Retropharyngeal Report of a MAJ Richard J.
Case
Perrotta, MC, USA, Robert W. Selfe,
\s=b\ We report the case of an infant with penetrating neck trauma followed by an upper respiratory tract obstruction. This case presented a diagnostic and management dilemma because of the development of an unusual retropharyngeal
collection of air. In our review of the medical literature, we were unable to find any other case with a similar manifestation and clinical course.
(Arch Otolaryngol 105:355-357, 1979)
Every puncture
case
of
penetrating trauma unique. The small¬
to the neck is
est
wound may result in
a
life-threatening situation, posing a multitude of challenges to the clini¬
cian and surgeon alike. We report a very unusual case of iatrogenic pene¬ trating trauma to the neck of an infant. REPORT OF A CASE
A 3-week-old girl was brought to her pediatrician because of persistent vomit¬ ing, diarrhea, and weight loss. During an attempt to obtain a blood sample, a left external jugular stick was unsuccessful.
Accepted for publication June 30, 1978. From the Department of Otorhinolaryngology, The New York Hospital, New York. Dr Perrotta is now with the US Army Hospital, Fort Polk, La.
Mediastinal Pneumatocele
Reprint requests to US Army Hospital, Fort Polk, LA 71459 (Dr Perrotta).
MD
This was followed by a right internal jugu¬ lar stick, which resulted in aspiration of 4 cc of air into the syringe. A 7-ml blood sample was then obtained from the left internal jugular vein. Two minutes follow¬ ing these procedures, the child stopped breathing and her heart rate dropped to three to four beats per minute. The child responded to external cardiac massage and mouth-to-mouth resuscitation. She was then transferred to the New York Hospital for further evaluation. Examination on the day of admission, Sept 12, 1977, showed normal findings except for poor tissue turgor. Temperature was 36.3 °C, respirations were 60/min, and pulse rate was 150 beats per minute. A chest x-ray film revealed normal lung fields and a small amount of subcutaneous air in the right lateral side of the neck. Her hematocrit reading was 37.7%; hemoglobin level was 12.6 g/dl. The patient did well for four days, but coughing and retractions developed shortly after feeding on Sept 16. Following chest x-ray, she became dyspneic with stridor, cyanosis, and bradycardia. An orotracheal tube was passed, and the child began treat¬ ment with a pressure-cycled respirator. The x-ray film taken during this episode showed a narrowing of the trachea at the thoracic inlet. However, on retrospective examination, there was a faint retropha¬ ryngeal collection of air present (Fig 1). Her hematocrit reading at this time was 19.6%; she received 30 ml of packed cells, and her posttransfusion hematocrit read¬ ing stabilized at 36%.
She was extubated within 48 hours, but had to be reintubated because of recurrent stridor and cyanosis. A chest x-ray film taken at the time of extubation on Sept 18 showed an increase in the size of the air shadow. Direct laryngoscopy and bronchoscopy performed 24 hours later disclosed a 50% narrowing of her trachéal lumen extending from 1 to 2 cm above the carina, resulting from nonpulsatile external com¬ pression or edema of the right lateral and posterior walls. The trachéal mucosa appeared normal. A nasotracheal tube was then passed to maintain the child's airway, and dexamethasone sodium phosphate injection, 0.5 mg every six hours, was administered in an attempt to decrease trachéal edema. A barium swallow performed a few hours after endoscopy showed a further increase in the size of the air pocket, which was situated posterior to the trachea and esophagus and displaced both structures to the left and anteriorly (Fig 2). No commu¬ nication between the esophagus and air pocket was demonstrated. A second barium swallow and tracheography performed three days later again failed to demon¬ strate any communication between the air pocket and trachea or esophagus. On Sept 28, the patient underwent a follow-up direct laryngoscopy and bronchoscopy that revealed only a minimal narrowing at the same trachéal level. She received positive-pressure ventilation by mask and bronchoscope during this proce¬ dure as well as during her first endoscopy. Because of the adequate airway, she was
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Fig 1 .—Faint air density in posterior part of trachea (arrows). Also note trachéal com¬ pression at thoracic inlet.
Fig 2.—Enlarging retropharyngeal air cavi¬ ty (arrows). Small amount of barium is in esophagus. Nasotracheal tube is in place.
Fig 3.—Further increase in size of pneuma¬ tocele with extension into mediastinum.
