대한응급의학회지 제 27 권 제 3 호 � 증례� Volume 27, Number 3, June, 2016

Medical A Case of Unknown Cause of Subcutaneous Emphysema Presented by Generalized

Sam Beom Lee, M.D.*, Jong Ha Kim, M.D., Sin Youl Park, M.D., Byung Soo Do, M.D.

Department of , Yeungnam University College of Medicine, Daegu, Korea

Subcutaneous emphysema and are above symptom was suddenly developed and accompa- commonly derived from trauma or of respiratory or nied by resting dyspnea and then progressively aggravat- gastrointestinal tracts, but occasionally the origin of air was ed, but no other symptoms such as , abdominal not determined at evaluation. We report on a case of pain, nausea and vomiting was developed. severe subcutaneous emphysema detected using simple He has been suffered from hypertension and myasthe- X-ray films in the emergency department, which extended nia gravis, but no history of lung and heart diseases or to almost all of the bodies, with a review of the literature. recent trauma or injury at chest. On physical examination, alert mentality and stable Key Words: Air, Origin, Pneumomediastinum vital signs were showed, but, his face, eyelid, neck and upper chest was showed edematous, swollen like bal- loon, even to abdominal wall and thigh after taking off Introduction his clothes. The sound of was heard during touching his body and the breathing sound was slightly Subcutaneous emphysema is defined as the presence of decreased on both lung fields. air in the subcutaneous tissues of the body. It can result After initial , simple chest X-ray was from puncture of parts of the respiratory or gastrointesti- taken, it showed large amount of subcutaneous emphyse- nal tracts, particularly in the chest and neck. The air may ma on his neck extending to both axilla, chest wall and become trapped as a result of or blunt abdominal wall (Fig. 1). trauma and subcutaneous emphysema can be caused by The whole body computed tomography was taken to medical procedures and medical conditions. But, sponta- detect the range and cause of subcutaneous emphysema, neous subcutaneous emphysema when the cause is not it showed mediastinal emphysema around the heart, clear used to be occasionally reported. Subcutaneous esophagus and aorta, and even neck and face, retroperi- emphysema is not typically dangerous in itself, but we toneum, lower , pelvic cavity and both thigh cir- report a case, which showed remarkable X-ray finding cumferentially (Fig. 2). accompanied severely extended physical finding at The collar incisions at both infraclavicular areas were almost of the bodies in the emergency department1,2). promptly done and squeezing the head and neck to downward, the abdomen and to upward was done Case Report to remove the air beneath the skin out to the body. The esophagogastroscopy and brochoscopy did not A 54-year-old male patient was admitted to emergency find the detectable cause of any ulceration or perforation department due to generalized edema. At 5-6 hours ago, of the esophagus, upper and the tra- cheobronchial tree. 책임저자: 이 삼 범 We decided to observe the patient closely without fur- 대구광역시 남구 현충로 170 ther diagnostic tests and specific treatment because his 영남대학교 의과대학 응급의학교실 condition was comfortable and vital signs were stable. Tel: 053) 620-3193, Fax: 053) 628-7106 E-mail: [email protected] Daily checked follow-up chest X-ray films during 접수일: 2016년 4월 5일, 1차 교정일: 2016년 4월 7일 admission showed rapidly resolving status of soft tissue 게재승인일: 2016년 5월 3일 284 Sam Beom Lee et al.: A Case of Unknown Cause of Subcutaneous Emphysema Presented by Generalized Edema / 285

emphysemas and after then 1 week later he was improved and discharged after general treatment with some residual lesion, but long term follow-up chest X- ray films showed no visible residual lesions after dis- charge (Fig. 3).

Discussion

Subcutaneous emphysema is when gas or air is in the layer under the skin. Subcutaneous refers to the tissue beneath the skin, and emphysema refers to trapped air. It is sometimes called tissue emphysema, or Sub Q air. Since the air generally comes from the chest cavity, sub- cutaneous emphysema usually occurs on the chest, neck Fig. 1. Initial chest x-ray film shows extensive soft tissue and face, where it is able to travel from the chest cavity emphysema on the neck, upper chest wall and along the fascia1). abdominal wall and also shows mediastinal emphy- Subcutaneous emphysema has a characteristic crack- sema along the diaphragm and the descending aorta.

A

Fig. 2. (A) Serial computed tomography (CT) scan shows soft tissue emphysema and also medi- astinal, peritoneal and retroperitoneal emphysema on the whole bodies. (B) Coronal CT scan shows extensive soft tissue emphysema and also mediastinal, peritoneal B and retroperitoneal emphysema. 286 / 대한응급의학회지: 제 27 권 제 3 호 2016 ling feel to the touch, a sensation that has been described emphysema can be uncomfortable and may interfere as similar to touching Rice Krispies; this sensation of air with breathing, and is often treated by removing air from under the skin is known as subcutaneous crepitation3). the tissues, for example by using large bore needles, skin Numerous etiologies of subcutaneous emphysema incisions (collar incision) or subcutaneous catheteriza- have been described1). Subcutaneous emphysema can tion6). result from puncture of parts of the respiratory or gas- Pneumomediastinum may be defined as the presence trointestinal systems. Particularly in the chest and neck, of free air or gas in the mediastinal structures. Air in the air may become trapped as a result of penetrating trauma human body is usually confined to the respiratory or the (e.g., gunshot wounds or stab wounds) or . gastrointestinal tract, but when the free air enters the (e.g., gas ) can cause gas to be trapped it’s termed as pneumomediastinum or in the subcutaneous tissues. Subcutaneous emphysema mediastinal emphysema. It’s an uncommon, but impor- can be caused by medical procedures and medical condi- tant condition found in healthy young adults and children tions that cause the pressure in the alveoli of the lung to presenting with chest pain and , which be higher than that in the tissues outside of them4). may involve the neck, face, chest, abdomen, legs and Its most common causes are and a chest arms. Pneumomediastinum commonly results from alve- tube that has become occluded by a blood clot or fibri- olar rupture, in which case air tracks along interstitial nous material. It can also occur spontaneously due to and vascular supporting tissues till it reaches the medi- rupture of the alveoli with dramatic presentation. The astinum1-2). term spontaneous subcutaneous emphysema is used Radiographically pneumomediastinum is manifested when the cause is not clear5). by lucent streaks or bubbles of gas in the mediastinal soft Subcutaneous emphysema is not typically dangerous tissues outlining mediastinal structures, and mediastinal in itself, however it can be a symptom of very dangerous air between the heart and the diaphragm results in underlying conditions, such as pneumothorax. Although “continuous diaphragm sign” on the frontal chest radi- the underlying conditions require treatment, subcuta- ograph7,8). neous emphysema usually does not; small amounts of air In this case we can find the continuous air shadow are reabsorbed by the body. However, subcutaneous along the diaphragm between the heart and the

