Rare Cause Of Respiratory Distress Research Articles

Empyema thoracic in a neonate co-infected with SARS-CoV-2 and staphylococcus arouse successfully treated with fibrinolysis: a brief report

BackgroundEmpyema as a rare cause of respiratory distress in neonatal period has not yet been reported after Corona virus disease 2019 (COVID-19) and even rarely in the context of bacterial infections is age group.Case PresentationThe first neonate of quadruplets, resulting from Cesarean-Section of a 30-year-old mother without a history of previous illness, born at 34 weeks of gestation with a birth weight of 1600 gram. All four newborns were hospitalized; while the first one underwent nasal continuous positive airway pressure (N-CPAP) with a positive end-expiratory pressure of 6 and fractional inspired oxygen of 0.6, due to respiratory distress, noting a respiratory score of five out of 12.She was resuscitated one hour later due to bradycardia and underwent ventilator support. She received a single dose of pulmonary surfactant, intravenous paracetamol, and phenobarbital due to respiratory distress syndrome, Pectus Ductus Arteriosus, and unilateral clonic movements, respectively. In the lack of positive blood culture, she extubated two days later and breast-feeding was started. On day eight, she underwent high flow nasal cannula. On day 12, she suddenly developed respiratory distress, mottling, and abdominal distension, leading to N-CPAP and re-evaluation. The nasopharyngeal sampling for severe acute respiratory syndrome coronavirus-2 (SARS-CoV-2) real time Polymerase chain reaction and the blood culture for staphylococcus aurous became positive. A large volume pleural effusion including septa and loculation in right hemi-thorax on chest computerized tomography scan and ultrasound was seen, necessitating to thoracotomy, which was not possible due to her general condition. Remdesivir was started and antibiotics changed to a more broad-spectrum coverage. Chest tube was inserted and Alteplase was injected for three consecutive days with beneficial effects. She had no problem for six-month follow up.ConclusionThis was a case of empyema thoracic in the context of SARS-CoV-2 and Staphylococcus arouses co-infection. In our experience, it can be treated by chest drainage and fibrinolysis in neonatal period.

PHRENIC NERVE PALSY: A RARE CAUSE OF RESPIRATORY DISTRESS IN NEWBORN

Phrenic nerve palsy is a rare cause of respiratory distress in newborns, and often under-diagnosed, It is often accompanied by brachial plexus palsy. The possibility of spontaneous recovery should be considered before choosing surgery. This rare cause of respiratory distress of the newborn can be easily overlooked among the many common etiologies of respiratory distress of the newborn if this entity is not kept in mind and a thorough workup is not performed.We report a newborn who was admitted to the neonatal unit with respiratory distress and paresis of the right upper limb. He had a dystocia labor. Chest x-ray and ultrasound showed an elevation of the right diaphragmatic dome. The diagnosis was phrenic nerve palsy associated with brachial plexus palsy secondary to birth trauma. The newborn was treated with continuous positive airway pressure (CPAP) ventilation without surgical intervention.The outcome was favorable with spontaneous recovery at the age of 2 months.The management of this association is not yet codified.Further investigations will be necessary to develop more definitive guidelines for the treatment of this condition.

The Clinical Features and Risk Factors of Subglottic Cysts in Children: A Clinical Experience Using the Spontaneous Respiration Technique.

Subglottic cysts (SGCs) are a rare cause of respiratory distress resulting from upper airway obstruction in infants and young children. Risk factors other than prematurity with a history of endotracheal intubation have not yet been well elucidated. Therefore, we aimed to describe the clinical features and analyze the risk factors of SGCs. We conducted a retrospective review of medical records of pediatric patients who underwent marsupialization for SGCs between January 2017 and March 2022. These records were then compared with those of controls with a history of neonatal intubation, with a case-to-control ratio of 1:3. Eleven patients (eight boys and three girls) diagnosed with SGCs and 33 control patients (26 boys and seven girls) were included. All patients had a history of premature birth and neonatal intubation. Symptoms of SGCs appeared at a mean age of 8.2 months (range, 1-14 months) after extubation. The mean duration of intubation was 21.5 days (range, 2-90 days), and the intubation period was longer in patients with SGCs than in controls (21.5±24.8 days vs. 5.3±7.1 days; P<0.001). Furthermore, gestational age (28.3±4.2 weeks vs. 33.8±4.4 weeks; P=0.001) and birth weight (1,134.1±515.1 g vs. 2,178.2±910.1 g; P=0.001) were significantly lower in patients with SGCs than in controls. Multivariable analysis identified the intubation period as an independent risk factor. This study showed that gestational age, birth weight, and the intubation period were significantly associated with the development of SGCs. Pediatric patients presenting with progressive dyspnea who have the corresponding risk factors should undergo early laryngoscopy for the differential diagnosis of SGC.

