scholarly journals Intracranial cavernous malformation in children: a single-centered experience with 30 consecutive cases

2013 ◽  
Vol 71 (4) ◽  
pp. 220-228 ◽  
Author(s):  
Marcelo Campos Moraes Amato ◽  
João Flávio Gurjão Madureira ◽  
Ricardo Santos de Oliveira

Objectives: To determine the clinical presentation and treatment outcome of pediatric intracranial cavernous malformation (CM) in a single-centered institution. Methods: Clinical data review of 30 patients under 18 years-old who had undergone surgery for cavernous malformation from January 1993 to December 2011. Results: The Study Group included 18 males and 12 females (mean age: 8.7 years-old). Symptoms at presentation were seizures (16/30, 53.3%), headache (15/30, 50.0%), and focal neurological deficits (11/30, 36.6%). Multiple cavernous malformations were found in 5/30 (16.6%). According to location, patients were classified in groups: (G1) brain-steam in 5/30 (16.6%), (G2) cerebellum in 2/30 (6.6%), (G3) supratentorial associated with seizures in 16/30 (53.3%), and (G4) supratentorial without seizures in 7/30 (23.3%). Surgical resection was performed in 26 out of 30 (86.6%) patients. The mean follow-up period was 4.1 years. Of 15 children followed-up with preoperative seizures, all were rendered seizure-free after surgery. Conclusions: For symptomatic solitary cavernous malformation, the treatment of choice is complete microsurgical excision preceded by careful anatomical and functional evaluation. For multiple cavernous malformation or asymptomatic patients, the treatment modalities must be cautiously considered.

Author(s):  
Gildas Patet ◽  
Andrea Bartoli ◽  
Torstein R. Meling

AbstractRadiation-induced cavernous malformations (RICMs) are delayed complications of brain irradiation during childhood. Its natural history is largely unknown and its incidence may be underestimated as RCIMS tend to develop several years following radiation. No clear consensus exists regarding the long-term follow-up or treatment. A systematic review of Embase, Cochrane Library, PubMed, Google Scholar, and Web of Science databases, following the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines, was performed. Based on our inclusion/exclusion criteria, 12 articles were included, totaling 113 children with RICMs, 86 were treated conservatively, and 27 with microsurgery. We were unable to precisely define the incidence and natural history from this data. The mean age at radiation treatment was 7.3 years, with a slight male predominance (54%) and an average dose of 50.0 Gy. The mean time to detection of RICM was 9.2 years after radiation. RICM often developed at distance from the primary lesion, more specifically frontal (35%) and temporal lobe (34%). On average, 2.6 RICMs were discovered per child. Sixty-seven percent were asymptomatic. Twenty-one percent presented signs of hemorrhage. Clinical outcome was favorable in all children except in 2. Follow-up data were lacking in most of the studies. RICM is most often asymptomatic but probably an underestimated complication of cerebral irradiation in the pediatric population. Based on the radiological development of RICMs, many authors suggest a follow-up of at least 15 years. Studies suggest observation for asymptomatic lesions, while surgery is reserved for symptomatic growth, hemorrhage, or focal neurological deficits.


2003 ◽  
Vol 93 (1) ◽  
pp. 6-10 ◽  
Author(s):  
Gregory Morris ◽  
Kelly Nix ◽  
Flair D. Goldman

Fracture of the second metatarsal is a cause of chronic midfoot pain that has not been thoroughly examined in the literature. A retrospective review of medical charts and imaging studies was undertaken to investigate this phenomenon. The clinical presentation, differential diagnosis, results of imaging studies, and treatment modalities are described for eight patients with midfoot pain who were treated for a mean of 3.7 months (range, 0 to 12 months) before imaging studies showed a nondisplaced transverse fracture of the second metatarsal base. Initial radiographs indicated fracture in only one patient. Two patients later had surgical bone grafting, two patients had asymptomatic nonunion, and three patients eventually healed. At the last follow-up examination, one patient was continuing treatment with immobilization and electrical bone stimulation. For the seven patients in whom symptoms resolved, the mean recovery period was 14 months (range, 5 to 23 months). Further research is needed to improve treatment and outcomes for this condition. (J Am Podiatr Med Assoc 93(1): 6-10, 2003)


2014 ◽  
Vol 13 (5) ◽  
pp. 471-483 ◽  
Author(s):  
Da Li ◽  
Shu-Yu Hao ◽  
Jie Tang ◽  
Xin-Ru Xiao ◽  
Gui-Jun Jia ◽  
...  

