• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

青少年起病的家族性反应性穿通性胶原病:1例罕见病例报告及文献复习

Familial Reactive Perforating Collagenosis with Adolescence-Onset: A Rare Case Report and Literature Review.

作者信息

Tsaqilah Laila, Nastiti Hedwika Advina, Usman Hermin Aminah, Avriyanti Erda, Dharmadji Hartati Purbo, Hidayah Risa Miliawati Nurul

机构信息

Department of Dermatology and Venereology, Faculty of Medicine, Universitas Padjadjaran - Dr. Hasan Sadikin General Hospital, Bandung, West Java, Indonesia.

Department of Anatomical Pathology, Faculty of Medicine, Universitas Padjadjaran - Dr. Hasan Sadikin General Hospital, Bandung, West Java, Indonesia.

出版信息

Clin Cosmet Investig Dermatol. 2024 Aug 23;17:1895-1904. doi: 10.2147/CCID.S468181. eCollection 2024.

DOI:10.2147/CCID.S468181
PMID:39193096
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11348990/
Abstract

Familial Reactive Perforating Collagenosis (FRPC) is a very rare form of benign dermatosis frequently presented during early childhood and not associated with systemic diseases. Less than 50 FRPC patients have been reported in the literature. Due to the limited number of cases, the pathophysiology of this unique entity remains elusive; moreover, no standard treatment has been agreed upon. Here, we report a case of FRPC in a 20-year-old male who was presented with generalized multiple discrete papules covered with central keratotic plugs in all regions of his body, particularly in the facial area, neck, abdominal, and extensor region of the extremities for more than 7 years. Similar symptoms were acknowledged in the patient's family members. Histopathological analyses identified the crateriform shape invagination in the epidermis filled with inflammatory lymphocytes and basophilic debris and perforated by basophilic collagen bundles from the underlying dermis. Based on the clinical and histopathological findings, the patient was diagnosed with FRPC. He was treated with topical desoximetasone 0.25% cream applied 2-3 times daily. A follow-up evaluation after 4 weeks revealed a near-complete resolution of skin papules. To our knowledge, this is the first report of FRPC case from Indonesia. Unlike the majority of FRPC patients who had their disease onsets during infancy or early childhood, FRPC skin manifestations in our patient started during the adolescence period. The resolution of skin manifestations after daily application of topical desoximetasone suggests that topical corticosteroids are a potential treatment option for FRPC patients.

摘要

家族性反应性穿孔性胶原病(FRPC)是一种非常罕见的良性皮肤病,常在儿童早期出现,且与全身性疾病无关。文献报道的FRPC患者不足50例。由于病例数量有限,这种独特疾病的病理生理学仍不清楚;此外,尚未达成标准治疗方案。在此,我们报告一例20岁男性FRPC病例,该患者全身各部位,特别是面部、颈部、腹部和四肢伸侧出现泛发性多个离散丘疹,中央有角质栓,病程超过7年。患者家庭成员也有类似症状。组织病理学分析发现表皮呈火山口状内陷,充满炎性淋巴细胞和嗜碱性碎屑,并被来自真皮下层的嗜碱性胶原束贯穿。根据临床和组织病理学表现,该患者被诊断为FRPC。给予其外用0.25%地索奈德乳膏,每日涂抹2 - 3次。4周后的随访评估显示皮肤丘疹几乎完全消退。据我们所知,这是印度尼西亚首例FRPC病例报告。与大多数在婴儿期或儿童早期发病的FRPC患者不同,我们的患者FRPC皮肤表现始于青春期。每日外用糖皮质激素后皮肤表现消退,提示外用糖皮质激素是FRPC患者的一种潜在治疗选择。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b3a9/11348990/f078d3a04464/CCID-17-1895-g0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b3a9/11348990/97a666fc5a14/CCID-17-1895-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b3a9/11348990/f078d3a04464/CCID-17-1895-g0002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b3a9/11348990/97a666fc5a14/CCID-17-1895-g0001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b3a9/11348990/f078d3a04464/CCID-17-1895-g0002.jpg

