Transvenous Phrenic Nerve Stimulation for Central Sleep Apnea.

RespiCardia remede System (Minnetonka, MN 2017), a transvenous phrenic nerve stimulator, is indicated to treat central sleep apnea (CSA) by stimulating the phrenic nerve to cause diaphragmatic contraction to restore normal breathing during sleep. CSA is associated with decreased patient quality of life and worsens cardiovascular outcomes. Systematic review was conducted according to the Preferred Reporting of Systematic Reviews and Meta-Analysis guidelines. PubMed/MEDLINE, Cochrane, EBSCO CINAHL, and Ovid databases were queried by 2 independent reviewers for English-language studies published between 2000 and 2021. The initial search screened for all occurrences of "remede" then was further refined to include studies evaluating use of the RespiCardia remede System as a treatment for CSA in multiple patients. A total of 124 articles were identified from the initial search results. A total of 37 articles were identified through screening of title and abstracts from initial results. Full-text review of all the articles was then completed. Of the 13 articles included, a total of 232 patients underwent device implantation. We sought to summarize the available evidence regarding patient selection for implantation, immediate and delayed complications, adherence to therapy, and polysomnographic evidence of efficacy. All 13 articles detailed significant decreases in central apnea index and many patients reported significant mild to marked improvement in quality of life. In conclusion, the remede System has been demonstrated to improve sleep and respiratory parameters, with few complications. This device demonstrates safe and effective treatment of moderate to severe CSA in adult patients, including those with heart failure.

Improving Nocturnal Hypoxemic Burden with Transvenous Phrenic Nerve Stimulation for the Treatment of Central Sleep Apnea.

Nocturnal hypoxemic burden is established as a robust prognostic metric of sleep-disordered breathing (SDB) to predict mortality and treating hypoxemic burden may improve prognosis. The aim of this study was to evaluate improvements in nocturnal hypoxemic burden using transvenous phrenic nerve stimulation (TPNS) to treat patients with central sleep apnea (CSA). The remede System Pivotal Trial population was examined for nocturnal hypoxemic burden. The minutes of sleep with oxygen saturation < 90% significantly improved in Treatment compared with control (p < .001), with the median improving from 33 min at baseline to 14 min at 6 months. Statistically significant improvements were also observed for average oxygen saturation and lowest oxygen saturation. Hypoxemic burden has been demonstrated to be more predictive for mortality than apnea-hypopnea index (AHI) and should be considered a key metric for therapies used to treat CSA. Transvenous phrenic nerve stimulation is capable of delivering meaningful improvements in nocturnal hypoxemic burden. There is increasing interest in endpoints other than apnea-hypopnea index in sleep-disordered breathing. Nocturnal hypoxemia burden may be more predictive for mortality than apnea-hypopnea index in patients with poor cardiac function. Transvenous phrenic nerve stimulation is capable of improving nocturnal hypoxemic burden. Graphical Abstract.

Images: Polysomnographic artifacts in a child with congenital central hypoventilation syndrome.

NONE: Diaphragm pacing (DP), a modality of ventilatory support in children with congenital central hypoventilation syndrome, generates respiration using the patient's own diaphragm as the respiratory pump. We report a 14-year-old boy with congenital central hypoventilation syndrome who uses DP with an uncapped tracheostomy during sleep. Polysomnography to titrate DP settings identified artifacts occurring in regular intervals coinciding with the onset of inspiration during all sleep stages in several channels including legs, snore, and electrocardiogram. Clinicians interpreting polysomnograms performed during DP should become familiar with the multichannel artifacts due to DP impulses. We also identified that our patient was hyperventilated on home DP settings that led to adjustment of DP settings during the polysomnogram to achieve optimal oxygenation and ventilation. Our case also highlights the utility of polysomnography to ensure optimal gas exchange during sleep in children with congenital central hypoventilation syndrome using DP.

Central Sleep Apnea in Patients with Heart Failure-How to Screen, How to Treat.

