(Chest. 2001;120:306-309.)
© 2001
American College of Chest Physicians
Cervical Emphysema, Pneumomediastinum, and Pneumothorax Following Self-induced Oral Injury*
Report of Four Cases and Review of the Literature
María F. López-Peláez, MD;
José Roldán, MD and
Salvador Mateo, MD
*
From the Departments of Radiology (Drs. López-Peláez and Roldán) and Internal Medicine (Dr. Mateo), Hospital Universitario "12 de Octubre," Madrid, Spain.
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Abstract
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Spontaneous rupture of the pulmonary alveoli after a sudden
increase in intra-alveolar pressure is a common cause of
pneumomediastinum, which is usually seen in healthy young men. Other
common causes are traumatic and iatrogenic rupture of the airway and
esophagus; however, pneumomediastinum following cervicofacial emphysema
is much rarer and is occasionally found after dental surgical
procedures, head and neck surgery, or accidental trauma. We present
four cases of subcutaneous emphysema and pneumomediastinum with two
secondary pneumothoraces after self-induced punctures in the oral
cavity. They constitute an uncommon clinical entity that, to our
knowledge, has not been reported in the literature. Its radiologic
appearance, clinical presentation, and diagnosis are
described.
Key Words: cervical emphysema • chest CT • chest radiography • penetrating oral trauma • pneumomediastinum • subcutaneous pneumothorax
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Introduction
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Spontaneous
pneumomediastinum commonly occurs in healthy young men or parturient
women in whom an increased intra-alveolar pressure (Valsalva maneuver,
cough, emesis) leads to the rupture of the marginal pulmonary
alveoli.1
2
3
The air ascends along the mediastinum toward
the subcutaneous space of the neck, causing cervicofacial subcutaneous
emphysema in 70 to 90% of cases.4
5
Inversely,
pneumomediastinum following cervicofacial emphysema is very rare and
has been reported in relation to dental surgical procedures, head and
neck surgery, or orofacial trauma.6
7
8
We review four cases of subcutaneous cervical emphysema and subsequent
pneumomediastinum secondary to self-induced injuries in the oral cavity
in four young men from the same penitentiary center. Two of them also
had pneumothorax demonstrated by chest radiography and CT. The
radiologic appearance of this uncommon entity is described, and the
English-language literature on the reported etiologies of
pneumomediastinum is reviewed.
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Case Reports
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Case 1
A 27-year-old man who smoked 10 cigarettes per day and had been
treated for epilepsy since childhood was admitted to the hospital on
November 3, 1997, complaining of sudden odynophagia, dyspnea, and
cervicofacial emphysema. No history of trauma or surgery was reported.
The initial physical examination showed important cervicofacial and
thoracic subcutaneous emphysema. Results of the esophagogram,
otolaryngologic examination, and bronchoscopy performed in the
emergency department were normal. The initial chest radiograph
showed subcutaneous emphysema in the cervicofacial, thoracic, and
axillary regions with no evidence of rib fracture. The chest CT
performed a few days later demonstrated air in the subcutaneous,
visceral, and carotid spaces of the neck (Fig 1
, top), extending along the anterior mediastinal space down to the
aortic arch. A small right pneumothorax that collapsed the middle lobe
slightly was also observed (Fig 1
, bottom; arrows).
Antibiotic therapy was administered to prevent
mediastinitis, and the patient’s condition improved, making it
possible for him to return to the prison.
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Figure 1.. Chest CT on the level of thyroid gland
(top) and lung bases (bottom) in Case 1.
Top: there is air in subcutaneous (SC), visceral (VS),
carotid (CS), and anterior cervical (ACS) spaces bilaterally.
Bottom: At this level, a small right pneumothorax
collapses slightly the middle lobe (arrows). PSC = posterior cervical
space.
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Case 2
A 26-year-old man who was a former drug abuser, and was
seropositive for HIV and hepatitis C, was admitted to hospital on
November 4, 1997, reporting sudden periorbital and cervicofacial edema,
chest pain, and dyspnea. He denied any autoprovocative maneuver. In the
initial physical examination, periorbital, cervical, and thoracic
emphysema were seen. The chest radiograph confirmed these findings,
demonstrating a small left pneumothorax (Fig 2 , arrows). The results of the esophagogram, otolaryngologic examination,
and bronchoscopy ruled out any abnormality. Antibiotic therapy was
administered to prevent mediastinitis, and his condition improved with
progressive decrease of the emphysema and the pneumothorax.
Case 3
A 31-year-old former drug abuser who was seropositive
for hepatitis C was admitted to hospital on January 5, 1998, with
sudden cervicofacial swelling and chest pain. The patient reported fish
ingestion the day before. In the initial physical examination,
cervicofacial emphysema was found. The chest radiograph revealed
important subcutaneous emphysema and pneumomediastinum (Fig 3
, arrows). However, results of upper GI studies, otolaryngologic
examination, and bronchoscopy performed in the emergency department
were normal. During the following days, the patient’s condition
improved notably, with almost total resolution of the cervical
emphysema and pneumomediastinum shown in the radiographs.
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Figure 3.. Posteroanterior chest radiograph in Case 3. Vast
cervical and axillary subcutaneous emphysema are seen. The radiolucent
lines beside the aorta indicate the mediastinal emphysema (arrows).
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Case 4
A 26-year-old former drug abuser arrived at the hospital on
February 12, 1998, presenting with generalized pain and subcutaneous
emphysema. He had reported having been punched in the prison. In the
physical examination, the only positive finding was orbital and facial
swelling, extending along the neck and anterior chest wall, down to the
iliac fossa. However, there was no evidence of a traumatic lesion. The
chest radiograph and CT confirmed the clinical findings and revealed
the presence of pneumomediastinum.