相似文献

1
Familial Reactive Perforating Collagenosis with Adolescence-Onset: A Rare Case Report and Literature Review.青少年起病的家族性反应性穿通性胶原病:1例罕见病例报告及文献复习
Clin Cosmet Investig Dermatol. 2024 Aug 23;17:1895-1904. doi: 10.2147/CCID.S468181. eCollection 2024.
2
A rare case of familial reactive perforating collagenosis.一例罕见的家族性反应性穿通性胶原病。
Indian J Dermatol. 2013 Sep;58(5):408. doi: 10.4103/0019-5154.117341.
3
Acquired reactive perforating collagenosis, a rare entity at uncommon site.获得性反应性穿孔胶原病,一种罕见部位的罕见疾病。
Indian J Pathol Microbiol. 2022 Oct-Dec;65(4):895-897. doi: 10.4103/ijpm.ijpm_333_21.
4
Acquired reactive perforating collagenosis with the histological features of IgG4-related sclerosing disease in a patient with Mikulicz's disease.米库利奇病患者中具有IgG4相关硬化性疾病组织学特征的获得性反应性穿通性胶原病。
Pathol Int. 2009 May;59(5):326-31. doi: 10.1111/j.1440-1827.2009.02374.x.
5
Reactive perforating collagenosis and systemic lupus erythematosus: A rare case report.反应性穿孔性胶原病合并系统性红斑狼疮:一例罕见病例报告。
Medicine (Baltimore). 2022 Dec 2;101(48):e32138. doi: 10.1097/MD.0000000000032138.
6
Reactive Perforating Collagenosis反应性穿通性胶原病
7
Acquired reactive perforating collagenosis: A report of a typical case.获得性反应性穿通性胶原病:1例典型病例报告。
Medicine (Baltimore). 2016 Jul;95(30):e4305. doi: 10.1097/MD.0000000000004305.
8
A Case of Noonan Syndrome and Kyrle Disease: Casualty or Causality?一例努南综合征合并 Kyrle 病:偶然还是必然?
Acta Dermatovenerol Croat. 2023 Dec;31(3):160-161.
9
A Case of Noonan Syndrome and Kyrle's Disease: Coincidence or Causality?努南综合征合并 Kyrle 病 1 例:巧合还是因果关系?
Acta Dermatovenerol Croat. 2024 Mar;32(1):71-72.
10
Generalized Lesions of Kyrle's Disease: A Rare Case.凯尔氏病的全身性病变:一例罕见病例。
Int Med Case Rep J. 2022 Apr 12;15:187-191. doi: 10.2147/IMCRJ.S358523. eCollection 2022.

本文引用的文献

1
Keratotic Papules and Scars in a Young Boy.一名小男孩身上的角化性丘疹和瘢痕
Indian J Dermatol. 2016 Sep-Oct;61(5):576-8. doi: 10.4103/0019-5154.190112.
2
Familial reactive perforating collagenosis: a report of two cases.家族性反应性穿通性胶原病:两例报告。
Indian J Dermatol. 2014 May;59(3):287-9. doi: 10.4103/0019-5154.131405.
3
Familial reactive perforating collagenosis in three siblings.三名兄弟姐妹患家族性反应性穿通性胶原病。
Indian J Dermatol Venereol Leprol. 2014 Jan-Feb;80(1):86-7. doi: 10.4103/0378-6323.125488.
4
A rare case of familial reactive perforating collagenosis.一例罕见的家族性反应性穿通性胶原病。
Indian J Dermatol. 2013 Sep;58(5):408. doi: 10.4103/0019-5154.117341.
5
Reactive perforating collagenosis in two siblings.两名兄弟姐妹患反应性穿通性胶原病。
BMJ Case Rep. 2013 Sep 26;2013:bcr2013009023. doi: 10.1136/bcr-2013-009023.
6
Familial reactive perforating collagenosis in a child: response to narrow-band UVB.一名儿童的家族性反应性穿通性胶原病:对窄谱中波紫外线的反应
Pediatr Dermatol. 2013 Nov-Dec;30(6):762-4. doi: 10.1111/j.1525-1470.2012.01736.x. Epub 2012 Apr 4.
7
Acquired reactive perforating collagenosis: a clinicopathologic study of 15 cases from Saudi Arabia.获得性反应性穿孔性胶原病:来自沙特阿拉伯的 15 例临床病理研究。
J Eur Acad Dermatol Venereol. 2010 Feb;24(2):223-7. doi: 10.1111/j.1468-3083.2009.03333.x.
8
Familial reactive perforating collagenosis.家族性反应性穿通性胶原病
Indian J Dermatol. 2009;54(4):334-7. doi: 10.4103/0019-5154.57608.
9
Familial reactive perforating collagenosis: a clinical, histopathological study of 10 cases.家族性反应性穿通性胶原病:10例临床及组织病理学研究
J Eur Acad Dermatol Venereol. 2007 Jul;21(6):766-70. doi: 10.1111/j.1468-3083.2006.02085.x.
10
Familial reactive perforating collagenosis: a case report.家族性反应性穿通性胶原病:一例报告。
J Dermatol. 1998 Jan;25(1):54-6. doi: 10.1111/j.1346-8138.1998.tb02347.x.