Dejen T Gebrewahd, Tsegazeab Laeke, Eyob Z Wendimagegnehu, Mestet Y Shiferaw, Tesfaye G Tefera, Ilili A Aliye, Taye J Robele, Bereket H Mekuria, Anteneh E Berga, Surafael M Mendere, Sebboona B Abelti
Background: Actinomycosis is a chronic suppurative infection caused by non-spore-forming, anaerobic, and filamentous gram-positive bacteria. Primary central nervous system involvement is rare, with no specific clinical features, causing a clinical diagnostic dilemma. Imaging can help in localizing and characterizing the lesion; however, a definitive diagnosis relies on culture and/or histopathology.
Observations: The authors describe a 29-year-old male farmer with a rare case of invasive and diffuse cranial actinomycosis with a dura-based mass mimicking a brain tumor. Brain magnetic resonance imaging showed a moderately enhanced right frontoparietal infiltrative dura-based mass with marked thickening of the skull and multiple scalp actinomycotic abscesses. He underwent microsurgical excision of the mass, orbital decompression, and debridement of the scalp abscess. Histopathology confirmed actinomycosis, and his postoperative course was uneventful.
Lessons: Invasive and diffuse cranial actinomycosis with a dura-based actinomycetoma is a rare presentation that poses a diagnostic challenge due to its nonspecific manifestations. Imaging is helpful in localizing and characterizing the lesion; however, histopathology remains the gold standard for diagnosing actinomycosis. A high index of suspicion is also warranted in patients with predisposing factors to promote an early diagnosis and the initiation of appropriate treatments to improve functional recovery and limit residual deficits. https://thejns.org/doi/10.3171/CASE24210.
{"title":"An invasive and diffuse cranial actinomycosis with a dura-based mass mimicking a brain tumor: illustrative case.","authors":"Dejen T Gebrewahd, Tsegazeab Laeke, Eyob Z Wendimagegnehu, Mestet Y Shiferaw, Tesfaye G Tefera, Ilili A Aliye, Taye J Robele, Bereket H Mekuria, Anteneh E Berga, Surafael M Mendere, Sebboona B Abelti","doi":"10.3171/CASE24210","DOIUrl":"10.3171/CASE24210","url":null,"abstract":"<p><strong>Background: </strong>Actinomycosis is a chronic suppurative infection caused by non-spore-forming, anaerobic, and filamentous gram-positive bacteria. Primary central nervous system involvement is rare, with no specific clinical features, causing a clinical diagnostic dilemma. Imaging can help in localizing and characterizing the lesion; however, a definitive diagnosis relies on culture and/or histopathology.</p><p><strong>Observations: </strong>The authors describe a 29-year-old male farmer with a rare case of invasive and diffuse cranial actinomycosis with a dura-based mass mimicking a brain tumor. Brain magnetic resonance imaging showed a moderately enhanced right frontoparietal infiltrative dura-based mass with marked thickening of the skull and multiple scalp actinomycotic abscesses. He underwent microsurgical excision of the mass, orbital decompression, and debridement of the scalp abscess. Histopathology confirmed actinomycosis, and his postoperative course was uneventful.</p><p><strong>Lessons: </strong>Invasive and diffuse cranial actinomycosis with a dura-based actinomycetoma is a rare presentation that poses a diagnostic challenge due to its nonspecific manifestations. Imaging is helpful in localizing and characterizing the lesion; however, histopathology remains the gold standard for diagnosing actinomycosis. A high index of suspicion is also warranted in patients with predisposing factors to promote an early diagnosis and the initiation of appropriate treatments to improve functional recovery and limit residual deficits. https://thejns.org/doi/10.3171/CASE24210.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 13","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418642/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142309522","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Zachary T Olmsted, Peter B Wu, Alireza Katouzian, Michael J Dorsi
Background: Spinal cord stimulation (SCS), including the percutaneous placement of epidural stimulation leads, has been increasingly utilized to treat chronic pain. Although lead migration is a well-characterized complication, few studies have reported on malpositioned leads in the intrathecal space. Here, the authors discuss two cases of intrathecal lead placement necessitating surgical revision.
Observations: This report is a two-case series on the inadvertent placement of percutaneous SCS leads in the intrathecal space. The authors describe the identification of malpositioned leads, describe the clinical presentation, characterize stimulation parameters, and report improvement following neurosurgical revision for each case. Two patients originally presenting with chronic low-back pain underwent percutaneous SCS lead implantation. Both patients presented with atypical pain symptoms in the acute to subacute postprocedural period, raising suspicion for malpositioned leads. Imaging was consistent with intrathecal malpositioning. Both patients underwent revision surgery resulting in symptomatic improvement.
