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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

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

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.

Open access

Adhesive arachnoiditis, subarachnoid hemorrhage, and intradural extramedullary thoracic cavernoma: illustrative case

Agne Andriuskeviciute, Michel Gustavo Mondragón-Soto, Nicolas Penet, and Juan Barges-Coll

BACKGROUND

Spinal arachnoiditis can result from various factors, including spinal subarachnoid hemorrhage (sSAH). In this paper, the authors describe a case of intradural extramedullary cavernoma with an initial presentation of subarachnoid hemorrhage leading to multilevel spinal arachnoiditis to discuss the pathophysiology and optimal treatment strategy.

OBSERVATIONS

Spinal intradural extramedullary cavernoma manifesting with sSAH is a rare clinical presentation; therefore, there is no clear strategy for the management of sSAH. Spinal arachnoiditis is a result of chronic inflammation of the pia arachnoid layer due to hematomyelia. No effective treatment that interrupts this inflammatory cascade and would also prevent the development of spinal arachnoiditis has been described to date.

LESSONS

Lumbar drainage could aid in sSAH management, relieve spinal cord compression, and restore the normal spinal cerebrospinal fluid circulation gradient. It could help to clear the blood degradation products rapidly and prevent early inflammatory arachnoiditis development. Mini-invasive intrathecal endoscopic adhesiolysis appears to be a reasonable approach for reducing the risk of aggravating spinal arachnoiditis with a mechanical-surgical stimulus. Whether a conservative approach should be applied in these patients with mild myelopathy symptoms is still debatable.

Open access

Mycobacterium xenopi vertebral osteomyelitis in a patient with systemic lupus erythematosus: illustrative case

Ryan Kelly, Josha Woodward, Jacob B Gerzenshtein, Mariam Aziz, and John E O’Toole

BACKGROUND

Mycobacterium xenopi is a common nontuberculous Mycobacterium (NTM) that is slow growing and an infrequent cause of infection. When infections do occur, it is by exposure to contaminated soil or water or to infectious aerosols. Nontuberculous mycobacterial infections in the spine are exceedingly rare. Risk factors can include immunosuppression, particularly human immunodeficiency virus; however, other systemic diseases such as systemic lupus erythematosus (SLE) have been reported.

OBSERVATIONS

The authors report a case of cord compression due to M. xenopi vertebral osteomyelitis with an epidural abscess in a patient with SLE on hydroxychloroquine and recent steroid use. The authors explore the presentation of a patient who developed acute neurological deficits concerning for spinal pathology secondary to NTM. Although considered a rare occurrence, patients with autoimmune pathologies are susceptible to infection by unusual organisms. Standard treatment of autoimmune diseases can predispose patients to infection and warrant surgical correction to prevent long-term neurological deficits.

LESSONS

There is still much work and research to be done in the exploration and understanding of nontuberculous mycobacterial infection, pathophysiology, and treatment in the immunocompetent population and in patients with autoimmune disorders.

Open access

Intraspinal cervicothoracic junction chondrosarcoma: illustrative case

Matthew T Carr, Margaret Pain, Kevin Kay, and John K Houten

BACKGROUND

Chondrosarcoma is an uncommon spinal tumor that can present as an extraskeletal mass. Rarely, these tumors present as dumbbell tumors through the neural foramina, mimicking schwannomas or neurofibromas.

OBSERVATIONS

A 46-year-old female presented with 2 years of worsening right-arm radiculopathy. Magnetic resonance imaging of the thoracic spine revealed a peripherally enhancing extramedullary mass through the right T1 foramen and compressing the spinal cord. Computed tomography showed the mass to be partially calcified. She underwent C7–T2 laminectomy and C6–T3 posterior instrumented fusion with gross-total resection of an extradural mass. Pathology revealed a grade I chondrosarcoma. Her symptoms improved postoperatively, with some residual right-arm radicular pain.

LESSONS

Intraspinal extradural dumbbell conventional chondrosarcoma is rare, with only 9 cases, including ours, reported. Patient ages range from 16 to 72 years old, and male sex is more common in these cases. The most common location is the thoracic spine, and our case is the only reported one in the cervicothoracic junction. These tumors often mimic schwannomas on imaging, but chondrosarcoma should remain in the differential diagnosis, because management of these tumors differs. Chondrosarcoma may benefit from more aggressive resection, including en bloc resection, and may require adjuvant radiotherapy.

Open access

Compressive myelopathy from diffuse spinal dural calcifications in a patient with end-stage renal disease: illustrative case

Alexis Malecki, Jacob Pawloski, Anthony Anzalone, Kelly Shaftel, Hassan Ali Fadel, and Ian Lee

BACKGROUND

Diffuse spinal dural calcification is a rare disorder associated with hyperparathyroidism, including the secondary forms associated with renal failure, osteodystrophy, and chronic hypocalcemia. Here, the authors report a rare case of diffuse dural calcification causing spinal cord compression with myelopathy, requiring decompressive surgery with duraplasty to achieve adequate decompression.

