Perioral Cyanosis Research Articles

A Case of Atypical Delayed Anaphylaxis to Rifampin

Rifampin is a mainstay in anti-tuberculosis therapy and is generally well tolerated and safe to take at therapeutic dosing. While anaphylactic reactions to rifampin are exceedingly rare, prompt recognition and awareness is vital to patient safety, and alternative medications should be considered desensitization has been associated with significant morbidity. Here we report a case of a severe anaphylactic reaction to rifampin. A 65-year-old Asian woman was diagnosed with active tuberculosis and initiated on rifampin, isoniazid, pyrazinamide, and ethambutol (RIPE). A couple of weeks after initiating RIPE therapy, she developed urticaria and angioedema and therapy was held. Upon resumption of RIPE, she developed emesis, fever, chills, perioral cyanosis, and hypotension within a few hours of the first dose. She was admitted to the intensive care unit for a rifampin graded dose challenge and subsequently developed a severe anaphylactic reaction to rifampin four hours after the final dose of the challenge. She tolerated desensitization to isoniazid and ethambutol well and was prescribed an alternative anti-tuberculosis regimen of isoniazid, ethambutol, levofloxacin, and pyridoxine for a total of 18 months to complete her treatment.

AN UNCOMMON PRESENTATION OF ANTISYNTHETASE SYNDROME-RELATED INTERSTITIAL LUNG DISEASE IN A YOUNG INDIAN MALE

AN UNCOMMON PRESENTATION OF ANTISYNTHETASE SYNDROME-RELATED INTERSTITIAL LUNG DISEASE IN A YOUNG INDIAN MALE

Non-convulsive febrile status epilepticus mimicking a postictal state after a febrile seizure: an ictal electroclinical and evolutive study.

Febrile status epilepticus evolves from a febrile seizure (FS) in 5% of cases. Its prompt recognition is challenging, especially when motor manifestations are absent or subtle. We describe the ictal electroclinical features of non-convulsive febrile status epilepticus (NCFSE) following an apparently concluded FS, initially misinterpreted as postictal obtundation and in some way mimicking the described "non-epileptic twilight state". We present an electroclinical study of 18 children, collected in our unit, who presented with NCFSE after an apparently resolved FS, longitudinally followed for one year to seven years and nine months (mean: four years and three months). The age at first NCFSE ranged between one year and two months and five years and eight months (mean: two years and six months). Patients were examined after spontaneous or rectal diazepam-induced resolution of a FS, while showing persisting impairment of awareness. A lack of responsiveness to painful stimulation, abnormal posturing and aphasia were present in all cases, variably associated with perioral cyanosis, hypersalivation, automatisms, gaze deviation and other lateralizing signs; eyes were open. The EEG recording started 20 to 140 minutes after the apparent resolution of the FS and was invariably characterized by delta or theta-delta pseudorhythmic activity, mainly involving the fronto-temporal regions, with hemispheric predominance in two thirds of the cases. The electroclinical condition, lasting 25 to 210 minutes, quickly recovered after intravenous diazepam. Follow-up revealed normal neurodevelopment and EEG in almost all patients (learning disability emerged in three). In five subjects, NCSE relapsed (twice in two). None presented afebrile seizures. Our series highlights the electroclinical features of focal NCFSE. Distinctive elements are a lack of reactivity, cyanosis, lateralizing clinical and EEG signs, and resolution clearly tied to intravenous benzodiazepine administration.

HP35: Takotsubo cardiomyopathy complicated with non-convulsive status epilepticus

Takotsubo cardiomyopathy (TTC) is a cardiac condition that causes an unexpected waning of the heart occurring during a stressful emotional event and potentially leading to acute heart failure [Ghadri J 2018]. TTC can be associated with various complications including cardiogenic shock, life-threatening arrhythmias and cardiac arrest [Templin C 2015]. Here we present a case of a young woman with TTC complicating with cardiac arrest and non-convulsive status epilepticus and a good neurological outcome Case report: A 38-years-old woman with unremarkable past medical history, after a strong emotional distress presented acute chest pain, dyspnea, and loss of consciousness with perioral cyanosis and noisy breathing. Cardiopulmonary resuscitation was immediately initiated, which continued for 50 minutes until the appearance of the sinus rhythm on the ECG. Dilated apex and mid-ventricular segment akinesis were recorded on echocardiography. Brain CT was unremarkable. Continuous video-EEG monitoring showed malignant EEG patterns, firstly, suppressed and non-reactive background, followed 24h later by frontally predominant generalized spike and spike-wave discharges. Administration of diazepam 20 mg did not modify her clinical and EEG state, while infusion of phenobarbital 1200 mg [20mg/kg] resulted in significant EEG improvement. Under combined antiepileptic medication (i.e., valproate 500 mg bid and levetiracetam 750 mg bid, her EEG and clinical status improved in few days. Conclusion: Although non-convulsive status epilepticus after cardiac arrest is associated with poor outcome, our patient survived, with a Cerebral Performance Category score of 2. Good recovery in this case might be related to young age, reversible cardiomyopathy, and timely management of complications.

