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资源有限环境下结节性硬化症复合体的诊断与管理——一名14岁赞比亚青少年女性的病例报告

Diagnosis and Management of Tuberous Sclerosis Complex in a Resource-Limited Setting-A Case Report of a 14-Year-Old Female Zambian Adolescent.

作者信息

Lienda Mwamba, Mwila Meek, Sichula Chilala, Kabengele Chishiba, Akombwa Moses, Zulu Christina, Banda Chihena Hansini, M'hango Hellen

机构信息

Lusaka Apex Medical University, Lusaka, Zambia.

University of Zambia School of Medicine, Lusaka, Zambia.

出版信息

Clin Med Insights Case Rep. 2025 Feb 18;18:11795476251321268. doi: 10.1177/11795476251321268. eCollection 2025.

DOI:10.1177/11795476251321268
PMID:39974288
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11837062/
Abstract

Tuberous sclerosis complex (TSC) is a rare multisystemic neurocutaneous syndrome with a wide spectrum of clinical manifestations. We present a case of a 14-year-old adolescent female who presented with a history of facial angiofibromas since the age of 8 months. Physical examination was remarkable for multiple angiofibromas on the face, and other multiple cutaneous manifestations of TSC. MRI of the head, and abdomen revealed cortical tubers, multiple bilateral periventricular and subependymal nodular lesions, calcifications, and bilateral kidney enlargement with multiple bilateral renal angiomyolipomas of varying sizes in a background of bilateral polycystic kidneys, MRI of the chest was unremarkable. A diagnosis of TSC was made using the clinical diagnostic criteria which consist of major and minor features. A diagnosis using genetic studies could not be made due to a lack of resources. Management was multidisciplinary and regular monitoring every 6 months will be required to monitor disease progression and manage complications as they arise. This case illustrates the multidisciplinary approach needed to address the diverse clinical manifestations of TSC and the diagnostic challenges, treatment limitations, and psychological impact of TSC in low-resource settings like Zambia where access to advanced therapies is limited.

摘要

结节性硬化症(TSC)是一种罕见的多系统神经皮肤综合征,临床表现多样。我们报告一例14岁青春期女性病例,该患者自8个月大起就有面部血管纤维瘤病史。体格检查发现面部有多个血管纤维瘤,以及TSC的其他多种皮肤表现。头部和腹部的MRI显示有皮质结节、多个双侧脑室周围和室管膜下结节性病变、钙化,以及双侧肾脏增大,在双侧多囊肾的背景下有多个大小不一的双侧肾血管平滑肌脂肪瘤,胸部MRI未见明显异常。根据由主要和次要特征组成的临床诊断标准做出了TSC的诊断。由于缺乏资源,无法通过基因研究做出诊断。治疗采用多学科方法,需要每6个月进行定期监测,以监测疾病进展并处理出现的并发症。该病例说明了应对TSC多样临床表现所需的多学科方法,以及在赞比亚这样资源有限、获得先进治疗手段受限的低资源环境中TSC的诊断挑战、治疗局限性和心理影响。

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本文引用的文献

1
Tuberous Sclerosis Complex and the kidneys: what nephrologists need to know.结节性硬化症与肾脏:肾科医生应知应会
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