Category Archives: Articles

Update: GLARIUS trial

GLARIUS trial

The GLARIUS trial which investigated the efficacy of bevacizumab (BEV)/irinotecan (IRI) as compared to standard temozolomide (TMZ) in the first-line therapy of MGMT promoter methylation glioblastoma showed that progression free survival was significantly prolonged by BEV/IRI while overall survival was similar in both arms 1).


A report focusses on quality of life (QoL) and Karnofsky performance score (KPS) during the whole course of the disease.

Patients (n=170) received standard radiotherapy and were randomized (2:1) for BEV/IRI or standard TMZ. At least every three months KPS was determined and QoL was measured using the EORTC QLQ-C30 and EORTC QLQ-BN20 questionnaires. A generalized estimating equation model (GEE) evaluated differences in the course of QoL and KPS over time. Also, the time to first deterioration and the time to postprogression deterioration was analyzed separately.

In all dimensions of QoL and KPS, GEE analyses and time to first deterioration analyses did not detect significant differences between the treatment arms. At progression, 82% of patients receiving second-line therapy in the standard arm received BEV second-line therapy. For the dimensions motor dysfunction and headaches, time to postprogression deterioration was prolonged in the standard arm receiving crossover second-line BEV in the vast majority of patients at the time of evaluation.

GLARIUS did not find indications for a BEV-induced detrimental effect on QoL in first-line therapy of MGMT-nonmethylated GBM patients. Moreover, GLARIUS provided some indirect corroborative data supporting the notion that BEV may have beneficial effects upon QoL in relapsed GBM 2).

1)

Herrlinger U, Schäfer N, Steinbach JP, Weyerbrock A, Hau P, Goldbrunner R, Friedrich F, Rohde V, Ringel F, Schlegel U, Sabel M, Ronellenfitsch MW, Uhl M, Maciaczyk J, Grau S, Schnell O, Hänel M, Krex D, Vajkoczy P, Gerlach R, Kortmann RD, Mehdorn M, Tüttenberg J, Mayer-Steinacker R, Fietkau R, Brehmer S, Mack F, Stuplich M, Kebir S, Kohnen R, Dunkl E, Leutgeb B, Proescholdt M, Pietsch T, Urbach H, Belka C, Stummer W, Glas M. Bevacizumab Plus Irinotecan Versus Temozolomide in Newly Diagnosed O6-Methylguanine-DNA Methyltransferase Nonmethylated Glioblastoma: The Randomized GLARIUS Trial. J Clin Oncol. 2016 May 10;34(14):1611-9. doi: 10.1200/JCO.2015.63.4691. Epub 2016 Mar 14. PubMed PMID: 26976423.

2)

Schäfer N, Proescholdt M, Steinbach JP, Weyerbrock A, Hau P, Grauer O, Goldbrunner R, Friedrich F, Rohde V, Ringel F, Schlegel U, Sabel M, Ronellenfitsch MW, Uhl M, Grau S, Hänel M, Schnell O, Krex D, Vajkoczy P, Tabatabai G, Mack F, Schaub C, Tzaridis T, Nießen M, Kebir S, Leutgeb B, Urbach H, Belka C, Stummer W, Glas M, Herrlinger U. Quality of life in the GLARIUS trial randomizing bevacizumab/irinotecan versus temozolomide in newly diagnosed, MGMT-nonmethylated glioblastoma. Neuro Oncol. 2017 Nov 7. doi: 10.1093/neuonc/nox204. [Epub ahead of print] PubMed PMID: 29121274.

Update: Chronic subdural hematoma recurrence

Chronic subdural hematoma recurrence

Epidemiology

Recurrence rates after chronic subdural hematoma (CSDH) evacuation with any of actual techniques twist drill craniostomy (TDC), burr hole craniostomy, craniotomy range from 5% to 30%. 1)

Risk factors

In the series of Han et al. independent risk factors for recurrence were as follows: age > 75 years (HR 1.72, 95% CI 1.03-2.88; p = 0.039), obesity (body mass index ≥ 25.0 kg/m2), and a bilateral operation 2).

Chon et al. shown that postoperative midline shifting (≥5 mm), diabetes mellitus, preoperative seizure, preoperative width of hematoma (≥20 mm), and anticoagulant therapy were independent predictors of the recurrence of chronic subdural hematoma.

According to internal architecture of hematoma, the rate of recurrence was significantly lower in the homogeneous and the trabecular type than the laminar and separated type 3).

see Chronic subdural hematoma and anticoagulant therapy.


The recurrence rate of chronic subdural hematoma cSDH seems to be related to the excessive neoangiogenesis in the parietal membrane, which is mediated via vascular endothelial growth factor (VEGF). This is found to be elevated in the hematoma fluid and is dependent on eicosanoid/prostaglandin and thromboxane synthesis via cyclooxygenase-2 (COX-2).


Antiplatelet therapy

Antiplatelet therapy significantly influences the recurrence of CSDH 4).

Pneumocephalus

Remaining pneumocephalus is seen as an approved factor of recurrence 5) 6).

Septation

Jack et al.found a 12% reoperation rate. CSDH septation (seen on computed tomogram scan) was found to be an independent risk factor for recurrence requiring reoperation (p=0.04). Larger post-operative subdural haematoma volume was also significantly associated with requiring a second drainage procedure (p<0.001). Independent risk factors of larger post-operative haematoma volume included septations within a CSDH (p<0.01), increased pre-operative haematoma volume (p<0.01), and a greater amount of parenchymal atrophy (p=0.04). A simple scoring system for quantifying recurrence risk was created and validated based on patient age (< or ≥80 years), haematoma volume (< or ≥160cc), and presence of septations within the subdural collection (yes or no).

Septations within CSDHs are associated with larger post-operative residual haematoma collections requiring repeat drainage. When septations are clearly visible within a CSDH, craniotomy might be more suitable as a primary procedure as it allows greater access to a septated subdural collection. The proposed scoring system combining haematoma volume, age, and presence of septations might be useful in identifying patients at higher risk for recurrence 7).

Membranectomy

Opening the internal hematoma membrane does not alter the rate of patients requiring revision surgery and the number of patients showing a marked residual hematoma six weeks after evacuation of a CSDH 8).

In the study of Lee et al, an extended surgical approach with partial membranectomy has no advantages regarding the rate of reoperation and the outcome. As initial treatment, burr-hole drainage with irrigation of the hematoma cavity and closed-system drainage is recommended. Extended craniotomy with membranectomy is now reserved for instances of acute rebleeding with solid hematoma 9).

Diabetes

Surgeons should consider informing patients with diabetes mellitus that this comorbidity is associated with an increased likelihood of recurrence

10) 11) 12).


Balser et al. report 11% recurrence, which included individuals who recurred as late as 3 years after initial diagnosis 13).

Close imaging follow-up is important for CSDH patients for recurrence prediction. Using quantitative CT volumetric analysis, strong evidence was provided that changes in the residual fluid volume during the ‘self-resolution’ period can be used as significantly radiological predictors of recurrence 14).

A structural equation model showed a significant association between increased antiinflammatory activity in hematoma fluid samples and a lower risk of recurrence, but this relationship was not statistically significant in venous blood samples. Moreover, these findings indicate that anti-inflammatory activities in the hematoma may play a role in the risk of a recurrence of CSDH 15).

Irrigation with artificial cerebrospinal fluid (ACF) decreased the rate of CSDH recurrence 16).

Treatment

There is no definite operative procedure for patients with intractable chronic subdural hematoma (CSDH).

Most recurrent hematomas are managed successfully with burr hole craniostomies with postoperative closed-system drainage. Refractory hematomas may be managed with a variety of techniques, including craniotomy or subdural-peritoneal shunt placement 17).

Although many studies have reported risk factors or treatments in efforts to prevent recurrence, those have focused on single recurrence, and little cumulative data is available to analyze refractory CSDH.

Matsumoto et al. defined refractory CSDH as ≥2 recurrences, then analyzed and compared clinical factors between patients with single recurrence and those with refractory CSDH in a cohort study, to clarify whether patients with refractory CSDH experience different or more risk factors than patients with single recurrence, and whether burr-hole irrigation with closed-system drainage reduces refractory CSDH.

Seventy-five patients had at least one recurrence, with single recurrence in 62 patients and ≥2 recurrences in 13 patients. In comparing clinical characteristics, patients with refractory CSDH were significantly younger (P=0.04) and showed shorter interval to first recurrence (P<0.001). Organized CSDH was also significantly associated with refractory CSDH (P=0.02). Multivariate logistic regression analysis identified first recurrence interval <1 month (OR 6.66, P<0.001) and age <71 years (OR 4.16, P<0.001) as independent risk factors for refractory CSDH. On the other hand, burr-hole irrigation with closed-system drainage did not reduce refractory CSDH.

When patients with risk factors for refractory CSDH experience recurrence, alternative surgical procedures may be considered as the second surgery, because burr-hole irrigation with closed-system drainage did not reduce refractory CSDH 18).

Implantation of a reservoir 19) 20) 21).

Subdural-peritoneal shunt 22).

Middle meningeal artery embolization

Embolization of the MMA is effective for refractory CSDH or CSDH patients with a risk of recurrence, and is considered an effective therapeutic method to stop hematoma enlargement and promote resolution 23) 24) 25) 26) 27) 28).

A pilot study indicated that perioperative middle meningeal artery (MMA) embolization could be offered as the least invasive and most effectual means of treatment for resistant patients of CSDHs with 1 or more recurrences 29).

Chihara et al. have treated three cases of CSDH with MMA embolization to date, but there was a postoperative recurrence in one patient, which required a craniotomy for hematoma removal and capsulectomy. MMA embolization blocks the blood supply from the dura to the hematoma outer membrane in order to prevent recurrences of refractory CSDH. Histopathologic examination of the outer membrane of the hematoma excised during craniotomy showed foreign-body giant cells and neovascular proliferation associated with embolization. Because part of the hematoma was organized in this case, the CSDH did not resolve when the MMA was occluded, and the development of new collateral pathways in the hematoma outer membrane probably contributed to the recurrence. Therefore, in CSDH with some organized hematoma, MMA embolization may not be effective. Magnetic resonance imaging (MRI) should be performed in these patients before embolization 30).

