RESUMEN
BACKGROUND: Although arterial stiffness is known as a biomarker for cardiovascular events and stroke, there is limited information in the literature regarding the stiffness of intracranial aneurysms. In this study, we aim to assess the stiffness of intracranial aneurysms using 4D Flow MRI. METHODS: A total of 27 aneurysms in 25 patients with internal carotid artery aneurysms were included in this study. Using 4D Flow MRI, we measured the arterial pulse wave form during a cardiac cycle at planes proximal and distal to the target aneurysm. The damping of these waveforms through the aneurysm was defined as the aneurysm damping index (ADI) and compared to the contralateral side. We also investigated the clinical factors related to the ADI. RESULTS: ADI assessment was successful in all cases. The average ADI was 1.18±0.28, which was significantly larger than 1.0 (P = 0.0027 [t-test]). The ADI on the aneurysm side was larger than on the contralateral side (1.19±0.30 vs 1.05±0.17, P = 0.029 [t-test]). On multivariate analysis, the use of beta-blockers (ß=0.46, P = 0.015) and smoking history (ß=-0.22, P = 0.024) showed a significant correlation with ADI. CONCLUSION: We have proposed a novel method to observe arterial pulse wave dumping through intracranial aneurysm using 4D Flow MRI. The damping can be quantitatively observed, and the ADI has correlations with clinical factors such as antihypertensive drugs and smoking. Further studies should focus more on evaluating aneurysm stiffness and its clinical applications.
RESUMEN
OBJECTIVE: The characteristics of pregnancy and delivery in patients with moyamoya disease (MMD) remain unclear. We retrospectively investigated perinatal outcomes in patients with MMD to evaluate the risks associated to this condition. MATERIALS AND METHODS: Clinical data of women with MMD who delivered at the University of Tokyo Hospital between 2000 and 2021 were collected. Maternal characteristics including genetic data, obstetric complications, method of delivery and anesthesia, neonatal outcomes, neurological events during pregnancy, delivery, and postpartum course, were reviewed. RESULTS: Thirteen pregnancies with MMD were identified. The median maternal age was 30 years. The initial clinical symptoms were identified as transient ischemic attack, infarction, and headache. Eight patients had a history of bypass surgery. The median gestational age at delivery was 37 weeks. DNA samples were collected from five patients, responsible for six pregnancies. Of these six cases, five had the RNF213 c.14429G > A (p.Arg4810Lys) heterozygous variant. Of the 13 pregnancies, seven had hypertensive disorder of pregnancy (HDP). Additionally, three of five pregnancy cases with RNF213 p.Arg4810Lys heterozygous variant presented with HDP. Nine patients underwent cesarean section, and four delivered vaginally with epidural anesthesia. One case of ischemic stroke was confirmed during the postpartum period. Regarding newborns, neither Apgar scores lower than 7 nor neonatal intensive care unit admissions were reported. CONCLUSIONS: This study suggests that the frequency of HDP is higher in patients with MMD compared to those with normal pregnancies. Strict blood pressure control should be performed in patients with MMD during pregnancy and postpartum period.
RESUMEN
BACKGROUND: Vestibular schwannomas (VS) present at variable size with heterogeneous symptomatology. Modern treatment paradigms for large VS include gross total resection, subtotal resection (STR) in combination with observation, and/or radiation to achieve optimal function preservation, whereas treatment is felt to be both easier and safer for small VS. The objective is to better characterize the presentation and surgical outcomes of large and small VS. METHODS: We collected data of patients who had surgically treated VS with a posterior fossa diameter of 4.0 cm or larger (large tumor group, LTG) and smaller than 1.0 cm in cisternal diameter (small tumor group, STG). Statistical significance was defined as p < 0.05. RESULTS: LTG included 48 patients (average tumor size: 44.9 mm) and STG 38 (7.9 mm). Patients in STG presented more frequently with tinnitus and sudden hearing loss. Patients in LTG underwent more STR than STG (50.0% vs. 2.6%, p < 0.0001). LTG had more complications (31.3% vs. 13.2%, p = 0.049). Postoperative facial nerve function in STG was significantly better than LTG. STG had better hearing preoperatively (p < 0.0001) and postoperatively than LTG (p = 0.0002). Postoperative headache was more common in STG (13.2% vs. 2.1%, p = 0.045). The rate of recurrence/progression needing treatment was not statistically different between the groups (12.5% in LTG vs. 7.9% in STG, p = 0.49). Those patients who required periprocedural cerebrospinal fluid diversion had higher risk of infection (20.8% vs 4.8%, p = 0.022). CONCLUSION: Large and small VS present differently. LTG showed more unsatisfactory outcomes in facial nerve function and postoperative hearing despite maximal efforts undertaken toward function-preservation strategy; however, similar tumor control was achieved.