Univentricular Heart

Introduction: The univentricular heart refers to a pathophysiological concept in which a single functional ventricle provides both pulmonary and systemic aortic flow in parallel. The aim of this study was to conduct an epidemiological study of the univentricular heart, to study the circumstances of discovery, to analyze the problems posed by the diagnostic and therapeutic management of these patients in our Moroccan context by evaluating the diagnostic role of Doppler echocardiography and finally evaluating the evolution of these operated children. Methods: This was a retrospective study in the cardiovascular surgery department of CHU IBN ROCHD in Casablanca, over a period of 3 years from January 2017 to December 2019 involving 4 patients, whose age varies between 6 months and 7 years. Results: The sex ratio was 1. The average age of the patients was 8 months and 6 years respectively for the pulmonary artery cerclage and for the Fontan treatment. Consanguinity, diabetes, hypertension and advanced maternal age were the main antecedents. Several circumstances of discovery have been highlighted, the most common are: perioral cyanosis in 3 patients, growth restriction with eating difficulties in 3 patients and dyspnea in 2 others. Pulmonary arterial hypertension and pulmonary stenosis were present in 50% of the patients. The diagnosis was made by echocardiography and the most common anatomical form of the single ventricle was tricuspid atresia in 2 patients. All patients were operated: 2 received pulmonary artery cerclage and 2 others received Fontan treatment, 1 of them had primary pulmonary artery cerclage in his history. In our study, no deaths were reported. Postoperative complications were dominated by atelectasis, infectious complications, bleeding and pleuro-pericardial effusion. The short and medium-term postoperative evolution was favorable for all our patients with weight gain, symptom reduction and improvement in O2 saturation.

Quando lo squat fa la differenza

The paper presents the case of a 1-year-old girl with severe respiratory distress, perioral cyanosis and severe desaturation (SpO2 35% with oxygen in nasal prongs). An echocardiogram was readily performed showing the distinctive features of Fallot’s tetralogy. The child was placed in a squatting position (knee-chest position). Subcutaneous morphine at a dose of 0.2 mg/kg, bolus of physiological solution in 30 minutes and therapy with oral propanolol were administered, with progressive clinical improvement. Subsequently, corrective cardiac surgery was performed with regular postoperative period and following good clinical conditions. Cardiogenic causes must be considered among the causes of respiratory distress especially in low resource countries where congenital heart disease can be misdiagnosed or diagnosed belatedly.

Синдром ятагана: подходы к диагностике и ведению

В статье представлен клинический случай синдрома ятагана у 3-летней девочки. Заболевание манифестировало в месячном возрасте с бледности, периорального цианоза и эпизода апноэ. Ребенок госпитализирован для дальнейшего обследования. У девочки наблюдался пери­оральный цианоз, SpO2 90 %, частота дыхательных движений 36/мин. Аускультативно в легких пуэрильное дыхание, ­ослабленное справа по передней поверхности грудной клетки. Сердечные тоны ритмичные, систолический шум 2/6 вверху левого края грудины и 4/6 в межлопаточной области слева. При эхокардиографии диагностированы некритическая коарктация аорты, дефект межпредсердной перегородки. При рентгенографии органов грудной клетки в области правого легочного поля интенсивная тень от верхушки до уровня V ребра с нечеткими краями. Тень сердца смещена вправо. Что у ребенка: декстрокардия, декстрапозиция, пневмония, ателектаз, тимомегалия? При бронхоскопии обнаружена обструкция бронхов верхней и средней доли правого легкого с густой мокротой белого цвета. Проведена санация. После бронхоскопии нормализовалась сатурация, но, несмотря на ликвидацию причины ателектаза, контрольная рентгенография оставалась без изменений. Проведена компьютерная томография органов грудной полости с контрастированием. У ребенка диагностирован синдром ятагана в сочетании с коарктацией аорты и аберрантной правой подключичной артерией. В возрасте 2,5 мес. выполнена баллонная дилатация коарктации аорты. В возрасте 2 лет проведена хирургическая коррекция — устранение аномального дренажа легочных вен и пластика дуги аорты. Интраоперационная анатомия: мезокардия, правые отделы сердца дилатированы, нижняя доля правого легкого гипоплазирована. Наша пациентка фактически не имела симптомов, характерных для синдрома ятагана, поскольку отсутствовали признаки сердечной недостаточности, девочка не болела респираторными заболеваниями. Диагноз в грудном возрасте был установлен благодаря эпизоду аспирации, но ателектаз и мезокардия помешали визуализации характерной рентгенологической картины синдрома ятагана. Поэтому полное исследование с использованием компьютерной томографии с контрастированием является обязательным для таких пациентов.

