Int J Clin Exp Med 2015;8(9):16117-16121 www.ijcem.com /ISSN:1940-5901/IJCEM0009522
Original Article Recurrent cervicodorsal spinal intradural enterogenous cyst: case report and literature review Zhi-Gang He, Zong-Fang Wu, Xiao-Hua Xia, Ai-Jun Xu, Tao Zhang, Hong-Bing Xiang Department of Anesthesiology and Pain Medicine, Tongji Hospital of Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei, P. R. China Received April 23, 2015; Accepted June 21, 2015; Epub September 15, 2015; Published September 30, 2015 Abstract: This report described a recurrent enterogenous cyst of the cervicodorsal spinal canal occurred in an 8-year-old boy who experienced cervical back pain at the age of 5. He had been operated for mass lesion at the same level 3 years ago. The cervical and thoracic spine MRI showed a large intradural cyst at C7-T1. The cyst was subtotally removed via posterior approach using a laminectomy. Based on the results of immunostaining, it was identified as an enterogenous cyst. A literature review related to spinal cyst is also included. Keywords: Recurrent enterogenous cyst, cervical spinal canal
Introduction The cause of cervical spinal cord compression consists of cervical disc protruding, traumatic cervical injury, cervical spine instability, and spinal canal mass lesion [1]. Several studies have demonstrated that intraspinal cyst usually affects young patient with cervical spinal mass lesion [2-6]. Embryologically, the cyst is suggested to be related to congenital maldevelopment of endodermal tissue in the spinal canal and the neural plaque [2, 7], thus, the endodermal cysts should be considered in the diagnosis of spinal solitary cystic mass. It is known that the endodermal cysts usually include the enterogenous and neuroepithelial cyst, and develop in the subdural space in the anterior spinal cord [8]. Because an endodermal cyst wall includes columnar epithelium, its layer and its cells have the ability to grow and produce secretions, the cyst should be subtotally excised [9]. We report here a case of a recurrent enterogenous cyst in the cervicodorsal spinal canal. Case report An 8-year old boy presented with cervical back pain for 1 week. His nocturnal pain symptom became progressively worse. He didn’t experi-
ence weakness and paraesthesia of double upper extremity. He had been operated for mass lesion at the cervicothoracic spine level 3 years ago, and a postoperative diagnosis of enterogenous cyst was made. A MRI film of the cervicothoracic spine in November 9, 2014 showed a cystic mass lesion ventral to the spinal cord and lying at the level from C7 to T1 (Figure 1A and 1B). Written informed consent was obtained from the patient and his family before preparation and submission of this manuscript. The patient underwent preoperative evaluation using a standard protocol. The following parameters were recorded during surgery: electrocardiogram, non-invasive blood pressure, pulse oximetry, body temperature, inspired and expired anesthetic concentration. In the operating room, after he was lightly sedated with 1 mg intravenous midazolam, anesthesia was induced with intravenous fentanyl 2 μg/kg, propofol 2.5 mg/kg. Intravenous administration of rocuronium 0.8 mg/kg was used to facilitate endotracheal intubation with a 5.0-mm-ID endotracheal tube (Cormack and Lehane grade I). 1%-1.5% isoflurane and 0.1-0.2 mg/kg/min propofol were used to maintain general anesthesia and PaCO2 between 35-40 mmHg [1017]. Intermittent doses of vecuronium were
Recurrent cervical spinal enterogenous cyst
Figure 1. Composite photographs of preoperative (A, B) and postoperative (C, D) MRI images in sagittal view (left) and axial view (right). Sagittal T2-weighted MRI of the cervical spine (A) showed a large cystic lesion at C7-T1 filling almost all of the entire spinal canal. The C7-T1 spinal cord has been displaced posteriorly by enterogenous cysts (blue arrows). 6 d after surgery, sagittal Tl-weighted sequence MRI images (C, D) showed that lesion at C7-T1 was dissected and removed.
