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PubMed1978

Myelomeningocele before birth.

The authors report a study of 92 human embryos and four fetuses with myeloschisis. The characteristics of embryonic myeloschisis compared with spina bifida cystica in infants are: 1) the lesion is often more diffuse, involving the whole spinal cord (12 embryos); 2) the cervical cord is frequently affected (23 of the remaining 80 embryos); 3) holoprosencephaly is frequently associated (18 embryos); 4) meningocele is not found; and 5) hydrocephalus and Arnold-Chiari malformation are not yet developed. Hydrocephalus and Arnold-Chiari malformation are found in myeloschistic fetuses. Almost all embryos with diffuse and cervical myeloschisis or with holoprosencephaly are extruded before birth by spontaneous abortion. Absence of meningocele in the embryonic period implies that its appearance is deferred to the fetal period. The development of hydrocephalus and Arnold-Chiari malformation also seems to be delayed until the fetal period. Our observation implies that myelomeningocele is induced by non-closure of the neural tube, not by rupture once it was closed. "Neural overgrowth" and disturbed "recanalization process" are discussed in relation to the pathogenesis of myelomeningocele.

Abnormalities, Multiple↗

PubMed1989

Magnetic resonance imaging in myelocystoceles. Report of two cases.

Two cases of terminal myelocystocele, a rare localized cystic dilatation of the caudal spinal central canal, are reviewed. Magnetic resonance imaging is a useful diagnostic tool for its evaluation. Terminal myelocystocele consists of the following: a myelocystocele which contains a "trumpet-like" flaring of the distal spinal cord central canal and thus is partially lined by ependymal tissue; a meningocele or dilated subarachnoid space located around the myelocystocele, which bulges into the subcutaneous region; and fibrolipomatous tissue surrounding the two cysts. This condition is usually associated with abnormalities of the vertebral column and sacrum as well as compression of the spinal cord and meningocele by a fibrous band. There is a possible relationship of the myelocystocele to teratogens such as loperamide HCl and retinoic acid, although the exact etiology of this entity is not known.

Cysts↗

PubMed2001

Complete familial Currarino triad. Report of three cases in one family.

The Currarino triad is a unique complex of congenital caudal anomalies including anorectal malformation, sacral bone abnormality, and presacral mass. In this report, the authors describe three cases with the complete Currarino triad in a family. The authors treated a 30-year-old mother with an anterior sacral meningocele, her 1-year-old son with a combination of anterior sacral meningocele and dermoid cyst, and her 4-year-old daughter with an epidermoid cyst. These three patients had associated sacral agenesis and anorectal malformations. To the authors' knowledge, this is the first report describing radiological and operative findings of complete familial Currarino triad in which a mother and her two children were affected.

Abnormalities, Multiple↗

PubMed2003

[Meningoencephalocele as rhinosurgical problem].

Cephalocele is a rare lesion mainly of congenital or traumatic origin. The lesion occurs as an extracranial hernia of dura mater that consists of cerebrospinal fluid only (meningocele) or cerebellar tissue (encephalocele). Some cephaloceles concern anterior cranial fossa and then are observed as hernias in nasal cavity or in paranasal sinuses. Three patients are presented. In 52 year woman, who was admitted due to idiopathic rhinorrhea (persisting 16 years) with periodical headache, the meningocele in right ethmoid sinus was diagnosed. The lesion was removed with the rhinosurgical approach and the defect in anterior cranial fossa was repaired with the free cartilaginous flap. 12 year boy was admitted due to the nasal tumor diagnosed by CT and MRI. The lesion was removed with the combined access: firstly the hernia sac was cut intracranially with the frontal craniotomy and then encephalocele was removed with lateral rhinotomy. Cranial fossa defect was repaired by the periosteum flap. In third case (8 year boy) encephalocele caused 5 incidences of purulent meningitis. CT and MRI showed the lesion in frontal sinus. Encephalocele was removed with rhinosurgical approach and the defect in posterior frontal wall was repaired with free cartilaginous and mucosal flaps. In all patients long term result of operations was good. Anatomic and pathologic conditions that influence on the choice of rhinosurgical or combined access to nasal and sinusal encephaloceles are presented and discussed.

Child↗

PubMed2003

[Anesthetic management for a radical operation in an infant with cloacal exstrophy].

We report the anesthetic management for a radical operation in an infant with cloacal exstrophy. Diagnosis of cloacal exstrophy with meningocele was made at the 26th week of gestation. Cesarian section was performed under general anesthesia in order to keep the infant in a state of apnea to prevent aerophagia, the so-called "sleeping baby", at the 38th week of gestation. As soon as the infant was born, she was intubated orotracheally and a radical operation (abdominal closure, removal of meningocele, and vesico-intestinal-pelvioplasty) was performed. Some complications occurred during the operation including hypotension, oliguria, hypothermia, hypoproteinemia, hyponatremia and hyperpotassemia. The operation time was 10 hours and 30 minutes. Mechanical ventilation was continued for 4 days in the NICU and the infant was discharged 37 days after the operation. Early prenatal diagnosis and simulation of the operation enabled an effective surgical procedure allowing us to minimize these intraoperative complications.