Dapsone-induced methemoglobinemia in a patient of lichen planus: A case report

Introduction: Methemoglobin (MetHb) is the rare form of hemoglobin (Hb) produced by abnormal oxidation of Hb. Methemoglobinemia may be hereditary or acquired after exposure to certain drugs, toxins, chemicals, food, etc., Case Presentation: Here, we report a case of methemoglobinemia induced by dapsone in a young girl who presented with respiratory distress and was treated successfully with methylene blue (MB). Conclusion: MetHb levels can be seen increased inpatient on dapsone. Methemoglobinemia is a rare cause of respiratory distress and tachypnea in a patient on dapsone. MB and Vitamin C are recommended treatments in such cases.

Was It Pneumothorax? A Case Report of a Neonate with Congenital Pulmonary Airway Malformation from Rural Nepal

Formerly called congenital cystic adenomatoid malformation (CCAM), congenital pulmonary airway malformation (CPAM) is a rare but most common congenital lung malformation that may manifest as an asymptomatic lesion at birth or in later life. The diagnosis may be difficult and is easily confused for pneumothorax often resulting incorrect ICD insertion on an emergency basis, which causes complications and delays accurate diagnosis. We report a case of a neonate, who presented with increasing respiratory distress at birth and hyperlucency in a chest radiograph initially misdiagnosed as pneumothorax and ICD inserted, later correctly diagnosed as CPAM type I with help of a CT chest. This case report is presented to highlight that CPAM is a rare cause of respiratory distress in newborns at birth.

Bilateral diaphragm paralysis: a clinical case

Diaphragm dysfunction is a rare cause of respiratory distress with a variety of clinical manifestations that complicate diagnosis and treatment. The given clinical case demonstrates the possibility of detecting bilateral diaphragm paralysis using available general clinical and instrumental diagnostic methods. Among the physical data, high standing of the lower borders of the lungs with limited mobility and paradoxical movement of the diaphragm during the Mueller test have a high diagnostic value. Chest X-ray demonstrates the high standing of both domes of the diaphragm and subsegmental atelectasis in the basal parts of the lungs. Severe hypoxemia developed: oxygen saturation in clino- and orthostasis was 72 and 96%, respectively. The tests of pulmonary function showed significant restrictive impairments, a decrease in the vital capacity of the lungs was also determined. Ultrasound examination of the diaphragm revealed hyperechogenicity, lack of inspiratory thickening, and respiratory mobility of the domes of the diaphragm. Electromyography confirmed gross right and left phrenic nerve axonopathy. An idiopathic variant of diaphragm dysfunction can be assumed based on the patient stabilization during CPAP therapy, physiotherapy exercises, chest massage, followed by the disappearance of signs of bilateral diaphragm paralysis. Conclusion. The presented case demonstrates the difficulties of diagnosing bilateral diaphragm paralysis. The final diagnosis was made through the use of specific research methods recommended for suspected diaphragm dysfunction. The prognosis of the idiopathic variant of bilateral diaphragm paralysis, as in this case, is favorable. Spontaneous remission was observed.

Spondylocostal dysplasia: a rare cause of respiratory distress in term neonates

Jarcho-Levin syndrome (JLS) also known as spondylocostal dysplasia is a rare congenital dysplastic disorder involving ribs and vertebrae. It as an autosomal recessive form of inheritance however dominant forms have also been reported. Multiple genes have been postulated to be causative, however exact genetic basis is not known. Infants usually present at birth with respiratory distress and have varying degree of thoracic and vertebral anomalies. We present a case of term male infant with a clinical diagnosis of JLS. The infant presented at birth with distress with no significant antenatal risk factors. On examination he was found to have a short neck, short trunk, accessory nipple with asymmetrical chest wall. X-ray suggested absent ribs (2-8) on left side with hemivertebrae (T3-T8). The baby was managed conservatively and is being followed up with neonatologists, orthopaedicians and pediatric surgeons.

A rare cause of respiratory distress in the critical care setting.

A rare cause of respiratory distress in the critical care setting.

A Rare Cause of Respiratory Distress in Infants: Double Aortic Arch

Vascular ring, which is a rare cause of respiratory distress in infants, results in respiratory and gastrointestinal findings due to tracheal and/or esophageal compression, but the diagnosis may be delayed because of the nonspecific symptoms. Double aortic arch is the most common vascular ring anomaly. A case of double aortic arch presented with respiratory distress, stridor, and feeding intolerance during infancy is discussed.