Figure 2. Photographs of microscopic cross-section from the lesion at C7-T1. Histological examination of the material from the lesion at C7-T1 revealed a cyst wall line by mucin-secreting columnar epithelium (hematoxylin & eosin staining, magnification × 400).
administered to maintain skeletal muscle paralysis [18, 19]. The depth of anesthesia was monitored by intraoperative recording bispectral index (BIS) [20-22]. 16118
Under general anesthesia, the boy was placed in prone position with the neck extended. He was subjected to operate on via posterior approach using a C6-T1 laminectomy. After opening the dura, the spinal cord and a well-demarcated encapsulated cyst were exposed by blunt dissection. It was observed that the cyst caused no obvious compression to spinal cord. The cyst was fragile and was removed along its axis as completely as possible and excised from their attachments using microscope. The operation lasted 5 h. After confirmation of neuromuscular recovery, the patient was extubated in supine position and transported to the post-anesthesia care unit (PACU). The patient was monitored for 1 h in the PACU. There were no complications. After he was conscious and his vital signs were stable, he was discharged to the ward. The preoperative pain symptoms disappeared 5 days after surgery. No additional signs related to the cervical nerves were observed. Pathologically, the mass lesion was diagnosed as an enterogenous cyst (Figure 2). With the aid of postoperative magnetic resonance imaging, the mass lesion within the C7-T1 spinal cord was removed (Figure 1C and 1D). The patient was discharged and followed up for 4 month, with no symptoms.
Discussion The neurenteric cyst usually located intradurally or extramedullary in the ventral cervicodorsal Int J Clin Exp Med 2015;8(9):16117-16121
Recurrent cervical spinal enterogenous cyst spinal canal is an uncommon congenital lesion that usually presents with focal neurological signs [12, 15, 23-30]. Cystic changes within spinal cord pose difficult diagnostic and therapeutic problem by using computed tomography (CT) or magnetic resonance imaging (MRI). The endodermal origin of the cyst may be distinguished from the enterogenous or neuroepithelial cyst. Pathological examination reveals that the cyst wall includes the outer layer (smooth muscle and connective tissue) and the inner layer (columnar epithelium cells that produce mucin and mucopolysaccharide). It is generally considered that the increase of secretion from the cells lining the endodermal cyst cause rapid enlargement of the cyst contents, leading to spinal cord compression, therefore, an endodermal cyst should be removed as completely as possible. The surgical approach to mass lesion in the spinal canal depends on the location and accessibility of the mass lesion. Many reports have shown that the neurenteric cyst has been operated on via a posterior approach using a laminectomy [31-34]. Despite the fact that the neurenteric cyst is usually located ventral to the spinal cord [35, 36], the study of Tuzun et al indicated that the complete cyst removal may result in damaging the spinal cord via posterior approach, and this method is generally considered to be easier and safer than the anterior approach, especially in children [31]. Takase et al reported a case of a recurrent intradural cervical neurenteric cyst operated on using an anterior approach in a 42-year-old woman [32], and she presented with a cervical neurenteric cyst that had recurred eight years after its partial removal via a posterior approach. The patient complained of pain on the lateral side of her upper arms, and a magnetic resonance imaging showed that the recurrent cyst was located ventral to the spinal cord and compressed the cord dorsally at the C4-6 level. It is known that enterogenous cysts are uncommon congenital lesions mostly located in the lower cervical region of the spinal canal and have only rarely been observed in the upper cervical and lumbar spine region. Tuzun et al reported a case of neurenteric cyst of the upper cervical spine in an 11-year-old female child [31], and she presented with the symptoms of neck pain that radiated to both shoulders and arms. Spine MRI scans revealed a cystic mass 16119
