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Open access

Ondine’s curse: clinical presentation with diaphragmatic pacing and spontaneous respiratory recovery. Illustrative case

Alexander J. Schupper, Alex Devarajan, Dong-Seok Lee, Enrique Perez, and Raj K. Shrivastava

BACKGROUND

The complexity of posterior fossa surgery can often lead to rare complications due to the anatomy involved. Vestibular schwannoma resection is a common pathology in the posterior fossa, often requiring surgical intervention. Given the proximity of this space to the brainstem, cranial nerve VII/VIII complex, and posterior inferior cerebellar artery (PICA), neurovascular complications are not infrequent. A rare vascular complication from this surgical approach is a lateral medullary infarction from injury to the lateral medullary segment of the proximal PICA, leading to central hypoventilation syndrome (CHS).

OBSERVATIONS

This report presents a unique case of a 51-year-old man who underwent a retrosigmoid craniectomy for resection of a vestibular schwannoma. Following surgery, the patient was unable to be weaned off the ventilator and was noted to become apneic while he slept, a clinical picture consistent with Ondine’s curse.

LESSONS

This report discusses the anatomical considerations of this surgical corridor leading to this complication and the management of a patient with acquired Ondine’s curse and reviews the scarce literature on this uncommon cause of acquired CHS.

In Journal of Neurosurgery: Case Lessons Volume 5 (2023): Issue 21 (May 2023)

Open access

Successful treatment with urgent revascularization and parent artery occlusion for a ruptured intratumoral aneurysm following prior meningioma surgery: illustrative case

Tomohiro Okuyama, Kota Kurisu, Masaaki Hokari, Kei Miyata, Kazuki Uchida, Katsuyuki Asaoka, Koji Itamoto, and Miki Fujimura

BACKGROUND

An intratumoral aneurysm encased within the associated intracranial tumor is rare, and hemorrhage caused by its rupture is even more rare. While urgent and adequate surgical treatment is important, the treatment can be difficult given the limited understanding of this rare condition.

OBSERVATIONS

A 69-year-old man who had undergone meningioma surgery 30 years prior presented with a disturbance in consciousness. Magnetic resonance imaging revealed massive intracerebral and subarachnoid hemorrhage. A round, partially calcified mass, which was diagnosed as recurrent meningioma, was also observed. Subsequent cerebral angiography revealed that the source of the hemorrhage was an intratumoral aneurysm in the dorsal internal carotid artery (ICA) encased within the recurrent meningioma. Urgent surgical ICA trapping and high-flow graft bypass were conducted. The postoperative course was uneventful, and he was referred to another hospital for rehabilitation.

LESSONS

This is the first case report of a ruptured intratumoral aneurysm being treated with urgent combined revascularization and parent artery trapping surgery. This surgical approach may be a feasible treatment option for such a challenging condition. Additionally, this case highlights the importance of diligent long-term follow-up after skull-base surgery, as minor intraoperative vascular wall injury may trigger the development and rupture of an intracerebral aneurysm.

In Journal of Neurosurgery: Case Lessons Volume 5 (2023): Issue 21 (May 2023)

Open access

Trigeminal neuralgia secondary to vascular compression and neurocysticercosis: illustrative case

Mao Vásquez, Luis J. Saavedra, Hector H. García, Evelyn Vela, Jorge E. Medina, Miguel Lozano, Carlos Hoyos, and William W. Lines-Aguilar

BACKGROUND

Trigeminal neuralgia (TN) is a frequent neurosurgical problem negatively influencing the quality of life of patients. The standard surgical treatment is microvascular decompression for primary cases and decompression of the mass effect, mainly tumors, for secondary cases. Neurocysticercosis (NCC) in the cerebellopontine angle is a rare etiology of TN. The authors report a case in which NCC cysts around the trigeminal nerve coexisted with a vascular loop, which compressed the exit of the trigeminal nerve from the pons.

