克拉曲滨治疗进展期播散性黄瘤3例
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摘要
报告3例克拉曲滨成功治疗进展期播散性黄瘤(XD)。患者均为男性,2例33~34岁,1例12岁,病程5~8年。皮肤科检查:皮疹均泛发全身且伴黏膜损害,褶皱部位受累明显。2例并发尿崩症与眼内损害,3例均有黏膜受累且病情进展,对多种治疗抵抗。诊断:XD。治疗:予静脉滴注克拉曲滨0.14 mg/(kg·d),连续5 d为一个疗程,每月1次,共6个疗程。治疗后3例患者病情均有不同程度的改善,起效时间在第2~3疗程间。其中2例患者>70%皮损消退,遗留色素沉着,其余皮损明显缩小。3例黏膜和(或)眼内症状及尿崩症均明显改善。治疗过程中均未发现严重不良反应,仅有短期可逆性血细胞下降。随访4年发现停药后皮损与黏膜情况仍在进一步好转中,尿崩症亦有所好转。
Three cases of progressive xanthoma disseminatum(XD) cured with cladribine are reported. Two males were 33 to34 years old, and one male was 12 years old, with 5-to 8-year history of XD. Physical examination showed generalized rashes all over the body with mucosal damage, and lesions were more severe in the flexural regions. All patients were in progressive stage, and resistant to multiple treatments. Two cases were complicated with diabetes insipidus and intraocular damage. All patients were diagnosed with XD. The patients were treated with intravenous cladribine at a dosage of 0.14 mg/(kg·d) for 5 days, repeated monthly for 6 months. After 2 to 3 months of treatments, all patients showed improvement. At the end of the treatment, over 70% of lesions disappeared in two patients. No patient showed any severe adverse effects, except for hyperpigmentation and temporary, reversible hypocytosis. After termination of treatment, all patients experienced further improvements in cutaneous and mucosal damages as well as diabetes insipidus during a 4-year follow up.
Three cases of progressive xanthoma disseminatum(XD) cured with cladribine are reported. Two males were 33 to34 years old, and one male was 12 years old, with 5-to 8-year history of XD. Physical examination showed generalized rashes all over the body with mucosal damage, and lesions were more severe in the flexural regions. All patients were in progressive stage, and resistant to multiple treatments. Two cases were complicated with diabetes insipidus and intraocular damage. All patients were diagnosed with XD. The patients were treated with intravenous cladribine at a dosage of 0.14 mg/(kg·d) for 5 days, repeated monthly for 6 months. After 2 to 3 months of treatments, all patients showed improvement. At the end of the treatment, over 70% of lesions disappeared in two patients. No patient showed any severe adverse effects, except for hyperpigmentation and temporary, reversible hypocytosis. After termination of treatment, all patients experienced further improvements in cutaneous and mucosal damages as well as diabetes insipidus during a 4-year follow up.
引文
[1]Ya ci B,Varan A,Altinok G,et al.Xanthoma disseminatum in a child with cranial bone involvement[J].J Pediatr Hematol Oncol,2008,30(4):310-312.
[2]Zak IT,Altinok D,Neilsen SS,et al.Xanthoma disseminatum of the central nervous system and cranium[J].AJNR Am J Neuroradiol,2006,27(4):919-921.
[3]徐晨琛,刘跃华.播散性黄瘤并发垂体、骨受累及尿崩症[J].临床皮肤科杂志,2015,44(6):380-382.
[4]James WD,Berger TG,Elston D.Andrews’Disease of the Skin Clinical Dermatology[M].10th ed.Philadelphis:Saunders,2006:717-718.
[5]Calverly DC,Wismer J,Rosenthal D,et al.Xanthoma disseminatum in an infant with skeletal and marrow involvement[J].J Pediatr Hematol Oncol,1995,17(1):61-65.
[6]Zelger BW,Sidoroff A,Orchard G,et al.Non-Langerhans cell histiocytoses.A new unifying concept[J].Am J Dermatopathol,1996,18(5):490-504.
[7]Caputo R,Veraldi S,Grimalt R,et al.The various clinical patterns of xanthoma disseminatum[J].Dermatology,1995,190(1):19-24.
