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Unusual manifestations of neonatal pharyngeal perforation
ClinicalRadiology (1980) 31,581-585 ©1980 Royal College of Radiologists
0009-9260/80/00830581502.00
Llnusual Manifestations of Neonatal Pharyngeal perforation ~RIC N. F A E R B E R * , A L A N M. S C H W A R T Z * , LEWIS W. P I N C H t and J O H N C. L E O N I D A S *
,Department of Pediatric Radiology, Boston Floating Hospital, Tufts New England Medical Center Hospital, Massachusetts, and ~Matthew Thornton Health Plan, Nashua, New Hampshire, U.S.A. The small series o f previously described cases o f n e o n a t a l pharyngeal perforation are reviewed and two additional cases w i t h atypical initial presentations are described. The c o n d i t i o n is more c o m m o n than is currently appreciated, as suggested by one o f our cases which r e m a i n e d u n d e t e c t e d until m a n y years later. We also wish to emphasise the high l o c a t i o n o f perforation despite an initial presentation which m a y appear to the contrary.
Traumatic perforation o f the p h a r y n x in the n e o n a t a l period is rare. The c o n d i t i o n m a y simulate oesophageal atresia b y presenting as a high obstruction to the passage o f a feeding tube. Two cases are described, b o t h w i t h atypical initial presentations. The invariably high l o c a t i o n of perforation site is emphasised, as this has n o t received adequate a t t e n t i o n in the literature.
found in the chest which culture proved sterile. Chest tube drainage of the mediastinum was instituted. The retro-oesophageal space in the neck was then explored through the left cervical approach, and the tube was found to be behind the entire cervical length of the oesophagus with the site of perforation being in the hypopharynx. The neck was drained with a small Penrose drain and the rent in the hypopharynx was allowed to heal without
CASE REPORT Case 1. The patient was born after a 28 week gestation to a 19-year-old mother who had received inadequate prenatal care. The labour was uncomplicated; birth was by vaginal delivery with a vertex presentation. The infant breathed spontaneously but subsequently developed mild respiratory distress, and an abdominal radiograph demonstrated the catheter tube tip within the stomach. Repeat passage of nasogastric tube was accomplished with difficulty. Chest and abdominal radiographs revealed right middle lobe volume loss. The nasogastric tube tip was noted to lie posteriorly at the level of L3 (Figs la, b). This low position of the catheter suggested perforation of the oesophagus at the intrathoracic level. An oesophagram was performed through a second nasogastric tube, using water soluble contrast medium. A high site of perforation within the pharynx was demonstrated. Contrast medium was noted to lie lateral to the oesophagus within the thorax (Fig. 2). There was no demonstrable contrast medium within the abdominal cavity, and no free air was noted under the hemidiaphragms. At surgery the mediastinum was explored for its entire length via a right posterior lateral thoracotomy. The oesophagus was intact and the nasogastric tube was found to be entering the mediastinum from the neck, and was outside for its entire thoracic course. A small amount of cloudy fluid was Address reprint requests to: Eric N. Faerber, MD Pediatric Radiology, Box 800, Boston Floating Hospital, New England Medical Center, 171 Harrison Avenue, Boston, Massachusetts 02111. 40
Fig. la - Anteroposterior view of the chest and abdomen demonstrating the nasogastric tube tip (arrow) at the level of L3.
582
CLINICAL R A D I O L O G Y
Fig. lb - Lateral view of the chest and abdomen demonstrating the nasogastric tube tip (arrow) at the level of L3.
