Retreatment of previously treated intracranial aneurysm: Procedural complications and risk factors for complications

ObjectiveIntracranial aneurysm (IA) is a frequent vascular malformation that can be managed by endovascular treatment (EVT) or microsurgery. A previously treated IA can recanalize, which may require further treatment. The aim of our study was to evaluate procedural complications related to IA retreatment and their risk factors.MethodsAll patients retreated for IA between 2007 and 2017 in 4 hospitals were included. We retrospectively reviewed the frequency of procedural complications of IA retreatment, defined as death or ≥ 1-point increase in modified Rankin score 24 h after the procedure. We then screened for risk factors of procedural complications by comparing the characteristics of patients with and without complications.ResultsDuring the inclusion period, 4,997 IAs were treated in our 4 institutions. Of these, 237 (4.7%) were retreated. 29 (12.2%) had ≥ 1 procedural complication. However, severe complications, defined as death or dependency at 1 month, occurred only in 3 patients (1.3%). The only risk factor for complications was microsurgical clipping as retreatment.ConclusionsProcedural complications during IA retreatment were frequent but, in most cases, retreatment did not lead to death or severe disability. The only risk factor for complications of IA retreatment was clipping as retreatment. However, the design of the study did not allow any conclusion to be drawn as to the optimal means of aneurysm retreatment, and further studies are needed.     

Outcomes according to treatment using an established protocol in patients with bilateral Wilms' tumor: A national Canadian population-based study

BackgroundBilateral Wilms tumor (BWT) is a rare entity. The goal of this study is to report outcomes (overall and event-free survival, OS/EFS) of BWT in a large cohort representative of the Canadian population since 2000. We focused on the occurrence of late events (relapse or death beyond 18 months), as well as outcomes of patients treated following the only protocol specifically designed for BWT to date, AREN0534, compared to patients treated following other therapeutic schemes.MethodsData was obtained for patients diagnosed with BWT between 2001 and 2018 from the Cancer in Young People in Canada (CYP-C) database. Demographics, treatment protocols, and dates for events were collected. Specifically, we examined outcomes of patients treated according to the Children's Oncology Group (COG) protocol AREN0534 since 2009. Survival analysis was performed.Results57/816 (7%) of patients with Wilms tumor had BWT during the study period. Median age at diagnosis was 2.74 years (IQR 1.37–4.48) and 35 (64%) were female; 8/57 (15%) had metastatic disease. After a median follow-up of 4.8 years (IQR 2.8–5.7 years, range 0.2–18 years), OS and EFS were 86% (CI 73–93%) and 80% (CI 66–89%), respectively. Less than 5 events were recorded after 18 months from diagnosis. Since 2009, patients treated according to the AREN0534 protocol had a statistically significant higher OS compared to patients treated with other protocols.ConclusionsIn this large Canadian cohort of patients with BWT, OS and EFS compared favorably to the published literature. Late events were rare. Patients treated according to a disease-specific protocol (AREN0534) had improved overall survival.Type of studyOriginal article.Level of evidenceLevel IV.     

MRI-based whole-tumor radiomics to classify the types of pediatric posterior fossa brain tumor

BackgroundDifferential diagnosis between medulloblastoma (MB), ependymoma (EP) and astrocytoma (PA) is important due to differing medical treatment strategies and predicted survival. The aim of this study was to investigate non-invasive MRI-based radiomic analysis of whole tumors to classify the histologic tumor types of pediatric posterior fossa brain tumor and improve the accuracy of discrimination, using a random forest classifier.MethodsMRI images of 99 patients, with 59 MBs, 13 EPs and 27 PAs histologically confirmed by surgery and pathology before treatment, were included in this retrospective study. Registration was performed between the three sequences, and high- throughput features were extracted from manually segmented tumors on MR images of each case. The forest-based feature selection method was adopted to select the top ten significant features. Finally, the results were compared and analyzed according to the classification.ResultsThe top ten contributions according to the classifier of wavelet features all came from the ADC sequence. The random forest classifier achieved 100% accuracy on the training data and validated the best accuracy (0.938): sensitivity = 1.000, 0.948 and 0.808, specificity = 0.952, 0.926 and 1.000 for EP, MB and PA, respectively.ConclusionA random forest classifier based on the ADC sequence of the whole tumor provides more quantitative information than TIWI and T2WI in differentiating pediatric posterior fossa brain tumors. In particular, the histogram percentile value showed great superiority, which added diagnostic value in pediatric neuro-oncology.     

