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Open access

Bilateral cerebellopontine angle lipomas in an infant with encephalocele: illustrative case

Xiaochun Zhao, Kiana Y Prather, Sherwin A Tavakol, and Joanna E Gernsback

BACKGROUND

Bilateral cerebellopontine angle (CPA) lipomas are extremely rare. Herein the authors present a case of bilateral CPA lipomas in an infant along with a literature review of bilateral CPA lipomas.

OBSERVATIONS

A newborn girl was incidentally found to have bilateral CPA lipomas during the workup for an occipital encephalocele. The encephalocele was repaired primarily on day 2 after birth. The patient demonstrated no symptoms associated with the bilateral CPA lipomas. Eight cases of bilateral CPA lipomas were identified in the literature review and are summarized. Conservative management is the consensus strategy, given minimum growth of the tumor and the high risk of surgical intervention.

LESSONS

This is the first reported case of bilateral CPA lipomas in an infant as well as the first with a coexisting intracranial malformation. Intracranial lipomas share an extremely low growth rate and typically do not cause severe symptoms. The management of asymptomatic or mildly symptomatic bilateral CPA lipomas is usually conservative.

In Journal of Neurosurgery: Case Lessons Volume 6 (2023): Issue 13 (Sep 2023)

Open access

Ischemic stroke in a young adult with a known epileptogenic arteriovenous malformation: illustrative case

Seiji Kuribara, Takuma Maeda, Taro Yanagawa, Eisuke Tsukagoshi, Shinya Kohyama, and Hiroki Kurita

BACKGROUND

Brain arteriovenous malformations (AVMs) usually manifest as hemorrhages or seizures. They rarely present with ischemic symptoms, especially in young patients. We present a case of an epileptogenic AVM that led to cerebral infarction due to paradoxical embolic occlusion of the middle cerebral artery (MCA) involving the main feeder of the lesion.

OBSERVATIONS

A 35-year-old male had been suffering from AVM-associated epilepsy for 10 years and was scheduled for surgery. He suddenly developed right-sided hemiconvulsions followed by hemiparalysis and impaired consciousness. Computed tomography revealed no intracerebral hemorrhage, and symptoms were initially thought to indicate epilepsy and Todd’s palsy. Because of his prolonged symptoms, he underwent magnetic resonance imaging, which revealed a large cerebral infarction due to occlusion of the MCA involving the main feeder of the AVM. The patient underwent AVM resection, and the partially thrombosed nidus was completely removed. Histopathological investigation revealed a fresh thrombus in totally occluded nonarteriosclerotic feeders. He had no atrial fibrillation; however, subsequent transesophageal echocardiography revealed a patent foramen ovale, suggesting a paradoxical embolism.

LESSONS

This case serves as a reminder that AVMs can present with considerable variability. Acute cerebral infarction should be considered a possible mechanism of seizures, even in patients with epileptogenic AVM.

In Journal of Neurosurgery: Case Lessons Volume 6 (2023): Issue 13 (Sep 2023)

Open access

The management of symptomatic hyperostotic bilateral spheno-orbital meningiomas: patient series

Lauren Harris, Jarnail S Bal, Evangelos Drosos, Samir Matloob, Nicola Y Roberts, Charlotte Hammerbeck-Ward, Omar Pathmanaban, Gareth Evans, Andrew T King, Scott A Rutherford, Jonathan Pollock, and Alireza Shoakazemi

BACKGROUND

The occurrence of hyperostotic bilateral spheno-orbital meningiomas (BSOMs) is very rare. Patients present with bilateral symptoms and require bilateral treatment. This series describes 6 patients presenting to 2 UK neurosurgical units and includes a literature review. To the best of the authors’ knowledge, this is the largest series documented.

OBSERVATIONS

This is a retrospective review of patients with BSOMs presenting between 2006 and 2023. Six females, whose mean age was 43 (range: 36–64) years, presented with features of visual disturbance. Bilateral sphen-oorbital meningiomas were identified. All patients underwent bilateral staged resections. The patients had an initial improvement in their symptoms. Extensive genetic testing was performed in 4 patients, with no variants in the NF2, LZTR1, SMARCB1, SMARCE1, and SMARCA4 genes or other variants detected. The mean follow-up was 100.3 (range: 64–186) months. Sixty-seven percent of patients had good long-term visual acuity. The progression rate was 75% and was particularly aggressive in 1 patient. Four patients required radiation therapy, and 2 needed further surgery.