repair. The postoperative course was uneventful. The patient remains asymptomatic at the age of 12 weeks. Case 2. A six-year-old boy presented with an upper respiratory tract infection. A posteroanterior (PA) chest radiograph demonstrated unusual prominence of the posterior mediastinal junction line and right para-oesophageal line (Fig 3a). On the lateral view a thin white line was noted extending from the cervical region to the diaphragm, parallel to the posterior wall of the oesophagus (Fig. 3b). Study of the patient's past history revealed that birth was by breech delivery, as the first of a set of twins. In the immediate neonatal period the ,patient had respiratory distress and was started on respiratory care. At seven days of age, a nasogastric tube was inserted for feeding purposes but there was difficulty in the tube placement. An upper gastrointestinal series was subsequently performed because of intermittent vomiting but no abnormalities were detected. Chest radiographs at the age of four months, demonstrated radiographic contrast medium in the mediastinum with two superior parallel lines and a more inferior transverse line crossing over the arch of the aorta and extending inferiorly, adjacent to the right oesophageal wall. Tliere was
Fig. 2 - Oesophagram demonstrating the extravasation of water soluble contrast medium lateral to the proximal oesophagus (upper arrow) and distal oesophagus (lower arrow), also a thin sliver of contrast medium adjacent to the medial wall of the left lung superiorly (Figs 4a, b). A film taken 20 min after the administration of 2 oz of water showed no clearing of contrast medium. Subsequent chest films taken at the age of 19 months demonstrated the contrast medium in the upper portion of the mediastinum to have merged at the level of the second posterior rib with two streaks remaining more superiorly, including the visualised lower cervical region (Fig. 5).
U N U S U A L M A N I F E S T A T I O N S OF N E O N A T A L P H A R Y N G E A L P E R F O R A T I O N
(a)
583
(b)
Fig. 3a, b - Age six years. (a) Postero-anterior view showing thickened posterior junction line and right para-oesophageal line
(arrowheads). (b) Theselines are never normallyvisibleon the lateralview (arrows). Based on the radiographic findings of persistence of barium in the para-oesophageal tissues and the neonatal history we concluded that an unsuspected traumatic perforation of the oesophagus had occurred with subsequent spontaneous healing.
due to the increased need to pass nasogastric tubes for diagnostic or therapeutic purposes (Lee and Kuhn, 1976). Perforation generally occurs at the pharyngooesophageal junction. The oesophageal introitus is the narrowest region of the oesophagus because of the DISCUSSION action of cricopharyngeus muscle (lee and Kuhn, 1976). Injury following instrumentation most Since the first report of pharyngeal perforation by commonly occurs at this point when th~ oesophageal Ekl6f et aL (1969) further small series have been wall is compressed against a cervical vertebra (Loop reported by Girdany et al. (1969); Astley and and Groves, 1970). Roberts (1970); Loop and Groves (1970), Ducharme Cricopharyngeal spasm caused by injury to the et al. (1971); Edison and Hollinger (1973); Wells et posterior pharyngeal wall has been postulated by al. (1974); de Espinosa and de Paredes (1974); Lynch Girdany e t al. (1969) to precede perforation of this et al. (1974); Lee and Kulm (1976) and Touloukian area. The spasm is considered to produce difficulty in etal. (1977). passage of a tube and is thus responsible for perTraumatic perforation of the pharynx or oeso- foration. Breech presentation has been associated phagus tends to occur in premature infants probably with pharyngeal perforation (Girdany et al. 1969;
584
CLINICAL RADIOLOGY
(a)
(b)
Fig. 4a, b - Age four months. (a) Following upper GI series. Barium is seen in the mediastinum surrounding upper oesophagus and right side of lower haft of oesophagus. Barium tracks to right because of aortic arch. (b) Lateral view showing barium
posterior to oesophagus. Wells et al. 1974). The obstetrician in delivering the diagnosis will be confirmed by plain film radiographs aftercoming head of a breech presentation may insert of the chest and abdomen and contrast medium his finger into the infant's mouth and tear the examinations. The lateral projection is particularly pharyngeal wall. Injury may also result from a suction helpful. On radiographic examination the presence of a catheter during clearing of secretions from the nasopharynx. Suctioning of the infant's mouth and pneumomediastinum and especially subcutaneous pharynx after birth is occasionally performed with a emphysema of the neck in a patient with suspected rigid tube in a hurry and with the neck extended, a oesophageal atresia should suggest the possibility of position