Anti-synthetase syndrome in a child with pneumomediastinum: a case report and literature review.

BACKGROUND: Anti-synthetase syndrome (ASS) is a group of rare clinical subtypes within inflammatory myopathies, predominantly affecting adult females. Instances of critical illness associated with ASS in children are even rarer. CASE PRESENTATION: We report the case of a 7-year-old boy finally diagnosed with ASS, combined with pneumomediastinum. He presented with intermittent fever persisting for 12 days, paroxysmal cough for 11 days, chest pain, and shortness of breath for 4 days, prompting admission to our hospital. Pre-admission chest CT revealed diffuse pneumomediastinum, subcutaneous pneumatosis in the neck and bilateral chest wall, consolidation, atelectasis, and reticular nodular shadowing in both lungs, as well as pericardial effusion and bilateral pleural effusions. Laboratory tests revealed a positive result for serum MP immunoglobulin M (MP-IgM) and MP immunoglobulin G (MP-IgG). The patient was initially diagnosed with mycoplasma pneumoniae (MP) infection, and following 3 days of antibiotic treatment, the patient's tachypnea worsened. Positive results in muscle enzyme antibody tests included anti-PL-12 antibody IgG, anti-Jo-1 antibody IgG, and anti-RO-52 antibody IgG. Ultrasonography detected moderate effusions in the right shoulder, bilateral elbow, and knee joints. Corticosteroids pulse therapy was initiated on the 27th day following disease onset, and continued for 3 days, followed by sequential therapy for an additional 12 days. The child was discharged on the 43rd day, and subsequent follow-up revealed a significant improvement in consolidation and interstitial lesions in both lungs. CONCLUSIONS: ASS in children may combine with rapidly progressive interstitial lung disease (RPILD) and pneumomediastinum. It is crucial to promptly identify concurrent immunologic abnormalities during the outbreak of MP, particularly when the disease exhibits rapid progression with ineffective conventional antibiotic therapy.

[A Case of Drug-Induced Pneumomediastinum Preceding Interstitial Pneumonitis Onset during Treatment with a First-Line Regimen of GnP in a Patient with Unresectable Pancreatic Cancer].

BACKGROUND: The highest prevalence of drug-induced interstitial pneumonitis(IP)occurs in patients receiving antineoplastic agents, such as cytotoxic chemotherapeutic drugs, molecular targeted drugs, and immune checkpoint inhibitors. A certain period of the treatment for IP requires discontinuation of the anticancer therapy, resulting in progression of the malignant status. CASE: A 70-year-old man was incidentally diagnosed with locally advanced unresectable pancreatic cancer in the course of his treatment for ventricular dysrhythmia. After the insertion of a pacing instrument, he was ensured to be eligible to receive combination chemotherapy with gemcitabine and nab-paclitaxel(GnP)as the primary regimen. Shortly after the second course of GnP, the patient had high fever and developed pneumomediastinum 3 days prior to the onset of IP. The GnP treatment was suspended, and the IP was treated with pulse steroid therapy. The respiratory disorder took approximately 3 months to resolve; however, this concomitantly led to aggravation of the malignancy, which developed multiple metastases to the liver. The patient was no longer allowed to receive antineoplastic treatment. CONCLUSION: Although GnP may be a key regimen for the treatment of unresectable pancreatic cancer, patients should be closely monitored to ensure early detection of adverse events, such as interstitial pneumonia. Furthermore, drug-induced pneumomediastinum may be a precursor to the onset of interstitial pneumonia.

Pneumocystis jirovecii-related spontaneous pneumothorax, pneumomediastinum and subcutaneous emphysema in a liver transplant recipient: a case report.

BACKGROUND: Pneumocystis pneumonia (PCP) is a common opportunistic infection caused by Pneumocystis jirovecii. Its incidence at 2 years or more after liver transplant (LT) is < 0.1%. PCP-related spontaneous pneumothorax and/or pneumomediastinum is rare in patients without the human immunodeficiency virus, with an incidence of 0.4-4%. CASE PRESENTATION: A 65-year-old woman who had split-graft deceased-donor LT for primary biliary cirrhosis developed fever, dyspnea and dry coughing at 25 months after transplant. Her immunosuppressants included tacrolimus, mycophenolate mofetil, and prednisolone. PCP infection was confirmed by molecular detection of Pneumocystis jirovecii,in bronchoalveolar lavage. On day-10 trimethoprim-sulphamethoxazole, her chest X-ray showed subcutaneous emphysema bilaterally, right pneumothorax and pneumomediastinum. Computed tomography of the thorax confirmed the presence of right pneumothorax, pneumomediastinum and subcutaneous emphysema. She was managed with 7-day right-sided chest drain and a 21-day course of trimethoprim-sulphamethoxazole before discharge. CONCLUSION: Longer period of PCP prophylaxis should be considered in patients who have a higher risk compared to general LT patients. High index of clinical suspicion, prompt diagnosis and treatment with ongoing patient reassessment to detect and exclude rare, potentially fatal but treatable complications are essential, especially when clinical deterioration has developed.

