ArticleInternational journal of hematology2013
Therapeutic outcome of multifocal Langerhans cell histiocytosis in adults treated with the Special C regimen formulated by the Japan LCH Study Group.
Article in International journal of hematology, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 19 papers.
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Who cites it
19 citing papers in PubMed, 39 citations in OpenAlex.
- Delayed Neurologic Response to Dabrafenib and Trametinib in the Case of Mixed Histiocytosis (LCH/ECD): Case Report and Literature Review.Reports (MDPI) · 2026Article
- Trametinib for a child with refractory Rosai-Dorfman-Destombes disease harboring a novel somatic mutation in MAP2K1.International journal of hematology · 2024Article
- Clinical and prognostic features of Langerhans cell histiocytosis in adults.Cancer science · 2023Article
- Langerhans cell histiocytosis in adolescent patients: a single-centre retrospective study.Orphanet journal of rare diseases · 2022Article
- Article
- A case of langerhans cell histiocytosis of the mandible that spontaneously regressed after biopsy in a child.Clinical case reports · 2021Article
- Treatment Outcomes of Langerhans Cell Histiocytosis: A Retrospective Study.Medicina (Kaunas, Lithuania) · 2021Article
- Central diabetes insipidus developing in a 6-year-old patient 4 years after the remission of unifocal bone Langerhans cell histiocytosis.Clinical pediatric endocrinology : case reports and clinical investigations : official journal of the Japanese Society for Pediatric Endocrinology · 2021Article
- Gastrointestinal lesion in adult-onset Langerhans cell histiocytosis.International journal of clinical oncology · 2020Article
- Clinical features and outcomes of adult Langerhans cell histiocytosis: a single-center experience.International journal of hematology · 2020Article
- Update on Pulmonary Langerhans Cell Histiocytosis.Frontiers in medicine · 2020Review
- Langerhans cell histiocytosis in adults: Advances in pathophysiology and treatment.Cancer science · 2018Review
- Ninety-year-old man with hypereosinophilia, lymphadenopathies and pruritus.BMJ case reports · 2018Article
- Vinblastine chemotherapy in adult patients with langerhans cell histiocytosis: a multicenter retrospective study.Orphanet journal of rare diseases · 2017Article
- An unusual case of adult-onset multi-systemic Langerhans cell histiocytosis with perianal and incident thyroid involvement.Endocrinology, diabetes & metabolism case reports · 2017Article
- High efficacy of the MACOP-B regimen in the treatment of adult Langerhans cell histiocytosis, a 20 year experience.BMC cancer · 2015Article
- Successful treatment of adult Langerhans cell histiocytosis with intensified chemotherapy.International journal of hematology · 2015Article
- Successful treatment of histiocytic sarcoma with induction chemotherapy consisting of dose-escalated CHOP plus etoposide and upfront consolidation auto-transplantation.International journal of hematology · 2014Article
- Adult Onset of BRAF(V600E)-Mutated Langerhans Cell Histiocytosis with Cutaneous Involvement Successfully Diagnosed by Immunohistochemical Staining.Case reports in dermatologyArticle
Corrections and comments
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Authors and funding
12 authors at 8 institutions in 3 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Little information is available regarding effective systemic therapies for adult Langerhans cell histiocytosis (LCH). The Japan LCH Study Group has formulated an ambulatory treatment regimen for adult patients with LCH. In total, 14 patients (median age 43 years, range 20-70 years) with multifocal LCH with biopsy-confirmed histology were enrolled. None had received cytoreductive agents for LCH previously. Four had single system (SS) and ten had multi system (MS) disease. All were treated with the Special C regimen, which consists of vinblastine/prednisolone and methotrexate with daily 6-mercaptopurine for 36 weeks. At the end of the therapeutic regimen, all SS patients achieved no active disease (NAD), and six of the ten MS patients showed a response (NAD in two, partial response in four). At the last follow-up (median 34 months), 11 patients were alive (NAD in eight and active disease in three). Of the three deceased, one died of hemorrhage during the Special C treatment, and two of infections during subsequent therapy. Although this study is limited by the small sample size, this ambulatory regimen shows signs of efficacy for adult LCH. This was particularly evident for patients with multifocal SS disease, but half of those with MS disease also benefited.
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