OBSERVATIONS

A 46-year-old male with a history of renal failure on dialysis presented with 2 months of progressive neuropathic pain, lower-extremity weakness, and nonsustained clonus. Spine imaging showed severe renal osteodystrophy with multilevel compression fractures and diffuse dural calcifications with areas of invagination causing severe spinal cord compression. Decompressive surgery was recommended. In surgery, a thickened and calcified dura was encountered with areas of buckling causing spinal cord compression. The invaginated area of the dura was resected and reconstructed with patch duraplasty. The patient’s neurological status remained unchanged postoperatively, and at the 6-month follow-up, the patient reported significant improvement in pain and muscle spasms.

LESSONS

Diffuse dural calcifications are a rare complication of prolonged dialysis and secondary hyperparathyroidism. When there is resultant spinal cord compression, this condition requires an intradural approach that addresses the thickened, calcified dura directly to obtain adequate spinal cord decompression.

Open access

Minimally invasive resection of a prominent transverse process in neurogenic thoracic outlet syndrome: new application for a primarily spinal approach. Illustrative case

Marc Hohenhaus, Johann Lambeck, Nico Kremers, Jürgen Beck, Christoph Scholz, and Ulrich Hubbe

BACKGROUND

The optimal surgical approach to treat neurogenic thoracic outlet syndrome (nTOS) depends on the individual patient’s anatomy as well as the surgeon’s experience. The authors present a minimally invasive posterior approach for the resection of a prominent transverse process to reduce local muscular trauma.

OBSERVATIONS

A 19-year-old female presented with painful sensations in the right arm and severe fine-motor skill dysfunction in the right hand, each of which had been present for several years. Further examination confirmed affected C8 and T1 areas, and imaging showed an elongated C7 transverse process displacing the lower trunk of the brachial plexus. Decompression of the plexus structures by resection of the C7 transverse process was indicated, owing to persistent neurological effects. Surgery was performed using a minimally invasive posterior approach in which the nuchal soft tissue was bluntly dissected by dilatators and resection of the transverse process was done microscopically through a tubular retractor. The postoperative course showed a sufficient reduction of pain and paresthesia.

LESSONS

The authors describe a minimally invasive posterior approach for the treatment of nTOS with the aim of providing indirect relief of strain on brachial plexus structures. The advantages of this technique include a small skin incision and minor soft tissue damage.

Open access

Angiomatoid fibrous histiocytoma: primary intracranial lesion with thoracic spine metastasis and a malignant course. Illustrative case

Audrey Demand, Sean Barber, Suzanne Powell, and Gavin Britz

BACKGROUND

Angiomatoid fibrous histiocytoma (AFH) is an exceptionally rare soft tissue neoplasm. This tumor primarily presents as a benign soft tissue lesion in children with an average age of 14 years. The standard treatment regimen is wide local excision with interval follow-up. However, newer reports have demonstrated malignant potential with the possibility of intracranial metastasis.

OBSERVATIONS

A 45-year-old male with no soft tissue primary tumor presented with a primary intracranial lesion and thoracic spine metastasis refractory to chemotherapy and radiation treatment.

LESSONS

This report illustrates the potential for a highly malignant nature of metastatic AFH. In addition, the authors demonstrate an incidence of AFH in a middle-aged male without a primary soft tissue or skin lesion. This report highlights the importance of prompt treatment and excision for AFH, as there is still little understanding of successful options for systemic therapy.

Open access

Spinal cord stimulation for chronic pain treatment following sacral chordoma resection: illustrative case

Khaled M Taghlabi, Taimur Hassan, Isuru A Somawardana, Sibi Rajendran, Ahmed Doomi, Lokeshwar S Bhenderu, Jesus G Cruz-Garza, and Amir H Faraji

BACKGROUND

Cancer-related or postoperative pain can occur following sacral chordoma resection. Despite a lack of current recommendations for cancer pain treatment, spinal cord stimulation (SCS) has demonstrated effectiveness in addressing cancer-related pain.

OBSERVATIONS

A 76-year-old female with a sacral chordoma underwent anterior osteotomies and partial en bloc sacrectomy. She subsequently presented with chronic pain affecting both buttocks and posterior thighs and legs, significantly impeding her daily activities. She underwent a staged epidural SCS paddle trial and permanent system placement using intraoperative neuromonitoring. The utilization of percutaneous leads was not viable because of her history of spinal fluid leakage, multiple lumbosacral surgeries, and previous complex plastic surgery closure. The patient reported a 62.5% improvement in her lower-extremity pain per the modified Quadruple Visual Analog Scale and a 50% improvement in the modified Pain and Sleep Questionnaire 3-item index during the SCS trial. Following permanent SCS system placement and removal of her externalized lead extenders, she had an uncomplicated postoperative course and reported notable improvements in her pain symptoms.

LESSONS

This case provides a compelling illustration of the successful treatment of chronic pain using SCS following radical sacral chordoma resection. Surgeons may consider this treatment approach in patients presenting with refractory pain following spinal tumor resection.