Ethanol combined with coil embolisation for the treatment of arteriovenous malformations in a patient with Parkes Weber syndrome

Parkes Weber syndrome is a rare congenital condition of the vascular system with severe symptoms and life-threatening complications. The challenge is to manage the arteriovenous malformations, and there is no consensus on optimal treatment. We report the case of an 18-year-old woman with Parkes Weber syndrome who was treated with ethanol combined with coil embolisation at an early stage. After two sessions of embolisation, a significant devascularisation was achieved. No sign of recurrence was observed two years after the initial procedure. The patient’s symptoms and signs were greatly relieved during the follow-up period. This case raises awareness of Parkes Weber syndrome and highlights the importance of timely intervention, as well as offering a promising therapeutic option for this condition.

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Massive haemorrhagic complications of ruptured pulmonary arteriovenous malformations: outcomes from a 12 years’ retrospective study

BMC Pulmonary Medicine ◽

10.1186/s12890-021-01604-5 ◽

2021 ◽

Vol 21 (1) ◽

Author(s):

Xu Ma ◽

Bing Jie ◽

Dong Yu ◽

Ling-Ling Li ◽

Sen Jiang

Keyword(s):

Arteriovenous Malformations ◽

Arterial Oxygen Saturation ◽

Arterial Oxygen ◽

Hereditary Haemorrhagic Telangiectasia ◽

Pulmonary Arteriovenous Malformations ◽

Imaging Data ◽

Peripheral Lung ◽

Life Threatening ◽

The Mean

Abstract Background The life-threatening haemorrhagic complications of pulmonary arteriovenous malformations (PAVMs) are extremely rare, and only described in isolated cases. This study was designed to comprehensively investigate management of ruptured PAVMs. Methods We retrospectively assessed clinical and imaging data of ruptured PAVMs to summarize incidence, clinical characteristics, and outcomes following embolisation between January 2008 and January 2021. Results Eighteen of 406 (4.4%) patients with PAVMs developed haemorrhagic complications. Twelve of 18 patients were clinically diagnosed with hereditary haemorrhagic telangiectasia (HHT). Haemorrhagic complications occurred with no clear trigger in all cases. Eight of 18 patients (44.4%) were initially misdiagnosed or had undergone early ineffective treatment. 28 lesions were detected, with 89.3% of them located in peripheral lung. Computed tomography angiography (CTA) showed indirect signs to indicate ruptured PAVMs in all cases. Lower haemoglobin concentrations were associated with the diameter of afferent arteries in the ruptured lesions. Successful embolotherapy was achieved in all cases. After embolotherapy, arterial oxygen saturation improved and bleeding was controlled (P < 0.05). The mean follow-up time was 3.2 ± 2.5 years (range, 7 months to 10 years). Conclusions Life threatening haemorrhagic complications of PAVMs are rare, they usually occur without a trigger and can be easily misdiagnosed. HHT and larger size of afferent arteries are major risk factors of these complications. CTA is a useful tool for diagnosis and therapeutic guidance for ruptured PAVMs. Embolotherapy is an effective therapy for this life-threatening complication.

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The long-term outcomes of radiosurgery for arteriovenous malformations in pediatric and adolescent populations

Journal of Neurosurgery Pediatrics ◽

10.3171/2015.1.peds14407 ◽

2015 ◽

Vol 16 (2) ◽

pp. 222-231 ◽

Cited By ~ 29

Author(s):

Shunya Hanakita ◽

Tomoyuki Koga ◽

Masahiro Shin ◽

Hiroshi Igaki ◽

Nobuhito Saito

Keyword(s):