Abnormalities, Multiple↗

PubMed2004

[Currarino triad: a case report].

The authors report a case of Currarino triad with a combination of anterior sacral meningocele and mature teratoma, sacral body deformity, anorectal stenosis, and tethered cord. A newborn girl suffered from vomiting, abdominal distension and constipation. Initially, a diverting colostomy was performed at the age of one month. 7 months later, at the age of 8 months, we performed posterior sagittal anorectoplasty (PSARP): As a result, extirpation of teratoma, excision of meningocele, untethering of the spinal cord, and anorectoplasty were achieved simultaneously without complication. We suggest the use of an MRI to specify the presence of anosacral and spinal cord anomalies in patients with intractable constipation and we recommend combined pediatric and neurosurgical assessment and management for all cases of Currarino triad.

Abnormalities, Multiple↗

PubMed2005

Currarino syndrome: report of two cases and review of the literature.

In the present paper the authors examine two cases of Currarino syndrome and review the existing literature on the disease. Both cases presented with chronic constipation. The first concerns a two year old male born with anorectal stenosis and diagnosed with Currarino syndrome when scimitar sacrum and anterior meningocele were finally detected. The second concerns a 25 year old female who suffered from chronic constipation but was not diagnosed until thorough examination revealed ectopic anus with Hirschsprung disease, scimitar sacrum and anterior meningocele. Because these patients were not diagnosed with Currarino syndrome when first seen, the authors reviewed its prevalence, embryogenesis, clinical manifestations, diagnosis and treatment. The authors' research supports the significance of prompt diagnosis in effective treatment and reduction of morbidity.

Abnormalities, Multiple↗

PubMed1989

Observations on spinal dysraphism.

An attempt has been made to look into the age at presentation, incidence and clinical presentation of 40 patients with spinal dysraphism. Thirteen cases of myelomeningocele and 11 cases of meningocele constituted the 'aperta' group and there were 16 'occulta' cases. Though among the 'aperta' cases male is to female ratio was 1:1, a marked male preponderance of 2.2:1 was seen among 'occulta' cases. First born child was found to be most frequently (44.50%) affected in the present series. Myelography remained a mainstay in deciding operation in 'occulta' cases. All 'aperta' cases and 11 out of 16 'occulta' cases were treated by operation. Regarding results of treatment, all cases with meningocele were fully cured, cases with myelomeningocele showed no improvement but further deterioration of the neurological status could be arrested. Hydrocephalus was associated in 8 out of 13 cases with myelomeningocele and ventriculoperitoneal (VP) shunt were instituted in all of them. Prognostic significance of presence of hydrocephalus was profoundly grave as was evident from the mortality rate (10%) in this series.

Adolescent↗

PubMed1988

[Sacral perineural cyst--report of a case].

The presence of cysts within the sacral spinal canal, so-called sacral cysts, is described in literature. These include 'sacral perineural cyst', 'sacral extradural cyst', 'occult intrasacral meningocele' and 'anterior sacral meningocele'. Sacral perineural cyst in these cystic disorders was first described as an incidental autopsy finding by Tarlov in 1938. Since then, several reports have been made describing the sign and symptom, neurological findings, roentgenographic diagnosis and cause and origin of the sacral perineural cysts, although many problems are not yet solved satisfactorily. This cyst occurs on the extradural components of sacral or coccygeal nerve roots. Although most are asymptomatic, these occasionally cause low back pain, sciatic and sacrococcygeal pain, sensory and motor disturbance in the lower extremities, and urinary dysfunction, which symptoms are similar to those brought on by lumbar disc herniation. In 1948, Tarlov reported a case of sciatic pain due to a perineural cyst, the removal of which relieved the symptoms. Symptoms occur because adjacent nerve roots are impinged upon by the thin-walled, fluid-filled cysts, which are formed in a space between the endoneurium and the perineurium. Microscopically, the cyst walls consist of peripheral nerve fibers or ganglionic cells covered with meningeal epithelium. Communication of the cyst with subarachnoid cerebrospinal fluid may be poor, but myelogram and CT myelogram demonstrate the cysts filling with contrast media. With the advent of magnetic resonance imaging (MRI), imaging of the sacral perineural cysts has improved. Recently we had the opportunity to evaluate a patient in whom perineural cysts had caused considerable erosion of the sacrum.(ABSTRACT TRUNCATED AT 250 WORDS)

Female↗

PubMed1986

[Modern technics for studying injuries of the lumbo-ischiatic plexus. Apropos of a new case].

