Antinuclear antibody–associated autoimmune cytopenia in childhood is a risk factor for systemic lupus erythematosus

Author:

Granel Jérôme12,Fernandes Helder1,Bader-Meunier Brigitte3,Guth Amandine4,Richer Olivier1,Pillet Pascal1,Leverger Guy25,Ducassou Stéphane12,Fahd Mony26,Pasquet Marlène7ORCID,Garnier Nathalie8,Barlogis Vincent9ORCID,Guitton Corinne10ORCID,Jeziorski Eric11ORCID,Thomas Caroline12,Bayart Sophie13,Cheikh Nathalie14ORCID,Paillard Catherine15,Abou Chahla Wadih16,Chastagner Pascal17,Neven Bénédicte3,Millot Frédéric18,Lejeune Julien19,Li-Thiao Te Valérie20,Armari-Alla Corinne21,Briandet Claire22,Carausu Liana23,Deparis Marianna24,Piguet Christophe25,Benadiba Joy26,Marie-Cardine Aude27,Stephan Jean-Louis28,Pellier Isabelle29ORCID,Pluchart Claire30,Doré Eric31,Michaux Katell32,Héritier Sébastien25ORCID,Leblanc Thierry26,Aladjidi Nathalie12ORCID

Affiliation:

1. 1Paediatric Clinical Immunology, Pellegrin Hospital, CIC1401, INSERM CICP, Bordeaux University Hospital, Bordeaux, France

2. 2Centre de Référence National des Cytopénies Auto-immunes de l’Enfant, Bordeaux, France

3. 3Paediatric Haematology-Immunology and Rheumatology Department, Hôpital Necker-Enfants Malades, Assistance Publique–Hôpitaux de Paris, Paris, France

4. 4Paediatric Department, Pontarlier Hospital, Pontarlier, France

5. 5Paediatric Oncology Immunology Haematology Unit, Armand-Trousseau University Hospital, Assistance Publique–Hôpitaux de Paris, Paris, France

6. 6Paediatric Haematology and Immunology Unit, Robert-Debré University Hospital, Assistance Publique–Hôpitaux de Paris, Paris, France

7. 7Paediatric Oncology Immunology Haematology Unit, Children’s University Hospital, Toulouse, France

8. 8Institute of Paediatric Haematology and Oncology, Hospices Civils de Lyon, Lyon, France

9. 9Department of Paediatric Haematology, La Timone Hospital, Marseille University Hospital, Marseille, France

10. 10Department of Paediatrics, Bicêtre University Hospital, Assistance Publique–Hôpitaux de Paris, Le Kremlin-Bicêtre, France

11. 11Paediatric Oncology Haematology Unit, Arnaud de Villeneuve University Hospital, Montpellier, France

12. 12Paediatric Haematology Unit, Nantes University Hospital, Nantes, France

13. 13Paediatric Haematology Unit, Rennes University Hospital, Rennes, France

14. 14Department of Paediatric Haematology-Oncology, Besançon University Hospital, Besançon, France

15. 15Department of Paediatric Haematology and Oncology, Hautepierre University Hospital, Strasbourg, France

16. 16Department of Paediatric Haematology, Jeanne de Flandre Hospital, Lille University Hospital, Lille, France

17. 17Department of Paediatric Haematology and Oncology, Children's University Hospital, Nancy, France

18. 18Department of Paediatric Haematology, Poitiers University Hospital, Poitiers, France

19. 19Department of Paediatric Haematology-Oncology, Clocheville Hospital, Tours University Hospital, Tours, France

20. 20Department of Paediatric Haematology/Oncology, Amiens University Hospital, Amiens, France

21. 21Paediatric Haematology-Oncology Department, Grenoble University Hospital, Grenoble, France

22. 22Department of Paediatrics, Dijon University Hospital, Dijon, France

23. 23Department of Paediatric Hematology, CHU de Brest, Brest, France

24. 24Paediatric Oncology- Haematology Unit Department, Caen University Hospital, Caen, France

25. 25Paediatric Oncology Hematology Unit, Limoges University Hospital, Limoges, France

26. 26Department of Haematology-Oncology Paediatrics, Nice University Hospital, Nice, France

27. 27Department of Paediatric Haematology and Oncology, Rouen University Hospital, Rouen, France

28. 28University Hospital of Saint Etienne, North Hospital, Department of Paediatric Oncology, Saint Etienne, France

29. 29Paediatric Unit, Angers University Hospital, Angers, France

30. 30Paediatric Haematology-Oncology Unit, Institut Jean Godinot, Reims University Hospital, Reims, France

31. 31Paediatric Unit, Clermont-Ferrand University Hospital, Clermont-Ferrand, France

32. 32Paediatric Unit, Martinique University Hospital, Fort-de-France, France

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.

Publisher

American Society of Hematology

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