Cases reported "Meningomyelocele"

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1/4. Unusual findings during abdominal placement of a ventriculoperitoneal shunt: report of three cases.

    The authors present three cases of infants born with myelodysplasia. Each infant underwent closure of a myelomeningocele and within 2 to 4 days placement of a ventriculoperitoneal (VP) shunt. In each case, on opening the peritoneal cavity, the authors observed egress of a dark or creamy dark fluid. None of the patients had a history of abdominal birth trauma. The decision was made to continue the procedures and send samples of the unusual fluids to the laboratory for culture and analysis. The cultures proved to be nondiagnostic and the characteristics of the fluid samples were most consistent with those of blood-tinged chyle. The authors hypothesize that, occasionally, the mechanical tautness that is created with repair of myelomeningoceles is sufficient to rupture small lymphatic vessels and accompanying blood vessels of the abdomen. An alternative hypothesis is that abdominal compression due to closure of the myelomeningocele may temporarily compress the liver, leading to raised intraportal pressures and resulting in weeping of chyle from the gastrointestinal tract. This abnormal fluid accumulation did not lead to chronic ascites, VP shunt infection, or dysfunction at long-term follow-up examination and abdominal visceral function has not been an issue.
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2/4. The OEIS complex: two case reports that illustrate the spectrum of abnormalities and a review of the literature.

    We present two cases of OEIS (omphalocele, exstrophy, imperforate anus, spinal defects) complex -MIM 258040 and a review of the literature. Case 1 was a 14-year-old girl who presented at 30 weeks' gestation. An ultrasound examination showed an omphalocele and spina bifida; the bladder was not visualised. She went into spontaneous labour two weeks later and the baby died shortly after birth. A full post-mortem examination was refused, but the mother did agree to an external examination, skin biopsy for fibroblast culture, X rays and MR imaging. The MR imaging showed a pelvic kidney, a large omphalocele containing the other kidney, liver, bowel and a fluid filled structure thought to represent an exstrophy of the bladder (EB). Case 2 was a 30-year-old woman who had an ultrasound examination at 20 weeks' gestation; this showed an omphalocele, but the bladder was not visualised. The pregnancy was subsequently terminated and a post-mortem examination showed a low set umbilical cord associated with a small omphalocele; there was an imperforate anus; a blind ending rectum terminated in the omphalocele. We conclude that these two cases illustrate the variability of the OEIS complex.
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3/4. Management of a child on CAPD with a ventriculoperitoneal shunt.

    Long term dialysis in children with multiple handicaps has become easier with the advent of CAPD. In December 1986, an 11 year old with myelomeningocele in end stage renal disease secondary to chronic pyelonephritis required dialysis. Placement of a right ventriculoperitoneal shunt had been done at 4 months of age. Poor family compliance required placement in medical foster care. The peritoneal catheter was replaced three times. Leakage and infection were the major complications resulting in catheter replacement. In March 1988 the child required shunt extension due to accelerated growth after which she developed relapsing peritonitis requiring removal of the peritoneal catheter and externalization of the ventriculoperitoneal shunt. A proximal tap of the ventriculoperitoneal shunt prior to revision had a negative culture but glucose of 471 when serum glucose was 91. Six days after externalization of the shunt and catheter removal, the shunt was converted to ventriculoatrial and the PD catheter replaced. Nasogastic feedings and cycler dialysis became necessary due to worsening nutritional status and hyperabsorption. Successful transplantation occurred 28 months after initial dialysis.
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4/4. haemophilus influenzae B meningitis in a patient with a ventriculoperitoneal shunt and meningomyelocele.

    A child with an implanted ventriculoperitoneal (VP) shunt and meningomyelocele had an infection of his cerebrospinal fluid (CSF). The organism cultured from the VP shunt apparatus and the meningomyelocele repair site was haemophilus influenzae B (HIB), a very unusual pathogen in this setting. The patient was treated with the appropriate antibiotics, administered intravenously in accordance with accepted practice and available literature, with no improvement. The site of the persistent infection was finally determined to be within the meningomyelocele repair tissue. The patient's clinical status improved dramatically following the removal and replacement of the total shunt apparatus. The three-part apparatus tip was cultured according to the standard laboratory practice, but did not yield any organism. We conclude that the management of HIB meningitis in the presence of a VP shunt should include prompt removal of the apparatus. Recovery of the organism may be enhanced by separating the apparatus components and culture of the connections, instead of merely the indwelling tip.
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