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一名小儿肾移植患者的播散性毛囊角化病采用局部西多福韦和口服来氟米特治疗

Management of Trichodysplasia Spinulosa in a Pediatric Kidney Transplant Patient With Topical Cidofovir and Oral Leflunomide.

作者信息

Hansen Annika M, Mehra Sonia, Bowen Anneli R, Sherbotie Joseph, Cipriano Sarah D

机构信息

Spencer Fox Eccles School of Medicine, Salt Lake City, Utah, USA.

Department of Pediatrics, University of Utah, Salt Lake City, Utah, USA.

出版信息

Pediatr Transplant. 2025 May;29(3):e70080. doi: 10.1111/petr.70080.

DOI:10.1111/petr.70080
PMID:40200820
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11979675/
Abstract

BACKGROUND

This case report describes a 10-year-old male with a history of kidney transplantation who developed trichodysplasia spinulosa (TS), a rare skin disorder associated with immunosuppression.

METHODS

The patient's condition, characterized by follicular papules and keratinous spines primarily on the face, was managed by a multidisciplinary team.

RESULTS

Treatment involved reducing immunosuppression and administering topical cidofovir alongside oral leflunomide, leading to significant clinical improvement over several months.

CONCLUSIONS

This case adds to the literature of TS management and highlights the importance of multidisciplinary care as well as the challenges of treatment accessibility due to insurance limitations.

摘要

背景

本病例报告描述了一名10岁男性肾移植受者,其患有一种与免疫抑制相关的罕见皮肤疾病——棘状毛发发育异常(TS)。

方法

该患者的病情以面部为主的毛囊丘疹和角质棘为特征,由多学科团队进行管理。

结果

治疗包括减少免疫抑制,并外用西多福韦和口服来氟米特,在数月内取得了显著的临床改善。

结论

本病例丰富了TS治疗的文献,并强调了多学科护理的重要性以及由于保险限制导致的治疗可及性挑战。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/b3f38226db1c/PETR-29-e70080-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/08acd184942a/PETR-29-e70080-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/f24cccb4f811/PETR-29-e70080-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/c21b85a62244/PETR-29-e70080-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/b3f38226db1c/PETR-29-e70080-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/08acd184942a/PETR-29-e70080-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/f24cccb4f811/PETR-29-e70080-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/c21b85a62244/PETR-29-e70080-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/372f/11979675/b3f38226db1c/PETR-29-e70080-g001.jpg

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1
Management of Trichodysplasia Spinulosa in a Pediatric Kidney Transplant Patient With Topical Cidofovir and Oral Leflunomide.一名小儿肾移植患者的播散性毛囊角化病采用局部西多福韦和口服来氟米特治疗
Pediatr Transplant. 2025 May;29(3):e70080. doi: 10.1111/petr.70080.
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本文引用的文献

1
Trichodysplasia spinulosa: a presentation of polyomavirus infection in immunosuppressed patients.棘状毛发发育异常:免疫抑制患者多瘤病毒感染的一种表现。
Dermatol Online J. 2022 Dec 15;28(6). doi: 10.5070/D328659724.
2
Trichodysplasia spinulosa: a comprehensive review of the disease and its treatment.棘层松解性外毛根鞘瘤:疾病及其治疗的全面综述。
J Eur Acad Dermatol Venereol. 2021 May;35(5):1067-1076. doi: 10.1111/jdv.17081. Epub 2021 Jan 6.
3
Pediatric trichodysplasia spinulosa: A report of 2 cases and review of the literature.
儿童棘状毛发发育异常:2例报告及文献复习
Pediatr Dermatol. 2020 Nov;37(6):1023-1029. doi: 10.1111/pde.14323. Epub 2020 Aug 12.
4
Primary trichodysplasia spinulosa polyomavirus infection in a kidney transplant child displaying virus-infected decoy cells in the urine.原发性毛发疏松性棘皮瘤多瘤病毒感染:一例肾移植患儿的尿液中存在病毒感染的诱饵细胞。
J Med Virol. 2019 Oct;91(10):1896-1900. doi: 10.1002/jmv.25519. Epub 2019 Jul 2.
5
Trichodysplasia spinulosa in a child: Identification of trichodysplasia spinulosa-associated polyomavirus in skin, serum, and urine.一名儿童的棘状毛发发育异常:在皮肤、血清和尿液中鉴定出与棘状毛发发育异常相关的多瘤病毒
Pediatr Dermatol. 2019 Sep;36(5):723-724. doi: 10.1111/pde.13857. Epub 2019 Jun 12.
6
Leflunomide for the treatment of trichodysplasia spinulosa in a liver transplant recipient.来氟米特用于治疗肝移植受者的棘状毛囊发育不良。
Transpl Infect Dis. 2017 Aug;19(4). doi: 10.1111/tid.12702. Epub 2017 May 15.
7
Primary Polyomavirus Infection, Not Reactivation, as the Cause of Trichodysplasia Spinulosa in Immunocompromised Patients.原发性多瘤病毒感染而非病毒再激活是免疫功能低下患者棘状毛囊发育不良的病因。
J Infect Dis. 2017 Apr 1;215(7):1080-1084. doi: 10.1093/infdis/jiw403.
8
Spiny Papules in an Immunosuppressed Child.一名免疫抑制儿童身上的棘状丘疹
Pediatr Dermatol. 2015 Nov-Dec;32(6):e296-7. doi: 10.1111/pde.12672. Epub 2015 Sep 4.
9
Insights into the mechanism of action of cidofovir and other acyclic nucleoside phosphonates against polyoma- and papillomaviruses and non-viral induced neoplasia.西多福韦及其他无环核苷膦酸盐对多瘤病毒和乳头瘤病毒以及非病毒诱导肿瘤形成的作用机制研究
Antiviral Res. 2015 Feb;114:21-46. doi: 10.1016/j.antiviral.2014.10.012. Epub 2014 Oct 30.
10
Trichodysplasia spinulosa: rare presentation of polyomavirus infection in immunocompromised patients.棘层松解性外毛根鞘瘤:免疫功能低下患者中多瘤病毒感染的罕见表现。
J Cutan Med Surg. 2014 Nov;18(6):430-5. doi: 10.2310/7750.2014.13189.