Asunto(s)
Neuroma Acústico , Cefalea , Audición , Humanos , Recurrencia Local de Neoplasia , Neuroma Acústico/cirugía , Procedimientos NeuroquirúrgicosRESUMEN
Distal anterior cerebral artery (DACA) aneurysms are rare, accounting for 1-9% of all intracranial aneurysms. Previous systematic reviews have highlighted that given the markedly increased incidence of major complications after endovascular treatment, microsurgical clipping is the more attractive treatment option with generally excellent clinical outcomes. Subcallosal DACA aneurysms constitute a rare subset of these aneurysms, requiring special anatomic considerations-particularly with regard to the approach. The aim of this study is to review the technical nuances of microsurgical treatment of subcallosal DACA aneurysms, including review of contemporary techniques through presentation of a microneurosurgical operative video. This is a retrospective case series and intraoperative microsurgical videos review. Three subcallosal DACA aneurysms were identified via retrospective query of our institutional neurosurgical database from December 2017 to May 2018. Two were female; median age was 74 years (range 70-83); all 3 underwent bifrontal craniotomy via bicoronal skin incision for aneurysm clipping. Aneurysms were located in left pericallosal-callosomarginal artery junction, bifurcation of azygos A2, and pericallosal artery related with azygos A2, and the anterior interhemispheric approach was used in all 3 operations. No acute stroke, hemorrhage, or major complications occurred, and all patients remained neurologically intact at the time of last follow-up (median 3 months, range 1-6). Although DACA aneurysms are rare, they represent an important variant for cerebrovascular neurosurgeons where microsurgical clipping can have better angiographic outcomes than endovascular treatment. Detail-oriented anterior interhemispheric arachnoid dissection through bifrontal craniotomy with its lower margin sitting at the superior orbital rim maximizes safe and effective clipping of subcallosal DACA aneurysms.
Asunto(s)
Aneurisma Intracraneal/cirugía , Microcirugia/métodos , Anciano , Anciano de 80 o más Años , Arteria Cerebral Anterior/cirugía , Craneotomía , Disección , Femenino , Humanos , Masculino , Estudios Retrospectivos , Resultado del TratamientoRESUMEN
Intracranial epidermoid cysts are benign cystic lesions that typically exhibit slow growth. Their malignant transformation into squamous cell carcinoma is rare. We report a 77-year-old woman who was admitted to our hospital because of a near-drowning incident due to a seizure sustained in her bathtub. Magnetic resonance imaging(MRI)revealed an extra-axial tumor occupying the right cerebellopontine angle. The lesion appeared hyperintense in diffusion-weighted images and exhibited contrast enhancement after gadolinium injection. Cerebrospinal fluid examination revealed noninfectious meningitis, presumably due to the ruptured epidermoid cyst. Tumor resection was performed and histopathological examination revealed squamous cell carcinoma, which was indicative of malignant transformation of the cyst. The patient underwent adjuvant radiotherapy and has no signs of recurrence 9 months postsurgery. Rapid neurological deterioration and contrast enhancement on MRI are key signs of malignant transformation of epidermoid cysts.
