Hu Hui-Min, Long Wei-Guo, Wang Xuan, Li Yu-Mei, Xu Hui
Department of Dermatology, Affiliated Hospital of Jiangsu University, Zhenjiang, China.
Department of Pathology, Affiliated Hospital of Jiangsu University, Zhenjiang, China.
AME Case Rep. 2021 Apr 25;5:20. doi: 10.21037/acr-20-95. eCollection 2021.
Infantile digital fibromatosis is a rare, benign fibrous tumor among infants usually limited to fingers and toes. Most cases present themselves with round eosinophilic inclusion bodies of different sizes, similar to erythrocyte in the parakarytoplasm. Although infantile digital fibromatosis had been observed with a tendency of spontaneous regression after a rapid phase of growth in some reports, the recurrence rate of early surgical intervention remains high. And the mechanism of recurrence is still unknown. Human papillomavirus (HPV), as a circular icosahedral double-stranded DNA virus, is famous for its remarkable significant correlation with cervical cancer. However, the reports about the possible relationship of recurrent infantile digital fibromatosis and HPV infection are rare and inconsistent. Here, we report a recurrent case of infantile digital fibromatosis after surgical resection. Pathological biopsy of the recurrent site not only identified the diagnosis of infantile digital fibromatosis again, but found the sign of HPV infection. Family history indicated that the patient's grandmother had a history of verruca plana. After complete resection of recurrence, the tumor recurred again and the case is still being followed up. The unique case may serve as a clue to the pathogenesis of the relationship between recurrent infantile digital fibromatosis and HPV infection.
婴儿指(趾)纤维瘤病是一种罕见的婴儿期良性纤维性肿瘤,通常局限于手指和脚趾。大多数病例在副核质中可见大小不一的圆形嗜酸性包涵体,类似于红细胞。尽管在一些报道中观察到婴儿指(趾)纤维瘤病在快速生长阶段后有自发消退的趋势,但早期手术干预后的复发率仍然很高。而且复发机制尚不清楚。人乳头瘤病毒(HPV)作为一种环状二十面体双链DNA病毒,因其与宫颈癌有显著的相关性而闻名。然而,关于复发性婴儿指(趾)纤维瘤病与HPV感染可能关系的报道很少且不一致。在此,我们报告一例手术切除后复发的婴儿指(趾)纤维瘤病病例。复发部位的病理活检不仅再次确诊为婴儿指(趾)纤维瘤病,还发现了HPV感染的迹象。家族史显示患者的祖母有扁平疣病史。在彻底切除复发肿瘤后,肿瘤再次复发,该病例仍在随访中。这一独特病例可能为复发性婴儿指(趾)纤维瘤病与HPV感染之间关系的发病机制提供线索。