PURPOSE OF REVIEW: Central sleep apnea occurs in up to 50% of heart failure patients and worsens outcomes. Established therapies are limited by minimal supporting evidence, poor patient adherence, and potentially adverse cardiovascular effects. However, transvenous phrenic nerve stimulation, by contracting the diaphragm, restores normal breathing throughout sleep and has been shown to be safe and effective. This review discusses the mechanisms, screening, diagnosis, and therapeutic approaches to CSA in patients with HF. RECENT FINDINGS: In a prospective, multicenter randomized Pivotal Trial (NCT01816776) of transvenous phrenic nerve stimulation with the remede System, significantly more treated patients had a ≥ 50% reduction in apnea-hypopnea index compared with controls, with a 41 percentage point difference between group difference at 6 months (p < 0.0001). All hierarchically tested sleep, quality of life, and daytime sleepiness endpoints were significantly improved in treated patients. Freedom from serious related adverse events at 12 months was 91%. Benefits are sustained to 36 months. Transvenous phrenic nerve stimulation improves quality of life in patients with heart failure and central sleep apnea. Controlled trials evaluating the impact of this therapy on mortality/heart failure hospitalizations and "real world" experience are needed to confirm safety and effectiveness.

Análisis fenoménico de la técnica neuromuscular funcional como tratamiento para el SAOS / Phenomenical Analysis of the Functional Neuromuscular Technique as a Treatment for OSA

Introducción: el objetivo de este estudio es develar si la técnica Neuromuscular Funcional (NMF) puede ser considerada como opción de tratamiento en el Síndrome de Apnea Obstructiva de Sueño (SAOS) y si se complementa con la Terapia Miofun-cional Orofacial o hace parte de ella.Métodos: la fenomenología trascendental de Husserl es el enfoque metodoló-gico que se adoptó para este estudio, siguiendo la ruta para su desarrollo: epojé, reducción trascendental fenomenológica y síntesis. El contenido real se obtuvo mediante dos entrevistas semiestructuradas, aplicadas a un informante clave que contó con los criterios de inclusión presupuestados. Resultados: en la primera etapa se identificaron 10 noesis y 266 noemas, que fue-ron relacionados entre sí; en la fase de reducción trascendental las noesis se redujeron a 5 y los noemas a 14. Análisis y discusión: el Método Chiavaro como fenómeno se decanta en las di-mensiones: (a) Enfoque Sistémico, (b) Técnica Neuromuscular Funcional y (c) Téc-nica Respiratoria. La prioridad del fenómeno NMF es la explicación de la alteración dentro del enfoque sistémico a partir de la lógica Función-Estructura-Función, de esta manera el SAOS se considera una manifestación de esas relaciones y como tal se le cataloga como adaptación o como función en disfunción.Conclusiones: la Técnica NMF hace parte de la neurorrehabilitación mientras que la TMO para intervenir el SAOS se inscribe en la fisiología del ejercicio

Introduction: the objective of this study is to reveal if the Functional Neuromuscu-lar technique (In Spanish: NMF) can be considered as a treatment option for OSA. Additionally, this paper seeks to assess if the aforementioned is derived from, a por-tion of or if it is complemented by Orofacial Myofunctional Therapy (OMT). Methods: Husserl's transcendental phenomenology is the methodological approach that was adopted for this study, following the path to its development: epoché, phenomenologi-cal transcendental reduction and synthesis. The actual content was obtained through two semi-structured interviews applied to a key informant who met the preconceived inclusion criteria.Results: in the first stage 10 noesis and 266 noems , which were related to each other, were identified; in the phase of transcendental reduction, the noesis were reduced to 5 and the noems to 14. Analysis and discussion: the Chiavaro Method as a phenomenon is composed of three dimensions: (a) Systemic Approach, (b) Functional Neuromuscular Technique and (c) Respi-ratory Technique. The priority of the NMF phenomenon is the explanation of the alteration within the systemic approach based on the Function-Structure-Function logic, ergo the OSA is considered a manifestation of these relationships and as such it is classified as adaptation or as a function in dysfunction.Conclusions: the NMF technique is a part of neurorehabilitation whilst OMT for the in-tervention of OSA is subscribed to the physiology of exercise.

How to implant a phrenic nerve stimulator for treatment of central sleep apnea?

BACKGROUND: Central sleep apnea (CSA) is a breathing disorder caused by the intermittent absence of central respiratory drive. Transvenous phrenic nerve stimulation is a new therapeutic option, recently approved by the FDA , for the treatment of CSA. OBJECTIVE: To describe the technique used to implant the transvenous phrenic nerve stimulation system (the remede System, Respicardia, Inc). METHODS: The remede System is placed in the pectoral region, typically on the right side. A single stimulation lead is placed in either the left pericardiophrenic vein (PPV) or the right brachiocephalic vein (RBC). A sensing lead is placed into the azygous vein to detect respiration. RESULTS: In the remede System Pivotal trial, 147 of 151 (97%) patients were successfully implanted with the system. Sixty-two percent of stimulation leads were placed in the PPV and 35% in the RBC. Mean procedure time was 2.7 ± 0.8 hours and 94% of patients were free from implant-related serious adverse events through 6 months. CONCLUSION: In patients with CSA, transvenous phrenic nerve stimulation is an effective and safe therapy with an implant procedure similar to that of cardiac implantable electronic devices.

