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Open access

Arteriovenous malformation presenting as complex regional pain syndrome: illustrative case

Dayna C Sloane, Diego D Luy, Atul K Mallik, Joseph C Serrone, and Douglas E Anderson

BACKGROUND

Complex regional pain syndrome (CRPS) is typically described as a peripheral nerve disorder in which exaggerated allodynia and hyperalgesia follow a minor injury. Some researchers propose a central mechanism, although current evidence is lacking.

OBSERVATIONS

A 14-year-old female presented with classic CRPS symptoms of left upper-extremity weakness and hyperalgesia after a bout of sharp pain in her thumb while shoveling snow. A possible seizure prompted magnetic resonance imaging, revealing a right frontal Spetzler-Martin grade II arteriovenous malformation (AVM) adjacent to the primary motor cortex. Brodmann areas 1, 3a, and 3b, which are responsible for localizing and processing burning and painful sensations, were also involved. The patient underwent transarterial Onyx embolization in two sessions and microsurgical resection, after which her CRPS symptoms completely resolved.

LESSONS

To our knowledge, this is the first reported case of a cerebral AVM presenting as CRPS, which supports a central mechanism. The authors propose that rapid growth of the AVM led to a vascular steal phenomenon of surrounding parenchyma, which disrupted the patient’s normal motor function and nociceptive processing. Further validation in other series is needed.

Open access

Intracranial solitary fibrous tumor in a 15-year-old girl: illustrative case

Kuan Lu, Xiaoqing Qu, Jingcheng Jiang, Quanjun Zheng, Yongsheng Ao, and Lihua Qiu

BACKGROUND

Solitary fibrous tumor (SFT) is an infrequent spindle cell tumor derived from mesenchymal tissue, which can manifest in diverse anatomical locations, primarily in the pleural cavity and infrequently in the central nervous system. SFT is predominantly observed in individuals aged between 40 and 50 years old, with a slightly higher occurrence in males than in females.

OBSERVATIONS

This case report describes a female, age 15, who had migraines for 2 months prior to the diagnosis of an intracranial tumor. Computed tomography and magnetic resonance imaging showed a mass located in the right parietooccipital region with surrounding edema and a compressed right lateral ventricle. Neurosurgery was utilized to successfully remove the mass, and single intracranial fibrous tumor (grade I) was identified by postoperative pathological analysis. During an 8-month follow-up period, the patient did not experience any recurrences.

LESSONS

SFT is often misdiagnosed as meningioma because of their similar imaging characteristics. However, identifying the distinctive features of SFT on magnetic resonance imaging can distinguish it from meningioma and help to select appropriate treatment. The complete preoperative imaging data for this case indicated a highly vascularized tumor. Preoperative vascular embolization treatment reduced any difficulties during the subsequent tumor resection and minimized intraoperative bleeding.

Open access

Laser interstitial thermal therapy as a radiation-sparing approach for central nervous system tumors in children with cancer predisposition syndromes: report of a child with Li-Fraumeni syndrome. Illustrative case

Sergio W Guadix, Abhinav Pandey, Carson Gundlach, Michael Walsh, Nelson S Moss, and Mark M Souweidane

BACKGROUND

Ionizing radiation and alkylating chemotherapies increase secondary malignancy risk in patients with cancer predisposition syndromes (CPSs), such as Li-Fraumeni syndrome. Laser interstitial thermal therapy (LITT) is a minimally invasive ablation technique that has not been associated with mutagenic risks. We describe the case of a child with LFS and a history of treated choroid plexus carcinoma (CPC) who developed a second primary glial tumor that was safely treated with magnetic resonance imaging (MRI)–guided LITT.

OBSERVATIONS

A 4-year-old male with left parietal World Health Organization grade III CPC associated with a TP53 germline mutation was evaluated. The patient underwent neoadjuvant platinum-based chemotherapy before near-total resection, followed by 131I-8H9 immunotherapy and 30 fractions of 54-Gy proton radiotherapy. He remained without evidence of disease for 2 years before developing a slow-growing mass adjacent to the left frontal ventricular horn. Stereotactic biopsy revealed a glial neoplasm. Given the nonsuperficial location and focality of the lesion, MRI-guided LITT was performed for ablative therapy. There were no complications, and 2 years of surveillance revealed continued retraction of the ablated tumor focus and no subsequent disease.

LESSONS

Alternatives to mutagenic therapies for brain tumors should be explored for patients with CPS. LITT paired with imaging surveillance is a logical strategy to ensure durable outcomes and mitigate treatment-related secondary neoplasms.

Open access

The complex treatment paradigms for concomitant tethered cord and scoliosis: illustrative case

Rose Fluss, Riana Lo Bu, Andrew J Kobets, and Jaime A Gomez

BACKGROUND

Scoliosis associated with tethered cord syndrome is one of the most challenging spinal deformities to manage. Multiple surgical approaches have been developed, including traditional staged and concomitant procedures, spine-shortening osteotomies, and individual vertebral column resections.

