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PubMed1998

Gastrocystoplasty: long-term followup.

PURPOSE: Gastrocystoplasty is no longer favored at many institutions due to complications, including the hematuria-dysuria syndrome and metabolic alkalosis. We reviewed our experience to determine the advantages and disadvantages of bladder augmentation using stomach body wall. MATERIALS AND METHODS: We retrospectively reviewed the medical records, urodynamic studies, x-rays and laboratory evaluations of 47 children who underwent gastrocystoplasty at our institution between 1986 and June 1997. Parents and patients were contacted by telephone for detailed interviews to validate the medical record and determine whether there had been any changes since the last visit. Followup ranged from 9 months to 11 years (mean 4.4 years). Bladder dysfunction was secondary to spinal dysraphism in 38 children. Other diagnoses included cloacal and bladder exstrophy, posterior urethral valves, a persistent urogenital sinus and bilateral ectopic ureters. RESULTS: Preoperative and postoperative ultrasound studies available for 79 renal units demonstrated a stable or improved upper tract in 75 (95%). Preoperative and postoperative serum sodium, potassium and creatinine levels showed no significant changes. Mean serum chloride decreased 2.7 mEq./l. and bicarbonate increased 3.3 mEq./l. In the 3 patients with renal insufficiency serum bicarbonate increased 8 mEq./l. Mean pressure specific bladder volume at less than 20 cm. water increased 177 cc and mean maximum bladder pressure decreased from 35 to 13 cm. water. Two-thirds of the patients had bacilluria and a fourth reported a symptomatic bladder infection. No patients had bothersome mucus or required routine bladder irrigation. Symptoms consistent with the hematuria-dysuria syndrome were present in 27% of the patients. No patients had symptoms more than once weekly. Symptoms occurred in 75% of the patients without a neurogenic bladder, such as exstrophy or posterior urethral valves, and in 14% of those with neurogenic bladder dysfunction. In 1 case a bladder stone developed 8 years after surgery. Five patients required reoperation for complications related to gastrocystoplasty. No patient had perforation. CONCLUSIONS: Our data show that even after a mean of 4.4 years gastrocystoplasty has significant advantages over intestinal augmentation, including decreased chloride reabsorption, mucous production and urinary infection, and an extremely low incidence of stones and perforation. The gastric patch is associated with metabolic alkalosis and the hematuria-dysuria syndrome, which may be avoided and medically treated with proper patient selection and close followup. The procedure should be avoided in sensate patients with sufficient bowel.

Child↗
PubMed1998

Fibronectin expression in the developing human spinal cord, nerves, and ganglia.

AIM: Analysis of developmental role of fibronectin during differentiation of the human spinal cord, nerves, and ganglia. METHODS: Seven normal human embryos and fetuses between the 7th and 9th developmental week and a 9-week fetus with cervical spina bifida were histologically examined on hematoxylin and eosin stained serial paraffin sections of thoracic axial segments. Monoclonal antibody to the human cell fibronectin fragment was used for immunohistochemical detection of fibronectin. RESULTS: In the 7th and 8th week of development, fibronectin was weakly expressed in the ventricular and intermediate zones of the spinal cord. Intense fibrillar expression was found in the marginal zone of the spinal cord - first over the ventral gray horns and later over the lateral and dorsal gray horns, and along the pathways of ventral and dorsal roots of the spinal nerves and in the spinal ganglia. At 9th week, fibronectin expression disappeared in the ventricular and intermediate zones a nd became weak and granular in the marginal zone of the spinal cord. In the spinal cord of a 9-week malformed fetus with cervical spina bifida, fibronectin expression was completely absent. Fibronectin was expressed in the nerves and ganglia throughout the investigated period, both in normal and malformed human conceptuses. CONCLUSION: Transient expression of fibronectin in the human spinal cord coincided with the most intense neuronal differentiation. Temporal and spatial expression of fibronectin during normal development, and its absence in a malformed human fetus suggests developmental role of fibronectin for the normal formation of the spinal cord.

Biomarkers↗
PubMed1999

Genitourinary anomalies in the CHARGE association.