OBSERVATIONS

A 78-year-old woman presented with a 3-year history of persistent severe pain in the left side of her face, refractory to medical treatment. On gadolinium-enhanced magnetic resonance imaging, cystic lesions were observed around the left trigeminal nerve and a vascular loop was also present and in contact with the nerve. A retrosigmoid approach for cyst excision plus microvascular decompression of the trigeminal nerve was successfully performed. There were no complications. The patient was discharged without facial pain.

LESSONS

Albeit rare, TN secondary to NCC cysts should be considered in the differential diagnosis in NCC-endemic regions. In this case, the cause of the neuralgia was probably both problems, because when both were treated, the patient improved.

In Journal of Neurosurgery: Case Lessons Volume 5 (2023): Issue 21 (May 2023)

Open access

Vascular anomaly, lipoma, and polymicrogyria associated with schizencephaly: developmental and diagnostic insights. Illustrative case

Kevin K. Kumar, Angus Toland, Nancy Fischbein, Martha Morrell, Jeremy J. Heit, Donald E. Born, and Gary K. Steinberg

BACKGROUND

Schizencephaly is an uncommon central nervous system malformation. Intracranial lipomas are also rare, accounting for approximately 0.1% of brain “tumors.” They are believed to be derived from a persistent meninx primitiva, a neural crest–derived mesenchyme that develops into the dura and leptomeninges.

OBSERVATIONS

The authors present a case of heterotopic adipose tissue and a nonshunting arterial vascular malformation arising within a schizencephalic cleft in a 22-year-old male. Imaging showed right frontal gray matter abnormality and an associated suspected arteriovenous malformation with evidence of hemorrhage. Brain magnetic resonance imaging revealed right frontal polymicrogyria lining an open-lip schizencephaly, periventricular heterotopic gray matter, fat within the schizencephalic cleft, and gradient echo hypointensity concerning for prior hemorrhage. Histological assessment demonstrated mature adipose tissue with large-bore, thick-walled, irregular arteries. Mural calcifications and subendothelial cushions suggesting nonlaminar blood flow were observed. There were no arterialized veins or direct transitions from the arteries to veins. Hemosiderin deposition was scant, and hemorrhage was not present. The final diagnosis was consistent with ectopic mature adipose tissue and arteries with meningocerebral cicatrix.

LESSONS

This example of a complex maldevelopment of derivatives of the meninx primitiva in association with cortical maldevelopment highlights the unique challenges from both a radiological and histological perspective during diagnostic workup.

In Journal of Neurosurgery: Case Lessons Volume 5 (2023): Issue 21 (May 2023)

Restricted access

Accuracy of robot-assisted stereotactic MRI–guided laser ablation in children with epilepsy

Keng Siang Lee, Kiran K. Seunarine, Nicola Barnes, M. Zubair Tahir, Sophia M. Varadkar, and Martin M. Tisdall

OBJECTIVE

Robot-assisted (RA) stereotactic MRI–guided laser ablation has been reported to be a safe and effective technique for the treatment of epileptogenic foci in children and adults. In this study the authors aimed to assess the accuracy of RA stereotactic MRI–guided laser fiber placement in children and to identify factors that might increase the risk of misplacement.

METHODS

A retrospective single-institution review of all children from 2019 to 2022 who underwent RA stereotactic MRI–guided laser ablation for epilepsy was undertaken. Placement error was calculated at the target by measuring the Euclidean distance between the implanted laser fiber position and the preoperatively planned position. Collected data included age at surgery, sex, pathology, date of robot calibration, number of catheters, entry position, entry angle, extracranial soft-tissue thickness, bone thickness, and intracranial catheter length. A systematic review of the literature was also performed using Ovid Medline, Ovid Embase, and the Cochrane Central Register of Controlled Trials.

