An infant with an infected spinal epidermoid cyst associated with a congenital dermal sinus developed recurrent complicated urinary tract infections after neurosurgical treatment, according to a case report. The 9-month-and-26-day-old girl initially presented with four weeks of intermittent fever, followed by acute flaccid paraplegia over three days. Magnetic resonance imaging identified a dermal sinus extending from a sacral skin dimple into the spinal canal and an intradural cystic mass from L1 to S2. After surgery and drainage of the lesion, she developed neurogenic bladder requiring clean intermittent catheterization every four to six hours. Serial cultures subsequently identified Klebsiella aerogenes, Candida glabrata and multidrug-resistant Enterococcus faecium, illustrating a prolonged infection-management challenge in a catheterized infant.
Congenital dermal sinus is a rare form of occult spinal dysraphism in which an epithelial-lined tract connects the skin surface with the spinal canal. The tract can provide a route for central nervous system infection, including meningitis, intramedullary abscess or infected spinal epidermoid cysts. An infected cyst may produce acute neurological deterioration and requires surgical management. This infant had previously been identified on magnetic resonance imaging as having spina bifida occulta. During the earlier febrile prodrome, she had no evident neurological signs, even though temperatures reached 39.6 degrees Celsius and antibiotics had produced only transient responses at a local hospital. A sacral dimple with localized hypertrichosis was noted when she was admitted. Her neurological state at that point was severe: examination showed flaccid paraplegia with no measurable muscle power, complete sensory loss below the umbilicus and absent reflexes.
Emergency imaging of the whole spine revealed diffuse spinal cord swelling and abnormal signal from the cervical through lumbar regions. Contrast-enhanced magnetic resonance imaging showed a linear tract extending from the sacral dimple into the spinal canal, together with a non-enhancing intradural mass between L1 and S2. Imaging also suggested a low-lying conus medullaris and a tethered cord. The lesion was hypointense on T1-weighted images, heterogeneously hyperintense on T2-weighted images and associated with peripheral enhancement. Follow-up imaging also showed local diffusion restriction and rim enhancement, described in the report as consistent with residual or recurrent infection. Because the original images were unavailable for de-identified publication, the radiological account was based on outside imaging reports and clinical records. The authors presented the four-week fever without earlier neurological signs as a possible warning of congenital dermal sinus, although this interpretation comes from a single case.
On day 18 after admission, surgeons at one hospital removed the intraspinal lesion, excised the dermal sinus and decompressed the spinal canal. A localized dural defect was identified, although the dura was not actively opened, and the sinus entered the subdural space through that defect. No cerebrospinal-fluid leak occurred. Histopathology confirmed an infected epidermoid cyst secondary to congenital dermal sinus. Purulent material from the cyst grew K. aerogenes that was resistant to ceftriaxone but susceptible to meropenem and cefoperazone-sulbactam. The infant developed neurogenic bladder after the operation and began clean intermittent catheterization every four to six hours. Because the neurogenic bladder was associated with incomplete emptying, the team monitored residual urine by ultrasound and maintained a bladder diary. The infant was initially treated empirically for possible immune-mediated myelitis with ceftriaxone, vancomycin, methylprednisolone and intravenous immunoglobulin, before therapy was changed to meropenem according to culture results.
The first positive urine culture was collected on day 25, approximately one week after surgery and before catheterization had begun. It yielded more than 100,000 colony-forming units per milliliter of K. aerogenes. The specimen was obtained by catheterization, pyuria was present at a reported 3-plus level, and the infant had a temperature of 38.5 degrees Celsius. The authors classified this episode as a true infection and treated her with meropenem for 14 days, after which the culture cleared. By day 45, a catheterized urine specimen contained 6.8 × 10^4 CFU/mL of C. glabrata. Pyuria, cloudy urine and persistent sediment were present even though she was afebrile. The isolate was fluconazole-susceptible dose dependent and susceptible to micafungin, and she received micafungin for 21 days. The case illustrates why a positive culture in a catheterized infant requires interpretation alongside pyuria, fever, urinary symptoms and other clinical evidence rather than colony count alone.
A subsequent series of four cultures collected from days 70 to 110 each yielded more than 100,000 CFU/mL of multidrug-resistant E. faecium. Two urinalyses were normal, the infant was asymptomatic, and no targeted antimicrobial therapy was administered. The clinical team therefore classified the repeated finding as colonization rather than infection. The report defined recurrent complicated urinary tract infection in this setting as at least two episodes with pyuria—more than 10 white blood cells per high-power field—and clinical symptoms such as fever or urinary symptoms, or a single episode with systemic involvement. Asymptomatic bacteriuria or candiduria without pyuria was not treated. This distinction was central to the case because repeated positive cultures in a chronically catheterized child do not, on their own, establish active infection.