Epidural pneumorrhachis, a rare complication of asthma exacerbation.

Introduction: An association between pneumorrhachis and asthma exacerbation is uncommon. However, we present a clinical case involving a patient with exacerbated asthma, subcutaneous emphysema, spontaneous pneumomediastinum (SPM), and pneumorrhachis. Case study: The patient was an 18-year-old male with asthma since childhood who only relied on salbutamol to control his asthma symptoms. Results: The patient suddenly experienced dyspnea, chest tightness, and paroxysmal coughing, which prompted him to visit the emergency department. Upon arrival, subcutaneous emphysema was detected on the face, neck, thorax, and left forearm. Chest X-ray showed air in the mediastinum, neck, left supraclavicular region, and chest, all of which were verified by a computed tomography scan that also revealed air in the epidural region. At the hospital, his treatment focused on preventing asthma exacerbation and managing associated symptoms. Conclusion: When a patient has asthma exacerbation that is accompanied by SPM and extensive emphysema, the presence of epidural pneumorrhachis should not be overlooked.

Spontaneous pneumomediastinum following pneumonia in a 23-year-old male patient.

Spontaneous pneumomediastinum (SPM) is defined as the presence of interstitial air in the mediastinum without any apparent precipitating factor. We present a case of 23 year old male patient, who has been referred to our outpatient clinic with the complaints of sudden chest pain, dyspnea followed by pneumonia and was diagnosed as SPM. The patient was treated with ampicillin sulbactam (4 gr/day) and methylprednisolon (20 mg/day) for 4 days. and oral intake was stopped during treatment. Post treatment, it was observed that the crepitations were disappeared thoroughly and vesicular sounds were heard by oscultation. The control values of arterial blood gas was as following: pH:7,39 pO2:95 mmHg, pCO2:37 mmHg, SaO2: %97. In the 5th day his oral intake was started and he was discharged.

IMAGING DIAGNOSIS-SPONTANEOUS PNEUMOMEDIASTINUM SECONDARY TO PRIMARY PULMONARY PATHOLOGY IN A DALMATIAN DOG.

A 1.5-year-old, 23 kg intact male Dalmatian dog was evaluated for acute respiratory insufficiency without a previous history of trauma or toxic exposition. Imaging revealed pneumomediastinum, pneumothorax, diffuse unstructured interstitial pulmonary pattern, pulmonary interstitial emphysema, and pneumoretroperitoneum. Histopathological evaluation of the lungs revealed perivascular and peribronchial emphysema, mild lymphocytic interstitial pneumonia with atypical proliferation of type II pneumocytes in bronchioles and alveoli. A lung disease resembling fibrosing interstitial pneumonia in man and cats has been previously reported in Dalmatians and should be included as a differential diagnosis for Dalmatians with this combination of clinical and imaging characteristics.

Survival analysis and risk factors for mortality in connective tissue disease-associated pneumomediastinum.

The aim of this study was to analyze the characteristics of patients with diffuse connective tissue diseases (CTDs) complicated by pneumomediastinum and identify the risk factors associated with increased mortality in these patients. Twenty-eight patients with CTD-associated pneumomediastinum, who were admitted to our hospital from January 1997 to June 2012, were prospectively studied. Their demographic characteristics, time to death, and potential risk factors were assessed. Survival curves were depicted by the Kaplan-Meier method. Univariate and multivariate survival analyses were performed by Cox regression. Of the 28 patients, 21 had dermatomyositis; two, polymyositis; three, systemic lupus erythematosus; one, polyarteritis nodosa; and one, undifferentiated CTD. The mean follow-up period was 1,461 days (54-5,264). The cumulative estimated Kaplan-Meier survival rate was 68 % at 1 week, 50 % at 1 month, and 43 % at 1 year. According to univariate analysis, higher serum albumin level (HR 0.87, 95 % CI 0.78-0.98), "slow air leak" (defined as time to progression of dyspnea [newly acquired respiratory failure, mechanical ventilation required, or decrease in PaO2 >30 mmHg after pneumomediastinum]) >3 days (HR 0.07, 95 % CI 0.02-0.34), and early initiation of immunosuppressive agents (within 1 month of steroid therapy; HR 0.27, 95 % CI 0.09-0.81) were associated with better prognosis. Final regression analysis revealed that slow air leak was associated with a lower mortality risk. We found that slow air leak was independently associated with better prognosis. Furthermore, most patients (86 %) who survived for at least 1 month following the pneumomediastinum event subsequently survived beyond 1 year.

