We report the case of a 9-year-old girl who presented to our hospital at the age of 6 years with recurrent febrile urinary tract infections started at 6 months, associated with nocturnal enuresis, daytime urinary frequency, and urge incontinence. Initial evaluation revealed a routine physical exam with no evidence of neurological abnormalities. Urinary tract ultrasound failed to demonstrate structural abnormities; micturition cystourethrogram documented the absence of vesicoureteral reflux. Accordingly, the urodynamic study demonstrated the presence of low bladder compliance and reduced bladder capacity. A whole spine magnetic resonance imaging showed isolated syringomyelia extending from T4 to T12 and measuring about 2.7mm in anterior-posterior (AP) diameter at the D8D9 level. After the failure of initial management of bladder overactivity using anticholinergic medications, the patient was managed by repeated cystoscopic injections of 200 international units of OnabotulinumtoxinA, with good symptomatic relief. After three years of follow-up 3 years, investigations and clinical findings failed to demonstrate neurological or urodynamic deterioration.

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