[8]Seaton ED,Pillai GJ,Chu AC.Treatment of xanthoma disseminatum with cyclophosphamide[J].Br J Dermatol,2004,150(2):346-349.
[9]Pruvost C,Picard-Dahan C,Bonnefond B,et al.Vinblastine treatment for extensive non-X histiocytosis(xanthoma disseminatum)[in French][J].Ann Dermatol Venereol,2004,131(3):271-273.
[10]Khezri F,Gibson LE,Tefferi A.Xanthoma disseminatum:effective therapy with 2-chlorodeoxyadenosine in a case series[J].Arch Dermatol,2011,147(4):459-464.
[11]Spurgeon S,Yu M,Phillips JD.Cladribine:not just another purine analogue[J].Expert Opin Investig Drugs,2009,18(8):1169-1181.
[12]Mottl H,StarJ,ChánováM,et al.Treatment of recurrent Langerhans cell histiocytosis in children with 2-chlorodeoxyadenosine[J].Leuk Lymphoma,2006,47(9):1881-1884.
[13]Adam Z,Szturz P,Pour L,et al.Cladribine is highly effective in the treatment of Langerhans cell histiocytosis and rare histiocytic disorders of the juvenile xanthogranuloma group[J].2012,58(6):455-465.
[14]Shani-Adir A,Chou P,Morgan E,et al.A child with both Langerhans and non-Langerhans cell histiocytosis[J].Pediatr Dermatol,2002,19(5):419-422.
[2]Zak IT,Altinok D,Neilsen SS,et al.Xanthoma disseminatum of the central nervous system and cranium[J].AJNR Am J Neuroradiol,2006,27(4):919-921.
[3]徐晨琛,刘跃华.播散性黄瘤并发垂体、骨受累及尿崩症[J].临床皮肤科杂志,2015,44(6):380-382.
[4]James WD,Berger TG,Elston D.Andrews’Disease of the Skin Clinical Dermatology[M].10th ed.Philadelphis:Saunders,2006:717-718.
[5]Calverly DC,Wismer J,Rosenthal D,et al.Xanthoma disseminatum in an infant with skeletal and marrow involvement[J].J Pediatr Hematol Oncol,1995,17(1):61-65.
[6]Zelger BW,Sidoroff A,Orchard G,et al.Non-Langerhans cell histiocytoses.A new unifying concept[J].Am J Dermatopathol,1996,18(5):490-504.
[7]Caputo R,Veraldi S,Grimalt R,et al.The various clinical patterns of xanthoma disseminatum[J].Dermatology,1995,190(1):19-24.
[8]Seaton ED,Pillai GJ,Chu AC.Treatment of xanthoma disseminatum with cyclophosphamide[J].Br J Dermatol,2004,150(2):346-349.
[9]Pruvost C,Picard-Dahan C,Bonnefond B,et al.Vinblastine treatment for extensive non-X histiocytosis(xanthoma disseminatum)[in French][J].Ann Dermatol Venereol,2004,131(3):271-273.
[10]Khezri F,Gibson LE,Tefferi A.Xanthoma disseminatum:effective therapy with 2-chlorodeoxyadenosine in a case series[J].Arch Dermatol,2011,147(4):459-464.
[11]Spurgeon S,Yu M,Phillips JD.Cladribine:not just another purine analogue[J].Expert Opin Investig Drugs,2009,18(8):1169-1181.
[12]Mottl H,StarJ,ChánováM,et al.Treatment of recurrent Langerhans cell histiocytosis in children with 2-chlorodeoxyadenosine[J].Leuk Lymphoma,2006,47(9):1881-1884.
[13]Adam Z,Szturz P,Pour L,et al.Cladribine is highly effective in the treatment of Langerhans cell histiocytosis and rare histiocytic disorders of the juvenile xanthogranuloma group[J].2012,58(6):455-465.
[14]Shani-Adir A,Chou P,Morgan E,et al.A child with both Langerhans and non-Langerhans cell histiocytosis[J].Pediatr Dermatol,2002,19(5):419-422.