Lessons: Indicators of malpositioned thoracic SCS leads in the intrathecal space include thoracoabdominal or flank pain exacerbated by movement, insufficient pain relief versus that in the SCS trial, very low electrode impedances, direct visualization on imaging, and lack of epidural lead visualization following laminectomy. Revision options include removal of the intrathecal leads and the surgical placement of a paddle electrode in the epidural space. https://thejns.org/doi/10.3171/CASE24275.
{"title":"Intrathecal placement of percutaneous spinal cord stimulation leads: illustrative cases.","authors":"Zachary T Olmsted, Peter B Wu, Alireza Katouzian, Michael J Dorsi","doi":"10.3171/CASE24275","DOIUrl":"10.3171/CASE24275","url":null,"abstract":"<p><strong>Background: </strong>Spinal cord stimulation (SCS), including the percutaneous placement of epidural stimulation leads, has been increasingly utilized to treat chronic pain. Although lead migration is a well-characterized complication, few studies have reported on malpositioned leads in the intrathecal space. Here, the authors discuss two cases of intrathecal lead placement necessitating surgical revision.</p><p><strong>Observations: </strong>This report is a two-case series on the inadvertent placement of percutaneous SCS leads in the intrathecal space. The authors describe the identification of malpositioned leads, describe the clinical presentation, characterize stimulation parameters, and report improvement following neurosurgical revision for each case. Two patients originally presenting with chronic low-back pain underwent percutaneous SCS lead implantation. Both patients presented with atypical pain symptoms in the acute to subacute postprocedural period, raising suspicion for malpositioned leads. Imaging was consistent with intrathecal malpositioning. Both patients underwent revision surgery resulting in symptomatic improvement.</p><p><strong>Lessons: </strong>Indicators of malpositioned thoracic SCS leads in the intrathecal space include thoracoabdominal or flank pain exacerbated by movement, insufficient pain relief versus that in the SCS trial, very low electrode impedances, direct visualization on imaging, and lack of epidural lead visualization following laminectomy. Revision options include removal of the intrathecal leads and the surgical placement of a paddle electrode in the epidural space. https://thejns.org/doi/10.3171/CASE24275.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 13","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418641/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142309523","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Kirby C Manigos, Joseph Erroll V Navarro, Kenny S Seng, Jose Carlos S Alcazaren, Oliver Ryan M Malilay, Tito Guillermo D Rejante, Kevin Paul Ferraris
Background: Fibrous dysplasia is a rare and benign skeletal lesion characterized by fibrous tissue proliferation due to an abnormal osteogenesis replacing normal bone.
Observations: An 18-year-old male with fibrous dysplasia of the left sphenoid, ethmoid, orbit, and frontal bones was managed with excision and skull base reconstruction. After complete removal of the tumor, skull base reconstruction was commenced by making a reverse temporalis flap and placing it over the opened paranasal sinuses for a robust vascularized graft, followed by an abdominal fat graft, and then a pedicled pericranal flap was added to complete the multilayer onlay graft. To recreate the skull base, a mirror image of the contralateral skull base was constructed using three-dimensional (3D) printing, and the 3D-printed model was sterilized prior to the surgery. Intraoperatively, the model was then pressed onto dental alginate gel to make a negative mold. This was used to make the definitive flap using polymethylmethacrylate. Temporoplasty was also performed using polymethylmethacrylate to fill the defect left by the temporalis graft. The patient recovered well following the procedure.
Lessons: Appropriate, personalized skull base reconstruction techniques can be successfully done with 3D printing using alternative low-cost materials and implements, especially following resection of cases like craniofacial fibrous dysplasia. https://thejns.org/doi/10.3171/CASE24262.
