Downloaded from www.ajronline.org by 117.253.238.148 on 11/08/15 from IP address 117.253.238.148. Copyright ARRS. For personal use only; all rights reserved
987
Case Report
Intrapulmonary Pneumothorax M. A. Matzinger,1
Bronchogenic
Bronchogenic Cyst: Spontaneous as the Presenting Symptom
F. R. Matzinger,2
cysts
are
thought
and H. J. Sachs3
to
result
from
budding of the tracheal diverticulum between sixth weeks of gestation [1 ] and consequently diastinal
or intrapulmonary
in location.
abnormal
the third and may be me-
The clinical
presenta-
tion is variable; some bronchogenic cysts are asymptomatic. Infants typically present with respiratory compromise due to airway compression, whereas older children present with superimposed
infection
We describe
[2]. Other
presentations
a case of a bronchogenic
girl in whom
the presenting
symptoms
caused
by pneu-
mothorax.
Case
A 16-year-old
girl was ice skating
and fell onto her anterior chest. her activity. The next day, after
and continued
a bout of laughing,
she began having
right-sided
chest
pain
with
to the scapula. revealed good with shortness
a right-sided
tension
suction
many
Chest auscultation in the emergency deair entry on both sides. The next day she of breath, and a chest radiograph showed pneumothorax. Despite prolonged chest tube
days,
there
was
persistent
air leak
into
the
right
pleural space. A chest radiograph and a CT scan showed two thinwalled cystic cavities in the right upper lobe (Fig. 1). The remainder of both lungs and the mediastinum were normal. Surgical exploration confirmed a 0.5-cm rupture of the cyst surface. Postoperative course after right upper lobectomy was uneventful. The pathologic diagnosis was intrapulmonary subpleural broncho-
Received September 5, i 99i ; accepted after revision October 30, i 99i. 1 Department of Radiology, Children’s Hospital of Eastern Ontario, 40i Smyth 2 Department of Radiology, Ottawa Civic Hospital, i 053 Caning Ave., Ottawa, 3
Department
AJR 158:987-988,
Bronchogenic
diastinum
cysts
of Surgery, May
Ottawa
Civic Hospital,
1992 036i-803X/92/i585-0987
1 053 Caning
Ave.,
© American
Ottawa, Roentgen
are
or lungs,
but
found
they
most
can
frequently
develop
in the
bronchogenic
the frequency
cysts
are
controversial.
[4] and by DiLorenzo
of mediastinal
bronchogenic
me-
in extrathoracic
locations, such as cervical, pericardial, paravertebral, and infradiaphragmatic sites [3]. The relative occurrence rates of intrapulmonary Reed and Sobonya
She felt no ill effects
over
Discussion
diastinal
Report
radiation partment presented
material.
are unusual.
cyst in a 16-year-old were
genic cyst, based on examination of a bibbed cyst lined by pseudostratified ciliated columnar respiratory epithelium, which overlay parallel bundles of smooth muscle and contained mucoid proteinaceous
or intraand meStudies
by
et al. [5] indicated cysts
was greater,
whereas the series reported by Rogers and Osmer [6] and by Ramenofsky et al. [3] showed a higher frequency of pulmonary
lesions.
Past reports asymptomatic.
to 14 years)
emphasize Among
studied
that bronchogenic
the 26 patients
by DiLorenzo
cysts
(age range,
et al. [5],
may be 2 months
27%
were
asymptomatic and 73% presented with respiratory symptoms such as cough, dyspnea, pain, fever, and pneumonia. Nearly all 20 patients studied by Ramenofsky et al. [31 were symptomatic, presenting with such signs and symptoms as fever, recurrent pneumonia, cervical mass, respiratory distress, frequent upper respiratory infections, and empyema.
Ad., Ottawa, Ontario, Canada Ontario, Canada Ki Y 4E9. Ontario,
Canada
Ray Society
Ki Y 4E9.
KiH
8Li.
MATZINGER
Downloaded from www.ajronline.org by 117.253.238.148 on 11/08/15 from IP address 117.253.238.148. Copyright ARRS. For personal use only; all rights reserved
988
ET AL.
