Search

STUDY OBJECTIVES: In multiple system atrophy, sleep-disordered breathing has a broad spectrum of phenotypes, among them inspiratory stridor and obstructive sleep apnea being most frequent. METHODS: We present a case of a 59-year-old woman with cerebellar-type multiple system atrophy, who showed transient paradoxical breathing effort during non–rapid eye movement sleep on diagnostic polysomnography. Because this thoraco-abdominal paradox was atypical for and did not coincide with upper airway obstruction, it most likely indicated central dysregulation of the diaphragm. RESULTS: Continuous positive airway pressure therapy with low pressure (5 cm H(2)O) was sufficient to completely resolve this type of respiratory dysregulation. CONCLUSIONS: This case extends the clinical spectrum of sleep-disordered breathing in multiple system atrophy. CITATION: Ha J, Park J, Youn J, Cho JW, Joo EY. Non-REM sleep–predominant reversible paradoxical breathing effort indicates dysregulation of diaphragm movements in multiple system atrophy. J Clin Sleep Med. 2021;17(7):1499–1502.

An asymptomatic 60-year-old woman presented to our outpatient clinic for a routine health screening as part of a government welfare program. Her medical history was unremarkable, however physical examination revealed hypoplasia of the left chest wall muscles, breast tissue, nipple, absence of axillary hair, absence of the costal cartilages of upper ribs on the left side of the chest and brachydactyly in the left hand. There was also paradoxical breathing at the levels of the 2nd, 3rdand 4thrib of the left chest. The rest of the examination was unremarkable. The chest radiograph revealed reduced upper left hemithorax volume with partial rib agenesis of the 2nd, 3rdand 4thrib, bilateral cervical ribs, dextroposition of the heart and mid thoracic scoliosis with convexity to the left. A 12-lead electrocardiogram revealed a sinus rhythm with a normal axis, lead 1 has upright P, QRS, and T waves and aVR is globally negative. The precordial leads show normal R Waves in V1-4, deep S waves in V1-2, small S waves in V3-4 and no S waves in V5-6. There is diminution of the precordial complexes in V3, 4, 5 and 6. These findings are consistent with a dextroposition of the heart. The blood investigations were unremarkable. The findings from physical examination, chest radiograph and 12 lead electrocardiogram are consistent with Poland´s Syndrome. As the patient was otherwise well and not bothered by her condition, no treatment was initiated for her.

Diaphragmatic dysfunction has been reported in congenital myopathies, muscular dystrophies, and occasionally, mitochondrial respiratory chain deficiency. Using a minimally invasive procedure in 3 young girls, 1 with a heteroplasmic MT-CYB mutation and 2 with biallelic pathogenic TK2 variants, we provided functional evidence of diaphragmatic dysfunction with global respiratory muscle weakness in mitochondrial respiratory chain deficiency. Analysis of respiratory muscle performance using esogastric pressures revealed paradoxical breathing and severe global inspiratory and expiratory muscle weakness with a sniff esophageal inspiratory pressure and a gastric pressure during cough averaging 50% and 40% of predicted values, respectively. This diaphragmatic dysfunction was responsible for severe undiagnosed nocturnal hypoventilation, requiring noninvasive ventilation. Our results underline the interest of this minimally invasive procedure for the evaluation of respiratory muscle performance and its potential value for the monitoring of future clinical trials in respiratory chain deficiency.

