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PubMed2006

Spectrum of nonterminal myelocystoceles.

OBJECTIVE: To present the magnetic resonance imaging features, clinical findings, and possible embryologic bases for nonterminal myelocystoceles, a distinct subset of closed spinal dysraphisms. METHODS: We retrospectively analyzed imaging series and clinical records from five newborns and one older child with skin-covered soft tissue masses along the posterior midline spine. Spinal (6 patients) and brain (5 patients) magnetic resonance imaging was performed before surgical repair and compared with clinical findings, observations at surgery, and final lesion histology. RESULTS: The lesions affected the cervical (n = 3), thoracic (n = 2), and lumbar (n = 1) regions. In each case, the dome of the mass was covered by thickened, dystrophic epithelium with no subcutaneous fat, whereas the base and lateral walls of the mass were covered by normal skin. All patients were neurologically intact at presentation. In three cases, a stalk emanated from the dorsal normal spinal cord, crossed a narrow posterior bony spina bifida, and coursed through a posterior meningocele to attach to the inner aspect of its dome. The other three cases showed dissection of a hydromyelic cavity into the stalk, converting it into a second "cyst" within the meningocele. Concurrent anomalies included focal hydromyelia immediately cranial to the origin of the posterior stalk (n = 2), mild Chiari II malformation (n = 3), triventricular hydrocephalus from aqueductal stenosis (n = 1), filar lipoma (n = 1), and presumed neurenteric cyst (n = 1). At surgery, the sac was resected in all cases, but intradural exploration and untethering was performed in only three children. Embryologic considerations indicate that the spectrum of these lesions likely arises from partial limited closure of the neural tube, failed disjunction of the cutaneous ectoderm, and variable degrees of hydromyelia. CONCLUSION: The nonterminal myelocystocele is a distinct form of closed spinal dysraphism characterized by a skin-covered meningocele, which is either crossed by a fibroneurovascular stalk that extends from the dorsal aspect of the spinal cord to attach to the dome of the meningocele (abortive form, or myelocystocele manqué) or contains a hydromyelic cavity that is continuous with the ependymal canal of the spinal cord (complete form).

Abnormalities, Multiple↗
PubMed1994

Occult dysraphism in adulthood: clinical course and management.

We present a series of 23 patients with dysraphic malformations and adult onset of symptoms (4 meningoceles, 19 spinal hamartomas). Mean age at presentation was 39 +/- 21 years (range 23-67 years). Patients were followed up for a mean period of 19 months (range 0.5-68 months). Only patients with progressive neurological disease were operated on (3 meningoceles and 16 spinal hamartomas). The remaining patients were treated conservatively and continue to be observed clinically. Two of three patients operated for meningoceles improved without recurrence of symptoms. Patients with spinal hamartomas could be divided into two groups according to their main symptom: paraparesis (group A, n = 8) or pain (group B, n = 11). Malformations in group B were typically associated with a tethered cord and tended to be more complex than in group A. The majority of patients in group A showed better long-term results than patients in group B, due to their considerably lower rate of recurrence.

Adult↗
PubMed1989

MR evaluation of brachial plexus injuries.

Ten cases of brachial plexus injury were subjected to magnetic resonance (MR) to demonstrate the roots, trunks, divisions or cord abnormalities. Both normal and abnormal brachial plexuses were imaged in sagittal, axial, coronal and axial oblique planes. Myelography, using water soluble contrast agents, was performed in seven cases. MR demonstrated one traumatic meningocele, one extradural cerebrospinal fluid (CSF) collection, trunk and/or root neuromas in four, focal root fibrosis in two and diffuse fibrosis in the remaining two cases. Results of MR were confirmed at surgery in four cases with neuromas, while myelography was normal in two and was not carried out in the remaining two. In two cases, where MR demonstrated diffuse fibrosis of the brachial plexus, myelography showed C7 and T1 traumatic meningocele in one and was normal in the other. Both these patients showed excellent clinical and electrophysiological correlation with MR findings and in one of them surgical confirmation was also obtained. In the other two cases with focal nerve root fibrosis, myelography was normal in one and showed a traumatic meningocele in another. Operative findings in these cases confirmed focal root fibrosis but no root avulsion was observed although seen on one myelogram. Focal fibrosis, however, was noted at operation in more roots than was observed with MR. Initial experience suggests that MR may be the diagnostic procedure of choice for complete evaluation of brachial plexus injuries.