Object The aim of this study was to investigate the clinical appearance of untreated pediatric brainstem cavernous malformations (CMs) and to identify the hemorrhage risks and functional outcomes. Methods All pediatric patients with a diagnosis of brainstem CM between 1985 and 2012 were registered. The clinical chart and radiographs were recorded, and follow-up evaluations were obtained prospectively. Results A total of 85 patients (69.4% male) were included with a mean age of 12.7 years. Sixty-seven patients (78.8%) had prior hemorrhage, and 6 patients (7.1%) were asymptomatic. There were 15 midbrain lesions, 53 pons lesions, and 17 medulla lesions. The mean lesion size was 1.9 cm. During a total of 401.6 patient-years of follow-up, 47 hemorrhages occurred in 37 patients, and the annual hemorrhage rate was 11.7% per patient-year. The mean hemorrhage interval was 47.8 months. The hemorrhage risk declined over time, especially after the first 2 years. Both a lesion size ≥ 2 cm (hazard ratio [HR] 2.122, p = 0.037) and the presence of perilesional edema (HR 2.192, p = 0.039) predicted future hemorrhage and were associated with a high annual hemorrhage rate. The hemorrhage-free survival at 6 months was 85.7%, and at 1, 5, 10, and 15 years was 71.5%, 49.4%, 27.5%, and 13.7%, respectively. At the most recent functional evaluation, 33 patients (38.8%) had improved, 32 (37.6%) had stabilized, and 20 (23.5%) had worsened, without any deaths. Twenty-two patients (25.9%) obtained a full recovery. Prospective hemorrhage (HR 0.191, p = 0.003) was the adverse predictor for full recovery. Full recovery primarily occurred within the first 12 months, after which the chance of full recovery decreased. The cumulative percentage of complete recovery at 6 months was 32.7%, and at 1, 3, and 5 years was 40.8%, 43.6%, and 49.2%, respectively. Conclusions In this study the hemorrhage rate was relatively high in pediatric brainstem CMs, although the functional outcome was acceptable. The decline in hemorrhage risk and the identified adverse predictors in this study were helpful for clinicians and patients when deciding on treatment. Referral bias and the insufficient follow-up period of the study were highlighted as limitations.


2012 ◽  
Vol 117 (Special_Suppl) ◽  
pp. 164-169 ◽  
Author(s):  
Cheng-Chia Lee ◽  
David Hung-Chi Pan ◽  
Wen-Yuh Chung ◽  
Kang-Du Liu ◽  
Huai-Che Yang ◽  
...  

Object The authors retrospectively reviewed the efficacy and safety of Gamma Knife surgery (GKS) in patients with brainstem cavernous malformations (CMs). The CMs had bled repeatedly and placed the patients at high risk with respect to surgical intervention. Methods Between 1993 and 2010, 49 patients with symptomatic CMs were treated by GKS. The mean age in these patients was 37.8 years, and the predominant sex was female (59.2%). All 49 patients experienced at least 2 instances of repeated bleeding before GKS; these hemorrhages caused neurological deficits including cranial nerve deficits, hemiparesis, hemisensory deficits, spasticity, chorea or athetosis, and consciousness disturbance. Results The mean size of the CMs at the time of GKS was 3.2 cm3 (range 0.1–14.6 cm3). The mean radiation dose directed to the lesion was 11 Gy with an isodose level at 60.0%. The mean clinical and imaging follow-up time was 40.6 months (range 1.0–150.7 months). Forty-five patients participated in regularly scheduled follow-up. Twenty-nine patients (59.2%) were followed up for > 2 years, and 16 (32.7%) were followed up for < 2 years. The pre-GKS annual hemorrhage rate was 31.3% (69 symptomatic hemorrhages during a total of 220.3 patient-years). After GKS, 3 episodes of symptomatic hemorrhage were observed within the first 2 years of follow-up (4.29% annual hemorrhage rate), and 3 episodes of symptomatic hemorrhage were observed after the first 2 years of follow-up (3.64% annual hemorrhage rate). In this study of 49 patients, symptomatic radiation-induced complications developed in only 2 patients (4.1%; cyst formation in 1 patient and perifocal edema with neurological deficits in the other patient). There were no deaths in this group. Conclusions Gamma Knife surgery is effective in reducing the rate of recurrent hemorrhage. In the authors' experience, it was possible to control bleeding using a low-dose treatment. In addition, there were few symptomatic radiation-induced complications. As a result, the authors believe that GKS is a good alternative treatment for brainstem CMs.


Neurosurgery ◽  
2009 ◽  
Vol 65 (3) ◽  
pp. 450-455 ◽  
Author(s):  
Juri Kivelev ◽  
Mika Niemelä ◽  
Riku Kivisaari ◽  
Reza Dashti ◽  
Aki Laakso ◽  
...  

