Orthopnea secondary to brachial plexitis with bilateral diaphragmatic paralysis.

BACKGROUND: Diaphragmatic paralysis can present with orthopnea. We report a unique presentation of bilateral diaphragmatic paralysis, an uncommon diagnosis secondary to an unusual cause, brachial plexitis. This report thoroughly describes the patient's presentation, workup, management, and outcome. It also reviews the literature on diaphragmatic paralysis and Parsonage-Turner syndrome. CASE PRESENTATION: A 50-year-old male patient developed insidious orthopnea associated with left shoulder and neck pain over three months with no associated symptoms. On examination, marked dyspnea was observed when the patient was asked to lie down; breath sounds were present and symmetrical, and the neurological examination was normal. The chest radiograph showed an elevated right hemidiaphragm. Echocardiogram was normal. There was a 63% positional reduction in Forced Vital Capacity and maximal inspiratory and expiratory pressures on pulmonary function testing. The electromyogram was consistent with neuromuscular weakness involving both brachial plexus and diaphragmatic muscle (Parsonage and Turner syndrome). CONCLUSIONS: Compared to unilateral, bilateral diaphragmatic paralysis may be more challenging to diagnose. On PFT, reduced maximal respiratory pressures, especially the maximal inspiratory pressure, are suggestive. Parsonage-Turner syndrome is rare, usually with unilateral diaphragmatic paralysis, but bilateral cases have been reported.

Bilateral Phrenic Nerve Palsy Associated With Neuralgic Amyotrophy.

Neuralgic amyotrophy (NA) is a multifocal inflammatory neuropathy accompanied by acute pain and muscle atrophy. NA commonly affects the upper extremities, but rarely affects the phrenic nerve. Here, we report a male with neck pain, orthopnea, difficulty sleeping in the supine position, and inability to slurp. His saturated oxygen level decreased from 97% to 86% in the supine position. His right shoulder showed muscle atrophy. Chest X-ray examination in the supine position and a nerve conduction study showed phrenic palsy. We diagnosed it as bilateral phrenic nerve palsy associated with NA. NA sometimes causes phrenic nerve palsy and may cause slurping difficulty.

Strange Saucer-Shaped Lung Collapse Successfully Salvaged in Spite of Delayed Intervention.

A middle-aged woman presented in November 2023 with exertional dyspnea and a chronic cough for three months. She had undergone a repeat mitral valve replacement (MVR) surgery five months prior. She had a tissue MVR in 2016, which degenerated, making her symptomatic, and hence had to be replaced with a metallic valve. As the respiratory symptoms recurred two months post-op, she was evaluated for the integrity of the newly placed mitral valve, which was found to be functioning well. The left ventricular function was well preserved, and she had no vegetation or clots. She was therefore investigated further for other possible causes. Chest X-ray showed a strange saucer-shaped (or disc-shaped) opacity above the middle portion of the right hemidiaphragm, which itself was found to be elevated. An ultrasound of the chest ruled out subpulmonic pleural effusion and confirmed right hemidiaphragm palsy. A computed tomography (CT) scan of the thorax was suggestive of a strange-shaped collapse of the right lower lobe with tortuous air bronchograms and a small intraluminal soft tissue shadow in the right lower lobe bronchus. A bronchoscopy confirmed collapsing segments of the right lower lobe due to external compression. It also ruled out any intrabronchial pathology causing obstruction, effectively confirming that the tissue shadow was probably just a mucus plug. A possible phrenic nerve injury during thoracotomy at the time of MVR was thus concluded. A diaphragmatic plication was advised considering that she had significant orthopnea and low peripheral oxygen saturation and that the collapsed lung would not possibly expand beyond six months or so and could in itself act as a focus for further mucus stagnation, leading to infection and further bronchiectasis, which had probably already started developing. It was difficult to persuade the patient for a third thoracotomy and she took much time to decide. Good counselling, rapport building, and assurance that, although the lung may not fully expand beyond six months, at least the orthopnea would significantly improve, she finally consented to diaphragmatic plication, which was done after 10 months of the MVR surgery. Aggressive post-operative chest physiotherapy and rehabilitation were promptly initiated. The lung completely expanded one month post-op and was thus successfully salvaged.

Not So Transient Effusive-Constrictive Pericarditis.

A 63-year-old male patient presented with chest pain and signs of volume overload. His work-up revealed a diagnosis of transient effusive constrictive pericarditis of idiopathic etiology. Despite treatment with optimal medical therapy, he continued to experience persistent symptoms eventually requiring radical pericardiectomy.

A Case of Chronic Aortic Dissection With Hemopericardium and Tamponade.