Adverse Events ◽

Arteriovenous Malformations ◽

Therapeutic Option ◽

Long Term Outcomes ◽

Bleeding Rate ◽

Study Institute ◽

Delayed Complications ◽

Risk Of Hemorrhage

OBJECT Although stereotactic radiosurgery (SRS) has been accepted as a therapeutic option for arteriovenous malformations (AVMs) in children and adolescents, substantial data are still lacking regarding the outcomes of SRS for AVMs in this age group, especially long-term complications. This study aimed to clarify the long-term outcomes of SRS for the treatment of AVM in pediatric patients aged ≤ 18 years. METHODS Outcomes of 116 patients who were aged 4–18 years when they underwent SRS between 1990 and 2009 at the study institute were analyzed retrospectively. RESULTS The median follow-up period after SRS was 100 months, with 6 patients followed up for more than 20 years. Actuarial obliteration rates at 3 and 5 years after SRS were 68% and 88%, respectively. Five hemorrhages occurred in 851 patient-years of follow-up. The annual bleeding rate after SRS before obliteration was calculated as 1.3%, which decreased to 0.2% after obliteration. Shorter maximum nidus diameter (p = 0.02) and higher margin dose (p = 0.03) were associated with a higher obliteration rate. Ten patients experienced adverse events after SRS. Of them, 4 patients presented with delayed complications years after SRS (range 9–20 years after SRS). CONCLUSIONS SRS can reduce the risk of hemorrhage in pediatric and adolescent AVMs, with an acceptable risk of complications in the long term. However, adverse events such as expanding hematoma and radiation necrosis that can occur after substantial follow-up should be taken into account at the time that treatment decisions are made and informed consent is obtained.

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Intraoperative Radiotherapy Is Not a Better Alternative to Whole Breast Radiotherapy as a Therapeutic Option for Early-Stage Breast Cancer

Frontiers in Oncology ◽

10.3389/fonc.2021.737982 ◽

2021 ◽

Vol 11 ◽

Author(s):

Linwei Wang ◽

Min Sun ◽

Shuailong Yang ◽

Yuanyuan Chen ◽

Tian Li

Keyword(s):

Breast Cancer ◽

Early Stage ◽

Therapeutic Option ◽

Meta Analysis ◽

Intraoperative Radiotherapy ◽

Recurrence Risk ◽

Early Stage Breast Cancer ◽

Breast Radiotherapy ◽

Whole Breast Radiotherapy

ObjectiveIntraoperative radiotherapy (IORT) in early-stage breast cancer has been studied over the years. However, it has not been demonstrated whether IORT is more suitable as a therapeutic option for early-stage breast cancer than whole breast radiotherapy (WBRT). Therefore, we performed a meta-analysis to compare the efficacy and safety of IORT to those of WBRT as therapeutic options for early-stage breast cancer patients receiving breast-conserving surgery (INPLASY2020120008).MethodsPubMed, Embase, and Cochrane Library databases were searched from inception to October 2021. Computerized and manual searches were adopted to identify eligible randomized control trials from online databases. Risk ratio (RR) and 95% confidence intervals (CI) were calculated by random-effect models to assess the relative risk. Potential publication bias was quantified by Begg’s and Egger’s tests.ResultsBased on our inclusion criteria, 10 randomized control trials involving 5,698 patients were included in this meta-analysis. This meta-analysis showed that the IORT group was associated with a higher local recurrence risk (RR = 2.111, 95% CI, 1.130–3.943, p = 0.0191), especially in the long-term follow-up subgroup or published after 2020 subgroup or Caucasian subgroup (RR = 2.404, 95% CI, 1.183–4.885, p = 0.0154). Subgroup analysis showed that the IORT group had a higher recurrence risk than the WBRT group in the polycentric randomized controlled trial subgroup (RR = 1.213, 95% CI, 1.030–1.428, p = 0.0204). Pooled analysis showed that there was no statistically significant difference in overall survival, recurrence-free survival, distant metastasis-free survival, and cancer-specific survival between IORT and WBRT groups. Additionally, the risk of skin toxicity was reduced, but the incidences of fat toxicity, edema, and scar calcification were significantly increased in the patients who underwent IORT in comparison to those who underwent WBRT.ConclusionThis meta-analysis revealed that IORT was not a better alternative to WBRT. More large-scale and well-designed clinical trials with longer follow-up periods are encouraged to further investigate the value of IORT.Systematic Review Registrationhttps://inplasy.com/inplasy-2020-12-0008/.

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Plasma Exchange as a Rescue Therapy for Acute Liver Failure

JOURNAL FOR CLINICAL AND DIAGNOSTIC RESEARCH ◽

10.7860/jcdr/2021/49865.15371 ◽

2021 ◽

Author(s):

Nupur B Patel ◽

Anand Sharma ◽

Itish Patnaik ◽

Ashok Kumar

Keyword(s):

Liver Failure ◽

Acute Liver Failure ◽

Plasma Exchange ◽

Resource Constraints ◽

Rescue Therapy ◽

Therapeutic Option ◽

Liver Function Tests ◽

Threatening Condition ◽

Life Threatening

Acute Liver Failure (ALF) is a life-threatening condition and often necessitates Liver Transplantation (LT). However, LT is not available to most patients in developing countries due to resource constraints. Here, authors presents a case of 30-year-old female with ALF and fulfilled the criteria for LT. The aetiology of ALF could not be diagnosed in her. Due to the lack of LT facilities, she was offered plasma exchange as a therapeutic option, which resulted in improvement in sensorium and Liver Function Tests (LFT) {bilirubin, International Normalised Ratio (INR), Aspartate Aminotransferase (AST), Alanine Aminotransferase (ALT)} over a period of two weeks. She was discharged and was doing well during follow-up. Plasma exchange is a less studied but potential treatment option for ALF when LT is not feasible.