Adolescent↗
PubMed2003

Currarino triad with dual pathology in the presacral mass: report of a case.

PURPOSE: Currarino triad, which comprises anorectal stenosis, anterior sacral defect, and a presacral mass, is an uncommon cause of constipation in children and adults. The presacral mass in this triad is most often caused by an anterior sacral meningocele, a teratoma, or an enterogenous cyst, but rarely may be caused by dual pathology. A neonate with Currarino triad and dual pathology in the presacral mass is described in this report. METHOD: A male Chinese neonate, who presented with abdominal distention and constipation on the second day of life, was found to have features of Currarino triad. Colostomy was done in the neonatal period, and the presacral mass was excised by posterior sagittal perineal approach at the age of six months. RESULTS: The excised presacral mass consisted of an anterior meningocele and a teratoma. The patient continued to have constipation during follow-up and required anorectoplasty to correct residual anorectal stenosis. At the time of this report the patient was three years old and growing normally with normal anorectal function. DISCUSSION: Of a total of about 200 cases of complete Currarino triad found in the literature, in only 22 patients did the presacral mass contain both meningocele and teratoma. The features of these 22 patients and the current views on the surgical management of Currarino triad are discussed.

Abnormalities, Multiple↗
PubMed1991

Nerve root avulsion of birth palsy: comparison of myelography with CT myelography and somatosensory evoked potential.

Findings at myelography and computed tomographic (CT) myelography were reviewed in 21 patients (22 limbs) with birth palsy; nerve root shadows were the focus of this study. Myelography demonstrated 51 completely avulsed roots (78%) and 14 incompletely avulsed roots (22%). A traumatic meningocele was detected at 38 roots on myelograms and 51 roots on CT myelograms. Thirteen avulsed roots (eight completely and five incompletely avulsed roots) (20%) were not associated with a meningocele. In nine patients who underwent brachial plexus exploration, myelographic findings were compared with root somatosensory evoked potential (SEP). SEP was not induced at 22 of 25 completely avulsed roots and was induced at all seven incompletely avulsed roots. Myelography and SEP were consistent in 29 of 32 roots (91%). It is concluded that myelography is indispensable for preoperative evaluation of cervical nerve root avulsion of birth palsy, because CT myelography is not sensitive to nerve root avulsion without a traumatic meningocele, and SEP cannot enable one to discriminate incomplete avulsion from intact roots.

Brachial Plexus↗
PubMed1979

Carcinoma associated with lipomeningocele: case report.

We report the case of a 41-year-old man with an epidermoid carcinoma that had developed on a meningocele with a fistulous tract that chronically drained cerebrospinal fluid. After a review of the literature, we discuss the cause of this rare occurrence. We think that the recurrent discharge of spinal fluid (never complicated by meningitis) may have caused a chronic irritation of the tissues and then the delayed development of a carcinoma, which is similar to a case found in the literature. In our case there was a "flow" of carcinomatous cells along the fistula, which entered the meningocele, invading the subdural space as far as the peduncle. Our patient, who had a benign lesion (meningocele), came under neurosurgical care only after the lesion had degenerated. The malignant degeneration of such a benign lesion emphasizes the need for surgical treatment as soon as possible.

Adult↗
PubMed1991

The nature of congenital posterior cervical or cervicothoracic midline cutaneous mass lesions. Report of eight cases.

