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一名患有鲁宾斯坦-泰比综合征患者的毛发上皮瘤:诊断和治疗线索

Pilomatricomas in a patient with Rubinstein-Taybi syndrome: diagnostic and therapeutic clues.

作者信息

Battaglia Laura, Ini' Corrado, Lo Bianco Manuela, Leonardi Roberta, Ini' Eleonora, Foti Pietro Valerio, Palmucci Stefano, Fichera Marco, Ruggieri Martino, Basile Antonio

机构信息

Department of Medical Surgical Sciences and Advanced Technologies 'G.F. Ingrassia'-Radiology I Unit, University Hospital Policlinico 'G. Rodolico-San Marco,' Catania, Italy.

Department of Medical Surgical Sciences and Advanced Technologies 'G.F. Ingrassia'-Radiology I Unit, University Hospital Policlinico 'G. Rodolico-San Marco,' Via Santa Sofia 78, Catania 95123, Italy.

出版信息

Ther Adv Rare Dis. 2025 Apr 12;6:26330040251330316. doi: 10.1177/26330040251330316. eCollection 2025 Jan-Dec.

Abstract

Pilomatricoma is a rare benign neoplasm originating from hair cortex cells and typically manifests in children as a slow-growing bluish-red, superficial and firm mass. Multiple pilomatricomas can be associated with genetic mutations and syndromic disorders, most commonly with Rubinstein-Taybi syndrome, Gardner syndrome, myotonic dystrophy, Turner syndrome, and Sotos syndrome. Ultrasound examination allows this tumor to be characterized, to assess the involvement of deeper structures and to plan treatment. Pilomatricoma shows some distinctive ultrasonographic features that aid in its diagnosis and it may be seen on ultrasound as an ovoid complex mass. Complications and malignant transformation of pilomatricomas have been described as a possible tumor evolution, and surgical resection is recommended. We present a rare case of a 17-year-old female patient with intellectual disability and microcephaly, and with the evidence of multiple pilomatricomas in the head-neck region on ultrasound examination. The syndromic features of the patient and genetic tests led to a diagnosis of Rubinstein-Taybi syndrome. We also focused on the association between pilomatricomas and genetic mutations in patients with Rubinstein-Taybi syndrome.

摘要

毛母质瘤是一种罕见的良性肿瘤,起源于毛皮质细胞,在儿童中通常表现为生长缓慢的蓝红色、浅表且质地坚硬的肿块。多发性毛母质瘤可能与基因突变和综合征性疾病相关,最常见的是与鲁宾斯坦-泰比综合征、加德纳综合征、强直性肌营养不良、特纳综合征和索托斯综合征有关。超声检查可以对这种肿瘤进行特征描述,评估深部结构的受累情况并制定治疗方案。毛母质瘤具有一些独特的超声特征,有助于其诊断,在超声上可能表现为椭圆形复合肿块。毛母质瘤的并发症和恶变已被描述为一种可能的肿瘤演变,建议手术切除。我们报告一例罕见病例,一名17岁女性患者,有智力障碍和小头畸形,超声检查显示头颈部区域有多个毛母质瘤。患者的综合征特征和基因检测导致诊断为鲁宾斯坦-泰比综合征。我们还关注了鲁宾斯坦-泰比综合征患者中毛母质瘤与基因突变之间的关联。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/29de/12033402/fa938ce21f90/10.1177_26330040251330316-fig1.jpg

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