Split notochord syndrome presenting with meningomyelocoele and dorsal enteric fistula

Gupta, Divya; Deodhar, Michael

doi:10.1016/s0022-3468(87)80251-8

Cited by 23 publications

(10 citation statements)

References 5 publications

Supporting

Mentioning

Contrasting

Unclassified

Order By: Relevance

“…7 Rarely, they can arise in the lumbosacral region and involve the hindgut. 2,8,9 In our case, the presacral cyst clearly communicated with the rectum; that communication was demonstrated with the gastrograffin enema. The failure to demonstrate communication between the presacral cyst and the epidural space could have been the result of insufficient pressure within the presacral cyst, because the contrast material was injected into the rectum and not into the cyst.…”

Section: Discussionsupporting

confidence: 61%

Presacral Neuroenteric Fistula in a Newborn Presenting With an Epidural Abscess: Case Report and Review of the Literature

Darwish

Stanley²,

Wickremesekera³

et al. 2004

Pediatrics

View full text Add to dashboard Cite

ABSTRACT. We describe a newborn infant (<24 hours of age) who presented with mild swelling on the back and buttocks attributable to a neuroenteric fistula complicated by an epidural parasacral abscess infected with mixed coliforms. Epidural abscesses in infancy are extremely rare, and one has not been observed previously in the newborn period. The infant was surprisingly mildly affected. Prompt intervention led to an excellent outcome. Coliforms may colonize the infant gut in <24 hours, even in the developed world. Unexplained swellings on the backs of infants should lead to a search for underlying malformations and an early surgical review, which is best conducted with a combined pediatric surgical and neurosurgical approach. N euroenteric cysts (fistulas) are rare, with Ͻ30 cases being described in the pediatric literature. Epidural abscesses presenting in infancy are even rarer, with the youngest patient reported being 20 days of age. 1 An epidural abscess complicating a congenital neuroenteric cyst appears not to have been reported previously. CASE REPORTA Ͻ1-day-old boy was admitted to the neonatal unit at a peripheral hospital with irritability, lethargy, and poor feeding. He had been delivered at 42 weeks of gestation through normal vaginal delivery and weighed 3250 g. His mother had an older healthy child and had remained in good health during this pregnancy. She admitted to smoking cigarettes and occasionally drinking alcoholic drinks. Results of ultrasound scanning performed in the first trimester had been reported as normal and consistent with dates. The infant's condition at birth was excellent, and he was allowed home, breastfeeding successfully, at 12 hours of age. In the examination, the infant was mildly jaundiced and lethargic. He had cool extremities, with a capillary return of 4 seconds, a heart rate of 150 beats per minute, a respiratory rate of 68 breaths per minute, and a skin temperature of 36.6°C. There was no sign of respiratory difficulty. The patient's anterior fontanelle was not distended, and his posture was normal. His abdomen was soft, without organomegaly. Movements were normal in all limbs, with normal tone and reflexes. The patient's anus was in a normal position, and he passed stools without difficulty. His back was normal, with no sign of sinuses or fistulas. Blood and urine cultures were obtained. Blood tests showed a C-reactive protein level of 25 g/mL (normal: Ͻ10 g/mL) and a white blood cell count of 10 300 cells per mm 3 (neutrophils: 8000 cells per mm 3 ). Results of a chest radiograph were normal. Lumbar punctures were performed as part of the septic screening and on 2 occasions revealed frank yellow-green pus, which showed large numbers of pus cells and mixed flora with Gram-staining and subsequently showed heavy growth of Escherichia coli and enterococci. A provisional diagnosis of mixed-organism meningitis was made, and the infant began broad-spectrum antibiotic treatment and was transferred to the neonatal intensive care unit of Wellington Hospital for additional in...

show abstract

Section: Discussionsupporting

confidence: 61%

Presacral Neuroenteric Fistula in a Newborn Presenting With an Epidural Abscess: Case Report and Review of the Literature

Darwish

Stanley²,

Wickremesekera³

et al. 2004

Pediatrics

View full text Add to dashboard Cite

show abstract

“…[21] Neurenteric cyst is classically reported as a solitary lesion in the cervical region, located anterior or anterolateral to the cord. [24,25] The bony abnormalities that may be associated with neurenteric cyst are likely to involve the anterior column. Wilkins and Odum initially reviewed 47 patients with reported neurenteric cysts.…”

Section: Discussionmentioning

confidence: 99%

Rare Cervical Spine Neuroenteric Idem Cyst With Intramedullary Extension

Gupta¹,

Wadhwa²,

Garg³

et al. 2015

jemds

View full text Add to dashboard Cite

56-year-old man presented to us with progressive weakness of upper and lower limbs. He had gradually progressive neurological deterioration in the form of quadriparesis. MRI was suggestive of cervical cord SOL. The clinical, radiological, and surgical findings are discussed together with a brief critical review of the literature. The patient had no other associated anomalies. The intradural extramedullary component of cyst was removed totally with debulking of intramedullary part with good neurological recovery. The clinical presentation was rather atypical for neurenteric cyst. Neurenteric cyst should be considered in the differential diagnosis of an intradural mass lesion regardless of the age, clinical presentation or location of the lesion. HOW TO CITE THIS ARTICLE:

show abstract

“…Defects in the lower cervical and upper thoracic region are said to be more common than those in the lumbosacral region [6]. A total of 21 cases with a lumbosacral defect mentioned earlier in the literature showed the presence of dorsal enteric fistula communicating with the GI tract and presenting with fecal discharge [2,5,6,7,8,9,10,11,12,13,14,15,16,17,18,19,20,21,22,23]. Thus, these cases usually presented in the neonatal period.…”

Section: Discussionmentioning

confidence: 99%

Dorsal Enteric Sinus with Spina Bifida: A Rare Form of Split Notochord Syndrome

Nanda¹,

Bawa²,

Kanojia³

et al. 2010

Pediatr Neurosurg

View full text Add to dashboard Cite

Split notochord syndrome is a rare congenital malformation that results in spinal anomalies associated with anomalies of the gastrointestinal tract and central nervous system. This report presents an infrequently described variant of this syndrome. A 9-month-old female presented with a dorsal midline mass since birth which was partially covered by skin, and part of the lesion gave a gross appearance of intestinal mucosa. MRI was suggestive of partial sacral agenesis with spinal dysraphism with tethered cord. Excision of the mass with repair of the spinal defect was done. Histopathology confirmed the presence of gastrointestinal mucosa.

show abstract

Split notochord syndrome presenting with meningomyelocoele and dorsal enteric fistula

Cited by 23 publications

References 5 publications

Presacral Neuroenteric Fistula in a Newborn Presenting With an Epidural Abscess: Case Report and Review of the Literature

Presacral Neuroenteric Fistula in a Newborn Presenting With an Epidural Abscess: Case Report and Review of the Literature

Rare Cervical Spine Neuroenteric Idem Cyst With Intramedullary Extension

Dorsal Enteric Sinus with Spina Bifida: A Rare Form of Split Notochord Syndrome

Contact Info

Product

Resources

About