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Anterior sacral meningocele in patient 1. Preoperative sagittal T2-weighted MRI (A, B) shows a large, well-defined cystic mass occupying nearly entire pelvis, compression of the uterus (B, red arrow) and bladder (B, red asterisk). After the second operation, sagittal T2-weighted MRI (C) shows complete disappearance of the meningocele, and decompression of the bladder (C, red asterisk) and uterus (C, red arrow). The schematic diagram illustrates the position of meningocele (D), the first surgical method (E), and the second surgical method (F).
Source publication
Rationale:
Anterior spinal meningoceles are rare neuroanatomic abnormality formed by protrusion of the spinal meninges through a defect in the vertebral column. Presently, therapeutic options for anterior spinal meningoceles are still controversial. The objective of this study is to discuss the individualized management of giant anterior spinal me...
Contexts in source publication
Context 1
... examination, a large, firm, nontender mass can be palpated at her lower abdomen. Cranial nerve, somatic motor and sensory examinations showed to be normal. Unremarkable were the laboratory test results found to be. MRI revealed a large, non-enhanced, cystic abdominal mass measuring about 11.6 Â 18.3 Â 12.4 cm, spanning across S1 segment and S3 (Fig. 1A,B). The neck of the cyst seemed to be in continuity with the lumbar cistern, and a 7-cm defect was noted in the right posterolateral part of the S2 vertebral body. The patient received her first operation via a dorsal trans-sacral approach. After draining the cystic fluid, the neck of the meningocele opening was repaired with muscle and ...
Context 2
... of the cyst seemed to be in continuity with the lumbar cistern, and a 7-cm defect was noted in the right posterolateral part of the S2 vertebral body. The patient received her first operation via a dorsal trans-sacral approach. After draining the cystic fluid, the neck of the meningocele opening was repaired with muscle and absorbable sutures (Fig. 1E). Her symptoms relieved significantly after the operation. However, her repeated MRI revealed an abdominal mass again after 3 months. So she received another operation via the same surgical approach as the first time. We drained the cystic fluid and reinforced the neck of the meningocele opening with muscle and absorbable sutures. ...
Context 3
... the operation. However, her repeated MRI revealed an abdominal mass again after 3 months. So she received another operation via the same surgical approach as the first time. We drained the cystic fluid and reinforced the neck of the meningocele opening with muscle and absorbable sutures. After that, lumboperitoneal shunts (LPS) was performed (Fig. 1F). The patient acquired rapid postoperative recovery and discharge 5 days after the operation. Follow-up MRI was performed 1 year after surgery showing no reaccumulation of cyst fluid (Fig. 1C). And she gave birth to a baby 2 years after the ...
Context 4
... cystic fluid and reinforced the neck of the meningocele opening with muscle and absorbable sutures. After that, lumboperitoneal shunts (LPS) was performed (Fig. 1F). The patient acquired rapid postoperative recovery and discharge 5 days after the operation. Follow-up MRI was performed 1 year after surgery showing no reaccumulation of cyst fluid (Fig. 1C). And she gave birth to a baby 2 years after the ...
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Citations
... Los reportes de meningocele sacro anterior (MSA) han sido escasos en el mundo (1,2) (casi 300 casos) (3) , y ninguno se ha estudiado en el Perú. El MSA puede aparecer de manera espontánea, pero también como parte de un defecto hereditario de la columna sacrococcígea en el síndrome de Currarino (2,4) . ...
... Los reportes de meningocele sacro anterior (MSA) han sido escasos en el mundo (1,2) (casi 300 casos) (3) , y ninguno se ha estudiado en el Perú. El MSA puede aparecer de manera espontánea, pero también como parte de un defecto hereditario de la columna sacrococcígea en el síndrome de Currarino (2,4) . Este cuadro fue descrito por primera vez en 1981 por Currarino, quien definió su tríada clásica: defecto óseo sacro, malformación anorrectal (MAR) y masa presacra (MP) (MSA es la más común) (5) . ...
... El primer artículo que describe el MSA pertenece a Coller y Jackson (6) , y en la literatura existen solo unos 300 casos reportados (3) . El MSA se caracteriza por presentar una hernia de la duramadre a través de un defecto congénito en la pared anterior del sacro (2,7,8) , resultado probable de una ectasia dural a causa del agrandamiento del espacio subaracnoideo por la pulsación del LCR. Representa, aproximadamente, el 5 % de todas las masas retrorrectales y es más prevalente en las mujeres (9) . ...
Anterior sacral meningocele is a protrusion of the dural sac through a bone defect in the sacrum anterior wall. It can occur in isolation or be associated with other malformations, such as the Currarino syndrome, which is part of the classic triad together with hemisacrum and anorectal malformation, although it has great variability. This is the case of an adult woman with chronic constipation, recurrent urinary infection and marked abdominal distension, diagnosed with giant anterior sacral meningocele and incomplete Currarino syndrome. The patient underwent a sacral laminectomy and thecal sac ligation at the level of the bone defect. This article describes this rare type of occult spinal dysraphism as well as the surgical technique used.
