SESSION TITLE: Pediatrics Case Report Posters
SESSION TYPE: Affiliate Case Report Poster
PRESENTED ON: Tuesday, October 27, 2015 at 01:30 PM - 02:30 PM
INTRODUCTION: We present 2 young children with intractable seizures who presented with massive hemoptysis found to be secondary to numerous aortopulmonary collaterals (APCs); this has never been previously described.
CASE PRESENTATION: Two children (ages 2 and 3), both with a history of full-term birth, global developmental delay, intractable seizures, and normal chromosomal microarray, presented within 2 months of each other with massive hemoptysis. Each required intubation for airway protection and underwent flexible bronchoscopy. Both were found to have pulmonary hemorrhage (case 1 in the right middle lobe; case 2 in the right lower lobe). No foreign body, mass, or vascular structure noted in the airways. Neither patient has cardiac nor pulmonary disease. Computed chest tomography angiography (CTA) revealed numerous collateral, tortuous vessels supplying both lungs. Coil embolization was performed, and neither has had further hemoptysis.
DISCUSSION: APCs usually develop in response to chronic hypoxia, and in pediatrics are most commonly seen in children with congenital heart disease. It is unclear why these two children developed so many collaterals. Neither patient has a history of pulmonary nor cardiac disease. Both patients were on multiple anti-epileptics including potassium bromide and trileptal, but there are no reported cases of APCs and anti-epileptics. Vascular endothelial growth factor (VEGF) is known to be a potent stimulator of angiogenesis, and it has been shown to increase in the neurons and glia of rats after seizures.(1) There is also evidence that adults with intractable epilepsy have chronically elevated VEGF levels in their seizure foci.(2) It has been demonstrated that VEGF levels in the serum of adults rise after electroconvulsive therapy. (3) Taken together, it is possibile there is a direct link between seizures and angiogenesis.
CONCLUSIONS: We theorize that intractable seizures likely leads to transient hypoxia, leading to increased VEGF levels and thus angiogenesis and formation of APCs. Further investigation is warranted.
Reference #1: Croll SD. Vascular Endothelial Growth Factor (VEGF) in Seizures: A Double-Edged Sword. NIH Public Access. Adv Exp Med Biol. 2004;548:57-68.
Reference #2: Rigau V. Angiogenesis is associated with blood-brain barrier permeability in temporal lobe epilepsy. Brain. 2007;130(7):1942-1956.
Reference #3: Minelli A. Association between baseline serum vascular endothelial growth factor levels and response to electroconvulsive therapy. Acta Psychiatr Scand. 2014;129(6):461-466. doi:10.1111/acps.12187.
DISCLOSURE: The following authors have nothing to disclose: Rachna Wadia, Stephanie Holm, Manisha Newaskar
No Product/Research Disclosure Information