AJR:i58,
May i992
Fig. 1.-16-year-old giri with intrapulmonary bronchogenic cyst. A, Chest radiograph on day chest tube was inserted shows two thin-walled right upper lobe cysts (arrows) and a moderately large pneumothorax. B, CT scan 11 days later shows two contiguous air-containing cavities in posterior aspect of right upper lobe. Visceral pleura overiying the more anterior cavity appears to be open (arrow), permitting cyst communication with pleural
space.
A
B
A literature search disclosed no previous report of spontaneous pneumothorax caused by rupture of an intrapubmonary bronchogenic Cyst. Kirwan et ab. [7] described catastrophic spontaneous rupture of an upper mediastinal bronchogenic
defined, thin-walled, round or oval lesion that may be air filled, have homogeneous water density, or contain an air-fluid level. CT provides optimal demonstration of cyst location, mor-
Cyst with leakage
choice for both the infected and uninfected cyst to firmly establish the diagnosis and prevent future complications [1].
into the pericardium
and cardiac
tampon-
ade. Adam et al. [8] described a 24-year-old woman with right pleuritic chest pain and a very small right pleural effusion presumed to result from leakage of the contents of a subcarinal cyst. Air embolism was reported in two men, with right bower lung Cysts, who underwent decompression after construCtion work in a tunnel [9]. Since no surgery or pathologic examination of the cysts was carried out, confirmation of their
congenital
Our case
and contents.
illustrates
Surgical
excision
the plain radiographic
is the treatment
and CT features
of
of
bronchogenic cyst and represents an atypical presentation of this relatively uncommon congenital lesion. Bronchogenic cyst is generally regarded as an unlikely cause of pneumothorax; perhaps, however, it is more common than intrapubmonary
previously
thought.
nature was not available.
Traumatic lung cyst the major considerations
patient.
phobogy,
Traumatic
and congenital pulmonary cyst were in the differential diagnosis of our
cyst typically
follows
blunt chest
trauma
with sudden compression of the elastic chest wall in a young person. Radiologically apparent within 1 2 to 24 hr of injury, traumatic Cyst undergoes complete resolution in 2 to 16 weeks [1]. Pneumothorax is a rare complication. Congenital Cysts tend to be multiple, either limited to a lobe, or generalized, involving one or both lungs. They may be asymptomatic, or the patient may present with complications of either infection or air trapping and tension [1 ]. Other possible causes of thin-walled, air-containing pulmonary cysts include (1) cavities resulting from mycotic, tuberculous, or Echinococcus infection and (2) pneumatoceles caused by Staphylococcus aureus or hydrocarbon ingestion. Chest radiography is the usual imaging method for initial detection of bronchogenic cysts, although the findings are not always diagnostic. On a chest radiograph, an intrapulmonary bronchogenic cyst typically appears as a sharply
REFERENCES i 2. 3. 4. 5. 6. 7. 8.
9.
.
Shamji FM Sachs HJ, Perkins DG. Cystic disease of the lungs. Surg Clin North Am 1988;68:581-620 Haddon MJ, Bowen A. Bronchopulmonary and neurenteric forms of foregut anomalies. Radiol Clin North Am 1991;29:24i-254 Ramenofsky ML, Leape LL, McCauley RGK. Bronchogenic cyst. J Pediatr Surg 1979;i4:2i9-224 Reed JC, Sobonya RE. Morphologic analysis offoregut cysts in the thorax. AJR 1974;120:851-860 DiLorenzo M, Collin PP, Vaillancourt A, Duranceau A. Bronchogenic cysts. J Pediatr Surg 1989;24:988-991 Rogers LF, Osmer JC. Bronchogenic cyst. A review of 46 cases. AJR 1964;9i :273-283 Kirwan WO, Walbaum PR, McCormack RJM. Cystic intrathoracic derivatives of the foregut and their complications. Thorax 1973;28:424-428 Adam A, MacSweeney JE, Whyte MKB, Smith PLC, nd PW. CT-guided extrapleural drainage of bronchogenic cyst. J Comput Assist Tomogr 1989;13: 1065-i 068 Campbell Golding F, Griffiths P, Hempleman HV, Paton WDM, Walder DN. Decompression sickness during construction of the Dartford tunnel. Br J IndMed 1960;i7:167-180
987
Case Report
Intrapulmonary Pneumothorax M. A. Matzinger,1
Bronchogenic
Bronchogenic Cyst: Spontaneous as the Presenting Symptom
F. R. Matzinger,2
cysts
are
thought
and H. J. Sachs3
to
result
from
budding of the tracheal diverticulum between sixth weeks of gestation [1 ] and consequently diastinal
or intrapulmonary
in location.
