[Spontaneous bilateral carotid-cavernous fistulas: about a case and review of the literature].
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ABSTRACT:
SUBMITTER: Belhachmi A
PROVIDER: S-EPMC5554656 | biostudies-other | 2017
REPOSITORIES: biostudies-other
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Project description:BACKGROUND:Vascular Ehlers-Danlos syndrome (vEDS) is a rare condition characterized by connective tissue fragility. Direct spontaneous carotid-cavernous fistula (sCCF) is reportedly pathognomonic of vEDS. We conducted this study to understand the possible mechanisms of occurrence of sCCF in this subset of patients. METHODS:We conducted a retrospective analysis of a monocentric vEDS cohort along with a literature review regarding sCCF in this condition. RESULTS:Of 133 patients regularly followed in our centre between 2000 and 2017, 13 (9.8%) had a diagnosis of direct sCCF (92.3% female, median age 33.0 years, interquartile range (IQR) [26.0-39.5]). There were 7 Glycine missense and 6 splice-site variants but no variant leading to haploinsufficiency. The literature search identified 97 vEDS patients with direct sCCF (79.4% female, 7.2% sex not reported, median age 31.0 years, IQR [24.0-39.0]). Increased carotid circumferential wall stress, higher carotid distensibility and lower carotid intima-media thickness could contribute to a higher risk for direct sCCF in vEDS. There is no predictive factor for the occurrence of sCCF apart from female sex in vEDS. CONCLUSIONS:In vEDS, anatomical and pathophysiological features of the intra-cavernous internal carotid artery make it prone to shunting in the cavernous sinus, due either to a spontaneous rupture or to a spontaneous dissection with pseudoaneurysm formation. Direct sCCF in seemingly healthy young individuals should be highly suggestive of vEDS and prompt further investigation.
| S-EPMC6019721 | biostudies-literature
Project description:Coronary artery fistula is an abnormal vascular communication of coronary artery with cardiac chambers or any segment of the systemic or pulmonary circulation. The prevalence is 0.9% of all coronary anomalies. Coronary artery fistula arises from the right coronary artery in approximately 50.0% of patients, from left coronary artery in approximately 42.0% of patients, and from both in approximately 5.0% of patients. Low-pressure structures are the most common sites of drainage of the coronary fistulas. If a large left-to-right shunt exists, it can be associated with potential complications, such as arterial aneurysm. Here we report an extremely rare case of a 76-year-old woman with bilateral coronary-to-pulmonary artery fistulas associated with giant aneurysms, detected by coronary angiography and confirmed with coronary computed tomography.
| S-EPMC6536618 | biostudies-literature
Project description:This report describes two cases of post-traumatic, high flow carotid-cavernous fistulas that demonstrated residual shunting after initial embolization with coils and Onyx, and that were successfully closed with pipeline embolization devices. Following their combined endovascular treatments, the patients experienced clinical improvement of symptoms with durable obliteration of the fistulous communications.
| S-EPMC4550904 | biostudies-other
Project description:PURPOSE:Patient-reported long-term follow-up after endovascular treatment of indirect carotid cavernous fistulas is important, but rarely addressed in literature. We report on this issue with a special focus on the patient's view and its impact on the indication evaluation process. METHODS:We retrospectively reviewed the records of all patients (n = 33) with a minimum follow-up interval of at least 36 and up to 166 months after endovascular treatment of an indirect carotid cavernous fistula (Barrow B-D) at our institution (treated from 01/2003 to 06/2015). We determined treatment details including primary therapy success and complication rate and quote the patient's subjective perception of the long-term treatment success using a standardized interview form. RESULTS:As a primary result the fistula was completely occluded in 25/33 cases (76%), while a downgrading was achieved in 8/33 (24%) of the cases. Secondary occlusion was observed in three out of eight patients (38%). In the long-term interview (response rate: 91%, median follow-up interval: 114 months) 87% of the patients reported high satisfaction with the long-term therapy result. Endovascular treatment achieved a sustainable relief from all eye-related symptoms in 89% and from pulsatile tinnitus in 57% of the cases. CONCLUSIONS:In addition to good results in terms of angiographic and clinical cure, endovascular treatment is also perceived as beneficial by most of the patients. This is another important argument in favor of an endovascular treatment.
| S-EPMC7147764 | biostudies-literature
Project description:Cavernous sinus angioleiomyoma (ALM) is extremely rare. Only three cases have been reported to be cavernous sinus ALM, and very few reports described characteristic findings for intracranial ALMs in detail. We report a new case of cavernous sinus ALM, with detailed information on the clinical presentation, radiology, pathology, and surgical approach. A 52-year-old woman had a 6-month history of right eye discomfort. Magnetic resonance imaging showed a right cavernous sinus tumor with heterogenous blush enhancement. Enhanced computed tomography scans and angiography showed small nodular enhancement in the tumor. Complete tumor resection was achieved via an extradural temporopolar approach. ALM was identified based on histologic examination. Intracranial ALMs are different from the ALMs that occur in the extremities based on our review of the literature. Intracranial ALMs appear more frequently in men than women. The cavernous type was the most common pathologically, and they occur often in the epiperidural location. Because cavernous sinus ALM occurs in the interdural space, an epidural approach should be selected. Therefore, it is important to include cavernous sinus ALM into a differential diagnosis of a cavernous sinus tumor. The blush enhancement and nodular enhancement within this lesion may be useful to distinguish cavernous sinus ALM from other differential diagnoses.