Case series

2017

A retrospective analysis of 756 consecutive patients with CSDH who underwent bur hole surgery at the Hanyang University Medical Center (Seoul and Guri) between January 1, 2004, and December 31, 2014. During the 6-month follow-up, 104 patients (13.8%) with recurrence after surgery for CSDH were identified. Independent risk factors for recurrence were as follows: age > 75 years (HR 1.72, 95% CI 1.03-2.88; p = 0.039), obesity (body mass index ≥ 25.0 kg/m2), and a bilateral operation.

This study determined the risk factors for recurrence of CSDH and their effects on outcomes. Further studies are needed to account for these observations and to determine their underlying mechanisms 31).

2016

Chronic subdural hematomas (cSDHs) have shown an increasing incidence in an ageing population over the last 20 years, while unacceptable recurrence rates of up to 30 % persist. The chronic subdural hematoma recurrence rate seems to be related to the excessive neoangiogenesis in the parietal membrane, which is mediated via vascular endothelial growth factor (VEGF). This is found to be elevated in the haematoma fluid and is dependent on eicosanoid/prostaglandin and thromboxane synthesis via cyclooxygenase-2 (COX 2). With this investigator-initiated trial (IIT) it was thought to diminish the recurrence rate of operated-on cSDHs by administering a selective COX-2 inhibitor (Celecoxib) over 4 weeks’ time postoperatively in comparison to a control group.

The thesis of risk reduction of cSDH recurrence in COX-2-inhibited patients was to be determined in a prospective, randomised, two-armed, open phase-II/III study with inclusion of 180 patients over a 2-year time period in four German university hospitals. The treated- and untreated-patient data were to be analysed by Fisher’s exact test (significance level of alpha, 0.05 [two-sided]).

After screening of 246 patients from January 2009 to April 2010, the study had to be terminated prematurely as only 23 patients (9.3 %) could be enrolled because of on-going non-steroid anti-rheumatic (NSAR) drug treatment or contraindication to Celecoxib medication. In the study population, 13 patients were treated in the control group (six women, seven men; average age 66.8 years; one adverse event (AE)/serious adverse event (SAE) needing one re-operation because of progressive cSDH (7.7 %); ten patients were treated in the treatment group (one woman, nine men; average age 64.7 years; five AEs/SAEs needing two re-operations because of one progressive cSDH and one wound infection [20 %]). Significance levels are obsolete because of insufficient patient numbers.

The theoretical advantage of COX-2 inhibition in the recurrent cSDH could not be transferred into the treatment of German cSDH patients as 66.6 % of the patients showed strict contraindications for Celecoxib. Furthermore, 55 % of the patients were already treated with some kind of COX-2 inhibition and, nevertheless, developed cSDH. Thus, although conceptually appealing, an anti-angiogenic therapy with COX-2 inhibitors for cSDH could not be realised in this patient population due to the high prevalence of comorbidities excluding the administration of COX2 inhibitors 32).

2010

Recurrence rates after chronic subdural hematoma (CSDH) evacuation with any of actual techniques twist drill craniostomy (TDC), burr hole craniostomy, craniotomy range from 5% to 30%. Use of drain has improved recurrence rates when used with burr-hole craniostomy. Now, we analyze predictors of recurrence of TDC with drain.

Three hundred twelve consecutive patients with CSDH have been studied in a retrospective study. Operative technique in all patients consisted in TDC with drain. Data recorded included any associated comorbidity. Radiologic measures of the CSDH before and after the procedure were studied. Clinical evaluation included Modified Rankin Scale, Glasgow Coma Scale (GCS), and neurological deficits. Two groups were compared: recurrence group and nonrecurrence group. Follow-up was for at least 1 year.

Twelve percent experienced recurrence. Preoperative CSDH width, preoperative midline shift, postoperative midline width, postoperative CSDH width, and residual CSDH 1 month later were significantly associated with CSDH recurrence. The logistic regression model for the multivariate analysis revealed that postoperative midline shift and postoperative neurological deficit were significantly associated with CSDH recurrence. The duration of treatment with dexamethasone was found not to be related with recurrence. Mortality before hospital discharge was 1%. Hospital stay was 2.5 days.

TDC with drain has similar results in recurrence rates, morbidity, mortality, and outcome as other techniques as burr-hole craniostomy with drain. Preoperative and postoperative hematoma width and midline shift are independent predictors of recurrence. Brain re-expansion and time of drain maintenance are important factors related with recurrence of CSDH. Future CSDH reservoirs must avoid negative pressure and sudden pressure changes inside the whole closed drain system 33).

Case reports

2016

Mewada et al. report a case with right hemiparesis and aphasia 1 month after a fall from a bicycle. Computed tomography scan of the head showed left chronic subdural hematoma, which was evacuated by burr-hole drainage. The postoperative course was complicated by reaccumulation within short period of time. On superselective digital subtraction angiography of MMA, iatrogenic dAVF was found on left side. We embolized successfully it using n-butyl cyanoacrylate after a third irrigation. No reaccumulation found in the postoperative period or at last follow-up. They proposed a treatment protocol based on the own experience and literature review.

Refractory chronic subdural hematoma with reaccumulation within a short interval should be subjected to digital subtraction angiography of the MMA. Embolization of ipsilateral MMA is safe, effective, and a useful option for the treatment of iatrogenic dAVF and resolution of hematoma 34).


An 85-year-old male presented with left CSDH, which recurred five times. The hematoma was irrigated and drained through a left frontal burr hole during the first to third surgery and through a left parietal burr hole during the fourth and fifth surgery. The hematoma had no septation and was well-evacuated during each surgery. Antiplatelet therapy for preventing ischemic heart disease was stopped after the second surgery, the hematoma cavity was irrigated with artificial cerebrospinal fluid at the third surgery, and the direction of the drainage tube was changed to reduce the postoperative subdural air collection at the fourth surgery. However, none of these interventions was effective. He was successfully treated by fibrin glue injection into the hematoma cavity after the fifth surgery.

This procedure may be effective for refractory CSDH in elderly patients 35).


A 67-year-old man with dural arteriovenous fistula (AVF) presenting as a non-traumatic chronic subdural hematoma (CSDH). This previously healthy patient was hospitalized due to progressive headache with subacute onset. He underwent burr-hole surgery twice for evacuating the left CSDH that was thickest at the posterior temporal area. The operative procedure and finding was not extraordinary, but subdural hematoma slowly progressed for days following the revision surgery. After investigation by super-selective external carotid angiography, a dural AVF found near the transverse-sigmoid sinus was diagnosed. Dural AVF was completely occluded with trans-arterial injecting polyvinyl alchol particles into the petrosquamosal branch of the middle meningeal artery. The patient showed a good neurological outcome with no additional intervention. Brain surgeons have to consider the possibility of dural AVF and perform cerebral angiogram if necessary when they manage the cases that have a spontaneously occurred and repeatedly recurring CSDH 36).

2007

Spontaneous intracranial hypotension (SIH) is reported to cause chronic subdural hematoma (SDH), however diagnosis of SIH in patients with SDH is not always easy.

Takahashi et al. report a case of chronic SDH refractory to repeated drainage, which was attributed to SIH. A forty-five-year-old man who had been suffering from orthostatic headache for one month was admitted to our hospital presenting with unconsciousness and hemiparesis. CT on admission revealed a chronic subdural hematoma, which was successfully treated once with subdural drainage. However, the patient fell into unconscious again with recurrence of the hematoma within several days. After two more sessions of drainage, SIH due to cerebrospinal fluid leakage was diagnosed with spinal magnetic resonance imaging (MRI) and radionuclide cisternography. Spinal MRI demonstrated abnormal fluid accumulation in the thoracic epidural space, and the radionuclide cisternogram showed early excretion of tracer into urine as well as absence of intracranial tracer filling. After treatment with epidural blood patching, the hematoma rapidly disappeared and he was discharged without symptoms. In the treatment of chronic SDH, especially in young to middle aged patient without preceding trauma or hematological disorders, physicians should pay attention to underlying SIH to avoid multiple surgery. MRI of the spine as well as radionuclide cisternography is useful in evaluation of this condition 37).

1) , 33)

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2) , 31)

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4)

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5)

Mori K, Maeda M (2001) Surgical treatment of chronic subdural hematoma in 500 consecutive cases: clinical characteristics, surgical outcome, complications, and recurrence rate. Neurol Med Chir (Tokyo) 41:371–381

6)

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7)

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9)

Lee JY, Ebel H, Ernestus RI, Klug N. Various surgical treatments of chronic subdural hematoma and outcome in 172 patients: is membranectomy necessary? Surg Neurol. 2004 Jun;61(6):523-7; discussion 527-8. PubMed PMID: 15165784.

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12)

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Balser D, Rodgers SD, Johnson B, Shi C, Tabak E, Samadani U. Evolving management of symptomatic chronic subdural hematoma: experience of a single institution and review of the literature. Neurol Res. 2013 Apr;35(3):233-42. doi: 10.1179/1743132813Y.0000000166. Review. PubMed PMID: 23485050.

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Update: Anterior cerebral artery infarct

Anterior cerebral artery infarct

Stroke in the anterior cerebral artery territory are much less common than either middle cerebral artery or posterior cerebral artery territory infarcts.

Epidemiology

ACA territory infarcts are rare, comprising ~2% of ischaemic strokes.

ACA territory infarcts are less common because if the A1 segment is occluded there is generally enough collateral flow via the contralateral A1 segment to supply the distal ACA territory.

Etiology

Embolic strokes (often with MCA involvement) are the most common cause.

Rarely, they are also seen as a complication of severe midline shift, where the ACA is occluded by mass effect or severe vasospasm.

An asymmetry of the A1 segment of the anterior cerebral artery (A1SA) was identified on digital subtraction angiography studies from 127 patients (21.4%) and was strongly associated with anterior communicating artery aneurysm (ACoAA) (p < 0.0001, OR 13.7). An A1SA independently correlated with the occurrence of ACA infarction in patients with ACoAA (p = 0.047) and in those without an ACoAA (p = 0.015). Among patients undergoing Anterior communicating artery aneurysm endovascular treatment, A1SA was independently associated with the severity of ACA infarction (p = 0.023) and unfavorable functional outcome (p = 0.045, OR = 2.4).

An A1SA is a common anatomical variation in SAH patients and is strongly associated with ACoAA. Moreover, the presence of A1SA independently increases the likelihood of ACA infarction. In SAH patients undergoing ACoAA coiling, A1SA carries the risk for severe ACA infarction and thus an unfavorable outcome. Clinical trial registration no.: DRKS00005486 (http://www.drks.de/) 1).