abnormal
the third and may be me-
The clinical
presenta-
tion is variable; some bronchogenic cysts are asymptomatic. Infants typically present with respiratory compromise due to airway compression, whereas older children present with superimposed
infection
We describe
[2]. Other
presentations
a case of a bronchogenic
girl in whom
the presenting
symptoms
caused
by pneu-
mothorax.
Case
A 16-year-old
girl was ice skating
and fell onto her anterior chest. her activity. The next day, after
and continued
a bout of laughing,
she began having
right-sided
chest
pain
with
to the scapula. revealed good with shortness
a right-sided
tension
suction
many
Chest auscultation in the emergency deair entry on both sides. The next day she of breath, and a chest radiograph showed pneumothorax. Despite prolonged chest tube
days,
there
was
persistent
air leak
into
the
right
pleural space. A chest radiograph and a CT scan showed two thinwalled cystic cavities in the right upper lobe (Fig. 1). The remainder of both lungs and the mediastinum were normal. Surgical exploration confirmed a 0.5-cm rupture of the cyst surface. Postoperative course after right upper lobectomy was uneventful. The pathologic diagnosis was intrapulmonary subpleural broncho-
Received September 5, i 99i ; accepted after revision October 30, i 99i. 1 Department of Radiology, Children’s Hospital of Eastern Ontario, 40i Smyth 2 Department of Radiology, Ottawa Civic Hospital, i 053 Caning Ave., Ottawa, 3
Department
AJR 158:987-988,
Bronchogenic
diastinum
cysts
of Surgery, May
Ottawa
Civic Hospital,
1992 036i-803X/92/i585-0987
1 053 Caning
Ave.,
© American
Ottawa, Roentgen
are
or lungs,
but
found
they
most
can
frequently
develop
in the
bronchogenic
the frequency
cysts
are
controversial.
[4] and by DiLorenzo
of mediastinal
bronchogenic
me-
in extrathoracic
locations, such as cervical, pericardial, paravertebral, and infradiaphragmatic sites [3]. The relative occurrence rates of intrapulmonary Reed and Sobonya
She felt no ill effects
over
Discussion
diastinal
Report
radiation partment presented
material.
are unusual.
cyst in a 16-year-old were
genic cyst, based on examination of a bibbed cyst lined by pseudostratified ciliated columnar respiratory epithelium, which overlay parallel bundles of smooth muscle and contained mucoid proteinaceous
or intraand meStudies
by
et al. [5] indicated cysts
was greater,
whereas the series reported by Rogers and Osmer [6] and by Ramenofsky et al. [3] showed a higher frequency of pulmonary
lesions.
Past reports asymptomatic.
to 14 years)
emphasize Among
studied
that bronchogenic
the 26 patients
by DiLorenzo
cysts
(age range,
et al. [5],
may be 2 months
27%
were
asymptomatic and 73% presented with respiratory symptoms such as cough, dyspnea, pain, fever, and pneumonia. Nearly all 20 patients studied by Ramenofsky et al. [31 were symptomatic, presenting with such signs and symptoms as fever, recurrent pneumonia, cervical mass, respiratory distress, frequent upper respiratory infections, and empyema.
Ad., Ottawa, Ontario, Canada Ontario, Canada Ki Y 4E9. Ontario,
Canada
Ray Society
Ki Y 4E9.
KiH
8Li.
MATZINGER
Downloaded from www.ajronline.org by 117.253.238.148 on 11/08/15 from IP address 117.253.238.148. Copyright ARRS. For personal use only; all rights reserved
988
ET AL.
