دورية أكاديمية

Clinical and pathological evaluation of cladribine treatment response in a case series of patients with xanthoma disseminatum.

التفاصيل البيبلوغرافية
العنوان: Clinical and pathological evaluation of cladribine treatment response in a case series of patients with xanthoma disseminatum.
المؤلفون: Zhou EY; Department of Dermatology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Yu F; Department of Hematology and Oncology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Wang Y; Department of Hematology and Oncology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Bi Y; Department of Dermatology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Tuan H; Department of Dermatology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Huang Y; Department of Hematology and Oncology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Wang B; Department of Hematology and Oncology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Li J; Department of Hematology and Oncology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Yin H; Department of Pathology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Li L; Department of Hematology and Oncology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China., Zhao Y; Department of Dermatology, Beijing Tsinghua Changgung Hospital, School of Clinical Medicine, Tsinghua University, Beijing, China, Photomedicine Laboratory, Institute of Precision Medicine, Tsinghua University, Beijing, China.
المصدر: European journal of dermatology : EJD [Eur J Dermatol] 2023 Jun 01; Vol. 33 (3), pp. 270-279.
نوع المنشور: Review; Journal Article
اللغة: English
بيانات الدورية: Publisher: John Libbey Eurotext Country of Publication: France NLM ID: 9206420 Publication Model: Print Cited Medium: Internet ISSN: 1952-4013 (Electronic) Linking ISSN: 11671122 NLM ISO Abbreviation: Eur J Dermatol Subsets: MEDLINE
أسماء مطبوعة: Original Publication: Montrouge, France : John Libbey Eurotext, c1991-
مواضيع طبية MeSH: Cladribine*/therapeutic use , Histiocytosis, Non-Langerhans-Cell*/drug therapy, Humans ; Female ; Male ; Retrospective Studies ; Antimetabolites
مستخلص: Background: Xanthoma disseminatum (XD) is a rare form of non-Langerhans histiocytosis with extensive cutaneous involvement. There is a paucity of evidence-based recommendations for treatment decision-making. Previous case reports have established purine analogues, especially cladribine, as a hopeful first-line treatment option, but characterization of the clinical and pathological responses is lacking.
Objectives: To characterize the clinical and pathological responses to cladribine monotherapy based on serial examinations in XD patients.
Materials & Methods: We retrospectively studied the clinical, pathological and laboratory data in a cohort of five XD patients who received intravenous cladribine monotherapy with serial examinations in our hospital. Compared with baseline characteristics, changes in clinical features and pathological patterns were identified and analysed. We also conducted a literature review of reported cases of cladribine treatment in XD patients.
Results: Four male and one female patient were involved in the study. All patients demonstrated satisfactory clinical responses to cladribine monotherapy after 5 to 10 cycles. We observed a pathological shift in pattern from classic xanthogranuloma to transitional fibrohistiocytic infiltration during the treatment, and pathological responses heralded persistent clinical improvement. Other than afebrile neutropenia, no prominent adverse events were identified. Sustainable lesion clearance was achieved in all five patients during the follow-up period, ranging from 19 to 66 months.
Conclusion: Cladribine monotherapy is an effective and well-tolerated therapeutic option for XD patients. Pathological transformation is a signature of the clinical response and possibly unveils the underlying histiocyte biology of diseases in the xanthogranuloma family.
فهرسة مساهمة: Keywords: chemotherapy; cladribine; juvenile xanthogranuloma; non-Langerhans histiocytosis cell; xanthoma disseminatum
المشرفين على المادة: 47M74X9YT5 (Cladribine)
0 (Antimetabolites)
تواريخ الأحداث: Date Created: 20230818 Date Completed: 20230821 Latest Revision: 20230821
رمز التحديث: 20230821
DOI: 10.1684/ejd.2023.4502
PMID: 37594335
قاعدة البيانات: MEDLINE
الوصف
تدمد:1952-4013
DOI:10.1684/ejd.2023.4502