Pulmonary veno-occlusive disease in childhood-a rare disease not to be missed.

Pulmonary veno-occlusive disease (PVOD) is a rare disease leading to pulmonary hypertension and potentially death related to right heart failure and/or respiratory insufficiency. Clinical symptoms are heterogenous and nonspecific: fatigue, decreased exercise tolerance, shortness of breath on exertion, cough, dizziness, chest pain with exercise, palpitations, syncope, as well as nonspecific symptoms such as headache, poor appetite, pallor or perioral cyanosis. Mutations in the EIF2AK4 (eukaryotic translation initiation factor 2-alpha kinase 4) have been recently described, other risk factors include exposure to organic solvent and trichloroethylene, tobacco exposure and chemotherapy. Echocardiography helps to estimate right ventricular systemic pressure, but further diagnostic workup includes cardiac catheterization to confirm pulmonary hypertension and increased pulmonary vascular resistance. High-resolution computed tomography reveals typical findings: centrilobular ground-glass nodules or opacities, septal lines, thickened interlobular septa, mosaic perfusion, and lymphadenopathy. Histology remains the gold standard, but carries risks for the patient. Proper workup is essential in order to avoid incorrect diagnosis. Pulmonary hypertension targeted treatment has been used in patients with PVOD, however, experience is limited, vasodilatory effects on pulmonary vasculature may lead to deterioration of the patients and should be used with great caution. Lung transplantation is currently the only valid treatment option for patients with PVOD. With prolonged waiting time and progression of the disease mechanical support could be considered.

ECG of the Month: Wide Complex Tachycardia in an Adult Patient with Surgically Corrected Congenital Heart Disease

A 27-year-old man with history of hypoplastic left-sided heart syndrome, status post-Fontan palliation surgery, and depressed right-sided ventricular function presented to the emergency department with acute-onset chest pain and shortness of breath while walking up steps to his home 90 minutes before presentation. He was ill appearing and diaphoretic on arrival. He was alert but had delayed capillary refill and perioral cyanosis. His initial vital signs were significant for pulse rate of 204 beats/min, respiratory rate of 22 breaths/min, blood pressure 132/75 mm Hg, and oxygen saturation of 88% on 3 L of nasal cannula oxygen.

Choking and Cyanotic Episodes in a 3-month-old Male.

1. Gabriel Paris, MS DO* 2. Nicholas L. Friedman, DO* 3. Jacob S. Hogue, MD* 1. *Madigan Army Medical Center, Tacoma, WA A previously healthy 3-month-old boy presents to the emergency department with a chief complaint of choking episodes with perioral cyanosis. The episodes started 4 weeks before and are increasing in frequency. During the episodes, his parents note rapid eye twitching, facial twitching, and a fine tremor in the extremities. He would appear to be choking with breath holding and cyanosis around the mouth. Some of the episodes are also accompanied by emesis. Many of the episodes occurred after feeding, so he was switched from breastfeeding to formula. Over the past month, parents have also noted a progressive decline in movement, head control, visual tracking, and increased sleepiness. Previously, he had been able to push his head and chest off a flat surface while prone and could roll from back to front but lost these skills. He also has regression in visual tracking, focusing on faces, and spontaneously smiling. He was born at 40 3/7 weeks’ estimated gestational age via spontaneous vaginal delivery to a gravida 3, para 3, mother without pregnancy or delivery complications. Maternal blood type was O+, and infant was A+ with direct antiglobulin test negative. Maternal laboratory studies were as follows: human immunodeficiency virus negative, hepatitis B surface antigen negative, Rubella immune, group B Streptococcus negative, and rapid plasma reagin negative. Newborn metabolic screen and hearing screen were normal. Family history is significant for a maternal uncle with Pelizaeus-Merzbacher disease (PMD). The details of the …