Fig 6.—Resolution
Fig 4—Elongation of pneumatocele during inspiration (arrows). extubated without difficulty, but a chest x-ray film obtained after this endoscopy showed a considerable increase in the size of the air pocket (Fig 3). On Sept 29, while undertaking fluoroscopic examination for a barium swallow, another observation was made. During inspiration (Fig 4), the air pocket was elongated and descended into the medias¬ tinum, and during expiration (Fig 5), it ascended into the retropharyngeal area. There was no apparent change in the volume of the air pocket, but rather, a discrete change in shape and location took place. Some air was also noted anterior to the trachea during this study. The air pocket slowly decreased in size until Oct 3, at which time a third direct laryngoscopy and bronchoscopy was per¬ formed with positive-pressure ventilation. No airway compression or obstruction was noted, but once again a follow-up x-ray
Fig 5.—Pneumatocele rising to during expiration (arrows).
base of
skull
film demonstrated an increase in the size of the air pocket. No further endoscopie procedures were performed thereafter. The evaluation of the air pocket was then accomplished solely by x-ray films, and on Oct 13 obvious decrease in its size was noted. Because of the child's progress, she was discharged on Oct 17, but she had to be readmitted on Oct 19 because of recurrent diarrhea that was subsequently controlled. She experienced no further respiratory difficulties,, and a chest x-ray film obtained on Oct 30 showed resolution of the air pocket (Fig 6).
COMMENT
Our review of the medical literature disclosed reports of some very inter¬ esting cervical and mediastinal lesions both in children and adults. Lotan et al1 described an anterior cervical
of
pneumatocele.
pneumatocele that resulted from a tracheotomy tube decannulation. Devgan and Brodeur- reported an apical pneumatocele originating from the right lung, and Bergstrom et al·1 reported a case of a migrating bronchogenic cyst in the superior medias¬ tinum. Killen and Collins' reported two cases of tracheoesophageal fistu¬ las secondary to blunt trauma to the chest. However,
we were
of
unable to
a patient any description with a history and roentgenographic manifestation similar to that of the patient described herein. We have chosen to call this lesion a pneumatocele because it developed into a well-circumscribed pocket of air contained within a specific anatomical region. The probable location of this pneumatocele would be in the "danger space" or space number 4 of Grodinsky and Holyoke, as described by Levitt."' This compartment is limited posteriorly by the prevertebral fascia and anteriorly by the alar fascia. It extends from the base of the skull superiorly, through the posterior meuncover
Downloaded From: http://archotol.jamanetwork.com/ by a University of Pennsylvania User on 06/21/2015
diastinum,
to the level of the dia¬
The length of this space would also allow for the free migration of the pneumatocele as shown in Fig 4 and 5. We believe that the most likely sequence of events in the development of the pneumatocele began with the three attempts at jugular venipuncture, one of which aspirated 4 cc of air. These resulted in a communication between the hypopharynx or trachea and the retropharyngeal region that acted as a one-way valve. This valve effect allowed the pneumatocele to enlarge after each episode of positivepressure ventilation either by respira¬ tor or during each endoscopie proce¬ dure. A congenital origin, such as a bron-
phragm inferiorly.
chogenic
or
gastroenteric cyst,
was
also considered. Such cysts can contain air fluid levels if there is a patent communication between the cyst and either a bronchus or esophagus," and these cysts may be located in the same anatomical region.1' But in this case,
the pneumatocele developed gradually after the penetrating trauma and while the patient was hospitalized. The first bronchoscopy did show a swelling within the trachéal lumen that subsequently resolved and that would explain the earlier episodes of respiratory obstruction. The onset of bradycardia and respiratory arrest at the time of venipuncture can also be accounted for by the trauma, since it is known that percutaneous catheterization of the internal jugular vein can produce cardiac arrhythmias.7 Conversely, bronchogenic cysts will commonly produce coincidental pulmonary changes such as a mediastinal shift and emphysematous changes of the obstructed lung.8·9 Gastroenteric cysts produce common symptoms of cough, dyspnea, vomit¬ ing, and poor weight gain, and they are frequently associated with verte¬ bral anomalies.1" Our patient had no substantial pulmonary involvement throughout her illness. Multiple esophagograms failed to show any extrinsic
or
intrinsic abnormalities of the No vertebral anomalies
esophagus. were seen.
The key to the successful treatment of this patient came with the realiza¬ tion that the air within the pneumato¬ cele was being maintained by the use of positive-pressure ventilation. After the third endoscopy, a retrospective examination of all the patient's x-ray films showed us the nature of the problem. Only when all positive-pres¬ sure ventilation was discontinued did the pneumatocele gradually decrease in size and eventually resolve (Fig
6).
