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An infant with congenital varicella syndrome due to maternal varicella zoster is presented. Unlike other reports in which such infection caused multiple congenital defects, our patient suffers from atonic bladder and anal dysfunction as the sole manifestation of congenital varicella syndrome. Attention is called for maternal varicella as a possible cause for limited neurologic damage in the fetus.
We studied the efficacy of a fourteen-day course of the fluoroquinolone, norfloxacin, in a group of 69 catheter-free spinal cord injury (SCI) outpatients who experienced a total of seventy-nine urinary tract infection (UTI) episodes. A total of 114 bacterial strains representing twenty species, including sixteen Pseudomonas aeruginosa, all susceptible to norfloxacin, were isolated from initial urine cultures. Mid-treatment cultures in 58/79 (73%) cases were negative. Cultures taken five to seven days after completing treatment in 42 cases (53%) were negative. In the remaining 37 there was persistence of initial or superinfecting pathogen(s), or early reinfection with new organisms. Cultures eight to twelve weeks later in 32 cases with initial eradication showed that 27 (84%) had either relapsed and/or become reinfected. Clinical cure occurred in 4/6 (67%) symptomatic episodes. Side effects occurred in 6 of 73 (8%) patients who received norfloxacin, 4 of whom required withdrawal from the study. Twenty of one hundred twenty-two (16%) bacterial strains isolated during or after treatment were resistant to norfloxacin. Given the limited oral treatment options and the constant risk of reinfection in the SCI population, norfloxacin appears to be a reasonable choice in many patients. However, as with other antimicrobials, when using norfloxacin in SCI patients, the emergence of resistant bacteria must be carefully monitored.
Urinary retention or incontinence is not an infrequent clinical finding in patients with neuroleptic malignant syndrome. We studied the pathophysiology of this voiding disorder by urodynamic testing. It revealed involuntary bladder contraction and rigidity of external sphincter (dyskinesia). These findings are analogous to those in Parkinson disease patients and support the dopamine deficiency theory as the cause of neuroleptic malignant syndrome.
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PURPOSE: We reviewed the long-term outcome of sigmoidocolocystoplasty. MATERIALS AND METHODS: The records of 86 patients who underwent sigmoidocolocystoplasty with a followup of more than 10 years were reviewed, focusing on histopathology, renal function, vesicoureteral reflux, bladder stones and urinary continence. We developed a urinary continence evaluation score, calculated as the postoperative minus the preoperative urinary continence score, including 0-incontinence, 1-severe leakage, 2-moderate leakage, 3-mild leakage, 4-almost dry and 5-dry, for patient self-assessment of urinary continence. A score of 2 or more meant improvement and a score of less than 2 meant no improvement. RESULTS: Mean followup was 13.1 years (range 10 to 21). Histopathology showed inflammation in all cases, hyperplasia in 9 and metaplasia in 4. In 80 of 86 cases creatinine clearance was normal. Ureteral reimplantation had been performed before sigmoidocolocystoplasty in 9 cases (13 kidneys) for grades III to V vesicoureteral reflux, which recurred as grade I in 1 (1 kidney). Ureteral reimplantation was performed during sigmoidocolocystoplasty in 31 cases (58 kidneys) and grades I to II vesicoureteral recurred in 5 (6 kidneys). Of the remaining 44 sigmoidocolocystoplasty cases there were 2 (2 kidneys) of grade I vesicoureteral reflux. On (99m)technetium-dimercapto-succinic acid scintigraphy there was increased renal scarring in 8 cases. Bladder stones were identified in 18 of 86 cases during followup but they were absent at review. The urinary continence evaluation score showed improvement in 61 cases and no improvement in 25. CONCLUSIONS: Our results suggest that sigmoidocolocystoplasty is safe and effective in the long term.