A 22 year old patient was admitted with multiple lumbosacral root meningoceles following a motorbike accident. Because of the marked neurologic deficit in left lower limb a radiculographic exploration was completed by a CT scan, with improvement in analysis of images, post-traumatic pseudo-meningoceles being neither an absolute proof nor the unique lesion responsible for the clinical picture. They may be missing whereas the root is absent. Combining these two examinations provides for total screening of lesions and more selective choice of therapy.

Accidents, Traffic↗

PubMed1979

[Meckel's syndrome: an unusual pedigree].

The authors report the case, in the same family, of two children with the Meckel's syndrome, of two mis-arriages (one foetus with anencephalia, the other one with occipital meningocele), and of two still-born children (one with anencephalia, the other one with occipital meningocele). The genetic problems raised by this unusual genealogy are discussed.

Anencephaly↗

PubMed1979

The prognosis of occipital encephalocele.

147 cases of occipital cranium bifidum have been followed: 32 had meningoceles and 115 encephaloceles. Females predominated (69%). 90% of the meningocele and 43% of the encephalocele patients survived. Of the former 48% are normal and 16% are both physically and mentally handicapped. Of the latter only 4% are normal and 26% are physically and mentally handicapped. Cerebral tissue in the sac and associated microcephaly are bad prognostic features.

Encephalocele↗

PubMed1984

Sacrococcygeal developmental abnormalities and tumors in children.

Lesions from the SC region of children examined histologically at the RAHC were: 1. Malformations almost always associated with spina bifida aperta or occulta: 183 myelomeningocele (MM), 32 meningocele (M), 35 lipoMM and lipoma, 19 dermoid cyst, six occult meningocele, two Pacinian hamartoma, one short filum, four hindgut cysts or sinuses, two tailgut cysts, and two epithelial heterotopia. 2. Neoplasms, usually without spina bifida: 56 teratomas (11 malignant), five ependymomas (two purely subcutaneous), and 14 miscellaneous primary malignancies, (most neuroblastoma and rhabdomyosarcoma). Distinction between MM with glial tissue and M without glial tissue is important as M had a much better prognosis, less than a third developing hydrocephalus, and 77% walking unaided. Of those with glial tissue, the eight without Arnold-Chiari malformation were myelocystocele associated with cloacal exstrophy (six), caudal regression syndrome (one), and microcephaly (one). Postsacral glial tissue without paraplegia may occur with a subcutaneous vestige of filum terminale, or with herniation of the nonfunctioning half of a diplomyelia. Of postsacral "lipomas" and dermoids, 70% had an intraspinal connection through an occult spina bifida. This posterior vertebral defect is easily overlooked as the arches normally may not ossify until after 6 years. Therefore, the pathologist receiving a postsacral specimen may wish to alert the clinician to the high incidence of late effects from an occult intraspinal component or tethering of the spinal cord. Transsacral hindgut herniations and cysts probably result from ectoendodermal adhesions. Presacral multicystic malformations with mixed squamous and mucus cell lining are probably tailgut remnants or anorectal duplications, and may be mistaken for dermoid or teratoma. In SC teratoma in infants, contrary to some reports on ovarian teratoma in adults, immature tissues do not indicate a worse prognosis. Malignancy is virtually confined to teratomas including a carcinomatous or "yolk sac" component. It is more common in predominantly presacral examples and rare before the age of 4 months. SC ependymoma differs from ependymoma elsewhere in that it may be primary outside the craniospinal cavity (presacral or postsacral), may have a myxopapillary pattern special to the region, and although low-grade and slow growing, is more likely to metastasize beyond the central nervous system. Postsacral examples arise from vestiges of the filum terminale which are normal in the subcutis there. Combinations of all these lesions occur with vertebral defects and with each other.(ABSTRACT TRUNCATED AT 400 WORDS)

Adolescent↗

PubMedDate not supplied

Sonographic evaluation of caudal spine anomalies in children.

Retrospective analysis of B-mode spinal sonography in 15 pediatric patients with spinal dysraphism and caudal anomalies documents a high degree of accuracy in identifying the presence, site, size, and configuration of meningoceles; and slightly less accuracy in identifying extension of neural tissue into the meningocele and presence of concurrent lipoma. B-mode sonography is capable of displaying spinal anatomy clearly enough to permit accurate retrospective interpretation of pathologic changes. When radiologists learn to interpret sonographic images well enough to achieve equal accuracy in prospective diagnosis, then B-mode spinal sonography will supplant computed tomography and myelography as the preferred screening procedure in the initial evaluation of children with selected anomalies of the caudal spine.