The involvement of the respiratory muscular pump makes SMA children prone to frequent hospitalization and morbidity, particularly in type 1. Progressive weakness affects ribcage muscles resulting in bell-shaped chest that was never quantified. The aims of the present work were: (1) to quantify the presence of bell-shaped chest in SMA infants and children and to correlate it with the action of ribcage muscles, assessed by the contribution of pulmonary ribcage to tidal volume (ΔVRC, p); (2) to verify if and how the structure of the ribcage and ΔVRC, p change after 1-year in SMA type 2. 91 SMA children were studied in supine position during awake spontaneous breathing: 32 with type 1 (SMA1, median age: 0.8 years), 51 with type 2 (SMA2, 3.7 years), 8 with type 3 (SMA3, 5.4 years) and 20 healthy children (HC, 5.2 years). 14 SMA2 showed negative ΔVRC, p (SMA2px), index of paradoxical inspiratory inward motion. The bell-shaped chest index was defined as the ratio between the distance of the two anterior axillary lines at sternal angle and the distance between the right and left 10th costal cartilage. If this index was < < 1, it indicated bell shape, if ~1 it indicated rectangular shape, while if >> 1 an inverted triangle shape was identified. While the bell-shaped index was similar between HC (0.92) and SMA3 (0.91), it was significantly (p < 0.05) reduced in SMA2 (0.81), SMA2px (0.74) and SMA1 (0.73), being similar between the last two. There was a good correlation (Spearman's rank correlation coefficient, ρ = 0.635, p < 0.001) between ribcage geometry and ΔVRC, p. After 1 year, ΔVRC, p reduced while bell-shaped chest index did not change being significantly lower than HC. The shape of the ribcage was quantified and correlated with the action of ribcage muscles in SMA children. The impaired ribcage muscles function alters the ribcage structure. HC and SMA3 show an almost rectangular ribcage shape, whereas SMA2, SMA2px and SMA1 are characterized by bell-shaped chest. In SMA, therefore, a vicious cycle starts since infancy: the disease progressively affects ribcage muscles resulting in reduced expansion of lung and ribcage that ultimately alters ribcage shape. This puts the respiratory muscles at mechanical disadvantage.

The aetiology of phrenic neuropathy is often unknown, but immune mechanisms may play a role. In a typical case of bilateral phrenic neuropathy with paradoxical breathing (video), an inflammatory pathogenesis was suggested by prolonged distal latency of phrenic nerve compound muscle action potentials in nerve conduction studies and a clear-cut albumin-cytologic dissociation. This encouraged us to treat the patient with a standard dose of intravenous immunoglobulin. After obtaining a strong improvement at spirometry, we repeated the second cycle of intravenous immunoglobulin and observed normalization of symptoms within few weeks and no relapse after 3 years. This case suggests that lumbar puncture should be performed in the acute phase of phrenic neuropathies to detect potential responders to immunomodulatory treatment.

Aim To present a new therapy for Poland syndrome (PS) using a novel surgical approach: the vertical expandable prosthetic titanium rib (VEPTR) system. Methods The VEPTR system rib-to-rib variant was used to enhance the chest wall and vertebral column support in a young patient before walking age. Case Report We present a 12-month-old infant diagnosed with left-sided PS at the age of 6 months associated with missing ribs, scoliosis, and absence of the left pectoral muscles. Because of four missing ribs, paradoxical breathing was present. In addition, the left scapula was protruding into the chest due to the missing rib support. Scoliosis was caused by a left-sided nonsegmented bar of the thoracic spine. Results We decided to use the VEPTR system before the patient reached walking age to prevent progression of column deformation and future pulmonary problems. To improve the spinal deformity, to stabilize the thorax, and to improve thoracic function, we performed the operation at 1 year of age. At 10-month follow-up, the patient was reevaluated. The construction was still stable and scoliosis had not deteriorated. Conclusion The VEPTR system is a choice of treatment in young patients with PS to prevent late complications after a child reaches walking age.

Resumen La parálisis diafragmática bilateral es infrecuente y puede ser idiopática o más comúnmente asociada a varias entidades. Describimos el caso de un varón de 61 años con antecedentes de asma bronquial quien desarrolló parálisis difragmática bilateral sin causa evidente, la cual es excepcional. El paciente presentó disnea progresiva y ortopnea, que no mejoraban con antibioterapia ni esteroides sistémicos, por lo que se descartaron diversas etiologías como neuralgia lateral amiotrófica y finalmente se le diagnosticó parálisis diafragmática lateral en base a la exploración física y las pruebas de imagen. Se inició ventilación mecánica no invasiva con gran mejoría clínica. Bilateral paralysis of the diaphragm is uncommon and can be either idiopathic or more frequently associated with several medical conditions. We describe the case of a 61-year-old man with a history of asthma who developed severe bilateral diaphragmatic paralysis without any obvius cause, which is exceptional. The patient manifested progressive dyspnea and debilitating orthopnea with no improvement with antibiotics or systemic steroids. Different etiologies were discarded as amyotrophic lateral neuralgia, and he was diagnosed of bilateral diaphragmatic paralysis based on physical examination and imaging tests. Noninvasive mechanical ventilation was started with great clinical improvement.

