- Email: [email protected]
Spontaneous Pneumomediastinum After Electronic Cigarette Use
Accepted Manuscript Spontaneous Pneumomediastinum Following Electronic Cigarette Use Rita Daniela Marasco, MD, Domenico Loizzi, MD, Nicoletta Pia Ardò, MD, Fabio Nicola Fatone, MD, Francesco Sollitto, MD PII:
S0003-4975(18)30058-4
DOI:
10.1016/j.athoracsur.2017.12.037
Reference:
ATS 31287
To appear in:
The Annals of Thoracic Surgery
Received Date: 21 November 2017 Accepted Date: 26 December 2017
Please cite this article as: Marasco RD, Loizzi D, Ardò NP, Fatone FN, Sollitto F, Spontaneous Pneumomediastinum Following Electronic Cigarette Use, The Annals of Thoracic Surgery (2018), doi: 10.1016/j.athoracsur.2017.12.037. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Spontaneous Pneumomediastinum Following Electronic Cigarette Use Running Head: A rare case of Pneumomediastinum
Rita Daniela Marasco, MD, Domenico Loizzi, MD, Nicoletta Pia Ardò, MD, Fabio Nicola Fatone,
RI PT
MD, and Francesco Sollitto, MD
General Thoracic Surgery Department, Azienda Ospedaliero-Universitaria “Ospedali Riuniti”,
SC
Foggia
M AN U
Corresponding Author: Rita Daniela Marasco, MD, General Thoracic Surgery Department, Azienda Ospedaliero-Universitaria “Ospedali Riuniti”, Via L. Pinto, 71122, Foggia (Italy). Email: [email protected].
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WORD COUNT: 1254
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Keywords: Electronic cigarette, Pneumomediastinum, Subcutaneous emphysema
1
ACCEPTED MANUSCRIPT ABSTRACT Spontaneous pneumomediastinum (SPM) is an uncommon condition typically occurring in young males presenting with pleuritic pain, dyspnea and subcutaneous emphysema. We reported an exceptional case of spontaneous pneumomediastinum following electronic cigarette use in an
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EP
TE D
M AN U
SC
RI PT
otherwise healthy young man.
2
ACCEPTED MANUSCRIPT Pneumomediastinum , or mediastinal emphysema, is a rare entity definined as the presence of free air - almost invariably originating from the alveolar space or the conducting airways - within the mediastinum1. We herein describe a case of spontaneous pneumomediastinum (SPM) occurring in
RI PT
a healthy young man after his first electronic cigarette smoking experience.
A 17-year-old white male was referred to the emergency department for dyspnea and disphagia. His past medical history was unremarkable, he had no tobacco exposure and took no medications. At
SC
the admission, the patient referred a moderate shortness of breath associated with painful swallowing; he denied preceding trauma, heavy lifting, coughing or recent vomiting, but upon
M AN U
further questioning, he admitted to have taken a deep inhalation from an electronic cigarette just before the symptoms occurred. Vital signs were normal. General examination was unremarkable, except for soft crepitation on the left side of his neck.
Standard thoracic radiography revealed streaking lucencies outlining mediastinal contours, such as
TE D
subcutaneous emphysema (Fig.1). Chest computed tomography scan showed the so called “ring sign” (due to air surrounding the pulmonary artery and its branches) and double bronchial wall (Fig. 2).
EP
The patient was hospitalized for observation and the clinical course was uneventful; chest X-ray within 48 hours did not detect any progression or complications. He was discharged on the 2nd day
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of hospitalization and referred to the General Thoracic Surgery Department Outpatient Clinic. During a two months follow up there was no recurrence of pneumomediastinum. The patient stated he had stopped vaping.
COMMENT First described by Laennec2 in 1819, pneumomediastinum can be classified as spontaneous (or primary) and secondary3.
Secondary pneumomediastinum may result from either blunt or
penetrating thoracic trauma, endobronchial or esophageal procedures, head and neck surgery, 3
ACCEPTED MANUSCRIPT hollow organ perforation, mechanical ventilation or other invasive procedures. Spontaneous pneumomediastinum (SPM) is a rare condition, more frequent in males on 2nd-4th decades of life, which rarely leads to clinically significant complications. Even though the true incidence is not known, the reviewed series report an incidence of 1:7000-1:45.000 hospital admissions4.
lung, pleural space) or extratoracic (head, neck, peritoneum).