Cyclic alternating pattern in infants with congenital hypothyroidism.

Congenital hypothyroidism is defined as thyroid hormone deficiency present at birth which is crucial for brain development. Recently, the cyclic alternating pattern, a rhythm present in electroencephalography recordings in non-Rapid eye movement sleep, has been related to brain development and cognition in different pediatric conditions. Therefore, we evaluated the cyclic alternating pattern rate in infants with congenital hypothyroidism, thyroxine supplementation, and healthy controls. The parameters of the cyclic alternating pattern were evaluated in 19 healthy infants (10 female, mean age 25.5 ±â€¯15.5 months) and 21 infants diagnosed with congenital hypothyroidism (19 female, mean age 24.3 ±â€¯19.0 months). We considered the transient electro-cortical activations (phase A of the cycle) in non-Rapid eye movement sleep and the subdivisions of the A phase in: A1, A2 and A3, based on their frequency content. All subjects were subjected to polysomnography recording in a standard laboratory setting. Sleep data were stored computer following the International 10-20 System. Data showed that congenital hypothyroidism infants exhibited higher frequency of central apnea, hypopnea, and arousals in comparison to controls. Particularly, central apnea index decreased with age in the control group but not in congenital hypothyroidism group. Regarding to cyclic alternating pattern measurements, congenital hypothyroidism infants exhibit a higher frequency in the percentage of A3 subtype (electroencephalographic desynchrony) and conversely a lower percentage of A1 subtype (electroencephalographic synchrony), than healthy infants. An important finding of this study is the positive correlation between A1 mean duration and age, which is bigger in control group than in congenital hypothyroidism group (time duration in control group (0.52 s/month) versus congenital hypothyroidism group (0.1 s/month). Infants with congenital hypothyroidism showed an increase of A3 subtype, of central apnea, and of arousals. The reduction of percentage and mean duration of A1 subtype could be a valuable indicator of sleep development in patients with congenital hypothyroidism and healthy infants.

Unusual case of central alveolar hypoventilation.

Central alveolar hypoventilation is rarely encountered. This case report describes a young woman who was recently diagnosed with hypertension and ischemic heart disease. She presented to the emergency room with hypercapnic respiratory failure, for which she was mechanically ventilated. This was preceded by an acute upper respiratory tract infection. She was initially suspected to have Guillain-Barré syndrome, but further investigations ruled out neuromuscular or autoimmune disorders. Sleep-related hypoventilation was suspected after she experienced recurrent apneas at night that resulted in re-intubation. Polysomnographic studies confirmed episodes of central apnea and hypopnea during sleep, with significant carbon dioxide retention and oxygen desaturations. She required nocturnal ventilation via a tracheostomy tube until a diaphragmatic pacer could be placed. Using bi-level positive airway pressure and average volume-assured pressure support together with the diaphragmatic pacer, adequate ventilation during sleep was achieved.

Long-Term Experience with First-Generation Implantable Neurostimulation Device in Central Sleep Apnea Treatment.

BACKGROUND: Sleep-disordered breathing (SDB) and Cheyne-Stokes respiration (CSR) are associated with shorter survival in patients with heart failure. A novel treatment method for this patient group is unilateral phrenic nerve stimulation by the remede® system (Respicardia Inc., Minnetonka, MN, USA), a transvenously implantable neurostimulation device, which has recently been studied in a large randomized, controlled trial. Previous literature has shown efficacy and safety of the treatment with this first-generation device, but hardly any data are available on long-term clinical parameters, the remede® device's battery lifetime, device exchangeability, lead position stability, surgical accessibility, and manageability. METHODS: We performed remede® device replacements in consecutive patients for battery depletion, and documented clinical parameters, longevity, operation procedure, complications, and difficulties. RESULTS: All patients were on neurostimulation treatment by phrenic nerve neurostimulation when device replacement became necessary. Apnea-hypopnea index (from 45 ± 4/h to 9 ± 4/h), oxygen-desaturation index (from 35 ± 7/h to 7 ± 6/h), and time spent with oxygen saturation of <90% (T < 90% from 5 ± 7% to 0 ± 0%) were improved and improvements remained constant throughout the 4-year follow-up. Mean battery life was 4.2 ± 0.2 years and mean replacement procedure time was 25 ± 5.1 minutes. Apart from conventional X-ray documentation of stable lead positions in a long-term setting, no radiation or contrast dye usage was needed and no major complications occurred. In addition, clinical exercise capacity and sleepiness symptoms improved. CONCLUSIONS: Novel remede® device shows sustained therapy efficacy and safety in terms of stable lead positions over 4 years. Long-term phrenic nerve neurostimulation therapy for central SDB/CSR appears feasible in a clinical routine setting.