Aim: The aim of this study was to assess and compare the validity and potential utility of two manual breathing assessment procedures: the Manual Assessment of Respiratory Motion (MARM) and the Hi Lo Breathing Assessment. A secondary aim was to gauge the relationships between experience and the ability to perform these assessment techniques, by comparing the performance of students with practitioners. Method: 56 osteopaths and osteopathic students were taught the MARM and the Hi Lo Breathing Assessment and trained to simulate breathing patterns. The participants, acting alternatively as breathers and examiners, then attempted to accurately determine whether the breathing patterns simulated by their partner were predominately abdominal, thoracic or, in the case of the Hi Lo, paradoxical. Participants were surveyed on their confidence in the use of each technique, their perceived ease in using each technique, and their intended future use of the techniques. Student and practitioner abilities to detect simulated breathing patterns were compared for the MARM and Hi Lo. Results: Overall scores for correctly determining breathing patterns were not significantly different for the MARM or the Hi Lo, and there was no notable moderation of this effect according to experience, with both practitioners and students demonstrating a high level of performance on both techniques. There were some differences in accuracy of performance across different breathing styles, with Hi Lo assessment of paradoxical breathing being more difficult to identify correctly. Ease of learning was similar for MARM and Hi Lo but confidence in using the techniques, and intended future use was higher for the MARM. There were some significant relationships between these utility measures and performance, particularly on the MARM. Conclusions: This study builds on our previous study to strengthen the evidence for the validity of the MARM and also supports the validity of the Hi Lo. Responses to the survey indicate that, overall, participants preferred the MARM to the Hi Lo. This study is a preliminary investigation of these techniques. Future studies to test the validity of these techniques should be performed in a clinical setting on individuals with actual rather than simulated breathing pattern disturbances. 2008 Published by Elsevier Ltd.

Approximately, 21,000 breaths per day and 10.5 million breaths per year are taken by average people. Breathing functions depend on the actions of the respiratory muscles on the chest wall. This study analyzes the motions of the humans’ chest compartments with a portable, low-cost and convenient monitoring system to be used in e-health applications. Two wearable MEMS sensors are used to capture the rib cage (RC) and abdomen (AB) movements, simultaneously. In addition to measuring the phase shift between the chest wall compartments, the impacts of different body positions on AB and RC motions have been investigated on four different respiration patterns with ten subjects. The performance of the presented system is evaluated and the average mean square error of 0.13 is achieved for three time parameters including inspiratory, expiratory, and total time of the respiratory cycle. Moreover, the overall errors of phase shifts for paradoxical and synchronous breath patterns are 0.13° ± 0.12° and 0.21° ± 0.08°, respectively, considering two vernier respiration monitor belts as our references. The system properly indicates a significant increase in the degree of RC and AB asynchrony in the paradoxical breathing compared to normal pattern. To complete our platform, we propose a new “Breathing Therapy” technique as an advanced way to assist people to learn the science of breath and help the patients successfully restore their health in a systematic way. Graphical signs are used to provide feedback on the quality of each breath taken in real-time. The performance of the system is evaluated based on five well-known yogic breathing patterns.

To demonstrate the elimination of pectus excavatum and promotion of more normal lung growth and chest wall development by the use of high-span positive inspiratory pressure plus positive end-expiratory pressure (PIP+PEEP), patients with spinal muscular atrophy type 1 with paradoxical breathing were placed on high-span PIP+PEEP when sleeping from the point of diagnosis of spinal muscular atrophy. Although the appearance of pectus excavatum is ubiquitous in untreated infants with spinal muscular atrophy type 1, after institution of high-span PIP+PEEP, pectus resolves and lungs and chest walls grow more normally. High-span PIP+PEEP is indicated for all infants diagnosed with spinal muscular atrophy who demonstrate paradoxical breathing for the purpose of promoting more normal lung and chest development.

Automated methods of real-time and un-obstructive patient's respiration and position monitoring have been subjects of interests in e-health applications. The present study implements a low-cost and convenient monitoring system for patients with breathing problems or sleep disorders. We have also captured the Rib Cage (RC) and Abdomen (AB) movements using accelerometer sensors. In addition to measurement of phase shift between the chest wall compartments, the impacts of different body positions on AB and RC motions have been investigated. The performance of the presented system is evaluated and the average Mean Square Error (MSE) of 0.14 is achieved for three breath timing variables. Moreover, the overall errors of phase angles for paradoxical and synchronous breath patterns are 0.25°±0.06 and 0.26°±6E-03, respectively. The system properly indicates a significant increase in the degree of ribcage and abdomen asynchrony in the paradoxical breathing compared to normal pattern.