Delayed recurrence of spontaneous intracranial hypotension syndrome mimicking a Chiari I malformation: Case report with a review of the literature

IntroductionCerebellar tonsils descent seen on brain MRI is, along with other findings, a recognized radiological sign of possible spontaneous intracranial hypotension (SIH). The short-term outcome of SIH is usually favorable with symptoms improvement and reversibility of the low-lying tonsils. Nevertheless, data on the long-term outcome are lacking or inconsistent.Case ReportA 32-year-old woman presented to her general practitioner with a six months history of non-specific headaches. An MRI brain with gadolinium showed a 12mm tonsillar descent with no other remarkable findings. Headaches were initially managed conservatively as migraines. Following the onset of progressive upper back and shoulder pain at rest, nausea, photophobia and fogging in her vision, the patient was referred to our Department with a suspicion of symptomatic Chiari I malformation. After an in-depth anamnesis, it emerged a previous history of SIH, 14 years earlier, successfully treated conservatively in another center. A whole spine MRI confirmed the suspicion of recurrent SIH showing an anterior cervico-thoracic epidural fluid collection. The patient underwent an epidural blood patch with complete resolution of the symptoms and radiological signs.DiscussionTo our knowledge, this case is the first report of delayed recurrence of a SIH successfully treated conservatively over 10 years earlier. The etiopathogenesis and management of this rarity with literature review is discussed.ConclusionAn isolated cerebellar tonsil descent with no other remarkable findings on brain MRI and a previous history of SIH should always alert the clinician of a possible late recurrence of a CSF leak and avoid unnecessary Chiari I malformation surgical procedures.     

Kidney and contrast media: Common viewpoint of the French Nephrology societies (SFNDT,FIRN, CJN) and the French Radiological Society (SFR) following ESUR guidelines

Contrast medium administration is classically considered to cause or worsen kidney failure, but recent data may moderate this assertion. The European Society of Urogenital Radiology recently published guidelines re-evaluating the precautions before administering contrast media. Kidney injury does not constitute a contra-indication to the administration of iodinated contrast medium, as long as the benefit–risk ratio justifies it. Intravenous hydration with 0.9% NaCl or 1.4% sodium bicarbonate is the only validated measure for the prevention of post-iodine contrast nephropathy. This is necessary for intravenous or intra-arterial administration of iodinated contrast agent without first renal pass when the glomerular filtration rate is less than 30 mL/min/1.73 m², for intra-arterial administration of iodinated contrast agent with first renal passage when the glomerular filtration rate is less than 45 mL/min/1.73 m², or in patients with acute renal failure. The use of iodinated contrast medium should allow the carrying out of relevant examinations based on an analysis of the benefit–risk ratio and the implementation of measures to prevent toxicity when necessary.     

