Lupus-associated hypoprothrombinemia syndrome in children: Differences between post-infectious and autoimmune forms

Thromb Res. 2025 Jan:245:109231. doi: 10.1016/j.thromres.2024.109231. Epub 2024 Nov 24.

Abstract

Introduction: Lupus-anticoagulant hypoprothrombinemia syndrome (LAHS) is a rare but potentially serious condition. LAHS can be of post-infectious (PI) or autoimmune (AI) origin. However, there is currently no clear data available on the differences between these two forms.

Method: A retrospective multicenter study of cases in France was performed, followed by a review of cases in the literature.

Result: A total of 84 patients were included in the study. Seventeen patients were selected from the French cohort, and 67 were selected from a systematic review of the literature. 95 % of patients presented with hemorrhagic symptoms, with nearly half of these cases being severe. PI or AI context was identified in 33 % and 53 % of cases. 54 % of patients were treated with corticosteroids, and 30 % received immunomodulatory therapy. Thrombopenia and lower factor V were associated with a higher risk of bleeding. The AI group consisted of older children and exhibited significantly more severe bleeding (p < 0.001). The treatment was more frequent and intensive, and the relapse rate was higher in the AI group (p < 0.001).

Conclusion: Post-infectious forms are transient and associated with a low risk of serious hemorrhage. The treatment must be adapted according to the clinical and biological context.

Keywords: Acquired coagulation disorder; Child; Children; Factor II inhibitor; Lupus anticoagulant; Lupus-associated hypoprothrombinemia.

Publication types

  • Multicenter Study
  • Systematic Review

MeSH terms

  • Adolescent
  • Autoimmune Diseases / blood
  • Autoimmune Diseases / complications
  • Child
  • Child, Preschool
  • Female
  • Hemorrhage / blood
  • Hemorrhage / etiology
  • Humans
  • Hypoprothrombinemias* / blood
  • Hypoprothrombinemias* / complications
  • Infant
  • Lupus Coagulation Inhibitor / blood
  • Lupus Erythematosus, Systemic / blood
  • Lupus Erythematosus, Systemic / complications
  • Male
  • Retrospective Studies

Substances

  • Lupus Coagulation Inhibitor