RI PT
The potential source of mediastinal air can be intrathoracic (trachea and major bronchi, esophagus,
Pathophysiology of spontaneous pneumomediastinum is based on alveolar rupture due to an
SC
increase of intrathoracic pressure, followed by air tracks through the peribronchial vascular sheaths into the mediasinum, toward the hilum of the lung (Macklin effect)5. Furthermore, the mediastinum
M AN U
communicates with the submandibular and retropharyngeal spaces, and also with the retroperitoneum through a tissue plane extending from the sternocostal attachment to the diaphragm, as well as through periaortic and periesophageal fascial planes; as a result, the air may dissect from the mediastinum through these tissue planes, causing pneumopericardium,
TE D
pneumothorax, pneumoperitoneum or pneumoretroperitoneum 6. The precipitating factors - events closely related to the development of SPM and generally due to an increase in intrathoracic pressure - include: Valsalva manoeuvre, emesis, cough, asthma exacerbation, defecation, physical
EP
exercise, childbirth, airway infections, sepsis-induced acute respiratory distress syndrome, inhaled drug use (either continuous and occasional abuse), chocking. The predisposing factors - previous
AC C
conditions favouring pneumomediastinum – are: asthma, interstitial and other lung diseases, corticosteroids, tobacco, inhaled drug use, inhalation of irritants. A classical clinical triad has been described, and consists of thoracic pain (usually retrosternal and pleuritic), subcutaneous emphysema (which is the most common sign) and dyspnea; less common symptoms are cough, fever, dysphonia, odynophagia and dysphagia. The Hamman sign (detected in 10-20% of cases) is pathognomonic and characterized by systolic crackles in the left sternal border, best heard in left lateral decubitus, described as “rubbing balloons”. Leukocytosis and/or neutrophilia are common, sometimes associated with low grade fever. 4
ACCEPTED MANUSCRIPT Chest radiography is the standard diagnostic procedure, showing a double line outlining the mediastinum. Diagnosis can be confirmed by chest computed tomography. Oral contrast study or bronchoscopy should be performed if esophageal and tracheobronchial tree rupture is suspected. Once confirmed the diagnosis, patient should be admitted for monitoring and treatment, consisting
RI PT
of avoidance of the trigger factors, oxygen and bed rest. Some Authors advocate antibiotic prophylaxis for mediastinitis. A chest tube is recommended in case of pneumothorax or hypertensive pneumomediastinum. Recurrence of pneumomediastinum is exetremely rare.
SC
Complications are uncommon as well, and include tension pneumomediastinum, eventuality in which elevated mediastinal pressure leads to diminished cardiac output because of direct cardiac
M AN U
compression or reduced venous return, hemodynamic and respiratory compromise. Airway compression may occur in presence of extensive subcutaneous and mediastinal emphysema. In Boerhaave syndrome (esophageal rupture following vomiting) mortality can be as high as 50-70% 7 Mediastinitis is also a serious complication, and morbidity and mortality are related to coexisting
TE D
illness.
There are no reports of mortality directly related to pneumomediastinum: spontaneous PM is a selflimited condition that rarely produces significant or life-threatening symptoms.
EP
In the present paper, we described an exceptional case of spontaneous pneumomediastinum occurring after inhalation from an electronic cigarette in a healthy young man.
AC C
The increasing use of electronic nicotine delivery systems, such as electronic cigarettes (especially among young never smokers) implies the exposure by users to the aerosols, that may contain propylene glycol and glycerol (whose aerosolised form produce throat irritation and dry cough), flavourings, other chemicals and nicotine 8. Aerosol exposure can be associated with a decrease in fractional exhaled nitric oxide, increase in respiratory impedance and respiratory flow and may also result in respiratory function impairment. Scientific evidence regarding the human health effects of electronic cigarettes is still limited, and studies evaluating whether they are less harmful than cigarettes are inconclusive. 5
ACCEPTED MANUSCRIPT As previously described, pneumomediastinum can rarely occur as a consequence of illicit and recreational drug use; nonetheless, to our knowledge, this is the first case of a spontaneous
AC C
EP
TE D
M AN U
SC
RI PT
pneumomediastinum occurring after the inhalation from an electronic cigarette.