Objective—To determine the strength of the relationship between paradoxical breathing (PB) and spontaneous pleural diseases in dyspneic dogs and cats. Design—Cross-sectional study. Animals—Dogs (n = 195) and cats (194) with a recorded diagnosis of dyspnea examined at the National Veterinary Schools of Alfort and Toulouse (France) between January 2001 and October 2009. Procedures—Dogs and cats were divided into 2 groups according to the presence or absence of PB. Stratified analysis by species was performed. Signalment of affected animals and occurrence of PB were recorded. The relationship between PB and pleural diseases among dyspneic dogs and cats was analyzed. Results—A strong relationship between PB and pleural diseases was highlighted in multivariate analysis (dogs, OR = 12.6 and 95% confidence interval = 4.6 to 31.2; cats, OR = 14.1 and 95% confidence interval = 6.0 to 33.5). Paradoxical breathing prevalence among dyspneic dogs and cats was 27% and 64%, respectively. Occurrence of pleural diseases in dyspneic animals with and without PB was 49% and 9% in dogs and 66% and 13% in cats, respectively. The sensitivity and specificity of PB as a predictor of pleural diseases were 0.67 and 0.83 in dyspneic dogs and 0.90 and 0.58 in dyspneic cats, respectively. The positive and negative predictive values of PB were 0.49 and 0.91 in dyspneic dogs and 0.66 and 0.87 in dyspneic cats, respectively. Age, sex, feline breeds, and canine morphotypes in patients with PB were not significantly different from those of other dyspneic animals. Conclusions and Clinical Relevance—PB was strongly associated with pleural diseases in dyspneic dogs and cats. The presence of this clinical sign should prompt small animal practitioners to implement appropriate emergency procedures and guide their diagnostic strategy.

OBJECTIVE This work describes and correlates plateau/maximum observed vital capacity (VC) with spinal muscular atrophy (SMA) severity and prognosis for autonomous breathing. DESIGN SMA severity was correlated with VC, onset, paradoxical breathing, age at definitive dependence on continuous mechanical ventilation (DDCV), and age at first respiratory hospitalization and gastrostomy. RESULTS Ten severe SMA 1A patients with DDCV before 6 mos of age had maximum observed and plateau VC of 100 ml or less, with plateaus for six at 10.3 (range, 3-48) mos. Another 120 had typical SMA 1B defined by any three of the following: acute respiratory failure before 12 mos of age, gastrostomy before 12 mos of age, DDCV before 10 yrs of age, and VC not exceeding 200 ml, with plateaus for 15 at 26.4 (6-138) mos. Fifteen were with mild type 1C defined by three of the following: VC exceeding 200 ml, acute respiratory failure after 1 yr of age, gastrostomy after 1 yr of age, and no DDCV before 10 yrs of age and had a plateau/maximum observed mean VC of 409 (range, 200-1175) ml at 8.9 (range, 7-10) yrs of age. Of 88 patients with SMA 2 and paradoxical breathing (2A), 16 had a mean plateau/maximum observed VC of 758 (range, 460-2100) ml. DDCV with no autonomous breathing always followed plateauing of VC. Types 2B, 3, and 4 patients attained normal VC. Intergroup VC differences were significant (P < 0.05) for 1A, 1B, 1C, 2A, and 2B-4. When intubated and "unweanable," 50-ml VC signaled the ability for autonomous breathing 1 to 21 days after extubation. CONCLUSIONS VC should be monitored from birth. It correlates with prognosis with SMA 1A VCs not exceeding 100 ml or 1B 200 ml. Patients who attained 200 ml at any time (milder 1C) retain some ability to breathe after 10 yrs of age.

Reflex Yoga is based on the observation that the unconscious mind has far greater control of behavior, posture and breathing than the conscious mind. There is an immense amount of unconscious activity involved in standing, sitting and breathing. With perseverance we can "stand up straight" and "breathe deeply and evenly," and these instructions can be internalized and habitualized. But we must ask if this is the purpose and goal of yoga, even when backed by scientific research and traditional authority.