COVID-19-associated apnea and circumoral cyanosis in a 3-week-old

BackgroundData regarding coronavirus disease 2019 (COVID-19) cases and outcomes in infants are sparse compared to older pediatric and adult populations.Case presentationWe present a three-week-old full-term male with a history of mild hypoxic ischemic encephalopathy (HIE) who was admitted as an inpatient twice for episodes of apnea and perioral cyanosis. The patient tested positive for COVID-19 and negative for other common respiratory viruses at both admissions.ConclusionsTo our knowledge, this is the first report of apnea and perioral cyanosis associated with COVID-19 in an infant. This case highlights a previously undocumented COVID-19 presentation and suggests that even mildly symptomatic infants warrant viral diagnostic testing in an effort to prevent further spread of the disease.

A 3 Year Old with Hypoxia other Treatments Besides Oxygen

Background: Glucose-6-phosphatase Dehydrogenase (G6PD) deficiency causes acute hemolysis when exposed to oxidative stress. Methemoglobinemia has been associated with G6PD deficiency, and recognition is important to avoid further complications. Case Presentation: A 3-year-old healthy boy presents to the pediatric emergency department with acute onset of difficulty in breathing, perioral cyanosis and hypoxia not responding to oxygen therapy. Transfusion of packed red blood cells was given with improvement of symptoms. Laboratory results were significant for anemia and methemoglobinemia. Further tests revealed deficiency in glucose-6-phosphatase dehydrogenase enzyme. Discussion: G6PD deficiency can cause acute hemolytic anemia. Cyanosis can be exacerbated by the presence of methemoglobinemia. Management must include transfusion of packed red blood cells and avoidance of methylene blue to prevent further hemolysis. Conclusion: G6PD deficiency can manifest after exposure to oxidative stressors with acute onset hemolytic anemia and hypoxia. Occasionally methemoglobinemia could be present contributing to worsening symptoms.

27-Year-Old Woman With Fever, Headache, and Anemia

27-Year-Old Woman With Fever, Headache, and Anemia

Cerebral arterial gas embolism in a patient with hypoplastic left heart syndrome treated with emergent hyperbaric oxygen: case report

A 30-year-old female with a history of seizure disorder and hypoplastic left heart syndrome treated with a Norwood procedure in 1986 followed by a modified non-fenestrated Fontan (Left SVC to IVC to pulmonary arteries) with a known baffle leak presented to the emergency department. On day of presentation, the patient became unresponsive, with perioral cyanosis, rightward gaze and a left facial droop near the end of a platelet transfusion. An emergent non-contrast head CT revealed intracranial air in the right MCA distribution. She was taken to the hyperbaric chamber and was treated with a U.S. Navy Table 6 in a multiplace chamber with no extensions. Ten minutes into the treatment patient became more alert and spontaneously asked questions. The following day she was treated with a U.S. Navy Table 5. Patient had repeat CT of the head, which showed resolution of intracerebral gas and small areas of ischemia in right frontal lobe and right caudate. On hospital day five neurologic exam was normal, with 5/5 strength and no residual deficits. Treating the patient was a concern because patient has a single ventricle, in which the pulmonary artery is connected directly to the vena cava. There is very little data regarding the effects of hyperbaric oxygen (HBO2)therapy on single-ventricle physiology. Only two case reports of three pediatric patients treated with HBO2 for CAGE in a similar setting are known. In these cases the patients had improvements in their symptoms following HBO2. These cases and ours indicate HBO2 is feasible and indicated for CAGE in patients with cyanotic congenital heart disease.

Cerebral arterial gas embolism in a patient with hypoplastic left heart syndrome treated with emergent hyperbaric oxygen: case report