AJR:i58,
May i992
Fig. 1.-16-year-old giri with intrapulmonary bronchogenic cyst. A, Chest radiograph on day chest tube was inserted shows two thin-walled right upper lobe cysts (arrows) and a moderately large pneumothorax. B, CT scan 11 days later shows two contiguous air-containing cavities in posterior aspect of right upper lobe. Visceral pleura overiying the more anterior cavity appears to be open (arrow), permitting cyst communication with pleural
space.
A
B
A literature search disclosed no previous report of spontaneous pneumothorax caused by rupture of an intrapubmonary bronchogenic Cyst. Kirwan et ab. [7] described catastrophic spontaneous rupture of an upper mediastinal bronchogenic
defined, thin-walled, round or oval lesion that may be air filled, have homogeneous water density, or contain an air-fluid level. CT provides optimal demonstration of cyst location, mor-
Cyst with leakage
choice for both the infected and uninfected cyst to firmly establish the diagnosis and prevent future complications [1].
into the pericardium
and cardiac
tampon-
ade. Adam et al. [8] described a 24-year-old woman with right pleuritic chest pain and a very small right pleural effusion presumed to result from leakage of the contents of a subcarinal cyst. Air embolism was reported in two men, with right bower lung Cysts, who underwent decompression after construCtion work in a tunnel [9]. Since no surgery or pathologic examination of the cysts was carried out, confirmation of their
congenital
Our case
and contents.
illustrates
Surgical
excision
the plain radiographic
is the treatment
and CT features
of
of
bronchogenic cyst and represents an atypical presentation of this relatively uncommon congenital lesion. Bronchogenic cyst is generally regarded as an unlikely cause of pneumothorax; perhaps, however, it is more common than intrapubmonary
previously
thought.
nature was not available.
Traumatic lung cyst the major considerations
patient.
phobogy,
Traumatic
and congenital pulmonary cyst were in the differential diagnosis of our
cyst typically
follows
blunt chest
trauma
with sudden compression of the elastic chest wall in a young person. Radiologically apparent within 1 2 to 24 hr of injury, traumatic Cyst undergoes complete resolution in 2 to 16 weeks [1]. Pneumothorax is a rare complication. Congenital Cysts tend to be multiple, either limited to a lobe, or generalized, involving one or both lungs. They may be asymptomatic, or the patient may present with complications of either infection or air trapping and tension [1 ]. Other possible causes of thin-walled, air-containing pulmonary cysts include (1) cavities resulting from mycotic, tuberculous, or Echinococcus infection and (2) pneumatoceles caused by Staphylococcus aureus or hydrocarbon ingestion. Chest radiography is the usual imaging method for initial detection of bronchogenic cysts, although the findings are not always diagnostic. On a chest radiograph, an intrapulmonary bronchogenic cyst typically appears as a sharply
REFERENCES i 2. 3. 4. 5. 6. 7. 8.
9.
.
Shamji FM Sachs HJ, Perkins DG. Cystic disease of the lungs. Surg Clin North Am 1988;68:581-620 Haddon MJ, Bowen A. Bronchopulmonary and neurenteric forms of foregut anomalies. Radiol Clin North Am 1991;29:24i-254 Ramenofsky ML, Leape LL, McCauley RGK. Bronchogenic cyst. J Pediatr Surg 1979;i4:2i9-224 Reed JC, Sobonya RE. Morphologic analysis offoregut cysts in the thorax. AJR 1974;120:851-860 DiLorenzo M, Collin PP, Vaillancourt A, Duranceau A. Bronchogenic cysts. J Pediatr Surg 1989;24:988-991 Rogers LF, Osmer JC. Bronchogenic cyst. A review of 46 cases. AJR 1964;9i :273-283 Kirwan WO, Walbaum PR, McCormack RJM. Cystic intrathoracic derivatives of the foregut and their complications. Thorax 1973;28:424-428 Adam A, MacSweeney JE, Whyte MKB, Smith PLC, nd PW. CT-guided extrapleural drainage of bronchogenic cyst. J Comput Assist Tomogr 1989;13: 1065-i 068 Campbell Golding F, Griffiths P, Hempleman HV, Paton WDM, Walder DN. Decompression sickness during construction of the Dartford tunnel. Br J IndMed 1960;i7:167-180