OBSERVATIONS

A 10-year-old female presented with congenital kyphoscoliosis with worsening curve progression, tethered spinal cord, and a history of enuresis. The scoliosis had progressed to a 26° coronal curve and 55° thoracolumbar kyphosis. Preoperative magnetic resonance imaging of the spine revealed a tethered cord between the levels of L3–4 and a large kyphotic deformity at L1. The patient underwent laminectomy, during which intraoperative motor signals were lost. A planned hemivertebrectomy at L1 was performed prior to an L4 laminectomy, untethering of the filum terminale, and posterior spinal fusion from T11 to L2. After surgery, the patient experienced transient lower-extremity weakness, with her neurological function improving from baseline over the next 2 months. Ultimately, the goal of this surgery was to halt the progressive decline in motor function, which was successfully achieved.

LESSONS

Much remains to be learned about the treatment of this complicated disease, especially in the setting of concomitant scoliosis. This case serves to exemplify the complex treatment paradigms that exist when attempting to manage this clinical syndrome and that more remains to be learned.

Open access

Nontraumatic symptomatic de novo arachnoid cyst in an adolescent patient treated with cystoperitoneal shunting: illustrative case

Angelica M Fuentes, Jonathan J Yun, and John A Jane Jr.

BACKGROUND

Arachnoid cysts are often congenital, asymptomatic lesions detected in the pediatric population. When seen in adults, they usually occur following trauma. De novo formation of arachnoid cysts is uncommon, with only a few instances cited in the literature and most of which occurred in the pediatric population. Treatment options for these lesions include observation, craniotomy for cyst resection, microsurgical/endoscopic fenestration, or shunting.

OBSERVATIONS

In this report, the authors describe a female patient with a de novo, symptomatic, enlarging middle cranial fossa arachnoid cyst detected at age 16 years. She was treated with the placement of a cystoperitoneal shunt. After surgery, she experienced clinical and radiological improvement.

LESSONS

We illustrate successful shunting of a de novo arachnoid cyst in a symptomatic teen patient. Although arachnoid cysts in certain intracranial locations are more likely to produce symptoms, those in the middle cranial fossa tend to be asymptomatic. To our knowledge, this is the first reported case of a symptomatic de novo arachnoid cyst located in the middle cranial fossa in a postpubertal patient. Although the current presentation is rare, the authors demonstrate an effective surgical treatment of a symptomatic, large, de novo arachnoid cyst in a postpubertal pediatric patient.

Open access

Surgical management of pediatric spinal aneurysmal bone cysts: patient series

Benjamin E Flyer, Erik B Vanstrum, Nicholas Chapman, Joseph H Ha, Jacob K Al-Husseini, Jason K Chu, J. Gordon McComb, Susan R Durham, Mark D Krieger, and Peter A Chiarelli

BACKGROUND

Aneurysmal bone cysts (ABCs) are rare, highly vascular osteolytic bone lesions that predominantly affect pediatric populations. This report evaluates the clinicopathological data of pediatric patients with spinal ABCs. The medical records for all patients at Children’s Hospital Los Angeles with biopsy-proven ABCs of the spine between 1998 and 2018 were evaluated.

OBSERVATIONS

Seventeen patients, 6 males and 11 females, were identified. The mean age at surgery was 10.4 years (range, 3.5–20 years). The most common presenting complaint was pain at the lesion site 16/17 (94%), followed by lower-extremity weakness 8/17 (47%). Resection and intralesional curettage were performed in all patients. Three (18%) of 17 patients underwent selective arterial embolization prior to resection. Spinal stability was compromised in 15 of 17 patients (88%), requiring instrumented fusion. Five (29%) of the 17 patients received additional therapy including radiation, calcitonin-methylprednisolone, or phenol. Four (23.5%) of 17 patients experienced a recurrence, and the mean time to recurrence was 15 months. The postoperative follow-up ranged from 6 to 108 months (median, 28 months). Reoperation occurred after an average of 35 months. At the recent follow-up, patients were free of disease.

LESSONS

Gross-total resection by intralesional curettage with case-dependent instrumented spinal fusion for instability remains an effective strategy for managing pediatric spinal ABCs. Long-term follow-up is necessary to detect tumor recurrence.

Open access

Arteriovenous malformation with unique drainage through the emissary vein of the foramen ovale: illustrative case

Xiaochun Zhao, Alexander R Evans, Ali Tayebi Meybodi, Nicholas Hopkins, Ira Bowen, Shyian S Jen, Mark C Preul, and Karl Balsara

BACKGROUND

As part of the laterotrigeminal venous system (LTVS), the emissary vein of the foramen ovale (EVFO) is an underrecognized venous structure communicating between the cavernous sinus and pterygoid plexus. The sphenobasal sinus is an anatomical variation of the sphenoparietal sinus that drains directly into the EVFO. The authors present the case of a ruptured arteriovenous malformation (AVM) with a unique drainage pattern through the sphenobasal sinus and EVFO.

