|Year : 2015 | Volume
| Issue : 1 | Page : 30-32
Spontaneous subcutaneous emphysema and pneumomediastinum in a child with bronchial asthma
Bindu T Nair, Sajith Surendran, Tarikjot Singh Brar
Department of Paediatrics, Army College of Medical Sciences, Delhi Cantt, New Delhi, India
|Date of Web Publication||21-Aug-2015|
Bindu T Nair
Department of Paediatrics, Army College of Medical Sciences, Delhi Cantt - 110 010, New Delhi
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Massive spontaneous subcutaneous emphysema is rare in the absence of trauma. An 11-year-old male child, a known case of poorly controlled bronchial asthma presented with sudden onset of respiratory distress after a sudden bout of cough. He also had painful swelling of the neck, chest, abdomen, and upper limbs. On clinical and radiological examination, he was found to have massive subcutaneous emphysema. A gastrografin study was done which showed no esophageal perforation. Computed tomography of his neck and thorax demonstrated pneumomediastinum with no lung or pleural pathology. Management was conservative with supplemental oxygen, nebulized bronchodilators, corticosteroids, and intravenous antibiotics. Child improved symptomatically without any surgical intervention and was discharged after 7 days.
Keywords: Bronchial asthma, pneumomediastinum, subcutaneous emphysema
|How to cite this article:|
Nair BT, Surendran S, Brar TS. Spontaneous subcutaneous emphysema and pneumomediastinum in a child with bronchial asthma. Ann Nigerian Med 2015;9:30-2
|How to cite this URL:|
Nair BT, Surendran S, Brar TS. Spontaneous subcutaneous emphysema and pneumomediastinum in a child with bronchial asthma. Ann Nigerian Med [serial online] 2015 [cited 2021 Apr 18];9:30-2. Available from: https://www.anmjournal.com/text.asp?2015/9/1/30/163333
| Introduction|| |
Spontaneous subcutaneous emphysema though rare has an association with whooping-cough, measles, diphtheritic laryngeal obstruction, and bronchopneumonia.  Rarely bronchial asthma has been attributed to the production of subcutaneous emphysema. Forceful paroxysms of coughing in asthma can lead to pneumomediastinum and subcutaneous emphysema. The first definitive case of spontaneous subcutaneous emphysema in a child due to asthma was reported in 1850.  Thereafter, even though there are case reports of subcutaneous emphysema and pneumomediastinum in adults with chronic obstructive pulmonary disease and asthma, there are no reports in children.
| Case Report|| |
An 11-year-old male child presented to the Pediatric Emergency Department of our hospital with a history of cough for 2 days and increasing breathing difficulty for 6 h. He also complained of swelling and pain over his neck, chest, abdomen, and upper limbs for the past 6 h). He gave a history of having had an upper respiratory tract infection for past 1-week. He also gave a history of frequent, forceful coughing episodes. There was no history suggestive of foreign body aspiration. He was a known case of bronchial asthma since the age of 5 years. He had been admitted earlier on a few occasions for acute exacerbations of asthma. He had been on metered dose inhalers of bronchodilators and corticosteroids off and on since the age of 5 years.
At the time of presentation in the Emergency Department, he had a respiratory rate of 40 breaths per minute, heart rate of 120 beats per minute, blood pressure of 110/76 mmHg. His Spo 2 was 86% in room air which increased to 94% with oxygen supplementation. He was noted to have subcutaneous swelling and crepitus over the neck from below the mandible, upper anterior chest region, abdomen and both upper limbs. Chest auscultation revealed polyphonic rhonchi bilaterally with prolonged expiratory phase. Clinical examination of other systems were essentially normal.
Laboratory tests showed a white cell count of 9.2 × 109/L (normal range: 4-10 × 109/L) with polymorphs of 70%, C-reactive protein was 2 g/dL (normal range <5 g/dL) with urea, creatinine, sodium, potassium, and liver enzymes all within normal limits. Gram-stain and Ziehl-Neelsen stain of sputum for organism was negative. Culture of sputum also did not show any growth for bacteria or mycobacterium tuberculosis.