Proximal aortic repair in dialysis patients: A national database analysis

ObjectivesDialysis is a well-established risk factor for morbidity and mortality after cardiovascular procedures. However, little is known regarding the outcomes of proximal aortic surgery in this high-risk cohort.MethodsPerioperative (in-hospital or 30-day mortality) and 10-year outcomes were analyzed for all the patients who underwent open proximal aortic repair with the diagnosis of nonruptured thoracic aortic aneurysm (aneurysm, n = 325) or type A aortic dissection (dissection, n = 461) from 1987 to 2015 using the US Renal Data System database.ResultsIn patients with aneurysm, perioperative mortality was 12.6%. The 10-year mortality was 81% ± 3%. Age 65 years or more (hazard ratio [HR], 1.35; 95% confidence interval [CI], 1.03 to 1.78; P = .03), chronic obstructive pulmonary disease (HR, 1.68; 95% CI, 1.01-2.82; P = .047), and Black race (HR, 1.46; 95% CI, 1.09-1.97; P = .01) were independently associated with worse 10-year mortality. In patients with dissection, perioperative mortality was 24.3% and 10-year mortality was 87.9% ± 2.2%. Age 65 years or more (HR, 1.49; 95% CI, 1.19-1.86; P < .001), congestive heart failure (HR, 1.39; 95% CI, 1.11-2.57; P = .004), and diabetes mellitus as the cause of dialysis (HR, 1.75; 95% CI, 1.2-2.57; P = .004) were independently associated with worse 10-year mortality. Black race (HR, 0.74; 95% CI, 0.6-0.92; P = .008) was associated with a better outcome.ConclusionsWe described challenging perioperative and 10-year outcomes for dialysis patients undergoing proximal aortic repair. The present study suggests the need for careful patient selection in the elective repair of proximal aortic aneurysm for dialysis-dependent patients, whereas it affirms the feasibility of emergency surgery for acute type A aortic dissections.     

Percutaneous thermal ablation of sacral metastases: Assessment of pain relief and local tumor control

PurposeTo retrospectively report on safety, pain relief and local tumor control achieved with percutaneous ablation of sacral bone metastases.Materials and methodsFrom February 2009 to June 2020, 23 consecutive patients (12 women and 11 men; mean age, 60 ± 8 [SD] years; median, 60; range: 48-80 years) with 23 sacral metastases underwent radiofrequency (RFA) or cryo-ablation (CA), with palliative or curative intent at our institution. Patients’ demographics and data pertaining to treated metastases, procedure-related variables, safety, and clinical evolution following ablation were collected and analyzed. Pain was assessed with numerical pain rating scale (NPRS).ResultsSixteen (70%) patients were treated with palliative and 7 (30%) with curative intent. Mean tumor diameter was 38 ± 19 (SD) mm (median, 36; range: 11-76). External radiation therapy had been performed on five metastases (5/23; 22%) prior to ablation. RFA was used in 9 (39%) metastases and CA in the remaining 14 (61%). Thermo-protective measures and adjuvant bone consolidation were used whilst treating 20 (87%) and 8 (35%) metastases, respectively. Five (22%) minor complications were recorded. At mean 31 ± 21 (SD) (median, 32; range: 2-70) months follow-up mean NPRS was 2 ± 2 (SD) (median, 1; range: 0–6) vs. 5 ± 1 (median, 5; range: 4–8; P < 0.001) at the baseline. Three metastases out of 7 (43%) undergoing curative ablation showed local progression at mean 4 ± 4 (SD) (median, 2; range: 1-8) months follow-up.ConclusionPercutaneous ablation of sacral metastases is safe and results in significant long-lasting pain relief. Local tumor control seems sub-optimal; however, further investigations are needed to confirm these findings due to paucity of data.     

Awake spinal anesthesia facilitates spine surgery in poor surgical candidates: A case series

