ArticleJournal of hematology2026
Patterns and Outcomes of Recurrence in Pediatric Langerhans Cell Histiocytosis.
Article in Journal of hematology, 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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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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5 authors.
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Abstract
Background: Langerhans cell histiocytosis (LCH) is a rare neoplasm with a heterogeneous clinical spectrum ranging from isolated lesions to multisystem disease, with recurrence reported in up to 50-60% of patients. Data on outcomes of recurrent LCH from resource-limited settings remain limited. Methods: We report a 12-year retrospective review of children (1-18 years) with biopsy-proven relapsed LCH treated between January 2014 and December 2025. Clinical characteristics, recurrence patterns, treatment, and long-term outcomes were analyzed. All patients had received first-line therapy according to LCH-III protocol. Salvage treatments included cladribine-based chemotherapy, lenalidomide-dexamethasone (Len-Dex), or surgery for isolated lesions. Follow-up was updated to December 31, 2025. Results: Seven patients with a median age of 22 months at diagnosis (range 6-57) were included. Four had risk-organ involvement at presentation. Diabetes insipidus was present in 4/7 (57%). The median time to first recurrence was 25.8 months (range 6-104). Recurrence involved bone alone in four patients and bone with skin in three patients; none had risk-organ involvement at relapse. Three patients experienced multiple recurrences. Salvage therapy included cladribine-based chemotherapy (n = 6), Len-Dex (n = 2), and surgery (n = 1). At a median follow-up of 114 months (range 63-169), overall and event-free survival were 100%. Long-term sequelae included persistent diabetes insipidus (3/4), sclerosing cholangitis (n = 1), and sensorineural hearing loss (n = 1). Conclusions: Children with recurrent LCH in this cohort demonstrated good long-term outcomes irrespective of the salvage treatment strategy. Len-Dex represents a feasible, cost-effective therapeutic option in resource-limited settings where access to targeted therapy is limited.
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