{"title":"A novel technique for skull base reconstruction in craniofacial fibrous dysplasia surgery using three-dimensional printing and a dental alginate mold: illustrative case.","authors":"Kirby C Manigos, Joseph Erroll V Navarro, Kenny S Seng, Jose Carlos S Alcazaren, Oliver Ryan M Malilay, Tito Guillermo D Rejante, Kevin Paul Ferraris","doi":"10.3171/CASE24262","DOIUrl":"10.3171/CASE24262","url":null,"abstract":"<p><strong>Background: </strong>Fibrous dysplasia is a rare and benign skeletal lesion characterized by fibrous tissue proliferation due to an abnormal osteogenesis replacing normal bone.</p><p><strong>Observations: </strong>An 18-year-old male with fibrous dysplasia of the left sphenoid, ethmoid, orbit, and frontal bones was managed with excision and skull base reconstruction. After complete removal of the tumor, skull base reconstruction was commenced by making a reverse temporalis flap and placing it over the opened paranasal sinuses for a robust vascularized graft, followed by an abdominal fat graft, and then a pedicled pericranal flap was added to complete the multilayer onlay graft. To recreate the skull base, a mirror image of the contralateral skull base was constructed using three-dimensional (3D) printing, and the 3D-printed model was sterilized prior to the surgery. Intraoperatively, the model was then pressed onto dental alginate gel to make a negative mold. This was used to make the definitive flap using polymethylmethacrylate. Temporoplasty was also performed using polymethylmethacrylate to fill the defect left by the temporalis graft. The patient recovered well following the procedure.</p><p><strong>Lessons: </strong>Appropriate, personalized skull base reconstruction techniques can be successfully done with 3D printing using alternative low-cost materials and implements, especially following resection of cases like craniofacial fibrous dysplasia. https://thejns.org/doi/10.3171/CASE24262.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 13","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-23","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418638/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142309521","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Vincenzo Mastropasqua, Marco Obersnel, Vito Stifano, Marco Gessi, Domenico Cristiano Corsi, Francesco Doglietto
Background: Pancreatoblastoma is a malignant neoplasm of the pancreas, occurring usually in children and rarely in adults. Treatment consists of surgery with a variable combination of adjuvant therapies. Liver metastases are common, whereas brain diffusion is exceptionally rare.
Observations: The authors report the case of a 42-year-old man with a 16-year history of metastatic pancreatoblastoma, previously treated with surgery, chemotherapy, and radiotherapy, demonstrating a partial response. He presented with headache and dizziness, and brain magnetic resonance imaging (MRI) showed a cerebellar lesion. A craniotomy was performed with complete tumor removal, and the postoperative course was uneventful. Brain MRI showed gross-total resection of the lesion, and the patient was discharged with an improvement of the preoperative symptoms. Histopathological analysis confirmed the diagnosis of metastasis from pancreatoblastoma. The patient received adjuvant stereotactic radiotherapy and showed further clinical improvement at the last follow-up.
Lessons: Brain metastases from pancreatoblastoma are exceptionally rare and poorly described in the literature. There is no standard therapy for this condition; hence, patients usually undergo treatments similar to those for other central nervous system metastases. All the described patients have had good clinical outcomes yet short-term follow-ups; therefore, further investigations are needed to better understand the best treatments for this condition. https://thejns.org/doi/10.3171/CASE23764.
{"title":"A rare case of central nervous system pancreatoblastoma metastasis: illustrative case.","authors":"Vincenzo Mastropasqua, Marco Obersnel, Vito Stifano, Marco Gessi, Domenico Cristiano Corsi, Francesco Doglietto","doi":"10.3171/CASE23764","DOIUrl":"10.3171/CASE23764","url":null,"abstract":"<p><strong>Background: </strong>Pancreatoblastoma is a malignant neoplasm of the pancreas, occurring usually in children and rarely in adults. Treatment consists of surgery with a variable combination of adjuvant therapies. Liver metastases are common, whereas brain diffusion is exceptionally rare.</p><p><strong>Observations: </strong>The authors report the case of a 42-year-old man with a 16-year history of metastatic pancreatoblastoma, previously treated with surgery, chemotherapy, and radiotherapy, demonstrating a partial response. He presented with headache and dizziness, and brain magnetic resonance imaging (MRI) showed a cerebellar lesion. A craniotomy was performed with complete tumor removal, and the postoperative course was uneventful. Brain MRI showed gross-total resection of the lesion, and the patient was discharged with an improvement of the preoperative symptoms. Histopathological analysis confirmed the diagnosis of metastasis from pancreatoblastoma. The patient received adjuvant stereotactic radiotherapy and showed further clinical improvement at the last follow-up.</p><p><strong>Lessons: </strong>Brain metastases from pancreatoblastoma are exceptionally rare and poorly described in the literature. There is no standard therapy for this condition; hence, patients usually undergo treatments similar to those for other central nervous system metastases. All the described patients have had good clinical outcomes yet short-term follow-ups; therefore, further investigations are needed to better understand the best treatments for this condition. https://thejns.org/doi/10.3171/CASE23764.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 12","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418033/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142304902","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Background: Extracranial carotid artery aneurysms (ECAAs) are rare, and treatment guidelines are lacking. Few reports on endovascular treatments performed for ECAAs exist.