Amyopathic dermatomyositis with lung involvement responsive to mycophenolate mofetil.

Amyopathic dermatomyositis (ADM) causes a severe pulmonary disease in 50% of patients and no validated therapeutic option is available. Mycophenolate mofetil permitted to control interstitial lung disease and pneumomediastinum in a patient with ADM. This drug has recently been used to treat interstitial lung disease in ADM, but its effectiveness has not been previously reported in case of pneumomediastinum. Patients with ADM need an accurate pulmonary surveillance and in case of pulmonary complications mycophenolate mofetil could be a new therapeutic option.

Integrated Chinese herbal medicine and Western medicine successfully resolves spontaneous subcutaneous emphysema and pneumomediastinum in a patient with severe COVID-19 in Taiwan: A case report.

CASE: Serious complications of severe coronavirus disease 2019 (COVID-19) include subcutaneous emphysema (SE) and pneumomediastinum, which are complicated to treat with conventional Western medicine. We report how combining Chinese herbal medicine (CHM) with Western medicine quickly resolved a patient's COVID-19-associated pulmonary complications, shortened hospital stay and improved quality of life. CLINICAL FEATURES AND OUTCOME: A 59-year-old male with a history of smoking and tumors was diagnosed with COVID-19 in May 2021. At hospitalization, his oxygen saturation (SpO2) was 80%, he had a continuous severe cough, rapid shallow breathing, spontaneous SE and pneumomediastinum. By Day 4 of hospitalization, his condition was worsening despite standard care, so CHM was added. After 3-5 days, his coughing had lessened and supplementary oxygen therapy was de-escalated. Nine days after starting CHM, the SE had completely resolved and the patient avoided intubation. His WHO OS 10-point Scale score had fallen from 6 to 3 points and the modified Medical Research Council Dyspnea Scale score from 4 to 2 points. He was hospitalized for 19 days. At 1 week post-discharge, the patient could handle most of his daily activities and experienced minor shortness of breath only when performing labor-intensive tasks. At 1 month, his work output was restored to pre-COVID-19 levels. CONCLUSION: CHM combined with standard Western medicine improved pulmonary function, respiratory rate, blood oxygen saturation and shortened the hospital stay of a patient with severe COVID-19 complicated by SE and pneumomediastinum.

Pneumorrhachis and pneumomediastinum in connective tissue disease-related interstitial lung disease: case series from a tertiary care teaching hospital in South India.

Pneumomediastinum has been described as a rare complication of connective tissue diseases. Here, we report four cases of pneumomediastinum: three of which are associated with dermatomyositis and one with mixed connective tissue disease. All our patients had interstitial lung disease. The first case of dermatomyositis described below was complicated by epidural emphysema (pneumorrhachis) in addition to pneumomediastinum. Pneumorrhachis is reported in many isolated case reports and series in the setting of asthma, pneumothorax, blunt chest trauma, etc. Less than 10% of pneumomediastinum cases develop this complication and vast majority of cases resolve spontaneously. The mechanism behind this has been postulated to be the passage of air through the intervertebral foramen. Others suggest entrapment of air which dissects between paraspinal soft tissues and along the vascular and nerve sheaths into the epidural space. This is the first ever reported case of epidural emphysema in connective tissue disease to the best of our knowledge.

[Visit at the dentist with consequences]. / Zahnarztbesuch mit Folgen.

A 52-year-old patient presented to the emergency department of the polyclinic with a cervicofacial swelling. The indolent swelling appeared after professional tooth cleaning. Computed tomography scanning of the cervical region and thorax revealed entrapped air extending from the frontotemporal to the mediastinal regions. The patient was diagnosed with cervicofacio mediastinal emphysema and subsequently treated with antibiotics. The symptoms subsided and the patient left hospital free of symptoms.

Newly formed cystic lesions for the development of pneumomediastinum in Pneumocystis jirovecii pneumonia.

BACKGROUND: Pneumocystis jirovecii, formerly named Pneumocystis carinii, is one of the most common opportunistic infections in human immunodeficiency virus (HIV)-infected patients. CASE PRESENTATIONS: We encountered two cases of spontaneous pneumomediastinum with subcutaneous emphysema in HIV-infected patients being treated for Pneumocystis jirovecii pneumonia with trimethoprim/sulfamethoxazole. CONCLUSION: Clinicians should be aware that cystic lesions and bronchiectasis can develop in spite of trimethoprim/sulfamethoxazole treatment for P. jirovecii pneumonia. The newly formed bronchiectasis and cyst formation that were noted in follow up high resolution computed tomography (HRCT) but were not visible on HRCT at admission could be risk factors for the development of pneumothorax or pneumomediastinum with subcutaneous emphysema in HIV-patients.