Between 4% and 8% of cases of spina bifida cystica occur in a cervical or cervicothoracic location. Despite a large body of literature concerning spinal dysraphism, there has been little written specifically about patients afflicted with this disorder in a cervical location. Eight children who presented at birth with posterior cervical or cervicothoracic lumps, all of which represented a dysraphic state, are discussed. Two types of abnormalities were noted. Three patients had hydromyelia with an associated myelocystocele herniating posteriorly into a meningocele sac. In these three patients there was an associated Chiari II malformation and hydrocephalus. The other five children had a meningocele in which a band of tissue extended from the posterior aspect of the spinal cord through a defect in the bone and fascia to the posterior part of the meningocele sac itself. No patient had a lesion that could be described as a meningomyelocele. The investigation and surgical management of these conditions are discussed and the need for intradural exploration to untether the spinal cord in the cervical region is stressed.

Female↗
PubMed1990

[A case of transsphenoidal meningoencephalocele].

A 6-year-old boy was sent to us due to basal meningoencephalocele detected by MRI. He had had a past history of strabismus and morning glory syndrome since 6 months of age, as well as hypopituitary dwarfism since he was 3 years old. On admission, physical examination revealed hypertelorism and left coloboma. Laryngoscopic examination revealed a round mass covered by normal nasal mucose in the midline of the tegmen of the choana. X-ray tomogram and CT scan demonstrated an isodense mass protruding into the nasopharyngeal cavity through a bony defect of the sella turcica. Right carotid angiogram showed stenosis of the right internal carotid artery and abnormal fine vessels in the basal ganglia similar to basal Moyamoya network. There was an irregular filling of the right anterior cerebral artery. Left carotid angiogram showed an irregular filling of the left middle cerebral artery. MRI showed an anterior part of the third ventricle descending into, and the meningocele protruding into the nasopharyngeal cavity. It also showed a stalk extending from the hypothalamus into the meningocele, but the pituitary gland was not recognized. The meningocele was of the same signal intensity as CSF in both T1 and T2 weighted images. Growth hormone deficiency was confirmed by radioimmunoassay of the peripheral blood. An operation was scheduled transcranial to prevent snoring during sleep at age 9. The stalk was observed behind the chiasm during the operation, but a radical operation was not performed because of a possibility of postoperative hypothalamic-pituitary dysfunction. Transsphenoidal encephalocele is rare. So far as we could see, only 30 cases have been reported.(ABSTRACT TRUNCATED AT 250 WORDS)

Child↗
PubMed1995

Tethered cord with anorectal malformation, sacral anomalies and presacral masses: an under-recognized association.

The association of congenital anal stenosis, scimitar-shaped sacral defect and a presacral mass is known as the Currarino triad. These patients present with constipation of variable severity which may result from a combination of compression by the mass, neurogenic dysfunction as well as anal stenosis itself. We have treated three patients who illustrate the spectrum of severity of this condition. All had an associated tethered cord. One was cured by a single anal dilatation, cord release and anterior meningocele repair. The second did not improve after anoplasty and neurosurgical intervention. A previously diagnosed rectal ectasia was felt to be contributory to the constipation and was treated by low anterior resection with excellent results. The third patient developed an ischiorectal abscess that required drainage and colostomy after unsuccessful dilatation of a very tight and deeply scarred anal stenosis. A subsequent tethered cord release and closure of the anterior meningocele was followed by an anoplasty. Colostomy was closed at a later date. In all patients with anal stenosis a search should be made for a presacral mass and its associated sacral defect. Release of the tethered cord, resection of the presacral mass and/or closure of the anterior meningocele are essential for optimal functional results.

Abnormalities, Multiple↗
PubMed1996

School-aged children with spina bifida in Western Australia--parental perspectives on functional outcome.