A 30-year-old female with a history of seizure disorder and hypoplastic left heart syndrome treated with a Norwood procedure in 1986 followed by a modified non-fenestrated Fontan (Left SVC to IVC to pulmonary arteries) with a known baffle leak presented to the emergency department. On day of presentation, the patient became unresponsive, with perioral cyanosis, rightward gaze and a left facial droop near the end of a platelet transfusion. An emergent non-contrast head CT revealed intracranial air in the right MCA distribution. She was taken to the hyperbaric chamber and was treated with a U.S. Navy Table 6 in a multiplace chamber with no extensions. Ten minutes into the treatment patient became more alert and spontaneously asked questions. The following day she was treated with a U.S. Navy Table 5. Patient had repeat CT of the head, which showed resolution of intracerebral gas and small areas of ischemia in right frontal lobe and right caudate. On hospital day five neurologic exam was normal, with 5/5 strength and no residual deficits. Treating the patient was a concern because patient has a single ventricle, in which the pulmonary artery is connected directly to the vena cava. There is very little data regarding the effects of hyperbaric oxygen (HBO2) therapy on single-ventricle physiology. Only two case reports of three pediatric patients treated with HBO2 for CAGE in a similar setting are known. In these cases the patients had improvements in their symptoms following HBO2. These cases and ours indicate HBO2 is feasible and indicated for CAGE in patients with cyanotic congenital heart disease.

КЛИНИКО-ЛАБОРАТОРНЫЕ ОСОБЕННОСТИ СИНДРОМА ЭНДОГЕННОЙ ИНТОКСИКАЦИИ У ДЕТЕЙ РАННЕГО ВОЗРАСТА С ПНЕВМОНИЕЙ

Objective: To study the clinical features of the course and the dynamics of some haematological indices in endogenous intoxication syndrome in children under 3 years of age suffering from pneumonia. Methods: There were 80 children under surveillance, of which 60 were pneumonia patients (main group), and 20 healthy children (control group). Laboratory survey included determining levels of leukocyte index of intoxication (LII), lactic and uric acids. The respiratory failure (RF) I is noted in 51.6%, RF II in 34.2% and RF III in 14.2% of cases. Results: Clinically and radiologically, 60.5% of children were diagnosed with bilateral processes in lungs, and in 39.5% the unilateral process. Signs of endogenous intoxication were: hyperhidrosis (59.4%), acrocyanosis (46.6%), perioral cyanosis (55.5%), mottled skin integument (49.2%), pronounced subcutaneous venous network (48.2%). The LII rate in the acute period had a strong tendency towards an increase of up to 5 days, with a gradual reduction to normal reference values by the end of the first week. It was found that high levels of lactic and uric acids were closely correlated with hypoxia. For example, in RF I, II and III levels of lactic acid were 1.3±0.1, 1.9±0.2 and 2.61±0.25 respectively. Children with RF III have degrees of lactic acid concentration that was statistically significant (p<0.001) higher than in the control group. Conclusions: Lactic and uric acid levels depend on the degree of RF and can indicate the severity of endogenous intoxication syndrome. The reduction of LII indices below 3.0 by the end of the first week of the disease, maybe a sign of a favorable outcome. Keywords: Pneumonia, respiratory failure, endogenous intoxication, leukocyte index of intoxication, lactic acid, uric acid.

ORAL MANIFESTATIONS AND CRANIOFACIAL CHARACTERISTICS OF HUTCHINSON-GILFORD PROGERIA SYNDROME: A CASE REPORT

Introduction: Hutchinson-Guilford progeria syndrome (HGPS) is a rare genetic disease with a characteristic phenotype of premature aging in young children caused by a mutation in the LMNA gene and consequent accumulation of progerin in the cell. Aim: Describe oral manifestations of Hutchinson-Guilford progeriasyndrome. Case Report: This is a case report of a six-year-old female patient with Hutchinson-Guilford Progeria syndrome. The physical examination revealed skin atrophy, lipodystrophy, hair rarefaction, prominent blood vessels of the scalp, craniofacial disproportion, perioral cyanosis and enlarged knee joints. The intraoral exam revealed limited mouth opening, mixed dentition with normal tooth anatomy and anteroinferior crowding. The eruption sequence and chronology were abnormal. The treatment plan included professional prophylaxis, the topical application of fluoride as well as both oral hygiene and dietarycounselling. Monitoring the development of dentition and an early and timely dental intervention contributed to the maintenance of child’s oral health. Conclusion: Early clinical and educational interventions can help patients with HGPS maintain adequate oral health status and improve their quality of life.