which may increase the risk of perforation an underlying perforation (Wells et al. 1974). Arrest of a catheter in the proximal oesophagus is consistent during instrumentation (Ekl6f et al., 1969). The clinical presentation is most often that of with oesophageal atresia. Lateral radiographs in upper oesophageal obstruction with excessive saliva- oesophageal atresia, however, demonstrate an air tion, choking, coughing, stridor, cyanosis and filled dilated blind upper pouch. An air-filled pouch inability to pass a catheter into the stomach. These has not been observed in a pseudodiverticulum. A small volume of barium introduced into the findings may be indistinguishable from those associated with oesophageal atresia, with or without upper pouch of an atretic oesophagus to confirm the tracheo-oesophageal fistula. Failure to pass a naso- diagnosis of atresia is easily aspirated back into the gastric tube may, however, be an unreliable indicator syringe almost in its entirety; failure to retrieve this of oesophageal obstruction. There may be dissection barium has been found to be a good indication of an and formation of false passage (pseudodiverticulum) existing pseudodiverticulum. The following features have also been found useextending from the hypopharynx along the variable length of the oesophagus (Wells et al. 1974). The ful by Ducharme et al. (1971) in the differential
UNUSUAL MANIFESTATIONS OF NEONATAL PHARYNGEAL PERFORATION
585
described with the subsequent development of a pneumopericardium (Touloukian etal. 1977). It must be emphasised, however, that despite a low position o f the catheter tube, perforation almost always occurs at a higher level, at or above the pharyngo-oesophageal junction (as demonstrated in the first case described here). Confirmation o f the perforation will be obtained by the injection of water soluble contrast m e d i u m under fluoroscopy. The volume of contrast m e d i u m used should be limited because o f the possibility o f over-tilling the false tract, with subsequent aspiration of contrast material into the tracheo-bronchial tree (Ducharme et al. 1971). The second case emphasises that the condition m a y be more common than is currently thought. Many cases undoubtedly remain undetected. Acknowledgement. We wish to acknowledge the assistance of Joseph A. Stetz, MD, chief resident in Pediatric Surgery, Boston Floating Hospital, for the preparation of the surgical notes of Case 1. REFERENCES
Fig. 5 - Nineteen months of age. The upper columns of barium join at the T2 level (arrowhead), probably secondary to growth of the lungs.
diagnosis between the two conditions. The opacified tract is usually elongated and more irregular with pharyngeal perforation, and there is an absence of tracheal compression. Atypical location of the catheter tip within the chest or abdomen on plain film studies may alert the physicians to the diagnosis of pharyngeal perforation in a newborn infant. An i m p o r t a n t finding indicative of perforation is a position of the catheter to the left of the oesophagus as it approaches the diaphragm, as seen on the frontal projection (Touloukian et al. 1977). A single case of pericardial perforation has been
Astley, R. & Roberts, K. D. (1970). Intubation perforation of the oesophagus in the newborn baby. British Journal of Radiology, 43, 219-223. de Espinosa, H. & de Paredes, C. G. (1974). Traumatic perforation of the pharynx in a newborn baby. Journal of Pediatric Surgery, 9, 247-248. Ducharme, J. C., Bertrand, R. & Debie, J. (1971). Perforation of the pharynx in the newborn. A condition mimicking esophageal atresia. Canadian Medical Association Journal, 104, 785-787. Edison, B. & HoUinger, P. (1973). Traumatic pharyngeal opseudodiverticulum in the newborn infant. Journal of Pediatrics, 82, 483-485. Ekl6f, O., Lohr, G. & Okian, L. (1969). Submucosal perforation of the oesophagus and the neonate. Acta radiologica, 8, 187-192. Girdany, B. R., Sieber, W. K. & Osman, M. Z. (1969). Traumatic pseudodiverticulums of the pharynx in newborn infants. New England Journal of Medicine, 280, 237240. Lee, S. B. & Kuhn, J. P. (1976). Oesophageal perforation in the neonate. American Journal of Children, 130, 325329. Loop, F. D. & Grooves, L. K. (1970). Oesophageal perforations. Annals of Thoracic Surgery, 10, 571-587. Lynch, F. P., Coran, A. G., Cohen, S. R. & Lee, F. A. (1974). Traumatic oesophageal pseudodiverticula in the newborn. Journal of Pediatric Surgery, 9, 675-681. qouloukian, R. J., Beardsley, G. P., Ablow, R. C. & Effmal,, E. L. (1977). Traumatic perforation of the pharynx in the newborn. Pediatrics, 59, SuppL 6 Part 2, 1019-1022. Wells, S. D., Leonidas, J. C., Conkle, D., Holder, T. M., Amoury, R. A. & Ashcraft, K. W. (1974). Traumatic prevertebral pharyngoesophageal pseudodiverticulum in the newborn infant. Journal of Pediatric Surgery, 9, 217 -222.
