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Open access

Adamantinomatous craniopharyngioma associated with a compromised blood–brain barrier: patient series

Eric W. Prince, Lindsey M. Hoffman, Trinka Vijmasi, Kathleen Dorris, Jennifer A. McWilliams, Kimberly R. Jordan, David M. Mirsky, and Todd C. Hankinson

BACKGROUND

Adamantinomatous craniopharyngioma (ACP) is a highly morbid adult and pediatric brain tumor derived from epithelial remnants of the craniopharyngeal canal (Rathke’s pouch), which gives rise to the anterior pituitary gland. Standard therapy includes maximal safe resection with or without radiation therapy. Systemic antitumor therapy remains elusive. Immune-related paracrine signaling involving the interleukin-6 receptor (IL-6R) may contribute to ACP pathogenesis. Tocilizumab, a recombinant humanized monoclonal antibody against IL-6R, is approved by the US Food and Drug Administration but does not cross an intact blood–brain barrier.

OBSERVATIONS

In a phase 0 trial design, a single dose of tocilizumab was delivered intravenously before clinically indicated surgical intervention in 3 children with ACP. The presence of tocilizumab was assayed in plasma, tumor tissue, tumor cyst fluid, and cerebrospinal fluid (n = 1) using a novel enzyme-linked immunosorbent assay. Tocilizumab reached ACP tumor tissue and/or cyst fluid after one systemic dose in every patient.

LESSONS

This finding helps explain extant data that indicate tocilizumab may contribute to ACP therapy. It further indicates that ACP does not reside behind an intact blood–brain barrier, dramatically broadening the range of potential antitumor therapies against this tumor. This has substantial implications for the design of future clinical trials for novel therapies against ACP in both children and adults.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Delayed-onset white cord syndrome after anterior and posterior cervical decompression surgery for symptomatic ossification of spinal ligaments: illustrative cases

Ranjit D. Singh, Mark P. Arts, and Godard C. W. de Ruiter

BACKGROUND

White cord syndrome is an extremely rare complication of cervical decompressive surgery, characterized by serious postoperative neurological deficits in the absence of apparent surgical complications. It is named after the characteristic ischemic-edematous intramedullary T2-hyperintense signal on postoperative magnetic resonance imaging and is believed to be caused by ischemic-reperfusion injury. Neurological deficits typically manifest immediately after surgery, and delayed occurrence has been reported only once.

OBSERVATIONS

The authors presented two cases of delayed white cord syndrome after anterior and posterior cervical decompression surgery for symptomatic ossification of the posterior longitudinal ligament and ligamentum flavum, respectively. Neurological deficits manifested on postoperative day 2 (case 1) and day 8 (case 2). The patients’ conditions were managed with high-dose corticosteroids, mean arterial pressure augmentation, and early physical therapy, after which they showed partial neurological recovery at discharge, which improved further by the 3-month follow-up visit.

LESSONS

The authors’ aim was to raise awareness among spine surgeons about this rare but severe complication of cervical decompressive surgery and to emphasize the mainstays of treatment based on current best evidence: high-dose corticosteroids, mean arterial pressure augmentation, and early physical therapy.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Endovascular treatment of a ruptured aneurysm arising from the proximal end of a partial vertebrobasilar duplication with a contralateral prominent persistent primitive hypoglossal artery: illustrative case

Nobuyuki Genkai, Kouichirou Okamoto, Toshiharu Nomura, and Hiroshi Abe

BACKGROUND

Ruptured aneurysms associated with a partial vertebrobasilar duplication or a persistent primitive hypoglossal artery (PPHA) have been reported. Only rarely has endovascular treatment of ruptured aneurysms in association with both vascular variations been reported.

OBSERVATIONS

A 66-year-old woman experienced the sudden onset of a severe headache caused by a subarachnoid hemorrhage. Cerebral angiograms demonstrated a prominent PPHA originating from the left internal carotid artery at the C2 vertebral level and a partial vertebrobasilar duplication between the hypoplastic right vertebral artery and proximal basilar artery with a small aneurysm at the proximal end of the duplication from where the anterior spinal artery originated. The left vertebral artery was aplastic. A microcatheter was introduced into the aneurysm via the PPHA under the control of high blood flow, using a balloon-assisted technique. The aneurysm was completely obliterated with a coil. Although small cerebellar and cerebral infarcts developed during the procedure, the patient was discharged without neurological symptoms.

LESSONS

To avoid serious neurological complications, precise analysis of the complex vascular anatomy, including the anterior spinal artery and hemodynamics, is clinically important for endovascular therapy of cerebral aneurysms in patients with an association between a partial vertebrobasilar duplication and a PPHA.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Iatrogenic neurological injury after radiofrequency ablation and epidural steroid injections: illustrative cases

Lisa B. E. Shields, Vasudeva G. Iyer, Yi Ping Zhang, and Christopher B. Shields

BACKGROUND

Neck pain is often chronic and disabling. Cervical facet joint injections and epidural steroid injections are frequently used to manage chronic neck pain and cervicogenic headaches. While minimal side effects are commonly associated with these treatments, severe complications are exceedingly rare.

