Department of Neurosurgery, Bagcilar Education and Research Hospital, University of Health Science, Istanbul, Turkey.
Department of Radiology, Gaziosmanpaşa Hospital, Istanbul Yeni Yüzyil University, Istanbul, Turkey.
Pediatr Neurosurg. 2021;56(4):379-384. doi: 10.1159/000516207. Epub 2021 May 7.
Trigonocephaly (TC), the tapering of the metopic suture toward the anterior, lateral inadequacy of supraorbital bar, hypoplasia of the ethmoid bone, and hypotelorism due to orbital medialization leads to a triangular shape in the head together with frontotemporal stenosis and widening of the biparietal diameter. Arachnoid cysts (ACs) are benign cysts, which are formed by cerebrospinal-fluid entrapment into the arachnoid membrane due to duplication or separation of the arachnoid membrane layers. ACs are typically located in the middle cranial fossa and Sylvian fissure region. They are mostly detected incidentally through neuroimaging. The coexistence of TC and AC is considerably rare; hence, its actual incidence is unknown. To our knowledge, the coexistence of nonsyndromic TC and AC has been revealed in only 2 publications in the literature. In this case study, 2 patients, one of whom were with unilateral and the other with bilateral temporally localized ACs, sizes of which increased following the reconstructive surgeries, and who underwent reconstructive surgery for TC, were presented.
Both of the 2 patients that we have presented in our study are 7 months old and male. Fronto-orbital advancement and calvarial remodeling operations were performed on both. ACs of the patients were not intervened. However, in the post-op follow-ups, the left temporal cyst of the 1st case and the right cyst of the 2nd case grew radiologically, and the left cyst became very small.
DISCUSSION/CONCLUSION: TC must be treated surgically. ACs may show changes in size secondary to the increase in intracranial volume following reconstructive surgery for TC. However, if it does not give rise to any complaints, it can be followed-up clinically and radiologically. Patients should be followed-up for many years, given that neurodevelopmental and behavioral problems may occur later in both pathologies.
三角头畸形（TC），即额缝向矢状面变尖，眶上缘外侧不足，筛骨发育不良，眶内移导致三角头畸形，同时伴有额颞部狭窄和双颞径增宽。蛛网膜囊肿（AC）是良性囊肿，由于蛛网膜层的复制或分离，脑脊液被捕获到蛛网膜膜中而形成。AC 通常位于中颅窝和大脑外侧裂区域。它们大多通过神经影像学偶然发现。TC 和 AC 同时存在的情况相当罕见；因此，其实际发病率尚不清楚。据我们所知，在文献中仅发现了 2 篇关于非综合征性 TC 和 AC 同时存在的报道。在本病例研究中，我们介绍了 2 名患者，其中 1 名患者存在单侧、另 1 名患者存在双侧颞部局限性 AC，这些囊肿在重建手术后增大，并接受了 TC 重建手术。
我们研究中介绍的 2 名患者均为 7 个月大的男性。他们均接受了额眶前移和颅骨重塑手术。患者的 AC 未进行干预。然而，在术后随访中，第 1 例患者的左侧颞部囊肿和第 2 例患者的右侧囊肿在影像学上增大，而左侧囊肿明显缩小。
讨论/结论：TC 必须进行手术治疗。AC 可能会因 TC 重建手术后颅内容积增加而导致大小发生变化。然而，如果它没有引起任何不适，可以进行临床和影像学随访。鉴于这两种病变可能会在以后出现神经发育和行为问题，患者应进行多年随访。