The management of
penetrating
neck, exploration or will forever be argued in observation, trauma to the
the medical literature
as
it was in this
specific case.111" However, it was our opinion that since the child remained afebrile, had no further respiratory distress, and the role of positive-pres¬ ventilation had become apparent, observation rather than exploration was the treatment of choice. sure
References 1. Lotan AN, Eliachar I, Joachims HZ: Pretracheal air cyst (late complication of tracheotomy). Arch Otolaryngol 103:596-597, 1977. 2. Devgan BK, Brodeur AE: Apical penumatocele. Arch Otolaryngol 102:121-123, 1976. Yost RV, Ford KT, et al: 3. Bergstrom Unusual roentgen manifestations of bronchogenic cysts. Radiology 107:49-54, 1973. 4. Killen DA, Collins HA: Tracheo-esophageal fistula resulting from nonpenetrating trauma to the chest. J Thorac Cardiovasc Surg 50:104-110, 1965. 5. Levitt GW: Cervical fascia and deep neck
JF,
infections. Laryngoscope 80:409-435, 1970. 6. Oldham HN Jr: Mediastinal tumors and cysts. Ann Thorac Surg 11:246-275, 1971. 7. Brown CS, Wallace CT: Chronic hematoma: A complication of percutaneous catheterization of the internal jugular vein. Anesthesiology 45:368-369, 1976. 8. Crawford DB, Oh KS, Shermeta DW: Neonatal respiratory distress due to a bronchogenic cyst. Br J Radiol 48:494-496, 1975. 9. Opsahl T, Berman EJ: Bronchogenic mediastinal cysts in infants: Case report and review of literature. Pediatrics 30:372-377, 1962.
10. Spock A, Schneider S, Baylin GJ: Mediastinal gastric cysts: A case report and review of the English literature. Am Rev Respir Dis 94:97-103, 1966. 11. Sheely CH, Mattox KL, Reul GJ, et al: Current concepts in the management of penetrating neck trauma. J Trauma 15:895-900, 1975. 12. Saletta JD, Lowe RJ, Lim LT, et al: Penetrating trauma of the neck. J Trauma 16:579-587, 1976.
Downloaded From: http://archotol.jamanetwork.com/ by a University of Pennsylvania User on 06/21/2015
Case
Perrotta, MC, USA, Robert W. Selfe,
\s=b\ We report the case of an infant with penetrating neck trauma followed by an upper respiratory tract obstruction. This case presented a diagnostic and management dilemma because of the development of an unusual retropharyngeal
collection of air. In our review of the medical literature, we were unable to find any other case with a similar manifestation and clinical course.
(Arch Otolaryngol 105:355-357, 1979)
Every puncture
case
of
penetrating trauma unique. The small¬
to the neck is
est
wound may result in
a
life-threatening situation, posing a multitude of challenges to the clini¬
cian and surgeon alike. We report a very unusual case of iatrogenic pene¬ trating trauma to the neck of an infant. REPORT OF A CASE
A 3-week-old girl was brought to her pediatrician because of persistent vomit¬ ing, diarrhea, and weight loss. During an attempt to obtain a blood sample, a left external jugular stick was unsuccessful.
Accepted for publication June 30, 1978. From the Department of Otorhinolaryngology, The New York Hospital, New York. Dr Perrotta is now with the US Army Hospital, Fort Polk, La.
Mediastinal Pneumatocele
Reprint requests to US Army Hospital, Fort Polk, LA 71459 (Dr Perrotta).
MD
This was followed by a right internal jugu¬ lar stick, which resulted in aspiration of 4 cc of air into the syringe. A 7-ml blood sample was then obtained from the left internal jugular vein. Two minutes follow¬ ing these procedures, the child stopped breathing and her heart rate dropped to three to four beats per minute. The child responded to external cardiac massage and mouth-to-mouth resuscitation. She was then transferred to the New York Hospital for further evaluation. Examination on the day of admission, Sept 12, 1977, showed normal findings except for poor tissue turgor. Temperature was 36.3 °C, respirations were 60/min, and pulse rate was 150 beats per minute. A chest x-ray film revealed normal lung fields and a small amount of subcutaneous air in the right lateral side of the neck. Her hematocrit reading was 37.7%; hemoglobin level was 12.6 g/dl. The patient did well for four days, but coughing and retractions developed shortly after feeding on Sept 16. Following chest x-ray, she became dyspneic with stridor, cyanosis, and bradycardia. An orotracheal tube was passed, and the child began treat¬ ment with a pressure-cycled respirator. The x-ray film taken during this episode showed a narrowing of the trachea at the thoracic inlet. However, on retrospective examination, there was a faint retropha¬ ryngeal collection of air present (Fig 1). Her hematocrit reading at this time was 19.6%; she received 30 ml of packed cells, and her posttransfusion hematocrit read¬ ing stabilized at 36%.