| S-EPMC4110136 | biostudies-other
Project description:ObjectiveTo describe clinical characteristics, imaging findings, morbidity, and mortality in a single-center cohort of 12 pediatric cavernous sinus thrombosis cases and to review all cases available in recent English literature.MethodsClinical data and radiographic studies on 12 cases from our institution were analyzed retrospectively. A literature search and review was conducted, with additional cases pooled with the new cohort for an aggregate analysis.ResultsTwelve cases of cavernous sinus thrombosis in children from the Children's Hospital of Philadelphia between January 1, 2000, and December 31, 2013, were reviewed. All patients survived to discharge; 3 of 12 (25%) experienced neurologic morbidity. Contrast-enhanced MRI and contrast-enhanced head CT were 100% sensitive for detecting cavernous sinus thrombosis, while noncontrast time-of-flight magnetic resonance venography (TOF MRV) and noncontrast head CT were 0% sensitive. Literature review produced an additional 40 cases, and the aggregate mortality rate was 4 of 52 (8%) and morbidity rate was 10 of 40 (25%). Outcomes did not vary by treatment or with unilateral vs bilateral cavernous sinus involvement. There was a trend toward worse outcomes with fungal infections.ConclusionOur case series demonstrates low morbidity and mortality with early, aggressive surgical, antimicrobial, and anticoagulation therapies. Although anticoagulation and surgery were not associated with significantly different outcomes, more study is needed.
| S-EPMC4553026 | biostudies-literature
Project description:BACKGROUND:Pampiniform plexus thrombosis is a very rare disease (only less than 25 published cases are available till date), and it is a diagnostic dilemma. The present case is an unusual condition of an elderly gentleman who was finally diagnosed as a case of spontaneous thrombosis of bilateral pampiniform plexus and was managed conservatively. Literature was reviewed to explore potential etiologies and therapeutic strategies. CASE PRESENTATION:A 65-year-old afebrile gentleman, laborer (in brick industry), and non-smoker with no previous major health problems was admitted with swelling in the bilateral inguinal region. The swelling had started one and half months ago. He had developed severe pain over the swelling for last 1?day with tenderness and indurations. Neither he had history of previous surgeries, chronic cough, dysuria, prostatism, and trauma nor he presented any thrombogenic factors. There was no history of vomiting, abdominal pain, and obstipation. Physical examination revealed normotensive person with BMI of 22.5, was significant only for one tender, movable, and firm to hard 10?cm?×?3?cm mass extending from the left deep inguinal ring up to the upper pole of the testis in the scrotum. Another 5 cm?×?3?cm mass of similar characteristics was found extending from deep inguinal ring up to the root of the scrotum on right side. The testes and prostate were normal on palpation. On the contrary to preoperative USG, which clinched suspicion of incarcerated inguinal hernia, a thrombosed pampiniform plexus without any evidence of hernia sac was found on the left side during inguino-scrotal exploration. Wound was closed without doing any further procedure. Contralateral inguino-scrotal exploration was spared considering same nature of disease. Postoperative Doppler ultrasonography confirmed the diagnosis of bilateral thrombosed pampiniform plexus. MDCT of whole abdomen revealed no abnormality other than bilateral spermatic cord thrombosis. Blood thrombophilia screening came normal. The subject had an uneventful postoperative hospital course. With 2 years of follow-up, the gentleman is doing well, remaining asymptomatic and had returned to his usual life. CONCLUSIONS:Due to extreme rarity, spontaneous thrombosis of the pampiniform plexus may be a diagnostic dilemma and requires a high index of suspicion. Doppler ultrasound is the initial investigation of choice. In the absence of other concomitant disease, beginning the treatment conservatively instead of excising the thrombosed segment is more suitable.
| S-EPMC7058780 | biostudies-literature
Project description:In this report, we are presenting a case of a 49-year-old female complaining of defective vision in the left eye. The main complaints were: pain, redness, mild proptosis, and high intraocular pressure. She had a history of uneventful phacoemulsification surgery 3 months prior to presenting to us. Investigations revealed a macular edema caused by central retinal vein occlusion and computed tomography angiography showed an early opacified left cavernous sinus with a dilated superior ophthalmic vein along with a fistula between the meningeal branches of the carotid arteries and the cavernous sinus. Improvement of ocular symptoms was achieved after endovascular treatment by transarterial and transvenous embolization.
| S-EPMC6637435 | biostudies-literature
Project description:BackgroundSchwannomas of the abducens nerve are a rare pathology and are encountered less within the cavernous sinus. We describe a case of sixth cranial nerve schwannoma, in the cavernous sinus.Case descriptionA 50-year-old lady, presented with 2 years history of double vision and left facial numbness that started 6 months before presentation, found to have hyperintense lobulated mass at the left cavernous sinus extending into Meckel's cave with bony remodeling on magnetic resonance imaging. She underwent left frontotemporal craniotomy, combined extra-intradural approach, gross total resection. She had a gradual recovery of the sixth cranial nerve function.ConclusionAbducens nerve schwannoma of the cavernous sinus is a rare and challenging tumor. However amenable to surgical intervention with favorable neurological outcome.
| S-EPMC7749956 | biostudies-literature
Project description: Not available
| S-EPMC5958930 | biostudies-literature