LESSONS

Hyperostotic BSOMs are extensive, challenging tumors causing significant disability. They can recur, with significant patient impact. Multidisciplinary management and indefinite long-term follow-up are essential. The biology of these tumors remains unclear. As molecular testing expands, the understanding of BSOM oncogenesis and potential therapeutic targets is likely to improve.

In Journal of Neurosurgery: Case Lessons Volume 6 (2023): Issue 13 (Sep 2023)

Open access

Spinal arteriovenous malformation with a calcified nodule: illustrative case

Ping-Chuan Liu, Chung-Chia Huang, and Ching-Lin Chen

BACKGROUND

This article describes a rare case of cervical spinal arteriovenous malformation (AVM) mimicking a neurogenic spinal tumor.

OBSERVATIONS

A 22-year-old female presenting with a C6–7 AVM with a calcification nodule experienced new-onset acute right upper radiculopathy associated with extradural compression of the spinal cord. Note that spinal AVMs with a calcified nodule are rare. Endovascular embolization is generally used to relieve the symptoms of AVM; however, this procedure cannot relieve cord compression, particularly in cases complicated by calcified nodules. This article discusses treatment options.

LESSONS

Decompression surgery is preferable to endovascular embolization because it alleviates symptoms while preventing cord compression and minimizing the risk of recurrence.

In Journal of Neurosurgery: Case Lessons Volume 6 (2023): Issue 13 (Sep 2023)

Open access

Three-stage correction of severe idiopathic scoliosis with limited skeletal traction during a humanitarian surgical mission: illustrative case

J. Manuel Sarmiento, Jordan Fakhoury, Angadh Singh, Cameron Hawk, Khalid Sethi, and Ravi Bains

BACKGROUND

Underprivileged and underserved patients from developing countries often present late with advanced, untreated spinal deformities. We report a three-stage all-posterior approach using limited skeletal traction with Gardner-Wells tongs (GWTs) for the management of severe idiopathic scoliosis during a humanitarian surgical mission trip.

OBSERVATIONS

A 17-year-old high-school female was previously diagnosed with juvenile idiopathic scoliosis (diagnosed at age 8) and progressed to a severe 135° kyphoscoliosis. Procedural stage 1 involved spinal instrumentation and posterior releases via posterior column osteotomies from T3 to L4. She then underwent 7 days of skeletal traction with GWTs in the intensive care unit as stage 2. In stage 3, rod engagement, posterior spinal fusion, and partial T10 vertebral column resection were performed. There were no changes in intraoperative neuromonitoring during either surgery and she woke up neurologically intact after both stages of the surgical procedure.

LESSONS

Skeletal traction with GWTs is a viable alternative to traditional halo-gravity traction in settings with limited resources. Three-stage spinal deformity correction using limited skeletal traction is a feasible and effective approach for managing severe scoliosis during humanitarian surgical mission trips.

In Journal of Neurosurgery: Case Lessons Volume 6 (2023): Issue 13 (Sep 2023)

Open access

Transvenous embolization via the facial vein for intraorbital dural arteriovenous fistula: illustrative case

Jumpei Ienaga, Tetsuya Tsukada, Toru Watanabe, Yosuke Sakai, Kenji Uda, Kazunori Shintai, Yoshio Araki, Tetsuya Nagatani, and Yukio Seki

BACKGROUND

Intraorbital arteriovenous fistula (IOAVF) is a rare type of intracranial fistula that presents with ocular signs similar to those of cavernous sinus dural arteriovenous fistula. The treatment of IOAVF is based on the vascular architecture of each case due to its infrequent occurrence. The authors report the case of an IOAVF that was successfully treated with embolization via the facial vein, with good outcomes.

OBSERVATIONS

A 78-year-old woman presented with left eyelid swelling, pulsatile ocular protrusion, and left ocular conjunctival hyperemia. Ophthalmological evaluation revealed elevated intraocular pressure; time-of-flight magnetic resonance angiography revealed a dilated left superior ophthalmic vein. Digital subtraction angiography showed an arteriovenous shunt in the left superior orbital fissure, which was treated using transvenous coil embolization. The patient experienced immediate improvement in left ocular protrusion and conjunctival hyperemia. Ophthalmological evaluation 1 month after treatment showed normal intraocular pressure in the left eye. No neurological symptoms were observed, and there was no recurrence of the arteriovenous shunt 3 months postoperatively.