lesion ventral to the C1-3 spinal cord without enhancement after gadolinium injection. No postoperative complications were noted after the cystic lesion was surgically removed via a posterior approach. Choi et al also reported a 15-year-old boy having an intraspinal neurenteric cyst located at cervical spine, and he presented with symptoms of neck pain and both shoulders pain for 2 months [37]. Magnetic resonance imaging of the patient showed an intradural extramedullary cystic mass at the C1-3 level without enhancement after gadolinium injection. After hemilaminectomy at the C1-3 levels was performed and the lesion was completely removed through a posterior approach, histological examination showed the cystic wall lined with ciliated pseudostratified columnar epithelium containing mucinous contents. Marchionni et al reported a case of upper cervical cord enterogenous cyst occurred in a 76-year-old woman presenting with tetraparesis, left-sided hemisensory loss and occasional neck pain [8]. Her cervical magnetic resonance imaging showed a partially cystic lesion compressing the C2-4 spinal cord. Complete cyst resection was performed. Pathological examination revealed that of an enterogenous cyst. Arslan et al reported a 24-year old woman with a recurrent enterogenous cyst in the L2 lumbar spinal canal [38], and she presented with lower back and left radicular leg pain for 1 year and underwent complete cyst resection via a posterior approach. She experienced an operation on for mass lesion at the same level 10 years ago. Shuangshoti et al reported a sudden paraparesis case induced by intraspinal cervicodorsal mass lesion [7]. He experienced sudden paraparesis in 6-year-old, and subsequently underwent repeated surgical explorations. At the age of 20 years, the mass lesion was subtotally removed, and the histopathological diagnosis was that the lesion was an intraspinal genuine endodermal epithelial cyst. Conclusions In conclusion, notwithstanding some discrepancies, complete cyst resection is generally not advocated via a posterior approach because there exists the high risk of the spinal cord injury. Further studies are needed to clarify the underlying mechanisms for the recurrence of the intraspinal enterogenous cysts after cyst aspiration and partly resection. Int J Clin Exp Med 2015;8(9):16117-16121
Recurrent cervical spinal enterogenous cyst Disclosure of conflict of interest None. Address correspondence to: Tao Zhang, Department of Anesthesiology and Pain Medicine, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei, P. R. China. E-mail: [email protected]
References [1]
Linge SO, Mardal KA, Helgeland A, Heiss JD and Haughton V. Effect of craniovertebral decompression on CSF dynamics in Chiari malformation type I studied with computational fluid dynamics: Laboratory investigation. J Neurosurg Spine 2014; 21: 559-564. [2] Chen IH, Kao KP, Penn IW and Ho DM. Double intraspinal enterogenous cysts. J Neurol Neurosurg Psychiatry 1995; 58: 110-111. [3] Agnoli AL, Laun A and Schonmayr R. Enterogenous intraspinal cysts. J Neurosurg 1984; 61: 834-840. [4] Holmes GL, Trader S and Ignatiadis P. Intraspinal enterogenous cysts. A case report and review of pediatric cases in the literature. Am J Dis Child 1978; 132: 906-908. [5] Lea ME, Sage MR, Bills D, Brophy B and Blumbergs P. Enterogenous cyst of the cervical spinal canal. Australas Radiol 1992; 36: 327329. [6] Miyagi K, Mukawa J, Mekaru S, Ishikawa Y, Kinjo T and Nakasone S. Enterogenous cyst in the cervical spinal canal. Case report. J Neurosurg 1988; 68: 292-296. [7] Shuangshoti S, Kasantikul V, Taecholarn C and Wangsuphachart S. Symptomatic intraspinal genuine endodermal epithelial cyst. J Med Assoc Thai 1989; 72: 295-301. [8] Marchionni M, Tzerakis N, Tsang K and Carey M. Upper cervical cord enterogenous cyst mimicking transient ischaemic attacks. BMJ Case Rep 2013; 2013. [9] Nakajima M, Miyajima M, Hishii M, Arai H, Sato K and Fujii H. Endodermal cyst of the quadrigeminal cistern: case report. Surg Neurol 2001; 56: 385-388; discussion 388-389. [10] Chaitanya G, Arivazhagan A, Sinha S, Reddy KR, Thennarasu K, Bharath RD, Rao MB, Chandramouli BA and Satishchandra P. Dexmedetomidine anesthesia enhances spike generation during intra-operative electrocorticography: A promising adjunct for epilepsy surgery. Epilepsy Res 2015; 109: 65-71. [11] Yao W, Qiu J, Zhou Z, Zhang L and Zhang C. Cervical spinal cord compression after thyroidectomy under general anesthesia. J Anesth 2014; 28: 125-127.