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Disfiguring high-flow cervicofacial arteriovenous malformations

Journal of Surgical Case Reports ◽

10.1093/jscr/rjaa435 ◽

2020 ◽

Vol 2020 (10) ◽

Author(s):

Alaa Nabawi ◽

Nader Abraham ◽

Ayman Nabawi

Keyword(s):

Oral Cavity ◽

Vascular Malformation ◽

Arteriovenous Malformations ◽

Case Series ◽

High Flow ◽

Vascular Anomalies ◽

Maxillofacial Region ◽

Clinical Onset ◽

Life Threatening

Abstract Arteriovenous malformations (AVMs) are congenital vascular anomalies resulting from defects in angiogenesis. Approximately 40% of AVMs go undetected after birth and only experience the delayed clinical onset of symptoms in adulthood. AVMs are rare, representing only 1.5% of all vascular anomalies. The most common sites for the aberrant vascular nidus are the oral cavity and maxillofacial region, which represent 50% of the cases. AVMs are the most challenging and life-threatening form of vascular malformation. Exsanguination, thrombus detachment and embolization are the most hazardous operative risks. Small case series revealed a 75% recurrence rate during a 5-year follow-up, which adds another layer of complexity to their management. Large lesions in the head and neck cause deformation to the patient and present a challenge to the surgeon during their excision among vital structures and reconstruction of the 3D complex defects.

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Focal fractionated radiotherapy for intramedullary spinal arteriovenous malformations: 10-year experience

Journal of Neurosurgery Spine ◽

10.3171/spi.2003.99.1.0034 ◽

2003 ◽

Vol 99 (1) ◽

pp. 34-38 ◽

Cited By ~ 14

Author(s):

Kazutoshi Hida ◽

Hiroki Shirato ◽

Toyohiko Isu ◽

Toshitaka Seki ◽

Rikiya Onimaru ◽

...

Keyword(s):

Radiation Dose ◽

Arteriovenous Malformations ◽

Therapeutic Option ◽

Fractionated Radiotherapy ◽

Contrast Enhanced ◽

Complete Obliteration ◽

History Of ◽

Spinal Arteriovenous Malformations ◽

Tomography Scanning

Object Radiosurgical treatment of spinal arteriovenous malformations (AVMs) is becoming a practical therapeutic option as methodology improves, but no comparative study has yet been published on focal fractionated radiotherapy. The authors report their experience with conventional and hypofractionated radiotherapy for spinal AVM. Methods Candidates for this study were patients who experienced symptoms due to an intramedullary AVM but were ineligible for embolization or surgery. Of 21 patients with spinal AVMs, 10 cases in a 10-year period met this criterion. Angiography and contrast-enhanced computerized tomography scanning were used for treatment planning in all cases. Fractionated radiotherapy was performed using a linear accelerator, extracranial immobilization system, and frequent orthogonal linacographic verification. The starting radiation dose was 32 Gy in two, 36 Gy in three, and 40 Gy in two patients, in a regimen involving 1.8 to 2—Gy daily fractions; this was recently changed to a hypofractionation schedule of 30 Gy (in eight sessions) in one and 20 Gy (in four sessions) in two patients. Results The follow-up period ranged from 26 to 124 months (median of 49 months). There were no hemorrhages nor any adverse reactions attributable to irradiation. Of the seven patients who consented to undergo follow-up angiography, the nidus size decreased in five, but complete obliteration did not occur in any patient. Conclusions Because no patient experienced adverse effects, the maximum tolerable radiation dose for the spinal cord associated with an AVM could not be identified, although it presumably is higher than those administered. The lack of rebleeding in patients in whom complete angiographic occlusion was absent suggests that the natural history of spinal AVMs may be less aggressive than previously reported.