RESULTS

In 28 children with epilepsy, the authors assessed 35 RA stereotactic MRI–guided laser ablation fiber placements. Twenty (71.4%) children had undergone ablation for hypothalamic hamartoma, 7 children (25.0%) for presumed insular focal cortical dysplasia, and 1 patient (3.6%) for periventricular nodular heterotopia. Nineteen children were male (67.9.%) and 9 were female (32.1%). The median age at the time of the procedure was 7.67 years (IQR 4.58–12.26 years). The median target point localization error (TPLE) was 1.27 mm (IQR 0.76–1.71 mm). The median offset error between the planned and actual trajectories was 1.04° (IQR 0.73°–1.46°). Patient age, sex, pathology and the time interval between date of surgery and robot calibration, entry position, entry angle, soft-tissue thickness, bone thickness, and intracranial length were not associated with the placement accuracy of the implanted laser fibers. However, the number of catheters placed did correlate with the offset angle error on univariate analysis (ρ = 0.387, p = 0.022). There were no immediate surgical complications. Meta-analysis indicated that the overall pooled mean TPLE was 1.46 mm (95% CI −0.58 to 3.49 mm).

CONCLUSIONS

RA stereotactic MRI–guided laser ablation for epilepsy in children is highly accurate. These data will aid surgical planning.

In Journal of Neurosurgery: Pediatrics Publish Before Print

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Association between urinary oxytocin secretion and natriuresis after transsphenoidal pituitary surgery

Paul E. Constanthin, Nathalie Isidor, Sophie De Seigneux, and Shahan Momjian

OBJECTIVE

Oxytocin (OXT) secretion has been shown to be abnormally elevated in patients who develop syndrome of inappropriate secretion of antidiuretic hormone (SIADH)–related hyponatremia after transsphenoidal pituitary surgery (TPS). While OXT was previously reported to increase natriuresis in the kidney, a potential role for this hormone in postoperative sodium balance and dysnatremias has not been studied. The objective of this study was to analyze the correlation between patients’ urinary output of OXT and natremia and natriuresis after TPS.

METHODS

The authors measured and correlated the urinary output of OXT with natriuresis and natremia in 20 consecutive patients who underwent TPS.

RESULTS

The ratio of urinary secretion of OXT between days 1 and 4 showed a strong, significant correlation with patient natriuresis at day 7 after pituitary surgery. Concomitantly, patient natremia showed a moderate, inverted correlation with OXT secretion in the urine.

CONCLUSIONS

Together, these results show for the first time that urinary OXT secretion correlates with patient natriuresis and natremia after pituitary surgery. This observation suggests a notable role for this hormone in sodium balance.

In Journal of Neurosurgery Publish Before Print

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Clinical outcomes of transforaminal lumbar interbody fusion using a modified posterior spinous process–splitting approach for lumbar degenerative or isthmic spondylolisthesis: a prospective cohort study

Guanyi Liu, Lihua Hu, Feng Shen, Yong Hu, and Weihu Ma

In the conventional posterior approach to the lumbar spine for transforaminal lumbar interbody fusion (TLIF), the paravertebral muscles are stripped from the spinous process. The authors developed a novel surgical procedure in which TLIF was performed via a modified spinous process–splitting (SPS) approach that enabled the preservation of the attachment of the paravertebral muscles to the spinous process. The SPS TLIF group comprised 52 patients with lumbar degenerative or isthmic spondylolisthesis who underwent surgery using a modified SPS TLIF technique, whereas the control group comprised 54 patients who underwent conventional TLIF. Compared with the control group, the SPS TLIF group had a significantly shorter operation time, smaller intra- and postoperative blood loss volumes, and shorter hospital stay and time to ambulation (p < 0.05). The SPS TLIF group had a lower mean visual analog scale score for back pain than the control group on both postoperative day 3 and at 2 years postoperatively (p < 0.05). Follow-up MRI showed changes in the paravertebral muscles in 46 of 54 patients (85%) in the control group and 5 of 52 patients (10%) in the SPS TLIF group (p < 0.001). This novel technique may be a useful alternative to the conventional posterior approach for TLIF.