Clinical features

Diagnosis

The features are those of cerebral infarction in the anterior cerebral artery vascular territory:

Paramedian frontoparietal cerebral cortex

Anterior corpus callosum.

Anterior limb of the internal capsule.

Inferior portion of the Caudate nucleus head.

Differential diagnosis

Case series

Kumral et al. studied 48 consecutive patients who admitted to the stroke unit over a 6-year period.

They performed magnetic resonance imaging (MRI) and magnetic resonance angiography (MRA) in all patients, and Diffusion weighted magnetic resonance imaging (DWI) in 21. In the stroke registry, patients with ACA infarction represented 1.3% of 3705 patients with ischemic stroke. The main risk factors of ACA infarcts was hypertension in 58% of patients, diabetes mellitus in 29%, hypercholesterolemia in 25%, cigarette smoking in 19%, atrial fibrillation in 19%, and myocardial infarct in 6%. Presumed causes of ACA infarct were large-artery disease and cardioembolism in 13 patients each, small-artery disease (SAD) in the territory of Heubner’s artery in two and atherosclerosis of large-arteries (<50% stenosis) in 16. On clinico-radiologic analysis there were three main clinical patterns depending on lesion side; left-side infarction (30 patients) consisting of mutism, transcortical motor aphasia, and hemiparesis with lower limb predominance; right side infarction (16 patients) accompanied by acute confusional state, motor hemineglect and hemiparesis; bilateral infarction (two patients) presented with akinetic mutism, severe sphincter dysfunction, and dependent functional outcome. Our findings suggest that clinical and etiologic spectrum of ACA infarction may present similar features as that of middle cerebral artery infarction, but frontal dysfunctions and callosal syndromes can help to make a clinical differential diagnosis. Moreover, at the early phase of stroke, DWI is useful imaging method to locate and delineate the boundary of lesion in the territory of ACA 3).

1)

Jabbarli R, Reinhard M, Roelz R, Kaier K, Weyerbrock A, Taschner C, Scheiwe C, Shah M. Clinical relevance of anterior cerebral artery asymmetry in aneurysmal subarachnoid hemorrhage. J Neurosurg. 2017 Nov;127(5):1070-1076. doi: 10.3171/2016.9.JNS161706. Epub 2016 Dec 23. PubMed PMID: 28009232.
3)

Kumral E, Bayulkem G, Evyapan D, Yunten N. Spectrum of anterior cerebral artery territory infarction: clinical and MRI findings. Eur J Neurol. 2002 Nov;9(6):615-24. PubMed PMID: 12453077.

Update: Acinetobacter baumannii ventriculitis

Acinetobacter baumannii ventriculitis

Even though meningitis caused by Acinetobacter baumannii is relatively rare, it is associated with high mortality rates especially in neurosurgery patients and represents a serious therapeutic problem due to the limited penetration of effective antibiotics into the cerebrospinal fluid.

Multidrug-resistant Acinetobacter baumannii ventriculitis is a serious clinical challenge for neurosurgeons 1).

Diagnosis

Matrix-assisted laser desorption/ionization time-of-flight mass spectrometry (MALDITOF) has been effectively used as a rapid method for microbial identification.

In a case report Brunetti et al. identified A. baumanni by MALDI-TOF technique directly from the CSF drawn from the external ventricular drainage of a patient with severe confusional state and signs of meningism. Simultaneously the antibiotic susceptibility test was performed by automated method from the pellet of the broth-enriched sample. The MALDI-TOF technique allowed microbial identification in less than 30 minutes, and the susceptibility test result was available in eight hours, thus allowing a fast diagnosis ready for prompt and targeted antimicrobial therapy 2).

Treatment

In 2013 a review of the available literature regarding intraventricular (IVT) or intrathecal (ITH) administration of colistin in multidrug-resistant (MDR) and extensively drug-resistant (XDR) A. baumannii ventriculitis/meningitis was conducted and a total of 83 episodes in 81 patients were identified (71 cases in adults and 10 in children and neonates). Colistin was administered via the IVT and ITH route in 52 and 22 cases, respectively, whilst in 7 cases the exact route was not identified. The median dose of local colistin was 125000 IU (10mg) with a range of 20000 IU (1.6 mg) to 500000 IU (40 mg) in adults, whilst a dose of 2000 IU/kg (0.16 mg/kg) up to 125000 IU (10mg) was used in the paediatric population. The median duration of treatment of IVT/ITH polymyxin E was 18.5 days, whilst the median time to achieve sterilisation of cerebrospinal fluid was 4 days. The rate of successful outcome was 89%, and toxicity related to treatment mainly manifested as reversible chemical ventriculitis/meningitis was reported in nine cases (11%). Nowadays, IVT and ITH colistin represents the last resort treatment of MDR and XDR A. baumannii ventriculitis/meningitis, offering a unique, rather safe and successful mode of therapy 3).

Case series

2016

Thirty-four patients presented nosocomial meningitis/ventriculitis; 11 (32.5 %) were included in the intravenous colistin (IVC group) and 23 (67.6 %) in the intrathecal/intraventricular colistin (ITC group). The most frequent isolated bacteria were Acinetobacter baumannii. The mean dose was 170,000 (±400) IU and the duration of intraventricular treatment was 16.0 (±8.3) days. The duration of intravenous treatment was 16.0 (±8.3) days in the ITC group and 15.3 ± 7.6 days in IVC group. Hospital mortality was significantly lower in the ITC group compared with the IVC group (13 vs. 72.7 %, p = 0.001).

The combination of intravenous plus intraventricular (IV-IVT) colistin therapy may improve outcomes in patients attending with meningitis/ventriculitis due to multi-drug resistance infections 4).


In an 11-year period, information on 18 consecutive patients with extensively drug-resistant A. baumannii ventriculomeningitis was collected. Infection was defined on the basis of (i) isolation of A. baumannii from the cerebrospinal fluid (CSF); (ii) laboratory evidence of CSF infection; (iii) signs/symptoms of central nervous system (CNS) infection. Patients were divided into group 1 (nine patients, IV colistin alone) and group 2 (nine patients, IV plus IVT colistin).

Cerebrospinal fluid sterilization was documented for 12 of 18 patients (66.6%). The CSF sterilization rate was 33.3% in group 1 and 100% in group 2 (P = 0.009). The mean time to CSF sterilization was 21 days (range 8-48). Five patients died due to A. baumannii CNS infection (all in group 1), and five deaths were unrelated to A. baumannii ventriculomeningitis. Intensive care unit mean length of stay was shorter in group 2 (20.7 vs. 41.6 days, P = 0.046). Crude relative risk ratio of cumulative incidence of persistent CNS infection in group 1 versus group 2 was 13. No cases of chemical meningitis due to intrathecal colistin administration were encountered 5).

2013

Treatment results of six post-neurosurgical ventriculitis and meningitis cases caused by extensively drug-resistant Acinetobacter baumannii after application of an intraventricular loading dose of 500000 IU (40 mg) of colistin followed by a dose of 125000-250000 IU (10-20 mg) every 24-48 h plus parenteral colistin are reported. Simultaneous bacteraemia with an identical Acinetobacter strain was observed in three patients. The mean duration of treatment was 17.2 days (range 15-21 days) and the median time of sterilisation of cerebrospinal fluid was 2.5 days (range 1-5 days). All patients were cured, however one patient presented with chemical meningitis and one with chemical ventriculitis, conditions that clinically and biochemically resemble bacterial meningitis 6).

2010

In a case series of seven Thai patients and 17 patients identified in the literature, clinical and microbiological cure rates with IT/IVT colistin therapy were 83% and 92%, respectively. Three patients (13%) developed chemical ventriculitis and one (4%) experienced treatment-associated seizures. Death was associated with delayed IT/IVT colistin therapy compared to survival (mean time from diagnosis to IT/IVT colistin, 7 vs. 2 days; p 0.01). The only independent predictor of mortality was the severity of illness (APACHE II score > 19, adjusted odds ratio 49.5; 95% CI 1.7-1428.6; p 0.02). This case series suggests that administration of primary or adjunctive IT/IVT colistin therapy was effective for drug-resistant A. baumannii CNS infection 7).

2009

López-Alvarez et al review the literature concerning intraventricular use of colistin (polymyxin E) for A. baumannii ventriculitis and add three cases successfully treated 8).

2007

Two patients with multiresistant Acinetobacter baumannii central nervous system infection, successfully treated with either intravenous and/or intraventricular colistin are presented. Unresolved issues such as dose and duration of intraventricular colistin are discussed 9).

2006

Five patients, all were admissions to the neurosurgical ICU and all were cured of their CNS infections. Three cases were complicated by drug-induced aseptic meningitis or ventriculitis.

This largest case series till 2006 shows that direct instillation of colistin into the CNS may cause chemical meningitis or ventriculitis but it is an effective treatment option for MRAB CNS infection. Further study of dosing regimens is needed 10).

Case reports

2016

Shrestha et al report a case of MDR Acinetobacter ventriculitis treated with intravenous and intraventricular colistin together with intravenous tigecycline. The patient developed nephrotoxicity and poor neurological outcome despite microbiological cure. Careful implementation of bundle of measures to minimize EVD-associated ventriculitis is valuable 11).

2015

Full remission in a patient with catheter-associated ventriculitis due to Acinetobacter baumannii treated with intrathecal and intravenous colistin besides coinfections with other multidrug-resistant bacteria 12).

2013

A case of meningitis due to extensively drug-resistant A baumannii in an Austrian patient who had undergone neurosurgery in northern Italy. The case illustrates the limits of therapeutic options in central nervous system infections caused by extensively drug-resistant pathogens 13).

2012

A Baby PR was delivered vaginally in a district hospital of central India at 32 weeks of gestation following the premature rupture of membranes for more than 72 hours and spontaneous onset of labor. His mother received two doses of betamethasone as well as antibiotics before delivery. He had an Apgar score of 6 at the 1st minute and 9 at the 5th minute and weighed 1.5 kilograms at birth. His initial neonatal course was relatively straightforward with administration of one dose of surfactant, ventilation for 12 hours and subsequent continuous positive airway pressure (CPAP) for one week, and IV amoxicillin and gentamicin for 48 hours, which were stopped as blood cultures were negative. The baby did not require total parenteral nutrition or umbilical catheterizations. His initial head scan on day 2 was normal. Beyond the first week of life, the baby was stable with full enteral feeding and no respiratory support.

