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Open access

Epidural lipomatosis with foci of hemorrhage and acute compression of the spinal cord in a child with CLOVES syndrome: illustrative case

Dmytro Ishchenko, Iryna Benzar, and Andrii Holoborodko

BACKGROUND

Congenital lipomatous overgrowth, vascular malformations, epidermal nevi, spinal/skeletal anomalies, and/or scoliosis (CLOVES) syndrome is the most recently described combined vascular anomaly characterized by congenital excessive growth of adipose tissue, vascular malformations, epidermal nevi, and skeletal deformities. This condition exhibits a significant variability in clinical manifestations and a tendency for rapid progression and affects extensive anatomical regions. Information regarding the association of epidural lipomatosis with low-flow venous lymphatic malformations is rare, with few reports in the literature.

OBSERVATIONS

The authors present a case of a 6-year-old girl who was admitted to the emergency department complaining of rapidly progressing weakness in her lower extremities and partial loss of sensation in the inguinal area. Radiologically, an extradural mass was identified at the T2–6 level, causing acute spinal cord compression. Urgent decompression and partial resection of the mass were performed. Despite satisfactory intraoperative hemo- and lymphostasis, postoperative lymphorrhea/seroma leakage was encountered as a delayed complication and was managed conservatively.

LESSONS

CLOVES syndrome is characterized by the combination of various clinical symptoms, not all of which are included in the abbreviation, as well as a progressively deteriorating course, the emergence of new symptoms, and complications throughout the patient’s life. This necessitates ongoing monitoring of such patients.

Open access

Metastatic intradural primary spinal osteosarcoma: illustrative case

Shreyas Thiruvengadam, Mark Lam, and Stephen Honeybul

BACKGROUND

Osteosarcomas are a common primary bone neoplasm among adolescents but represent 0.2% of all malignancies with an incidence of two to four cases per million persons annually worldwide. Although known to have significant metastatic potential, its rare incidence, treatment resistance, and poor prognosis have rendered it a poorly understood and infrequently documented pathology.

OBSERVATIONS

Herein the authors present the first documented case of lumbosacral intradural metastasis of a primary osteosarcoma in a young patient, possibly via intradural dissemination following pinhole durotomy in a prior thoracic surgery.

LESSONS

Osteosarcomas remain a difficult pathology to treat, particularly upon metastatic dissemination. The utility of adjuvant radiotherapy after resection of an osteosarcoma is increasingly evident in the reduction of local recurrence. In the context of intraoperative pinhole durotomies in resections of high-grade lesions, due consideration should be given to whole-spine radiation, although this remains an evidence-free zone.

Open access

A rare case of atypical intradural extramedullary glioblastoma diagnosed utilizing next-generation sequencing and methylation profiling: illustrative case

William J Shelton, Andrew P Mathews, Karrar Aljiboori, J. Stephen Nix, Murat Gokden, and Analiz Rodriguez

BACKGROUND

Primary spinal cord tumors, especially primary spinal cord glioblastoma multiforme (PSC-GBM), are exceptionally rare, accounting for less than 1.5% of all spinal tumors. Their infrequency and aggressive yet atypical presentation make diagnosis challenging. In uncertain cases, a surgical approach for tissue diagnosis is often optimal.

OBSERVATIONS

A 76-year-old male presented with a rapidly progressing clinical history marked by worsening extremity weakness, urinary retention, and periodic fecal incontinence alongside diffuse changes on neuraxis imaging. The patient, in whom subacute polyneuropathy was initially diagnosed, received multiple rounds of steroids and intravenous immunoglobulin without clinical improvement. Histopathological review of the biopsy tissue yielded an initial diagnosis of spindle cell neoplasm. Next-generation sequencing (NGS) is done routinely on all neuropathology specimens at the authors’ institution, and methylation profiling is pursued in difficult cases. Ultimately, NGS and methylation profiling results were essential to an integrated final diagnosis of GBM.

LESSONS

PSC-GBM is a rare but highly aggressive occurrence of this tumor. Prolonged back pain, rapid neurological decline, and imaging changes warrant the consideration of lesional biopsy for precise disease characterization. In inconclusive cases, NGS has proved invaluable for clinical clarification and diagnosis, underscoring its importance for integrated diagnoses in guiding appropriate treatment strategies.

Open access

Thoracic spinal cord injury after surgical removal of a ruptured cerebellar arteriovenous malformation in a patient in the Concorde position: illustrative case

Kohei Ishikawa, Hideki Endo, Yasufumi Ohtake, Toshiichi Watanabe, and Hirohiko Nakamura

BACKGROUND

Thoracic spinal cord injury after posterior cranial fossa surgery in younger patients is a rare complication. There have been reports of this complication in tumor and spine fields but not in vascular surgery.