PURPOSE: The diagnostic criteria for secondary tethering of the spinal cord in children with prior closure of an open spinal defect are not well defined. Urological, neurological or orthopedic deterioration during followup remains the only strong indicator for the diagnosis of this condition. However, the outcome of untethering surgery cannot always be predicted. The aim of this study was to determine neurourological outcome after secondary untethering surgery in children with myelomeningocele. MATERIALS AND METHODS: Of 401 children with myelomeningocele followed at our multidisciplinary spina bifida clinic between 1996 and 2005, 56 (14%) were diagnosed with secondary tethering of the spinal cord (median age at diagnosis 4.1 years). The diagnosis was based on urological and neuro-orthopedic deterioration in 58% and 42% of children, respectively. Preoperative urological findings were compared with 6-month postoperative findings. RESULTS: Febrile urinary tract infection, upper tract dilatation and vesicoureteral reflux were preoperatively documented in 48.2%, 19% and 30% of the 56 children, respectively. At 6 months postoperatively urine cultures were sterile in 34% of patients and upper tract dilatation completely resolved in 45%. For all grades of vesicoureteral reflux complete resolution occurred in 47% of patients. Urodynamic parameters in terms of cystometric bladder capacity and detrusor leak point pressure substantially improved 6 months after untethering surgery (125 vs 170 ml and 69.1 vs 47.5 cm H2O, respectively, p < 0.05). Assessment of urodynamic findings in 19 children at 1 year failed to demonstrate a significant change in these parameters. A subgroup analysis according to patient age at untethering surgery revealed urodynamic improvement was more significant in children who were diagnosed and treated before age 7 years. CONCLUSIONS: Secondary tethering of the spinal cord is a major risk factor for urological and neurological deterioration in children with myelomeningocele. Close urological surveillance remains the most valuable tool in the early diagnosis. Our study shows that secondary untethering surgery may significantly improve urological outcome.
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BACKGROUND/PURPOSE: Spinal dysraphism and neurovesical dysfunction (NVD) frequently are associated in children with anorectal malformations (ARM). This study compares the urodynamic data from a selected group of patients with the results of their spinal and urologic imaging studies. METHODS: Twenty-six children (20 with isolated imperforate anus and six with persistent cloacal malformations) were investigated. All patients were evaluated with leak point pressures (LPP), renal ultrasound scan, and voiding cystourethrography. Eight children had urodynamics performed before and after posterior sagittal anorectoplasty (PSARP). The spinal cord was assessed using ultrasonography or magnetic resonance imaging. Current urologic status was obtained to provide long-term follow-up. RESULTS: Twenty-one of 26 children demonstrated elevated LPPs above the established normal value of 40 cm H2O, and 15 of these children had normal spinal imaging study findings. Uroradiographic studies findings showed that 12 of 21 children with elevated LPPs had hydronephrosis or vesicoureteral reflux with seven of these patients having normal spinal cords. LPPs in the eight patients with pre- and postoperative studies were 74 +/- 14.7 cm H2O and 68 +/- 31.8 cm H2O (mean +/- SD), respectively. CONCLUSIONS: These urodynamic and radiographic data confirm that NVD (elevated LPP) is common in patients with anorectal malformations despite normal spinal cords. Bladder dysfunction does not appear to be a sequelae of a properly performed PSARP. Patients with ARM and any uroradiographic or clinical urologic abnormality should undergo urodynamic testing even though the spinal studies are normal.
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PURPOSE: SCT treatment in newborns consists of surgery and selective chemotherapy. Few reports document urological sequelae using this approach. This review focuses on the urological and neurourological findings following SCT treatment in the newborn period. MATERIALS AND METHODS: We reviewed the records of all infants with SCT resected in early infancy who underwent urodynamic evaluation for bladder dysfunction between 1986 and 2004. The radiological, neurological and urodynamic findings, and postoperative incontinence management were analyzed. RESULTS: We analyzed UDS of 14 patients who presented with urinary infection or incomplete bladder emptying after SCT resection. At the time of UDS an abnormal neurological examination was noted in 5 patients (36%). Detrusor overactivity was seen in 8 patients, underactivity in 2 and normal activity in 4. Abnormal urethral sphincter EMG potentials were observed in 7 of 13 patients (54%). Five of 13 patients (38%) had sphincter dyssynergia during voiding. Consequently, CIC was needed in 11 of the 14 patients (79%) to empty the bladder, of whom 5 also required anticholinergics to improve detrusor compliance and dryness. Only 3 patients voided spontaneously with normal bladder and sphincter function, of whom 2 were toilet trained. Hydronephrosis was seen in 6 patients and reflux was noted in 7 (including 5 of 6 with hydronephrosis). Antireflux surgery was performed in 6 patients, all of whom had up to grade 4 reflux due to recurrent urinary tract infection. One girl with grade 2 reflux had spontaneous resolution. CONCLUSIONS: SCT and its treatment can produce neurourological dysfunction of the lower urinary tract with high grade reflux, and abnormal bladder and urethral function. Complete assessment, including urodynamic studies, is imperative preoperatively and postoperatively. Constant vigilance is required to maintain as near normal bladder function as possible and to prevent upper urinary tract injury.