摘要: 目的：探讨蛛网膜囊肿合并慢性硬膜下血肿的临床特点与外科治疗方法。方法：回顾分析苏州大学附属第一医院神经外科及伊犁医院自2021年1月至2024年3月收治蛛网膜囊肿合并慢性硬膜下血肿患者3例，其中1例患者行开颅囊肿剥除 + 硬膜下血肿清除术，另1例患者行神经内镜下囊肿切除 + 硬膜下血肿清除术，最后1例患者行单侧硬膜下血肿钻孔引流术。回顾性分析患者的临床资料和疗效，并且结合文献(对PubMed、Embase、Cochrane Library、Medline、中国知网、万方、维普等数据库进行系统的文献检索)进行总结。结果：选择硬膜下血肿钻孔引流术与神经内镜下囊肿切除 + 硬膜下血肿清除术的两例患者为儿童，术后随访2年，蛛网膜囊肿与血肿均无复发；1例行开颅囊肿剥除 + 硬膜下血肿清除术患者为青年男性，术后复查CT存在少量硬膜外血肿，经引流后，患者术后恢复良好。通过文献复习蛛网膜囊肿合并慢性硬膜下血肿的病人，总结其临床特征及外科治疗方法。IAC合并CSDH少见，通常发生于青年人，常发生于囊肿的同侧，外伤、剧烈运动是发生慢性硬膜下血肿的主要诱因；蛛网膜囊肿合并慢性硬膜下血肿患者的治疗一般选择钻孔引流术；对于囊肿或者血肿复发的患者，无需急于再次钻孔或者急于行针对囊肿的手术，可随访观察；对于出血前囊肿就有症状或者囊肿反复出血的患者，可以在血肿清除同时切除囊肿。结论：蛛网膜囊肿合并慢性硬膜下血肿患者的外科治疗包括钻孔引流术、神经内镜下囊肿切除 + 硬膜下血肿清除、显微镜下囊肿切除 + 硬膜下血肿清除，可以取得较好的手术疗效。
Abstract: Objective: To investigate the clinical characteristics and surgical treatment of arachnoid cyst combined with chronic subdural hematoma. Methods: Review analysis of the first affiliated hospital of Suzhou university neurosurgery and Yili hospital from January 2021 to March 2024 treated arachnoid cyst with chronic subdural hematoma 3 patients, including one patient with craniotomy cyst removal + subdural hematoma removal, another patient with nerve endoscopic cyst removal + subdural hematoma removal, the last one patient with unilateral subdural hematoma drilling drainage. The clinical data and efficacy of patients were retrospectively analyzed, and the literature (systematic literature search of PubMed, Embase, Cochrane Library, Medline, CNKI, CNER, Wanfang, VIP and other databases was conducted) was summarized. Results: Two patients with drilling and drainage of subdural hematoma and neuroendoscopic cyst removal + subdural hematoma removal were selected as children, with no arachnoid cyst or hematoma recurrence; The patient was a young man, and the patient recovered well after drainage. Patients with arachnoid cysts combined with chronic subdural hematoma were reviewed through the literature, and their clinical characteristics and surgical treatment methods were summarized. IAC with CSDH is rare, usually occurs in young people, often occurring in the same side of the cyst, trauma and strenuous exercise are the main cause of chronic subdural hematoma; arachnoid cyst patients with chronic subdural hematoma generally choose drilling drainage; for patients with cyst or hematoma recurrence, there is no need to drill again or cyst surgery, follow-up observation; for patients with the cyst symptoms or repeated bleeding before bleeding, the cyst can be removed from the hematoma at the same time. Conclusion: The surgical treatment of patients with arachnoid cyst combined with chronic subdural hematoma includes drilling and drainage, neuroendoscopic cyst removal and subdural hematoma removal, microscopic cyst removal and subdural hematoma removal, which can achieve good surgical effect.