The baby had a septic deterioration at the end of the second week with recurrent apneas requiring ventilation. Blood tests revealed an increase in C-reactive protein (CRP: 80 mg/L; normal: <5 mg/L) and white blood cell count (23,000/mm3; normal: 4000–11000/mm3). All of the sepsis screening was performed and treatment was commenced with IV cefotaxime and gentamicin for late-onset sepsis. Blood culture obtained on that day showed the growth of MDRAB. The organism was sensitive to ceftazidime, polymyxin B, trimethoprim, colistin, netilmicin, and amikacin, but was resistant to all other 3rd and 4th generation cephalosporins, other aminoglycosides, quinolones, carbapenems, modified and enhanced penicillins (also with sulbactam), aztreonam, and chloramphenicol. Lumbar CSF examination revealed severe pyogenic meningitis. There was elevated CSF protein level – 1014 mg/dl (normal: 65-150 mg/dl), low CSF glucose – 20 mg/dl (normal: 24-63 mg/dl), and high white blood cell count – 500 cells/mm3 with 100% polymorphs (normal: 0-29 lymphocytes/mm3).

However, CSF obtained was insufficient for culture. The patient was referred to our tertiary neonatal unit on the 18th day of life because of a further increase in CRP (220 mg/L) and the positive blood culture results.

Antibiotic treatment was changed to IV ceftazidime and amikacin as per the blood culture results. The patient showed a clinical improvement over the next 48 hours with successful extubation and decline in his CRP.

Unfortunately, there was further deterioration on the 22nd day of life with bulging anterior fontanel and increasing head circumference. Cranial ultrasonography was suggestive of hydrocephalus and ventriculitis. Ventricular CSF examination on that day showed elevated proteins – 863 mg/dl, low sugar – 13 mg/dl, and a high white cell count – 2,360/mm3 with 90% polymorphs. CSF culture showed the growth of PDRAB that was sensitive only to polymyxin B and netilmicin, but resistant to all other antibiotics as listed above.

Further advice was sought at this point from the microbiology and neurosurgical teams. IV netilmicin (5 mg/kg 12 hourly for 6 weeks) and polymyxin B (20,000 units 12 hourly for 6 weeks) were started on day 26 of life. IVT polymyxin B (40,000 units per dose) was given by alternate day ventricular puncture for four weeks (14 doses) as the family did not consent to the insertion of a ventricular reservoir.

Ventricular CSF examination after four weeks of therapy showed improvement: protein – 124 mg/dl, sugar – 35 mg/dl, and cell counts – 48/ mm3 (mainly lymphocytes). CSF culture was negative. IV therapy was continued for another two weeks.

Computed tomography of the brain performed after the treatment showed massive communicating hydrocephalus but no evidence of ventriculitis. Ventriculoperitoneal shunting was performed four weeks after stopping treatment, and the baby was discharged at the chronologic age of 4.5 months.

On subsequent follow-up at the corrected (for prematurity) age of two years, his head was growing consistently along the 25th percentile on the World Health Organization (WHO) growth chart and weight and height were just below the 10th percentile. His automated brain evoked audiometry was normal. He had a Bayley’s Motor Development Index (MDI) of 80 and a Pervasive Development Index (PDI) of 65.

Bayley Scales of Infant Development-II (1993) assess the attainment of key developmental milestones in children from 1 to 42 months in two main domains: MDI and PDI as above. It is a tester-observed score only and although parental reports can be recorded, they do not contribute to the final scores. Raw scores are adjusted for the chronological age and index scores are obtained. Despite a lack of standardization in premature babies, this test is widely accepted as a reliable measure of development 14).

2011

A 38-year-old, 84-kg Caucasian woman with a recent history of craniotomy was admitted with nausea, fever, headache, photophobia, and drainage from her craniotomy incision. She underwent a repeat craniotomy on hospital day 4 with abscess debridement and repair of a cerebrospinal fluid leak. Cultures grew MDR A. baumannii, coagulase-negative Staphylococcus species, and methicillin-resistant Staphylococcus aureus. Based on the limited published pharmacokinetic and pharmacodynamic data for colistin, we determined a favorable outcome with i.v. colistin monotherapy was unlikely and decided to treat the patient with simultaneous i.v. and intraventricular colistin, as well as intraventricular tobramycin and i.v. rifampin. She was treated with a total of 36 days of intraventricular colistin, 40 days of intraventricular tobramycin, 51 days of i.v. colistin and rifampin, and 56 days i.v. vancomycin for infection that persisted despite multiple debridements. The patient had subsequent improvement in clinical manifestations and eradication of infection. She was subsequently discharged to an acute rehabilitation facility on hospital day 77 with posttreatment sequelae including mental impairment and renal failure requiring hemodialysis. Follow-up visits revealed significant improvement in her mental status, speech, and strength on the side not affected by the stroke.

Prolonged combination therapy with intraventricular colistin and tobramycin plus i.v. colistin, rifampin, and vancomycin led to the resolution of a persistent central nervous system infection caused by MDR A. baumannii 15).

2010

A case of a 42-year-old male patient affected by low-grade ependymoma who developed AB-MDR post-neurosurgical ventriculitis. Initially, because of in vitro susceptibility, De Pascale et al used a combination of intravenous colistin and tigecycline. This treatment resulted in the improvement of the patient’s initial condition. However, soon after, the infection relapsed; tigecycline was stopped and treatment with intrathecal colistin was initiated. Cure was achieved by continuing this treatment for approximately three weeks, without adverse effects 16).

2009

A 2-month-old girl with ventriculitis caused by MDRAB is reported. Despite therapy with intravenous (IV) colistin ventricular fluid, cultures remained positive for MDRAB. Institution of combination therapy with IV and intraventricular colistin resulted in a successful clinical and microbiological outcome. Intraventricular/intrathecal and IV colistin might be the best therapeutic option in the treatment of central nervous system infection caused by MDRAB. Further studies are required to evaluate pharmacokinetic and pharmacodynamic parameters of combined IV and intraventricular/intrathecal colistin administration, especially in children 17).

2008

A case of a 40-year old man was admitted to the intensive care unit due to subarachnoid haemorrhage. The patient developed a ventriculitis due to A.baumannii treated successfully with sulbactam IV and intrathecal amikacin 18).

1)

Ganjeifar B, Zabihyan S, Baharvahdat H, Baradaran A. Multidrug-resistant Acinetobacter baumannii ventriculitis: a serious clinical challenge for neurosurgeons. Br J Neurosurg. 2016 Jul 8:1-2. [Epub ahead of print] PubMed PMID: 27387018.
2)

Brunetti G, Ceccarelli G, Giordano A, Navazio AS, Vittozzi P, Venditti M, Raponi G. Fast and reliable diagnosis of XDR Acinetobacter baumannii meningitis by matrix-assisted laser desorption/ionization time-of-flight mass spectrometry. New Microbiol. 2017 Nov 7;40(4). [Epub ahead of print] PubMed PMID: 29112767.
3)

Karaiskos I, Galani L, Baziaka F, Giamarellou H. Intraventricular and intrathecal colistin as the last therapeutic resort for the treatment of multidrug-resistant and extensively drug-resistant Acinetobacter baumannii ventriculitis and meningitis: a literature review. Int J Antimicrob Agents. 2013 Jun;41(6):499-508. doi: 10.1016/j.ijantimicag.2013.02.006. Epub 2013 Mar 16. Review. PubMed PMID: 23507414.
4)

Fotakopoulos G, Makris D, Chatzi M, Tsimitrea E, Zakynthinos E, Fountas K. Outcomes in meningitis/ventriculitis treated with intravenous or intraventricular plus intravenous colistin. Acta Neurochir (Wien). 2016 Mar;158(3):603-10; discussion 610. doi: 10.1007/s00701-016-2702-y. Epub 2016 Jan 23. PubMed PMID: 26801512.
5)

De Bonis P, Lofrese G, Scoppettuolo G, Spanu T, Cultrera R, Labonia M, Cavallo MA, Mangiola A, Anile C, Pompucci A. Intraventricular versus intravenous colistin for the treatment of extensively drug resistant Acinetobacter baumannii meningitis. Eur J Neurol. 2016 Jan;23(1):68-75. doi: 10.1111/ene.12789. Epub 2015 Jul 31. PubMed PMID: 26228051.
6)

Karaiskos I, Galani L, Baziaka F, Katsouda E, Ioannidis I, Andreou A, Paskalis H, Giamarellou H. Successful treatment of extensively drug-resistant Acinetobacter baumannii ventriculitis and meningitis with intraventricular colistin after application of a loading dose: a case series. Int J Antimicrob Agents. 2013 May;41(5):480-3. doi: 10.1016/j.ijantimicag.2013.02.010. Epub 2013 Apr 6. PubMed PMID: 23566531.
7)

Khawcharoenporn T, Apisarnthanarak A, Mundy LM. Intrathecal colistin for drug-resistant Acinetobacter baumannii central nervous system infection: a case series and systematic review. Clin Microbiol Infect. 2010 Jul;16(7):888-94. doi: 10.1111/j.1469-0691.2009.03019.x. Epub 2009 Aug 17. Review. PubMed PMID: 19686281.
8)

López-Alvarez B, Martín-Láez R, Fariñas MC, Paternina-Vidal B, García-Palomo JD, Vázquez-Barquero A. Multidrug-resistant Acinetobacter baumannii ventriculitis: successful treatment with intraventricular colistin. Acta Neurochir (Wien). 2009 Nov;151(11):1465-72. doi: 10.1007/s00701-009-0382-6. Epub 2009 May 8. PubMed PMID: 19424656.
9)

Paramythiotou E, Karakitsos D, Aggelopoulou H, Sioutos P, Samonis G, Karabinis A. Post-surgical meningitis due to multiresistant Acinetobacter baumannii. Effective treatment with intravenous and/or intraventricular colistin and therapeutic dilemmas. Med Mal Infect. 2007 Feb;37(2):124-5. Epub 2007 Jan 30. PubMed PMID: 17270377.
10)