LESSONS

The authors report a rare case of IOAVF treated with embolization via the facial vein with a good outcome. A thorough understanding of the vascular architecture using three-dimensional images is useful for determining endovascular access and procedures.

In Journal of Neurosurgery: Case Lessons Volume 6 (2023): Issue 13 (Sep 2023)

Open access

Upper cervical intramedullary schwannoma of the spinal cord presenting with myelopathy: illustrative case

Shyam Duvuru, Vivek Sanker, Naureen Syed, Shubham Mishra, Sayantika Ghosh, and Tirth Dave

BACKGROUND

Intramedullary schwannomas account for 1.1% of all spinal schwannomas. Preoperative diagnosis is best accomplished by thoroughly evaluating clinical and radiological characteristics, accompanied by a high index of suspicion. The authors report a case of C2–3 intramedullary schwannoma in a young male who presented with neck pain and vertigo. The current literature is also reviewed.

OBSERVATIONS

The authors reviewed the data of a young male with a 2-month history of neck pain and vertigo. Magnetic resonance imaging of the brain and cervical spine showed an intramedullary mass at C2–3 with a syrinx extending into the cervicomedullary junction. Laminectomy, myelotomy, and microsurgical excision of the mass under intraoperative neurological monitoring (IONM) were done. Postoperative pathology reported the specimen as a schwannoma.

LESSONS

Gross-total resection of a schwannoma using IONM is the treatment of choice because of the lesion’s benign nature, a better prognosis, and defined cleavage plane. Schwannomas should be included in the differential diagnosis of intramedullary spinal tumors. Because of its progressive nature, early surgery is recommended in symptomatic patients.

In Journal of Neurosurgery: Case Lessons Volume 6 (2023): Issue 13 (Sep 2023)

Restricted access

Calvarial vault remodeling including pterional decompression for the treatment of nonsyndromic scaphocephaly patients: technical note and retrospective case series

Vincent Matthijs, Jelle Vandersteene, and Edward Baert

OBJECTIVE

Surgical techniques to correct scaphocephaly often rely on the implantation of foreign material and/or postoperative helmet therapy and possibly result in minimal correction of frontal bossing. Moreover, foreign material and helmet therapy are associated with extra medical care and financial costs. Frontal bossing is perceived as a prominent, disfiguring feature of scaphocephaly. Herein, authors present the results of a total cranial vault remodeling technique that corrects scaphocephaly features without relying on foreign material or postoperative helmet therapy. It includes frontal release and pterional decompression, which aim to correct frontal bossing.

METHODS

All patients who had been operated on for isolated scaphocephaly at a single institution between January 2011 and December 2020 were included in this retrospective review. Operation time, transfusion volume, hospital stay, complications, cephalic index (CI), and bossing angle (BA) were analyzed.

RESULTS

Sixty-five patients with nonsyndromic scaphocephaly were included in this analysis. Imaging to calculate the CI and BA preoperatively, immediately postoperatively, and 1 year postoperatively was available in 22 and 20 patients, respectively. The mean CI increased from 69.2% preoperatively to 74.6% postoperatively and 75.5% 1 year postoperatively. The mean BA decreased from 114.5° preoperatively to 111.6° postoperatively and 108.9° 1 year postoperatively. The mean operating time was 2 hours 4 minutes, and the median blood transfusion volume was 100 ml. There were no major complications or reoperations.

CONCLUSIONS

The described total cranial vault remodeling technique is a safe procedure that mitigates total treatment burden, as no helmet therapy or implantation of foreign material is needed. It is effective in correcting CI and results in significant frontal bossing correction. The latter is attributed to a distinctive feature of the technique: frontal release and pterional decompression.

In Journal of Neurosurgery: Pediatrics Publish Before Print

Restricted access

Halo-gravity traction combined with growing rod treatment: an effective preoperative management for severe early-onset scoliosis

Xiaojiang Pu, Bo Yang, Qingshuang Zhou, Haojie Chen, Bin Wang, Zezhang Zhu, Yong Qiu, and Xu Sun

OBJECTIVE

The aim of this study was to investigate the effectiveness of preoperative halo-gravity traction (HGT) with subsequent growing rod (GR) treatment in patients with severe early-onset scoliosis (EOS).