OBSERVATIONS

The authors report 4 cases of iatrogenic neurological injury after radiofrequency ablation (RFA) and epidural steroid injections. One patient experienced left shoulder, scapular, and arm pain with left arm and hand weakness that developed immediately after RFA for chronic neck pain. Electromyography/nerve conduction velocity (EMG/NCV) studies confirmed denervation changes in the left C8–T1 distribution. Three patients complained of numbness and weakness of the hands immediately after an interlaminar cervical epidural block. One of these patients underwent EMG/NCV that confirmed denervation changes occurring in the left C8–T1 distribution.

LESSONS

Spine surgeons and pain management specialists should be aware of neurological injuries that may occur after cervical RFA and epidural steroid injections, especially after a multilevel cervical procedure and with severe cervical spinal stenosis. EMG/NCV studies plays an important role in detecting and localizing neurological injury and in differentiating from conditions that mimic cervical root injuries, including brachial plexus trauma due to positioning and Parsonage-Turner syndrome.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Paroxysmal otalgia treated with microvascular decompression of the intermediate nerve: illustrative case

Leonie Witters, Anton Lukes, and Tomas Menovsky

BACKGROUND

Intermediate nerve neuralgia is a rare type of cranial neuralgia that causes clinical, therapeutic, and diagnostic challenges. Studies have described pharmacological and surgical treatment options. Surgical treatment ranges from sectioning of neural structures to microvascular decompression. Given the rareness of the disease, there are no clear recommendations concerning treatment.

OBSERVATIONS

Reported is the case of a patient with typical intermediate nerve neuralgia. In this particular case, decision-making toward surgical decompression in an earlier stage of the disease could have been beneficial. The authors found excellent results using only microvascular decompression without sectioning of neural structures.

LESSONS

Knowledge of intermediate nerve anatomy is essential to understand this complex pain syndrome. This case illustrates that surgery should not only be regarded as a last resort in case of failure of conservative treatment but also should be considered early in the disease course, especially in the case of a clear neurovascular conflict. When no evident cause is found, surgery could be considered as an exploratory option to depict a neurovascular conflict intraoperatively. Microvascular decompression of the intermediate nerve without sectioning of neural structures can obtain excellent results. Since neural structures are saved, postoperative sequelae can be limited.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

A retro-odontoid pseudotumor treated with fixation and tumor resection by the lateral approach: illustrative case

Yoshiaki Oda, Takamitsu Tokioka, and Toshifumi Ozaki

BACKGROUND

A retro-odontoid pseudotumor is not a condition that requires resection. However, pathological diagnosis is required when a tumor such as a meningeal tumor or chordoma is suspected. The authors report a case of a large lesion treated with posterior fixation and tumor resection using a lateral approach.

OBSERVATIONS

A 77-year-old man visited the authors’ department complaining of neck pain and decreased dexterity of the upper extremities. Magnetic resonance imaging showed a large, beak-shaped lesion behind the dens and severe compression of the spinal cord. Surgery consisted of occipitocervical–C2 fixation, followed by tumor resection with a left lateral approach. The pathological diagnosis was consistent with a retro-odontoid pseudotumor. The tumor was resected to a relatively large extent and shrank over time, leading to complete disappearance.

LESSONS

Pathological examination is also possible with a posterior approach if the tumor can be reached through the lateral edge of the dura. In that situation, the amount of resection is limited, and there is a risk of spinal cord compression. Intradural dissemination of tumors is a concern with the transdural approach. If tumor resection by the posterior approach is difficult, the lateral approach can facilitate tumor resection.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Right hemidiaphragmatic paralysis after cervical transforaminal epidural steroid injection: illustrative case

Molly Farrell, Ezek Mathew, Martin Weiss, and Rob Dickerman

BACKGROUND

Cervical radiculopathy is a common cause of neck pain, with radiation into the upper extremity in a dermatomal pattern. Corticosteroid injection is a conservative management option with a low risk of major adverse events. No reviewed literature or case reports have implicated phrenic nerve injury secondary to cervical transforaminal epidural steroid injection (CTFESI).

OBSERVATIONS

A 45-year-old man with severe right C6 radiculopathy secondary to a large right-sided C5–6 herniated intervertebral disc presented to the pain management clinic, where he received a right-sided C6 CTFESI. An hour after injection, the patient experienced shortness of breath, which was found to be caused by right diaphragmatic paralysis. The patient underwent a C5–6 anterior cervical discectomy and fusion, which provided complete relief of his radicular symptoms. However, the right hemidiaphragmatic paralysis remained at the 1-year postoperative visit.

