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Infection of Arachnoid Cyst Associated with Vasospasm and Stroke in a Pediatric Patient: Case Report Jenna R

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Infection of Arachnoid Cyst Associated with Vasospasm and Stroke in a Pediatric Patient: Case Report Jenna R CASE REPORT J Neurosurg Pediatr 26:594–598, 2020 Infection of arachnoid cyst associated with vasospasm and stroke in a pediatric patient: case report Jenna R. Gale, BA, Kamil W. Nowicki, MD, PhD, Rachel M. Wolfe, BS, Roberta K. Sefcik, MD, MSCR, and Taylor J. Abel, MD Department of Neurological Surgery, University of Pittsburgh School of Medicine, Pittsburgh, Pennsylvania Arachnoid cysts are relatively common and benign intraarachnoid membrane outpouchings containing CSF-like fluid. The majority of arachnoid cysts remain stable and asymptomatic and do not require intervention in the pediatric population. Here, the authors present the first reported case of an infected arachnoid cyst in a pediatric patient resulting in severe vasospasm of the left terminal internal carotid artery, left A1 segment, and left M1 branches with a left middle cerebral artery infarct. Their experience suggests that close monitoring is warranted for this condition and that the pediatric population may be at higher risk for vasospasm. https://thejns.org/doi/abs/10.3171/2020.5.PEDS20419 KEYWORDS pediatric; arachnoid cyst; meningitis; abscess; vasospasm; vascular disorders NTRACRANIAL arachnoid cysts (IACs) are CSF-filled transfer from a local hospital because of acute on chronic outpouchings that occur on the arachnoid membrane hearing loss, headache, fever, and difficulty ambulating. within the skull. Arachnoid cysts are the most common His laboratory workup was remarkable for profound leu- Itype of brain cysts, with a reported prevalence between kocytosis with a white blood cell (WBC) count at 46.8 × 1.4% and 2.6% in the adult and pediatric populations, re- 109/L with 42% neutrophils. On admission, the patient was spectively.1 There are several reported cases of familial febrile and demonstrated bilateral hearing loss but other- IACs and several genetic syndromes in which IACs occur wise had an unremarkable neurological examination. He including autosomal dominant polycystic kidney disease, was awake, alert, and oriented and had Medical Research acrocallosal syndrome, and Aicardi syndrome.2–5 Typical- Council (MRC) 5/5 strength in all major muscle groups ly, IACs are asymptomatic and found incidentally on imag- and full sensation in all extremities without hyperreflexia ing.6 However, there are documented cases of cyst enlarge- bilaterally. Initial head CT and MRI revealed sinusitis and ment, and larger cysts and recent head trauma have been an incidental left temporal arachnoid cyst measuring 4.4 identified as risk factors for rupture and hemorrhage.7–9 cm (Fig. 1A). Neurology and neurosurgery were consulted, Treatment options can range from observation to medical but the cyst was monitored as an incidental finding. The management with acetazolamide to surgical management patient was admitted for presumed bacterial meningitis involving cyst fenestration and/or shunting with or without and treated with 500 mg vancomycin and 2 g ceftriaxone. endoscopy.10 IACs can complicate other medical illnesses A lumbar puncture was notable for an opening pressure 3 such as meningitis, but only a few such cases have been greater than 50 cm H2O, 1247/mm WBCs, and hypogly- reported. One group reported a case of IAC drainage in the corrhachia, but no organism was identified in the setting of setting of meningitis, but no evidence of abscess formation, antibiotic pretreatment. Although he was started on 4.5 mg such as frank purulent material, cyst wall enhancement, or intravenous dexamethasone every 6 hours for 48 hours for cultured organism, was noted.11 Here, we report the case neuroprotection and to attempt preservation of hearing, an of a 7-year-old boy with meningitis and a superinfected audiology consult revealed profound bilateral sensorineu- arachnoid cyst resulting in infection-induced vasculitis and ral hearing loss, and an evaluation for cochlear implants a middle cerebral artery (MCA) infarct. was initiated. One week after admission, MRI was performed due Case Report to persistent fever and worsening headache. The MRI re- vealed worsening leptomeningeal enhancement of the pre- History and Examination vious meningitis as well as new abscess formation within A previously healthy 7-year-old boy presented as a the patient’s previously detected arachnoid cyst (Fig. 1B) ABBREVIATIONS IAC = intracranial arachnoid cyst; ICA = internal carotid artery; MCA = middle cerebral artery; MRC = Medical Research Council. SUBMITTED May 19, 2020. ACCEPTED May 22, 2020. INCLUDE WHEN CITING Published online August 28, 2020; DOI: 10.3171/2020.5.PEDS20419. 