Langerhans cell histiocytosis with central nervous system involvement-complete response to 2-chlorodeoxyadenosine after failure of tyrosine kinase inhibitor therapies with sorafenib and imatinib

2011 ◽  
Vol 30 (2) ◽  
pp. 101-104 ◽  
Author(s):  
Michael Baumann ◽  
Thomas Cerny ◽  
Andreas Sommacal ◽  
Dieter Koeberle
Keyword(s):  
Central Nervous System ◽  
Nervous System ◽  
Tyrosine Kinase ◽  
Tyrosine Kinase Inhibitor ◽  
Langerhans Cell Histiocytosis ◽  
Kinase Inhibitor ◽  
Central Nervous System Involvement ◽  
Complete Response ◽  
Langerhans Cell ◽  
System Involvement

Langerhans cell histiocytosis with central nervous system involvement: Follow-up by FDG-PET during treatment with cladribine

2004 ◽  
Vol 44 (3) ◽  
pp. 286-288 ◽  
Author(s):  
Tomas Büchler ◽  
Libor Cervinek ◽  
Otakar Belohlavek ◽  
Iva Kantorova ◽  
Marek Mechl ◽  
...  
Keyword(s):  
Central Nervous System ◽  
Nervous System ◽  
Fdg Pet ◽  
Langerhans Cell Histiocytosis ◽  
Central Nervous System Involvement ◽  
Langerhans Cell ◽  
System Involvement

scholarly journals Results of TETimaX Trial of Langerhans Cell Histiocytosis Treatment and Perspectives on the Role of Imatinib Mesylate in the Era of MAPK Signaling

Biomedicines ◽  
2021 ◽  
Vol 9 (12) ◽  
pp. 1759
Author(s):  
Liliana Montella ◽  
Margaret Ottaviano ◽  
Vittorio Riccio ◽  
Fernanda Picozzi ◽  
Gaetano Facchini ◽  
...  
Keyword(s):  
Tyrosine Kinase ◽  
Tyrosine Kinase Inhibitor ◽  
Langerhans Cell Histiocytosis ◽  
Kinase Inhibitor ◽  
Growth Factor Receptor ◽  
Mapk Signaling ◽  
Langerhans Cell ◽  
Mitogen Activated Protein Kinase ◽  
Mitogen Activated Protein

Langerhans cell histiocytosis (LCH) is a rare disease that has a variable clinical presentation and unpredictable behavior. Until recently, therapeutic options were limited. Insights into the role of mitogen-activated protein kinase (MAPK) signaling have allowed the increased use of targeted treatments. Before the advent of drugs that interfere with this pathway, investigations concerning the tyrosine kinase inhibitor imatinib opened the way to a rationale-based therapeutic approach to the disease. Imatinib block the binding site of ATP in the BCR/ABL protein and is also a platelet-derived growth factor receptor (PDGFR) and a KIT (CD117) kinase inhibitor. A case of refractory LCH with brain involvement was reported to be successfully treated with imatinib. Thereafter, we further explored the role of this tyrosine kinase inhibitor. The present study is composed of an immunohistochemical evaluation of PDGFRβ expression and a clinical evaluation of imatinib in a series of LCH patients. In the first part, a series of 10 samples obtained from LCH patients was examined and a strong immunohistochemistry expression of PDGFRβ was found in 40% of the cases. In the clinical part of the study, five patients were enrolled. Long-lasting disease control was obtained. These results may suggest a potential role for this drug in the current age.

scholarly journals Phase I/II study of tirabrutinib, a second-generation Bruton’s tyrosine kinase inhibitor, in relapsed/refractory primary central nervous system lymphoma

2020 ◽  
Author(s):  
Yoshitaka Narita ◽  
Motoo Nagane ◽  
Kazuhiko Mishima ◽  
Yasuhito Terui ◽  
Yoshiki Arakawa ◽  
...  
Keyword(s):  
Central Nervous System ◽  
Nervous System ◽  
Tyrosine Kinase ◽  
Phase I ◽  
Tyrosine Kinase Inhibitor ◽  
Second Generation ◽  
Kinase Inhibitor ◽  
Central Nervous System Lymphoma ◽  
Bruton’S Tyrosine Kinase ◽  
Bruton's Tyrosine Kinase

