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Hereditary Hemorrhagic Telangiectasia and a Recurrent Brain Abscess
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A6999 - Hereditary Hemorrhagic Telangiectasia and a Recurrent Brain Abscess
Author Block: M. E. Rosenthal, S. V. Cherian, R. M. Estrada-Y-Martin; Pulmonary, Critical Care and Sleep Medicine, University of Texas Health-McGovern Medical School, Houston, TX, United States.
Introduction: Patients with hereditary hemorrhagic telangiectasia (HHT) can have pulmonary arteriovenous malformations (AVMs) which potentially predispose them to develop brain abscesses. We describe a case of HHT with a delayed diagnosis of recurrent brain abscess.
Case report: A 37-year-old woman with HHT presented to the Emergency department complaining of headache, right ear pain and dizziness for one week. Her headaches increased in severity and frequency over the previous 2 months. Eight months prior to admission she underwent a dental procedure. Physical examination was unremarkable. Brain magnetic resonance imaging (MRI) revealed a 1.5x1.3cm solitary ring enhancing lesion in the left occipital lobe, which was treated with craniotomy with resection of abscess and antibiotics. Chest computed tomography (CT) revealed multiple AVMs. A pulmonary angiography identified and successfully occluded a 1cm right lower lobe lesion. The patient remained asymptomatic for 3 years but repeat chest CT with contrast showed a new large left lower lobe AVM which could not be embolized successfully and she was advised to take prophylaxis prior to dental work. Unfortunately, she was lost to follow up. Five years later, she presented with oozing from a right posterior molar tooth for two months but denied any recent procedures, dental instrumentation or endoscopies. On examination, her left temporal visual field was decreased. Brain MRI further demonstrated a cluster of ring enhancing lesions measuring 3x3.4x2.7cm suspicious for large multi-loculated abscess and craniotomy with resection was performed. A chest CT showed an 8x15mm right upper lobe AVM, which was successfully managed with coil embolization.
Discussion: HHT is an inherited disorder characterized by AVMs which may occur in multiple organs. Approximately 20% of HHT patients develop a pulmonary AVM and brain abscesses may occur in up to 10% of these individuals. However, recurrent brain abscesses secondary to pulmonary AVMs with HHT are rare with few previously reported cases. The small number of affected patients limit the evidence in reducing recurrence aside from antibiotic prophylaxis prior to dental and surgical interventions. Coil embolization is the preferred treatment of pulmonary AVMs greater than 3mm in diameter. Patients remain at risk for brain abscess secondary to AVMs which are too small to embolize and reperfusion of a previously treated AVMs. Therefore, importance of close follow up with repeat imaging must be stressed to patients to avoid catastrophic complications.
Author Block: M. E. Rosenthal, S. V. Cherian, R. M. Estrada-Y-Martin; Pulmonary, Critical Care and Sleep Medicine, University of Texas Health-McGovern Medical School, Houston, TX, United States.
Introduction: Patients with hereditary hemorrhagic telangiectasia (HHT) can have pulmonary arteriovenous malformations (AVMs) which potentially predispose them to develop brain abscesses. We describe a case of HHT with a delayed diagnosis of recurrent brain abscess.
Case report: A 37-year-old woman with HHT presented to the Emergency department complaining of headache, right ear pain and dizziness for one week. Her headaches increased in severity and frequency over the previous 2 months. Eight months prior to admission she underwent a dental procedure. Physical examination was unremarkable. Brain magnetic resonance imaging (MRI) revealed a 1.5x1.3cm solitary ring enhancing lesion in the left occipital lobe, which was treated with craniotomy with resection of abscess and antibiotics. Chest computed tomography (CT) revealed multiple AVMs. A pulmonary angiography identified and successfully occluded a 1cm right lower lobe lesion. The patient remained asymptomatic for 3 years but repeat chest CT with contrast showed a new large left lower lobe AVM which could not be embolized successfully and she was advised to take prophylaxis prior to dental work. Unfortunately, she was lost to follow up. Five years later, she presented with oozing from a right posterior molar tooth for two months but denied any recent procedures, dental instrumentation or endoscopies. On examination, her left temporal visual field was decreased. Brain MRI further demonstrated a cluster of ring enhancing lesions measuring 3x3.4x2.7cm suspicious for large multi-loculated abscess and craniotomy with resection was performed. A chest CT showed an 8x15mm right upper lobe AVM, which was successfully managed with coil embolization.
Discussion: HHT is an inherited disorder characterized by AVMs which may occur in multiple organs. Approximately 20% of HHT patients develop a pulmonary AVM and brain abscesses may occur in up to 10% of these individuals. However, recurrent brain abscesses secondary to pulmonary AVMs with HHT are rare with few previously reported cases. The small number of affected patients limit the evidence in reducing recurrence aside from antibiotic prophylaxis prior to dental and surgical interventions. Coil embolization is the preferred treatment of pulmonary AVMs greater than 3mm in diameter. Patients remain at risk for brain abscess secondary to AVMs which are too small to embolize and reperfusion of a previously treated AVMs. Therefore, importance of close follow up with repeat imaging must be stressed to patients to avoid catastrophic complications.
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