Ng J, Gosbell IB, Kelly JA, Boyle MJ, Ferguson JK. Cure of multiresistant Acinetobacter baumannii central nervous system infections with intraventricular or intrathecal colistin: case series and literature review. J Antimicrob Chemother. 2006 Nov;58(5):1078-81. Epub 2006 Aug 17. PubMed PMID: 16916866.
11)

Shrestha GS, Tamang S, Paneru HR, Shrestha PS, Keyal N, Acharya SP, Marhatta MN, Shilpakar S. Colistin and tigecycline for management of external ventricular device-related ventriculitis due to multidrug-resistant Acinetobacter baumannii. J Neurosci Rural Pract. 2016 Jul-Sep;7(3):450-2. doi: 10.4103/0976-3147.176194. PubMed PMID: 27365967; PubMed Central PMCID: PMC4898118.
12)

Dersch R, Robinson E, Beume L, Rauer S, Niesen WD. Full remission in a patient with catheter-associated ventriculitis due to Acinetobacter baumannii treated with intrathecal and intravenous colistin besides coinfections with other multidrug-resistant bacteria. Neurol Sci. 2015 Apr;36(4):633-4. doi: 10.1007/s10072-014-2031-y. Epub 2014 Dec 11. Review. PubMed PMID: 25501805.
13)

Hoenigl M, Drescher M, Feierl G, Valentin T, Zarfel G, Seeber K, Krause R, Grisold A. Successful management of nosocomial ventriculitis and meningitis caused by extensively drug-resistant Acinetobacter baumannii in Austria. Can J Infect Dis Med Microbiol. 2013 Fall;24(3):e88-90. PubMed PMID: 24421838; PubMed Central PMCID: PMC3852464.
14)

Piparsania S, Rajput N, Bhatambare G. Intraventricular polymyxin B for the treatment of neonatal meningo-ventriculitis caused by multi-resistant Acinetobacter baumannii–case report and review of literature. Turk J Pediatr. 2012 Sep-Oct;54(5):548-54. Review. PubMed PMID: 23427525.
15)

Patel JA, Pacheco SM, Postelnick M, Sutton S. Prolonged triple therapy for persistent multidrug-resistant Acinetobacter baumannii ventriculitis. Am J Health Syst Pharm. 2011 Aug 15;68(16):1527-31. doi: 10.2146/ajhp100234. PubMed PMID: 21817084.
16)

De Pascale G, Pompucci A, Maviglia R, Spanu T, Bello G, Mangiola A, Scoppettuolo G. Successful treatment of multidrug-resistant Acinetobacter baumannii ventriculitis with intrathecal and intravenous colistin. Minerva Anestesiol. 2010 Nov;76(11):957-60. Epub 2010 May 6. PubMed PMID: 20445494.
17)

Dalgic N, Ceylan Y, Sancar M, Telhan L, Kafadar I, Cavusoglu H, Ceylan O, Hasim O. Successful treatment of multidrug-resistant Acinetobacter baumannii ventriculitis with intravenous and intraventricular colistin. Ann Trop Paediatr. 2009 Jun;29(2):141-7. doi: 10.1179/146532809×440761. PubMed PMID: 19460268.
18)

Barrou L, Charra B, Hachimi A, Idali B, Benslama A, Motaouakkil S. Intrathecal use of amikacin: a case report. Braz J Infect Dis. 2008 Dec;12(6):546. PubMed PMID: 19287849.

Update: Bariatric surgery for idiopathic intracranial hypertension

Bariatric surgery for idiopathic intracranial hypertension

Bariatric surgery (BS) has been suggested as idiopathic intracranial hypertension treatment (IIH) associated with morbid obesity.

Reviews

2017

A systematic review and meta-analyses of surgical and non-surgical studies in 2017:

Bariatric surgery achieved 100% papilloedema resolution and a reduction in headache symptoms in 90.2%. Non-surgical methods offered improvement in papilloedema in 66.7%, visual field defects in 75.4% and headache symptoms in 23.2%. Surgical BMI decrease was 17.5 vs. 4.2 for non-surgical methods.

Whilst both bariatric surgery and non-surgical weight loss offer significant beneficial effects on IIH symptomatology, future studies should address the lack of prospective and randomised trials to establish the optimal role for these interventions 1).

2015

A comprehensive literature search was conducted using the following databases: MEDLINE, EMBASE, PubMed, Scopus, Web of Sciences, and the Cochrane Library. No restrictions were placed on these searches, including the date of publication.

A total of 85 publications were identified, and after initial appraisal, 17 were included in the final review. Overall improvement in symptoms of IIH after bariatric surgery was observed in 60 of the 65 patients observed (92%). Postoperative lumbar puncture opening pressure was shown to decrease by an average of 18.9 cmH2O in the 12 patients who had this recorded.

Bariatric surgery for weight loss is associated with alleviation of IIH symptoms and a reduction in intracranial pressure. Furthermore, an improvement was observed in patients where conventional treatments, including neurosurgery, were ineffective. Further prospective randomized studies with control groups and a larger number of participants are lacking within the published studies to date. There is, therefore, a strong rationale for the use of bariatric surgery in individuals with IIH for the effective treatment of this condition, as well as the efficacy of weight loss for various other obesity co-morbidities 2).

2011

Fridley et al. published in 2011 a review:

Eleven relevant publications (including 6 individual case reports) were found, reporting on a total of 62 patients. The Roux-en-Y gastric bypass was the most common bariatric procedure performed. Fifty-six (92%) of 61 patients with recorded postoperative clinical history had resolution of their presenting IIH symptoms following bariatric surgery. Thirty-four (97%) of 35 patients who had undergone pre- and postoperative funduscopy were found to have resolution of papilledema postoperatively. Eleven (92%) of 12 patients who had undergone pre- and postoperative formal visual field testing had complete or nearly complete resolution of visual field deficits, and the remaining patient had stabilization of previously progressive vision loss. In 13 patients both pre- and postoperative CSF pressures were recorded, with an average postoperative pressure decrease of 254 mm H(2)O. Changes in weight loss and body mass index varied depending on the reported postoperative follow-up interval.

The published Class IV evidence suggests that bariatric surgery may be an effective treatment for IIH in obese patients, both in terms of symptom resolution and visual outcome. Prospective, controlled studies are necessary for better elucidation of its role 3).


In a study Roth et al. describe a high rate of overdrainage (OD) seen in patients following shunts and BS.

Patients with IIH that undergo shunt surgery and BS (not concomitantly) may suffer from OD symptoms, necessitating multiple shunt revisions, and valve upgrades. Despite BS being a valid primary treatment for some patients with IIH, among shunted patients, BS may not lead to resolution of IIH-related symptoms and patients may remain shunt-dependent 4).


Hoang et al. present a report of 3 patients with adolescent-onset IIH that was treated at the Duke University in whom bariatric surgery was pursued successfully. The patients had previously undergone CSF shunting at ages 12, 15, and 23 years. They were shunt dependent for a collective average of 3.3 years prior to bariatriwc surgery. All patients reported “low-pressure” or postural headaches after bariatric surgery that correlated with dramatic reduction in their weight. Two of the 3 patients had their shunts removed and continued to be shunt free 1.5 years later at last follow-up; the third patient remained shunt dependent with the pressure set at 200 mm H2O. Given the significant complications inherent to multiple shunt revisions, earlier intervention for weight loss, including bariatric surgery, in these patients might have prevented complications and the associated health care burden. The authors recommend a multidisciplinary approach for IIH treatment with early consideration for weight loss interventions in select patients 5).


Findings support the notion that caloric restriction represents an important mechanism to explain the very early anti-diabetic effects observed after bariatric surgery. However, exclusion of the upper gastrointestinal tract also provides further metabolic improvements, possibly mediated by gastrointestinal hormonal responses and altered postprandial glucose absorption 6).

Case reports

2017

A 46-year-old woman presented at our service with idiopathic intracranial hypertension that had been diagnosed two years earlier, which had led to chronic refractory headache and an estimated 30% loss of visual acuity, associated with bilateral papilledema. She presented partial improvement of the headache with acetazolamide, but the visual loss persisted. Her intracranial pressure was 34 cmH2O. She presented a body mass index of 39.5 kg/m2, also associated with high blood pressure. Computed tomography of the cranium with endovenous contrast did not show any abnormalities. She underwent Roux-en-Y gastric bypass with uneventful postoperative evolution. One month following surgery, she presented a 24% excess weight loss. An ophthalmological examination revealed absence of visual loss and remission of the papilledema. There were no new episodes of headache following the surgery. There was also complete resolution of high blood pressure. The intracranial pressure decreased to 24 cmH2O, six months after the surgery 7).

1)

Manfield JH, Yu KK, Efthimiou E, Darzi A, Athanasiou T, Ashrafian H. Bariatric Surgery or Non-surgical Weight Loss for Idiopathic Intracranial Hypertension? A Systematic Review and Comparison of Meta-analyses. Obes Surg. 2017 Feb;27(2):513-521. doi: 10.1007/s11695-016-2467-7. Review. PubMed PMID: 27981458; PubMed Central PMCID: PMC5237659.

2)

Handley JD, Baruah BP, Williams DM, Horner M, Barry J, Stephens JW. Bariatric surgery as a treatment for idiopathic intracranial hypertension: a systematic review. Surg Obes Relat Dis. 2015 Nov-Dec;11(6):1396-403. doi: 10.1016/j.soard.2015.08.497. Epub 2015 Aug 12. Review. PubMed PMID: 26499350.

3)

Fridley J, Foroozan R, Sherman V, Brandt ML, Yoshor D. Bariatric surgery for the treatment of idiopathic intracranial hypertension. J Neurosurg. 2011 Jan;114(1):34-9. doi: 10.3171/2009.12.JNS09953. Epub 2010 Jan 22. Review. PubMed PMID: 20095788.

4)

Roth J, Constantini S, Kesler A. Over-drainage and persistent shunt-dependency in patients with idiopathic intracranial hypertension treated with shunts and bariatric surgery. Surg Neurol Int. 2015 Dec 8;6(Suppl 27):S655-60. doi: 10.4103/2152-7806.171230. eCollection 2015. PubMed PMID: 26713173; PubMed Central PMCID: PMC4683795.

5)

Hoang KB, Hooten KG, Muh CR. Shunt freedom and clinical resolution of idiopathic intracranial hypertension after bariatric surgery in the pediatric population: report of 3 cases. J Neurosurg Pediatr. 2017 Sep 29:1-6. doi: 10.3171/2017.6.PEDS17145. [Epub ahead of print] PubMed PMID: 28960170.