6
ACCEPTED MANUSCRIPT FIGURE LEGENDS Figure 1: Chest radiography.
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EP
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M AN U
SC
RI PT
Figure 2: Chest Computed Tomography showing pneumomediastinum.
7
ACCEPTED MANUSCRIPT REFERENCES 1. A. E. Newcomb, C. P. Clarke. Spontaneous Pneumomediastinum: A Benign Curiosity or a Significant Problem?. CHEST 2005; 128:3298–3302;
RI PT
2. J.B. Jougon, M. Ballester, Delcambre F., T. MacBride et al. Assessment of spontaneous pneumomediastinum: experience with 12 patients. Ann Thorac Surg 75:2003 (1711-1714); 3. E. Panacek, A. J. Singer, B. W. Sherman, W.F. Rutherford. Spontaneous pneumomediastinum: Clinical and natural history. Ann Emerg Med 1992;1222-1227;
SC
4. R. Jones, B. Willis, J.N. Johneson.Spontaneous pneumomediastinum.West J Emerg Med 2008 Nov; 9 (4):217-218;
M AN U
5. S.Murayama, S. Gibo. Spontaneous pneumomediastinum and Macklin effect: Overview and appearance on computed tomography. World J Radiol 2014 Nov 28; 6(11): 850–854. 6. V. Kouritas, K. Papagiannopulos, G. Lazaridis. Pneumomediastinum. J Thorac Dis. 2015 Feb; 7(Suppl1):S44-S49; 7. F. Al-Mufarrej, J. Badar, F. Gharagozloc. Spontaneous pneumomediastinum: diagnostic and therapeutic interventions..J Cardiothorac Surg,
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8. L. F. Chun, F. Moazed, C. S. Calfel, M. A. Matthay, J. E. Gotts. Pulmonary toxicity of electronic cigarettes: more doubts than certainties. Am J Physiol Lung Cell Mol Physiol 2017 Nov 1; 313(5):L964-L965;
8
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ACCEPTED MANUSCRIPT
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SC
RI PT
ACCEPTED MANUSCRIPT
S0003-4975(18)30058-4
DOI:
10.1016/j.athoracsur.2017.12.037
Reference:
ATS 31287
To appear in:
The Annals of Thoracic Surgery
Received Date: 21 November 2017 Accepted Date: 26 December 2017
Please cite this article as: Marasco RD, Loizzi D, Ardò NP, Fatone FN, Sollitto F, Spontaneous Pneumomediastinum Following Electronic Cigarette Use, The Annals of Thoracic Surgery (2018), doi: 10.1016/j.athoracsur.2017.12.037. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
ACCEPTED MANUSCRIPT Spontaneous Pneumomediastinum Following Electronic Cigarette Use Running Head: A rare case of Pneumomediastinum
Rita Daniela Marasco, MD, Domenico Loizzi, MD, Nicoletta Pia Ardò, MD, Fabio Nicola Fatone,
RI PT
MD, and Francesco Sollitto, MD
General Thoracic Surgery Department, Azienda Ospedaliero-Universitaria “Ospedali Riuniti”,
SC
Foggia
M AN U
Corresponding Author: Rita Daniela Marasco, MD, General Thoracic Surgery Department, Azienda Ospedaliero-Universitaria “Ospedali Riuniti”, Via L. Pinto, 71122, Foggia (Italy). Email: [email protected].
AC C
EP
WORD COUNT: 1254
TE D
Keywords: Electronic cigarette, Pneumomediastinum, Subcutaneous emphysema
1
ACCEPTED MANUSCRIPT ABSTRACT Spontaneous pneumomediastinum (SPM) is an uncommon condition typically occurring in young males presenting with pleuritic pain, dyspnea and subcutaneous emphysema. We reported an exceptional case of spontaneous pneumomediastinum following electronic cigarette use in an
AC C
EP
TE D
M AN U
SC
RI PT
otherwise healthy young man.