At 18 to 20 weeks, C. glabrata was isolated again, this time at 2,800 CFU/mL with pyuria and urinary sediment. The organism was resistant to fluconazole, with a reported minimum inhibitory concentration of 128, but remained susceptible to micafungin and intermediate to caspofungin. The infant again received micafungin, and the subsequent culture cleared. The change in antifungal susceptibility raised concern that prolonged broad-spectrum treatment and treatment of what may have been colonization could have selected for resistant C. glabrata. The authors cited guidance against routinely treating asymptomatic candiduria except in defined higher-risk groups, including neutropenic patients, very-low-birth-weight infants and patients undergoing urologic manipulation. They presented the episode as a cautionary antimicrobial-stewardship narrative rather than a general treatment protocol, and the reported resistance emergence could not be assigned conclusively to a single antimicrobial exposure.
Recovery depended on combining serial, susceptibility-guided antimicrobial therapy with management of urinary stasis and potential infection reservoirs. Beyond the targeted courses of meropenem and micafungin, the clinical team used temporary indwelling catheterization and intravenous antibiotics during recurrent infection before returning to clean intermittent catheterization. Bladder stones were detected at 12 weeks, and transurethral cystoscopic lithotripsy was performed on February 10, 2026. The stones could have provided a persistent reservoir for microorganisms and biofilm, but their composition was not analyzed, and the report could not establish a causal relationship between them and the infections. Micafungin was selected despite its limited urinary excretion because the Candida isolate was fluconazole-resistant and amphotericin B was avoided because of its nephrotoxicity. The authors explicitly declined to attribute successful clearance to micafungin alone, instead implicating the combined effects of culture-guided therapy, catheterization management and stone removal.
At the final assessment, when the child was 1 year and 3 months old, proximal lower-limb strength had improved to grade 4 out of 5, while distal strength remained grade 2 out of 5. She retained bilateral foot drop, more pronounced on the left, and her neurogenic bladder continued to require clean intermittent catheterization. Constipation was present without fecal incontinence and was managed with lactulose and glycerin enemas. The anal reflex was normal, while pathological reflexes were consistently absent. Somatosensory evoked potentials showed normal bilateral ankle-cortex and wrist-cortex pathways, although compound muscle action-potential amplitudes changed in several nerves. No further febrile urinary tract infections occurred after stone removal through the reported follow-up period, but this was an observational result without a comparison group. Long-term prognosis remained dependent on bladder function, and the investigators stated that lifelong urological follow-up, catheterization, renal ultrasound and monitoring for stones or recurrent infection would be required.
The report is limited by its single-patient design, incomplete symptom and urinalysis data for some episodes, variable specimen-collection methods and the absence of stone analysis. Urodynamic studies were not performed because of the patient’s age, the need for sedation and a lack of standardized normative pediatric data, so bladder management was guided by ultrasound, residual urine and clinical symptoms. Imaging was performed outside the treating institutions and could not be independently retrieved for de-identified publication. The dura was not actively opened, although a localized dural defect was found during surgery, and no cerebrospinal-fluid leak occurred. These constraints prevent the findings from establishing how frequently neurogenic bladder leads to recurrent complicated urinary tract infection after congenital dermal sinus surgery, or proving that stone removal, catheterization practices and any particular antimicrobial produced the observed clearance. Nevertheless, the case provides detailed longitudinal culture and susceptibility data, showing how bacterial and fungal pathogens can alternate in a catheterized neurogenic bladder and why repeated cultures must be interpreted alongside pyuria, symptoms and the clinical course.
Subject of Research: Recurrent complicated urinary tract infections in an infant with postoperative neurogenic bladder and congenital dermal sinus
Article Title: Prolonged fever as a harbinger: the diagnostic and therapeutic challenge of recurrent complicated urinary tract infection following congenital dermal sinus excision in an infant: a case report
Article References: Liao, L., & Peng, W. (2026). Prolonged fever as a harbinger: the diagnostic and therapeutic challenge of recurrent complicated urinary tract infection following congenital dermal sinus excision in an infant: a case report. Frontiers in Pediatrics, 14, Article 1900708. https://doi.org/10.3389/fped.2026.1900708
Image Credits: AI Generated
DOI: 10.3389/fped.2026.1900708
Keywords: congenital dermal sinus, neurogenic bladder, recurrent urinary tract infection, infant, Candida glabrata, Enterococcus faecium, antimicrobial stewardship, clean intermittent catheterization, spinal epidermoid cyst, pediatric surgery, Prolonged, fever
News Source: Ophelia Keating. (October 21, 2026). Single infant case links spinal surgery, neurogenic bladder and recurrent UTIs. Scienmag.



