Oshima Akito, Sakata Katsumi, Ishikawa Kosuke, Manaka Hiroshi, Kawasaki Takashi
Department of Neurosurgery, Yokohama City University Medical Center, Yokohama, Kanagawa, Japan.
NMC Case Rep J. 2021 Jun 25;8(1):319-324. doi: 10.2176/nmccrj.cr.2020-0324. eCollection 2021.
Endodermal cyst (EC) is a rare congenital cyst of endodermal origin, but the pathogenesis of this entity remains uncertain. Supratentorial EC is particularly uncommon, but some cases have been reported. Here, we report a case of supratentorial EC that developed at the frontal base which indicates posttraumatic development rather than a congenital origin.
A 65-year-old man who had a history of orbital bone fracture without rhinorrhea sustained in a traffic accident presented with gradually enlarging frontal-base cystic lesions. Multiple cystic lesions were removed via left frontal craniotomy. The cysts showed no communication with the frontal sinus. Histological examination identified EC. Postoperative course was uneventful and no recurrences have been identified as of 2 years later.
According to reported cases, unlike ECs in other intracranial locations, frontal base ECs tend to present at advanced ages. The present case also presented with EC enlargement at an advanced age and two lesions located at the bone hiatus in the frontal base that were presumably caused by trauma. It is possible that sinus communication was repaired as the bone fracture was remodeled, and the remnant sinus epithelial tissues developed into ECs over time. This situation makes it reasonable to presume a posttraumatic rather than a congenital origin. In conclusion, as for frontal base ECs, contrary to the traditional theory, the developmental mechanisms may not necessarily be congenital.
内胚层囊肿(EC)是一种罕见的内胚层起源的先天性囊肿,但其发病机制仍不明确。幕上EC尤其少见,但已有一些病例报道。在此,我们报告一例发生于额底部的幕上EC病例,提示其为创伤后形成而非先天性起源。
一名65岁男性,有交通事故导致眼眶骨折但无鼻漏病史,出现额底部囊性病变逐渐增大。通过左额开颅手术切除多个囊性病变。囊肿与额窦无相通。组织学检查确诊为EC。术后过程顺利,截至2年后未发现复发。
根据已报道的病例,与颅内其他部位的EC不同,额底部EC往往在高龄时出现。本病例也在高龄时出现EC增大,且两个病变位于额底部的骨裂孔处,推测由创伤引起。随着骨折重塑,鼻窦相通可能得以修复,残余的鼻窦上皮组织随时间发展为EC。这种情况使得推测其为创伤后起源而非先天性起源成为合理推断。总之,对于额底部EC,与传统理论相反,其发育机制不一定是先天性的。