Subacute Horizontal Diplopia, Jaw Dystonia, and Laryngospasm.

Jaw dystonia and laryngospasm in the context of subacute brainstem dysfunction have been described in a small number of diseases, including antineuronal nuclear antibody type 2 (ANNA-2, also known as anti-Ri) paraneoplastic neurologic syndrome. Severe episodes of laryngospasms causing cyanosis are potentially fatal. Jaw dystonia can also cause eating difficulty, resulting in severe weight loss and malnutrition. In this report, we highlight the multidisciplinary management of this syndrome associated with ANNA-2/anti-Ri paraneoplastic neurologic syndrome and discuss its pathogenesis.

A Case of Suspected Negative Pressure Pulmonary Edema after General Anesthesia.

Negative pressure pulmonary edema (NPPE) can occur rapidly after the release of an upper airway obstruction. In general anesthesia, NPPE can be caused by laryngospasm after extubation. This report describes a case in which NPPE was thought to have occurred after extubation during general anesthesia in a disabled person. The patient was a 28-yearold man, 160 cm in height and 56 kg in weight, who was scheduled for dental caries treatment under ambulatory general anesthesia due to intellectual disability. After induction of general anesthesia, nasal intubation was performed after sufficient oral suctioning to remove a large amount of serous secretion. After completion of dental treatment, pressurized extubation was performed after oral suctioning as sufficient spontaneous breathing and body movement were observed. Immediately after extubation, SpO2 dropped to 80%, subsequently recovering to 99% under oxygen administration at 5 liter/min with an oxygen mask. It dropped to approximately 85% again, however, when administration of oxygen was discontinued. Although communication with the patient was difficult, no expression of anguish or dyspnea was observed. A chest radiograph showed symmetric middle-lobe and lingular segment infiltrates, and the patient was transferred to the nearest general hospital. No obvious clinical findings other than a decrease in SpO2 were observed, suggesting NPPE as a result of airway narrowing due to secretions.

Laryngopharyngeal reflux induced sleep-related laryngospasm.

BACKGROUND: Sleep-related laryngospasm (SRL) has been defined as the sustained closure of the vocal cords during sleep. Studies have suggested that it is a rare manifestation of laryngopharyngeal reflux (LPR). Difficulties in diagnosing SRL and LPR have led to the condition being under-recognised in the clinical setting. AIMS: The aim of this study was to determine if LPR was the cause of the SRL symptoms seen in our patients. METHODS: A retrospective chart assessment of patients with SRL. Patients with risk factors for LPR were identified. These included smoking status, alcohol intake, a history of dyspepsia or history of gastroesophageal reflux disease, a history of late-night eating and a history of eating spicy or fatty foods before bed. A clinical diagnosis based on the history and response to management was made for the diagnosis of LPR. All were advised to refrain from late meals and those with signs of nasopharyngitis were commenced on proton pump inhibitor therapy. RESULTS: Nineteen patients (mean age ± SD: 57.21 ± 15.18) were included in the study. All had at least one risk factor for LPR. Ten (52.6%) had signs of nasopharyngitis on nasendoscopy. Following treatment, 17 (89.5%) reported no further SRL symptoms at 1-year follow-up. CONCLUSION: SRL is a largely unknown and under-diagnosed condition. We believe this study provides supportive evidence for the causal relationship between LPR and SRL.

Negative-pressure pulmonary edema after mammoplasty: a case report.

Negative-pressure pulmonary edema (NPPE) is a rare but life-threatening postoperative complication that occurs due to the acute obstruction of the upper airway. In our case report, we present a 25-year-old female patient who underwent elective mammoplasty under general anesthesia and developed NPPE 4 hours after extubation. The patient had a preoperative mallampati score of 3. After routine anesthesia induction, the patient was intubated with an endotracheal tube with a guide wire. Aspiration wasn't observed during extubation. The patient was followed in the post-anesthesia care unit (PACU) for 30 minutes with a saturation of 95% and was subsequently transferred to the service. Four hours after the operation, the patient was re-examined due to dyspnea and shortness of breath. Due to oxygen saturation of 88% and pO2of 56mmHg despite mask ventilation, the patient was admitted to the intensive care unit (ICU). A computed tomography (CT) scan revealed extensive diffuse ground-glass opacities and consolidations in both lungs. She did not respond to mask ventilation and was given non-invasive ventilation with continuous positive airway pressure (CPAP). Forced diuresis was induced with furosemide. Tachypnea resolved within 2 hours after CPAP was initiated, the patient did not require oxygen support and COVID-19 polymerase chain reaction (PCR) testing was negative. Subsequently, the patient was discharged to the clinical ward on postoperative day 1. When considering NPPE, early diagnosis and respiratory support are associated with reduced mortality and rapid recovery. Patients who develop laryngospasm during extubation must be closely monitored, and in the case of pulmonary edema, NPPE should be considered in the differential diagnosis.