In Journal of Neurosurgery: Spine Publish Before Print

Open access

Comparison of pedicle subtraction osteotomy and vertebral column decancellation for the correction of thoracolumbar kyphotic deformity in ankylosing spondylitis: a systematic review and meta-analysis

Haopeng Luan, Cong Peng, Kai Liu, and Xinghua Song

OBJECTIVE

The goal in this study was to compare the efficacy and safety outcomes of vertebral column decancellation (VCD) and pedicle subtraction osteotomy (PSO) for patients with ankylosing spondylitis (AS) with thoracolumbar kyphotic deformity.

METHODS

This study was registered on the International Prospective Register of Systematic Reviews (PROSPERO). The authors conducted a computer search of PubMed, EMBASE, Web of Science, Cochrane Library, China National Knowledge Infrastructure (CNKI), Wan Fang Database, and Wei Pu Database to collect controlled clinical studies on the efficacy and safety of VCD and PSO for patients with AS with thoracolumbar kyphotic deformity. The search covered the period from database establishment to March 2023. Two researchers screened the literature, extracted data, and evaluated the risk of bias of the included studies; these researchers recorded the authors and the sample size, and they extracted data on the intraoperative blood loss, Oswestry Disability Index, spine sagittal parameters, operation time, and complications in each study. Meta-analysis was performed using RevMan 5.4 software provided by Cochrane Library.

RESULTS

A total of 6 cohort studies with a total of 342 patients were included in this study, including 172 patients in the VCD group and 170 patients in the PSO group. The VCD group had lower intraoperative blood loss than the PSO group (mean difference [MD] −274.92, 95% CI −506.63 to −43.20, p = 0.02); significant correction of the sagittal vertical axis compared with the PSO group (MD 7.32, 95% CI −1.24 to 15.87, p = 0.03), and the operation time was shorter than that of the PSO group (MD −80.28, 95% CI −150.07 to −10.48, p = 0.02).

CONCLUSIONS

This systematic review and meta-analysis showed that VCD had more advantages than PSO in correcting the sagittal imbalance in the treatment of AS with thoracolumbar kyphotic deformity, and VCD had less intraoperative blood loss, shorter operation time, and satisfactory results in improving the quality of life.

In Journal of Neurosurgery: Spine Publish Before Print

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Correlation between degree of sagittal suture fusion and surrogates of elevated intracranial pressure in sagittal craniosynostosis

Dillan F. Villavisanis, Jessica D. Blum, Carlos E. Barrero, Christopher L. Kalmar, Natalie M. Plana, Daniel Y. Cho, Sameer Shakir, Scott P. Bartlett, Jesse A. Taylor, and Jordan W. Swanson

OBJECTIVE

Sagittal craniosynostosis constricts transverse skull growth, with possible neurocognitive sequelae. While the degree of sagittal suture fusion has been shown to influence the degree of dysmorphology, it is unknown if it impacts functional findings, including elevated intracranial pressure (ICP). The purpose of this study was to determine associations between the degree of sagittal suture fusion and optical coherence tomography (OCT) surrogates suggestive of increased ICP in patients with nonsyndromic sagittal craniosynostosis.

METHODS

Three-dimensional CT head images of patients with sagittal craniosynostosis were analyzed in Materialise Mimics and parietal bones were manually isolated to determine the percentage fusion of the sagittal suture. Retinal OCT was performed prior to the cranial vault procedure with analysis for thresholds that correlate with elevated ICP. The degree of sagittal suture fusion was compared with OCT retinal parameter measurements using Mann-Whitney U-tests, Spearman’s correlations, and multivariate logistic regression models controlled for age.