The chest radiograph showed low set hemidiaphragms and hyperinflation suggesting the known asthmatic state. There was a linear shadow of lucency on both borders of the heart [Figure 1]. There were areas of lucency in the subcutaneous tissue of the neck and both axillary regions and sides of the chest wall. There was no free air under his diaphragm. CT scan of the chest demonstrated marked subcutaneous emphysema with pneumomediastinum [Figure 2]. The CT scan did not show any pneumonia, pneumothorax, perforated viscous or any fluid collections.
|Figure 1: Computed tomography scan showing subcutaneous emphysema and pneumomediastinum (shown by arrows)|
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On the basis of the history, examination and radiological findings, a diagnosis of acute exacerbation of bronchial asthma with pneumomediastinum and subcutaneous emphysema was made. He was admitted in Pediatric Intensive Care Unit and was managed with salbutamol nebulization, parenteral hydrocortisone and humidified oxygen by nasal prongs. He was also started on intravenous antibiotics (injection piptaz and clindamycin) empirically which was discontinued after 5 days. After about 72 h of admission, he made remarkable improvement with resolution of the initial symptoms of respiratory distress. He was discharged after 7 days of admission on long acting bronchodilators and steroid inhalers. On follow-up, the child has remained stable.
| Consent|| |
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of the journal.
| Discussion|| |
Spontaneous pneumomediastinum associated with subcutaneous emphysema is a rare condition. It is also known as Hamman's syndrome or Macklin's syndrome. It has to be distinguished from more sinister pathology or entities like Boerhaave syndrome.  The clinical course of Hamman syndrome is usually benign and self-limiting. Therefore, management of this condition is often conservative.  Subcutaneous emphysema occurs when air is trapped under the skin. Both penetrating and blunt trauma to the respiratory or gastrointestinal tract can cause subcutaneous emphysema. 
Subcutaneous emphysema is also a rare complication of acute severe asthma. It may occur in association with spontaneous pneumomediastinum, pneumopericardium or pneumoperitoneum.  Spontaneous pneumomediastinum occurs as a result of sudden rise in intra-alveolar pressure (asthma, vasalva manoeuvre, cough, emesis, and other forms of barotrauma) which results in the rupture of marginal alveoli. Air forced into the interstitial tissues around the pulmonary vasculature travels back toward the hilum, leading to pneumomediastinum. This air eventually tracts into the soft tissue of the neck, face, and chest wall. 
Subcutaneous emphysema is usually a self-limiting condition if not massive. If the subcutaneous emphysema is massive, then it can rarely lead to airway and cardiovascular compromise around the thoracic inlet by compression of structures in the neck.  There is a paucity of case reports that describe such conditions. The case we report here is rare as the patient gave no history of trauma or any systemic illness like pneumonia. Our patient was well except for a history of asthma. Perforation of a cervical viscous has to be suspected in the absence of any signs of soft tissue infection, pulmonary disease or trauma in a patient with no relevant medical history. Recognition of this condition requires a high index of suspicion and may be difficult unless kept in mind.
The usual clue to diagnosis of subcutaneous emphysema is the presence of subcutaneous air in the neck, chest, upper limbs, and over the abdomen, as in our case. The diagnosis is confirmed by a chest radiograph which will show multiple thin, lucent streaks outlining mediastinal structures extending into the neck or chest wall. Computed tomography can be done if findings on plain radiograph are inconclusive.  Recently, ultrasound has been used to diagnose pneumomediastinum as a bedside procedure especially in critically ill patients. 
Management of spontaneous emphysema is largely conservative. However, administration of high concentration of oxygen may enhance faster resorption of air from extrapulmonary tissues. Needle aspiration and/or surgical decompression may be useful if mediastinal structures are getting compressed. , The time taken for resolution varies greatly depending on the amount of subcutaneous emphysema, adequacy of control of escaping air, and method of treatment.