Addition of magnesium sulfate to bupivacaine improves analgesic efficacy after tonsillectomy: A randomized trial and a CONSORT analysis.

OBJECTIVES: The main objective of this study was to compare the wound infiltration (peritonsillar fossa) of magnesium sulphate combined with bupivacaine, bupivacaine alone and saline solution on post-tonsillectomy pain in children. The accessory objectives were to evaluate the effect of magnesium sulphate infiltration on prevention of laryngospasm and occurrence of nausea/vomiting. METHODS: This study is a prospective; double blinded and randomized clinical trial. Seventy-five children undergoing tonsillectomy were enrolled. Patients were randomized into three groups using closed envelop technique. Group 1 (N=24) received saline solution (NaCl), group 2 (N=25) received 0.25% bupivacaine (1mg/kg) and group 3 received magnesium sulphate (5mg/kg) and 0.25% bupivacaine (1mg/kg) after tonsillectomy using three-point technique. Pain was evaluated using mCHEOPS scale. The occurrence of laryngospasm, nausea and vomiting was monitored. RESULTS: The mCHEOPS scores of the group 3 were significantly lower than those of the group 2 and 1 (P<0.001). Time to first analgesic administration was longer for the group 3 than for the groups 2 and 1 (P<0.001). The mean consumption of additional analgesic drugs was lower for the group 3 than the other groups (P<0.001). There were no episodes of laryngospasm in the group 3 in comparison with the other groups. The difference of the incidence of nausea and vomiting was not statistically significant (P=0.628). CONCLUSION: The adjunction of magnesium sulphate to bupivacaine proved to provide more efficient pain control than bupivacaine alone. However, the small number of participants and the absence of sampling at the P level of 0.005 do not allow to conclude with absolute certainty.

Complications during Fiberoptic Endoscopic Evaluation of Swallowing in 5,680 Examinations.

OBJECTIVE: To evaluate retrospectively the incidence of complications during fiberoptic endoscopic evaluation of swallowing (FEES) in 5,680 examinations. PATIENTS AND METHODS: 5,680 patients were evaluated at the Department of Otorhinolaryngology, Audiology and Phoniatrics of Pisa University Hospital between January 2014 and December 2018, involving both inpatients and outpatients. Most common comorbidities included neurological pathologies such as stroke (11.8%), neurodegenerative diseases (28.9%) and a history of previous head and neck surgery (24.6%). The evaluation was conducted by clinicians with experience in swallowing for a minimum of 10 years with the assistance of one or more speech-language pathologists. RESULTS: In all patients studied the endoscope insertion was tolerated, and it was possible to visualize the pharyngolaryngeal structures. Three subjects refused to undergo the procedure after being informed regarding the protocol and were therefore not included in this study. Most patients reported discomfort (70.1%) and gagging (20.8%). In a minority of patients complications were recorded, such as anterior epistaxis (0.1%), posterior epistaxis (0.02%), vasovagal crises (0.08%) and laryngospasm (0.04%). Especially laryngospasm was recorded in patients affected by amyotrophic lateral sclerosis. Multivariate binary logistic regression showed that discomfort (OR 9.944; CI 7.643-12.937), chronic gastrointestinal diseases (OR 2.003; CI 1.518-2.644), neurodegenerative diseases (OR 1.550; CI 1.302-1.846) and brain tumors (OR 1.577; CI 1.179-2.111) were risk factors associated with minor complications. CONCLUSIONS: FEES proved to be easy to perform, well tolerated by the patients and cost-effective. It can be performed at the patient's bedside, and it is characterized by a low rate of complications. As a matter of fact, normally only discomfort, gagging and/or vomiting are reported. Complications occurred only rarely, such as anterior or posterior epistaxis episodes or vasovagal crises, but these are still easily managed. Exceptionally, more severe complications are reported: adverse drug reactions to substances such as blue dye (methylene blue) and local anesthetics (not used in our protocol), and laryngospasm.

Laryngospasm in amyotrophic lateral sclerosis.