Type A aortic dissection involves the separation of the wall of the ascending aorta into a true lumen and a false lumen. The finding of an aortic dissection in a patient experiencing mild to moderate symptoms for several weeks may be surprising for clinicians, given the severity of the underlying process. Here, we present an 88-year-old patient who was admitted to our hospital due to orthopnea and leg swelling for the past two to three weeks and was found to have a chronic dissection of the ascending aorta, complicated by hemopericardium and tamponade. The existing literature very rarely reports chronic type A aortic dissection with tamponade on presentation.

Instant orthopnea and pseudo-cardiac dilatation due to anterior mediastinal tumor.

A 45-year-old woman presented with instant orthopnea and enlarged cardiomediastinal silhouette in her chest radiograph. Although anterior mediastinal tumor can be misdiagnosed as heart failure due to orthopnea with enlarged cardiomediastinal silhouette, "instant orthopnea" may be a useful sign to distinguish these conditions.

Prediction of respiratory collapse among pediatric patients with mediastinal tumors during induction of general anesthesia.

PURPOSE: Fatality resulting from respiratory collapse (RC) during general anesthesia (GA) induction in children with mediastinal tumors has been reported. We explored potentially useful parameters for predicting the risk of RC based on objective imaging results. METHODS: We retrospectively reviewed the records of 31 patients (median age: 4years; range: 4months-15years) with mediastinal tumors treated between 2000 and 2015. Comparing those with RC under GA induction to those without (RC group vs. non-RC group), we analyzed a variety of factors that might predict respiratory obstruction during GA induction, including our new parameter, the standardized tumor volume (STV), which is adapted from the formula for the volume of an ellipsoid. RESULTS: All eight patients in the RC group had large tumors in the anterior mediastinum, including lymphoma, teratoma, and germ cell tumor. The mean STV value of the RC group was significantly larger than that of the non-RC group (3.6±1.4 vs. 1.4±1.0, p=0.006). Using an STV cut-off value of 2.5, the sensitivity and specificity for predicting RC under GA induction were both 0.86, making STV more useful than previously reported risk factors. CONCLUSION: Anterior mediastinal tumors in children can often cause airway obstruction under GA induction. Measuring STV can help predict the respiratory risk during GA among pediatric patients with anterior mediastinal tumors. LEVEL OF EVIDENCE: III.

An unusual case of orthopnea.

Bilateral diaphragmatic paralysis is a known cause of respiratory failure. Diagnosis can be difficult, particularly in the acute setting. We present the case of a gentleman diagnosed with bilateral diaphragmatic paralysis secondary to phrenic neuropathy in the setting of cervical spondylosis.

Mechanisms of Orthopnea in Stable Obese Subjects.

BACKGROUND: The present study explored the role of closing volume as a determinant of orthopnea in stable obese subjects. We hypothesized that: (1) increase in closing volume in supine position would be greater in orthopneic than in non-orthopneic subjects, and (2) the relationship of change in closing volume to change in dyspnea with position would be dependent on expiratory flow limitation in the sitting position. METHODS: In stable obese subjects, in sitting and supine positions, we measured the Borg dyspnea score, static lung volumes, expiratory flow limitation during tidal breathing, and single-breath nitrogen expiration test. From the latter, we determined closing volume and closing capacity, slope of phase III, and opening capacity. Orthopnea was defined as any increase in the Borg score in the supine position from its value in the sitting position. RESULTS: Twenty-one subjects (13 women), median age (interquartile range) 55 (49-57) y and with body mass index of 39 (38-42) kg/m(2) were included, of whom 12 were orthopneic and 11 had expiratory flow limitation while seated. In the sitting position, orthopneic and non-orthopneic subjects were similar for age, body mass index, and pulmonary function tests, including single-breath nitrogen expiration test-derived variables. In the orthopneic subjects, there were no changes in any respiratory variable between positions. In the non-orthopneic subjects, there was a significant decrease in slope of phase III in the supine position from 1.67 (1.33-3.60) to 1.40 (1.25-1.66)%/L (P = .008). Overall, the subjects' Borg score significantly correlated with the slope of phase III (r = 0.63, P = .002) and opening capacity (r = -0.47, P = .03). In 10 subjects without expiratory flow limitation, it correlated with slope of phase III (r = 0.68, P = .03). CONCLUSIONS: In stable obese subjects, magnitude of orthopnea correlated with an increase in the slope of phase III in subjects without expiratory flow limitation. Expiratory flow limitation should be taken into account in obese patients.