We describe a non-invasive breath monitoring method using spring coil which is used to detect the movement of the body surface due to breathing. In this method, subjectwares a vest with the spring coil on the inner side. Therefore, it is compressed by inspiration or prolonged by expiration. This change in length of the spring coil can be detected as change in electrical inductance of the coil. Based on this principle, we made a monitoring system of respiration. This system has three detecting positions, that is, the chest, abdomen, and back. Monitoring experiments of breath timing were performed for some healthy subjects. As results, this sensing system is able to detect the breath timings of subject sleeping on the back or abdomen and to check the sleeping position on the abdomen or back, simultaneously. It was confirmed that this system can also detect the paradoxical breathing in which phase of movement of the abdomen is opposite to that of the chest.

Paradoxical breathing or "paradoxical inward rib cage movements" without frank sleep apnea is a clinical de- scription of the "upper airway resistance syndrome". Upper airway resistance syndrome is thought to be a distinct entity and not just a part of the continuum of obstructive sleep apnea; paradoxical breathing in children appears to be a separate entity from the classic patient with marked adenotonsillar hypertrophy causing obstructive apnea. The treatment also dif- fers. We present a case of paradoxical breathing in a child who resolved spontaneously over a two year period.

A 53-year-old man rapidly developed quadriplegia and tachypnoea (figure 1). Electrophysiologic studies showed demyelinating features consistent with Guillain-Barre syndrome. He required a tracheostomy and gastrostomy. Paradoxical breathing, also known as thoracoabdominal asynchrony, was seen (see online video). Normally during inspiration, the abdomen and chest expand in a synchronised fashion. During inspiration a downward movement of diaphragm pushes the abdominal contents out as the ribs are lifted and moved out, causing both chest and abdomen to rise. With diaphragmatic paralysis, the diaphragm moves up rather than down during inspiration, and the abdomen moves in during chest rise.1 ,2 The neuromuscular respiratory failure follows a predictable pattern: failure of diaphragm and intercostal …

The call came, as such calls always do, at dinner time. It was Jenasi, the very capable nurse from Seghe clinic in Marovo Lagoon, on the radio. A man had just been brought in, having been attacked by a barracuda earlier in the day. He had a large gash in his chest, through which air was passing in and out as he breathed, but his observations were stable and he was not cyanosed. Hmmm. The sun had just set, which in the Solomon Islands means no evacuation, by air or otherwise, until first light. Should the wound be packed to stop the movement of air or not? We conducted a straw poll among the doctors: our ex-army officer said it definitely should be, as otherwise he was in danger of developing paradoxical breathing; our general practitioner surgeon said probably not; and the Swiss orthopaedic surgeon in the capital, who we contacted by phone, said definitely not as there was no possibility of putting a drain in should he develop a tension pneumothorax and he was currently well. (My own contribution was only to …

Introduction: Reconstruction of chest wall defects remains a very challenging issue and often requires prosthetic repair to prevent flail chest and paradoxical breathing. Case Report: We report a case of chest wall reconstruction following resection of a long standing, massive, well differentiated, primary chondrosarcoma of the sternum which was involving the entire body of sternum and adjoining costal cartilages. Excision of entire sternum (except manubrium) and 3rd to 8 th ribs left a large defect (20x15 cm) in the chest wall which was reconstructed with a simple and new three layer technique using Prolene-Vicryl mesh, long split rib grafts and lattissimus dorsi muscle (LD) flap. Patient was weaned off the ventilator on seventh post operative day and discharged after four weeks. Conclusion: This is a simple chest wall reconstruction technique for wide chest wall defects and is effective, safe, low cost and easily reproducible.

AUTOMOBILE, industrial and farm-machinery accidents are accountable for a large percentage of deaths occurring in the young and middle-aged groups. Approximately 25 per cent of these deaths are due to nonpenetrating thoracic injuries. The majority of such deaths are the result of deranged pulmonary function. Extensive rib fractures with paradoxical breathing, obstructed air passages, the "wet lung" and hemopneumothorax comprise the majority of the serious thoracic injuries or complications. More unusual injuries or complications are trauma to the heart and great vessels, the tracheobronchial tree, the esophagus, the thoracic duct and the diaphragm. A search of the literature reveals 1 . . .