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Open access

Acute worsening of CADASIL in a patient with COVID-19 infection: illustrative case

Jared S. Rosenblum, Jessa M. Tunacao, Matthew A. Nazari, Halle Ronk, Danielle D. Dang, Chad Downing, Zhengping Zhuang, John D. Heiss, James G. Smirniotopoulos, Avraham Bluestone, James Badia, and Joseph White

BACKGROUND

Reports of cerebrovascular ischemia and stroke occurring as predominant neurological sequelae of severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection, which causes coronavirus disease 2019 (COVID-19), are increasingly evident within the literature. While various pathophysiological mechanisms have been postulated, including hypercoagulability, endothelial invasion, and systemic inflammation, discrete mechanisms for viral neurotropism remain unclear and controversial.

OBSERVATIONS

The authors present a unique case study of a 64-year-old male with acute COVID-19 infection and acute worsening of previously stable cerebral autosomal dominant arteriopathy with subcortical infarcts and leukoencephalopathy (CADASIL), a rare heritable arteriopathy due to mutation in the Notch3 gene, which is critical for vascular development and tone. Delayed cranial neuropathies, brainstem fluid-attenuated inversion recovery signal, and enhancement of olfactory and vagus nerves on magnetic resonance neurography in this patient further support viral neurotropism via cranial nerves in addition to cerebral vasculature.

LESSONS

To the authors’ knowledge, this is the first case in the literature that not only demonstrates the consequences of COVID-19 infection in a patient with altered cerebrovascular autoregulation such as CADASIL but also highlights the tropism of SARS-CoV-2 for (1) cranial nerves as a mode of entry to the central nervous system and (2) vessels as a cause of cerebrovascular ischemia.

Open access

Pituitary macroadenoma causing vision loss in Wyburn-Mason syndrome: illustrative case

Nicholas F. Hug, David A. Purger, Heather E. Moss, and Robert L. Dodd

BACKGROUND

Wyburn-Mason syndrome (WMS) is a neurocutaneous disorder consisting of vascular malformations of the brain, eye, and skin. These include characteristically high-flow intracranial and intraorbital arteriovenous malformations (AVMs) that present commonly with visual deterioration, headache, and hemiplegia. Complete removal of these lesions is challenging. Most patients are followed closely, and intervention occurs only in the setting of worsening symptoms secondary to AVM growth or hemorrhage. Here the authors present the first known case of a patient with WMS and a pituitary macroadenoma.

OBSERVATIONS

A 62-year-old man with a 30-year history of WMS with right basal ganglia and orbital AVMs and right eye blindness presented for new-onset left-sided vision loss. A pituitary adenoma was identified compressing the optic chiasm and left optic nerve. Magnetic resonance imaging and digital subtraction angiography studies were obtained for surgical planning, and the patient underwent an endoscopic transnasal transsphenoidal resection, with significant postoperative vision improvement.

LESSONS

Given the variable presentation and poor characterization of this rare syndrome, patients with WMS presenting with new symptoms must undergo evaluation for growth and hemorrhage of known AVMs, as well as new lesions. Further, in patients undergoing intracranial surgery, extensive preoperative imaging and planning are crucial for safe and successful procedures.

Open access

Skull base chondroblastoma with aneurysmal bone cyst–like changes treated with percutaneous radiofrequency ablation and doxycycline sclerotherapy: illustrative case

Madeline I. Foo, Kathleen Nicol, and James W. Murakami

BACKGROUND

Chondroblastomas (CBs) are rare benign bone tumors that are often difficult to treat because of their locations. CBs can be even more challenging to successfully manage when they present alongside aneurysmal bone cyst (ABC)-like changes. To minimize operative morbidity, especially in hard-to-reach lesions, percutaneous approaches for both lesions have been individually described. We present a skull base CB with associated ABC-like changes treated by combining two different previously described percutaneous modalities.

OBSERVATIONS

The authors report successful percutaneous treatment of a skull base CB with adjacent ABC-like changes in a 17-year-old male. The CB was treated with radiofrequency ablation (RFA) and the adjacent ABC area with doxycycline sclerotherapy. After 3 years of follow-up, there has been no clinical or radiological evidence of recurrence.

LESSONS

CBs occur in the skull base and, as elsewhere in the body, can be associated with ABC-like changes. Successful percutaneous treatment of such a CB with ABC-like changes is possible by combining previously described techniques of RFA and doxycycline sclerotherapy.

Open access

Bilateral blindness following flow diverter placement in a giant thrombosed cavernous carotid aneurysm: illustrative case

Basant K. Misra, Alay V. Khandhar, and Reema Pradhan

BACKGROUND

The best treatment option for giant intracranial aneurysms (GIAs) is still debated. The authors report a case of a giant thrombosed cavernous carotid artery (CCA) aneurysm for which two sessions of flow diverter (FD) placement failed, leading to bilateral blindness.