2
ACCEPTED MANUSCRIPT Pneumomediastinum , or mediastinal emphysema, is a rare entity definined as the presence of free air - almost invariably originating from the alveolar space or the conducting airways - within the mediastinum1. We herein describe a case of spontaneous pneumomediastinum (SPM) occurring in
RI PT
a healthy young man after his first electronic cigarette smoking experience.
A 17-year-old white male was referred to the emergency department for dyspnea and disphagia. His past medical history was unremarkable, he had no tobacco exposure and took no medications. At
SC
the admission, the patient referred a moderate shortness of breath associated with painful swallowing; he denied preceding trauma, heavy lifting, coughing or recent vomiting, but upon
M AN U
further questioning, he admitted to have taken a deep inhalation from an electronic cigarette just before the symptoms occurred. Vital signs were normal. General examination was unremarkable, except for soft crepitation on the left side of his neck.
Standard thoracic radiography revealed streaking lucencies outlining mediastinal contours, such as
TE D
subcutaneous emphysema (Fig.1). Chest computed tomography scan showed the so called “ring sign” (due to air surrounding the pulmonary artery and its branches) and double bronchial wall (Fig. 2).
EP
The patient was hospitalized for observation and the clinical course was uneventful; chest X-ray within 48 hours did not detect any progression or complications. He was discharged on the 2nd day
AC C
of hospitalization and referred to the General Thoracic Surgery Department Outpatient Clinic. During a two months follow up there was no recurrence of pneumomediastinum. The patient stated he had stopped vaping.
COMMENT First described by Laennec2 in 1819, pneumomediastinum can be classified as spontaneous (or primary) and secondary3.
Secondary pneumomediastinum may result from either blunt or
penetrating thoracic trauma, endobronchial or esophageal procedures, head and neck surgery, 3
ACCEPTED MANUSCRIPT hollow organ perforation, mechanical ventilation or other invasive procedures. Spontaneous pneumomediastinum (SPM) is a rare condition, more frequent in males on 2nd-4th decades of life, which rarely leads to clinically significant complications. Even though the true incidence is not known, the reviewed series report an incidence of 1:7000-1:45.000 hospital admissions4.
lung, pleural space) or extratoracic (head, neck, peritoneum).
RI PT
The potential source of mediastinal air can be intrathoracic (trachea and major bronchi, esophagus,
Pathophysiology of spontaneous pneumomediastinum is based on alveolar rupture due to an
SC
increase of intrathoracic pressure, followed by air tracks through the peribronchial vascular sheaths into the mediasinum, toward the hilum of the lung (Macklin effect)5. Furthermore, the mediastinum
M AN U
communicates with the submandibular and retropharyngeal spaces, and also with the retroperitoneum through a tissue plane extending from the sternocostal attachment to the diaphragm, as well as through periaortic and periesophageal fascial planes; as a result, the air may dissect from the mediastinum through these tissue planes, causing pneumopericardium,
TE D
pneumothorax, pneumoperitoneum or pneumoretroperitoneum 6. The precipitating factors - events closely related to the development of SPM and generally due to an increase in intrathoracic pressure - include: Valsalva manoeuvre, emesis, cough, asthma exacerbation, defecation, physical
EP
exercise, childbirth, airway infections, sepsis-induced acute respiratory distress syndrome, inhaled drug use (either continuous and occasional abuse), chocking. The predisposing factors - previous
AC C
conditions favouring pneumomediastinum – are: asthma, interstitial and other lung diseases, corticosteroids, tobacco, inhaled drug use, inhalation of irritants. A classical clinical triad has been described, and consists of thoracic pain (usually retrosternal and pleuritic), subcutaneous emphysema (which is the most common sign) and dyspnea; less common symptoms are cough, fever, dysphonia, odynophagia and dysphagia. The Hamman sign (detected in 10-20% of cases) is pathognomonic and characterized by systolic crackles in the left sternal border, best heard in left lateral decubitus, described as “rubbing balloons”. Leukocytosis and/or neutrophilia are common, sometimes associated with low grade fever. 4
ACCEPTED MANUSCRIPT Chest radiography is the standard diagnostic procedure, showing a double line outlining the mediastinum. Diagnosis can be confirmed by chest computed tomography. Oral contrast study or bronchoscopy should be performed if esophageal and tracheobronchial tree rupture is suspected. Once confirmed the diagnosis, patient should be admitted for monitoring and treatment, consisting
RI PT
of avoidance of the trigger factors, oxygen and bed rest. Some Authors advocate antibiotic prophylaxis for mediastinitis. A chest tube is recommended in case of pneumothorax or hypertensive pneumomediastinum. Recurrence of pneumomediastinum is exetremely rare.
