Tomita Yoshitaka, Morioka Takato, Murakami Nobuya, Noguchi Yushi, Sato Yuka, Suzuki Satoshi O
Department of Neonatology, Fukuoka Children's Hospital, Fukuoka, Japan.
Department of Neurosurgery, Fukuoka Children's Hospital, Fukuoka, Japan,
Pediatr Neurosurg. 2019;54(2):125-131. doi: 10.1159/000495810. Epub 2019 Jan 17.
Limited dorsal myeloschisis (LDM) and congenital dermal sinus (CDS) originate from incomplete disjunction between the cutaneous and neural ectoderms. Some LDM stalks have been found to have elements of a CDS or dermoid cyst. We surgically treated a saccular lesion in the lumbosacral region of a 7-day-old male neonate. Although fetal magnetic resonance imaging (MRI) failed to reveal a stalk, postnatal MRI including three-dimensional heavily T2-weighted imaging demonstrated a stalk originating from the lumbar cord and extending caudally to enter the lumbosacral meningocele sac. During untethering surgery, we found that the stalk was slender, with a diameter of 0.7-0.8 mm, but otherwise appeared to be a typical LDM stalk. Histopathological examination revealed that the fibrocollagenous stalk contained glial fibrillary acidic protein-immunopositive neuroglial tissues and stratified squamous epithelium. The present report describes the first documented case of a stalk with combined features of saccular LDM and CDS in a neonate. Since cutaneous ectodermal tissue is likely to remain in the remnant stalk, this patient requires careful monitoring to detect the potential development of a dermoid cyst.
局限性脊髓脊膜膨出(LDM)和先天性皮样窦(CDS)起源于皮肤外胚层和神经外胚层之间的不完全分离。已发现一些LDM蒂含有CDS或皮样囊肿的成分。我们对一名7日龄男婴腰骶部的囊状病变进行了手术治疗。尽管胎儿磁共振成像(MRI)未能显示出蒂,但包括三维重T2加权成像在内的产后MRI显示有一个蒂起源于腰髓并向尾侧延伸进入腰骶部脊膜膨出囊。在松解手术过程中,我们发现该蒂很细,直径为0.7 - 0.8毫米,但在其他方面似乎是一个典型的LDM蒂。组织病理学检查显示,纤维胶原性蒂含有胶质纤维酸性蛋白免疫阳性的神经胶质组织和复层鳞状上皮。本报告描述了首例新生儿中具有囊状LDM和CDS联合特征的蒂的病例。由于皮肤外胚层组织可能残留在残余的蒂中,该患者需要密切监测以检测皮样囊肿的潜在发展。