Mapping for Acute Transvenous Phrenic Nerve Stimulation Study (MAPS Study).

BACKGROUND: Central sleep apnea syndrome, correlated with the occurrence of heart failure, is characterized by periods of insufficient ventilation during sleep. This acute study in 15 patients aims to map the venous system and determine if diaphragmatic movement can be achieved by phrenic nerve stimulation at various locations within the venous system. METHODS: Subjects underwent a scheduled catheter ablation procedure. During the procedural waiting time, one multielectrode electrophysiology catheter was subsequently placed at the superior and inferior vena cava and the junctions of the left jugular and left brachiocephalic vein and right jugular and right brachiocephalic vein, for phrenic nerve stimulation (1-2 seconds ON/2-3 seconds OFF, 40 Hz, pulse width 210 µs). Diaphragmatic movement was assessed manually and by a breathing mask. During a follow-up assessment between 2 and 4 weeks postprocedure, occurrence of adverse events was assessed. RESULTS: In all patients diaphragmatic movement was induced at one or more locations using a median threshold of at least 2 V and maximally 7.5 V (i.e., e 3.3 mA, 14.2 mA). The lowest median current to obtain diaphragmatic stimulation without discomfort was found for the right brachiocephalic vein (4.7 mA). In 12/15 patients diaphragmatic movement could be induced without any discomfort, but in three patients hiccups occurred. CONCLUSION: Diaphragmatic stimulation from the brachiocephalic and caval veins is feasible. Potential side effects should be eliminated by adapting the stimulation pattern. This information could be used to design a catheter, combining cardiac pacing with enhancing diaphragm movement during a sleep apnea episode.

Design of the remede System Pivotal Trial: A Prospective, Randomized Study in the Use of Respiratory Rhythm Management to Treat Central Sleep Apnea.

BACKGROUND: Central sleep apnea is common in patients with cardiovascular disease and worsens outcomes. There is a lack of established therapies for central sleep apnea, and those available are limited by poor patient adherence and potentially adverse cardiovascular effects, at least in a subset of patients. The remede System (Respicardia, Minnetonka, Minnesota) is a new physiologic treatment that uses transvenous phrenic nerve stimulation to contract the diaphragm, thereby stabilizing gas exchange and restoring normal breathing throughout the sleep period. METHODS: This is a prospective multicenter randomized trial with blinded end points evaluating the safety and efficacy of the remede System. Up to 173 patients with central sleep apnea will be randomized 1:1 to remede System therapy initiated at 1 month after implantation (treatment) or to an implanted remede System that will remain inactive for 6 months (control). Primary efficacy end point is the percentage of patients who experience a reduction in apnea-hypopnea index by a ≥ 50% at 6 months (responder analysis). Primary safety end point is freedom from serious adverse events through 12 months. Secondary end points include sleep-disordered breathing parameters, sleep architecture, Epworth Sleepiness Scale score, and Patient Global Assessment. CONCLUSIONS: This is the 1st randomized controlled trial of the safety and efficacy of the remede System for the treatment of central sleep apnea.

Airway obstruction in congenital central hypoventilation syndrome.

Congenital central hypoventilation syndrome (CCHS) is the failure of the autonomic system to control adequate ventilation while asleep with preserved ventilatory response while awake. We report a case of a patient with CCHS who presented with intrathoracic and extrathoracic airway obstruction after tracheostomy tube decannulation and phrenic nerve pacer placement. Nocturnal polysomnography (NPSG) revealed hypoxia, hypercapnia and obstructive sleep apnoea, which required bilevel positive airway pressure titration. Airway endoscopy demonstrated tracheomalacia and paretic true vocal cords in the paramedian position during diaphragmatic pacing. Laryngeal electromyography demonstrated muscular electrical impulses that correlated with diaphragmatic pacer settings. Thus, we surmise that the patient's upper and lower airway obstruction was secondary to diaphragmatic pacer activity. Thorough airway evaluation, including NPSG and endoscopy, may help identify the side effects of diaphragmatic pacing, such as airway obstruction, in patients with CCHS.