LESSONS

Thorough literature review showed no established explanations for phrenic nerve injury after CTFESI. In this study, the authors explored the suspected mechanisms of possible injury to the phrenic nerve. Epidural corticosteroid injection is considered to be a safe option for conservative management of cervical radiculopathy. This study unveiled a unique and important adverse event that should be considered before a patient receives CTFESI.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Side-to-side reverse superficial temporal artery to M4 middle cerebral artery bypass for common carotid artery occlusion with bonnet collaterals: illustrative case

Lekhaj C. Daggubati, Varun Padmanaban, and Ephraim W. Church

BACKGROUND

The bonnet bypass was initially described for common carotid artery occlusion. Considered a second-generation bypass, it augments intracranial perfusion with contralateral external carotid artery flow through an interposition graft running over the scalp vertex. However, the traditional first-generation low-flow superficial temporal artery (STA)-M4 middle cerebral artery (MCA) bypass may be enhanced by performing a side-to-side (S-S) bypass with an intraluminal suture technique (fourth-generation bypass) to increase perfusion through antegrade and retrograde flow.

OBSERVATIONS

The authors present a reimagined S-S STA-M4 bypass in the case of a patient with symptomatic common carotid occlusion, in which the ipsilateral STA filled in a reverse fashion from the contralateral external carotid branches over the scalp vertex (bonnet collaterals). By performing an S-S anastomosis, the authors were able to improve cerebral perfusion and avoid the multiple anastomosis sites of the bonnet bypass.

LESSONS

The patient had a good recovery with resolution of his preoperative symptoms. Follow-up angiography showed a patent bypass supplying the MCA territory through retrograde flow in the frontal and parietal limbs of the STA, converging at the anastomosis site. In this report, the authors present a new fourth-generation bypass dubbed the “S-S reverse STA-M4 MCA bypass.”

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Spontaneous regression and near disappearance of a calcified herniated thoracic disc in a 44-year-old male: illustrative case

Andrew Utter, David Cavanaugh, Kelly F. Van Schouwen, Madison Mascagni, Madeline Walker, and Marcus Stone

BACKGROUND

Herniation of an intervertebral disc (IVD) is found predominantly in the lumbar and cervical spine of both children and adults, but herniated IVDs of the thoracic spine are a rare occurrence. However, approximately 40% of herniated thoracic disc cases are calcified. Approximately 0.65% of all spinal herniations are calcified herniated thoracic discs (CHTDs). CHTDs can be treated conservatively or invasively, depending on the symptoms and degree of neurological deficit present.

OBSERVATIONS

The authors report a 44-year-old male with near complete reabsorption and disappearance of a CHTD. A review of the available literature indicates that there are only seven adult patients in whom this phenomenon has been reported.

LESSONS

Determining the best form of invasive treatment is a challenge for surgeons given the complexity of this condition. While the disappearance of calcified herniated discs of the lumbar and cervical spine has been reported, reports of the regression of CHTDs are rare. The disappearance of CHTDs is more commonly reported in children who undergo conservative treatment, while surgery is reserved for children who experience progressive pain and neurological deficit. Given the success of conservative treatment of CHTDs in children, conservative treatment methods should be considered when treating mildly symptomatic adults.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)

Open access

Three decades of progress from surgery to medical therapy for isolated neuroaxis BRAF V600E–positive Langerhans cell histiocytosis management: illustrative case

Nallammai Muthiah, Kamil W. Nowicki, Jennifer L. Picarsic, Michael P. D’Angelo, Daniel F. Marker, Edward G. Andrews, Edward A. Monaco III, and Ajay Niranjan

BACKGROUND

“Langerhans cell histiocytosis” (LCH) is a term that encompasses single-system or multisystem disorders traditionally characterized by a proliferation of clonal CD1a+/CD207+ myeloid-derived histiocytes. In most cases of LCH, mitogen-activated protein kinase (MAPK) pathway somatic mutations lead to near universal upregulation of phosphorylated extracellular signal-regulated kinase expression. The clinical manifestations of LCH are numerous, but bone involvement is common. Intracranial lesions, especially as isolated manifestations, are rare.

OBSERVATIONS

The authors presented the case of a long-term survivor of exclusive intracranial LCH that manifested with isolated craniofacial bone and intraparenchymal central nervous system recurrences, which were managed with 3 decades of multimodal therapy. The patient was initially diagnosed with LCH at age 2 years, and the authors documented the manifestations of disease and treatment for 36 years. Most of the patient’s treatment course occurred before the discovery of BRAF V600E. Treatments initially consisted of chemotherapy, radiosurgery, and open resections for granulomatous LCH lesions. Into young adulthood, the patient had a minimal disease burden but still required additional radiosurgical procedures and open resections.

LESSONS

Surgical treatments alleviated the patient’s immediate symptoms and allowed for tumor burden control. However, surgical interventions did not cure the underlying, aggressive disease. In the current era, access to systemic MAPK inhibitor therapy for histiocytic lesions may offer improved outcomes.

In Journal of Neurosurgery: Case Lessons Volume 1 (2021): Issue 19 (May 2021)