ArticlePloS one2012
Optimal therapy for adults with Langerhans cell histiocytosis bone lesions.
Article in PloS one, 2012. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 57 papers.
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Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
57 citing papers in PubMed, 126 citations in OpenAlex.
- Clofarabine for relapsed/refractory LCH and non-LC histiocytosis.Blood advances · 2026Trial
- Phase 2 study using low dose cytarabine for adult patients with newly diagnosed Langerhans cell histiocytosis.Leukemia · 2024Trial
- Trial
- Isolated Esophageal Langerhans Cell Histiocytosis in an Adult: A Case Report.Clinical case reports · 2026Article
- What to do to beat Langerhans cell histiocytosis of bone? A narrative review and case series of radiofrequency ablation.EFORT open reviews · 2026Review
- [Chinese expert consensus on the diagnosis and treatment of adult Langerhans cell histiocytosis (2025)].Zhonghua xue ye xue za zhi = Zhonghua xueyexue zazhi · 2025Article
- [A case of adult temporal bone langerhans cell histiocytosis presenting as posterior canal dehiscence syndrome and literature review].Lin chuang er bi yan hou tou jing wai ke za zhi = Journal of clinical otorhinolaryngology head and neck surgery · 2025Review
- Case Report: Systematic endoscopic characterization of synchronous esophageal, gastric, and colorectal involvement in multisystem Langerhans cell histiocytosis.Frontiers in medicine · 2025Article
- Management of Spinal Langerhans Cell Histiocytosis in Children: A Systematic Review.International journal of spine surgery · 2024Article
- Solitary Skull Langerhans Cell Histiocytosis Presenting With a Pus Draining Fistula: An Unusual Presentation and Review of Literature.Brain tumor research and treatment · 2024Article
- Mutant PIK3CA is a targetable driver alteration in histiocytic neoplasms.Blood advances · 2023Article
- Clinical characteristics, genomic profiling and outcomes of single system multifocal Langerhans cell histiocytosis in adults with bone involvement.Blood cancer journal · 2023Article
- Cladribine and cytarabine in children refractory high risk multisystem Langerhans cell histiocytosis.Heliyon · 2023Article
- Clinical and prognostic features of Langerhans cell histiocytosis in adults.Cancer science · 2023Article
- Adult-onset Langerhans cell histiocytosis - A Trojan horse of oral cavity: A case report with rare clinical presentation.Journal of oral and maxillofacial pathology : JOMFP · 2023Article
- Langerhans cell histiocytosis involving only the thymus in an adult: A case report.World journal of clinical cases · 2022Article
- Langerhans cell histiocytosis in adolescent patients: a single-centre retrospective study.Orphanet journal of rare diseases · 2022Article
- Article
- Adult Langerhans Cell Histiocytosis and the Skeleton.Journal of clinical medicine · 2022Review
- Histiocytic disorders.Nature reviews. Disease primers · 2021Review
Corrections and comments
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Authors and funding
6 authors at 4 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundThere is little data on treatment of Langerhans cell histiocytosis (LCH) in adults. Available data is on small numbers of patients with short follow-up times and no comparison of results from different treatment regimens. We analyzed the responses of adult LCH patients with bone lesions to three primary chemotherapy treatments to define the optimal one. METHODS AND
findingsFifty-eight adult patients with bone lesions, either as a solitary site or as a component of multisystem disease, were analyzed for disease location and response to surgery, curettage, steroids, radiation, vinblastine/prednisone, 2-Chlorodeoxyadenosine (2-CdA), or cytosine arabinoside (ARA-C). The mean age of patients was 32 years, with equal gender distribution. Twenty-nine patients had 1 lesion; 16, 2 lesions; 5, 3 lesions; and 8 had 4 or more. Most bone lesions were in the skull, spine, or jaw. Chemotherapy, surgery, curettage, or radiation, but not steroids alone, achieved improvement or resolution of lesions in a majority of patients. Comparison of the three chemotherapy regimens revealed 84% of patients treated with vinblastine/prednisone either did not respond or relapsed within a year, whereas 59% of patients treated with 2-CdA and 21% treated with ARA-C failed. Toxicity was worse with the vinblastine/prednisone group as 75% had grade 3-4 neuropathy. Grade 3-4 cytopenias occurred in 37% of the 2-CdA -treated patients and 20% of the ARA-C-treated patients. The major limitation of this study is it is retrospective and not a clinical trial.
conclusionsARA-C is an effective and minimally toxic treatment for LCH bone lesions in adults. In contrast, vinblastine/prednisone results in poor overall responses and excessive toxicity.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.