Asunto(s)
Ángulo Pontocerebeloso/cirugía , Quiste Epidérmico , Anciano , Transformación Celular Neoplásica , Neoplasias Cerebelosas , Quiste Epidérmico/cirugía , Femenino , Humanos , Imagen por Resonancia Magnética , Recurrencia Local de NeoplasiaRESUMEN
PURPOSE: Cavernous sinus syndrome is a rare phenomenon, characterized by simultaneous neuropathies of cranial nerves III-VI. Various pathological processes have been reported as precipitating etiologies, including infection, inflammation, vascular lesions, and neoplasms. PURPOSE: We report a unique case series of cavernous sinus syndrome attributable to prolonged Trendelenburg or prone positioning during non-cranial procedures and review the pertinent literature to enlighten on this rare but catastrophic phenomenon. METHODS: Retrospective case series. RESULTS: In the past year we encountered two patients who presented with acute cavernous sinus syndrome upon awakening from non-cranial operations. One patient underwent an extensive urologic resection of a bladder malignancy positioned in Trendelenburg for approximately 4 h. The second patient underwent a lumbar laminectomy and discectomy in prone position. Both patients were discovered to have infarcted large pituitary macroadenomas as the etiology of their acute ophthalmoplegias, and transnasal, transsphenoidal resection was performed acutely to decompress the cavernous sinus contents. Pathologic analysis of the resected specimens in each case confirmed necrotic, infarcted pituitary adenoma. Both patients made a complete recovery with no evidence of residual or recurrent tumor in short term follow-up. CONCLUSION: We report a brief case series of acute cavernous sinus syndrome resulting from dependent positioning during non-cranial operations in patients with pituitary macroadenoma. Although rare, this highlights a potential danger of "head down" positioning in patients with intracranial pathology-particularly in or around the sella and cavernous sinus. Despite multiple cranial neuropathies upon presentation, both patients made complete recovery following surgical decompression of the cavernous sinuses.
Asunto(s)
Seno Cavernoso/cirugía , Neoplasias Hipofisarias/cirugía , Seno Cavernoso/patología , Trombosis del Seno Cavernoso/cirugía , Humanos , Procedimientos Neuroquirúrgicos/métodos , Estudios RetrospectivosRESUMEN
BACKGROUND: The frontal bridging vein, which is the venous drainage route of the frontal cortex into the superior sagittal sinus (SSS), sometimes poses an obstacle in the anterior interhemispheric approach during surgery for anterior cerebral artery aneurysms. Although severe complications including venous infarction or edema due to damage to the bridging vein are well known, only a few reports have discussed how to avoid venous injury when we must sacrifice the bridging vein to obtain an appropriate surgical field. This report describes a microvascular technique performed in two patients who underwent rerouting of the bridging vein to obtain an appropriate anterior interhemispheric surgical corridor to treat a ruptured anterior cerebral artery aneurysm. The hindering bridging vein was resected from the entrance to the SSS and anastomosed toward the adjacent cortical vein. METHODS: A 65-year-old male and a 43-year-old male were admitted to our hospital for sudden headache. Computed tomography, magnetic resonance angiogram, or digital subtraction angiography demonstrated a subarachnoid hemorrhage and an anterior cerebral artery aneurysm in both patients. In both cases, a relatively robust bridging vein, which appeared problematic to sacrifice, was draining into the SSS, resulting in a limited surgical corridor. Thus, we performed cortical vein reconstruction, and the aneurysms were successfully clipped under a wider surgical view. RESULTS: We confirmed completed clipping without postoperative venous complications. One patient demonstrated patency of reconstructed venous flow by digital subtraction angiography. No apparent cognitive impairment was seen in either patient. CONCLUSIONS: This technique may be useful for obtaining an appropriate surgical corridor when the frontal bridging vein may be damaged.
Asunto(s)
Lóbulo Frontal/cirugía , Aneurisma Intracraneal/cirugía , Hemorragia Subaracnoidea/cirugía , Seno Sagital Superior/cirugía , Procedimientos Quirúrgicos Vasculares/métodos , Adulto , Anciano , Angiografía de Substracción Digital , Lóbulo Frontal/irrigación sanguínea , Lóbulo Frontal/diagnóstico por imagen , Humanos , Aneurisma Intracraneal/diagnóstico por imagen , Masculino , Microcirugia , Hemorragia Subaracnoidea/diagnóstico por imagen , Seno Sagital Superior/diagnóstico por imagen , Instrumentos Quirúrgicos , Tomografía Computarizada por Rayos XRESUMEN
This video demonstrates combined two separate craniotomies for two difficult unruptured cerebral aneurysms. The anterior communicating artery (ACOM) aneurysm existed at a high position, projected posteriorly, and thus necessitated an interhemispheric approach. Left middle cerebral artery (MCA) aneurysm with complex figure was treated through a separate pterional approach. Meticulous micro-cisternal opening under high magnification enabled safe and effective exposure of both aneurysms with minimal brain retraction, which alleviated brain damage as shown in postoperative images. The video can be found here: http://youtu.be/mBYsaAVekCA .
