Postictal neurogenic pulmonary edema: Case report and brief literature review

    Postictal neurogenic pulmonary edema: Case report and brief literature review Oscar Romero Osorio, Juan Abaunza, Pieralessandro Lasalvia, Diana Sandoval Brice˜no, Daniel Nari˜no Gonzalez PII: DOI: Reference:

S2213-3232(17)30109-3 doi:10.1016/j.ebcr.2017.09.003 EBCR 241

To appear in:

Epilepsy & Behavior Case Reports

Received date: Revised date: Accepted date:

30 July 2017 13 September 2017 20 September 2017

Please cite this article as: Osorio Oscar Romero, Abaunza Juan, Lasalvia Pieralessandro, Brice˜ no Diana Sandoval, Gonzalez Daniel Nari˜ no, Postictal neurogenic pulmonary edema: Case report and brief literature review, Epilepsy & Behavior Case Reports (2017), doi:10.1016/j.ebcr.2017.09.003

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ACCEPTED MANUSCRIPT POSTICTAL NEUROGENIC PULMONARY EDEMA: CASE REPORT AND BRIEF LITERATURE REVIEW.

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Abstract

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We present a case of a 34-year-old woman with a history of focal epilepsy since adolescence who presented self-limited pulmonary edema following a generalized seizure. Authors:

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1. - Oscar Romero Osorio: Neurologist. Javeriana University. 2. - Juan Abaunza: Medical student Javeriana University. 3.- Pieralessandro Lasalvia: Physician Javeriana University. 4. - Diana Sandoval Briceño: Physician Javeriana University. 5. - Daniel Nariño Gonzalez: Neurophysiologist Javeriana University.

Clinical Case

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A 34-years-old female patient visits the emergency room. She had just presented with an episode consisting of initial déjà-vu, palpitation, epigastric feeling of emptiness. Afterwards she had leftward ocular and cephalic version, followed by tonic extension posture of all four extremities for 1 minute. All symptoms then resolved leaving her in a state of confusion approximately for 40 minutes. She has had epilepsy for 9 years prior to this event. Her typical ictal pattern included consisted in self-limited episodes of déjàvu sensation and anxiety every year. The last episode was 3 years ago, in this occasion she presented respiratory failure associated with seizure and required orotracheal intubation and intensive care unit. She was currently taking 400 mg daily of carbamazepine. With further inquire; she admitted interruption of the previous dose and sleep deprivation as probable causes of the convulsive episode. On initial examination, she was awake with normal vital signs, with no cardiovascular or neurological abnormality. However, she referred mild dyspnea during examination. Blood chemistries showed slight leukocytosis attributable to postictal state and normal arterial blood gases. An electrocardiogram (figure 1A) was performed showing sinus tachycardia, S1Q3T3 pattern and negative T waves, and chest radiography was normal (figure1B). Meanwhile the patients referred worsening dyspnea and de novo low flow oxygen requirement. We then performed a chest computed tomography angiogram, ruling out pulmonary embolism and showing signs of bilateral pulmonary edema (figure1C). The patient then showed favorable clinical course, with no dyspnea or oxygen requirement after 48 hours and was discharged. In the outpatient control, she was asymptomatic and her transthoracic echocardiogram did not show any alteration. Diagnosis of neurogenic pulmonary edema was considered as the cause of her dyspnea after ruling out other cardiac or pulmonary causes.

ACCEPTED MANUSCRIPT Discussion

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Neurogenic pulmonary edema (NPE) is defined as pulmonary edema or alveolar occupation produced very shortly after an event involving central nervous system (CNS) injury (1). The complexity of presentation of these cases makes difficult full understanding of their pathophysiology. However, hemodynamic, systemic inflammatory and CNS changes have been identified (1). Several possible NPE triggers have been described, including aneurysmal subarachnoid hemorrhage, cranial trauma, spinal cord injury, stroke, epilepsy and postoperative intracranial injury. The prevalence of NPE varies between the studies, reaching 71% in patients with intracranial hemorrhage (1,2). Clinically, NPE should be suspected in any patients suffering a CNS injury followed by acute onset of dyspnea and decreased PaO2 and FiO2 (1). In the context of epilepsy, NPE is associated with recurrent seizures and longer epileptic seizure. Possible differential diagnosis include atelectasis and aspiration pneumonia (3,4). Sudden unexpected death in epilepsy (SUDEP) is a relevant entity in epileptic patients. Its possible etiologies are central apnea, autonomic dysregulation and NPE. The latter seems to be the most frequent given that 52 out of 72 autopsies in patients with SUDEP had marked pulmonary edema or congestion (3,4).