OBSERVATIONS

A 66-year-old man presented to an outside center with a history of rapid-onset right-sided retro-orbital pain, visual deterioration, and restricted eye movements associated with headache, vomiting, and diminished sensations on the right side of the face. He was diagnosed with an unruptured thrombosed giant CCA aneurysm and was treated twice with unsuccessful FDs. At follow-up, he developed blindness in both eyes. After 1.5 years, he presented to the authors’ institution with headache, vomiting, and epistaxis, for which he underwent high-flow external carotid artery–M2 segment of the middle cerebral artery bypass grafting using the radial artery as a conduit and ligation of the internal carotid artery in the neck without any added neurological deficits.

LESSONS

This dreadful complication of bilateral blindness after being treated with repeated unsuccessful FDs has not been reported in the literature. It could have been avoided if microsurgery had been the primary modality of treatment.

Open access

Bow hunter’s syndrome: temporary obstruction of blood flow in the affected vertebral artery during posterior occipitocervical fusion. Illustrative case

Takeru Yokota, Koji Otani, Junichi Handa, Takuya Nikaido, Takao Kojima, Naoki Sato, and Shinichi Konno

BACKGROUND

Bow hunter’s syndrome (BHS) is a rare condition characterized by mechanical impingement of a vertebral artery (VA) during neck rotation followed by vertebrobasilar insufficiency. Posterior fusion is a typical surgical method for BHS.

OBSERVATIONS

The case of a 70-year-old Japanese man who presented with presyncope that occurred during right cervical rotation is reported. Given the possibility of vertebrobasilar insufficiency, digital subtraction angiography and computed tomography angiography were performed and showed a hypoplastic right VA and severe stenosis of the left VA over the occiput (O)–C2 level. The blood flow of the left VA was interrupted by right cervical rotation, with resumption of blood flow on left cervical rotation. BHS was diagnosed, and posterior fusion at the O–C2 level was performed. Immediately after implant fixation, selective arteriography confirmed disruption of blood flow in the left VA. The rods were removed immediately; resumption of blood flow was confirmed; and the rods were refixed, anatomically bent with slight left cervical rotation. Then, sustained blood flow in the left VA was confirmed.

LESSONS

Posterior fixation for BHS can induce VA occlusion due to minor changes in cervical spine alignment. Intraoperative selective arteriography is a necessary tool to identify occlusion of the affected VA.

Open access

A complete thrombotic large aneurysm at the nonbranching segment of the distal anterior cerebral artery mimicking a cavernous malformation: illustrative case

Yu Hirokawa, Yuhei Michiwaki, Tatsuya Tanaka, Masatou Kawashima, Tomihiro Wakamiya, Ryohei Sashida, Kazuaki Shimoji, Eiichi Suehiro, Keisuke Onoda, Fumitaka Yamane, and Akira Matsuno

BACKGROUND

Thrombotic aneurysms at the nonbranching segment of the distal anterior cerebral artery (ACA) are extremely rare and difficult to differentiate from cavernous malformations by radiographic features alone.

OBSERVATIONS

Computed tomography and magnetic resonance imaging of a 30-year-old female patient with a chronic headache complaint revealed a 22-mm frontal lobe mass. The mass showed heterogeneous mixed intensity and hemosiderin deposits on magnetic resonance images. It was not visualized by conventional angiography, indicating that the mass and ACA/other vessels were not connected. The patient was preoperatively diagnosed with a cavernous malformation. However, during resection, the mass surface was white and smooth, different from a cavernous malformation. Although the mass was adherent to the pericallosal artery branch, no luminal continuity was observed. After detachment, the mass was completely resected. Pathological and immunohistochemical findings indicated a vessel wall and interior thrombus. The patient was rediagnosed with a thrombotic aneurysm at the distal ACA nonbranching segment and discharged 10 days postsurgery without neurological deficits.

LESSONS

Because radiographic findings of thrombotic aneurysm and cavernous malformation are similar, mass lesions in contact with major arteries should be differentiated as thrombotic aneurysms, even when the artery lumen appears disconnected from the mass.

Open access

A deceptive mass on neonatal ultrasound and a fetal brain MRI-confirmed complex dural arteriovenous fistula postnatally: illustrative case

Elliot T. Varney, Charlotte S. Taylor, Allen G. Crosthwait, Kristin Weaver, and Todd Nichols

BACKGROUND

Dural arteriovenous fistulas (dAVFs) are direct, aberrant connections between dural arteries and cerebral veins. In neonates, delayed diagnosis results in grim outcomes. Treatment involves endovascular management because of its success and tolerability. Here, the authors present a case of a complex dAVF initially recognized with an in utero neurosonogram and fetal magnetic resonance imaging (MRI).

OBSERVATIONS

A 21-week fetal ultrasound of a nonspecific brain mass was confirmed with fetal MRI as a 2.7-cm T1-hyperintense posterior fossa mass. Although a large flow void in the left middle cranial fossa was present, postnatal computed tomography angiography ultimately revealed a high-flow dAVF communicating with the left transverse sinus. In the early postnatal period, the patient developed hydrocephalus. After successful partial embolization, 6-week postangiogram brain MRI indicated disease progression with the development of a venous varix causing brainstem compression. Repeat embolization resulted in complete cessation of early venous drainage.