Asunto(s)
Craneotomía/métodos , Lateralidad Funcional , Aneurisma Intracraneal/cirugía , Procedimientos Neuroquirúrgicos/métodos , Humanos , Masculino , Persona de Mediana Edad , PosturaRESUMEN
BACKGROUND: Acute extracranial internal carotid artery (ICA) occlusion by a huge cardiogenic embolus is rare, but can be catastrophic. METHODS: Seven patients with acute ischemic stroke due to embolic occlusion of extracranial ICA who underwent emergent cervical surgical embolectomy were retrospectively reviewed. Diagnosis was made in six patients with magnetic resonance imaging (MRI) with optional digital subtraction angiography (DSA), while computed tomography (CT) and DSA were used in one patient with an implanted pacemaker. Clinical outcomes, including recanalization rate, recanalization time, complications, modified Rankin scale (mRS) at 3 months, and National Institute of Health Stroke Scale (NIHSS) score improvement at 1 month were evaluated. RESULTS: Complete recanalization was obtained in seven patients (100 %). Median recanalization time from symptom onset and from start of surgery was 402 and 40 min, respectively. All seven patients showed severe left ventricular hypertrophy (LVH) according to an increased cardiothoracic ratio (CTR) ≥50 %. Complications included recurrence of cardioembolic stroke with the right middle cerebral artery occlusion, minimal expansion of infarction, and aggravation of heart failure, each in one patient, respectively. Four (57.1 %) patients had a history or postoperative recurrence of cardioembolic stroke. Median NIHSS at 1 month was 2 (range, 0-30). Median mRS at 3 months was 2 (range, 0-5). Five patients (71.4 %) had a favorable outcome (mRS2). CONCLUSIONS: Cervical surgical embolectomy for acute extracranial ICA occlusion resulted in a high complete recanalization rate with an acceptable safety profile. A possible association between severe cardiac illness and huge embolus occluding proximal large artery was suggested.
Asunto(s)
Arteriopatías Oclusivas/cirugía , Enfermedades de las Arterias Carótidas/cirugía , Arteria Carótida Interna/cirugía , Embolectomía/efectos adversos , Cardiopatías/etiología , Accidente Cerebrovascular/etiología , Anciano , Anciano de 80 o más Años , Arteria Carótida Interna/patología , Embolectomía/métodos , Femenino , Humanos , Masculino , Persona de Mediana EdadRESUMEN
BACKGROUND: The white-collar sign (WCS) is represented by the formation of neointimal tissue at the level of the aneurysm neck as the successful outcome on follow-up angiography after coil embolization. WCS has been reported only in aneurysms treated with Matrix® coils. This is the first study to report WCS emergence in aneurysms treated with bare platinum coils, and potential factors associated with WCS emergence were evaluated. METHOD: Total 130 unruptured (female: male ratio, 100: 30; mean age, 60 years) cerebral aneurysms were treated with coil embolization. Embolization status was assessed immediately and 1 year after treatment, and emergence of WCS in follow-up angiography was assessed. We evaluated the association between WCS emergence and aneurysm location, dome diameter, neck diameter, dome-neck ratio, and type of coil used (bare platinum or bioactive). RESULTS: WCS appeared in nine aneurysms (6.9%), of which six were treated only with bare platinum coils. Neck diameter was significantly smaller in the WCS-positive group than in the WCS-negative group. The proportion of aneurysms treated with bioactive coils was not significantly different between the groups. Immediate embolization status in the WCS-positive group tended to be slightly better than that in the WCS-negative group. No aneurysmal morphological characteristics other than small neck diameter were associated with WCS emergence. CONCLUSIONS: WCS is not specific to bioactive coil usage. Small neck diameter was significantly associated with WCS emergence in our series. Further investigations to clarify the predictors of WCS will contribute to progress of aneurysmal embolization.