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[12] Liu TT, He ZG, Tian XB, Liu C, Xiang HB and Zhang JG. Hypothesis: Astrocytes in the central medial amygdala may be implicated in sudden unexpected death in epilepsy by melanocortinergic signaling. Epilepsy Behav 2015; 42: 4143. [13] Ye DW, Liu C, Liu TT, Tian XB and Xiang HB. Motor cortex-periaqueductal gray-spinal cord neuronal circuitry may involve in modulation of nociception: a virally mediated transsynaptic tracing study in spinally transected transgenic mouse model. PLoS One 2014; 9: e89486. [14] Xiang HB, Liu TT, Tian XB and Zhu WZ. Therapeutic mechanism of subthalamic nucleus stimulation for refractory epilepsy involved in melanocortin-4 receptor signaling. Molecular & Cellular Epilepsy 2014; 1: 13-18. [15] Xiang HB, Liu C, Liu TT and Xiong J. Central circuits regulating the sympathetic outflow to lumbar muscles in spinally transected mice by retrograde transsynaptic transport. Int J Clin Exp Pathol 2014; 7: 2987-2997. [16] Liu TT, He ZG, Tian XB and Xiang HB. Neural mechanisms and potential treatment of epilepsy and its complications. Am J Transl Res 2014; 6: 625-630. [17] Liu C, Liu TT, He ZG, Shu B and Xiang HB. Inhibition of itch-related responses by selectively ablated serotonergic signals at the rostral ventromedial medulla in mice. Int J Clin Exp Pathol 2014; 7: 8917-8921. [18] Iwasaki H, Takahoko K, Otomo S, Sasakawa T and Kunisawa T. Monitoring of neuromuscular blockade in one muscle group alone may not reflect recovery of total muscle function in patients with ocular myasthenia gravis. Can J Anaesth 2013; 60: 1222-1227. [19] Eikermann M, Groeben H, Husing J and Peters J. Accelerometry of adductor pollicis muscle predicts recovery of respiratory function from neuromuscular blockade. Anesthesiology 2003; 98: 1333-1337. [20] Girgirah K and Quinn A. Reliability of bispectral index analysis in patients undergoing Caesarean section. Br J Anaesth 2015; 114: 530. [21] Kim H, Lim BG and Lee SY. Transcranial electrical stimulations given for motor-evoked potentials as the cause for elevated bispectral index and entropy during spine surgery. J Neurosurg Anesthesiol 2013; 25: 217-219. [22] Witkowska M, Karwacki Z, Rzaska M, Niewiadomski S and Sloniewski P. Comparison of target controlled infusion and total intravenous anaesthesia with propofol and remifentanil for lumbar microdiscectomy. Anaesthesiol Intensive Ther 2012; 44: 138-144. [23] Visvanathan V, Crowther J and Taylor W. Neurenteric cyst presenting with stridor and dysphagia: case report and literature review. Scott Med J 2015; 60: e4-7.