Observations: A 73-year-old woman with a left giant cervical internal carotid artery aneurysm was treated with overlapping closed-cell stents. The aneurysm regrew 1 year after the treatment, and then a covered stent was deployed. Angioscopy was performed to confirm neointimal development to determine the appropriate stent position before the retreatment, and it revealed that the stent struts were embedded in thick neointima for the most part but that the neointima was thin around the aneurysm neck. Multiple holes connecting to the aneurysm were observed between the stent struts. A covered stent overlapped inside the closed-cell stents, and blood flow into the aneurysm completely disappeared.
Lessons: When deploying the covered stent for recurrent aneurysms, angioscopy is useful for confirming neointimal development and determining the appropriate stent length and position. Angioscopic observations suggest that using stents with a higher mesh density and smaller pore size can reduce the neck hole size of the aneurysm and may achieve complete occlusion of the aneurysm. https://thejns.org/doi/10.3171/CASE24383.
{"title":"Covered stent deployment for a recurrent cervical internal carotid artery aneurysm referencing angioscopy: illustrative case.","authors":"Hiromichi Hayami, Kenji Fukutome, Shuta Aketa, Junji Fukumori, Takaaki Mitsui, Yuki Shiraishi, Ryuta Matsuoka, Naoki Mori, Rinsei Tei, Yasushi Shin, Yasushi Motoyama","doi":"10.3171/CASE24383","DOIUrl":"10.3171/CASE24383","url":null,"abstract":"<p><strong>Background: </strong>Extracranial carotid artery aneurysms (ECAAs) are rare, and treatment guidelines are lacking. Few reports on endovascular treatments performed for ECAAs exist.</p><p><strong>Observations: </strong>A 73-year-old woman with a left giant cervical internal carotid artery aneurysm was treated with overlapping closed-cell stents. The aneurysm regrew 1 year after the treatment, and then a covered stent was deployed. Angioscopy was performed to confirm neointimal development to determine the appropriate stent position before the retreatment, and it revealed that the stent struts were embedded in thick neointima for the most part but that the neointima was thin around the aneurysm neck. Multiple holes connecting to the aneurysm were observed between the stent struts. A covered stent overlapped inside the closed-cell stents, and blood flow into the aneurysm completely disappeared.</p><p><strong>Lessons: </strong>When deploying the covered stent for recurrent aneurysms, angioscopy is useful for confirming neointimal development and determining the appropriate stent length and position. Angioscopic observations suggest that using stents with a higher mesh density and smaller pore size can reduce the neck hole size of the aneurysm and may achieve complete occlusion of the aneurysm. https://thejns.org/doi/10.3171/CASE24383.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 12","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418031/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142304904","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Daniel D Cummins, Hyun Joo Park, Jenna Tosto-Mancuso, David Putrino, Fedor E Panov, Christopher P Kellner, Roshini Kalagara, Margaret H Downes
{"title":"Letter to the Editor Response.","authors":"Daniel D Cummins, Hyun Joo Park, Jenna Tosto-Mancuso, David Putrino, Fedor E Panov, Christopher P Kellner, Roshini Kalagara, Margaret H Downes","doi":"10.3171/CASE24305","DOIUrl":"10.3171/CASE24305","url":null,"abstract":"","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 12","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418029/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142304905","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Abdullah K Afridi, Alexander G Steele, Catherine Martin, Dimitry G Sayenko, Sean M Barber
Background: Spinal cord stimulation (SCS) has demonstrated potential as a therapy to enhance motor functional recovery after spinal cord injury (SCI). Epidural SCS for motor recovery is traditionally performed via the dorsal electrode. While ventral epidural stimulation may provide more direct and specific stimulation of the ventral motor neurons involved in motor control, it is largely unstudied, and its role in motor recovery after SCI is unclear. In order to profile the safety and feasibility of ventral epidural spinal stimulation (VSS), the authors present a patient who underwent VSS following a corpectomy to treat SCI related to metastatic epidural cord compression.
Observations: A patient underwent transpedicular corpectomy for spinal cord decompression, as well as the placement of 2 ventral epidural electrodes, followed by concurrent physical therapy and ventral epidural stimulation. He was nonambulatory preoperatively but was able to walk over 300 feet with the assistance of a rolling walker at the conclusion of the 3-week study period. VSS was noted to produce improvements in muscle contraction when stimulation was on.