BackgroundAnnually, hundreds of thousands of patients undergo surgery for degenerative spine disease (DSD). This represents only a fraction of patients that present for surgical consideration. Procedures are often avoided due to comorbidities that make patients poor candidates for general anesthesia (GA) and its associated risks. With increasing interest in awake surgery under spinal anesthesia (SA), the authors have observed that SA may facilitate spine surgery in patients with relative contraindications to GA. With this in mind, the authors set out to summarize the outcomes of a series of highly comorbid patients who received surgery under SA.MethodsCase logs of a single surgeon were reviewed, and patients undergoing spine surgery under SA were identified. Within this group, patients were identified with relative contraindications to GA, such as advanced age and medical comorbidities. For these patients, for whom surgery was facilitated by SA, the medical records were consulted to report demographic information and patient outcomes.ResultsTen highly comorbid patients were identified who received lumbar spine surgery for DSD under SA. Comorbidities included octogenarian status, obesity, and chronic health conditions such as heart disease. The cohort had a mean age of 75.5 and a mean American Society of Anesthesiologists Physical Status (ASA-PS) score of 3.1. The patients were predicted to have a 2.74-fold increase of serious complications compared to the average patient. There were no adverse events.ConclusionFor patients with symptomatic, refractory DSD and relative contraindications to GA, SA may facilitate safe surgical intervention with excellent outcomes.     

Neurosurgical management of penetrating brain injury during World War I: A historical cohort

During World War I, 25% of penetrating injuries were in the cephalic region. Major Henri Brodier described his surgical techniques in a book in which he reported every consecutive penetrating brain injury (PBI) that he operated on from August 1914 to July 1916. The aim was to collate his data and discuss significant differences in management between soldiers who survived and those who died. We conducted a retrospective survey that included every consecutive PBI patient operated on by Henri Brodier from August 1914 to April 1916 and recorded in his book. We reported medical and surgical management. Seventy-seven patients underwent trepanation by Henri Brodier for PBI. Regarding injury mechanism, 66 procedures (86%) were for shrapnel injury. Regarding location, 21 (30%) involved the whole convexity. Intracranial venous sinus wound was diagnosed intraoperatively in 11 patients (14%). Postoperatively, 7 patients (9%) had seizures, 5 (6%) had cerebral herniation, 3 (4%) had cerebral abscess, and 5 (6%) had meningitis. No patients with abscess or meningitis survived. No significant intergroup differences were found for injury mechanism or wound location, including the venous sinus. Extensive initial surgery with debridement must be prioritized. Infectious complications must not be neglected. We should not forget the lessons of the past when managing casualties in present-day and future conflicts.     

Enhanced recovery after surgery (ERAS) for craniotomies in the treatment of brain tumors: A systematic review

IntroductionPostoperative complications after craniotomy for brain tumors include pain, nausea/vomiting, and infection. A standardized enhanced recovery after surgery (ERAS) protocol is not widely accepted for this common neurosurgical procedure. Few studies have explored its application.MethodsA literature search of PubMed, Cochrane, and Google Scholar databases was performed between January 1992 and March 2023. Original studies that implemented an ERAS protocol for patients that underwent craniotomy for brain tumors were included. The following variables were evaluated: hospital length of stay (LOS), postoperative pain, postoperative nausea and vomiting (PONV) prophylaxis, non-opioid analgesia, and quality of life (QOL).ResultsTwelve studies with a total of 1309 patients met inclusion criteria, including ten randomized controlled trials, one nonrandomized controlled trial, and one quality control study. Most frequently assessed metrics included hospital LOS, PONV prophylaxis, and non-opioid analgesia. A significant reduction in postoperative LOS was observed in 7 studies with ERAS or ERAS components. ERAS was significantly associated with pain reduction on the visual analog scale and verbal numerical rating scale (n = 8). Non-opioid analgesia in ERAS improved postoperative pain control (n = 4) and decreased the duration of pain (n = 1). Three of six studies found no difference in PONV in ERAS vs. control. No studies reported an increase in postoperative complications using ERAS vs. control. One study showed greater patient satisfaction at 30-day follow-up with improved QOL.ConclusionImplementing ERAS protocol may enhance outcomes and quality of life in patients with moderate evidence for improved recovery in those undergoing craniotomy for brain tumors.     

