Clinical and pathological evaluation of cladribine treatment response in a case series of patients with xanthoma disseminatum (notice n° 604687)

détails MARC
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fixed length control field 02989cam a2200337 4500500
005 - DATE AND TIME OF LATEST TRANSACTION
control field 20250121155425.0
041 ## - LANGUAGE CODE
Language code of text/sound track or separate title fre
042 ## - AUTHENTICATION CODE
Authentication code dc
100 10 - MAIN ENTRY--PERSONAL NAME
Personal name Zhou, Eray Yihui
Relator term author
245 00 - TITLE STATEMENT
Title Clinical and pathological evaluation of cladribine treatment response in a case series of patients with xanthoma disseminatum
260 ## - PUBLICATION, DISTRIBUTION, ETC.
Date of publication, distribution, etc. 2023.<br/>
500 ## - GENERAL NOTE
General note 59
520 ## - SUMMARY, ETC.
Summary, etc. 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.
690 ## - LOCAL SUBJECT ADDED ENTRY--TOPICAL TERM (OCLC, RLIN)
Topical term or geographic name as entry element juvenile xanthogranuloma
690 ## - LOCAL SUBJECT ADDED ENTRY--TOPICAL TERM (OCLC, RLIN)
Topical term or geographic name as entry element cladribine
690 ## - LOCAL SUBJECT ADDED ENTRY--TOPICAL TERM (OCLC, RLIN)
Topical term or geographic name as entry element xanthoma disseminatum
690 ## - LOCAL SUBJECT ADDED ENTRY--TOPICAL TERM (OCLC, RLIN)
Topical term or geographic name as entry element chemotherapy
690 ## - LOCAL SUBJECT ADDED ENTRY--TOPICAL TERM (OCLC, RLIN)
Topical term or geographic name as entry element non-Langerhans histiocytosis cell
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Yu, Fan
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Wang, Yanying
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Bi, Yalan
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Tuan, Hsiaohan
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Huang, Yuehua
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Wang, Bianhong
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Li, Jingxian
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Yin, Hongfang
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Li, Lihong
Relator term author
700 10 - ADDED ENTRY--PERSONAL NAME
Personal name Zhao, Yi
Relator term author
786 0# - DATA SOURCE ENTRY
Note European Journal of Dermatology | 33 | 3 | 2023-05-01 | p. 270-279 | 1167-1122
856 41 - ELECTRONIC LOCATION AND ACCESS
Uniform Resource Identifier <a href="https://shs.cairn.info/revue-european-journal-of-dermatology-2023-3-page-270?lang=en&redirect-ssocas=7080">https://shs.cairn.info/revue-european-journal-of-dermatology-2023-3-page-270?lang=en&redirect-ssocas=7080</a>

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