A questionnaire mailed to the parent(s) of all 86 school-aged children with spina bifida resident in Western Australia in 1992 who were attending a spinal dysfunction clinic or were members of the Spina Bifida Association was returned by 72 parents. All 72 of these children (55 with myelomeningocele, 17 with meningocele) lived at home with their parent(s). All but two children with meningocele were mobile without aids, whereas only 42% of the children with myelomeningocele were. Twenty one per cent of the children were naturally continent of urine day and night, and 36% were naturally continent of faeces. 76% attended a mainstream school although performance at school was rated below average for 40%. Nine children with meningocele (56%) and 42 with myelomeningocele (76%) were reported to have learning difficulties. This information will be useful in counselling parents of unborn and newborn children with spina bifida, and in allocating resources for children and young adults with spina bifida and their families.

Adolescent↗
PubMed2003

Recurrent meningitis associated with complete Currarino triad in an adult--case report.

A 58-year-old woman presented with Currarino triad manifesting as recurrent meningitis. Currarino triad is a combination of a presacral mass, a congenital sacral bony abnormality, and an anorectal malformation, which is caused by dorsal-ventral patterning defects during embryonic development. She had a history of treatment for anal stenosis in her childhood. Radiographic examinations demonstrated the characteristic findings of Currarino triad and a complicated mass lesion. The diagnosis was recurrent meningitis related to the anterior sacral meningocele. Neck ligation of the meningocele was performed via a posterior transsacral approach after treatment with antibiotics. At surgery, an epidermoid cyst was observed inside the meningocele. The cyst content was aspirated. She suffered no further episodes of meningitis. The meningitis was probably part of the clinical course of Currarino triad. Radiography of the sacrum and magnetic resonance imaging are recommended for patients with meningitis of unknown origin. The early diagnosis and treatment of this condition are important.

Abnormalities, Multiple↗
PubMed1985

Terminal myelocystocele.

Terminal myelocystoceles constitute approximately 5% of skin-covered lumbosacral masses and are especially common in patients with cloacal exstrophy. Pathologically, terminal myelocystocele consists of (a) a skin-covered lumbosacral spina bifida (b) an arachnoid-lined meningocele that is directly continuous with the spinal subarachnoid space; and (c) a low-lying, hydromyelic spinal cord that traverses the meningocele and then expands into a large terminal cyst. The terminal cyst bulges into the extraarachnoid compartment caudal to the meningocele and forms a distal sac that does not communicate with the subarachnoid space. The terminal cyst is lined by ependyma and dysplastic glia, is directly continuous with the dilated central canal of the cord, and probably represents a ballooned terminal ventricle. Patients with terminal myelocystocele have normal intellectual potential and are usually born without neurological deficit, so these defects must be identified and repaired early, before the onset or progression of lower extremity pareses.

Female↗
PubMed1983

[Traumatic elongation-traction lesions of the roots of the sciatic nerve, secondary to fractures of the pelvis].

Three cases of complex pelvic fractures associated with lumbo sacral roots lesions are reported. The authors stress this possible association should be suspected through clinical and conventional radiological findings. The contrast sacco-radiculography brings up confirmation. However the so called pseudo-meningocele is by far of less topographical value than the same myelographic findings at cervical level (for brachial plexus avulsion injuries). Direct and concise evaluation is performed by surgical approach of roots during a homolateral hemi-laminectomy. This procedure allows to appreciate the anatomical lesions and to clarify the anatomo-radiological discrepancies. Moreover endodural closure and reduction of volume of meningoceles and removal of arachnoidal adhesions is to pain relief value and may prevent delayed complications of neighboring roots compression related to increase in volume of meningocele. According to the literature and our findings, in future, the aim should be microsurgical repair of such roots lesions as they ar not avulsed from the conus but merely more distally ruptured on the cauda equina. We could not so far achieve this procedure, because of difficulties to identify the distal part of root, often retracted far away from its spinae foramen.

Adolescent↗
PubMed1999

Transnasal endoscopic repair of congenital defects of the skull base in children.