She was extubated within 48 hours, but had to be reintubated because of recurrent stridor and cyanosis. A chest x-ray film taken at the time of extubation on Sept 18 showed an increase in the size of the air shadow. Direct laryngoscopy and bronchoscopy performed 24 hours later disclosed a 50% narrowing of her trachéal lumen extending from 1 to 2 cm above the carina, resulting from nonpulsatile external com¬ pression or edema of the right lateral and posterior walls. The trachéal mucosa appeared normal. A nasotracheal tube was then passed to maintain the child's airway, and dexamethasone sodium phosphate injection, 0.5 mg every six hours, was administered in an attempt to decrease trachéal edema. A barium swallow performed a few hours after endoscopy showed a further increase in the size of the air pocket, which was situated posterior to the trachea and esophagus and displaced both structures to the left and anteriorly (Fig 2). No commu¬ nication between the esophagus and air pocket was demonstrated. A second barium swallow and tracheography performed three days later again failed to demon¬ strate any communication between the air pocket and trachea or esophagus. On Sept 28, the patient underwent a follow-up direct laryngoscopy and bronchoscopy that revealed only a minimal narrowing at the same trachéal level. She received positive-pressure ventilation by mask and bronchoscope during this proce¬ dure as well as during her first endoscopy. Because of the adequate airway, she was
Downloaded From: http://archotol.jamanetwork.com/ by a University of Pennsylvania User on 06/21/2015
Fig 1 .—Faint air density in posterior part of trachea (arrows). Also note trachéal com¬ pression at thoracic inlet.
Fig 2.—Enlarging retropharyngeal air cavi¬ ty (arrows). Small amount of barium is in esophagus. Nasotracheal tube is in place.
Fig 3.—Further increase in size of pneuma¬ tocele with extension into mediastinum.
Fig 6.—Resolution
Fig 4—Elongation of pneumatocele during inspiration (arrows). extubated without difficulty, but a chest x-ray film obtained after this endoscopy showed a considerable increase in the size of the air pocket (Fig 3). On Sept 29, while undertaking fluoroscopic examination for a barium swallow, another observation was made. During inspiration (Fig 4), the air pocket was elongated and descended into the medias¬ tinum, and during expiration (Fig 5), it ascended into the retropharyngeal area. There was no apparent change in the volume of the air pocket, but rather, a discrete change in shape and location took place. Some air was also noted anterior to the trachea during this study. The air pocket slowly decreased in size until Oct 3, at which time a third direct laryngoscopy and bronchoscopy was per¬ formed with positive-pressure ventilation. No airway compression or obstruction was noted, but once again a follow-up x-ray
Fig 5.—Pneumatocele rising to during expiration (arrows).
base of
skull
film demonstrated an increase in the size of the air pocket. No further endoscopie procedures were performed thereafter. The evaluation of the air pocket was then accomplished solely by x-ray films, and on Oct 13 obvious decrease in its size was noted. Because of the child's progress, she was discharged on Oct 17, but she had to be readmitted on Oct 19 because of recurrent diarrhea that was subsequently controlled. She experienced no further respiratory difficulties,, and a chest x-ray film obtained on Oct 30 showed resolution of the air pocket (Fig 6).
COMMENT
Our review of the medical literature disclosed reports of some very inter¬ esting cervical and mediastinal lesions both in children and adults. Lotan et al1 described an anterior cervical
of
pneumatocele.