INTRODUCTION: Laryngospasm is an involuntary, sustained closure of sphincter musculature that leads to an unpleasant subjective experience of dyspnea and choking. It is an underreported symptom in amyotrophic lateral sclerosis (ALS). In this study we aimed to better characterize the prevalence and clinical characteristics of laryngospasm in ALS patients. METHODS: The medical records of 571 patients with ALS followed between 2008 and 2018 were searched for evidence of laryngospasm. A total of 23 patients with laryngospasm were identified and the data related to patient and laryngospasm characteristics were extracted. RESULTS: Laryngospasm was reported in 4% of ALS patients. Females comprised 57% of patients and their mean age was 63.4 years. Laryngospasm frequently manifested in patients with moderate bulbar dysfunction and seemed independent of respiratory function. Among laryngospasm patients, 26% were cigarette smokers and 13% had a history of gastroesophageal reflux. The most common reported trigger was excessive saliva irritating the vocal cords (35%) followed by eating a meal (17%). There was significant variation in laryngospasm frequency (up to 5 per hour) and duration (seconds to minutes). Most patients could not identify an effective coping mechanism, although 13% reported that drinking water was effective. DISCUSSION: Despite its low prevalence in ALS, laryngospasm should be included in the symptom inquiry. The present findings may improve patient care through increased recognition of the clinical features of laryngospasm in ALS patients, identifying a link between laryngospasm and moderate bulbar dysfunction, and highlighting trigger avoidance as a management strategy. Additional research is required to understand the pathophysiology and optimal treatment.

Sulcus vocalis in spasmodic dysphonia-A retrospective study.

BACKGROUND: Spasmodic dysphonia (SD) is a neurological condition of the larynx characterised by task specific, involuntary spasms of the intrinsic laryngeal muscles causing frequent voice breaks during speech. The current treatment modality involves Botulinum Toxin injections into the affected group of muscles. This has yielded satisfactory results in Adductor SD (ADSD) and mixed SD but not in Abductor SD (ABSD). Sulcus vocalis is a morphological condition of the vocal folds with invagination of the superficial epithelium into the lamina propria or deeper layers. It is characterised by breathiness in voice and hypophonia. In our voice clinic, patients diagnosed with SD were occasionally found to have a sulcus on flexible stroboscopy. Studies have revealed an asymmetric stimulation of both the adductor and abductor group of muscles in ABSD and a predominant possibly symmetric stimulation of the adductor group of muscles in ADSD. Our objective was to study any significant association between vocal fold sulcus and two groups within SD; group one being ADSD and group two being both ABSD and Mixed SD. A literature review did not reveal any studies suggesting an association between SD and vocal fold sulcus to date. METHODS: A retrospective review of the stroboscopic video recordings as well as file records of all patients diagnosed with SD between January 2016 and September 2019 was conducted at our voice clinic. The first author was the laryngologist who had diagnosed SD and its type on the basis of hearing the voice and making the patient perform various vocal tasks with and without flexible videostroboscopy. The SD patients were divided into two groups with the first group consisting of ADSD patients and the second group consisting of ABSD as well as Mixed SD patients. The presence or absence of vocal fold sulcus was noted in all the SD patients. Odds ratio was used to establish statistical significance of the presence of vocal fold sulcus in the two SD groups. RESULTS: Among the 106 patients of SD, 62 patients were males and 44 were females. A total of 84 patients were diagnosed as ADSD, 10 as ABSD and 12 as Mixed SD patients. Vocal fold sulcus was noted in 5 out of 84 patients of ADSD, 4 out of 10 patients of ABSD, and in 3 out of 12 patients of mixed SD. Odds Ratio of 7.37 (C.I. = 2.063-26.35) was obtained for the second group of patients i.e. ABSD and Mixed SD. CONCLUSION: Our study revealed a significant association between patients of SD having an abductor component (ABSD and mixed SD) and vocal fold sulcus. The two hypothesis proposed for this are the possibility of asymmetrical adductor and abductor muscle stimulation in SD being responsible for the development of a vocal fold sulcus or the primary presence of a vocal fold sulcus contributing to altered sensory feedback resulting in SD. Further study to evaluate this, as well as a study of the vocal response to medialisation procedures for patients of ABSD with sulcus is recommended.

Systematic Review of Negative Pressure Pulmonary Edema in Otolaryngology Procedures.

