ArticleThe oncologist2026
Watchful waiting in children with unifocal osseous Langerhans cell histiocytosis.
Article in The oncologist, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
backgroundLangerhans cell histiocytosis (LCH) is a rare inflammatory neoplasm most commonly involving bone. However, data on single-system, single-site LCH (SS-s-LCH), particularly with unifocal bone disease, remain limited. Management strategies for unifocal osseous LCH vary globally, and the safety of a watchful-waiting approach is not well-defined. This study aimed to evaluate whether patients with osseous SS-s-LCH can be managed with observation alone without compromising long-term outcomes.
methodsIn this retrospective, multicenter study, we enrolled children with biopsy-proven osseous SS-s-LCH from 7 centers in the Chinese Children's Histiocytosis Group, excluding those with central nervous system (CNS)-risk bone lesions. The primary endpoint was 3-year event-free survival (EFS). Events were defined as disease progression or relapse.
resultsThe cohort included 134 patients with a median follow-up of 36.0 months. Involved sites were cranial bones (n = 37), vertebrae (n = 33), and other bones (n = 64). At final follow-up, 16 patients experienced an event, yielding a 3-year EFS of 88.1% (95% CI: 81.5%-92.5%). Events included new lesions (n = 14) and primary lesion expansion (n = 2), with 93.8% (15/16) occurring within the first 12 months after diagnosis. EFS did not differ significantly by involved site or mutation status. However, patients staged by PET-CT at diagnosis had a significantly higher 3-year EFS (97.6%) than those staged by conventional imaging (83.7%) (P < .05). Long-term quality of life was satisfactory in all assessed patients.
conclusionsWatchful waiting is a safe and effective management strategy for children with unifocal osseous SS-s-LCH. Initial staging with PET-CT may be associated with superior EFS, highlighting its potential value in refining risk assessment.
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