6)

Michaud A, Grenier-Larouche T, Caron-Dorval D, Marceau S, Biertho L, Simard S, Richard D, Tchernof A, Carpentier AC. Biliopancreatic diversion with duodenal switch leads to better postprandial glucose level and beta cell function than sleeve gastrectomy in individuals with type 2 diabetes very early after surgery. Metabolism. 2017 Sep;74:10-21. doi: 10.1016/j.metabol.2017.06.005. Epub 2017 Jun 21. PubMed PMID: 28764844.

7)

Cazzo E, Gestic MA, Utrini MP, Mendonça Chaim FD, Mendonça Chaim FH, Cândido EC, da Silveira Jarolavsky LB, de Almeida AMN, Pareja JC, Chaim EA. Bariatric surgery as a treatment for pseudotumor cerebri: case study and narrative review of the literature. Sao Paulo Med J. 2017 May 29:0. doi: 10.1590/1516-3180.2016.0305060117. [Epub ahead of print] PubMed PMID: 28562736.

Update: Middle cerebral artery aneurysm endovascular treatment with Flow Diverter

Middle cerebral artery aneurysm endovascular treatment with Flow Diverter

Flow diverter for middle cerebral artery aneurysm treatment should be considered an alternative when traditional treatment methods are not feasible 1).

When performed in a select treatment group, high rates of aneurysm occlusion and protection against re-rupture can be achieved 2).

Longer angiographic follow-ups are needed to assess the morphologic outcome; immediate subtotal occlusions do not seem to be related to rupture 3).

Findings suggest that complete occlusion after endovascular treatment with FDD can be delayed (>6 months). Ischemic complications may occur as early or delayed, particularly at clopidogrel interruption 4).

The Pipeline Embolization Device provides a safe and effective treatment alternative for wide-neck MCA aneurysms that give rise to a bifurcating or distal branch when other endovascular techniques are thought to be unfeasible or more risky 5).

WEB flow disruption seems to be a promising technique for the treatment of complex MCA aneurysms, particularly those with a wide neck or unfavorable dome-to-neck ratio 6).

For Caroff et al. compared with other available therapeutic options, the flow-diverter stent does not appear to be a suitable solution for the treatment of saccular MCA bifurcation aneurysms 7).

Unsatisfactory occlusion rate in bifurcation aneurysms likely results from residual filling of the aneurysms in cases in which the jailed side branch remains patent 8).

Systematic review and meta-analysis

A systematic search of PubMed, MEDLINE, and Embase was performed for studies published from 2008 to May 2017.

According to the Preferred Reporting Items for Systematic Reviews and MetaAnalyses, Cagnazzo et al. selected studies with >5 patients describing angiographic and clinical outcomes after flow-diversion treatment of MCA aneurysms.

Random-effects metaanalysis was used to pool the following outcomes: aneurysm occlusion rate, procedure-related complications, rupture rate of treated aneurysms, and occlusion of the jailed branches.

Twelve studies evaluating 244 MCA aneurysms were included in this meta-analysis. Complete/near-complete occlusion was obtained in 78.7% (95% CI, 67.8%-89.7%) of aneurysms. The rupture rate of treated aneurysms during follow-up was 0.4% per aneurysm-year. The rate of treatment-related complications was 20.7% (95% CI, 14%-27.5%), and approximately 10% of complications were permanent. The mortality rate was close to 2%. Nearly 10% (95% CI, 4.7%-15.5%) of jailed arteries were occluded during follow-up, whereas 26% (95% CI, 14.4%-37.6%) had slow flow. Rates of symptoms related to occlusion and slow flow were close to 5%.

Small and retrospective series could affect the strength of the reported results.

Given the not negligible rate of treatment-related complications, flow diversion for MCA aneurysms should be considered an alternative treatment when traditional treatment methods are not feasible. However, when performed in this select treatment group, high rates of aneurysm occlusion and protection against re-rupture can be achieved 9).

Case series

2017

Consecutive patients treated from January 2010 to December 2014 by Iosif et al. by using endovascular flow-diverting stents for MCA bifurcation aneurysms were evaluated retrospectively with prospectively maintained data. All patients had been followed for at least 12 months after treatment, with at least 2 control angiograms; regional flow-related angiographic modifications were registered by using a new angiographic outcome scale for flow diverters. Data were analyzed with emphasis on procedure-related events, angiographic results, and clinical outcome.

Fifty-eight patients were included in the study, with 63 MCA bifurcation aneurysms; 13 of these were large and giant. Pretreatment mRS was 0 for 12 patients (20.7%), 1 for 41 (70.7%), and 2 for 5 patients (8.6%). Six-month control revealed mRS 0-2 for 57 (98.3%) patients and 3 for 1 (1.7%) patient. Procedure-related morbidity and mortality were 8.6% (5/58) and 0%, respectively. From 95% of still circulating immediate postprocedure angiographic outcomes, 68% progressed to aneurysm occlusion at 6 months and 95%, to occlusion at 12 months, with a 0% aneurysm rupture rate.

Flow diverters seem to be an effective treatment alternative for complex MCA bifurcation aneurysms, with reasonable complication rates. Longer angiographic follow-ups are needed to assess the morphologic outcome; immediate subtotal occlusions do not seem to be related to rupture 10).


Bhogal et al. retrospectively reviewed there prospectively maintained database to collect information for all patients with unruptured saccular bifurcation MCA aneurysms treated with FDS between January 2010 and January 2016. In addition to demographic data, they recorded the location, aneurysm characteristics, previous treatments, number and type of FDS, complications, and clinical and angiographic follow-up.

The search identified 13 patients (7 males) with an average age of 61.7 years (47-74 years). All patients had a single bifurcation aneurysm of the MCA, and none of the aneurysms were acutely ruptured. The average fundus size of the saccular aneurysms was 3 mm (range 1.5-10 mm). Follow-up studies were available for 12 patients. Based on the most recent follow-up angiograms, six aneurysms (50%) were totally occluded; five aneurysms (41.7%) showed only a small remnant; and one aneurysm (8.3%) remained unchanged. One patient suffered from an ischemic stroke with resultant permanent hemiparesis (mRS 3). In another case, there was an in-stent thrombosis during the intervention, which resolved upon intra-arterial infusion of Eptifibatide (mRS 0). There were no intra-operative vessel or aneurysm ruptures and no mortalities. Angiography of the covered MCA branches showed no change in the caliber or flow of the vessel in six (50%), a reduction in caliber in five (41.7%), and a complete occlusion in one (8.3%). All caliber changes and occlusions of the vessels were asymptomatic.

91.7% of treated MCA bifurcation aneurysms were either completely occluded or showed only a small remnant with a good safety profile. Flow diversion of MCA bifurcation aneurysms should be considered as an alternative treatment strategy when microsurgical clipping or alternative endovascular treatment options are not feasible 11).

2016

Patients with MCAAs were treated by flow diversion if surgical or other endovascular treatment modalities had failed or were deemed likely to fail. Angiographic and clinical outcome of these patients was assessed retrospectively. Aneurysm location on MCA was defined as M1 segment, “true bifurcation” (classical bifurcation of MCA into superior and inferior trunks), “variant bifurcation” (bifurcation of early frontal or early/distal temporal branches), or M2 segment. Aneurysm morphology was classified as saccular versus dissecting/fusiform.

Treatment was attempted in 29 MCAAs. Technical failure rate was 3.4% (1/29). Thirteen of aneurysms were fusiform. Of the bifurcation aneurysms, most (10/16) were the variant type. Overall and procedure-related mortality/permanent morbidity rates were 10.3% (3/29) and 3.5% (1/29). Total occlusion rates (mean angiographic follow-up 10.3 months) for saccular and fusiform aneurysms were 40% and 75%, respectively. In bifurcation aneurysms, occlusion was strongly associated with side-branch occlusion (P < 0.005).

In this series, flow diversion for the treatment of MCAAs was safe, was effective in the treatment of fusiform MCAAs, and was not as effective at mid-term for MCA bifurcation aneurysms. Unsatisfactory occlusion rate in bifurcation aneurysms likely results from residual filling of the aneurysms in cases in which the jailed side branch remains patent 12).


Fourteen patients with 15 aneurysms were included in the study. Ischemic complications, as confirmed by MR imaging, occurred in 6 patients (43%). Procedure-related morbidity and mortality at last follow-up were 21% and 0%, respectively. Angiographic follow-up was available for 13 aneurysms, with a mean follow-up of 16 months. Complete occlusion was obtained for 8 aneurysms (62%).

Compared with other available therapeutic options, the flow-diverter stent does not appear to be a suitable solution for the treatment of saccular MCA bifurcation aneurysms 13).


From February 2010 to December 2013, 14 patients (10 women; mean age 59 years) with 15 small MCA aneurysms were treated with FDD. All procedures were performed with the Pipeline embolization device (PED).

Complete occlusion was obtained in 12/15 aneurysms (80%) and partial occlusion in 3 (20%). Among 13 aneurysms with a side branch, this was patent at the angiographic control in 4 cases, showed decreased filling in 6, and was occluded in 3 (with neurological deficits in 2). All PEDs were patent at follow-up. Post-procedural ischemic complications occurred in 4 (27%) procedures with permanent neurological deficit (modified Rankin score 2) in 3 (21%). No early or delayed aneurysm rupture, no subarachnoid or intraparenchymal hemorrhage and no deaths occurred.

Endovascular treatment with FDD is a relatively safe treatment for small MCA aneurysms resulting in a high occlusion rate. The findings suggest that complete occlusion after endovascular treatment with FDD can be delayed (>6 months). Ischemic complications may occur as early or delayed, particularly at clopidogrel interruption 14).

2014

Twenty-five aneurysms located at the MCA bifurcation (n = 21) or distal (n = 4) were treated. Of these, 22 were small and 3 were large. A single device was used in all but 2. No deaths occurred in the series. All patients had at least 1 control angiographic study, 21 of which were DSA (3-30 months), which showed that 12 of the rising branches were patent whereas 6 were filling in reduced caliber and 3 were occluded asymptomatically. According to the last angiographic follow-up, complete occlusion was revealed in 21 of 25 aneurysms (84%).

