Fujimoto Tatsufumi, Hasegawa Kana, Kumamaru Wataru, Chikui Toru, Yanai Yuta, Shibuya Minami, Fujii Shinsuke, Yoshiura Kazunori, Kawano Shintaro, Kiyoshima Tamotsu
Laboratory of Oral Pathology, Division of Maxillofacial Diagnostic and Surgical Sciences, Faculty of Dental Science, Kyushu University, Fukuoka, Japan.
Sections of Oral and Maxillofacial Surgery, Division of Maxillofacial Diagnostic and Surgical Sciences, Faculty of Dental Science, Kyushu University, Fukuoka, Japan.
Medicine (Baltimore). 2025 Jun 20;104(25):e42936. doi: 10.1097/MD.0000000000042936.
Composite lesions of neurogenic tumors with vascular malformations, which were once included among hemangiomas, are extremely rare and can be classified into 2 types: conjoined and discrete associations, with the former representing a single lesion of vascular malformation within tumor tissue. To date, we have found 24 composite lesions (conjoined association type) of schwannomas coexisting with vascular malformations. However, such composite lesions should be interpreted with caution.
A 29-year-old woman had recognized swelling of the tongue 5 years prior to her initial hospital visit and the lesion had recently been slowly growing.
Based on clinical examinations, including imaging studies, the tumorous lesion was clinically diagnosed as a schwannoma, but a benign salivary gland tumor and venous malformation (VM) were not completely ruled out.
The lesion was excised with safety margin.
The excised sample revealed 2 intriguing features; however, the lesion was diagnosed as an ancient schwannoma with VM-like features because of the lack of definitive findings that would make these lesions a composite of independent lesions. There was no evidence of recurrence or distant metastasis at the 16-month follow-up after excision.
Ancient schwannomas of the oral cavity are rare. In particular, schwannomas that appear as composite lesions of schwannomas with vascular malformations are extremely rare. Here, we report the first case of an ancient schwannoma with VM-like features of the tongue. It is important for clinicians to accumulate information on these rare cases to make an accurate preoperative diagnosis and to plan appropriate treatment.
神经源性肿瘤与血管畸形的复合性病变曾被归入血管瘤,极为罕见,可分为两种类型:联体和离散性关联,前者表现为肿瘤组织内单一的血管畸形病变。迄今为止,我们已发现24例与血管畸形共存的神经鞘瘤复合性病变(联体关联型)。然而,对此类复合性病变的解读应谨慎。
一名29岁女性在初次就诊前5年就发现舌头肿胀,且病变最近一直在缓慢生长。
基于包括影像学检查在内的临床检查,该肿瘤性病变临床诊断为神经鞘瘤,但未完全排除良性涎腺肿瘤和静脉畸形(VM)。
将病变完整切除。
切除的样本显示出两个有趣的特征;然而,由于缺乏明确的证据表明这些病变是独立病变的复合体,该病变被诊断为具有VM样特征的陈旧性神经鞘瘤。切除后16个月的随访中,无复发或远处转移的证据。
口腔陈旧性神经鞘瘤罕见。特别是表现为神经鞘瘤与血管畸形复合性病变的神经鞘瘤极为罕见。在此,我们报告首例舌部具有VM样特征的陈旧性神经鞘瘤。临床医生积累这些罕见病例的信息对于进行准确的术前诊断和制定合适的治疗方案很重要。