Radiomics for classification of bone mineral loss: A machine learning study

PurposeThe purpose of this study was to develop predictive models to classify osteoporosis, osteopenia and normal patients using radiomics and machine learning approaches.Materials and methodsA total of 147 patients were included in this retrospective single-center study. There were 12 men and 135 women with a mean age of 56.88 ± 10.6 (SD) years (range: 28–87 years). For each patient, seven regions including four lumbar and three femoral including trochanteric, intertrochanteric and neck were segmented on bone mineral densitometry images and 54 texture features were extracted from the regions. The performance of four feature selection methods, including classifier attribute evaluation (CLAE), one rule attribute evaluation (ORAE), gain ratio attribute evaluation (GRAE) and principal components analysis (PRCA) along with four classification methods, including random forest (RF), random committee (RC), K-nearest neighbor (KN) and logit-boost (LB) were evaluated. Four classification categories, including osteopenia vs. normal, osteoporosis vs. normal, osteopenia vs. osteoporosis and osteoporosis + osteopenia vs. osteoporosis were examined for the defined seven regions. The classification model performances were evaluated using the area under the receiver operator characteristic curve (AUC).ResultsThe AUC values ranged from 0.50 to 0.78. The combination of methods RF + CLAE, RF + ORAE and RC + ORAE yielded highest performance (AUC = 0.78) in discriminating between osteoporosis and normal state in the trochanteric region. The combinations of RF + PRCA and LB + PRCA had the highest performance (AUC = 0.76) in discriminating between osteoporosis and normal state in the neck region.ConclusionThe machine learning radiomic approach can be considered as a new method for bone mineral deficiency disease classification using bone mineral densitometry image features.     

Survival outcomes of breast cancer patients with brain metastases: A multicenter retrospective study in Korea (KROG 16–12)

PurposeThis study evaluated the influence of prognostic factors and whole brain radiotherapy (WBRT) on overall survival (OS) of breast cancer (BC) patients with brain metastases (BM).Methods and materialsMedical records of 730 BC patients diagnosed with BM from 2000 to 2014 at 17 institutions were retrospectively reviewed. OS was calculated from BM diagnosis. Median follow-up duration was 11.9 months (range, 0.1–126.2).ResultsMedian OS was 15.0 months (95% CI: 14.0–16.9). Patients with different BC-specific graded prognostic assessment (GPA) scores showed significant differences (p < 0.001) in OS. In multivariate analysis, histologic grade 3 (p = 0.014), presence of extracranial metastasis (p < 0.001), the number of BM (>4; p = 0.002), hormone receptor negativity (p = 0.005), HER2-negativity (p = 0.003), and shorter time interval (<30 months) between BC and BM diagnosis (p = 0.007) were associated with inferior OS. By summing the β-coefficients of variables that were prognostic in multivariate analyses, we developed a prognostic model that stratified patients into low-risk (≤0.673) and high-risk (>0.673) subgroups; the high-risk subgroup had poorer median OS (10.1 months, 95% CI: 7.9–11.9 vs. 21.9 months, 95% CI: 19.5–27.1, p < 0.001). Univariate and multivariate analyses of propensity score-matched patients diagnosed with BM ≥ 30 months after BC diagnosis (n = 389, “late BM”) revealed that WBRT-treated patients showed superior OS compared to non-WBRT-treated patients (p = 0.070 and 0.030, respectively).ConclusionOur prognostic model identified high-risk BC patients with BM who might benefit from increased surveillance; if validated, our model could guide treatment selection for such patients. Patients with late BM might benefit from WBRT as initial local treatment.     