Abstract Background The safety, tolerability, efficacy, and pharmacokinetics of tirabrutinib, a second-generation, highly selective oral Bruton’s tyrosine kinase inhibitor, were evaluated for relapsed/refractory primary central nervous system lymphoma (PCNSL). Methods Patients with relapsed/refractory PCNSL, Karnofsky performance status ≥70, and normal end-organ function received tirabrutinib 320 and 480 mg once daily (q.d.) in phase I to evaluate dose-limiting toxicity (DLT) within 28 days using a 3 + 3 dose escalation design and with 480 mg q.d. under fasted conditions in phase II. Results Forty-four patients were enrolled; 20, 7, and 17 received tirabrutinib at 320, 480, and 480 mg under fasted conditions, respectively. No DLTs were observed, and the maximum tolerated dose was not reached at 480 mg. Common grade ≥3 adverse events (AEs) were neutropenia (9.1%), lymphopenia, leukopenia, and erythema multiforme (6.8% each). One patient with 480 mg q.d. had grade 5 AEs (pneumocystis jirovecii pneumonia and interstitial lung disease). Independent review committee assessed overall response rate (ORR) at 64%: 60% with 5 complete responses (CR)/unconfirmed complete responses (CRu) at 320 mg, 100% with 4 CR/CRu at 480 mg, and 53% with 6 CR/CRu at 480 mg under fasted conditions. Median progression-free survival was 2.9 months: 2.1, 11.1, and 5.8 months at 320, 480, and 480 mg under fasted conditions, respectively. Median overall survival was not reached. ORR was similar among patients harboring CARD11, MYD88, and CD79B mutations, and corresponding wild types. Conclusion These data indicate favorable efficacy of tirabrutinib in patients with relapsed/refractory PCNSL. Trial registration JapicCTI-173646.

scholarly journals Topotecan Central Nervous System Penetration Is Altered by a Tyrosine Kinase Inhibitor

2006 ◽  
Vol 66 (23) ◽  
pp. 11305-11313 ◽  
Author(s):  
Yanli Zhuang ◽  
Charles H. Fraga ◽  
K. Elaine Hubbard ◽  
Nikolaus Hagedorn ◽  
John C. Panetta ◽  
...  
Keyword(s):  
Central Nervous System ◽  
Nervous System ◽  
Tyrosine Kinase ◽  
Tyrosine Kinase Inhibitor ◽  
Kinase Inhibitor ◽  
Central Nervous System Penetration

scholarly journals Solitary tibial lesion as the initial presentation of Langerhans cell histiocytosis: report of two cases and literature review

2021 ◽  
Vol 49 (1) ◽  
pp. 030006052098282
Author(s):  
Chih-Yang Lin ◽  
Chia-Che Lee ◽  
Kuan-Wen Wu ◽  
Chang-Tsu Yuan ◽  
Ken-Nan Kuo ◽  
...  
Keyword(s):  
Central Nervous System ◽  
Nervous System ◽  
Langerhans Cell Histiocytosis ◽  
Paediatric Oncology ◽  
Bone Diseases ◽  
Langerhans Cell ◽  
Oncology Group ◽  
Preventive Chemotherapy ◽  
System Risk ◽  
Risk Patients

The various presentations of osseous Langerhans cell histiocytosis (LCH) make it difficult to distinguish from other bone diseases. In addition, there is no universally accepted protocol for managing osseous LCH for single non-central nervous system-risk lesions. Here, the rare cases of two paediatric patients, aged 1 and 2 years, who presented with a solitary tibial lesion at time of LCH diagnosis, are reported. One patient progressed to multiple lesions after curettage of the original lesion. Subsequently, both patients received preventive chemotherapy using the Taiwan Paediatric Oncology Group (TPOG) revised protocol for treating low risk patients with LCH, namely, TPOG LCH2002-LR. After receiving this treatment, which included a schedule of prednisolone and vincristine for 6 weeks, followed by prednisolone, vincristine and 6-mercaptopurine for a further 48 weeks, both patients are free from recurrence or progression.

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