PURPOSE: We identified the incidence and types of genital and urinary anomalies, and established a plan for evaluating the urinary system in the CHARGE association. MATERIALS AND METHODS: We retrospectively reviewed the charts of 32 patients in whom the CHARGE association was diagnosed. RESULTS: Of the 32 patients identified 22 (69%) had genitourinary abnormalities. Genital anomalies, including micropenis, penile agenesis, hypospadias, chordee, cryptorchidism, a bifid scrotum, atresia of the uterus, cervix and vagina, and hypoplastic labia majora, labia minora and clitoris, were present in 18 patients (56%). Of the 24 patients who underwent renal ultrasound 10 (42%) were diagnosed with urinary tract anomalies including a solitary kidney, hydronephrosis, renal hypoplasia and duplex kidneys. Further evaluation revealed vesicoureteral reflux, neurogenic bladder secondary to spinal dysraphism, nephrolithiasis, ureteropelvic junction obstruction and a nonfunctioning upper pole in both duplex kidneys. CONCLUSIONS: There is a high incidence of genitourinary anomalies in the CHARGE association. Because of this high incidence of anomalies, patients with this condition should undergo a careful genitourinary evaluation, including renal and bladder ultrasound, and voiding cystourethrography screening.

Abnormalities, Multiple↗
PubMed1983

[Spinal hemangioblastoma in infants with dysraphic malformation of the CNS].

Spinal hemangioblastoma in early infancy is an extreme rarity. In the case presented here such a hamartomatous tumor is combined with dysraphic malformation of the CNS in a male infant aged seven months. The morphological, epidemiological and clinical aspects are discussed with special reference to the possibility of congenital occurrence.

Age Factors↗
PubMed1979

Spinal computed tomography: limitations and applications.

A comprehensive review of 188 spinal computed tomography (CT) scans done over a 9-month period was performed to establish both the shortcomings and applications of the technique. The value of intrathecal enhancement was reviewed in an attempt to establish indications for its use. The data base comprised 51 abnormal scans including cases of spinal trauma, dysraphism, intra- and extraspinal neoplasm, and spinal stenosis. Results indicate that: (1) the introduction of intrathecal contrast media greatly extends the morphologic capabilities of spinal CT in practically all cases where altered anatomy of the spinal canal and its contents is suspected; (2) conventional screening procedures such as plain films of the spine and myelography should be obtained prior to spinal CT in most cases because of their value in defining the location and longitudinal extent of the pathologic process; (3) spinal CT examinations tailored to specific areas of interest result in better characterization of traumatic, dysraphic, and stenotic conditions conceivably leading to better surgical management and more accurate prognosis. At present, spinal CT has its greatest diagnostic usefulness in the evaluation of trauma and its sequela.

Constriction, Pathologic↗
PubMed1982

Comparative prenatal development of the spinal cord in normal and dysraphic dogs: embryonic stage.

The prenatal development of the canine spinal cord was examined by light microscopy in 8 normal embryos and compared with the development of the spinal cord in 10 embryos obtained by mating severely dysraphic Weimaraner dogs. Dysraphic lesions were found in 80% of the embryos examined from dysraphic matings. The primary lesion was aberrantly positioned mantle cells ventrad to the central canal in the floor plate area. In 30% of the embryos of this group, there was a division of the terminal neural tube. The dysraphic embryos had significantly shorter gray matter and spinal cord transverse diameters. This confirmed the observation that the mantle cells in the dysraphic specimens were less differentiated, being more compact and appearing more basophilic when hematoxylin and eosin stains were used.

Animals↗
PubMed2003

Dysembryogenetic spinal tumours in adults without dysraphism.

The authors report 15 patients with spinal intradural dysembryogenetic tumours with clinical onset in adult age in the absence of clinical and radiological signs of dysraphism. The series includes seven lipomas, four epidermoid cysts, three dermoid cysts and one teratoma. The tumour site was the thoracic region in three cases, the lumbar cord and conus in six, the cauda equina in four and the filum terminale in two. Among 14 patients operated upon, the surgical removal was complete in eight cases, subtotal in two and partial in four. Recurrence was noticed only in one subtotally resected thoracic epidermoid cyst. Magnetic resonance imaging allows a precise diagnosis of these lesions, mainly of small lipomas and dermoids of the conus and filum, where a tethered conus is responsible for clinical symptoms. Dysembryogenetic spinal rumours that become symptomatic in adult age may require surgical treatment. Reduction of the mass and release of any associated tethered neural elements are the goal of surgery for spinal lipomas, whereas epidermoid and dermoid cysts require a more radical treatment. However, even partial resections to avoid neural damage result in a good clinical outcome and very low risk of recurrence.

Adult↗