LESSONS

Neonatal dAVFs are exceedingly rare and result in futile outcomes; however, detection in utero is possible. Although definitive therapy must be performed postnatally, constant monitoring and early delivery can prevent complications. Attention to fetal ultrasound is essential, and knowledge of fetal MRI in the detection of these complex lesions can significantly improve outcomes.

Open access

Spontaneous obliteration of brain arteriovenous malformations: illustrative cases

Kevin Agyemang, Anna Rose, Olatomiwa Olukoya, Jennifer Brown, and Edward Jerome St George

BACKGROUND

Spontaneous angiographic obliteration of a brain arteriovenous malformation (AVM) is considered a rare outcome, with most cases in the literature related to prior hemorrhage in small brain AVMs. The authors present a prospective, single center, consecutive case series. The clinical course and radiographic features of four cases with spontaneous obliteration of brain AVM were analyzed.

OBSERVATIONS

The median age of patients in this series was 47.6 years, with an equal gender split. The median maximum brain AVM diameter was 2 cm. The median time to spontaneous obliteration was 26 months, with hemorrhage preceding this in three out of four cases and a prolonged latency in the only case with a nidus size larger than 3 cm and no hemorrhage.

LESSONS

The present study provides additional information to allow clinicians to counsel patients about the rare outcomes of conservative management. This work extends our understanding of when this phenomenon can occur by reporting on the differences associated with spontaneous obliteration of larger AVMs.

Open access

Dural arteriovenous fistulas misdiagnosed as intracranial neoplasms: illustrative case

Tobias Rossmann, Michael Veldeman, Ville Nurminen, Rahul Raj, and Mika Niemelä

BACKGROUND

Dural arteriovenous fistulas (dAVF) may induce imaging findings attributable to various disease entities including malignant neoplasms. In these cases, diagnosis and adequate treatment are often delayed and patients may be exposed to spurious treatments in addition to the risks inherent to an untreated dAVF with cortical venous drainage.

OBSERVATIONS

The authors report a case of a patient referred for surgical treatment of a supratentorial high-grade glioma. Thorough review of imaging data challenged the initial radiological diagnosis and led to proper angiographic workup. As a result, a high-grade dAVF was confirmed and successfully embolized. In addition to this case, we provide an extensive literature review on dAVF initially diagnosed as cerebral neoplasms, including clinical, imaging and follow-up data.

LESSONS

The literature provides diagnostic criteria for dAVF on magnetic resonance imaging; however, those criteria may be only partly applicable in many cases. Misdiagnosis of a neoplasm due to dAVF has been reported but remains rare, especially in supratentorial lesions. Digital subtraction angiography should be pursued to rule out an underlying vascular pathology if any doubt. This may prevent unnecessary interventions such as biopsies, pharmacological treatment and a delay in dAVF treatment, given its associated risk of hemorrhage and nonhemorrhagic neurological deficits.

Open access

High-resolution micro-Doppler imaging during neurosurgical resection of an arteriovenous malformation: illustrative case

Sadaf Soloukey, Luuk Verhoef, Pieter Jan van Doormaal, Bastian S. Generowicz, Clemens M. F. Dirven, Chris I. De Zeeuw, Sebastiaan K. E. Koekkoek, Pieter Kruizinga, Arnaud J. P. E. Vincent, and Joost W. Schouten

OBJECTIVE

Given the high-risk nature of arteriovenous malformation (AVM) resections, accurate pre- and intraoperative imaging of the vascular morphology is a crucial component that may contribute to successful surgical results. Surprisingly, current gold standard imaging techniques for surgical guidance of AVM resections are mostly preoperative, lacking the necessary flexibility to cater to intraoperative changes. Micro-Doppler imaging is a unique high-resolution technique relying on high frame rate ultrasound and subsequent Doppler processing of microvascular hemodynamics. In this paper the authors report the first application of intraoperative, coregistered magnetic resonance/computed tomograpy, micro-Doppler imaging during the neurosurgical resection of an AVM in the parietal lobe.

OBSERVATIONS

The authors applied intraoperative two-dimensional and three-dimensional (3D) micro-Doppler imaging during resection and were able to identify key anatomical features including draining veins, supplying arteries and microvasculature in the nidus itself. Compared to the corresponding preoperative 3D-digital subtraction angiography (DSA) image, the micro-Doppler images could delineate vascular structures and visualize hemodynamics with higher, submillimeter scale detail, even at significant depths (>5 cm). Additionally, micro-Doppler imaging revealed unique microvascular morphology of surrounding healthy vasculature.

LESSONS

The authors conclude that micro-Doppler imaging in its current form has clear potential as an intraoperative counterpart to preoperative contrast-dependent DSA, and the microvascular details it provides could build new ground to further study cerebrovascular pathophysiology.