The Pipeline Embolization Device provides a safe and effective treatment alternative for wide-neck MCA aneurysms that give rise to a bifurcating or distal branch when other endovascular techniques are thought to be unfeasible or more risky 15).

2013

Thirty-three patients with 34 MCA aneurysms were treated with the Woven EndoBridge (WEB) in 5 European centers. The ability to successfully deploy the WEB, procedure- and device-related adverse events, morbidity and mortality of the treatment, and short-term angiographic follow-up results were analyzed.

Most treated aneurysms were unruptured (85.3%) and were between 5 and 10 mm (85.3%) with a neck size ≥  4 mm (88.2%). The treatment failed in 1 of the 34 aneurysms (2.9%) owing to a lack of appropriate device size. Treatment was performed exclusively with the WEB in 29 of 33 aneurysms (87.9%). Additional treatment (coiling and/or stenting) was used in 4 of 33 aneurysms (12.1%). Mortality of the treatment was 0.0% and morbidity was 3.1% (intraoperative rupture with modified Rankin Scale score of 3 at the 1-month follow-up). In short-term follow-up (range, 2-12 months), adequate occlusion (total occlusion or neck remnant) was observed in 83.3% of aneurysms.

WEB flow disruption seems to be a promising technique for the treatment of complex MCA aneurysms, particularly those with a wide neck or unfavorable dome-to-neck ratio 16).

Case reports

Burrows et al. present the case of an adolescent with a middle cerebral artery (MCA) fusiform aneurysm which recurred following clip reconstruction and bypass. The aneurysm was successfully treated with endovascular flow diversion 17).

1) , 11)

Bhogal P, AlMatter M, Bäzner H, Ganslandt O, Henkes H, Aguilar Pérez M. Flow Diversion for the Treatment of MCA Bifurcation Aneurysms-A Single Centre Experience. Front Neurol. 2017 Feb 2;8:20. doi: 10.3389/fneur.2017.00020. eCollection 2017. PubMed PMID: 28210239; PubMed Central PMCID: PMC5288345.

2) , 9)

Cagnazzo F, Mantilla D, Lefevre PH, Dargazanli C, Gascou G, Costalat V. Treatment of Middle Cerebral Artery Aneurysms with Flow-Diverter Stents: A Systematic Review and Meta-Analysis. AJNR Am J Neuroradiol. 2017 Oct 5. doi: 10.3174/ajnr.A5388. [Epub ahead of print] PubMed PMID: 28982785.

3) , 10)

Iosif C, Mounayer C, Yavuz K, Saleme S, Geyik S, Cekirge HS, Saatci I. Middle Cerebral Artery Bifurcation Aneurysms Treated by Extrasaccular Flow Diverters: Midterm Angiographic Evolution and Clinical Outcome. AJNR Am J Neuroradiol. 2017 Feb;38(2):310-316. doi: 10.3174/ajnr.A5022. Epub 2016 Dec 15. PubMed PMID: 27979794.

4) , 14)

Briganti F, Delehaye L, Leone G, Sicignano C, Buono G, Marseglia M, Caranci F, Tortora F, Maiuri F. Flow diverter device for the treatment of small middle cerebral artery aneurysms. J Neurointerv Surg. 2016 Mar;8(3):287-94. doi: 10.1136/neurintsurg-2014-011460. Epub 2015 Jan 20. PubMed PMID: 25603808.

5) , 15)

Yavuz K, Geyik S, Saatci I, Cekirge HS. Endovascular treatment of middle cerebral artery aneurysms with flow modification with the use of the pipeline embolization device. AJNR Am J Neuroradiol. 2014 Mar;35(3):529-35. doi: 10.3174/ajnr.A3692. Epub 2013 Sep 26. PubMed PMID: 24072620.

6) , 16)

Pierot L, Klisch J, Cognard C, Szikora I, Mine B, Kadziolka K, Sychra V, Gubucz I, Januel AC, Lubicz B. Endovascular WEB flow disruption in middle cerebral artery aneurysms: preliminary feasibility, clinical, and anatomical results in a multicenter study. Neurosurgery. 2013 Jul;73(1):27-34; discussion 34-5. doi: 10.1227/01.neu.0000429860.04276.c1. PubMed PMID: 23615104.

7) , 13)

Caroff J, Neki H, Mihalea C, D’Argento F, Abdel Khalek H, Ikka L, Moret J, Spelle L. Flow-Diverter Stents for the Treatment of Saccular Middle Cerebral Artery Bifurcation Aneurysms. AJNR Am J Neuroradiol. 2016 Feb;37(2):279-84. doi: 10.3174/ajnr.A4540. Epub 2015 Sep 24. PubMed PMID: 26405085.

8) , 12)

Topcuoglu OM, Akgul E, Daglioglu E, Topcuoglu ED, Peker A, Akmangit I, Belen D, Arat A. Flow Diversion in Middle Cerebral Artery Aneurysms: Is It Really an All-Purpose Treatment? World Neurosurg. 2016 Mar;87:317-27. doi: 10.1016/j.wneu.2015.11.073. Epub 2015 Dec 23. PubMed PMID: 26723288.

17)

Burrows AM, Zipfel G, Lanzino G. Treatment of a pediatric recurrent fusiform middle cerebral artery (MCA) aneurysm with a flow diverter. J Neurointerv Surg. 2013 Nov;5(6):e47. doi: 10.1136/neurintsurg-2012-010478.rep. Epub 2012 Nov 27. PubMed PMID: 23188788.

Update: Optic chiasma cavernous malformation

Optic chiasma cavernous malformation

Epidemiology

Suprasellar occurrences of cavernous malformations (CM) in the optic chiasm are extremely uncommon, representing less than 1% of all CNS CMs 1) 2).

To the best of the knowledge of Abou-Al-Shaar et al. less than 80 cases have been reported in the literature 3).

Clinical features

Patients with these lesions typically present with chiasmal apoplexy, characterized by sudden visual lossacute headaches, retroorbital pain, and nausea 4).

These symptoms typically occur after a period of transient blurry vision and headaches. In addition, hypopituitarism from direct compression of the pituitary stalk has been reported in the literature 5) 6).

Diagnosis

On CT scan, optic pathway CMs appear as well-demarcated hyperdense lesions with or without calcifications 7).

MRI

MR imaging of cavernous hemangioma of the optic chiasm 8)

MRI is considered the most sensitive and specific imaging modality for the diagnosis of CM 9).

On T1-weighted images, CMs of the optic pathway demonstrate a hypointense to isointense appearance, whereas on T2-weighted images, they appear as heterogeneous “popcorn” lesions with mixed hyperintense and hypointense signals.

The hypointensity can be delineated further in the gradient-echo T2* images due to hemosiderin deposition in and around the CM. In addition, following intravenous gadolinium administration, minimal or no enhancement can be observed in the CM 10) 11).

It has been reported that CMs of the optic nerve and tract may show nerve thickening on coronal views, whereas CMs of the optic chiasm often appear as focal round masses 12).

Angiography is usually not helpful in diagnosing CMs because it does not delineate the lesion due to the low internal flow and high incidence of thrombosis 13).

Differential diagnosis

CMs of the optic pathway are commonly misdiagnosed as optic neuritisoptic gliomameningiomacraniopharyngiomavenous angiomaarteriovenous malformation, thrombosed intracranial aneurysm, and pituitary apoplexyhistiocytosishypothalamic gliomatuber cinereum hamartoma and metastasis 14) 15).

Cavernoma should be considered when a solid suprasellar mass has hemorrhage (mimicking cystic- adamantinomatous craniopharingioma).


Cavernoma and suprasellar meningioma are rarely associated. Holland and Symon report a patient, whose recovery after removal of the meningioma was complicated by haemorrhage from the cavernoma. This occurrence has not been previously reported 16).

Treatment

Surgical removal is the recommended treatment to restore or preserve vision and to eliminate the risk of future hemorrhage. However, the anatomical location and eloquence of nearby neural structures can make these lesions difficult to access and remove.

The surgical approach should allow optimal exposure of the lesion using the shortest route and with minimal brain retraction. Various surgical approaches have been reported in the literature including pterional, orbitozygomatic, supraorbital, subfrontal, and transbasal interhemispheric approaches. Almost half of the cases reported in the literature were managed through the frontotemporal approach 17).

Biopsy is contraindicated for these lesions due to the high risk of bleeding and symptomatic worsening 18) 19).

Reviews

2006

In their meticulous review of the literature, Lehner et al. found 42 previously reported patients with vascular malformations within optic nerves, chiasm, or optic tracts, 30 of them being cavernous hemangiomas. The optic chiasma was involved in 38 patients (90.5%) and a total excision of the tumor was performed in 21 cases 20).

Case reports

2016

A 33-year-old female presented 3 months postpartum with a headache of moderate severity and progressive visual loss in both eyes. On examination, the patient’s Glasgow coma scale (GCS) was 15/15. Visual field examination showed left homonymous incomplete hemianopia. Her visual acuity was 20/25 in the right eye and 20/30 in the left eye. Her discs and macula were healthy bilaterally. Extraocular movements were intact and pupils were reactive. The rest of her examination was unremarkable. Complete endocrine workup was normal.

Magnetic resonance imaging (MRI) revealed a large heterogeneous, hyperintense, hemorrhagic right suprasellar extra-axial complex cystic structure measuring 31 × 30 × 90 mm on T1-weighted images. There was mass effect on the adjacent hypothalamus and third ventricle displacing them toward the left and superiorly in addition to the optic pathway. The pituitary stalk was displaced toward the left. The lesion encased the right posterior cerebral artery and displaced the right carotid artery laterally.

Computed tomography (CT) arteriography demonstrated a completely thrombosed center. The imaging findings were compatible with suprasellar CM.

The patient underwent right frontal craniotomy and gross total resection of her suprasellar intrachiasmatic large infiltrative hemorrhagic CM. Organizing blood clots with reactive fibrohistiocytic and inflammatory reaction admixed with some ectatic vascular channels suggestive of a vascular malformation were noted. There were small foci admixed with granulation tissue, showing some dilated cavernous spaces that would be compatible with a vascular malformation such as cavernous angioma. On immunohistochemistry, the lesion was CD163+, CD20 rare, CD3+, CD34+, CD31+, CD38+, CTK−, EMA plasma cells, GFAP−, S100 dendritic cells, SMA vascular smooth muscle.