OBJECTIVE: To examine imaging findings and methods of endoscopic treatment of congenital skull base defects in children. DESIGN: Retrospective study and case series. SETTING: Academic tertiary care center. PATIENTS: Four patients (aged 12 and 14 months and 8 and 13 years) were included from 1995 to 1997. Three presented with a nasal glioma, which was recurrent in 1 case. The fourth patient presented with bacterial meningitis due to a spontaneous cerebrospinal fluid leak. Computed tomography and magnetic resonance imaging were used to locate the defect of the skull base. INTERVENTION: Transnasal endoscopic resection of the glioma or the meningocele, with immediate repair of the skull base defects using free mucosal flaps and/or pediculized mucosal flaps and/or conchal cartilage together with fibrin glue and nasal packing during a 3-week period. RESULTS: None of the 4 patients has experienced recurrent cerebrospinal fluid leaks or postoperative meningitis. CONCLUSIONS: The transnasal endoscopic repair of congenital meningoceles is a reliable technique in select pediatric patients. Computed tomography and magnetic resonance imaging provide information that can be used to help the surgical procedure.

Adolescent↗
PubMed1975

Pathogenesis of diastematomyelia and spina bifida.

Three cases of spina bifida (SB) associated with diastematomyelia (DM) are described and possible differences in pathogenesis are discussed. The frequent simultaneous occurrence of both malformations and the possibility that the cord can be split above, at the level of or below the SB indicate a common pathogenesis for SB and DM. Splitting of the cord symmetrically, asymmetrically, bilateraly or even antero-posteriorly can be explained only by the existence of a destructive phenomenon which is responsible for the bifurcation of the neural tube or its anlage. The existence of such a phenomenon indicates that myelocele can develop due to re-opening of a previously closed neural tube. Meningocele may be a manifestation of early embryonic antero-posterior DM in which the anterior tubule develops into an almost normal spinal cord, whereas the posterior tubule provides the wall of the cystic sac of meningocele in which most or all the neural tissue undergoes fibrous replacement.

Humans↗
PubMed1994

Meningeal diverticula treated with ventriculoperitoneal shunt.

An infant with Chiari malformation developed respiratory distress due to progressive intrathoracic enlargement of multiple spinal meningeal diverticulae. Ventriculoperitoneal shunting of the associated hydrocephalus temporarily halted the expansion of the intrathoracic diverticulae and permanently relieved respiratory symptoms. The role of hydrostatic pressure in the enlargement of meningoceles and other spinal meningeal diverticulae is discussed. Ventriculoperitoneal shunting is suggested as a surgical alternative for the treatment of symptomatic meningoceles.

Arnold-Chiari Malformation↗
PubMed1989

Post-traumatic intradiploic pseudomeningocele in children.

Eight children with post-traumatic pseudomeningocele are reported. In this paper the mean age at the time of injury was one year and ten months. All of them gave a history of a fall from a height. In six patients the meningocele was located in the parieto-occipital region. The frontal bone was involved in one and the roof of the orbit in another. All these patients had localized progressive swelling of the skull associated with a bony defect. Four patients had a history of convulsion and three had hemiparesis. The meningocele was excised, the dural defect repaired and a cranioplasty was performed in all. Good recovery was observed in all of them.

Adolescent↗
PubMed2000

Diagnostic steps and staged operative approach in Currarino's triad: a case report and review of the literature.

The Currarino triad is a combination of a presacral mass, a congenital sacral bony abnormality and an anorectal malformation. It mostly presents with constipation. Rectal examination, plain radiographs and magnetic resonance imaging are the main tools for the diagnosis. If the mass is a meningocele, colostomy and neurosurgical exploration should precede anoplasty due to the risk of meningitis. A 14-month-old female patient with anal stenosis, a sacral scimitar defect and an anterior meningocele is presented in this report.

Constriction, Pathologic↗