Abstract:
:Pulmonary arteriovenous malformations (PAVMs) are relatively rare in children. They may occur both as single isolated anomalies and, much more commonly, as one of many widely distributed arteriovenous malformations (AVMs) in other organs and anatomic locations such as the liver, brain, nose and spine. The latter phenotypically defines the condition known as Hemorrhagic Hereditary Telengiectasia (HHT). We report a case of a 10 year-old female with an isolated endobronchial right lower lobe PAVM. The patient's chest radiograph and fine-cut contrast-enhanced CT (CECT) of the neck and thorax were both uncharacteristically normal and, hence, non-diagnostic. Surgical lobectomy was the only means by which to both diagnose and treat this life-threatening condition. This unique case points out the dilemma facing the physician in such a case with only two management options neither of which is ideal: to either obtain a diagnosis with biopsy which is minimally invasive yet life-threatening due to the potential for fatal hemorrhage, or to perform a pulmonary lobectomy which entails removal of an entire lobe of the lung without a pre-operatively confirmed indication to justify the procedure. To our knowledge, this is the first reported case of a child with an endobronchial PAVM who did not carry the diagnosis of HHT, had normal chest radiography, and had a normal fine-cut CECT of the neck and thorax where such a management dilemma arose. This case points out that a high index of suspicion for PAVMs must be maintained despite normal chest radiography and CECT of the chest. In such cases, empiric lobectomy becomes the sole therapeutic and definitive diagnostic intervention. We hope that such a case published in the literature serves as a guide to physicians confronting similar circumstances as the rarity of such a constellation of variables precludes higher levels of evidence reporting such as a retrospective case series or prospective randomized controlled clinical trials.
journal_name
Int J Pediatr Otorhinolaryngoljournal_title
International journal of pediatric otorhinolaryngologyauthors
Villardo RJ,Rosas-Salazar C,Spahr JE,Dohar JEdoi
10.1016/j.ijporl.2011.08.008subject
Has Abstractpub_date
2011-11-01 00:00:00pages
1463-7issue
11eissn
0165-5876issn
1872-8464pii
S0165-5876(11)00395-8journal_volume
75pub_type
杂志文章abstract::A 2-year-old patient with a recurrent endodermal sinus tumor of the nasopharynx is presented. After the first attempt to surgically excise the tumor it recurred, causing upper airway obstruction within 2 months. Combined treatment with surgery, radiotherapy and chemotherapy resulted in a disease-free survival of four ...
journal_title:International journal of pediatric otorhinolaryngology
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journal_title:International journal of pediatric otorhinolaryngology
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 传,历史文章,杂志文章,评审
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
doi:10.1016/j.ijporl.2003.11.021
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
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journal_title:International journal of pediatric otorhinolaryngology
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journal_title:International journal of pediatric otorhinolaryngology
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
doi:10.1016/j.ijporl.2009.07.012
更新日期:2009-10-01 00:00:00
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
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更新日期:2003-05-01 00:00:00
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
doi:10.1016/j.ijporl.2008.09.013
更新日期:2009-01-01 00:00:00
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更新日期:2019-05-01 00:00:00
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
doi:10.1016/j.ijporl.2011.12.026
更新日期:2012-04-01 00:00:00
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doi:10.1016/j.ijporl.2009.10.022
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更新日期:2018-01-01 00:00:00
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
doi:10.1016/j.ijporl.2003.11.002
更新日期:2004-03-01 00:00:00
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pub_type: 杂志文章
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更新日期:2003-08-01 00:00:00
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
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更新日期:2003-12-01 00:00:00
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pub_type: 杂志文章,评审
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pub_type: 杂志文章
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章
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更新日期:2009-05-01 00:00:00
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journal_title:International journal of pediatric otorhinolaryngology
pub_type: 杂志文章,评审
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更新日期:1998-01-01 00:00:00