Int J Clin Exp Med 2015;8(9):16117-16121
Recurrent cervical spinal enterogenous cyst [24] Savage JJ, Casey JN, McNeill IT and Sherman JH. Neurenteric cysts of the spine. J Craniovertebr Junction Spine 2010; 1: 58-63. [25] Rotondo M, D’Avanzo R, Natale M, Pasqualetto L, Bocchetti A, Agozzino L and Scuotto A. Intramedullary neurenteric cysts of the spine. Report of three cases. J Neurosurg Spine 2005; 2: 372-376. [26] Lippman CR, Arginteanu M, Purohit D, Naidich TP and Camins MB. Intramedullary neurenteric cysts of the spine. Case report and review of the literature. J Neurosurg 2001; 94: 305-309. [27] Felix RW, Seidel GK, Murphy EB and Rosenblum ML. Conus medullaris enterogenous cyst. PM R 2012; 4: 698-700. [28] Balasubramaniam S, Tyagi DK and Sawant HV. Intramedullary enterogenous cyst of the conus medullaris presenting as lower limb pain. J Craniovertebr Junction Spine 2011; 2: 46-48. [29] Xu LJ, Liu TT, He ZG, Hong QX and Xiang HB. Hypothesis: CeM-RVLM circuits may be implicated in sudden unexpected death in epilepsy by melanocortinergic-sympathetic signaling. Epilepsy Behav 2015; 45: 124-127. [30] Ye DW, Liu C, Tian XB and Xiang HB. Identification of neuroanatomic circuits from spinal cord to stomach in mouse: retrograde transneuronal viral tracing study. Int J Clin Exp Pathol 2014; 7: 5343-5347. [31] Tuzun Y, Izci Y, Sengul G, Erdogan F and Suma S. Neurenteric cyst of the upper cervical spine: excision via posterior approach. Pediatr Neurosurg 2006; 42: 54-56.
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[32] Takase T, Ishikawa M, Nishi S, Aoki T, Wada E, Owaki H, Katsuki T and Fukuda H. A recurrent intradural cervical neurenteric cyst operated on using an anterior approach: a case report. Surg Neurol 2003; 59: 34-39; discussion 39. [33] Chavda SV, Davies AM and Cassar-Pullicino VN. Enterogenous cysts of the central nervous system: a report of eight cases. Clin Radiol 1985; 36: 245-251. [34] Kim CY, Wang KC, Choe G, Kim HJ, Jung HW, Kim IO, Chi JG and Cho BK. Neurenteric cyst: its various presentations. Childs Nerv Syst 1999; 15: 333-341. [35] Arai Y, Yamauchi Y, Tsuji T, Fukasaku S, Yokota R and Kudo T. Spinal neurenteric cyst. Report of two cases and review of forty-one cases reported in Japan. Spine (Phila Pa 1976) 1992; 17: 1421-1424. [36] Brooks BS, Duvall ER, el Gammal T, Garcia JH, Gupta KL and Kapila A. Neuroimaging features of neurenteric cysts: analysis of nine cases and review of the literature. AJNR Am J Neuroradiol 1993; 14: 735-746. [37] Choi DY, Lee HJ, Shin MH and Kim JT. Solitary cervical neurenteric cyst in an adolescent patient. J Korean Neurosurg Soc 2015; 57: 135139. [38] Arslan E, Cakir E, Kuzeyli K, Guvercin AR, Gazioglu G, Arslan EA and Reis A. Recurrent lumbar spinal intradural enterogenous cyst: a case report. Turk Neurosurg 2010; 20: 402405.