Background
Neurofibromatosis type 1 is a genetic disease that affects multiple organs and systems, leading to various clinical manifestations. In Neurofibromatosis type 1, rare intrathoracic meningoceles often occur alongside bone dysplasia. These meningoceles contain cerebrospinal fluid and can be mistakenly diagnosed as ‘pleural effusion’.
Case presentation
In this case report, we mistakenly identified ‘cerebrospinal fluid’ as ‘pleural effusion’ and proceeded with drainage. This error posed significant risks to the patient and holds valuable implications for the future diagnosis and treatment of similar patients.
Conclusions
In patients with Neurofibromatosis type 1 complicated by spinal deformity, there is a high incidence of intrathoracic meningoceles. Treatment strategies may differ based on the specific features of the lesions, and collaboration among multiple disciplines can significantly improve patient outcomes.
The first documented description of an anterior sacral meningocele was Bryant’s in 1823. Anterior sacral meningocele patients have constipation as a universal symptom; urinary incontinence is also common. All the symptoms are directly related to the pressure from a pelvic mass on adjacent structures. When the patient stands, a headache often develops because the spinal fluid pressure decreases as the meningocele sac fills. Finally, a scimitar-shaped sacrum on a neuroradiological anteroposterior plain assessment is pathognomonic. The coccyx may be absent, and the lower sacral laminae may be absent or incomplete. The surgical options for this rare clinical condition are still matter of debate.
Anterior sacral meningocele is a pathology that lacks a current classification and neurosurgical therapeutic standards, even though a similar dynamic has been shown by the related traumatic pseudomeningocele. Anterior approaches (retro- and transperitoneal meningocele neck occlusion with internal cerebrospinal fluid (CSF) cyst drainage) and posterior approaches (posterior sacral laminectomy, dural sac ligation, and CSF cyst drainage) are the available surgical strategies.
We now report the case of an adult patient for whom a posterior approach was suggested and performed and report her postoperative surgical follow-up. The surgical rationale is also discussed.
Background:
Anterior sacral meningocele (ASM) is an uncommon variant of spinal dysraphism. Surgical correction for this condition is challenging and optimal corrective approaches are uncertain.
Objective:
To share our experience of managing this rare condition using the posterior trans-sacral approach and provide a contemporary review of the literature.
Methods:
Retrospective review of case notes, operative records, and imaging of eligible patients treated via the posterior trans-sacral approach between 2006 and 2020 at our regional neurosciences centre.
Results:
Three patients, two females and one male with a mean age of 30 years (range 16-38), were treated. Presenting symptoms included lower abdominal pain and recurrent miscarriages. Patients underwent corrective surgery using the posterior approach involving a sacral laminectomy, durotomy and closure of the communicating fistula. A single patient required reoperation due to early recurrence. Another patient proved challenging because of a very large sacral fistula and required two procedures due to the development of high-pressure headaches secondary to a recurrence. All patients improved symptomatically postoperatively and remained symptom free at the last clinic follow-up and have been discharged. Following review of the literature, only two other non-syndromic cases have been described.
Conclusions:
ASM is an uncommon congenital abnormality, typically presenting with mass effect symptoms secondary to a presacral cystic mass. Surgical management using a posterior approach to close the meningeal sac is feasible and less invasive than an anterior approach. Long term clinical outcomes in our series were satisfactory.
Background
69% of thoracic meningoceles are associated with Neurofibromatosis type 1(NF1). Pathology is debated between being a saccular protrusion of dysplastic meninges through a vertebral defect, or a forme fruste of neurofibroma, with no pathological evidence to the later. Treatment is usually conservative, however surgical intervention is sometimes indicated. Surgical options include thoracotomy, endoscopic plication of the cyst, or CSF diversion. After excision of the meningocele, watertight closure of the dura is difficult and meningoceles often reform, so dural reinforcement is indicated.
Objective
A literature review on thoracic meningocele in NF1 patients: pathological hypothesis, presentation including rupture and hemothorax in large meningoceles, management strategies, and surgical challenges. The review also includes an illustrative case discussing the surgical challenges with giant meningoceles.
Case presentation
A 46 year old male, NF1, with a known thoracic meningocele, presented with dyspnoea due to enlarging thoracic meningocele. The meningocele was successfully excised via thoracotomy in a challenging surgery with a novel surgical technique and unique insight on histology.
Results
10 cases of giant meningoceles in NF1 patients have been reported in literature, surgery was performed in 9 of them. 1 patient died, and there was recurrence or no change in 6 them. Histology in our illustrative case showed diffuse population of schwann cells. Also, Buttress plates achieved reinforced dural closure with no recurrence.
Conclusion
Our study concluded that in in large thoracic meningocele(s) surgery via thoracotomy is advised versus CSF diversion, and dural reinforcement with buttress plates proved to be successful. Our study also concluded that meningoceles in NF1 patients is likely a forme fruste of neurofibroma, or a giant cystic neurofibroma rather than due to dysplastic dura.