0009-9260/80/00830581502.00
Llnusual Manifestations of Neonatal Pharyngeal perforation ~RIC N. F A E R B E R * , A L A N M. S C H W A R T Z * , LEWIS W. P I N C H t and J O H N C. L E O N I D A S *
,Department of Pediatric Radiology, Boston Floating Hospital, Tufts New England Medical Center Hospital, Massachusetts, and ~Matthew Thornton Health Plan, Nashua, New Hampshire, U.S.A. The small series o f previously described cases o f n e o n a t a l pharyngeal perforation are reviewed and two additional cases w i t h atypical initial presentations are described. The c o n d i t i o n is more c o m m o n than is currently appreciated, as suggested by one o f our cases which r e m a i n e d u n d e t e c t e d until m a n y years later. We also wish to emphasise the high l o c a t i o n o f perforation despite an initial presentation which m a y appear to the contrary.
Traumatic perforation o f the p h a r y n x in the n e o n a t a l period is rare. The c o n d i t i o n m a y simulate oesophageal atresia b y presenting as a high obstruction to the passage o f a feeding tube. Two cases are described, b o t h w i t h atypical initial presentations. The invariably high l o c a t i o n of perforation site is emphasised, as this has n o t received adequate a t t e n t i o n in the literature.
found in the chest which culture proved sterile. Chest tube drainage of the mediastinum was instituted. The retro-oesophageal space in the neck was then explored through the left cervical approach, and the tube was found to be behind the entire cervical length of the oesophagus with the site of perforation being in the hypopharynx. The neck was drained with a small Penrose drain and the rent in the hypopharynx was allowed to heal without
CASE REPORT Case 1. The patient was born after a 28 week gestation to a 19-year-old mother who had received inadequate prenatal care. The labour was uncomplicated; birth was by vaginal delivery with a vertex presentation. The infant breathed spontaneously but subsequently developed mild respiratory distress, and an abdominal radiograph demonstrated the catheter tube tip within the stomach. Repeat passage of nasogastric tube was accomplished with difficulty. Chest and abdominal radiographs revealed right middle lobe volume loss. The nasogastric tube tip was noted to lie posteriorly at the level of L3 (Figs la, b). This low position of the catheter suggested perforation of the oesophagus at the intrathoracic level. An oesophagram was performed through a second nasogastric tube, using water soluble contrast medium. A high site of perforation within the pharynx was demonstrated. Contrast medium was noted to lie lateral to the oesophagus within the thorax (Fig. 2). There was no demonstrable contrast medium within the abdominal cavity, and no free air was noted under the hemidiaphragms. At surgery the mediastinum was explored for its entire length via a right posterior lateral thoracotomy. The oesophagus was intact and the nasogastric tube was found to be entering the mediastinum from the neck, and was outside for its entire thoracic course. A small amount of cloudy fluid was Address reprint requests to: Eric N. Faerber, MD Pediatric Radiology, Box 800, Boston Floating Hospital, New England Medical Center, 171 Harrison Avenue, Boston, Massachusetts 02111. 40
Fig. la - Anteroposterior view of the chest and abdomen demonstrating the nasogastric tube tip (arrow) at the level of L3.
582
CLINICAL R A D I O L O G Y
Fig. lb - Lateral view of the chest and abdomen demonstrating the nasogastric tube tip (arrow) at the level of L3.
repair. The postoperative course was uneventful. The patient remains asymptomatic at the age of 12 weeks. Case 2. A six-year-old boy presented with an upper respiratory tract infection. A posteroanterior (PA) chest radiograph demonstrated unusual prominence of the posterior mediastinal junction line and right para-oesophageal line (Fig 3a). On the lateral view a thin white line was noted extending from the cervical region to the diaphragm, parallel to the posterior wall of the oesophagus (Fig. 3b). Study of the patient's past history revealed that birth was by breech delivery, as the first of a set of twins. In the immediate neonatal period the ,patient had respiratory distress and was started on respiratory care. At seven days of age, a nasogastric tube was inserted for feeding purposes but there was difficulty in the tube placement. An upper gastrointestinal series was subsequently performed because of intermittent vomiting but no abnormalities were detected. Chest radiographs at the age of four months, demonstrated radiographic contrast medium in the mediastinum with two superior parallel lines and a more inferior transverse line crossing over the arch of the aorta and extending inferiorly, adjacent to the right oesophageal wall. Tliere was
Fig. 2 - Oesophagram demonstrating the extravasation of water soluble contrast medium lateral to the proximal oesophagus (upper arrow) and distal oesophagus (lower arrow), also a thin sliver of contrast medium adjacent to the medial wall of the left lung superiorly (Figs 4a, b). A film taken 20 min after the administration of 2 oz of water showed no clearing of contrast medium. Subsequent chest films taken at the age of 19 months demonstrated the contrast medium in the upper portion of the mediastinum to have merged at the level of the second posterior rib with two streaks remaining more superiorly, including the visualised lower cervical region (Fig. 5).