Asunto(s)
Embolización Terapéutica , Aneurisma Intracraneal/terapia , Anciano , Anciano de 80 o más Años , Aneurisma Roto/diagnóstico , Aneurisma Roto/terapia , Angiografía Cerebral/métodos , Embolización Terapéutica/métodos , Femenino , Humanos , Aneurisma Intracraneal/diagnóstico , Masculino , Persona de Mediana Edad , Platino (Metal)/uso terapéutico , Resultado del TratamientoRESUMEN
BACKGROUND: Executive functions are complex cognitive control functions that include cognitive flexibility, inhibition (self-control, self-regulation), and working memory. Bilateral frontal lobe ischemia is associated with cognitive impairment, especially in the context of dysexecutive syndrome. This report describes two patients who underwent bilateral anterior cerebral artery (ACA) reconstruction by A3-A3 anastomosis in conjunction with superficial temporal artery (STA)-radial artery (RA) graft-A3 bypass to treat bilateral ACA steno-occlusive hemodynamic ischemia accompanying cognitive and executive dysfunction. METHOD: A 74-year-old woman and a 73-year-old woman were admitted to our hospital for unilateral cerebral infarction in the frontal lobe. Magnetic resonance angiogram (MRA) demonstrated severe bilateral ACA steno-occlusive pathology in both patients. Considering the presence of impaired cognitive function with dysexecutive syndrome as well as hemodynamic compromise shown by single photon emission computed tomography (SPECT), we proceeded with A3-A3 anastomosis in conjunction with STA-RA-A3 bypass. Various neuropsychiatric tests were performed before and after the surgery. RESULTS: We confirmed good bypass patency without periprocedural complications. One patient recovered from apallic and bedridden status and regained ambulatory condition and ability to take in an oral diet. Another patient demonstrated improved scores in several cognitive tests with some persistent executive dysfunction. CONCLUSIONS: Bilateral ACA revascularization was technically feasible in two patients. This bypass surgery could have some positive effects in some basic cognitive function, such as memory, attention, and concentration by bilateral ACA hemodynamic improvement, although executive function, which is specific to prefrontal function, might not be reversible.
Asunto(s)
Arteria Cerebral Anterior/cirugía , Infarto Cerebral/cirugía , Revascularización Cerebral/métodos , Trastornos del Conocimiento/cirugía , Arteria Radial/trasplante , Arterias Temporales/trasplante , Anciano , Infarto Cerebral/etiología , Trastornos Cerebrovasculares/complicaciones , Trastornos Cerebrovasculares/cirugía , Trastornos del Conocimiento/etiología , Constricción Patológica , Función Ejecutiva , Femenino , HumanosRESUMEN
BACKGROUND: Transcarotid artery revascularization (TCAR) has emerged as an alternative to carotid artery stenting (CAS). TCAR demonstrated its superiority by avoiding femoral artery puncture and establishing proximal protection without crossing the stenotic lesion. In the TCAR era, we focused on the possibility of a trans-distal radial approach (DRA). A balloon-guide catheter was navigated via DRA to establish proximal protection before lesion crossing. The forearm subcutaneous vein was used as the flow-reversal circuit. METHODS: Six internal carotid artery stenosis patients underwent CAS using "the forearm flow reversal technique." Every procedure was performed under continuous flow reversal from the common carotid artery to the forearm cephalic vein. RESULTS: Successful revascularization was achieved without ischemic or access-site complications. The distal radial artery was patent at discharge in all cases. CONCLUSIONS: Trans-distal radial CAS with forearm flow reversal is a feasible and less invasive technical option.