Pedal Edema as an Indicator of Early Heart Failure in the Community: Prevalence and Associations With Cardiac Structure/Function and Natriuretic Peptides (MESA [Multiethnic Study of Atherosclerosis]).

BACKGROUND: The prevalence of pedal edema (PE) and its associations with abnormal cardiac structure/function, natriuretic peptides, and incident heart failure (HF) is unknown, especially in community-dwelling adults without a history of cardiovascular disease. METHODS AND RESULTS: Out of 5004 MESA (Multiethnic Study of Atherosclerosis) participants who had cardiac magnetic resonance imaging, 4196 had complete data and were included in this analysis (3501 for the right ventricle analysis). Logistic regression and Cox proportional hazard analyses were used to assess the associations among self-reported PE, 2-pillow orthopnea, paroxysmal nocturnal dyspnea, left and right ventricular structure and function, natriuretic peptide levels, and incident HF. PE was present in 28% of the participants. PE was not associated with overt left or right ventricular systolic dysfunction (ejection fraction <50%). PE was associated with 2-pillow orthopnea (odds ratio 1.66; 95% confidence interval [CI], 1.30-2.12), paroxysmal nocturnal dyspnea (odds ratio 1.95; 95% CI, 1.55-2.44), and abnormal N-terminal pro-B-type natriuretic peptide levels (defined as >400 pg/mL; odds ratio 1.80; 95% CI, 1.21-2.68) in adjusted models. After a mean of 10.2 years of follow-up, 184/4196 (4.4%) participants had an adjudicated incident HF hospitalization. PE was associated with incident HF hospitalization in models adjusted for age, sex, and race (hazard ratio 1.44; 95% CI, 1.05-1.97). This association persisted after adding additional covariates, including comorbidities, baseline left ventricular ejection fraction, and antecedent myocardial infarction (hazard ratio 1.43; 95% CI, 1.02-1.99). The association of PE with incident HF was attenuated by further adjustment for N-terminal pro-B-type natriuretic peptide. CONCLUSIONS: PE is prevalent in community-dwelling adults without clinically recognized cardiovascular disease and associated with future hospitalized HF.

Chiari I malformation associated with atlanto-occipital assimilation presenting as orthopnea and cough syncope.

Although it is not uncommon for patients with Chiari I malformations to present with respiratory complaints, cough syncope is a rare presenting symptom. We report an adult patient who had both a Chiari I malformation and atlanto-occipital assimilation, and complained of cough syncope, orthopnea, and central sleep apnea. The patient underwent decompressive craniectomy of the posterior fossa and a cervical level 2 laminectomy. However, due to an initial under-appreciation of the profound narrowing of the foramen magnum as a result of these concomitant pathologies, the patient had continued impaired cerebrospinal fluid flow, leading to a symptomatic pseudomeningocele and required a more extensive decompression that included a cervical level 3 laminectomy as well as a temporary lumbar drain. On 2 year follow-up, he remained asymptomatic.

Chiari I malformation associated with atlanto-occipital assimilation presenting as orthopnea and cough syncope: a case report and review of literature.

Although it is not uncommon for patients with Chiari I malformations to present with respiratory complaints, cough syncope is a rare presenting symptom. We report an adult patient who harbored both a Chiari I malformation and atlanto-occipital assimilation who complained of cough syncope, orthopnea, and central sleep apnea. The patient underwent decompressive craniectomy of the posterior fossa and cervical level 2 laminectomy. However, due to a possible initial underappreciation of the profound narrowing of the foramen magnum as a result of these concomitant pathologies, the patient may have had continued impaired cerebrospinal fluid flow, leading to a symptomatic pseudomeningocele and requiring a more extensive decompression that included a cervical level 3 laminectomy as well as a temporary lumbar drain. On 2-year follow-up, he has remained asymptomatic.

Pericardial hydatid cyst in oblique sinus, obstructing all pulmonary veins: a rare presentation.

Hydatid cyst of the heart is an uncommon presentation of hydatidosis. We present a case of pericardial hydatid cyst in pericardial oblique sinus with extension to posterior wall of left atrium (LA), occluding all pulmonary vein ostia in a 35 year-old female with progressive dyspnea and severe orthopnea.

[Platypnea-orthodeoxia syndrome: case report]. / Le syndrome de platydéoxie-orthopnée : à propos d'un cas.

We report the case of an 80-year-old woman with symptomatic postural hypoxaemia caused by a right-to-left shunt through a patent foramen ovale. The hypoxaemia was enhanced by the supine position and disappeared in upright position. Potential mechanisms underlying postural variations of the shunt seemed to be similar to those describe in platypnea-orthodeoxia syndrome. Patient became asymptomatic after shunt resolution.