Fig. 3. Serial follow-up chest X-ray films show rapidly resolving status of soft tissue emphysemas. Sam Beom Lee et al.: A Case of Unknown Cause of Subcutaneous Emphysema Presented by Generalized Edema / 287 diaphragm, the most typically visible on Day 2 film. Sometimes increased intrathoracic pressure during REFERENCES cough and straining is known to precipitate the condition as noted in common respiratory , , bron- 01. Available at: https://en.wikipedia.org/wiki/Subcutaneous chitis, foreign body choking, parturition and volutrauma _emphysema. Accessed March 4, 2016. during mechanical ventilation9-12). 02. Byun CS, Choi JH, Hwang JJ, Kim DH, Cho HM, Seok It can occur with blunt thoracic trauma, esophageal JP. Vacuum-assisted closure therapy as an alternative treatment of subcutaneous emphysema. Korean J Thorac rupture during vomiting or esophagoscopy, therapeutic Cardiovasc Surg. 2013;46:383-7. bronchoscopy, acute lung injury due to Mendelson’s 03. Lefor A. Critical Care on Call. New York: Lange Medical syndrome and mediastinitis by gas forming organisms Books/McGraw-Hill Medical; 2002. p.238-40. and also reported in cases of pulmonary tuberculosis, 04. Maunder RJ, Pierson DJ, Hudson LD. Subcutaneous and pneumocystic carinii pneumonia and ecstasy abuse4,13). mediastinal emphysema. Pathophysiology, diagnosis, and No preexisting cause may be apparent in some cases. management. Arch Intern Med. 1984;144:1447-53. In this case we could not find the detectable cause of any 05. Parker GS, Mosborg DA, Foley RW, Stiernberg CM. ulceration or perforation of the esophagus, upper gas- Spontaneous cervical and mediastinal emphysema. trointestinal tract and the tracheobronchial tree by the Laryngoscope. 1990;100:938-40. esophagogastroscopy and bronchoscopy. 06. Brooks DC. Current review of minimally invasive surgery. Philadelphia: Current Medicine. 1998. p.36. As in such there is no specific treatment of this condi- 07. CM Adya, Vinay Maurya, VD Charan, AKS Bairaria. tion apart from the fact that an incision, so called “collar Spontaneous pneumomediastinum in an adolescent. Med J incision” or are placed in the skin at both infra- Armed Forces India. 2005;61:192-4. clavicular areas in order to release the air confined in the 08. Levin B. The continuous diaphragm sign: a newly-recog- chest cavity. In this case prompt collar incision and nized sign of pneumomediastinum. Clin Radiol. 1973;24: squeezing the air at both infraclavicular areas was done 337-8. because of the severely extended lesion with the patient’s 09. Romero KJ, Trujillo MH. Spontaneous pneumomedi- uncomfortable symptom. While not in severe cases, sup- astinum and subcutaneous emphysema in asthma exacer- plemental oxygen is only provided via masks to combat bation: The Macklin effect. Heart Lung. 2010;39:444-7. the shortness of breath as felt by the patient. But, in 10. Findlay CA, Morrissey S, Paton JY. Subcutaneous emphy- sema secondary to foreign-body aspiration. Pediatr severe cases which can be massive and severely compro- Pulmonol. 2003;36:81-2. mised the airway, negative pressure wound therapy as 11. McGregor A, Ogwu C, Uppal T, Wong MG. Spontaneous known as vacuum-assisted closure therapy is effective subcutaneous emphysema and pneumomediastinum during treatment modality and surgical repair is required for second stage of labour. BMJ Case Rep. 2011; 2011: bcr invasive method2,7,9,14,15). 0420114067. We would like to present this case of severe subcuta- 12. Raymond LW. Pulmonary and related events neous emphysema detected by simple X-ray films in the in divers. Chest. 1995;107:1648-52. emergency department, because even though it is not 13. MacLennan FM. Maternal mortality from Mendelson’s typically dangerous in itself, but it shows remarkable syndrome: an explanation? Lancet. 1986;15:1(8481):587- simple X-ray film and also accompanied severely 9. 14. Cerfolio RJ, Bryant AS, Maniscalco LM. Management of extended physical findings for the purpose of instructing subcutaneous emphysema after pulmonary resection. Ann students and young doctors. Thorac Surg. 2008;85:1759-63. 15. Schintler MV. Negative pressure therapy: theory and prac- tice. Diabetes Metab Res Rev. 2012;28 Suppl 1:72-7.