OBSERVATIONS

A 9-year-old female initially presented with loss of consciousness and was subsequently found to have a ruptured AVM in the left basal frontal area. She underwent an immediate decompressive hemicraniectomy, with a computed tomography angiogram demonstrating a unique anatomical variation in which the sphenobasal sinus communicated with the EVFO and LTVS. The final venous drainage returned to the pterygoid plexus and external jugular vein. Postoperatively, the patient made a substantial recovery, with generalized right-sided weakness remaining as the sole deficit.

LESSONS

The authors present the case of a ruptured AVM with unique venous drainage into the sphenobasal sinus and EVFO, for which the current literature remains limited. As exemplified by this illustrative case, technique modification may be warranted in the setting of this unique anatomical variation to avoid venous sinus injury.

Open access

Continuous direct intraarterial treatment of meningitis-induced vasospasm in a pediatric patient: illustrative case

Aubrey C Rogers, Aditya D Goyal, and Alexandra R Paul

BACKGROUND

Bacterial meningitis–induced ischemic stroke continues to cause significant long-term complications in pediatric patients. The authors present a case of severe right internal carotid artery terminus and M1 segment vasospasm in a 9-year-old with an infected cholesteatoma, which was refractory to multiple intraarterial treatments with verapamil and milrinone. This is the first report of continuous intraarterial antispasmodic treatment in a pediatric patient as well as the first report of continuous treatment in an awake and extubated patient.

OBSERVATIONS

Arterial narrowing was successfully treated by continuous direct intraarterial administration of both a calcium channel blocker (verapamil) and a phosphodiesterase-3 inhibitor (milrinone). The patient recovered remarkably well and was discharged home with no neurological deficit (National Institutes of Health Stroke Scale score 0) and ambulatory without assistance after 22 days. The authors report a promising outcome of this technique performed in a pediatric patient.

LESSONS

This represents a novel treatment option for the prevention of stroke in pediatric bacterial meningitis. Continuous, direct intraarterial administration of antispasmodic medications can successfully prevent long-term neurological deficit in pediatric meningitis-associated vasospasm. The described method has the potential to significantly improve outcomes in severe pediatric meningitis-associated vasospasm.

Open access

Overcoming the challenge of a thin skull in a 2-year-old patient undergoing laser interstitial thermal therapy using an individualized stereotactic platform: illustrative case

Spencer Lau, Joseph Menousek, Tyler Pistone, Arnett Klugh III, and Afshin Salehi

BACKGROUND

Ependymoma is the third most common pediatric brain tumor that can present with headaches, cranial nerve deficits, nausea, vomiting, and ataxia. Current treatment is maximal safe resection followed by radiation therapy. More recently, laser interstitial thermal therapy (LITT) has become an alternative to traditional resection. In this report, the authors describe the utilization of a single-use, patient-specific stereotactic platform for the treatment of supratentorial ependymoma with LITT.

OBSERVATIONS

A 2-year-old female had a complex history of supratentorial ependymoma after multiple craniotomies for repeated tumor progression and ventriculoperitoneal shunt placement. Imaging demonstrated an enlarging, complex, enhancing mass in the right occipital region. LITT was decided on for treatment. Given the thinness of the patient’s skull, which precluded traditional means of stereotaxy, the authors elected to use a personalized stereotactic platform. Immediate postoperative imaging captured complete laser ablation of the tumor, with long-term imaging demonstrating a decreased tumor size.

LESSONS

Individualized stereotactic platforms are increasingly used in adult populations, but pediatric use continues to be infrequent. In this report, the authors present the youngest reported case using a personalized stereotactic platform and show the effectiveness of this system for performing LITT in the youngest of populations with very thin skulls.

Open access

Chronic subdural hematoma associated with type II and type III Galassi arachnoid cysts: illustrative cases

Bac Thanh Nguyen, Van Dinh Tran, Jehan Bista, and Trung Van Trinh

BACKGROUND

Arachnoid cysts (ACs) are congenital abnormalities that can be located anywhere within the subarachnoid space along the cerebrospinal axis, although they are most often found on the left side in the temporal fossa and sylvian fissure. ACs comprise approximately 1% of all intracranial space-occupying lesions and are considered potential risk factors for subdural hematoma (SDH) in individuals of all age groups who have experienced traumatic brain injury. Although it is uncommon for an intracystic hemorrhage of an AC to occur without evidence of head trauma, it may be more common among children and young adults. Here, the authors present three cases of spontaneous AC intracystic hemorrhage with chronic SDH. Additionally, they provide a thorough review of the existing literature.

OBSERVATIONS

All three patients with AC were adolescent males. In all cases, AC was identified using the Galassi classification (type II or III) and associated with spontaneous intracystic hemorrhage and chronic SDH as seen on imaging.

LESSONS

Spontaneous intracystic hemorrhage is a rare complication and occurs most commonly on the left side. Surgery is the definitive treatment, requiring either craniotomy or burr hole for hematoma evacuation and microsurgical fenestration to drain the cyst into the subarachnoid cisterns.