OBSERVATIONS

A 22-year-old-man experienced cerebellar arteriovenous malformation rupture, and the malformation was surgically removed with the man in the Concorde position. After surgery, the man had severe paraplegia, and a thoracic spinal cord injury was diagnosed.

LESSONS

In younger patients, cervical hyperflexion in the Concorde position can cause thoracic spinal cord injury even in surgery for cerebrovascular disease.

Open access

Migratory schwannoma of the cauda equina with a change in radicular pattern: illustrative case

Matthew S Parr, Sanford R Williams, James H Mooney, Travis J Atchley, and Mamerhi O Okor

BACKGROUND

Intradural spinal tumors are an uncommon entity with a variety of pathologies and symptom patterns. Few cases reports in the literature have described tumor migration within the spinal canal.

OBSERVATIONS

A 38-year-old male presented with bilateral upper lumbar radicular symptoms of anterior thigh pain, with an enhancing tumor of the cauda equina initially located at L1–2. He declined surgery initially, and at a follow-up 3 years later, his symptoms were unchanged but the tumor was now located at T12–L1. He again declined surgery, but 3 months later, he had a significant change in his pain distribution, which was now along his posterolateral right leg to his foot with associated dorsiflexion and extensor hallicus longus weakness. At this time, the tumor had migrated to L2–3. He underwent laminectomy and tumor resection with resolution of his radicular symptoms and improvement in his strength back to baseline by the 1-month follow-up. Pathology was consistent with a World Health Organization grade I schwannoma.

LESSONS

Migratory schwannoma is a rare entity but should be considered when radicular symptoms acutely change in the setting of a known intradural tumor. Repeat imaging should be performed to avoid wrong-level surgery. Intraoperative imaging can also be used for tumor localization.

Open access

Successful coil embolization of a ruptured pseudoaneurysm of the superior gluteal artery after a percutaneous awake robot-assisted sacroiliac joint fusion: illustrative case

Samah Morsi, Alyssa M Bartlett, Andrew A Hardigan, Mounica Paturu, Shawn W Adams, Malcolm R DeBaun, Waleska Pabon-Ramos, and Muhammad M Abd-El-Barr

BACKGROUND

Robot-assisted sacroiliac joint (SIJ) fusion has gained popularity, but it carries the risk of complications such as injury to the superior gluteal artery (SGA). The authors present the case of an awake percutaneous robot-assisted SIJ fusion leading to an SGA pseudoaneurysm.

OBSERVATIONS

An 80-year-old male, who had undergone an awake percutaneous robot-assisted SIJ fusion, experienced postoperative left hip pain and bruising. Subsequent arteriography demonstrated an SGA branch pseudoaneurysm requiring coil embolization.

LESSONS

An SGA injury, although uncommon (1.2% incidence), can arise from percutaneous screw placement, aberrant anatomy, or hardware contact. Thorough preoperative imaging, precise robot-assisted screw insertion, and soft tissue protection are crucial to mitigate risks. Immediate angiography aids in prompt diagnosis and effective intervention. Comprehensive knowledge of anatomical variants is essential for managing complications and optimizing preventative measures in robot-assisted SIJ fusion.

Open access

Cervicothoracic ventral-dorsal rhizotomy for bilateral upper-extremity hypertonia in cerebral palsy: illustrative case

Ryan Kelly, Hanna R Kemeny, Sunny Abdelmageed, Robin Trierweiler, Tim Krater, Melissa A LoPresti, and Jeffrey S Raskin

BACKGROUND

Management of medically refractory limb-specific hypertonia is challenging. Neurosurgical options include deep brain stimulation, intrathecal baclofen, thalamotomy, pallidotomy, or rhizotomy. Cervical dorsal rhizotomy has been successful in the treatment of upper-extremity spasticity. Cervical ventral and cervical ventral-dorsal rhizotomy (VDR) has been used in the treatment or torticollis and traumatic hypertonia; however, the use of cervicothoracic VDR for the treatment of upper-extremity mixed hypertonia is not well described.

OBSERVATIONS

A 9-year-old girl with severe quadriplegic mixed hypertonia secondary to cerebral palsy (CP) underwent cervicothoracic VDR. Modified Ashworth Scale scores, provision of caregiving, and examination improved. Treatment was well tolerated.