Infantile Pompe disease treatment with Myozyme in Chaharmahal and Bakhtiari: A case report

Pompe disease is a lysosomal storage disorder that results from an inborn error of metabolism which involves abnormal glycogen storage. Infantile-onset Pompe disease is the most severe phenotype that manifests with the findings of hypotonia, generalized muscle weakness, feeding difficulties, a failure to thrive, hearing loss, hypertrophic cardiomyopathy with systolic dysfunction, and respiratory distress. There is no effective treatment for Pompe disease and clinical control includes primary support health care. However, recent studies have reported that enzyme replacement therapy (ERT) can effectively decrease the symptoms or severity of the disease. The present study is a case report of a 24-day patient with infantile-onset Pompe disease who presented coughing and respiratory distress. The infant experienced fatigue during breastfeeding from the birth and developed dry and nonproductive coughs and perioral cyanosis from two weeks after the birth. The echocardiogram demonstrated left and right ventricular enlargement, mitral and tricuspid regurgitation, and pulmonary hypertension. After definitive diagnosis, the patient was administered with 20 mg/kg of alglucosidase 50 mg vial once every two weeks. He experienced significant improvement in body weight and echocardiogram and symptoms such as fatigue during breastfeeding and perioral cyanosis disappeared completely while he was under Myozyme treatment. In general, the observations confirmed the efficacy of Myozyme in a patient with infantile Pompe disease. Therefore, early diagnosis and quick treatment of Pompe disease with Myozyme can lead to acceptable outcomes, improve conditions, and finally, increase the chance of survival in these patients.

Generalized tonic seizures with autonomic signs are the hallmark of SCN8A developmental and epileptic encephalopathy

Developmental and epileptic encephalopathy (DEE) due to SCN8A gene variants is characterized by drug-resistant early onset epilepsy associated with severe intellectual disability. Different seizure types have been reported, and a sequence of autonomic manifestations such as brady-/tachycardia, irregular breathing, and cyanosis. Nevertheless, an exhaustive video-polygraphic documentation is still lacking. In this study, we reviewed the ictal electroencephalograms (EEGs) of five patients with SCN8A-DEE followed-up at the Neuroscience Department at Bambino Gesù Children's Hospital in Rome. We identified generalized tonic seizure as the major seizure type at epilepsy onset. Seizure severity could vary from subtle to marked clinical manifestations, depending from the extent and groups of muscles involved and association with autonomic modifications. We found autonomic signs in 80% of seizures in our cases, and we were able to identify a stereotyped sequence of ictal events for most of seizures. Autonomic signs occurred in rapid sequence: flushing of the face, sometimes associated with sialorrhea, bradycardia, and hypopnea appeared within the first 1–2 s. Tachycardia, polypnea, perioral cyanosis, and pallor occurred later in the course of the seizure.Generalized tonic seizures are rarely described in other genetic epileptic conditions of early infancy because of ion channel mutations, such as in DEE due to KCNQ2 or SCN2A gene mutations, where seizures are most frequently reported as focal to bilateral tonic. Therefore, generalized symmetric tonic seizures with autonomic signs can be considered a clinical hallmark for diagnosis of SCN8A-related DEE and relevant for therapeutic implications.

BREATHING CESSATION IN CHILD WITH ASTHMA AND INDUCIBLE LARYNGEAL OBSTRUCTION: WHEN TWO DIAGNOSES AREN'T ENOUGH

Introduction It is important to consider the differential of breathing cessation in a patient with established diagnoses, such as asthma and inducible laryngeal obstruction (ILO). Uncommon etiologies may masquerade as asthma or ILO exacerbations. Case Description A 5-year-old boy with history of asthma was referred for evaluation of recurrent respiratory episodes presumed to be from ILO. These episodes were characterized by complete absence of airflow, lasting 5-60 seconds with spontaneous resolution, occurring up to 20 times a day, every 3-4 days, during the day and at night, which would wake him up from sleep. Institution of pursed lip breathing was ineffective. Continuous laryngoscopy during exercise demonstrated moderate-to-severe ILO although characteristic field symptoms weren't reproduced. During 24-hour impedance probe study, he had several episodes of breathing cessation, perioral cyanosis, oxygen desaturation to the mid-60s and full-body limpness, resolving spontaneously after 45-60 seconds. Further evaluation included an electroencephalogram during an episode with no seizure activity, normal brain MRI, and normal rigid bronchoscopy. Impedance study demonstrated 100% correlation with reflux and breathing cessation, and he was diagnosed with reflux laryngospasm. A proton pump inhibitor (PPI) and promotility agent provided no relief. After Nissen fundoplication with post-surgical use of PPI and H2 blocker, there were no further respiratory events. Discussion In cases of apnea during wakefulness and sleep, it is important to consider etiologies such as seizure disorders, sleep disorders and reflux laryngospasm. Patients with gastroesophageal reflux can develop laryngospasm, a rare cause of abrupt absence of airflow caused by prolonged forceful apposition of the vocal cords.