Asunto(s)
Estenosis Carotídea , Procedimientos Endovasculares , Accidente Cerebrovascular , Humanos , Estenosis Carotídea/cirugía , Estenosis Carotídea/complicaciones , Antebrazo/cirugía , Arteria Radial/cirugía , Factores de Riesgo , Stents/efectos adversos , Arteria Carótida Común , Resultado del Tratamiento , Estudios Retrospectivos , Accidente Cerebrovascular/etiología , Procedimientos Endovasculares/efectos adversosRESUMEN
BACKGROUND: The emergence of flow disruptors has brought a dynamic transition in the selection of treatment for middle cerebral artery (MCA) aneurysms, and the number of MCA aneurysms clipped is acceleratingly decreasing. Still, retreatment after endovascular treatment is still a dilemma, which may necessitate surgical clipping. It is all the more important to elucidate characteristics of MCA aneurysms that make clipping unfavorable. Thus, the practical characteristics of MCA aneurysms treated endovascularly in a clip-favored institution before the usage of flow disruption devices were investigated. METHODS: This is a retrospective, single-center observational study. The clinical and imaging characteristics of treated MCA aneurysms from January 2012 to May 2022 were analyzed. RESULTS: A total of 83 aneurysms were included; 70 aneurysms (84%) were clipped, and 13 (16%) were treated endovascularly. Eighteen aneurysms (22%) were ruptured (clipping, 12; endovascular, 6). The reasons for an endovascular treatment were as follows: distal (3); subacutely ruptured with burdens of spasm (2); multiple aneurysms (6: another clipped in 3, coiled in 1, and conservatively managed in 2); no access to the operating room due to COVID-19 (1); and retreatment after coiling (1). Endovascular group aneurysms were smaller (maximum diameter 5.2 vs 7.3 mm, p < 0.01, as well as dome, neck, and height) without differences in the dome/neck and aspect (height/neck) ratios. In a subgroup analysis of 78 MCA bifurcation aneurysms, the endovascular group was still smaller (dome 4.4 vs 5.8 mm, p = 0.025; neck 2.8 vs 3.9, p = 0.03). CONCLUSION: In a limited series from a clip-favored institution before the flow disruption era, factors guided to endovascular treatments on MCA aneurysms were rather anatomical and clinical factors such as distal location, subacutely ruptured, multiple, or retreatment after coiling, than morphological factors such as dome/neck and aspect ratios albeit smaller size.
RESUMEN
Three-dimensional vessel model reconstruction from patient-specific magnetic resonance angiography (MRA) images often requires some manual maneuvers. This study aimed to establish the deep learning (DL)-based method for vessel model reconstruction. Time of flight MRA of 40 patients with internal carotid artery aneurysms was prepared, and three-dimensional vessel models were constructed using the threshold and region-growing method. Using those datasets, supervised deep learning using 2D U-net was performed to reconstruct 3D vessel models. The accuracy of the DL-based vessel segmentations was assessed using 20 MRA images outside the training dataset. The dice coefficient was used as the indicator of the model accuracy, and the blood flow simulation was performed using the DL-based vessel model. The created DL model could successfully reconstruct a three-dimensional model in all 60 cases. The dice coefficient in the test dataset was 0.859. Of note, the DL-generated model proved its efficacy even for large aneurysms (> 10 mm in their diameter). The reconstructed model was feasible in performing blood flow simulation to assist clinical decision-making. Our DL-based method could successfully reconstruct a three-dimensional vessel model with moderate accuracy. Future studies are warranted to exhibit that DL-based technology can promote medical image processing.
Asunto(s)
Aprendizaje Profundo , Imagenología Tridimensional , Angiografía por Resonancia Magnética , Humanos , Angiografía por Resonancia Magnética/métodos , Imagenología Tridimensional/métodos , Masculino , Femenino , Persona de Mediana Edad , Anciano , Adulto , Arteria Carótida Interna/diagnóstico por imagen , Procesamiento de Imagen Asistido por Computador/métodos , Aneurisma Intracraneal/diagnóstico por imagen , Aneurisma Intracraneal/fisiopatologíaRESUMEN
BACKGROUND: Moyamoya disease is a bilateral steno-occlusive disease involving the cerebral vasculature. While some patients are affected by procedure-related ipsilateral ischemia, ischemic complications contralateral to the revascularization are rarely observed. METHODS: We retrospectively investigated 135 hemispheres (103 patients) that underwent revascularization in our institution between April 2006 and September 2022. Revascularization surgery comprised single superficial temporal artery-middle cerebral artery anastomosis and encephalo-myo-synangiosis. Certain patients aged under 10 years underwent indirect revascularization. Bilateral revascularization was performed with an interval of >3 months. Medical records and neuroimages were reviewed, and patients with contralateral ischemic complications were identified. Some cases underwent genetic analysis. RESULTS: The mean age was 34.5 (range: 5-71) years, and 95 cases (70.4%) were in women. Of the 102 cases examined for the RNF213 c.14429 G > A (p.Arg4810Lys) variant, 33 (32.4%) and 69 (67.6%) showed the GG and GA genotype, respectively. Three cases (2.2%, all female, age range 44-71 years) were complicated with contralateral infarction. The infarcted area distributions of the 2 cases with RNF213 c.14429 G > A variant were patchy and peripheral. The other case showed on magnetic resonance imaging (MRI) angiography total occlusion of the internal carotid artery where patency had been confirmed preoperatively. CONCLUSIONS: Contralateral ischemia after revascularization occurred in 2.2% of cases. We classified them into peripheral and central types: peripheral type, an infarction owing to hemodynamic insufficiency or intracranial blood flow redistribution; central type, total occlusion of the contralateral internal carotid artery. Intensive preoperative management can minimize the risk of peripheral types, and neurosurgeons should beware of severe central types.