LESSONS

Cervicothoracic VDR can afford symptomatic and quality of life improvement in patients with medically refractory limb hypertonia. Intraoperative positioning and nuances in surgical techniques are particularly important based on spinal cord position as modified by scoliosis. Here, the first successful use of cervicothoracic VDR for the treatment of medically refractory upper-limb hypertonia in a pediatric patient with CP is described.

Open access

Isolated unilateral alar ligamentous injury: illustrative cases

Benjamin C Reeves, Marcus Valcarce-Aspegren, Stephanie M Robert, Aladine A Elsamadicy, Alexander M Tucker, Phillip B Storm, Michael L DiLuna, and Adam J Kundishora

BACKGROUND

Isolated unilateral alar ligament injury (IUALI) is a rare and likely underreported occurrence after upper cervical trauma, with only 16 cases documented in the literature to date. Patients generally present with neck pain, and definitive diagnosis is typically made by magnetic resonance imaging (MRI). Unfortunately, likely due in part to its rarity, there are no formal guidelines for the treatment of an IUALI. Furthermore, there is a limited understanding of the long-term consequences associated with its inadequate treatment.

OBSERVATIONS

Here, the authors report on three pediatric patients, each found to have an IUALI after significant trauma. All patients presented with neck tenderness, and two of the three had associated pain-limited range of neck motion. Imaging revealed either a laterally deviated odontoid process on cervical radiographs and/or MRI evidence of ligamentous strain or discontinuity. Each patient was placed in a hard cervical collar for 1 to 2 months with excellent resolution of symptoms. A comprehensive review of the literature showed that all patients with IUALI who had undergone external immobilization with either rigid cervical collar or halo fixation had favorable outcomes at follow-up.

LESSONS

For patients with IUALI, a moderate course of nonsurgical management with rigid external immobilization appears to be an adequate first-line treatment.

Open access

Posterior-only 2-level vertebrectomy and fusion in a medically complex patient with lumbar metastasis: illustrative case

Ryan Johnson, Annabelle Shaffer, Ashley Tang, Kathryn Tsai, Gina Guglielmi, and Paul M Arnold

BACKGROUND

Spinal metastases are commonly seen in patients with cancer and often indicate a poor prognosis. Treatment can include curative or palliative surgery, chemotherapy, and radiation therapy. The surgical approach varies widely on the basis of the affected region of the spine, the location of the tumor (anterior versus posterior), the goal of surgery, the health of the patient, and surgeon preference.

OBSERVATIONS

The authors present a case of a 68-year-old male with intractable lower-back pain and substantially diminished ambulation. Diagnostic imaging revealed a lumbar metastasis from a cholangiocarcinoma primary at L2–3 (4.5 cm anteroposterior × 5.7 cm transverse × 7.0 cm craniocaudal). The patient underwent a 2-level vertebrectomy with expandable cage placement and T10 to S2 fusion via a posterior-only approach. The patient regained much of his mobility and quality of life after the surgery.

LESSONS

Although this was a high-risk surgery, the authors show that a posterior-only approach can be used for lumbar vertebrectomies and fusion when necessary. Palliative surgeries carrying a high risk, especially in the setting of a limited prognosis, should include multidisciplinary deliberations and a thorough discussion of the risks and outcome expectations with the patient.

Open access

Thoracic root–related intradural extramedullary cavernoma presenting with subarachnoid hemorrhage: illustrative case

Vicente de Paulo Martins Coelho Junior, Nathaniel Toop, Peter Kobalka, and Vikram B Chakravarthy

BACKGROUND

Just 5% of all cavernomas are located in the spine. Thoracic root–related subtypes are the rarest, with a total of 14 cases reported in the literature to date. Among them, only 4 presented with subarachnoid hemorrhage (SAH).

OBSERVATIONS

A 65-year-old female presented after an ictus of headache with no neurological deficits. Computed tomography (CT) demonstrated sulcal SAH, with the remainder of the workup nondiagnostic for etiology. Three weeks later, she re-presented with acute thoracic back pain and thoracic myelopathy. CT and magnetic resonance imaging suggested dubiously a T9–10 disc herniation with spinal cord compression. Surgical decompression and resection were then planned. Intraoperative ultrasound (IUS) demonstrated an intradural extramedullary lesion, confirmed to be cavernoma. Complete resection was achieved, and the patient was discharged a few days postoperatively to inpatient rehabilitation.

LESSONS

Although spine imaging is deemed to be low yield in the evaluation of cryptogenic SAH, algorithms can be revisited even in the absence of spine-related symptoms. Surgeons can be prepared to change the initial surgical plan, especially when preoperative imaging is unclear. IUS is a powerful tool to assess the thecal sac after its exposure and to help guide this decision, as in this rare entity.