OBJECTIVE(S): Negative pressure pulmonary edema (NPPE) is a rare perioperative complication with a potentially fatal outcome. The aim of this study was to perform a systematic review of NPPE in adult otolaryngology procedures with the goal of identifying risk factors, clinical presentation, diagnosis, management and outcomes. METHODS: Systematic review performed using PubMed, Scopus, Web of Science, and Cochrane databases. RESULTS: Sixty-nine studies including data from 87 individual patients were included in this review. Fifty-six (68%) patients were male and the average patient age was 37 years old. Type 1 NPPE occurred in 63 (72%) cases, while type 2 NPPE accounted for 20 (23%) cases. The most common procedures leading to NPPE were septoplasty, rhinoplasty or sinus surgery (n = 22, 25%), directly laryngoscopy or bronchoscopy (n = 13, 15%), and tracheostomy or cricothyroidotomy (n = 11, 13%). The most employed treatment options included diuretics (n = 55, 63%) and mechanical ventilation (n = 54, 62%). Seventy-eight (90%) patients made a full recovery with an average time to NPPE resolution of 33 hours and an average length of hospitalization of 5.6 days. Five (6%) patients had a long-term morbidity and four (5%) patients died, with age and ICU stay increasing risk for death and long-term morbidity (OR 1.044 and 7.42, respectively, P < .05). CONCLUSION: Septoplasty, rhinoplasty and sinus surgery account for the majority of NPPE cases in adult otolaryngology procedures. Young, healthy patients are the most commonly involved with a slight male predominance. The vast majority of patients recover fully, however there is a significant risk for morbidity and mortality.

Successful use of VV ECMO in managing negative pressure pulmonary edema.

The indications for extracorporeal membrane oxygenation (ECMO) are expanding. Postobstructive pulmonary edema, also known as negative pressure pulmonary edema, can result in severe respiratory compromise and acute respiratory distress syndrome. We present a case of a 26-year-old female with laryngeal papillomatosis and laryngospasm after direct laryngoscopy, who developed severe NPPE refractory to mechanical ventilator support, which was successfully treated with veno-venous ECMO.

Concurrent Glossopharyngeal Neuralgia and Hemi-Laryngopharyngeal Spasm (HeLPS): A Case Report and a Review of the Literature.

BACKGROUND AND IMPORTANCE: Hemi-laryngopharyngeal spasm (HeLPS) has been recently described but is not yet widely recognized. Patients describe intermittent coughing and choking and can be cured following microvascular decompression of their Xth cranial nerve. This case report and literature review highlight that HeLPS can co-occur with glossopharyngeal neuralgia (GN) and has been previously described (but not recognized) in the neurosurgical literature. CLINICAL PRESENTATION: A patient with GN and additional symptoms compatible with HeLPS is presented. The patient reported left-sided, intermittent, swallow-induced, severe electrical pain radiating from her ear to her throat (GN). She also reported intermittent severe coughing, throat contractions causing a sense of suffocation, and dysphonia (HeLPS). All her symptoms resolved following a left microvascular decompression of a loop of the posterior inferior cerebellar artery that was pulsating against both the IXth and Xth cranial nerves. A review of the senior author's database revealed another patient with this combination of symptoms. An international literature review found 27 patients have been previously described with symptoms of GN and the additional (but not recognized at the time) symptoms of HeLPS. CONCLUSION: This review highlights that patients with symptoms compatible with HeLPS have been reported since 1926 in at least 4 languages. This additional evidence supports the growing recognition that HeLPS is another neurovascular compression syndrome. Patients with HeLPS continue to be misdiagnosed as conversion disorder. The increased recognition of this new medical condition will require neurosurgical treatment and should alleviate the suffering of these patients.

Negative-pressure pulmonary edema after laparoscopic appendectomy.

Negative-pressure pulmonary edema (NPPE) is acute-onset bilateral pulmonary interstitial edema. This condition can be caused by significant negative intrathoracic pressure generated by large inspiratory effort against acute upper airway obstruction. Postoperative NPPE is rare but potentially life-threatening if not recognized and treated promptly. This article describes a patient who developed postoperative NPPE following a laparoscopic appendectomy.

Population-Based Incidence of Potentially Life-Threatening Complications of Hypocalcemia and the Role of Vitamin D Deficiency.