Acute burn care in resource-limited settings: A cohort study on treatment and outcomes in a rural regional referral hospital in Tanzania

BackgroundOnly a few papers are published on the safety and effectiveness of acute burn care in low-income countries. A cohort study was therefore carried out to determine such outcomes.MethodsThe study was conducted in a rural Tanzanian hospital in 2017–2018. All patients admitted with burns were eligible. Complications were scored during admission as an indication for safety. Survivors of severe burn injuries were evaluated for time of reepithelialization, graft take, disability (WHODAS2.0) and quality of life (EQ5D-3L) up to 3 months post-injury, as an indication of effectiveness.ResultsPatients presented on average at 5 days after injury (SD 11, median 1, IQR 0–4). Three patients died at admission. The remaining 79 were included in the cohort. Their median age was 3 years (IQR 2–9, range 0.5–49), mean TBSA burned 12% (SD10%) and mortality rate 11.4%. No surgery-related mortality or life-threatening complications were observed. Skin grafting was performed on 29 patients at a delayed stage (median 23 days, IQR 15–47). Complications of skin grafts included partial (25% of procedures) and complete graft necrosis (8% of procedures). The mean time to reepithelialization was 52 (SD 42) days after admission. Disability and quality of life improved from admission to 3 months after injury (p<0.001, p<0.001, respectively).ConclusionIn this resource-limited setting patients presented after a delay and with multiple complications. The mortality during the first two weeks after admission was high. Surgery was found to be safe and effective. A significant improvement in disability and quality of life was observed.     

Antibody-drug conjugates targeting TROP-2: Clinical development in metastatic breast cancer

Antibody drug conjugates (ADCs) combine the potent cytotoxicity of chemotherapy with the antigen -specific targeted approach of antibodies into one single molecule. Trophoblast cell surface antigen 2 (TROP-2) is a transmembrane glycoprotein involved in calcium signal transduction and is expressed in multiple tumor types. TROP-2 expression is higher in HER2-negative breast tumors (HR+/HR-) and is associated with worse survival. Sacituzumab govitecan (SG) is a first-in-class TROP-2-directed ADC with an anti-TROP-2 antibody conjugated to SN-38, a topoisomerase inhibitor via a hydrolysable linker. This hydrolysable linker permits intracellular and extracellular release of the membrane permeable payload enabling the “bystander effect” contributing to the efficacy of this agent. There was significant improvement in progression free survival (PFS) and overall survival (OS) with SG versus chemotherapy in pretreated metastatic triple negative breast cancer (TNBC), resulting in regulatory approval. Common adverse events (AE) reported were neutropenia and diarrhea. SG also demonstrated clinical activity versus chemotherapy in a phase III trial of HR+/HER2-metastatic breast cancer (MBC) and is under evaluation in first-line metastatic and early stage TNBC as well. Datopotamab deruxtecan (Dato-DXd) is a TROP-2 ADC that differs from SG in that it has a cleavable tetrapeptide linker and a more potent topoisomerase inhibitor payload. This construct is highly stable in circulation with a longer half-life than SG, and undergoes cleavage in presence of intracellular lysosomal proteases. Dato-DXd demonstrated preliminary efficacy in unselected metastatic TNBC, with common AEs of low-grade nausea and stomatitis. Dato-DXd is being investigated in phase III studies in metastatic TNBC and HR+/HER2- MBC. These novel TROP-2 ADCs have the potential to deliver enhanced efficacy with reduced toxicity in MBC and possibly in early stage breast cancer (EBC).     

Acute total hip arthroplasty for older patients with acetabular fractures: A meta-analysis

ObjectiveMultiple treatment options for acetabular fractures in geriatric patients exist. However, no large-scale studies have reported the outcomes of acute total hip arthroplasty (THA) in this patient population. We systematically evaluated all available evidence to characterize clinical outcomes, complications, and revisions of acute THA for acetabular fractures in geriatric patients.MethodsMeta-analysis of 21 studies of 430 acetabular fractures with mean follow-up of 44 months (range, 17−97 months). Two independent researchers searched and evaluated the databases of Ovid, Embase, and United States National Library of Medicine using a Boolean search string up to December 2019. Population demographics and complications, including presence of heterotopic ossification (HO), dislocation, infection, revision rate, neurological deficits, and venous thromboembolic event (VTE), were recorded and analyzed.ResultsWeighted mean Harris Hip Score was 83.3 points, and 20% of the patients had reported complications. The most common complication was HO, with a rate of 19.5%. Brooker grade III and IV HO rates were lower at 6.8%. Hip dislocation occurred at a rate of 6.1%, 4.1% of patients developed VTE, deep infection occurred in 3.8%, and neurological complications occurred in 1.9%. Although the revision rate was described in most studies, we were unable to perform a survival analysis because the time to each revision was described in only a few studies. The revision rate was 4.3%.ConclusionsAcute THA is a viable option for treatment of acetabular fracture and can result in acceptable clinical outcomes and survivorship rates in older patients but with an associated complication rate of approximately 20%. Considering the limited treatment options, THA might be a viable alternative for appropriately selected patients.     