Int J Clin Exp Med 2015;8(9):16117-16121
Original Article Recurrent cervicodorsal spinal intradural enterogenous cyst: case report and literature review Zhi-Gang He, Zong-Fang Wu, Xiao-Hua Xia, Ai-Jun Xu, Tao Zhang, Hong-Bing Xiang Department of Anesthesiology and Pain Medicine, Tongji Hospital of Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei, P. R. China Received April 23, 2015; Accepted June 21, 2015; Epub September 15, 2015; Published September 30, 2015 Abstract: This report described a recurrent enterogenous cyst of the cervicodorsal spinal canal occurred in an 8-year-old boy who experienced cervical back pain at the age of 5. He had been operated for mass lesion at the same level 3 years ago. The cervical and thoracic spine MRI showed a large intradural cyst at C7-T1. The cyst was subtotally removed via posterior approach using a laminectomy. Based on the results of immunostaining, it was identified as an enterogenous cyst. A literature review related to spinal cyst is also included. Keywords: Recurrent enterogenous cyst, cervical spinal canal
Introduction The cause of cervical spinal cord compression consists of cervical disc protruding, traumatic cervical injury, cervical spine instability, and spinal canal mass lesion [1]. Several studies have demonstrated that intraspinal cyst usually affects young patient with cervical spinal mass lesion [2-6]. Embryologically, the cyst is suggested to be related to congenital maldevelopment of endodermal tissue in the spinal canal and the neural plaque [2, 7], thus, the endodermal cysts should be considered in the diagnosis of spinal solitary cystic mass. It is known that the endodermal cysts usually include the enterogenous and neuroepithelial cyst, and develop in the subdural space in the anterior spinal cord [8]. Because an endodermal cyst wall includes columnar epithelium, its layer and its cells have the ability to grow and produce secretions, the cyst should be subtotally excised [9]. We report here a case of a recurrent enterogenous cyst in the cervicodorsal spinal canal. Case report An 8-year old boy presented with cervical back pain for 1 week. His nocturnal pain symptom became progressively worse. He didn’t experi-
ence weakness and paraesthesia of double upper extremity. He had been operated for mass lesion at the cervicothoracic spine level 3 years ago, and a postoperative diagnosis of enterogenous cyst was made. A MRI film of the cervicothoracic spine in November 9, 2014 showed a cystic mass lesion ventral to the spinal cord and lying at the level from C7 to T1 (Figure 1A and 1B). Written informed consent was obtained from the patient and his family before preparation and submission of this manuscript. The patient underwent preoperative evaluation using a standard protocol. The following parameters were recorded during surgery: electrocardiogram, non-invasive blood pressure, pulse oximetry, body temperature, inspired and expired anesthetic concentration. In the operating room, after he was lightly sedated with 1 mg intravenous midazolam, anesthesia was induced with intravenous fentanyl 2 μg/kg, propofol 2.5 mg/kg. Intravenous administration of rocuronium 0.8 mg/kg was used to facilitate endotracheal intubation with a 5.0-mm-ID endotracheal tube (Cormack and Lehane grade I). 1%-1.5% isoflurane and 0.1-0.2 mg/kg/min propofol were used to maintain general anesthesia and PaCO2 between 35-40 mmHg [1017]. Intermittent doses of vecuronium were
Recurrent cervical spinal enterogenous cyst
Figure 1. Composite photographs of preoperative (A, B) and postoperative (C, D) MRI images in sagittal view (left) and axial view (right). Sagittal T2-weighted MRI of the cervical spine (A) showed a large cystic lesion at C7-T1 filling almost all of the entire spinal canal. The C7-T1 spinal cord has been displaced posteriorly by enterogenous cysts (blue arrows). 6 d after surgery, sagittal Tl-weighted sequence MRI images (C, D) showed that lesion at C7-T1 was dissected and removed.
Figure 2. Photographs of microscopic cross-section from the lesion at C7-T1. Histological examination of the material from the lesion at C7-T1 revealed a cyst wall line by mucin-secreting columnar epithelium (hematoxylin & eosin staining, magnification × 400).