OBJECTIVES: To determine the incidence of potentially life-threatening complications of hypocalcemia in infants and children in Olmsted County, Minnesota; and to determine if vitamin D deficiency contributed to these events and was, at the time of clinical presentation, considered as a possible cause. STUDY DESIGN: In this population-based descriptive study, data were abstracted from the Rochester Epidemiology Project, a medical record linkage system covering 95% of patients in Olmsted County, Minnesota. Participants were children aged 0-5 years who resided in Olmsted County between January 1, 1996 and June 30, 2017, and who received diagnoses of seizures, cardiomyopathy, cardiac arrest, respiratory arrest, laryngospasm, and/or tetany. The incidence of hypocalcemia plus a potentially life-threatening complication was calculated. RESULTS: Among 15 419 patients aged 0-5 years in Olmsted County during the study period, 1305 had eligible complications: 460 had serum calcium checked within 14 days of presentation and 85 had hypocalcemia. Patients were excluded when causes other than hypocalcemia likely triggered the complication, leaving 16 children whose complication was attributed to hypocalcemia. Three of these 16 patients had a serum 25-hydroxyvitamin D measurement and 2 were deficient (≤6 ng/mL [15 nmol/L]). Among children aged 0-5 years, the incidence of hypocalcemia plus a potentially life-threatening complication was 6.1 per 100 000 person-years (95% CI, 3.5-10.0). CONCLUSIONS: Vitamin D deficiency is an underinvestigated cause of complications of hypocalcemia in children. Serum calcium and 25-hydroxyvitamin D should be measured in children with these complications to identify possibly life-threatening vitamin D deficiency.

Negative pressure pulmonary edema: report of case series and review of the literature / Edema pulmonar por pressão negativa: relato de casos e revisão da literatura

Abstract Background and objectives: Negative pressure pulmonary edema occurs by increased intrathoracic negative pressure following inspiration against obstructed upper airway. The pressure generated is transmitted to the pulmonary capillaries and exceeds the pressure of hydrostatic equilibrium, causing fluid extravasation into the pulmonary parenchyma and alveoli. In anesthesiology, common situations such as laryngospasm and upper airway obstruction can trigger this complication, which presents considerable morbidity and requires immediate diagnosis and propaedeutics. Upper airway patency, noninvasive ventilation with positive pressure, supplemental oxygen and, if necessary, reintubation with mechanical ventilation are the basis of therapy. Case report: Case 1: Male, 52 years old, undergoing appendectomy under general anesthesia with orotracheal intubation, non-depolarizing neuromuscular blocker, reversed with anticholinesterase, presented with laryngospasm after extubation, followed by pulmonary edema. Case 2: Female, 23 years old, undergoing breast reduction under general anesthesia with oro-tracheal intubation, non-depolarizing neuromuscular blocker, reversed with anticholinesterase,presented with inspiration against closed glottis after extubation, was treated with non-invasiveventilation with positive pressure; after 1 hour, she had pulmonary edema. Case 3: Male, 44 yearsold, undergoing ureterolithotripsy under general anesthesia, without neuromuscular blocker,presented with laryngospasm after laryngeal mask removal evolving with pulmonary edema. Case 4: Male, 7 years old, undergoing crude fracture reduction under general anesthesia withorotracheal intubation, non-depolarizing neuromuscular blocker, presented with laryngospasmreversed with non-invasive ventilation with positive pressure after extubation, followed bypulmonary edema. Conclusions: The anesthesiologists should prevent the patient from perform a forced inspirationagainst closed glottis, in addition to being able to recognize and treat cases of negative pressurepulmonary edema.

Resumo Justificativa e objetivos: O edema pulmonar por pressão negativa ocorre por aumento da pressão negativa intratorácica após inspiração contra via aérea superior obstruída. A pressão gerada é transmitida aos capilares pulmonares e supera a pressão de equilíbrio hidrostático, o que causa extravasamento de líquido para o parênquima pulmonar e alvéolos. Em anestesiologia, situações comuns como laringoespasmo e obstrução de via aérea superior podem desencadear essa complicação, que apresenta considerável morbidade e exige diagnóstico e propedêutica imediatos. A desobstrução das vias aéreas superiores, ventilação não invasiva com pressão positiva, oxigênio suplementar e, se necessário reintubação com ventilação mecânica são a base da terapia. Relato de caso: Caso 1: Masculino, 52 anos, submetido a apendicectomia sob anestesia geral com intubação orotraqueal, uso de bloqueador neuromuscular adespolarizante, revertido com anticolinesterásico; apresentou laringoespasmo após extubação, seguido de edema pulmonar. Caso 2: Feminino, 23 anos, submetida a mamoplastia redutora sob anestesia geral com intubação orotraqueal, bloqueador neuromuscular adespolarizante revertido com anticolinesterásico, apresentou inspiração contra glote fechada após extubação, tratada com ventilação não invasiva com pressão positiva; após uma hora apresentou edema pulmonar. Caso 3: Masculino, 44 anos, submetido a ureterolitotripsia sob anestesia geral, sem bloqueador neuromuscular, apresentou laringoespasmo após retirada de máscara laríngea e evoluiu com edema pulmonar. Caso 4: Masculino, sete anos, submetido a redução cruenta de fratura sob anestesia geral com intubação orotraqueal, uso de bloqueador neuromuscular adespolarizante; apresentou laringo-espasmo revertido com ventilação não invasiva com pressão positiva após extubação, seguidode edema pulmonar. Conclusões: O anestesiologista deve evitar que o paciente faça inspiração forçada contra glotefechada, além de ser capaz de reconhecer e tratar os casos de edema pulmonar por pressãonegativa.