Phrenic nerve palsy: A rare cause of respiratory distress in newborn: A literature review

Phrenic nerve palsy could result from multiple etiologies including birth injuries. It could often be associated with brachial plexus palsy. The phrenic nerve plays a major role in innervating the diaphragm. It is the only motor supply to the diaphragm and also acts a sensory nerve. In this palsy, the patient may present with diaphragmatic dysfunction, unilateral diaphragmatic paralysis, or bilateral diaphragmatic paralysis. This diaphragmatic paralysis caused by phrenic nerve injury could lead to respiratory distress in the newborn. Thus, a newborn with respiratory distress and an elevated hemidiaphragm should be suspected for phrenic nerve palsy in the imaging studies and also in the differential diagnosis of respiratory distress in a newborn presenting with a history of traumatic delivery or brachial plexus palsy. We also herein report a case of phrenic nerve palsy in a newborn presenting with respiratory distress.

Diaphragmatic paralysis in young children: A literature review.

Diaphragmatic paralysis (DP) is a rare cause of respiratory distress in young children. In the first years of life, the main cause is phrenic nerve injury after cardiothoracic surgery or obstetrical trauma. DP usually presents as respiratory distress. Asymmetrical thorax elevation, difficulty weaning from mechanical ventilation, pulmonary atelectasis, and repeated pulmonary infections are other suggestive signs or complications. DP is usually suspected on chest X-ray showing abnormal hemidiaphragm elevation. Although fluoroscopy was considered the gold standard for DP confirmation, it has gradually been replaced by ultrasound, which can be done at the bedside. Some electrophysiological tools may be useful for a better characterization of phrenic nerve injury and chance of recovery. The management of DP is mainly based on clinical severity. In mild asymptomatic cases, DP may only require close monitoring. In more severe cases, adequate ventilatory support and/or surgical diaphragmatic plication may be needed. Electrophysiological tools may help clinicians assess the ideal timing for diaphragmatic plication.

Congenital lobar emphysema: a rare cause of respiratory distress in a neonate

Congenital lobar emphysema (CLE) is a congenital condition characterized by distension and air trapping of the affected lobe of the lung. It is one of the causes of infantile respiratory distress, which may require surgical resection of affected lobe. Case characteristics: 3-day-old neonate with ventilation refractory respiratory distress. Imaging was suggestive of decreased lung tissue on the right side with ipsilateral mediastinal shift. Intervention/ outcome: Early surgical lobectomy was done to improve lung functions and the child improved dur to early intervention. Message: An early diagnosis with high index of suspicion helps patients with this rare congenital anomaly. Early intervention is the key to good long-term outcome. More awareness about the entity and treatment options available would greatly help improving the outcome and disease burden.

Surgical management of congenital lobar emphysema: A 15 years experience in a tertiary thoracic surgery unit

Backgroundcongenital lobar emphysema is a rare cause of respiratory distress in neonates and infants. It was treated by lobectomy since 1943.The aim of this study was to present our experience in the diagnosis and surgical treatment of congenital lobar emphysema cases and highlight the effect of multi-displinary team work on the outcome. MethodsWe retrospectively analyzed data of patients operated for congenital lobar emphysema at cardiothoracic surgery department, Sohag university hospital over the period of 15 years starting from January 2000 and ending in December 2015. ResultsTotal number of cases was fifty three. Males were 39 (74%) while females were 14 (26%). Mean age was 12 weeks (3 weeks - 15 months). Six patients (11%) had associated cardiac anomalies. Main clinical presentations were acute respiratory distress and recurrent chest infection. Left upper lobe was affected in 35 cases (66%), right upper lobe in 10 cases (19%) and right middle lobe in 8 cases (15%). All cases were subjected to anatomical lobectomy by thoracotomy. One patient was subjected to patent ductus arteriosus ligation simultaneously with left upper lobectomy at the same operative setting. Post operative complications were reported in 8 cases (15%) and one patient of them died from sepsis and respiratory failure (1.9%). Postoperatively, all surviving patients were followed up for one year in the outpatient clinic. Conclusionscongenital lobar emphysema is a life threatening disease. Early diagnosis and surgical management by a multi-displinary team is the golden key for favorable and safe outcome.