Lessons: VSS appears to be safe, feasible, and well tolerated. VSS, as compared to standard-of-care therapy for SCI, can be used in conjunction with physical therapy and may lead to improvements in motor function. https://thejns.org/doi/10.3171/CASE24155.
{"title":"Ventral epidural stimulation for motor recovery after spinal cord injury: illustrative case.","authors":"Abdullah K Afridi, Alexander G Steele, Catherine Martin, Dimitry G Sayenko, Sean M Barber","doi":"10.3171/CASE24155","DOIUrl":"10.3171/CASE24155","url":null,"abstract":"<p><strong>Background: </strong>Spinal cord stimulation (SCS) has demonstrated potential as a therapy to enhance motor functional recovery after spinal cord injury (SCI). Epidural SCS for motor recovery is traditionally performed via the dorsal electrode. While ventral epidural stimulation may provide more direct and specific stimulation of the ventral motor neurons involved in motor control, it is largely unstudied, and its role in motor recovery after SCI is unclear. In order to profile the safety and feasibility of ventral epidural spinal stimulation (VSS), the authors present a patient who underwent VSS following a corpectomy to treat SCI related to metastatic epidural cord compression.</p><p><strong>Observations: </strong>A patient underwent transpedicular corpectomy for spinal cord decompression, as well as the placement of 2 ventral epidural electrodes, followed by concurrent physical therapy and ventral epidural stimulation. He was nonambulatory preoperatively but was able to walk over 300 feet with the assistance of a rolling walker at the conclusion of the 3-week study period. VSS was noted to produce improvements in muscle contraction when stimulation was on.</p><p><strong>Lessons: </strong>VSS appears to be safe, feasible, and well tolerated. VSS, as compared to standard-of-care therapy for SCI, can be used in conjunction with physical therapy and may lead to improvements in motor function. https://thejns.org/doi/10.3171/CASE24155.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 12","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418034/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142304907","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
{"title":"Letter to the Editor. The need for a documented therapeutic effect before recommending vagus nerve stimulation for recovery after cerebral bleeding.","authors":"Josef Finsterer","doi":"10.3171/CASE24263","DOIUrl":"10.3171/CASE24263","url":null,"abstract":"","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 12","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418030/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142304906","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Background: Chondrosarcoma is typically a slow-growing tumor, and intratumoral hemorrhage is rare. Acute brainstem hemorrhage due to chondrosarcoma has rarely been reported.
Observations: A 77-year-old man presented with the sudden onset of headache and vomiting followed by a declining level of consciousness, progressive right hemiparesis, and left ophthalmoplegia. Magnetic resonance imaging showed pontine hemorrhage and a mass in the retroclival space compressing the brainstem. Emergency endoscopic endonasal surgery was performed. Intraoperative observation revealed that a hematoma was located in the pons and subdural space around the tumor mass, suggesting that the hematoma had likely been caused by the rupture of small vessels around the pons, not by intratumoral hemorrhage. The pathological diagnosis was chondrosarcoma. The patient recovered well and underwent radiotherapy.
Lessons: This report describes a case of sudden neurological deterioration due to hemorrhage in a patient with chondrosarcoma of the skull base. An emergency endoscopic endonasal approach for mass reduction and hematoma removal was effective in the acute setting. This approach revealed the suspected etiology of peritumoral hemorrhage, not intratumoral hemorrhage. https://thejns.org/doi/10.3171/CASE2460.