administered to maintain skeletal muscle paralysis [18, 19]. The depth of anesthesia was monitored by intraoperative recording bispectral index (BIS) [20-22]. 16118
Under general anesthesia, the boy was placed in prone position with the neck extended. He was subjected to operate on via posterior approach using a C6-T1 laminectomy. After opening the dura, the spinal cord and a well-demarcated encapsulated cyst were exposed by blunt dissection. It was observed that the cyst caused no obvious compression to spinal cord. The cyst was fragile and was removed along its axis as completely as possible and excised from their attachments using microscope. The operation lasted 5 h. After confirmation of neuromuscular recovery, the patient was extubated in supine position and transported to the post-anesthesia care unit (PACU). The patient was monitored for 1 h in the PACU. There were no complications. After he was conscious and his vital signs were stable, he was discharged to the ward. The preoperative pain symptoms disappeared 5 days after surgery. No additional signs related to the cervical nerves were observed. Pathologically, the mass lesion was diagnosed as an enterogenous cyst (Figure 2). With the aid of postoperative magnetic resonance imaging, the mass lesion within the C7-T1 spinal cord was removed (Figure 1C and 1D). The patient was discharged and followed up for 4 month, with no symptoms.
Discussion The neurenteric cyst usually located intradurally or extramedullary in the ventral cervicodorsal Int J Clin Exp Med 2015;8(9):16117-16121
Recurrent cervical spinal enterogenous cyst spinal canal is an uncommon congenital lesion that usually presents with focal neurological signs [12, 15, 23-30]. Cystic changes within spinal cord pose difficult diagnostic and therapeutic problem by using computed tomography (CT) or magnetic resonance imaging (MRI). The endodermal origin of the cyst may be distinguished from the enterogenous or neuroepithelial cyst. Pathological examination reveals that the cyst wall includes the outer layer (smooth muscle and connective tissue) and the inner layer (columnar epithelium cells that produce mucin and mucopolysaccharide). It is generally considered that the increase of secretion from the cells lining the endodermal cyst cause rapid enlargement of the cyst contents, leading to spinal cord compression, therefore, an endodermal cyst should be removed as completely as possible. The surgical approach to mass lesion in the spinal canal depends on the location and accessibility of the mass lesion. Many reports have shown that the neurenteric cyst has been operated on via a posterior approach using a laminectomy [31-34]. Despite the fact that the neurenteric cyst is usually located ventral to the spinal cord [35, 36], the study of Tuzun et al indicated that the complete cyst removal may result in damaging the spinal cord via posterior approach, and this method is generally considered to be easier and safer than the anterior approach, especially in children [31]. Takase et al reported a case of a recurrent intradural cervical neurenteric cyst operated on using an anterior approach in a 42-year-old woman [32], and she presented with a cervical neurenteric cyst that had recurred eight years after its partial removal via a posterior approach. The patient complained of pain on the lateral side of her upper arms, and a magnetic resonance imaging showed that the recurrent cyst was located ventral to the spinal cord and compressed the cord dorsally at the C4-6 level. It is known that enterogenous cysts are uncommon congenital lesions mostly located in the lower cervical region of the spinal canal and have only rarely been observed in the upper cervical and lumbar spine region. Tuzun et al reported a case of neurenteric cyst of the upper cervical spine in an 11-year-old female child [31], and she presented with the symptoms of neck pain that radiated to both shoulders and arms. Spine MRI scans revealed a cystic mass 16119