METHODS

The authors retrospectively reviewed a cohort of patients with severe EOS who had received preoperative HGT with subsequent GR treatment at their center between January 2008 and January 2020. Patients with a Cobb angle in the coronal or sagittal plane that was > 90° were included. All patients received at least 6 weeks of HGT before GR placement. Results of pulmonary function tests (PFTs) and blood gas tests were compared before and after HGT. Radiological parameters were compared pre-HGT, post-HGT, postindex surgery, and at the latest follow-up.

RESULTS

A total of 28 patients (17 boys and 11 girls, mean age 6.1 ± 2.3 years) were included in this study. After a mean of 65.2 ± 22.9 days of traction, the Cobb angle decreased from 101.4° ± 12.5° to 74.5° ± 19.3° (change rate 26.5%), and the kyphosis angle decreased from 71.1° ± 21.2° to 42.7° ± 9.5° (change rate 39.9%). There was a significant improvement in BMI but a decrease in hemoglobin levels following HGT. No HGT-related complications were recorded except pin site infections in 2 patients. Statistically significant improvements in PFTs after HGT were observed in forced vital capacity (FVC) (p = 0.011), the percentage predicted FVC (p = 0.007), FEV1 (p = 0.015), and the percentage predicted forced expiratory volume in 1 second (FEV1) (p = 0.005). Fourteen patients received assisted ventilation due to preoperative hypoxia, alveolar hypoventilation, or hypercapnia. Significant improvement was seen in PaCO2 (p = 0.008), PaO2 (p = 0.005), actual bicarbonate (p = 0.005), and oxygen saturation (p = 0.012) in these patients. After the index surgery, the Cobb angle decreased to 49.5° ± 18.9° and the kyphosis angle decreased to 36.2° ± 25.8°. After a mean of 4.3 ± 1.4 lengthening procedures, the Cobb angle was 56.5° ± 15.8°, and the kyphosis angle was 38.8° ± 19.7°. Surgical complications occurred in 14 (50%) patients, but none of these patients required revision surgery at the latest follow-up.

CONCLUSIONS

Preoperative HGT notably improved both spinal deformity and pulmonary function in patients with severe EOS. GR treatment after HGT is a safe and effective strategy for these patients.

In Journal of Neurosurgery: Spine Publish Before Print

Open access

Impact of subthalamic nucleus stimulation on urinary dysfunction and constipation in Parkinson’s disease

Asra Askari, Brandon J. Zhu, Jordan L. W. Lam, Kara J. Wyant, Kelvin L. Chou, and Parag G. Patil

OBJECTIVE

The effect of subthalamic nucleus (STN) deep brain stimulation (DBS) on urinary dysfunction and constipation in Parkinson’s disease (PD) is variable. This study aimed to identify potential surgical and nonsurgical variables predictive of these outcomes.

METHODS

The authors used the Movement Disorder Society–Unified Parkinson’s Disease Rating Scale (MDS-UPDRS) Part I to assess urinary dysfunction (item 10) and constipation (item 11) preoperatively and at 6–12 months postoperatively. A multiple linear regression model was used to investigate the impact of global cerebral atrophy (GCA) and active electrode contact location on the urinary dysfunction and constipation follow-up scores, controlling for age, disease duration, baseline score, motor improvement, and levodopa-equivalent dose changes. An electric field model was applied to localize the maximal-effect sites for constipation and urinary dysfunction compared with those for motor improvement.

RESULTS

Among 74 patients, 23 improved, 28 deteriorated, and 23 remained unchanged for urinary dysfunction; 25 improved, 15 deteriorated, and 34 remained unchanged for constipation. GCA score and age significantly predicted urinary dysfunction follow-up score (R2 = 0.36, p < 0.001). Increased GCA and age were independently associated with worsening urinary symptoms. Disease duration, baseline constipation score, and anterior active electrode contacts in both hemispheres were significant predictors of constipation follow-up score (R2 = 0.31, p < 0.001). Higher baseline constipation score and disease duration were associated with worsening constipation; anterior active contact location was associated with improvement in constipation.

CONCLUSIONS

Anterior active contact location was associated with improvement in constipation in PD patients after STN DBS. PD patients with greater GCA scores before surgery were more likely to experience urinary deterioration after DBS.

In Journal of Neurosurgery Publish Before Print