With observation alone, it can take several weeks for significant subcutaneous emphysema to resolve as air is gradually reabsorbed by soft tissues.  A "blowhole" incision is widely used by some surgeons to allow the release of trapped subcutaneous air. It also helps to minimize further progression of air dissection into the face and neck. 
This case report emphasises that though spontaneous subcutaneous emphysema and pneumomediastinum is a known clinical entity, it is not commonly seen. Extra-pulmonary extravasations of air can be seen as subcutaneous emphysema and pneumomediastinum very rarely in cases of acute asthma. Though rare, this is a very important complication of acute exacerbation of bronchial asthma and is amenable to conservative management. This case report makes us aware of this rare entity.
A high index of suspicion can recognize this condition and appropriate management given in the emergency department. The outcome of pneumomediastinum varies from a benign course to a malignant and progressive one.  When the mediastinal pressure interferes with the circulation and lung expansion, it can have a fatal outcome. Early recognition and management of the underlying cause along with good supportive care helps in resolution of subcutaneous emphysema and pneumomediastinum.
| References|| |
Maunder RJ, Pierson DJ, Hudson LD. Subcutaneous and mediastinal emphysema. Pathophysiology, diagnosis, and management. Arch Intern Med 1984;144:1447-53.
McGovern JP, Ozkaragoz K, Roett K, Haywood TJ, Hensel AE Jr. Mediastinal and subcutaneous emphysema complicating atopic asthma in infants and children. Review of the literature and report of occurrence in an infant two years of age. Pediatrics 1961;27:951-60.
Chalumeau M, Le Clainche L, Sayeg N, Sannier N, Michel JL, Marianowski R, et al.
Spontaneous pneumomediastinum in children. Pediatr Pulmonol 2001;31:67-75.
Damore DT, Dayan PS. Medical causes of pneumomediastinum in children. Clin Pediatr (Phila) 2001;40:87-91.
Chapdelaine J, Beaunoyer M, Daigneault P, Bérubé D, Bütter A, Ouimet A, et al.
Spontaneous pneumomediastinum: Are we overinvestigating? J Pediatr Surg 2004;39:681-4.
Shenfine J, Dresner SM, Vishwanath Y, Hayes N, Griffin SM. Management of spontaneous rupture of the oesophagus Br J Surg 2000;87:362-73.
Kogan I, Celli BR. Pneumomediastinum in a 63-year-old woman with asthma exacerbation. Chest 2000;117:1778-81.
Kaneki T, Kubo K, Kawashima A, Koizumi T, Sekiguchi M, Sone S. Spontaneous pneumomediastinum in 33 patients: Yield of chest computed tomography for the diagnosis of the mild type. Respiration 2000;67:408-11.
Testa A, Candelli M, Pignataro G, Costantini AM, Pirronti T, Silveri NG. Sonographic detection of spontaneous pneumomediastinum. J Ultrasound Med 2008;27:1507-9.
Newcomb AE, Clarke CP. Spontaneous pneumomediastinum: A benign curiosity or a significant problem? Chest 2005;128:3298-302.
Dontu VS, Kramer D. Spontaneous pneumothorax, pneumomediastinum, and epidural emphysema presenting as neck pain suspicious for meningitis. Pediatr Emerg Care 2007;23:469-71.
Williams DJ, Jaggar SI, Morgan CJ. Upper airway obstruction as a result of massive subcutaneous emphysema following accidental removal of an intercostal drain. Br J Anaesth 2005;94:390-2.
Caceres M, Ali SZ, Braud R, Weiman D, Garrett HE Jr. Spontaneous pneumomediastinum: A comparative study and review of the literature. Ann Thorac Surg 2008;86:962-6.
Perna V, Vilà E, Guelbenzu JJ, Amat I. Pneumomediastinum: Is this really a benign entity? When it can be considered as spontaneous? Our experience in 47 adult patients. Eur J Cardiothorac Surg 2010;37:573-5.
[Figure 1], [Figure 2]