594 J Neurosurg Pediatr Volume 26 • November 2020 ©AANS 2020, except where prohibited by US copyright law Unauthenticated | Downloaded 09/27/21 04:54 AM UTC Gale et al. FIG. 1. A: Axial MRI scans obtained on admission, showing a left anterior temporal subarachnoid cyst with mild dural enhance- ment. B: Axial MRI scans obtained 1 week later, showing diffusion-weighted imaging (DWI) restriction within the subarachnoid cyst with increasing leptomeningeal enhancement concerning for cyst superinfection. ADC = apparent diffusion coefficient. and septic emboli in the left-sided white matter (Supple- A second head CT study was performed several days mentary Fig. 1A). His physical examination had remained later, which revealed the previously noted left arachnoid stable since admission, and he was at his neurological cyst without evidence of intracranial abscess. Three weeks baseline. He communicated by writing on a board. after admission, the patient was discharged to an in-pa- tient rehabilitation unit where he remained for a period of Operation several months. At the time of discharge to rehabilitation, Given the presence of an abscess in the arachnoid cyst, he exhibited a mild right facial droop, had complete deaf- the patient underwent an emergency left craniotomy with ness bilaterally, moved his left upper and lower extremities cyst fenestration, washout for drainage of the abscess, and spontaneously and against gravity but had no spontaneous drain placement. There was purulence on the periphery movement in his right upper or lower extremities. Repeat and surface of the cyst, and the entirety of cyst fluid had a brain MRI with and without contrast 1 month after dis- cloudy and proteinaceous appearance. charge showed a decreased size of the arachnoid cyst with some residual cyst wall enhancement but no evidence of Postoperative Course abscess (Fig. 4). One month later, the patient underwent Following surgery, examination revealed that the pa- surgery without complications for bilateral cochlear im- tient was at his preoperative baseline. He was awake, alert, plants, and his hearing improved significantly over the had full sensation, and moved all extremities with full course of several months (Fig. 5). strength in the major muscle groups on physical examina- At the last follow-up, 5 months after his initial presen- tion. Postoperative head CT showed expected decompres- tation for bacterial meningitis complicated by infection sion. CTA was performed postoperatively to evaluate for of an arachnoid cyst, infection-induced vasculitis, and vasospasm because preoperative MRI/MRA had revealed left MCA infarct, the patient was continuing to make im- possible asymmetrical mild left internal carotid artery provements in therapy. On physical examination, he had (ICA) and M narrowing. It was unclear from the initial slight right-sided facial weakness, normal strength and 1 tone throughout all major muscle groups in his left upper study if this was attributable to mass effect due to the cyst and lower extremities, and right hemiparesis. Deep tendon and empyema or vasospasm; hence, CTA was ordered. A reflexes were MRC 2+ bilaterally at the biceps and pa- nimodipine drip was started out of concern for the cere- tella with no clonus. Despite his right-sided hemiparesis, bral vasospasm seen on CTA (Supplementary Fig. 1C). the patient demonstrated a stable gait, ambulating with a Formal cerebral DSA was subsequently performed, which solid ankle-foot orthotic and Lofstrand crutch along his showed severe vasospasm in the left terminal ICA, left A1, left arm. Close follow-up and repeat head CT scans were and left M1 that did not respond to a 4-mg verapamil and recommended given the unusual nature of this patient’s 1-mg nicardipine injection into the left ICA (Fig. 2). After clinical course. DSA, on physical examination, the patient exhibited de- creased movement of his right upper and lower extremi- ties, prompting a second MRI/MRA study, which showed Discussion a progressive infarct in the left MCA territory and no flow Arachnoid cysts are intraarachnoid membrane out- in the left MCA (Fig. 3). A beaded appearance was also pouchings containing CSF-like fluid.12 Their prevalence in noted at the branch point of the anterior cerebral artery the pediatric population is approximately 2.6%, they are and ICA. The patient was then started on methylpredniso- often detected as an incidental finding on imaging, and lone out of concern for infection-induced CNS vasculitis. an overwhelming majority are stable from the time of ini- J Neurosurg Pediatr Volume 26 • November 2020 595 Unauthenticated | Downloaded 09/27/21 04:54 AM UTC Gale et al. FIG. 2. Left anterior circulation vasospasm (A). DSA showed severe vasospasm in the left terminal ICA (thin arrow, B), left A1 (ar- rowhead), and left M1 (broad arrow). Despite intraarterial infusion of nicardipine and verapamil, no improvement in vasospasm was observed (C). tial diagnosis.1,6 It is estimated that only 6.8%
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