On February 13, 1998, the patient was found unconscious with response
only to intense painful stimulation. Naloxone and flumazenil were
applied, and orotracheal intubation was performed. In the ICU, the
patient continued to be unresponsive, hypotensive, and severely
hypoxemic. As the results of cranial CT were normal and no
toxins were found in the urine and stomach fluids, ischemic
encephalopathy of unknown origin was suspected. Twenty-four hours
later, the patient recovered consciousness with complete normalization
of his neurologic functions. He progressed uneventfully until his total
recovery.
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Discussion
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Spontaneous pneumomediastinum is usually seen in healthy young men
or parturient women resulting from the rupture of peripheral pulmonary
alveoli1
2
due to sudden increase of intra-alveolar
pressure after exaggerated Valsalva maneuvers.2
3
Barotrauma during mechanical ventilation, ascent phase of a dive, or
hyperbaric treatment are other possible causes. Spontaneous
pneumomediastinum may also complicate obstructive airway processes such
as asthma4
or foreign bodies.1
Subsequently,
air descends along the connective tissue planes and vascular sheaths
toward the mediastinum, ascending up to the communicating cervical
spaces, producing subcutaneous cervical emphysema in 70 to 90% of
cases.5
In 31% of cases, it has no known precipitating
cause.5
Pneumomediastinum following cervicofacial emphysema is rare. It has
been found in the literature after dental extractions,6
7
head and neck surgery, or craniofacial trauma8
in which
the air passes from the air-filled spaces of the head (oral, nasal, and
pharyngeal cavities) through the neck down to the mediastinum.
Air-producing mechanisms play an important role in forcing the air down
to the mediastinum. These can be air compressing a dental
extraction,7
high positive-pressure
ventilation,9
or even variations in aircraft cabin
pressure on descent during a flight.10
However, spontaneous pneumomediastinum and cervicofacial emphysema
following self-induced injuries within the oral cavity have not been
described in the English-language literature. One case of accidental
penetrating oral trauma in a 18-month-old child11
has been
reported; however, pneumothorax was not present in that patient. In our
patients, subcutaneous emphysema and pneumomediastinum were confirmed
by chest radiograph and thoracic CT. There were also small
pneumothoraces in two patients demonstrated by chest radiograph and
thoracic CT, respectively.
The roots of the first, second, and third molars communicate directly
with the sublingual and submandibular spaces.7
12
In
addition, the visceral space of the neck communicates with the
parapharyngeal, sublingual, and submandibular spaces anteriorly and
superiorly11
and inferiorly directly with the mediastinum,
between the trachea and great vessels (anteriorly) and down to the
fourth thoracic vertebra (posteriorly). Thus, blood, pus, or air
entering any of these spaces can migrate downwards into the
mediastinum; likewise, air can ascend within the mediastinal space up
to the root of the neck, producing subcutaneous
emphysema.11
This diagnosis of pneumomediastinum is dependent on radiologic imaging:
standard posteroanterior and lateral radiographs are usually sufficient
for diagnosis, as posteroanterior chest radiographs typically
demonstrate a radiolucent line between the left heart border and the
mediastinal pleura.11
14
Other findings may include
"highlighting" of the aortic knob and the "contiguous
diaphragm" sign.1
11
However, posteroanterior chest
radiographs by themselves may overlook 50% of cases, so that lateral
chest radiographs should always be performed, which increases
sensitivity to nearly 100%.3
13
With lateral
views, air is visualized in the retrosternal space or as lucent streaks
outlining the aorta and other mediastinal structures.11
In
addition, radiographs may detect associated
pneumothoraces.15
Lateral decubitus radiographs may
sometimes be useful to differentiate a pneumothorax from
pneumomediastinum: air will ascend to the highest point possible in the
pneumothorax; however, mediastinal air shows little positional
variations as it is relatively confined.1
11
Complementary diagnostic procedures (esophagogram, esophagoscopy,
bronchoscopy, chest CT) are often performed following conventional
radiographic imaging to rule out spontaneous (Boerhaave syndrome) or
traumatic rupture of esophagus and tracheobronchial tree,
among different causes of secondary pneumomediastinum.13
In our patients, results of the esophagogram, otolaryngologic
examination, and bronchoscopy were normal. The chest CT was performed
in two patients and confirmed pneumomediastinum in both patients and
pneumothorax in one.
Initially, the four patients denied self-mutilation behavior. Two of
them admitted their intentional insertion of sharp objects (needles and
fish bones) into the oral cavity. Self-mutilation was subsequently
confirmed in the other two patients. After the sublingual injuries were
produced, all patients did the Valsalva maneuver. As no etiology for
subcutaneous emphysema and pneumomediastinum was found, and the time
frame of hospital admissions was so similar, the self-induced origin
was initially suspected and subsequently confirmed. Such action could
certainly suggest a possible collusion among the group of convicts from
which the plan was elaborated, due to the implied intention of escaping
from the penitentiary center by the simulation of an emergency.
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Footnotes
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Corrrespondence to: María F. López-Peláez,
MD, Department of Radiology, Hospital Universitario "12 de
Octubre," C/Seseña 34, 10.D, 28024 Madrid, Spain; e-mail:
fernandez lm @ eresmas.com
Received for publication January 21, 2000.
Accepted for publication January 10, 2001.
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Abstract
Introduction