Acute Postobstructive Pulmonary Edema Following Laryngospasm in Elderly Patients: A Case Report.

Only a few cases regarding postobstructive pulmonary edema following laryngospasm in older patients aged more than 60 years have been reported; however, acute pulmonary edema or pulmonary hemorrhage would be more deadly to elderly patients who have cerebrovascular disease than young healthy adults. After review of the literature, we report an unusual case of a 67-year-old man with ischemic cerebrovascular disease, who underwent carotid angioplasty and stenting and experienced severe pulmonary edema and hemorrhage secondary to laryngospasm after general anesthesia with laryngeal mask airway. The patient required positive-pressure ventilation, supportive treatment, and active cerebroprotection in the intensive care setting for 3 days before the edema resolved, and subsequently made a complete recovery without new onset of neurologic sequelae. The possible pathophysiological mechanisms, precaution, and preventative strategy of postobstructive pulmonary edema in older patients are discussed.

[Negative pressure pulmonary edema: report of case series and review of the literature]. / Edema pulmonar por pressão negativa: relato de casos e revisão da literatura.

BACKGROUND AND OBJECTIVES: Negative pressure pulmonary edema occurs by increased intrathoracic negative pressure following inspiration against obstructed upper airway. The pressure generated is transmitted to the pulmonary capillaries and exceeds the pressure of hydrostatic equilibrium, causing fluid extravasation into the pulmonary parenchyma and alveoli. In anesthesiology, common situations such as laryngospasm and upper airway obstruction can trigger this complication, which presents considerable morbidity and requires immediate diagnosis and propaedeutics. Upper airway patency, noninvasive ventilation with positive pressure, supplemental oxygen and, if necessary, reintubation with mechanical ventilation are the basis of therapy. CASE REPORT: Case 1: Male, 52 years old, undergoing appendectomy under general anesthesia with orotracheal intubation, non-depolarizing neuromuscular blocker, reversed with anticholinesterase, presented with laryngospasm after extubation, followed by pulmonary edema. Case 2: Female, 23 years old, undergoing breast reduction under general anesthesia with orotracheal intubation, non-depolarizing neuromuscular blocker, reversed with anticholinesterase, presented with inspiration against closed glottis after extubation, was treated with non-invasive ventilation with positive pressure; after 1 hour, she had pulmonary edema. Case 3: Male, 44 years old, undergoing ureterolithotripsy under general anesthesia, without neuromuscular blocker, presented with laryngospasm after laryngeal mask removal evolving with pulmonary edema. Case 4: Male, 7 years old, undergoing crude fracture reduction under general anesthesia with orotracheal intubation, non-depolarizing neuromuscular blocker, presented with laryngospasm reversed with non-invasive ventilation with positive pressure after extubation, followed by pulmonary edema. CONCLUSIONS: The anesthesiologists should prevent the patient from perform a forced inspiration against closed glottis, in addition to being able to recognize and treat cases of negative pressure pulmonary edema.

Ictal laryngospasm monitored by video-EEG and polygraphy: a potential SUDEP mechanism.

A 56-year-old man with refractory bitemporal epilepsy was monitored in the Epilepsy Monitoring Unit (EMU). In a video-EEG captured seizure, brief oroalimentary automatisms were followed by increased inspiratory effort, accompanied by prominent, visible tracheal movements and audible inspiratory stridor. The patient's oxygen saturation rapidly declined to 62%; persistent severe hypoxemia ended with spontaneous effective respiration commencing at seizure end. Subsequent seizures necessitated intensive care unit admission for respiratory distress, and ventilator support. This case suggests that ictal laryngospasm, a rare seizure manifestation, may represent another potential mechanism of sudden unexpected death in epilepsy (SUDEP). [Published with video sequence on www.epilepticdisorders.com].