Asunto(s)
Revascularización Cerebral , Enfermedad de Moyamoya , Humanos , Femenino , Anciano , Adulto , Persona de Mediana Edad , Enfermedad de Moyamoya/diagnóstico por imagen , Enfermedad de Moyamoya/cirugía , Enfermedad de Moyamoya/complicaciones , Estudios Retrospectivos , Revascularización Cerebral/métodos , Isquemia/complicaciones , Infarto , Adenosina Trifosfatasas , Ubiquitina-Proteína LigasasRESUMEN
BACKGROUND: Recent anatomical studies have reported the feasibility of the endoscopic endonasal approach to the middle fossa. However, its clinical applicability has been discussed in only a few cases. This article describes the case of a middle fossa epidural abscess successfully drained through a fully endoscopic endonasal corridor and discusses the key technical points. OBSERVATIONS: The authors describe an 8-year-old boy who presented with worsening headache, fatigue, emesis, and fever and was diagnosed with a left middle fossa epidural abscess associated with sphenoid sinusitis. Following endoscopic sinus surgery performed by a rhinologist, the middle fossa was accessed by removing the maxillary strut through the transmaxillary transpterygoid corridor. Complete drainage of the abscess was confirmed on postoperative magnetic resonance imaging. The patient tolerated the surgery without neurological deficit and demonstrated prompt symptom improvement. He was discharged home after completing a 6-week course of antibiotic therapy and remained free from recurrence at 1 year following surgery. LESSONS: The endoscopic endonasal approach may be applicable to a middle fossa epidural abscess, resulting in prompt clinical improvement. The maxillary strut is a key structure for entering the middle fossa. https://thejns.org/doi/10.3171/CASE24288.
RESUMEN
BACKGROUND: We aimed to comprehensively analyze the epidemiology, natural history, stroke events and their risk factors, and the RNF213 p.Arg4810Lys variant in older patients with moyamoya disease (MMD). METHODS: We enrolled patients with MMD followed-up at our hospital between 2000 and 2023. Those who developed MMD at age ≥60 years or were diagnosed at a younger age and followed-up after age 60 years were included. Baseline characteristics, onset type, radiologic features, and RNF213 p.Arg4810Lys variant status were investigated. RESULTS: Among 56 patients with 100 affected hemispheres, 62 were asymptomatic, 26 experienced ischemic onset, and 12 had hemorrhagic onset. A higher incidence of anterior choroidal artery (AchA) dilatation and lower proportion of favorable modified Rankin scale scores were detected in hemorrhagic onset, whereas greater prevalence of bypass surgery in ischemic onset. Of 76 asymptomatic hemispheres at the age of 60 years, subsequent stroke events occurred in 10 hemispheres, comprising 8 hemorrhages and 2 ischemias. Risk factors for de novo hemorrhage in asymptomatic hemispheres included AchA dilatation and choroidal anastomosis. Comparison of the RNF213 p.Arg4810Lys variant status showed no significant differences in baseline characteristics, onset types, or imaging findings, except for a higher percentage of patients in the GA group with a family history of MMD. CONCLUSIONS: Hemorrhagic events were the most prevalent and prognostically deteriorating factors in older patients with MMD aged ≥60 years. AchA dilatation and choroidal anastomosis were predictors of de novo hemorrhage in asymptomatic nonsurgical hemispheres in older patients with MMD.
