Immunology Unit, Robert-Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.; Pasquet M; Paediatric Oncology Immunology Haematology Unit, Children's University Hospital, Toulouse, France.; Garnier N; Institute of Paediatric Haematology and Oncology, Hospices Civils de Lyon, Lyon, France.; Barlogis V; Department of Paediatric Haematology, La Timone Hospital, Marseille University Hospital, Marseille, France.; Guitton C; Department of Paediatrics, Bicêtre University Hospital, Assistance Publique-Hôpitaux de Paris, Le Kremlin-Bicêtre, France.; Jeziorski E; Paediatric Oncology Haematology Unit, Arnaud de Villeneuve University Hospital, Montpellier, France.; Thomas C; Paediatric Haematology Unit, Nantes University Hospital, Nantes, France.; Bayart S; Paediatric Haematology Unit, Rennes University Hospital, Rennes, France.; Cheikh N; Department of Paediatric Haematology-Oncology, Besançon University Hospital, Besançon, France.; Paillard C; Department of Paediatric Haematology and Oncology, Hautepierre University Hospital, Strasbourg, France.; Abou Chahla W; Department of Paediatric Haematology, Jeanne de Flandre Hospital, Lille University Hospital, Lille, France.; Chastagner P; Department of Paediatric Haematology and Oncology, Children's University Hospital, Nancy, France.; Neven B; Paediatric Haematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, Assistance Publique-Hôpitaux de Paris, Paris, France.; Millot F; Department of Paediatric Haematology, Poitiers University Hospital, Poitiers, France.; Lejeune J; Department of Paediatric Haematology-Oncology, Clocheville Hospital, Tours University Hospital, Tours, France.; Li-Thiao Te V; Department of Paediatric Haematology/Oncology, Amiens University Hospital, Amiens, France.; Armari-Alla C; Paediatric Haematology-Oncology Department, Grenoble University Hospital, Grenoble, France.; Briandet C; Department of Paediatrics, Dijon University Hospital, Dijon, France.; Carausu L; Department of Paediatric Hematology, CHU de Brest, Brest, France.; Deparis M; Paediatric Oncology- Haematology Unit Department, Caen University Hospital, Caen, France.; Piguet C; Paediatric Oncology Hematology Unit, Limoges University Hospital, Limoges, France.; Benadiba J; Department of Haematology-Oncology Paediatrics, Nice University Hospital, Nice, France.; Marie-Cardine A; Department of Paediatric Haematology and Oncology, Rouen University Hospital, Rouen, France.; Stephan JL; University Hospital of Saint Etienne, North Hospital, Department of Paediatric Oncology, Saint Etienne, France.; Pellier I; Paediatric Unit, Angers University Hospital, Angers, France.; Pluchart C; Paediatric Haematology-Oncology Unit, Institut Jean Godinot, Reims University Hospital, Reims, France.; Doré E; Paediatric Unit, Clermont-Ferrand University Hospital, Clermont-Ferrand, France.; Michaux K; Paediatric Unit, Martinique University Hospital, Fort-de-France, France.; Héritier S; Centre de Référence National des Cytopénies Auto-immunes de l'Enfant, Bordeaux, France.; Paediatric Oncology Immunology Haematology Unit, Armand-Trousseau University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.; Leblanc T; Centre de Référence National des Cytopénies Auto-immunes de l'Enfant, Bordeaux, France.; Paediatric Haematology and Immunology Unit, Robert-Debré University Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France.; Aladjidi N; Paediatric Clinical Immunology, Pellegrin Hospital, CIC1401, INSERM CICP, Bordeaux University Hospital, Bordeaux, France.; Centre de Référence National des Cytopénies Auto-immunes de l'Enfant, Bordeaux, France.
Source
Publisher: Elsevier Country of Publication: United States NLM ID: 7603509 Publication Model: Print Cited Medium: Internet ISSN: 1528-0020 (Electronic) Linking ISSN: 00064971 NLM ISO Abbreviation: Blood Subsets: MEDLINE
Subject
Language
English
Abstract
Abstract: Autoimmune cytopenia (AIC) in children may be associated with positive antinuclear antibodies (ANA) and may progress to systemic lupus erythematosus (SLE). We evaluated the risk of progression to SLE of childhood-onset ANA-associated AIC. In the French national prospective OBS'CEREVANCE cohort, the long-term outcome of children with ANA-associated AIC (ANA titer ≥1/160) and a subgroup of children who developed SLE were described. ANA were positive in 355 of 1803 (20%) children with AIC. With a median follow-up of 5.8 (range, 0.1-29.6) years, 79 of 355 (22%) patients developed SLE at a median age of 14.5 (1.1-21.4) years; 20% of chronic immune thrombocytopenic purpura, 19% of autoimmune hemolytic anemia, and 45% of Evans syndrome. None of the patients with ANA-negative test developed SLE. Severe manifestations of SLE were observed in 21 patients, and 2 patients died. In multivariate analysis including patients with positive ANA within the first 3 months after AIC diagnosis, age >10 years at AIC diagnosis (relative risk [RR], 3.67; 95% confidence interval [CI], 1.18-11.4; P = .024) and ANA titer >1/160 (RR, 5.28; 95% CI, 1.20-23.17; P = .027) were associated with the occurrence of SLE after AIC diagnosis. ANA-associated AIC is a risk factor for progression to SLE, especially in children with an initial ANA titer >1/160 and an age >10 years at AIC diagnosis. ANA screening should be recommended in children with AIC, and patients with ANA should be monitored long-term for SLE, with special attention to the transition period. This trial was registered at www.ClinicalTrials.gov as #NCT05937828.
(© 2024 American Society of Hematology. Published by Elsevier Inc. All rights are reserved, including those for text and data mining, AI training, and similar technologies.)
(© 2024 American Society of Hematology. Published by Elsevier Inc. All rights are reserved, including those for text and data mining, AI training, and similar technologies.)