1. 引言
蛛网膜囊肿(ICA)发生率仅占颅内占位性病变的1%，往往由于胚胎发育异常，使得蛛网膜复制和分裂异常而形成，部分由于头部外伤、颅内感染、脑出血等导致颅内蛛网膜囊肿[1]；好发于儿童，大多患者无症状，部分患者因颅内占位效应或脑积水引起症状，慢性硬膜下血肿(Chronic subdural hematoma, CSDH)是陈旧血液的包裹性聚集，大部分或完全液化，位于硬脑膜和蛛网膜之间，好发于老年人，其发生的相关危险因素主要包括酗酒、糖尿病、高龄、脑萎缩、蛛网膜囊肿、凝血功能障碍、抗凝和抗血小板治疗等[2] [3]。蛛网膜囊肿合并CSDH较为少见，多见于青年患者，常发生于囊肿的同侧，外伤、剧烈运动是发生慢性硬膜下血肿的主要诱因。本文通过回顾苏州大学附属第一医院神经外科收治的3例患者，结合文献总结蛛网膜囊肿合并慢性硬膜下血肿的临床特点与外科治疗方法。
2. 临床资料与方法
2.1. 临床资料
ICA合并CSDH共3例，均为男性，2例为儿童，1例为中年，年龄11~38岁。
病例1：38岁男性，因 突发头痛1月余，加重伴头晕呕吐1周来我科就诊，入院体格检查未见明显阳性体征，头部MR (见图1)示左外侧裂蛛网膜囊肿，中线轻度向右移位；
病例2：12岁儿童，因 间断头痛1年，加重3天 入院，入院体格检查未见明显阳性体征，头颅CT示左侧额颞顶部慢性硬膜下血肿，中线轻度移位，左颞骨骨折，MR (见图2)示左额颞顶慢性硬膜下血肿，且囊肿内存在慢性血肿信号；
病例3：11岁儿童，因间断头痛伴恶心、呕吐1月入院，头颅CT (见图3)示左侧额颞顶部慢性硬膜下血肿，中线轻度右偏，侧脑室受压。
Figure 1. Case 1: Figure A shows preoperative CT, where a left sylvian fissure arachnoid cyst is visible as a low-density lesion; Figure B preoperative MR (T2) and Figure C suggest a left sylvian fissure arachnoid cyst, with T1 low signal and T2 high signal in the left frontal-temporal-parietal subdural space, indicating a possible subdural effusion mixed with a small amount of chronic hematoma, with slight displacement of the midline structures; Figure D is a postoperative CT, showing the disappearance of the chronic subdural hematoma, good re-expansion of the brain tissue, and the midline structures are centered
图1. 病例1：图A示术前CT，可见左侧侧裂区蛛网膜囊肿，呈低密度；图B术前MR (T2)和图C，提示左侧侧裂区蛛网膜囊肿，左额颞顶硬膜下腔T1低信号、T2高信号影，考虑混有少量慢性血肿的硬膜下积液可能，中线结构轻度移位；图D为术后CT，可见慢性硬膜下血肿消失，脑组织复张良好，中线结构居中
Figure 2. Figure A shows the surgical incision; Figure B shows a preoperative CT indicating a chronic subdural hematoma in the left frontotemporal-parietal region with a right-shifted midline; Figure C shows a preoperative MR (T2) indicating a chronic subdural hematoma in the left frontotemporal-parietal region, with an associated arachnoid cyst in the left temporal pole, and chronic blood clot signals within the cyst; Figure D shows a postoperative CT two years later indicating the resolution of the mass effect from the arachnoid cyst, with good re-expansion of the surrounding brain tissue
图2. 图A为手术切口；图B为术前CT示左额颞顶慢性硬膜下血肿，中线右偏；图C为术前MR (T2)左额颞顶慢性硬膜下血肿，合并左侧颞极蛛网膜囊肿，且囊肿内存在慢性血肿信号；图D为术后两年CT示蛛网膜囊肿占位效应解除，囊肿周围脑组织复张良好