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Emphysematous pyelonephritis: is conservative management effective? Our experience and review of literature

International Surgery Journal ◽

10.18203/2349-2902.isj20200531 ◽

2020 ◽

Vol 7 (3) ◽

pp. 849

Author(s):

Atul Kumar Khandelwal

Keyword(s):

Percutaneous Drainage ◽

Early Stage ◽

Abdominal Mass ◽

Review Of Literature ◽

Emphysematous Pyelonephritis ◽

Life Threatening ◽

Laboratory Investigations ◽

Time Of Presentation ◽

Time Of Admission

Background: Emphysematous pyelonephritis (EPN) is a urologic emergency caused by a life-threatening necrotizing infection of the kidney leading to an accumulation of gas in the renal parenchyma and perirenal tissue. we present the clinical details and outcome of twelve patients of managed at our institute and discuss their management and outcomes.Methods: Twelve consecutive patients with EPN were managed in our institute from July 2014 to July 2018. Data on demographic profile, clinical features, laboratory investigations, imaging studies, outcome of patients and follow up details were recorded.Results: Out of 12 patients with EPN, nine were female and three were male. Ten patients were diabetic (83%). All the diabetic patient had raised blood sugar at the time of admission ureteric stone was present in two nondiabetic patients. All the patients had fever at the time of presentation while localized flank pain was present in 6 (50%) patients. On examination, renal angle tenderness was present in ten patients while abdominal mass found in three patients. Pyuria was found in all patients while leukocytosis found in 10 patients. Two patients had thrombocytopenia while 4 had deranged renal parameters at the time of admission. Urine culture showed Escherichia coli in 8 patients and Klebsiella in two patients. Four patients required percutaneous drainage. Interval nephrectomy was done in one patient due to non-functioning kidney.Conclusions: Majority of patients diagnosed as emphysematous pyelonephritis were managed conservatively due to diagnosed at an early stage. Percutaneous drainage is successfully utilized in patients with more advanced disease.

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Diagnosis and Transvascular Embolization for Ruptured Pulmonary Arteriovenous Malformations: Outcomes from a 12 Years’ Retrospective Study

10.21203/rs.3.rs-100835/v1 ◽

2020 ◽

Author(s):

Xu Ma ◽

Bing Jie ◽

Dong Yu ◽

Ling-Ling Li ◽

Sen Jiang

Keyword(s):

Arteriovenous Malformations ◽

Simple Type ◽

Pulmonary Arteriovenous Malformations ◽

Imaging Data ◽

Test Analysis ◽

Kaplan Meier ◽

Hemorrhagic Complications ◽

Successful Resolution ◽

Life Threatening

Abstract Background Pulmonary arteriovenous malformations (PAVMs) are rare, and the life-threatening hemorrhagic complications, including hemoptysis and hemothorax, are extremely uncommon. However, the management of large series of ruptured PAVMs has not been described in recent years. Methods We retrospectively reviewed patients who developed ruptured PAVMs between January 2008 and December 2019. We assessed the clinical and imaging data to summarize the incidence and characteristics of ruptured PAVMs, and outcomes following embolization with coils or plugs. A paired-sample t-test analysis, Kaplan–Meier method and the simple linear regression were accessed, as appropriate.Results Eighteen (5.49%) of 328 patients diagnosed with PAVMs developed hemorrhagic complications. Twelve of these 18 patients were clinically diagnosed with hereditary hemorrhagic telangiectasia (HHT) (incidence, 66.67%). Eleven of 18 patients were female. Eight of the 11 females were diagnosed with HHT. Twenty-eight lesions were detected, and the simple type was more common (82.14%) than the complex type. 89.29% of lesions were located in the peripheral lung. Computed tomography angiography (CTA) showed indirect signs to predict the ruptured PAVMs in cases of hemothorax. Technical success of embolotherapy was achieved in all cases. Two days after treatment, pulmonary function was significantly improved and bleeding was controlled (P<0.05), and no patients showed recanalization of PAVMs. Two patients in the hemoptysis group died of other reasons during follow-up and the mean follow-up time was 3.22±2.52 years (range, 7 months to 10 years). The hemorrhage volume was linearly associated with the diameter of the afferent arteries in the ruptured lesions.Conclusions CTA was able to diagnose ruptured PAVMs, and embolotherapy led to successful resolution of symptoms in all 18 patients who had ruptured PAVMs. Ruptured PAVMs must be considered when managing patients with hemothorax or hemoptysis, especially female patients with HHT.