A novel therapeutic approach for the treatment of central sleep apnea: The remede® system.

Central sleep apnea (CSA) occurs primarily in cardiovascular patients and is associated with high morbidity and mortality. The disorder often is unrecognized due to the overlap of symptoms with those of the underlying cardiac disease. CSA can be easily diagnosed with a sleep study. Following optimization of all co-morbidities, the therapeutic approach available currently focuses on mask-based therapies which suffer from poor patient adherence. A new therapy, the remede® System, has been developed; it utilizes a transvenous, fully implantable system providing phrenic nerve stimulation intended to restore a more normal breathing pattern. The therapy demonstrated promising results based on an initial chronic study and a randomized trial is underway to further evaluate safety and efficacy of this novel system in patients with CSA.

Reversal of pulmonary hypertension after diaphragm pacing in an adult patient with congenital central hypoventilation syndrome.

INTRODUCTION: Patients with the congenital central hypoventilation syndrome (CCHS) suffer from life-threatening hypoventilation when asleep, making them dependent on mechanical ventilation (MV) at night or during naps. State-of-art respiratory management consists of intermittent positive-pressure ventilation via a tracheotomy or mask. In some patients hypoventilation is permanent, in which case ventilatory support must be extended to the waking hours. Diaphragm pacing can prove useful in such situations. 
 METHODS AND RESULTS: This report describes the case of a 26-year-old woman with CCHS in whom failure to achieve adequate MV led to life-threatening pulmonary hypertension (PH), with a systolic pulmonary artery pressure (PAP) of 80 mmHg and right ventricular hypertrophy, despite optimization of all possible measures and despite extensive therapeutic education efforts. Diaphragm pacing using laparoscopically implanted intradiaphragmatic phrenic nerve stimulation electrodes corrected alveolar hypoventilation and lastingly reversed PH (systolic PAP below 40 mmHg after 2 months, sustained after 2 years). Diaphragm pacing induced shoulder pain, however, involving the chronic use of analgesics. The pacing had to be stopped for tolerance reasons after two years, leading to PH worsening and the need for diurnal MV. 
 CONCLUSIONS: Diaphragm pacing appears likely effective to restore alveolar ventilation and reverse PH in adult CCHS patients.

Threshold currents of platinum electrodes used for functional electrical stimulation of the phrenic nerves for treatment of central apnea.

BACKGROUND: Stability of threshold currents during long-term use of phrenic nerve stimulation has been questioned. METHODS AND RESULTS: Between January 5, 1988, and March 5, 2008, 49 patients with functional C2-tetraplegia received an Atrostim PNS (Atrotech Ltd., Tampere, Finland) as treatment of their respiratory insufficiency; a follow-up of 35 of such patients was carried out exclusively in our institution for 6.3 (4.44) 0.04-15.75 years (mean [standard deviation (SD)] range). The device employed four-pole sequential nerve stimulation, which provided four threshold currents subsequently evaluated for each phrenic nerve. Stimulation data were prospectively recorded. The differences between threshold currents recorded 1 year after implantation and the last recorded values were 0.33-0.43 (0.44-0.63) 0-2.9 mA. After having excluded the data of eight patients with values >1 mA (= mean + SD), we registered the differences for the remaining patients of 0.15-0.24 (0.14-0.24) 0-0.95 mA, which is just twice the adjustment accuracy of the device. Out of the eight problem cases one had, and two were suspected to have, surgical trauma; all three nerves recovered. In two cases the values steadily increasing over years might have been caused by unspecific foreign body reaction. Two cases with values >1 mA for different durations at different electrodes might be caused by biofilm, and one patient displaying steadily increasing values lived, unwilling to live, only 2 years after the implantation. CONCLUSION: Thus, there was no permanent nerve injury and in 77% of the presented cases threshold currents remained stable.

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Ondine's curse with accompanying trigeminal and glossopharyngeal neuralgia secondary to medullary telangiectasia.