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Two major mechanisms seems to produce NPE.

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The first one involve intense pulmonary and systemic vasoconstriction as a response to adrenergic release by brain injury (3,4) leading to an increase in capillary hydrostatic pressure and capillary permeability. Neurons located in the areas A1 in the hypothalamus and A5 in the upper region of the bulb have generate a seizureassociated catecholamine discharge through their projections to sympathetic spinal centers (1,2). Myocardial dysfunction may then ensue resulting in dilated cardiomyopathy, volume overload, increased cardiac work, arrhythmia and , eventually, cardiac arrest (1). The second mechanism involves inflammatory mediators. When arterial transmural pressure exceeds 40 mm Hg, endothelial damage causes release of tumor necrosis factor alpha, interleukin 1 and 6, which cause additional damage, and endothelin 1, which increases capillary permeability leading to edema (1). This milieu modifies nuclear expression of additional inflammatory mediators in T lymphocytes and macrophages, further augmenting the process (1). Animal models of epilepsy show destruction of alveolar cells mediated by mitogen-activated protein kinase (MAPK). Deregulation of apoptotic and anti-apoptotic pathways happen, leading to further damage to alveolar lining (5). Treatment of NPE centers on resolution of CNS injury and ventilation support. Control of ictal frequency and length is essential. Specific treatments for NPE have been proposed. Endothelin converting enzyme inhibitors or sympathectomy have been successfully tested in rats (6,7). Evidence is however insufficient for human treatment (2). In conclusion, NPE is a relevant condition in patients with epilepsy. Recurrent exposure to CNS injury puts them in danger respiratory dysfunction and fatal outcomes. Further research should focus on finding predictors of respiratory compromise in these patients and possible prophylactic interventions.

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Figure 1. Electrocardiogram (A), chest radiography (B) and chest computed tomography angiogram (C) performed during emergency room attention.

ACCEPTED MANUSCRIPT REFERENCES

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1. Baumann A, Audibert G, McDonnell J, Mertes PM. Neurogenic pulmonary edema. Acta Anaesthesiol Scand [Internet]. 2007 Apr [cited 2016 Jun 24] ;51(4):447–55. Available from: http://www.ncbi.nlm.nih.gov/pubmed/17378783

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2. Dutta G, Demetis S, Dutta G, Demetis S. Neurogenic Pulmonary Edema Associated with Underlying Lung Disease after a Breakthrough Seizure. Case Rep Med [Internet]. Hindawi Publishing Corporation; 2012 [cited 2016 Jun 24];2012:1–3. Available from: http://www.hindawi.com/journals/crim/2012/560942/

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3. Zhao H, Lin G, Shi M, Gao J, Wang Y, Wang H, et al. The mechanism of neurogenic pulmonary edema in epilepsy. J Physiol Sci [Internet]. Springer Japan; 2014 Jan 20 [cited 2016 Jun 24];64(1):65– 72. Available from: http://link.springer.com/10.1007/s12576-013-0291-6

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4. Kennedy JD, Hardin KA, Parikh P, Li C-S, Seyal M. Pulmonary edema following generalized tonic clonic seizures is directly associated with seizure duration. Seizure [Internet]. NIH Public Access; 2015 Apr [cited 2016 Jun 24];27:19–24. Available from: http://www.ncbi.nlm.nih.gov/pubmed/25844030

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5. Colice GL, Matthay MA, Bass E, Matthay RA. Neurogenic Pulmonary Edema1,2. Am Rev Respir Dis. American Lung Association ; 2015.

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6. Herbst C, Tippler B, Shams H, Simmet T. A role for endothelin in bicuculline-induced neurogenic pulmonary oedema in rats. Br J Pharmacol [Internet ]. 1995 Jul [cited 2016 Jun 25];115(5):753–60. Available from: http://www.ncbi.nlm.nih.gov/pubmed/8548173

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7. Poulat P, Couture R. Increased pulmonary vascular permeability and oedema induced by intrathecally injected endothelins in rat. Eur J Pharmacol[Internet]. 1998 Mar 5 [cited 2016 Jun 25];344(2–3):251– 9. Available from: http://www.ncbi.nlm.nih.gov/pubmed/9600661

ACCEPTED MANUSCRIPT Highlights

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Neurogenic pulmonary edema can be fatal and its early identification allows the initiation of medical surveillance and management. Seizures are a rare cause of pulmonary edema and may be recurrent. NPE is associated with recurrent seizures and longer epileptic seizure. Treatment of NPE centers on resolution of CNS injury and ventilation support.

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