lesion ventral to the C1-3 spinal cord without enhancement after gadolinium injection. No postoperative complications were noted after the cystic lesion was surgically removed via a posterior approach. Choi et al also reported a 15-year-old boy having an intraspinal neurenteric cyst located at cervical spine, and he presented with symptoms of neck pain and both shoulders pain for 2 months [37]. Magnetic resonance imaging of the patient showed an intradural extramedullary cystic mass at the C1-3 level without enhancement after gadolinium injection. After hemilaminectomy at the C1-3 levels was performed and the lesion was completely removed through a posterior approach, histological examination showed the cystic wall lined with ciliated pseudostratified columnar epithelium containing mucinous contents. Marchionni et al reported a case of upper cervical cord enterogenous cyst occurred in a 76-year-old woman presenting with tetraparesis, left-sided hemisensory loss and occasional neck pain [8]. Her cervical magnetic resonance imaging showed a partially cystic lesion compressing the C2-4 spinal cord. Complete cyst resection was performed. Pathological examination revealed that of an enterogenous cyst. Arslan et al reported a 24-year old woman with a recurrent enterogenous cyst in the L2 lumbar spinal canal [38], and she presented with lower back and left radicular leg pain for 1 year and underwent complete cyst resection via a posterior approach. She experienced an operation on for mass lesion at the same level 10 years ago. Shuangshoti et al reported a sudden paraparesis case induced by intraspinal cervicodorsal mass lesion [7]. He experienced sudden paraparesis in 6-year-old, and subsequently underwent repeated surgical explorations. At the age of 20 years, the mass lesion was subtotally removed, and the histopathological diagnosis was that the lesion was an intraspinal genuine endodermal epithelial cyst. Conclusions In conclusion, notwithstanding some discrepancies, complete cyst resection is generally not advocated via a posterior approach because there exists the high risk of the spinal cord injury. Further studies are needed to clarify the underlying mechanisms for the recurrence of the intraspinal enterogenous cysts after cyst aspiration and partly resection. Int J Clin Exp Med 2015;8(9):16117-16121
Recurrent cervical spinal enterogenous cyst Disclosure of conflict of interest None. Address correspondence to: Tao Zhang, Department of Anesthesiology and Pain Medicine, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan 430030, Hubei, P. R. China. E-mail: [email protected]
References [1]
Linge SO, Mardal KA, Helgeland A, Heiss JD and Haughton V. Effect of craniovertebral decompression on CSF dynamics in Chiari malformation type I studied with computational fluid dynamics: Laboratory investigation. J Neurosurg Spine 2014; 21: 559-564. [2] Chen IH, Kao KP, Penn IW and Ho DM. Double intraspinal enterogenous cysts. J Neurol Neurosurg Psychiatry 1995; 58: 110-111. [3] Agnoli AL, Laun A and Schonmayr R. Enterogenous intraspinal cysts. J Neurosurg 1984; 61: 834-840. [4] Holmes GL, Trader S and Ignatiadis P. Intraspinal enterogenous cysts. A case report and review of pediatric cases in the literature. Am J Dis Child 1978; 132: 906-908. [5] Lea ME, Sage MR, Bills D, Brophy B and Blumbergs P. Enterogenous cyst of the cervical spinal canal. Australas Radiol 1992; 36: 327329. [6] Miyagi K, Mukawa J, Mekaru S, Ishikawa Y, Kinjo T and Nakasone S. Enterogenous cyst in the cervical spinal canal. Case report. J Neurosurg 1988; 68: 292-296. [7] Shuangshoti S, Kasantikul V, Taecholarn C and Wangsuphachart S. Symptomatic intraspinal genuine endodermal epithelial cyst. J Med Assoc Thai 1989; 72: 295-301. [8] Marchionni M, Tzerakis N, Tsang K and Carey M. Upper cervical cord enterogenous cyst mimicking transient ischaemic attacks. BMJ Case Rep 2013; 2013. [9] Nakajima M, Miyajima M, Hishii M, Arai H, Sato K and Fujii H. Endodermal cyst of the quadrigeminal cistern: case report. Surg Neurol 2001; 56: 385-388; discussion 388-389. [10] Chaitanya G, Arivazhagan A, Sinha S, Reddy KR, Thennarasu K, Bharath RD, Rao MB, Chandramouli BA and Satishchandra P. Dexmedetomidine anesthesia enhances spike generation during intra-operative electrocorticography: A promising adjunct for epilepsy surgery. Epilepsy Res 2015; 109: 65-71. [11] Yao W, Qiu J, Zhou Z, Zhang L and Zhang C. Cervical spinal cord compression after thyroidectomy under general anesthesia. J Anesth 2014; 28: 125-127.
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