Laryngocoele with contralateral laryngopyocoele - a rare cause of respiratory distress

Laryngocoele with contralateral laryngopyocoele - a rare cause of respiratory distress

BRONCHOGENIC CYST: A RARE CAUSE OF RESPIRATORY DISTRESS IN NEONATE - A CASE REPORT

Bronchogenic cyst is an unusual congenital pulmonary malformation which may occur at various sites of tracheobronchial tree as well as unusual ectopic site like cervical, scapular region, pericardium and abdomen. Newborn with large cyst can develop respiratory distress, cyanosis and feeding difficulty. A 37 day old male infant presented with failure to thrive, feeding difficulty, respiratory distress and cyanosis. His chest radiograph was suggestive of shift of mediastinum and heart with hyperinflatted left lung. CT thorax showed minimal enhancing cystic lesion posterior to trachea and its bifurcation on right side, it was compressing the right main bronchus. The cyst was successfully surgically excised which was adhering to carina and intrabronchial extension and its histopathological finding confirmed the bronchogenic cyst. Though, bronchogenic cyst is rare cause of respiratory distress, cyanosis, and failure to thrive and feeding difficulty, it should be considered one of the differential diagnoses.

Congenital idiopathic chylothorax: A very rare case

Chylothorax is a very rare cause of respiratory distress in the neonates, defined as a collection of chyle in the pleural space. Causes of chylothorax in neonates are congenital lymphatic malformations, associated syndrome or birth trauma. Here, we are presenting a neonate presented with severe respiratory distress require intubation and ventilator support and on evaluation no cause of chylothorax found. Baby managed with intercostal drainage and octreotide, improved and asymptomatic in 8 months of follow-up.

A rare cause of respiratory distress after transthoracic oesophagectomy.

Transthoracic oesophagectomy is a standard surgical procedure for oesophageal cancer. Because of thoracotomy and lung handling, perioperative pulmonary complications make such procedures challenging. The issues related to respiratory complications may be predicted and managed accordingly. However, we report two cases of respiratory compromise caused due to a peculiar iatrogenic component.

A Case of Palytoxin Poisoning in a Home Aquarium Enthusiast and His Family

Inhalational exposure to palytoxin is an extremely rare cause of respiratory distress. This little-known marine toxin has the potential to cause significant morbidity and mortality. Toxicity has been best documented in cases of ingestion but has also been seen in cases of dermal exposure and inhalation of vapors. Palytoxin has been found in several coral species, some of which are favored by home aquarium enthusiasts and are commercially available. We report a case of a family who were exposed to the aerosolized toxin following the cleaning of a coral in their home aquarium. It is important that clinicians be aware of this source of toxic exposure to provide necessary care to these patients.

Patient selection in congenital pyriform aperture stenosis repair – 14 year experience and systematic review of literature

PurposeCongenital nasal pyriform aperture stenosis (CNPAS) is a rare cause of respiratory distress in neonates that may necessitate early surgical intervention. Restenosis and granulation are postoperative concerns that may prompt a return to the operating room. Reoperation places children at increased risk of perioperative complications and prolonged hospital stays. We are presenting a review of our institutional experience of 16 patients treated for CNPAS over a 14 year period and a systematic review with pooled data analysis to determine the effect of craniofacial and neurologic anomalies on surgical success. MethodsRetrospective chart review of all cases of CNPAS treated at our tertiary children's hospital between 1999 and 2013. Systematic review of English language literature was conducted adhering to the PRISMA statement to determine the effect of neurologic anomalies and craniofacial dysmorphism (CFD) on surgical failure for CNPAS treatment. Univariate and exact multiple logistic regression were used for analysis of an individual patient data analysis. Results10 patients had surgery and 6 were treated medically. Average pyriform apertures were 5.71±1.72mm for the surgical group and 4.83±1.26mm for the medical group (p=0.38). 31% had neurological impairments. 31% had craniofacial dysmorphisms (CFD). 2 patients developed restenosis and 1 required tracheotomy. Both of these patients had other CFDs. Literature review captured 63 surgical patients and 9 failures in 6 series of CNPAS. 4.6% of patients without CFD and 36.8% of patients with CFD required surgical revision (p=0.023, OR13.8). ConclusionWhen repairing CNPAS, co-morbidities must be considered. Impaired respiration, central neurologic deficits and extensive craniofacial anomalies may require additional surgeries or an alternative approach.

Hypoplasia of the Maxilla: Differential Diagnosis of Nasal Obstruction in Infants

Introduction: The maxillary hypoplasia (MH) is a rare cause of respiratory distress in newborns (NB), which may be associated with genetic disorders such as holoprosencephaly and Apert syndrome. This pathology is part of the differential diagnosis of congenital nasal obstruction in neonates, along with choanal atresia and stenosis of the piriform aperture and nasolacrimal duct cyst.