Figure 3. Figures (A) and (B) are preoperative CT scans showing a chronic subdural hematoma in the left frontal-temporal-parietal region, presenting as isodense, with significant rightward midline shift. Figures (C) and (D) are postoperative CT scans, showing the disappearance of the subdural hematoma and good cerebral re-expansion; however, an isodense chronic hematoma is still visible at the temporal pole, which is blood accumulation within an arachnoid cyst. It is considered that due to the lack of communication between the arachnoid and the subdural space, the hematoma is confined within the cyst cavity, and therefore the subdural drainage tube did not fully drain the hematoma within the cyst
图3. 图(A)、(B)为术前CT，可见左额颞顶慢性硬膜下血肿，呈等密度，中线右偏明显。图(C)、(D)为术后CT，可见硬膜下血肿消失，脑复张良好；但颞极仍可见等密度的慢性血肿，为蛛网膜囊肿内积血，考虑因蛛网膜与硬膜下腔未沟通，而局限于囊肿腔内，故硬膜下引流管未能将囊肿内血肿充分引流
2.2. 外科治疗方法
常规开颅，尽可能多地切除外侧囊肿壁，如囊肿壁与血管等重要结构粘连不甚紧密则争取全切囊肿；同时尽可能广泛地打通囊腔与周围大的脑池或脑室之间的交通，使囊内液体充分参与脑脊液循环。
病例1：开颅囊肿剥除 + 硬膜下血肿清除术。
病例2：神经内镜下开颅血肿清除 + 蛛网膜囊肿切除术。
病例3：钻孔引流术。
3. 结果
中年男性患者术中打通颈内动脉池(见图4)，术后患者复查CT提示少量硬膜外血肿，脑组织复张良好，中线结构居中，患者间断性头晕、头痛，经引流后，患者术后恢复良好，头痛、恶心呕吐症状同样消失，囊壁组织送病理检查，见囊肿壁为纤维结缔组织和少量蛛网膜细胞。11岁儿童行单纯硬膜下钻孔引流术，术后患者头痛缓解，随访2年余，未见复发；12岁儿童术中打开硬脑膜，可见暗红色陈旧性液体流出，使用神经内镜四周探查，可见颞部蛛网膜囊肿包裹慢性硬膜下血肿，镜下全切(见图5)，术后随访1年，影像学检查提示囊肿占位效应解除，囊肿周围脑组织复张良好。
Figure 4. Case 1, upon opening the dura mater, a typical chronic subdural hematoma wall structure was visible. The wall layer was incised along the bone window, and the chronic subdural hematoma wall structure was completely resected. The arachnoid cyst wall layer was separated and completely resected, revealing multiple venous attachments on the wall layer, which were considered to be the responsible vessels for the chronic hematoma. The visceral layer of the arachnoid cyst was separated and resected, and the arachnoid around the internal carotid artery was opened, allowing communication between the cyst and the suprasellar cistern
图4. 病例1：打开硬膜可见典型的慢性硬膜下血肿壁层结构，沿骨窗切开壁层，完整切除慢性硬膜下血肿壁层结构，分离蛛网膜囊肿壁层，完整切除蛛网膜囊肿壁层，可见壁层结构多处静脉附着，考虑为慢性血肿的责任血管，分离、切除蛛网膜囊肿脏层，打通颈内动脉周围蛛网膜，使囊肿与鞍上池沟通
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Figure 5. Shows intraoperative images: after opening the dura mater, the chronic subdural hematoma was suctioned, revealing the vascularized wall layer. Visible brain surface veins were adhered to the proliferated wall layer, which were coagulated and cut. The arachnoid cyst wall was incised, and the hematoma inside the cyst was largely suctioned as it communicated with the subdural space. The thickened arachnoid cyst wall layer was resected, and the accumulated blood inside the cyst was suctioned, revealing the structures within the subdural and arachnoid cysts. The cyst wall was further resected, and veins attached to the arachnoid cyst wall layer were visible. The responsible blood vessels were considered, as the tension of the cyst caused high tension in the veins, leading to bleeding. The front showed a passage formed by the communication between the cyst and the subdural cavity