Acute total hip arthroplasty for older patients with acetabular fractures: A meta-analysis

ObjectiveMultiple treatment options for acetabular fractures in geriatric patients exist. However, no large-scale studies have reported the outcomes of acute total hip arthroplasty (THA) in this patient population. We systematically evaluated all available evidence to characterize clinical outcomes, complications, and revisions of acute THA for acetabular fractures in geriatric patients.MethodsMeta-analysis of 21 studies of 430 acetabular fractures with mean follow-up of 44 months (range, 17−97 months). Two independent researchers searched and evaluated the databases of Ovid, Embase, and United States National Library of Medicine using a Boolean search string up to December 2019. Population demographics and complications, including presence of heterotopic ossification (HO), dislocation, infection, revision rate, neurological deficits, and venous thromboembolic event (VTE), were recorded and analyzed.ResultsWeighted mean Harris Hip Score was 83.3 points, and 20% of the patients had reported complications. The most common complication was HO, with a rate of 19.5%. Brooker grade III and IV HO rates were lower at 6.8%. Hip dislocation occurred at a rate of 6.1%, 4.1% of patients developed VTE, deep infection occurred in 3.8%, and neurological complications occurred in 1.9%. Although the revision rate was described in most studies, we were unable to perform a survival analysis because the time to each revision was described in only a few studies. The revision rate was 4.3%.ConclusionsAcute THA is a viable option for treatment of acetabular fracture and can result in acceptable clinical outcomes and survivorship rates in older patients but with an associated complication rate of approximately 20%. Considering the limited treatment options, THA might be a viable alternative for appropriately selected patients.     

Ganglioglioma revealed by spontaneous intracerebral hematoma: A cohort study

BackgroundGangliogliomas are rare tumors of the central nervous system. Intracerebral hemorrhage (ICH) is a frequent pathological event, rarely related to intracranial tumor. Here we report a cohort of 14 gangliogliomas, 3 of which were revealed by ICH. We also performed a systematic review of the literature on gangliogliomas revealed by ICH.Patients and methodsWe retrospectively collected data for all gangliogliomas operated on in our department between January 2015 and December 2019: clinical history, radiological data and outcome, with a special focus on gangliogliomas revealed by spontaneous ICH. All cases had pathological validation. For the systematic review, relevant studies were identified by systematic search of the scientific literature in PubMed.ResultsFifteen patients underwent surgery for ganglioglioma during the study period. Six cases were revealed by seizures, 3 by headache, 1 by hydrocephaly and 1 by developmental disorder. Three patients (21%) had hemorrhagic presentation. All patients were male, aged 30 to 48 years, diagnosed with atypical ICH without vascular abnormality on cerebral angiogram. Conservative management was first decided. Diagnosis of ganglioglioma was made within 3 months for 2 patients and 9 years later for 1, after surgical removal and histological analysis. All hemorrhagic gangliogliomas were BRAF wild type. The systematic review of the literature identified 8 other cases of ganglioglioma revealed by ICH.ConclusionICH may be a mode of revelation of ganglioglioma, and ganglioglioma is a possible etiology of atypical ICH in young patients. Long-term imaging follow-up is mandatory in patients with unexplained ICH.