The patient had an uneventful operative course. Her visual acuity improved to 20/20 in both eyes. Extraocular muscles showed mild limitation of both eyes in an upward gaze. Otherwise, she was stable with no neurological deficits. Follow-up MRI at 12 months revealed complete removal of the suprasellar hemorrhagic CM with no evidence of a residual lesion or recurrence 21).


Cavernous malformation of the optic chiasm: Neuro-endoscopic removal 22).


Trentadue et al. report a case in which the finding was incidentally detected in a 49-year-old man. They describe the imaging characteristics of the lesion in such a rare location, highlighting the role of magnetic resonance imaging (MRI) (specifically 3 Tesla) in the management of asymptomatic patients 23).

2015

A 48-year-old female presented with an insidious history of progressive visual loss. Magnetic resonance imaging (MRI) showed a CM in the suprasellar region. The patient was operated via a right pterional approach with a complete lesion removal. The postoperative course was uneventful. Early postoperative ophthalmological examination revealed minimal improvement of the vision in the left eye 24).

2014

The case of a 60-year-old woman from our institution with acute-on-chronic visual disturbance secondary to visual pathway CM is presented. Including the current patient, 70 cases of anterior visual pathway CM have been published to our knowledge. The average patient age is 34.8 ± standard deviation of 14.2 years, with a female preponderance (n = 37, 52.9%). The majority of patients had an acute (n = 44; 62.9%; 95% confidence interval [CI] 0.51-0.73) onset of symptoms. In at least 55.6% (n = 40) of patients, the cause of visual disturbance was initially misdiagnosed. The majority (91.4%; n = 64) of patients underwent craniotomy, with complete resection and subtotal resection achieved in 53.1% (n = 34; 95%CI 0.41-0.65) and 17.2% (n = 11; 95%CI 0.10-0.28) of all surgical patients, respectively. Comparing surgically managed patients, complete resection improved visual deficits in 59.0% (n = 20; 95%CI 0.42-0.75), while subtotal resection improved visual deficits in 50.0% (n = 5; 95%CI 0.24-0.76; p = 0.62). CM is an important differential diagnosis for suprasellar lesions presenting with visual disturbance. A high index of suspicion is required in its diagnosis. Expeditious operative management is recommended to improve clinical outcomes 25).

2012

Ning et al. report a 28-year-old male presenting with left homonymous hemianopsia. Magnetic resonance imaging (MRI) revealed an occupied lesion located in the right side of the optic chiasm, and a clinical diagnosis of chiasmal CM was made. Microsurgical excision was performed via anterolateral pterional craniotomy. The patient showed good recovery with slight improvement of the visual field deficits after the operation. No CM recurrence was discovered during the follow-up MRI scans 26).

2011

Rheinboldt and Blase report the case of a 31-year-old male who presented to the ER with a 1-week history of progressively worsening, throbbing, left retro-orbital headache, ptosis, and subjective worsening of short-term memory function. Initial review of systems and laboratory data were noncontributory. Non-contrasted CT demonstrated a large hyperdense mass centered in the suprasellar cistern without evidence of dissecting extra-axial hemorrhage. Though the initial appearance mimicked a basilar tip aneurysm or another primary extra-axial suprasellar pathology such as a hemorrhagic or proteinaceous craniopharyngioma, germinoma, or optic glioma, a second smaller, clearly intra-axial, hyperdense lesion was observed in the left periventricular forceps major white matter. Consideration for multiple cavernomas versus hypervascular metastatic disease such as renal malignancy, thyroid malignancy, or melanoma was raised. CTA confirmed normal intracranial vasculature. Subsequent MRI images showed an acutely hemorrhagic mass centered at the left paramedian hypothalamus and tuber cinereum with numerous secondary foci, demonstrating mature hemorrhagic elements and confirming the diagnosis of multiple cavernomas 27).

2008

A 33-year-old female who suffered from a recurrence of an intrachiasmatic cavernous malformation is presented. She had already undergone surgery in 1991 and 2001 and was admitted to our hospital with reduced vision in the right eye. After MRI, and diagnosis of recurrence of the cavernoma, a neurosurgical operation was performed using the pterional approach. The intraoperative situation was documented with micro photographs. The postoperative course was uneventful. The female described a minimal improvement of her vision. No postoperative complications were observed. To our knowledge, microsurgically complete extirpation of a recurrence of an intrachiasmatic cavernoma has not yet been reported in the literature 28).

2007

Santos-Ditto et al. present the case of a female patient who developed chiasmatic apoplexy and menstrual alterations. CT scanning showed a suprasellar hemorrhage. She underwent surgery with the presumptive diagnosis of pituitary tumor. At surgery, we find a brown-grayish lesion involving left optic nerve and chiasm. Cavernous angioma was diagnosed by histopathology. Cavernous angiomas constitute nearly 15% of all central nervous system vascular malformations. Location at the optic pathway is very rare, but must to be ruled out in the diagnosis of a patient with chiasmatic and/or optic apoplexy. Surgery is useful in preventing worsening of the previous deficit or a new visual defect 29).


A 15-year-old boy presented with an extremely rare optochiasmatic cavernous angioma. He was admitted to a special hospital with the complaint of blurred vision persisting for 1 month. Magnetic resonance imaging and biopsy of the lesion were inconclusive. He was admitted to the neurosurgical clinic after worsening of the visual symptoms 9 months later. Repeat magnetic resonance imaging showed optochiasmatic cavernous angioma which had doubled in size. The lesion was removed completely without any problem. Postoperatively his visual complaints remained stable, but had improved after 1 year. Optochiasmatic cavernous malformation should be treated by surgical excision, whereas biopsy is useless and may result in enlargement 30).


A 38-year-old male patient who suffered from acute onset of severe headache and progressive loss of vision. The vascular malformation of the optic pathways was completely removed via a pterional approach. This is the first reported instance of complete resection of a cavernoma involving the optic nerve, the chiasm, and the optic tract 31).

2006

Muta et al. report a 14-year-old boy with cavernous malformation of the optic chiasm. He had a 2-year history of gradually worsening visual disturbance. Computed tomography (CT) and magnetic resonance imaging (MRI) revealed a suprasellar mass, findings compatible with craniopharyngioma. The mass was biopsied and histological examination confirmed cavernous malformation. On the second day after the biopsy, he suffered chiasmal apoplexy due to intratumoural haemorrhage, lost visual acuity and developed a field cut. Cavernous malformations arising from the optic nerve and chiasm are extremely rare; only 29 cases have been reported to date. Most patients manifested acute visual acuity and visual field disturbances. Although MRI findings of cavernous malformations in the brain parenchyma have been reported, MRI findings on the optic nerve and chiasm may not be completely diagnostic. Of the 29 documented patients, 16 underwent total resection of the lesion without exacerbation of their preoperative symptoms; in some cases, resection was complicated by risk of damage to the surrounding neural tissue. As patients may suffer intratumoural haemorrhage after biopsy or partial removal of the lesion, the advisability of surgical treatment of cavernous malformations of the optic nerve and chiasm must be considered carefully 32).


In their meticulous review of the literature, Lehner et al. found 42 previously reported patients with vascular malformations within optic nerves, chiasm, or optic tracts, 30 of them being cavernous hemangiomas. The optic chiasma was involved in 38 patients (90.5%) and a total excision of the tumor was performed in 21 cases. Lehner et al. published a patient with a cavernous haemangioma of the optic chiasma and left optic tract who presented with an acute defect of the right visual field and severe retro-orbital pain. They succeeded in total excision of the malformation via a neuronavigationally guided approach. In the postoperative course, vision of our patient improved immediately and was found to be completely normal three months after the surgical intervention. Considering this patient and the published cases in the literature, they are of the opinion that microsurgical excision is a safe and efficient treatment for these rare pathologies 33).

2005

Shkarubo et al. describe a rare case of chiasmatic apoplexy whose cause was chiasmatic cavernoma. In addition to acute visual disorders suggesting the involvement of the left optic nerve, chiasma, and left visual pathway, 23-year-old patient had endocrine disorders as polyuria, polydipsia, which first suggests craniopharyngioma and glioma of the chiasma. A capsule and hematomic clots were removed from the thickened left optic nerve and left chiasmatic half during surgery. Only did a morphological study involving immunohistochemical analysis permit identification of the process as hemorrhage from cavernous micromalformation with the formation of hematoma 34).

1989

Three patients with cavernomas of the optic nerve, chiasm, or optic tract are presented. All suffered progressive visual loss due to local hemorrhage and the space-occupying effects of the vascular malformation. Computed tomography scans revealed small lesions with mild contrast enhancement in the suprasellar and parasellar cisterns, whereas angiography was unremarkable. Magnetic resonance imaging was helpful in our cases both for diagnosis and for planning surgical approach, showing typical signs of cavernomas as confirmed by subsequent surgery and histological examination. The clinical and intraoperative findings are presented 35).

1984

Buonaguidi et al. report a very rare case of an intrasellar cavernous hemangioma mimicking, clinically and neuroradiologically, the presence of a nonfunctioning pituitary adenoma. It was possible to diagnose this benign, congenital vascular malformation only through a histological examination36).

1) , 4) , 12) , 14) , 17)

Liu JK, Lu Y, Raslan AM, Gultekin SH, Delashaw JB Jr. Cavernous malformations of the optic pathway and hypothalamus: analysis of 65 cases in the literature. Neurosurg Focus. 2010 Sep;29(3):E17. doi: 10.3171/2010.5.FOCUS10129. Review. PubMed PMID: 20809758.
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Tan T, Tee JW, Trost N, McKelvie P, Wang YY. Anterior visual pathway cavernous malformations. J Clin Neurosci. 2015 Feb;22(2):258-67. doi: 10.1016/j.jocn.2014.07.027. Epub 2014 Nov 11. Review. PubMed PMID: 25439746.
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Abou-Al-Shaar H, Bahatheq A, Takroni R, Al-Thubaiti I. Optic chiasmal cavernous angioma: A rare suprasellar vascular malformation. Surg Neurol Int. 2016 Aug 1;7(Suppl 18):S523-6. doi: 10.4103/2152-7806.187495. eCollection 2016. PubMed PMID: 27583178; PubMed Central PMCID: PMC4982351.
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Buonaguidi R, Canapicci R, Mimassi N, Ferdeghini M. Intrasellar cavernous hemangioma. Neurosurgery. 1984 Jun;14(6):732-4. PubMed PMID: 6462408.
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