Asunto(s)
Enfermedad de Moyamoya , Accidente Cerebrovascular , Humanos , Enfermedad de Moyamoya/epidemiología , Enfermedad de Moyamoya/genética , Enfermedad de Moyamoya/cirugía , Enfermedad de Moyamoya/diagnóstico por imagen , Enfermedad de Moyamoya/complicaciones , Masculino , Femenino , Persona de Mediana Edad , Factores de Riesgo , Anciano , Accidente Cerebrovascular/epidemiología , Accidente Cerebrovascular/etiología , Adulto , Ubiquitina-Proteína Ligasas/genética , Adenosina Trifosfatasas/genética , AdolescenteRESUMEN
OBJECTIVE: The genetic basis underlying the pathophysiology of quasi-moyamoya disease (qMMD) is unclear. Herein, the authors aimed to comprehensively analyze genetic variants in qMMD and investigate their association with clinical phenotypes, focusing on RNF213 and other moyamoya angiopathy (MMA)-related genes. METHODS: The authors evaluated 14 consecutive cases of qMMD, whose underlying conditions included autoimmune disease, head irradiation, meningitis/pachymeningitis, and Turner syndrome, and 9 cases of hyperthyroidism-associated MMD (hMMD). The frequencies of RNF213 p.Arg4810Lys in qMMD and hMMD were each compared to those in healthy controls and in patients with MMD. Whole-exome sequencing was performed, and rare variants (RVs) or damaging variants were analyzed in RNF213 and 36 MMA-related genes. RESULTS: The frequencies of p.Arg4810Lys were significantly higher in patients with qMMD (28.6%) and hMMD (33.3%) than in controls (1.1%; p < 0.001) and lower in the two former groups than in the MMD group (67.6%; p = 0.003 and 0.065, respectively). In qMMD, no significant clinical differences were observed based on the presence of p.Arg4810Lys. A novel RNF213 RV was identified in four cases with qMMD. These same cases also presented with significant worsening of intracranial main artery stenosis, which suggests a possible association between RNF213 RVs and the severe progression of qMMD. Among the 36 MMA-related genes, no variants correlated with specific phenotypes. CONCLUSIONS: While the clinical implications of p.Arg4810Lys in cases with qMMD were not identified, the study findings suggest a potential association between RNF213 RVs and the significant progression of intracranial artery stenosis. Genetic analysis should not focus solely on p.Arg4810Lys but instead consider a comprehensive analysis of RNF213 for more accurate clinical prognostication of qMMD.
RESUMEN
Robust postoperative bypass development is a characteristic of moyamoya disease (MMD); however, genetic factors mediating this phenomenon remain incompletely understood. Therefore, we aimed to elucidate the relationship between postoperative donor artery development and genetic variants. We retrospectively enrolled 63 patients (79 hemispheres) who underwent combined revascularization surgery. Postoperative development of the superficial temporal artery (STA), middle meningeal artery, and deep temporal artery (DTA) was assessed using the caliber-change ratio determined from magnetic resonance angiography measurements. We analyzed RNF213 and 36 other moyamoya angiopathy-related genes by whole-exome sequencing and extracted rare or damaging variants. Thirty-five participants carried RNF213 p.Arg4810Lys (all heterozygotes), whereas 5 had RNF213 rare variants (RVs). p.Arg4810Lys was significantly associated with postoperative DTA development, while age at surgery, hypertension, and hyperlipidemia were inversely associated. Multiple regression analysis revealed that age and p.Arg4810Lys held statistical significance (P = 0.044, coefficient - 0.015, 95% confidence interval (CI) - 0.029 to 0.000 and P = 0.001, coefficient 0.670, 95% CI 0.269 to 1.072, respectively). Those with RNF213 RV without p.Arg4810Lys exhibited a significant trend toward poor DTA development (P = 0.001). Hypertension demonstrated a significant positive association with STA development, which remained significant even after multiple regression analysis (P = 0.001, coefficient 0.303, 95% CI 0.123 to 0.482). Following Bonferroni correction for multiple comparisons, targeted analyses of RNF213 and 36 moyamoya angiopathy-related genes showed a significant association of only RNF213 p.Arg4810Lys with favorable DTA development (P = 0.001). A comprehensive analysis of RNF213, considering both p.Arg4810Lys and RVs, may provide a clearer prediction of postoperative DTA development.