A Rare Case of Spontaneous Arachnoid Cyst Rupture Presenting As Right Hemiplegia and Expressive Aphasia in a Pediatric Patient
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children Case Report A Rare Case of Spontaneous Arachnoid Cyst Rupture Presenting as Right Hemiplegia and Expressive Aphasia in a Pediatric Patient Anne Bryden 1,*, Natalie Majors 2, Vinay Puri 3 and Thomas Moriarty 4 1 Department of Neurology and Pediatrics, University of Louisville SOM, Louisville, KY 40202, USA 2 Department of Neurology and Pediatrics, Vanderbilt University, Nashville, TN 37420, USA; [email protected] 3 Department of Neurology and Pediatrics, University of Louisville, Louisville, KY 40202, USA; [email protected] 4 Department of Neurological Surgery and Pediatrics, University of Louisville, Louisville, KY 40202, USA; [email protected] * Correspondence: [email protected]; Tel.: +1-(570)-951-2998 Abstract: This study examines an 11-year-old boy with a known history of a large previously asymptomatic arachnoid cyst (AC) presenting with acute onset of right facial droop, hemiplegia, and expressive aphasia. Shortly after arrival to the emergency department, the patient exhibited complete resolution of right-sided hemiplegia but developed headache and had persistent word- finding difficulties. Prior to symptom onset while in class at school, there was an absence of reported jerking movements, headache, photophobia, fever, or trauma. At the time of neurology consultation, the physical exam showed mildly delayed cognitive processing but was otherwise unremarkable. The patient underwent MRI scanning of the brain, which revealed left convexity subdural hematohygroma Citation: Bryden, A.; Majors, N.; and perirolandic cortex edema resulting from ruptured left frontoparietal AC. He was evaluated by Puri, V.; Moriarty, T. A Rare Case of neurosurgery and managed expectantly. He recovered uneventfully and was discharged two days Spontaneous Arachnoid Cyst after presentation remaining asymptomatic on subsequent outpatient visits. The family express Rupture Presenting as Right concerns regarding increased anxiety and mild memory loss since hospitalization. Hemiplegia and Expressive Aphasia in a Pediatric Patient. Children 2021, 8, Keywords: pediatrics; arachnoid cysts; spontaneous rupture; aphasia; neurological deficits 78. https://doi.org/10.3390/ children8020078 Academic Editor: Marco Carotenuto 1. Introduction Received: 15 November 2020 Arachnoid cysts (ACs) are cerebral spinal fluid (CSF) collections between two layers Accepted: 22 January 2021 Published: 24 January 2021 of arachnoid membrane that are primarily congenital in origin or acquired secondary to trauma, infection, or skull-based surgery. ACs are a relatively common developmental anomaly that are frequently diagnosed incidentally by intracranial imaging in pediatric Publisher’s Note: MDPI stays neutral with regard to jurisdictional claims in patients [1–3]. They represent 1% of all intracranial masses [4] with a prevalence of around published maps and institutional affil- 2.6% in children [1] and male predominance [1,5–7]. In general, arachnoid cysts have a iations. benign course with a large proportion of patients remaining asymptomatic. However, they can be associated with clinical symptoms, such as chronic headache, seizure, asymmetric macrocrania, or hydrocephalus [8,9]. Arachnoid cysts may also present with symptoms as a consequence of cyst rupture causing acute subdural hygroma, subdural hematoma, and/or intracystic hemorrhage [2]. Copyright: © 2021 by the authors. Larger arachnoid cyst size, a median size of 2.2 × 2.6 × 2.5 cm [5], and recent head trauma Licensee MDPI, Basel, Switzerland. This article is an open access article are risk factors for symptomatic rupture [10]. Spontaneous or nontraumatic rupture appear distributed under the terms and as a rare event, with a total of only 57 reported cases of spontaneous AC rupture [11]. conditions of the Creative Commons The most frequently reported symptom following spontaneous rupture is headache (82%) Attribution (CC BY) license (https:// and nausea/vomiting (35.3%). Neurological deficits and altered consciousness are rarely creativecommons.org/licenses/by/ reported [11]. 4.0/). Children 2021, 8, 78. https://doi.org/10.3390/children8020078 https://www.mdpi.com/journal/children Children 2021, 8, x FOR PEER REVIEW 2 of 6 Children 2021, 8, 78 and nausea/vomiting (35.3%). Neurological deficits and altered consciousness are rarely2 of 5 reported [11]. This case describes a rare spontaneous AC rupture, and, to our knowledge, this is the firstThis spontaneous case describes AC rupture a rare spontaneous that presents AC with rupture, acute and,onset to focal our knowledge,neurological this signs, is thenamely first spontaneousfacial nerve injury, AC rupture expressive that presents aphasia, withand hemiplegia. acute onset focal neurological signs, namely facial nerve injury, expressive aphasia, and hemiplegia. 