U N U S U A L M A N I F E S T A T I O N S OF N E O N A T A L P H A R Y N G E A L P E R F O R A T I O N
(a)
583
(b)
Fig. 3a, b - Age six years. (a) Postero-anterior view showing thickened posterior junction line and right para-oesophageal line
(arrowheads). (b) Theselines are never normallyvisibleon the lateralview (arrows). Based on the radiographic findings of persistence of barium in the para-oesophageal tissues and the neonatal history we concluded that an unsuspected traumatic perforation of the oesophagus had occurred with subsequent spontaneous healing.
due to the increased need to pass nasogastric tubes for diagnostic or therapeutic purposes (Lee and Kuhn, 1976). Perforation generally occurs at the pharyngooesophageal junction. The oesophageal introitus is the narrowest region of the oesophagus because of the DISCUSSION action of cricopharyngeus muscle (lee and Kuhn, 1976). Injury following instrumentation most Since the first report of pharyngeal perforation by commonly occurs at this point when th~ oesophageal Ekl6f et aL (1969) further small series have been wall is compressed against a cervical vertebra (Loop reported by Girdany et al. (1969); Astley and and Groves, 1970). Roberts (1970); Loop and Groves (1970), Ducharme Cricopharyngeal spasm caused by injury to the et al. (1971); Edison and Hollinger (1973); Wells et posterior pharyngeal wall has been postulated by al. (1974); de Espinosa and de Paredes (1974); Lynch Girdany e t al. (1969) to precede perforation of this et al. (1974); Lee and Kulm (1976) and Touloukian area. The spasm is considered to produce difficulty in etal. (1977). passage of a tube and is thus responsible for perTraumatic perforation of the pharynx or oeso- foration. Breech presentation has been associated phagus tends to occur in premature infants probably with pharyngeal perforation (Girdany et al. 1969;
584
CLINICAL RADIOLOGY
(a)
(b)
Fig. 4a, b - Age four months. (a) Following upper GI series. Barium is seen in the mediastinum surrounding upper oesophagus and right side of lower haft of oesophagus. Barium tracks to right because of aortic arch. (b) Lateral view showing barium
posterior to oesophagus. Wells et al. 1974). The obstetrician in delivering the diagnosis will be confirmed by plain film radiographs aftercoming head of a breech presentation may insert of the chest and abdomen and contrast medium his finger into the infant's mouth and tear the examinations. The lateral projection is particularly pharyngeal wall. Injury may also result from a suction helpful. On radiographic examination the presence of a catheter during clearing of secretions from the nasopharynx. Suctioning of the infant's mouth and pneumomediastinum and especially subcutaneous pharynx after birth is occasionally performed with a emphysema of the neck in a patient with suspected rigid tube in a hurry and with the neck extended, a oesophageal atresia should suggest the possibility of position which may increase the risk of perforation an underlying perforation (Wells et al. 1974). Arrest of a catheter in the proximal oesophagus is consistent during instrumentation (Ekl6f et al., 1969). The clinical presentation is most often that of with oesophageal atresia. Lateral radiographs in upper oesophageal obstruction with excessive saliva- oesophageal atresia, however, demonstrate an air tion, choking, coughing, stridor, cyanosis and filled dilated blind upper pouch. An air-filled pouch inability to pass a catheter into the stomach. These has not been observed in a pseudodiverticulum. A small volume of barium introduced into the findings may be indistinguishable from those associated with oesophageal atresia, with or without upper pouch of an atretic oesophagus to confirm the tracheo-oesophageal fistula. Failure to pass a naso- diagnosis of atresia is easily aspirated back into the gastric tube may, however, be an unreliable indicator syringe almost in its entirety; failure to retrieve this of oesophageal obstruction. There may be dissection barium has been found to be a good indication of an and formation of false passage (pseudodiverticulum) existing pseudodiverticulum. The following features have also been found useextending from the hypopharynx along the variable length of the oesophagus (Wells et al. 1974). The ful by Ducharme et al. (1971) in the differential