Abstract OBJECTIVE Multiple cerebral cavernous malformations (MCCMs) typically occur in patients with a family history of these lesions. Literature on MCCMs is scarce, and little is known about their natural history. METHODS Of 264 consecutive patients with cerebral cavernomas treated at the Department of Neurosurgery, Helsinki University Central Hospital, in the past 27 years, 33 patients had MCCMs. Lesions were categorized according to the Zabramski classification scale. Follow-up questionnaires were sent to all patients. Outcome was assessed using the Glasgow Outcome Scale, and amelioration of epilepsy was assessed using the Engel scale. All clinical data were analyzed retrospectively. RESULTS The mean age of patients at diagnosis was 44 years. Sex presentation was almost equal. Nine percent of all patients had a family history of the disease. Patients presented with epilepsy, acute headache, and focal neurological deficits. MCCMs were incidental findings in 2 patients. Altogether, 416 cavernomas were found: 70% supratentorial and 30% infratentorial. Fifteen patients had symptomatic hemorrhage before admission to our department. Surgery was performed on 18 patients. In most cases, the largest cavernoma was removed. Postoperatively, 1 patient experienced temporary hemiparesis, and another developed permanent motor dysphasia. No mortalities occurred. The mean follow-up time was 7.7 years. Twenty-six patients (79%) were in good condition. Among patients with epilepsy who underwent lesionectomy, 70% had an Engel class I outcome. On follow-up magnetic resonance imaging, 52 de novo cavernomas were found. CONCLUSION Surgical treatment of patients with MCCMs is safe. An extirpation of the clinically active cavernoma prevents further bleedings and improves outcome of epilepsy.


2019 ◽  
Vol 103 (10) ◽  
pp. 1453-1459 ◽  
Author(s):  
Stephanie Ming Young ◽  
Kyung Hwan Kim ◽  
Yoon-Duck Kim ◽  
Stephanie S Lang ◽  
Ji Woong Park ◽  
...  

PurposeTo evaluate the efficacy and safety of multisession gamma knife radiosurgery (GKRS) for orbital apex venous cavernous malformation causing optic neuropathy.MethodsRetrospective cohort study in a single tertiary institution from January 2007 to December 2016 on patients who underwent multisession GKRS for orbital apex venous cavernous malformations causing optic neuropathy.ResultsThere were 12 patients included in our study. The mean age was 40.2±14.5 years, and men comprised 66.7% (n=8). Decrease in visual acuity (83.3%) was the most common symptom at presentation. The mean clinical follow-up was 28.5 months. Ten (83.3%) of the 12 patients had improvement in best corrected visual acuity. Of the 10 patients with pre-existing relative afferent pupillary defect (RAPD), 6 (60%) had complete resolution of RAPD. Of the 12 patients with visual field defect, 7 (58.3%) had complete resolution, 3 (25%) had partial improvement, while 2 (16.7%) remained unchanged due to optic atrophy from long-standing compressive optic neuropathy. Mean proptosis reduced from 2.3±1.7 mm pre-GKRS to 0.5±1.3 mm post-GKRS (p=0.005). Tumour shrinkage was observed in all patients. The mean tumour volume at the time of GKRS was 3104 mm3 (range 221–8500 mm3), which reduced to 658 mm3 (range 120–3350 mm3) at last follow-up. None of the patients experienced GKRS-related ocular morbidity during the follow-up period.ConclusionMultisession GKRS has shown to be an effective and safe option for the treatment of orbital apex venous cavernous malformations causing optic neuropathy, with significant improvement in ophthalmic outcomes and reduction in tumour volume.


2019 ◽  
Vol 1 (1) ◽  
pp. V16 ◽  
Author(s):  
Ken Matsushima ◽  
Michihiro Kohno ◽  
Helmut Bertalanffy

Hemorrhagic brainstem cavernous malformations carry a high risk of progressive neurological deficits owing to recurrent hemorrhages and hence require complete surgical resection while minimizing damage to the dense concentration of nuclei and fibers inside the brainstem. To access lesions inside the lower pons, the senior author (H.B.) has preferred to approach the lesions via the “perifacial zone” through the pontomedullary sulcus from the inferior surface of the pontine bulge for more than 20 years.1,2 This video demonstrates a case of a cavernous malformation inside the lower pons, which was surgically treated via the pontomedullary junction through the retrosigmoid supracondylar approach in a half-sitting position. The lesion was completely removed in piecemeal fashion through a tiny incision on the sulcus, which did not cause any new neurological deficits. The modified Rankin Scale improved from 4 before the surgery to 1, and the patient had no recurrence during the 2 years of follow-up. The advantage of this access and the dissection techniques for this challenging lesion are introduced, based on our experience with more than 230 surgeries of brainstem cavernoma.The video can be found here: https://youtu.be/0H_XqkQgQ9I.