RESULTS

Forty patients (31 males) with nonsyndromic sagittal craniosynostosis at a mean (± SD) age of 3.4 ± 0.4 months were included in this study. OCT surrogates of elevated ICP (maximal retinal nerve fiber layer [RNFL] thickness and maximal anterior projection [MAP]) were not associated with total sagittal suture fusion (p > 0.05). Maximal RNFL thickness was positively associated with increased percentage of posterior one-half (rho = 0.410, p = 0.022) and posterior one-third (rho = 0.417, p = 0.020) sagittal suture fusion. MAP was also positively associated with increased percentage of posterior one-half (rho = 0.596, p < 0.001) and posterior one-third (rho = 0.599, p < 0.001) sagittal suture fusion. Multivariate logistic regression models revealed increased percentage of posterior one-half (p = 0.048) and posterior one-third (p = 0.039) sagittal suture fusion predicted ICP > 20 mm Hg.

CONCLUSIONS

Increased percentage fusion of the posterior sagittal suture, but not total suture, was positively associated with retinal changes indicative of increased ICP. These findings suggest suture fusion leading to increased ICP may be region specific.

In Journal of Neurosurgery: Pediatrics Publish Before Print

Restricted access

Does axial cervical pain improve with surgical fusion? A meta-analysis

James S. Harrop, Glenn A. Gonzalez, Reyan K. Qasba, Guilherme Porto, John V. Wainwright, Sara Thalheimer, Merrie Schorsch, Alexander R. Vaccaro, and Zoher Ghogawala

OBJECTIVE

Axial neck pain is a prevalent condition that causes significant morbidity and productivity loss. This study aimed to review the current literature and define the impact of surgical intervention on the management of cervical axial neck pain.

METHODS

A search was conducted of three databases (Ovid MEDLINE, Embase, and Cochrane) for randomized controlled trials and cohort studies written in the English language with a minimum 6-month follow-up. The analysis was limited to patients with axial neck pain/cervical radiculopathy and preoperative/postoperative Neck Disability Index (NDI) and visual analog scale (VAS) scores. Literature reviews, meta-analyses, systematic reviews, surveys, and case studies were excluded. Two patient groups were analyzed: the arm pain predominant (pAP) cohort and the neck pain predominant (pNP) cohort. The pAP cohort had preoperative VAS neck scores that were lower than the arm scores, whereas the pNP cohort was defined as having preoperative VAS neck scores higher than the arm scores. A 30% reduction in patient-reported outcome measure (PROM) scores from the baseline represented the minimal clinically important difference (MCID).

RESULTS

Five studies met the inclusion criteria, involving a total of 5221 patients. Patients with pAP showed a slightly higher percent reduction in PROM scores from baseline than those with pNP. The NDI reduction in patients with pNP was 41.35% (mean change in NDI score 16.3/mean baseline NDI score 39.42) (p < 0.0001), whereas those with pAP had a reduction of 45.12% (15.86/35.15) (p < 0.0001). Surgical improvement was slightly but similarly greater in pNP patients compared with pAP patients (16.3 vs 15.86 points, respectively; p = 0.3193). Regarding VAS scores, patients with pNP had a greater reduction in neck pain, with a change from baseline of 53.4% (3.60/6.74, p < 0.0001), whereas those with pAP had a change from baseline of 50.3% (2.46/4.89, p < 0.0001). The difference in VAS scores for neck pain improvement was significant (3.6 vs 2.46, p < 0.0134). Similarly, patients with pNP had a 43.6% (1.96/4.5) improvement in VAS scores for arm pain (p < 0.0001), whereas those with pAP had 66.12% (4.43/6.7) improvement (p < 0.0001). The VAS scores for arm pain were significantly greater in patients with pAP (4.43 vs 1.96 points, respectively; p < 0.0051).

CONCLUSIONS

Overall, despite significant variations in the existing literature, there is mounting evidence that surgical intervention can lead to clinically meaningful improvements in patients with primary axial neck pain. The studies suggest that patients with pNP tend to have better improvement in neck pain than in arm pain. In both groups, the average improvements exceeded the MCID values and reached substantial clinical benefit in all studies. Further research is necessary to identify which patients and underlying pathologies will benefit most from surgical intervention for axial neck pain because it is a multifaceted condition with many causes.

In Journal of Neurosurgery: Spine Publish Before Print