UNUSUAL MANIFESTATIONS OF NEONATAL PHARYNGEAL PERFORATION
585
described with the subsequent development of a pneumopericardium (Touloukian etal. 1977). It must be emphasised, however, that despite a low position o f the catheter tube, perforation almost always occurs at a higher level, at or above the pharyngo-oesophageal junction (as demonstrated in the first case described here). Confirmation o f the perforation will be obtained by the injection of water soluble contrast m e d i u m under fluoroscopy. The volume of contrast m e d i u m used should be limited because o f the possibility o f over-tilling the false tract, with subsequent aspiration of contrast material into the tracheo-bronchial tree (Ducharme et al. 1971). The second case emphasises that the condition m a y be more common than is currently thought. Many cases undoubtedly remain undetected. Acknowledgement. We wish to acknowledge the assistance of Joseph A. Stetz, MD, chief resident in Pediatric Surgery, Boston Floating Hospital, for the preparation of the surgical notes of Case 1. REFERENCES
Fig. 5 - Nineteen months of age. The upper columns of barium join at the T2 level (arrowhead), probably secondary to growth of the lungs.
diagnosis between the two conditions. The opacified tract is usually elongated and more irregular with pharyngeal perforation, and there is an absence of tracheal compression. Atypical location of the catheter tip within the chest or abdomen on plain film studies may alert the physicians to the diagnosis of pharyngeal perforation in a newborn infant. An i m p o r t a n t finding indicative of perforation is a position of the catheter to the left of the oesophagus as it approaches the diaphragm, as seen on the frontal projection (Touloukian et al. 1977). A single case of pericardial perforation has been
Astley, R. & Roberts, K. D. (1970). Intubation perforation of the oesophagus in the newborn baby. British Journal of Radiology, 43, 219-223. de Espinosa, H. & de Paredes, C. G. (1974). Traumatic perforation of the pharynx in a newborn baby. Journal of Pediatric Surgery, 9, 247-248. Ducharme, J. C., Bertrand, R. & Debie, J. (1971). Perforation of the pharynx in the newborn. A condition mimicking esophageal atresia. Canadian Medical Association Journal, 104, 785-787. Edison, B. & HoUinger, P. (1973). Traumatic pharyngeal opseudodiverticulum in the newborn infant. Journal of Pediatrics, 82, 483-485. Ekl6f, O., Lohr, G. & Okian, L. (1969). Submucosal perforation of the oesophagus and the neonate. Acta radiologica, 8, 187-192. Girdany, B. R., Sieber, W. K. & Osman, M. Z. (1969). Traumatic pseudodiverticulums of the pharynx in newborn infants. New England Journal of Medicine, 280, 237240. Lee, S. B. & Kuhn, J. P. (1976). Oesophageal perforation in the neonate. American Journal of Children, 130, 325329. Loop, F. D. & Grooves, L. K. (1970). Oesophageal perforations. Annals of Thoracic Surgery, 10, 571-587. Lynch, F. P., Coran, A. G., Cohen, S. R. & Lee, F. A. (1974). Traumatic oesophageal pseudodiverticula in the newborn. Journal of Pediatric Surgery, 9, 675-681. qouloukian, R. J., Beardsley, G. P., Ablow, R. C. & Effmal,, E. L. (1977). Traumatic perforation of the pharynx in the newborn. Pediatrics, 59, SuppL 6 Part 2, 1019-1022. Wells, S. D., Leonidas, J. C., Conkle, D., Holder, T. M., Amoury, R. A. & Ashcraft, K. W. (1974). Traumatic prevertebral pharyngoesophageal pseudodiverticulum in the newborn infant. Journal of Pediatric Surgery, 9, 217 -222.