2014 ◽  
Vol 2 (11_suppl3) ◽  
pp. 2325967114S0022
Author(s):  
Yusuf Aksoy ◽  
Mehmet Arazi ◽  
Sezgin Şimşek

Objectives: The conservative treatment of unstable fractures of the distal clavicle associated with higher nonunion rates. Although many treatment modalities have been described for these unstable fractures, there isn’t a golden standard one. Although osteosynthesis of the fractures with hook plate known a treatment option its special problems regarded the design of the implant. Aim of the study: To show f effectiveness of the hook plate osteosynthesis in treatment of unstable fractures of the distal clavicle with early removal of the implant to prevent possible complications. Methods: Between July 2011 and September 2013, seven patients with unstable fractures of the distal clavicle were treated with open reduction and internal fixation with hook plate. The mean age of the patients was 36 (range: 21-58) and there were two female and 5 male. Routine plate removal was planned at six months after the index surgery and the patients were informed for this second surgery. All fractures were unilateral and 5 in dominant extremity. Routine follow up were done after two, six and 18 weeks after the index surgery. The healing of the fracture was evaluated with radiological and clinical examinations. The Constant scoring system was used for functional evaluation of the patients. Results: All fractures healed without any mechanical problems related to the plate. The average follow-up of the patients were 22 months (range: 8 - 35). After the complete healing all plates were removed. The mean Constant score was 93 (rang 82 to 98 points) at the final follow up of the patients. Conclusion: The osteosynthesis of the unstable fractures of the distal clavicle with hook plate is seems to be an alternative and effective option for surgical treatment. To prevent implant related mechanical problems, we advise to remove the plate after the healing of the fracture as a routine manner.


2006 ◽  
Vol 21 (1) ◽  
pp. 1-8 ◽  
Author(s):  
Jean G. de Oliveira ◽  
Aziz Rassi-Neto ◽  
Fernando A. P. Ferraz ◽  
Fernando M. Braga

Object The aim of this study was to analyze cerebellar cavernous malformations (CMs) with respect to epide-miological, clinical, radiological, and therapeutic aspects. Methods Between 1984 and 2004, 100 patients were surgically treated for intracranial CMs at the Division of Neurosurgery of Federal University of São Paulo. The authors reviewed the records of 10 patients whose lesions were located in the cerebellum. There were four male and six female patients (ratio 1:1.5) whose ages ranged from 14 to 45 years (mean age 33 years). Clinical presentation was sudden or acute in all cases, and neuroimaging examinations performed in all patients demonstrated signs of bleeding. The mean size of the malformations was 4.6 cm, and in all but one patient the lesions were totally removed without complications. After a mean follow-up period of 70 months, all patients were considered to be in good or excellent clinical condition. Conclusions Cerebellar CMs should be analyzed separately from other posterior fossa CMs. These lesions can reach large sizes and cause massive hemorrhages, resulting in acute or sudden presentation. Surgery is a safe and effective option that provides a curative treatment when a complete removal is achieved.


2010 ◽  
Vol 29 (3) ◽  
pp. E11 ◽  
Author(s):  
Edward A. Monaco ◽  
Aftab A. Khan ◽  
Ajay Niranjan ◽  
Hideyuki Kano ◽  
Ramesh Grandhi ◽  
...  

Object The authors performed a retrospective review of prospectively collected data to evaluate the safety and efficacy of stereotactic radiosurgery (SRS) for the treatment of patients harboring symptomatic solitary cavernous malformations (CMs) of the brainstem that bleed repeatedly and are high risk for resection. Methods Between 1988 and 2005, 68 patients (34 males and 34 females) with solitary, symptomatic CMs of the brainstem underwent Gamma Knife surgery. The mean patient age was 41.2 years, and all patients had suffered at least 2 symptomatic hemorrhages (range 2–12 events) before radiosurgery. Prior to SRS, 15 patients (22.1%) had undergone attempted resection. The mean volume of the malformation treated was 1.19 ml, and the mean prescribed marginal radiation dose was 16 Gy. Results The mean follow-up period was 5.2 years (range 0.6–12.4 years). The pre-SRS annual hemorrhage rate was 32.38%, or 125 hemorrhages, excluding the first hemorrhage, over a total of 386 patient-years. Following SRS, 11 hemorrhages were observed within the first 2 years of follow-up (8.22% annual hemorrhage rate) and 3 hemorrhages were observed in the period after the first 2 years of follow-up (1.37% annual hemorrhage rate). A significant reduction (p < 0.0001) in the risk of brainstem CM hemorrhages was observed following radiosurgical treatment, as well as in latency period of 2 years after SRS (p < 0.0447). Eight patients (11.8%) experienced new neurological deficits as a result of adverse radiation effects following SRS. Conclusions The results of this study support a role for the use of SRS for symptomatic CMs of the brainstem, as it is relatively safe and appears to reduce rebleeding rates in this high-surgical-risk location.


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