图5. 术中图片示：打开硬膜，吸除硬膜下慢性血肿，可见血管增生的壁层，脑表面静脉可见与壁层增生黏连的系带，予以电凝切断，切开蛛网膜囊肿壁，其囊内血肿因与硬膜下相通，清除硬膜下血肿时已大部吸除，切除增厚的蛛网膜囊肿壁层囊壁，吸除囊肿内积血，可见硬膜下和蛛网膜囊肿内结构；扩大切除囊壁，蛛网膜囊肿壁层上可见附着的静脉，考虑责任血管可能，因囊肿张力导致静脉高张力，进而出血，前方为囊肿和硬膜下腔形成沟通的孔道
4. 讨论
本文回顾分析医院收治的蛛网膜囊肿合并慢性硬膜下血肿患者，共3例，选择硬膜下血肿钻孔引流术与神经内镜下囊肿切除 + 硬膜下血肿清除术的两例患者为儿童，术后随访2年，蛛网膜囊肿与血肿均无复发；1例行开颅囊肿剥除 + 硬膜下血肿清除术患者为青年男性，术后复查CT存在少量硬膜外血肿，经引流后，患者术后恢复良好。
对蛛网膜囊肿的的第一次描述可以追溯到19世纪的一份尸检报告[4]，蛛网膜囊肿通常是偶然发现的，一般无明显症状，轻微外伤后可出现硬膜下血肿、硬膜下积液、囊内出血等并发症[5]，通常发生于儿童和青年人，慢性硬膜下血肿较为少见，外伤、剧烈运动是发生慢性硬膜下血肿的主要诱因，结合本文3例术中所见，囊肿壁层通常有静脉附着或黏连，囊肿扩张使静脉张力增高，故蛛网膜囊肿患者轻微外伤后可出现硬膜下血肿、硬膜下积液、囊内出血等并发症；老年人中慢性硬膜下血肿常见的诱发因素还包括弥漫性脑萎缩和抗凝药物的使用，但这些因素在儿童人群中很少见[3]。影像学上颞部膨出和或颅骨变薄是蛛网膜囊肿合并硬膜下血肿的诊断性表现，提示可能伴有囊内或硬膜下出血[6] [7]，对于常规体检发现蛛网膜囊肿的青年患者，尤其是影像学检查显示颅骨变薄或外凸的患者，应当提醒其避免发生创伤性脑损伤，避免导致硬膜下血肿[8]，在某些情况下，CT很难将AC与亚急性或慢性囊内或硬膜下血肿区分开来，MR是首选的放射学检查方法[9]；极少数情况下，硬膜下血肿会在头部外伤后消散，或在无症状病例的随访过程中自发消失，最常见的位置是中颅窝，部分病例会有一定的症状，可能与较大囊肿的占位效应或颅内压升高有关，颅内压升高是由于球阀机制或囊腔内蛛网膜细胞的分泌活动导致囊内液体与蛛网膜下腔连通所致[10]。
慢性硬膜下血肿是神经外科常见疾病之一，大多数SDH发生在创伤引起的桥静脉破裂，不到5%的SDH可能会自发发生，血管畸形、动脉瘤和瘘管形成可能是一小部分自发性SDH的基础[11]，其发生的相关危险因素还包括酗酒、糖尿病、高龄、脑萎缩、蛛网膜囊肿、凝血功能障碍、抗凝和抗血小板治疗等，蛛网膜囊肿的存在，可能会增加硬膜下血肿发生的风险[12]。
蛛网膜囊肿合并慢性硬膜下血肿的治疗取决于临床表现并考虑囊肿的大小和位置。头痛是最常见的症状，可出现在任何部位的囊肿，幕上囊肿常与癫痫发作有关[13]。无症状患者可以保守观察，而有症状患者则适合手术治疗。对于并发的早期硬膜下血肿可能需要手术减压。尽管颅内蛛网膜囊肿的手术治疗存在一定争议[14]，大多数情况下，蛛网膜囊肿合并慢性硬膜下血肿患者的治疗一般选择钻孔引流术；对于囊肿或者血肿复发的患者，无需急于再次钻孔或者急于行针对囊肿的手术，可随访观察；对于出血前囊肿就有症状或者囊肿反复出血的患者，且囊肿直径 ≥ 5 cm，可以在血肿清除同时切除囊肿，打通囊肿腔与蛛网膜下腔[15]；本文第二例虽存在囊肿内积血，但囊肿较小，仅作壁层切除有效降低附着的静脉张力，也获得较好的手术效果；本文第一例患者行开颅手术，快速清除血肿并切除薄膜，沟通囊肿腔与脑脊液循环，避免了囊肿再次复发。部分学者认为手术适应症还包括：局部脑组织受压移位，有明显颅内压增高症状；存在局灶性神经系统体征；有癫痫发作者；囊肿有增大趋势者。
综上所述，随着CT和MRI技术的广泛运用，偶发性蛛网膜囊肿的诊断率以及对其潜在并发症的预测能力得到了显著提升。认识到硬膜下出血可能与蛛网膜囊肿破裂相关，这一点至关重要。因此，对于蛛网膜囊肿合并慢性硬膜下血肿的患者，无症状者可采取密切观察的策略，而出现症状的患者则更适合进行手术治疗。外科治疗手段包括钻孔引流术、神经内镜下囊肿切除加硬膜下血肿清除术以及显微镜下囊肿切除加硬膜下血肿清除术，这些方法均能获得良好的手术效果。
然而，在选择具体手术方式时，需综合考虑患者囊肿的大小、位置、是否复发以及是否存在明显的临床症状等因素。例如，对于囊肿较小且无明显临床症状的患者，采取钻孔引流术即可有效缓解病情；而对于囊肿较大、有复发趋势或伴有明显颅内压增高症状的患者，则可能需要采取更为复杂的手术方式，如神经内镜下囊肿切除加硬膜下血肿清除术或显微镜下囊肿切除加硬膜下血肿清除术。
此外，术后随访也至关重要。通过定期随访观察，可以及时发现和处理可能的并发症，如囊肿复发、硬膜下血肿再次形成等，从而进一步提高手术治疗的效果和患者的预后。
总之，对于蛛网膜囊肿合并慢性硬膜下血肿的患者，应根据具体情况制定个性化的治疗方案，并在术后进行密切的随访观察，以确保患者的治疗效果和生命安全。
然而，由于病例的罕见，本文仅有3例病例，无法进行有效的统计分析，难以得出可靠的结论。
该病例报道已获得病人的知情同意。
NOTES
*共同第一作者。
#通讯作者。