Dermatomyositis, complicated with pneumomediastinum, successfully treated with cyclosporine A: a case report and review of literature.

We report a rare case of patient with dermatomyositis (DM) who developed spontaneous pneumomediastinum (PnM) and subcutaneous emphysema. She was successfully treated with oral prednisolone and cyclosporine A (CsA). We reviewed the cases of PnM in patients with DM treated with CsA. A review of four previously reported cases revealed that treatment with systemic glucocorticoid and CsA was effective for the DM and PnM. We indicate that initial and early treatment of the patients with DM and PnM with CsA enabled rapid tapering of the dose of glucocorticoid and improved the disease.

Spontaneous pneumomediastinum and epidural pneumatosis in an adolescent precipitated by weight lifting: a case report and review.

Spontaneous pneumomediastinum is an uncommon condition in athletes. The most common cause of spontaneous pneumomediastinum is alveolar rupture into the bronchovascular sheath as a result of increased intrathoracic pressure. Epidural pneumatosis (pneumorrhachis) has been rarely associated with spontaneous pneumomediastinum. In this article, we describe a case of a 17-year-old male who presented with neck and chest pain that started 14 hours after a weight lifting session. He developed both a pneumomediastinum and epidural pneumatosis--an association that is rarely reported in a setting without trauma. To our knowledge, there have been only 5 case reports of pneumomediastinum precipitated by weight lifting. Improper breathing technique during weight lifting can increase the intrathoracic pressure and the risk of pneumomediastinum; hence, it is important that physicians and trainers who work with athletes provide instructions regarding proper breathing techniques during weight lifting. In addition to the case discussion, this article reviews spontaneous pneumomediastinum and epidural pneumatosis.

A case report of rheumatoid arthritis complicated with rapidly progressive interstitial pneumonia, multiple bullae and pneumomediastinum, which was successfully treated with tacrolimus.

A 64-year-old woman had been treated with prednisolone (PSL) for interstitial pneumonia (IP) of unknown origin since 1988. The IP progressed gradually, however, and home oxygen therapy was instituted in 1993. In 2002, persistent arthritis of the hands appeared and diagnosis of rheumatoid arthritis (RA) was finally established based on radiological and pathological findings. Salazosulfapyridine was given with only partial effect. On October 2002, she was hospitalized because of back pain followed by dyspnea. Chest X-ray revealed multiple giant bullae on bilateral upper lung fields, accompanied by deterioration of IP. Methyl-prednisolone pulse therapy followed by 30 mg/day of PSL was instituted and the bullae were diminished with gradual improvement of IP and synovitis. On the 55th hospital day, she complained of chest oppression, and chest X-ray revealed a complication of pneumomediastinum. Since IP was still active and serum KL-6 remained high, 3 mg/day of tacrolimus was added to control IP further and to reduce the dosage of PSL which was recognized as one of the aggravation factors of pneumomediastinum. As a result, pneumomediastinum disappeared gradually along with amelioration of IP. PSL was successfully tapered to 15 mg/day by the 87th hospital day and the patient was discharged. Although the efficacy of tacrolimus on IP complicated with polymyositis / dermatomyositis and other autoimmune diseases has been reported, this case first suggests its efficacy on IP associated with RA.

Pneumomediastinum caused by occult paraquat poisoning: Case report.

RATIONALE: Paraquat is a widely applied contact herbicide that is highly poisonous. About 20% of patients with paraquat poisoning develop pneumomediastinum as a complication with a mortality rate of almost 100%. PATIENT CONCERNS: A 15-year-old boy presented with a 1-month history of retrosternal chest pain with no obvious cause. High-resolution computed tomography showed pneumomediastinum. DIAGNOSES: After all likely causes of pneumomediastinum were eliminated, the diagnosis of occult paraquat poisoning was made when serum paraquat concentration was revealed at 467.40 ng/mL, despite the patient's denial of ingestion or contact. INTERVENTIONS: Hemoperfusion, intravenous glucocorticoid, and ulinastatin was administered for 3 days with other routine treatment against paraquat poisoning. The serum paraquat concentration decreased to zero. OUTCOMES: Despite the general high mortality and poor prognosis of paraquat poisoning, the patient recovered and was completely asymptomatic at his 3-month follow-up. LESSONS: Paraquat poisoning should be suspected as a differential diagnosis when patients present with pneumomediastinum without recognizable cause.