pneumatocele that resulted from a tracheotomy tube decannulation. Devgan and Brodeur- reported an apical pneumatocele originating from the right lung, and Bergstrom et al·1 reported a case of a migrating bronchogenic cyst in the superior medias¬ tinum. Killen and Collins' reported two cases of tracheoesophageal fistu¬ las secondary to blunt trauma to the chest. However,
we were
of
unable to
a patient any description with a history and roentgenographic manifestation similar to that of the patient described herein. We have chosen to call this lesion a pneumatocele because it developed into a well-circumscribed pocket of air contained within a specific anatomical region. The probable location of this pneumatocele would be in the "danger space" or space number 4 of Grodinsky and Holyoke, as described by Levitt."' This compartment is limited posteriorly by the prevertebral fascia and anteriorly by the alar fascia. It extends from the base of the skull superiorly, through the posterior meuncover
Downloaded From: http://archotol.jamanetwork.com/ by a University of Pennsylvania User on 06/21/2015
diastinum,
to the level of the dia¬
The length of this space would also allow for the free migration of the pneumatocele as shown in Fig 4 and 5. We believe that the most likely sequence of events in the development of the pneumatocele began with the three attempts at jugular venipuncture, one of which aspirated 4 cc of air. These resulted in a communication between the hypopharynx or trachea and the retropharyngeal region that acted as a one-way valve. This valve effect allowed the pneumatocele to enlarge after each episode of positivepressure ventilation either by respira¬ tor or during each endoscopie proce¬ dure. A congenital origin, such as a bron-
phragm inferiorly.
chogenic
or
gastroenteric cyst,
was
also considered. Such cysts can contain air fluid levels if there is a patent communication between the cyst and either a bronchus or esophagus," and these cysts may be located in the same anatomical region.1' But in this case,
the pneumatocele developed gradually after the penetrating trauma and while the patient was hospitalized. The first bronchoscopy did show a swelling within the trachéal lumen that subsequently resolved and that would explain the earlier episodes of respiratory obstruction. The onset of bradycardia and respiratory arrest at the time of venipuncture can also be accounted for by the trauma, since it is known that percutaneous catheterization of the internal jugular vein can produce cardiac arrhythmias.7 Conversely, bronchogenic cysts will commonly produce coincidental pulmonary changes such as a mediastinal shift and emphysematous changes of the obstructed lung.8·9 Gastroenteric cysts produce common symptoms of cough, dyspnea, vomit¬ ing, and poor weight gain, and they are frequently associated with verte¬ bral anomalies.1" Our patient had no substantial pulmonary involvement throughout her illness. Multiple esophagograms failed to show any extrinsic
or
intrinsic abnormalities of the No vertebral anomalies
esophagus. were seen.
The key to the successful treatment of this patient came with the realiza¬ tion that the air within the pneumato¬ cele was being maintained by the use of positive-pressure ventilation. After the third endoscopy, a retrospective examination of all the patient's x-ray films showed us the nature of the problem. Only when all positive-pres¬ sure ventilation was discontinued did the pneumatocele gradually decrease in size and eventually resolve (Fig
6).
The management of
penetrating
neck, exploration or will forever be argued in observation, trauma to the
the medical literature
as
it was in this
specific case.111" However, it was our opinion that since the child remained afebrile, had no further respiratory distress, and the role of positive-pres¬ ventilation had become apparent, observation rather than exploration was the treatment of choice. sure
References 1. Lotan AN, Eliachar I, Joachims HZ: Pretracheal air cyst (late complication of tracheotomy). Arch Otolaryngol 103:596-597, 1977. 2. Devgan BK, Brodeur AE: Apical penumatocele. Arch Otolaryngol 102:121-123, 1976. Yost RV, Ford KT, et al: 3. Bergstrom Unusual roentgen manifestations of bronchogenic cysts. Radiology 107:49-54, 1973. 4. Killen DA, Collins HA: Tracheo-esophageal fistula resulting from nonpenetrating trauma to the chest. J Thorac Cardiovasc Surg 50:104-110, 1965. 5. Levitt GW: Cervical fascia and deep neck
JF,
infections. Laryngoscope 80:409-435, 1970. 6. Oldham HN Jr: Mediastinal tumors and cysts. Ann Thorac Surg 11:246-275, 1971. 7. Brown CS, Wallace CT: Chronic hematoma: A complication of percutaneous catheterization of the internal jugular vein. Anesthesiology 45:368-369, 1976. 8. Crawford DB, Oh KS, Shermeta DW: Neonatal respiratory distress due to a bronchogenic cyst. Br J Radiol 48:494-496, 1975. 9. Opsahl T, Berman EJ: Bronchogenic mediastinal cysts in infants: Case report and review of literature. Pediatrics 30:372-377, 1962.
10. Spock A, Schneider S, Baylin GJ: Mediastinal gastric cysts: A case report and review of the English literature. Am Rev Respir Dis 94:97-103, 1966. 11. Sheely CH, Mattox KL, Reul GJ, et al: Current concepts in the management of penetrating neck trauma. J Trauma 15:895-900, 1975. 12. Saletta JD, Lowe RJ, Lim LT, et al: Penetrating trauma of the neck. J Trauma 16:579-587, 1976.
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