2. Patient Description 2. PatientThe Descriptionpatient, an 11-year-old male with known history of a previously asymptomatic, left frontalThe patient, arachnoid an 11-year-old cyst, found male incidentally with known on imaging history after of a previouslypersistent headaches asymptomatic, at age left4, was frontal in arachnoidhis normal cyst, state found of health incidentally until experiencing on imaging aftersudden-onset persistent dizziness headaches while at age in 4,class. was inShortly his normal after, statehe developed of health until a right-si experiencingded lower sudden-onset facial droop, dizziness right hemiplegia, while in class. and Shortlyexpressive after, aphasia. he developed There awas right-sided no history lower of trauma, facial droop, seizures, right or hemiplegia, other neurological and expres- con- siveditions, aphasia. and the There patient was nowas history otherwise of trauma, in good seizures, health. These or other symptoms neurological began conditions, to resolve anden route the patient to a pediatric was otherwise tertiary in goodcare center health. vi Thesea a medical symptoms stat beganflight. toUpon resolve arrival, en route the tore- amaining pediatric symptoms tertiary care included center some via a medicalright arm stat we flight.akness, Upon mild-to-modera arrival, the remainingte dysarthria, symp- flat- tomstened included nasolabial some fold, right and arm facial weakness, asymmetry mild-to-moderate on smiling. There dysarthria, was near-complete flattened nasolabial symp- fold,tom andresolution facial asymmetryat the time onof neurology smiling. There consultation was near-complete 5 min after symptominitial exam resolution and 70 min at thefollowing time of neurologysymptom onset. consultation The neurological 5 min after exam initial showed exam and mildly 70 min delayed following cognitive symptom pro- onset.cessing, The but neurological the cranial exam nerve, showed motor, mildly sensory, delayed coordination, cognitive and processing, gait exams but thewere cranial unre- nerve,markable. motor, There sensory, was coordination,an absence of and witnesse gait examsd/reported were unremarkable.convulsions, abnormal There was move- an absencements, loss of witnessed/reported of consciousness, headache, convulsions, photop abnormalhobia, movements,or fever. There loss was of consciousness, no evidence of headache,external trauma photophobia, on physical or fever. exam There or report was no of evidence trauma preceding of external and/or trauma during on physical symp- examtoms. or report of trauma preceding and/or during symptoms. FiguresFigures1 1and and2 reveal2 reveal a changea change in signalin signal of arachnoidof arachnoid cyst cyst suggestive suggestive of intervalof interval hemorrhagehemorrhage with with a a thin thin subdural subdural hematoma hematoma along along its its inferior inferior aspect. aspect. Minimal Minimal edema edema in in thethe subjacent subjacent Rolandic Rolandic cortex cortex was was present. present. Given Given the the patient’s patient’s stroke-like stroke-like presentation, presentation, an an MRAMRA of of head head and and neck neck was was administered, administered, which which was was found found to to be be normal. normal. The The findings, findings, withwith aa comparisoncomparison of the previous previous MRI MRI of of th thee patient patient at atage age 4 (Figures 4 (Figures 3 and3 and 4),4 ),are are con- consistentsistent with with a ruptured a ruptured arachnoid arachnoid cyst. cyst. No Noacute acute neurosurgical neurosurgical intervention intervention was was per- performedformed due due to to the the absence absence of of raised raised intracranial intracranial pressure and reductionreduction ofof neurological neurological symptoms. A routine EEG performed in the emergency department exhibited interictal symptoms. A routine EEG performed in the emergency department exhibited interictal epileptiform discharges over the left and right central–temporal region during drowsy and epileptiform discharges over the left and right central–temporal region during drowsy sleep states, suggestive of increased risk for partial seizures from these regions. and sleep states, suggestive of increased risk for partial seizures from these regions. FigureFigure 1. 1.MRI MRI T2WI, T2WI, sagittal sagittal section section in in 2019 2019 (11 (11 years years of of age). age). The patient received 500 mg levetiracetam twice daily for seizure prophylaxis. On day 2 of admission, a rapid-sequence MRI brain scan showed a stable thin subdural hematoma and a ruptured arachnoid cyst at the left frontoparietal vertex with the interval resolution of edema in the subjacent cortex. At discharge on day 3, the patient’s neurological