BACKGROUND: Central hypoventilation syndrome ("Ondine's Curse") is an infrequent disorder that can lead to serious acute or chronic health consequences. This syndrome, especially in adults, is rare, and even less frequent in the absence of clear pathogenic lesions on MRI. In addition, we are not aware of any previously reported cases with associated cranial nerve neuralgias. METHODS: We describe a patient with baseline trigeminal and glossopharyngeal neuralgia, admitted with episodes of severe hypoventilatory failure of central origin, consistent with "Ondine's Curse". After evaluation, she was found to have a medullary capillary telangiectasia, thought to be the causative lesion, and which could explain her complete neurologic and hypoventilatory syndrome. The patient was treated with placement of a diaphragmatic pacing system, which has been effective thus far. RESULTS: This case illustrates the need for investigation of centrally mediated apnea, especially when co-occurring cranial nerve neuralgia is present and cardiopulmonary evaluation is negative. It provides an example of capillary telangiectasia as the causative lesion, one that to our knowledge has not been reported before. CONCLUSIONS: Placement of a diaphragmatic pacing system was warranted and became lifesaving as the patient was deemed to be severely incapacitated by chronic ventilatory insufficiency.

Era uma vez Ondina: relato de caso / Once upon a time Ondina...case report

Objetivo: relatar o caso de uma criança recém-nascida (RN) com comprometimento do sistema nervoso autônomo caracterizado por insensibilidade em detectar aumento de CO2 e diminuição de O2, denominado de Maldição de Ondina ou Síndrome da Hipoventilação Central Congênita ou Hipoventilação Alveolar Primária, com ênfase ao acompanhamento fonoaudiológico. Discussão: o diagnóstico de Maldição de Ondina deve ser aventado quando da presença de episódios de apneias em RN, sem que se consiga elucidar a causa e que seja afastada a possibilidade de crise epiléptica, distúrbio pulmonar e/ou cardíaco, lesão de tronco ou de outra região encefálica. A evolução respiratória desta criança foi de dependência do respirador por 24 horas até o final do terceiro mês de vida. Progressivamente, foi possível deixá-la com cateter nasal com fluxo contínuo de oxigênio enquanto se mantinha acordada, e respirador em modo controlado nos períodos de sono. O tratamento específico, com instalação de marca-passo diafragmático, é a única terapia existente até o momento.

Purpose: To report the case of a newborn (NB) with an autonomic nervous system disorder characterized by insensitivity to detect increased CO2 and decreased O2, referred to as Ondine's curse, Primary Hypoventilation Syndrome, or Congenital Central Alveolar Hypoventilation, with emphasis on speech therapy. Discussion: The diagnosis of Ondine's curse should be considered upon presence of episodes of apnea in newborns, with no clear indication of the cause and with no indications of seizures, severe pulmonary or cardiac injury of the brainstem or another cerebral region. The respiratory development characteristic of this child was being ventilator dependent for 24 hours a day until the end of the third month of life. Gradually, it was possible to leave the device with a continuous nasal oxygen flow while awake, and breathing with a ventilator under control during her periods of sleep. Specific treatment with installation of diaphragmatic pacemaker, is the only treatment available to date.

Objetivo: relatar el caso de un recién nacido (RN) con comprometimiento del sistema nervioso autónomo caracterizado por falta de sensibilidad para detectar el aumento de la emisión de CO2 y la disminución de O2, conocido como La Maldición de Ondina, o Síndrome de Hipoventilación Central Congénita o Hipoventilación Alveolar Primaria, con énfasis al acompañamiento fonoaudiológico. Discusión: El diagnóstico de la Maldición de Ondine debe ser considerado en la presencia de episodios de apnea en recién nacidos, sin que se pueda aclarar la causa y que sea rechazada la posibilidad de crisis epiléptica, trastorno pulmonar y/o cardiaco severo, lesión de tronco o otra región encefálica. El desarrollo respiratorio de este niño fue de dependencia del respirador por 24 horas hasta el final del tercer mes de vida. Progresivamente fue posible dejarla con un flujo nasal continuo de oxígeno mientras se mantenía despierta, y con respirador en forma controlada durante los períodos de sueño. El tratamiento específico con la instalación de un marcapasos diafragmático, es el único tratamiento disponible hasta la fecha.

Induction of central-type sleep apnea by vagus nerve stimulation.

Vagus nerve stimulation (VNS) is an acceptable and effective adjunctive therapy for pharmacoresistant epilepsy. It is generally well tolerated and the most frequent side effects reported include respiratory dysfunction. We report the case of a female patient with intractable epilepsy who was implanted with the device and achieved a significant reduction in the number of her seizures. However, she developed central-type sleep apnea documented polysomnographically. Upon reduction of her VNS parameters, the apnea resolved and her sleep study reverted to normal. To our knowledge, this is the first case reported with polysomnographic evidence of VNS induction of central-type sleep apnea.