Child↗

PubMedDate not supplied

[Amipaque myeloradiculography in the diagnosis of brachial plexus root rupture].

Myelography with a water-soluble contrast medium amipak (metrisamide) was applied in a complex with other diagnostic methods of examination in 22 patients with injury to the roots and primary trunks of the brachial plexus. Five of these patients had a clinical picture of Erb-Duchenne paralysis, in 17 injury to all the primary trunks with predominance of Djerine-Klumpke paralysis in the clinical picture was found. The results obtained provide evidence that myelography with amipak is a reliable method of the diagnosis of preganglionic (intraforaminal) injury to the roots of the brachial plexus. The most informative signs of such injury are. (a) traumatic meningocele; (b) deformity of the dura mater infundibula; (c) filling defects and constriction of the cerebrospinal fluid spaces. The presence of solitary meningocele is not a contraindication for operative exploration of the brachial plexus because of clinical picture of total paralysis may be caused by injury to the primary trunks in the more distal parts of the plexus.

Brachial Plexus↗

PubMed1996

Uncommon craniospinal dysraphism.

The uncommon variants amongst 619 cases of craniospinal dysraphism have been studied between 1987 to 1993. Lateral cervical meningocele is an extremely rare condition as to date only ten cases are traceable in the world literature. Its pathogenesis is discussed in detail. Similarly, this case of lateral frontal encephalocele is the second such example. Orbital meningocele may be congenital or following trauma to the orbital roof. Besides other unusual locations like the vertex, posterior fontanelle and anterior sacral are also encountered. Thus this paper focuses on the aetiology, clinical presentation and management of uncommon types of craniospinal meningo-encephaloceles.

Child↗

PubMed2006

Prenatal diagnosis of episodic tachypnea in an infant with OFD VI.

Episodic tachypnea is a typical neonatal presentation of Joubert syndrome, but may also occur in infants with other anomalies of the cerebellar vermis. Even though fetuses at risk for Joubert syndrome are usually closely followed by ultrasound, this respiratory pattern has only once been described prenatally. We describe a patient who presented prenatally with posterior meningocele, Dandy-Walker cyst, and four limb polydactyly on ultrasound. Amniocentesis showed a normal male karyotype and normal 7DHC. At 31 weeks, episodic fetal tachypnea was noted on ultrasound. The working diagnosis was Joubert syndrome or oro-facio-digital syndrome type VI (OFD VI). At birth, in addition to the findings noted prenatally, he had multiple nodules of his tongue, a Y-shaped metacarpal and micropenis. His respiratory pattern was notable for alternating tachypnea and apnea with respiratory rates up to 200 followed by apnea and bradycardia. Magnetic resonance image showed Dandy-Walker with massive 4th ventricle, complete absence of the cerebellar vermis, hypoplastic brainstem, and small meningocele. Development is profoundly delayed and he remains ventilator dependent. Both the previously described Joubert patient with prenatally recognized tachypnea, and our patient are atypical for Joubert syndrome since they have polydactyly (which occurs in only 8% of Joubert patients) and hamartomas of the tongue (which occur in 2%). Despite the tongue hamartomas, these patients are not entirely typical for OFD VI, since their polydactyly is postaxial. The observation of prenatal tachypnea in these two patients, but not in typical Joubert patients, suggests they have either a variant of OFD VI or a new Joubert or OFD-like syndrome.

Adult↗

PubMed1989

Central nervous system malformations induced by triamcinolone acetonide in nonhuman primates: pathology.

Triamcinolone acetonide (TAC) was administered to pregnant macaques (Macaca mulatta [15] and M. radiata [7]) during gestational days (GD) 23 to 41 using various dosing schedules. A daily dose of 10 mg/kg is approximately equal to 100 x the human dose equivalent. The brains of the fetuses and infants were studied grossly and histologically. All cases displayed either the mild form of the TAC-induced syndrome (craniofacial dysmorphia, cranium bifidum occultum, meningocele, and mild distortion of the midbrain) or the more severe form (occipital encephalocele, hydrocephalus, severe distortion of the midbrain or midbrain "beaking," shunting of cerebrospinal fluid, and craniofacial malformations). The dysmorphology was dose-related, with severity increasing at higher doses or with increased numbers of treatments. Individual cases were assessed for the severity of the syndrome by comparison of like components between groups. The lesions observed were morphologically comparable to those described in spontaneous human cases; the TAC-induced occipital encephaloceles were associated with brainstem and cerebellar abnormalities, and, with the less severe form of the syndrome, brainstem abnormalities were occasionally present, with occipital meningoceles. Controversy exists concerning the significance and temporal development of the midbrain changes. However, the associated alteration in aqueduct conformation may have been responsible for functional compromise and ensuing hydrocephalus.

Abnormalities, Drug-Induced↗