{"title":"Acute peritumoral brainstem hemorrhage of a clival chondrosarcoma successfully treated via endoscopic endonasal surgery: illustrative case.","authors":"Koki Mitani, Masahiro Tanji, Yuji Kitada, Masahiro Kikuchi, Akihiko Sugimoto, Masakazu Fujimoto, Yoshiki Arakawa","doi":"10.3171/CASE2460","DOIUrl":"10.3171/CASE2460","url":null,"abstract":"<p><strong>Background: </strong>Chondrosarcoma is typically a slow-growing tumor, and intratumoral hemorrhage is rare. Acute brainstem hemorrhage due to chondrosarcoma has rarely been reported.</p><p><strong>Observations: </strong>A 77-year-old man presented with the sudden onset of headache and vomiting followed by a declining level of consciousness, progressive right hemiparesis, and left ophthalmoplegia. Magnetic resonance imaging showed pontine hemorrhage and a mass in the retroclival space compressing the brainstem. Emergency endoscopic endonasal surgery was performed. Intraoperative observation revealed that a hematoma was located in the pons and subdural space around the tumor mass, suggesting that the hematoma had likely been caused by the rupture of small vessels around the pons, not by intratumoral hemorrhage. The pathological diagnosis was chondrosarcoma. The patient recovered well and underwent radiotherapy.</p><p><strong>Lessons: </strong>This report describes a case of sudden neurological deterioration due to hemorrhage in a patient with chondrosarcoma of the skull base. An emergency endoscopic endonasal approach for mass reduction and hematoma removal was effective in the acute setting. This approach revealed the suspected etiology of peritumoral hemorrhage, not intratumoral hemorrhage. https://thejns.org/doi/10.3171/CASE2460.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 12","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11418032/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142304903","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Paul M Harary, Yusuke S Hori, Amit R L Persad, Armine Tayag, Louisa Ustrzynski, Sara C Emrich, Elham Rahimy, David J Park, Gordon Li, Steven D Chang
Background: While genetic testing of tumors is commonly used to inform the selection of systemic therapies, there is limited evidence for the application of radiotherapy for brain cancer. Recent studies have shown that Kelch-like ECH-associated protein 1 (KEAP1), a key regulator of cellular responses to oxidative and electrophilic stress, is associated with radioresistance in multiple cancer types. Several studies have reported the clinical significance of KEAP1 mutation in brain metastasis; however, the effect of KEAP1 mutations on radioresponse in meningioma has never been reported.
Observations: The authors present the case of a 40-year-old female with a KEAP1 mutation-positive atypical meningioma that was initially treated with resection followed by intensity-modulated radiation therapy (IMRT). Recurrence was observed at 15 months, requiring reoperation and adjuvant stereotactic radiosurgery (SRS). An excellent treatment response was observed at 7 months post-SRS with an improvement in reported symptoms, although bevacizumab was required for the resolution of radiation necrosis observed 2 months post-SRS.
Lessons: To the authors' knowledge, this is the first report of KEAP1-mutant meningioma, including its clinical course after comprehensive management. Notably, treatment included multimodal radiotherapy with IMRT followed by SRS. SRS led to an excellent treatment response at the 7-month follow-up. However, radiation necrosis developed after both radiotherapy treatments, suggesting that radiological modification can be beneficial in patients with KEAP1 mutations. https://thejns.org/doi/10.3171/CASE24387.
{"title":"KEAP1-mutant atypical meningioma: illustrative case.","authors":"Paul M Harary, Yusuke S Hori, Amit R L Persad, Armine Tayag, Louisa Ustrzynski, Sara C Emrich, Elham Rahimy, David J Park, Gordon Li, Steven D Chang","doi":"10.3171/CASE24387","DOIUrl":"https://doi.org/10.3171/CASE24387","url":null,"abstract":"<p><strong>Background: </strong>While genetic testing of tumors is commonly used to inform the selection of systemic therapies, there is limited evidence for the application of radiotherapy for brain cancer. Recent studies have shown that Kelch-like ECH-associated protein 1 (KEAP1), a key regulator of cellular responses to oxidative and electrophilic stress, is associated with radioresistance in multiple cancer types. Several studies have reported the clinical significance of KEAP1 mutation in brain metastasis; however, the effect of KEAP1 mutations on radioresponse in meningioma has never been reported.</p><p><strong>Observations: </strong>The authors present the case of a 40-year-old female with a KEAP1 mutation-positive atypical meningioma that was initially treated with resection followed by intensity-modulated radiation therapy (IMRT). Recurrence was observed at 15 months, requiring reoperation and adjuvant stereotactic radiosurgery (SRS). An excellent treatment response was observed at 7 months post-SRS with an improvement in reported symptoms, although bevacizumab was required for the resolution of radiation necrosis observed 2 months post-SRS.</p><p><strong>Lessons: </strong>To the authors' knowledge, this is the first report of KEAP1-mutant meningioma, including its clinical course after comprehensive management. Notably, treatment included multimodal radiotherapy with IMRT followed by SRS. SRS led to an excellent treatment response at the 7-month follow-up. However, radiation necrosis developed after both radiotherapy treatments, suggesting that radiological modification can be beneficial in patients with KEAP1 mutations. https://thejns.org/doi/10.3171/CASE24387.</p>","PeriodicalId":94098,"journal":{"name":"Journal of neurosurgery. Case lessons","volume":"8 11","pages":""},"PeriodicalIF":0.0,"publicationDate":"2024-09-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11404106/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"142304996","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}