SC
Complications are uncommon as well, and include tension pneumomediastinum, eventuality in which elevated mediastinal pressure leads to diminished cardiac output because of direct cardiac
M AN U
compression or reduced venous return, hemodynamic and respiratory compromise. Airway compression may occur in presence of extensive subcutaneous and mediastinal emphysema. In Boerhaave syndrome (esophageal rupture following vomiting) mortality can be as high as 50-70% 7 Mediastinitis is also a serious complication, and morbidity and mortality are related to coexisting
TE D
illness.
There are no reports of mortality directly related to pneumomediastinum: spontaneous PM is a selflimited condition that rarely produces significant or life-threatening symptoms.
EP
In the present paper, we described an exceptional case of spontaneous pneumomediastinum occurring after inhalation from an electronic cigarette in a healthy young man.
AC C
The increasing use of electronic nicotine delivery systems, such as electronic cigarettes (especially among young never smokers) implies the exposure by users to the aerosols, that may contain propylene glycol and glycerol (whose aerosolised form produce throat irritation and dry cough), flavourings, other chemicals and nicotine 8. Aerosol exposure can be associated with a decrease in fractional exhaled nitric oxide, increase in respiratory impedance and respiratory flow and may also result in respiratory function impairment. Scientific evidence regarding the human health effects of electronic cigarettes is still limited, and studies evaluating whether they are less harmful than cigarettes are inconclusive. 5
ACCEPTED MANUSCRIPT As previously described, pneumomediastinum can rarely occur as a consequence of illicit and recreational drug use; nonetheless, to our knowledge, this is the first case of a spontaneous
AC C
EP
TE D
M AN U
SC
RI PT
pneumomediastinum occurring after the inhalation from an electronic cigarette.
6
ACCEPTED MANUSCRIPT FIGURE LEGENDS Figure 1: Chest radiography.
AC C
EP
TE D
M AN U
SC
RI PT
Figure 2: Chest Computed Tomography showing pneumomediastinum.
7
ACCEPTED MANUSCRIPT REFERENCES 1. A. E. Newcomb, C. P. Clarke. Spontaneous Pneumomediastinum: A Benign Curiosity or a Significant Problem?. CHEST 2005; 128:3298–3302;
RI PT
2. J.B. Jougon, M. Ballester, Delcambre F., T. MacBride et al. Assessment of spontaneous pneumomediastinum: experience with 12 patients. Ann Thorac Surg 75:2003 (1711-1714); 3. E. Panacek, A. J. Singer, B. W. Sherman, W.F. Rutherford. Spontaneous pneumomediastinum: Clinical and natural history. Ann Emerg Med 1992;1222-1227;
SC
4. R. Jones, B. Willis, J.N. Johneson.Spontaneous pneumomediastinum.West J Emerg Med 2008 Nov; 9 (4):217-218;
M AN U
5. S.Murayama, S. Gibo. Spontaneous pneumomediastinum and Macklin effect: Overview and appearance on computed tomography. World J Radiol 2014 Nov 28; 6(11): 850–854. 6. V. Kouritas, K. Papagiannopulos, G. Lazaridis. Pneumomediastinum. J Thorac Dis. 2015 Feb; 7(Suppl1):S44-S49; 7. F. Al-Mufarrej, J. Badar, F. Gharagozloc. Spontaneous pneumomediastinum: diagnostic and therapeutic interventions..J Cardiothorac Surg,
AC C
EP
TE D
8. L. F. Chun, F. Moazed, C. S. Calfel, M. A. Matthay, J. E. Gotts. Pulmonary toxicity of electronic cigarettes: more doubts than certainties. Am J Physiol Lung Cell Mol Physiol 2017 Nov 1; 313(5):L964-L965;
8
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ACCEPTED MANUSCRIPT
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ACCEPTED MANUSCRIPT