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124 Repeat Stereotactic Radiosurgery for Incompletely Obliterated Arteriovenous Malformations: An International Multicenter Study

Neurosurgery ◽

10.1093/neuros/nyx417.124 ◽

2017 ◽

Vol 64 (CN_suppl_1) ◽

pp. 227-228

Author(s):

Hideyuki Kano ◽

Nathaniel Sisterson ◽

Dale Ding ◽

Jason P Sheehan ◽

Roberto Martinez-Alvarez ◽

...

Keyword(s):

Stereotactic Radiosurgery ◽

Arteriovenous Malformations ◽

Target Volume ◽

Volume Reduction ◽

Factors Associated ◽

Initial Procedure ◽

International Multicenter Study ◽

Second Procedure ◽

Risk Of Hemorrhage

Abstract INTRODUCTION To evaluate the outcomes and risks of repeat stereotactic radiosurgery (SRS) for incompletely obliterated cerebral arteriovenous malformations (AVM). METHODS Six participating centers of the International Gamma Knife Research Foundation (IGKRF) identified 335 patients who had incompletely obliterated AVMs at a median follow-up of 45.6 months after initial SRS (range, 15–283 months). The median AVM target volume at initial SRS was 4.3 cc but was reduced to 1.4cc at the time of the second procedure. The median margin dose at both initial SRS and repeat SRS was 20 Gy. The median follow-up after repeat SRS was 64 months (range, 6–218 months). RESULTS >The actuarial rate of total obliteration by angiography or MRI after repeat SRS was 31%, 50%, 61%, and 80% at 3, 4, 5, and 10 years, respectively. Factors associated with a higher rate of AVM obliteration were smaller residual AVM target volume, and >50% of volume reduction after the initial procedure. The cumulative actuarial rates of new AVM hemorrhage after repeat SRS were 1.8%, 4.5%, 6.6%, 8.9% and 14.9% at 1, 2, 3, 5, and 10 years. Factors associated with a higher risk of hemorrhage after repeat SRS were a larger number of a prior hemorrhages, larger AVM target volume, and initial AVM volume reduction <50%. Symptomatic adverse radiation effect (ARE) developed in 29 patients (8.6%) after initial SRS and 27 patients (8.1%) after repeat SRS. CONCLUSION Repeat SRS for incompletely obliterated AVMs increases the eventual obliteration rate but patients remain at risk of hemorrhage until obliteration occurs. The best results for incompletely obliterated AVMs were seen in patients with a smaller residual AVM volume and no prior hemorrhages.

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Clinical Significance of Pulmonary Arteriovenous Malformation Reperfusion

Journal of Clinical Interventional Radiology ISVIR ◽

10.1055/s-0037-1604096 ◽

2017 ◽

Vol 01 (03) ◽

pp. 156-160 ◽

Cited By ~ 3

Author(s):

Claire Kaufman ◽

Katharine Henderson ◽

Jeffrey Pollak

Keyword(s):

Arteriovenous Malformation ◽

Clinical Significance ◽

Arteriovenous Malformations ◽

Clinical History ◽

Pulmonary Arteriovenous Malformation ◽

Initial Procedure ◽

Time Of Presentation ◽

First Time ◽

Embolization Procedure

AbstractThe purpose of this study was to assess the clinical significance of reperfused pulmonary arteriovenous malformations (rPAVM) after embolization. This study is a retrospective review of all patients from a single HHT center who underwent pulmonary arteriovenous malformation (PAVM) embolization between January 1, 2006 and December 30, 2007. Clinical history, imaging findings (including reperfusion), and symptoms seen in the setting of PAVMs were reviewed both at the time of embolization and during the subsequent follow-up through December 30, 2013. During the 2-year treatment period, 101 patients underwent PAVM embolization. During the initial embolization, 27 patients were found to have reperfusion of a previously embolized PAVM. During the follow-up period, none of these patients had recurrence of the rPAVM. Three patients who underwent an embolization procedure for the first time at the initial procedure were subsequently found to have rPAVM during the follow-up period. This resulted in a total of 30 patients with rPAVMs. Eighteen (60%) of the 30 patients were found to be symptomatic at the time of presentation with rPAVM. However, 14 had at least one other significant PAVM at the time of diagnosis of reperfusion. Only two symptomatic patients had only rPAVM, both with mild manifestations. Two patients were excluded from the study as their symptoms were attributed to other medical comorbidities. The majority of symptomatic patients with rPAVM had additional PAVMs that required embolization. Neither of the symptomatic patients with an isolated rPAVM had severe manifestations.

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