International Journal of Pediatric Otorhinolaryngology 78 (2014) 5–9
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Review Article
Pharyngeal perforation after blunt cervical trauma in child Mariana Svetlikova a, Ivo Starek b, Michaela Spenerova a, Jan Potesil a, Igor Sulla c, Csaba Hucko b, Vladimir Mihal a, Vladimir Balik d,* a
Department of Pediatrics, University Hospital Olomouc and Faculty of Medicine and Dentistry, Palacky University, I. P. Pavlova 6, 775 20 Olomouc, Czech Republic b Department of Otorhinolaryngology, University Hospital Olomouc and Faculty of Medicine and Dentistry, Palacky University, I. P. Pavlova 6, 775 20 Olomouc, Czech Republic c Department of Surgery, Hospital of Slovak Railways and Institute of Neurobiology, Slovak Academy of Sciences, Soltesovej 4, 040 01 Kosice, Slovak Republic d Department of Neurosurgery, University Hospital Olomouc and Faculty of Medicine and Dentistry, Palacky University, I. P. Pavlova 6, 775 20 Olomouc, Czech Republic
A R T I C L E I N F O
A B S T R A C T
Article history: Received 2 September 2013 Accepted 6 October 2013 Available online 15 November 2013
Pharyngeal perforation caused by non-penetrating cervical trauma is an extremely rare clinical entity both in adults and children. Data concerning management of this type of injury are quite rare in surgical and even scarcer in pediatric literature. Since delay in treatment may be associated with life-threatening complications, prompt diagnosis coupled with appropriate therapy is essential for achieving favorable clinical outcome. To the best of authors’ knowledge, the present study illustrates for the first time the experience with successful treatment of pharyngeal perforation caused by a blunt cervical trauma in a child. ß 2013 Elsevier Ireland Ltd. All rights reserved.
Keywords: Pharyngeal perforation Blunt non-penetrating cervical trauma Pediatrics
Contents 1. 2. 3.
4.
Introduction . . . . . . . . . . . . . . . . . . . . . . . Case report . . . . . . . . . . . . . . . . . . . . . . . . Discussion . . . . . . . . . . . . . . . . . . . . . . . . Incidence . . . . . . . . . . . . . . . . . . . . 3.1. 3.2. Etiology . . . . . . . . . . . . . . . . . . . . . Pathophysiological mechanism . . 3.3. Clinical presentation . . . . . . . . . . . 3.4. Diagnostic method consideration. 3.5. 3.6. Treatment . . . . . . . . . . . . . . . . . . . Follow-up . . . . . . . . . . . . . . . . . . . 3.7. Conclusion . . . . . . . . . . . . . . . . . . . . . . . . References . . . . . . . . . . . . . . . . . . . . . . . .
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1. Introduction
* Corresponding author. Tel.: +420 775 031 127/421 903 890 652; fax: +420 58 844 2539. E-mail addresses: [email protected] (M. Svetlikova), [email protected] (I. Starek), [email protected] (M. Spenerova), [email protected] (J. Potesil), [email protected] (I. Sulla), [email protected] (C. Hucko), [email protected] (V. Mihal), [email protected] (V. Balik). 0165-5876/$ – see front matter ß 2013 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijporl.2013.10.060
Rupture of the pharyngo-esophageal (PE) region following nonpenetrating blunt cervical trauma (BCT) is a rare phenomenon in adults and extremely rare in pediatric practice - only two pediatric cases including the one present here have been reported, so far (Table 1) [1]. The injury may lead to serious consequences in terms of morbidity and mortality, especially if not diagnosed and treated timely [2–8] with reported mortality rate for unrecognized injury as high as 92% [9]. The size and location of the perforation of
M. Svetlikova et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 5–9 Excellent No No Amoxicillin and clavulan acid 30 mg/kg every 8 hours four days intravenously and subsequently ten days per os No Computed tomography of the neck, chest X-ray, bronchoscopy, indirect laryngoscopy Fall out of bed
pharynx is crucial for determining appropriate treatment strategy. The infrequent occurrence of this high-risk injury in children; diagnostic embarrassment arising from the recognition of especially small defect; a fact that the vast majority of published work deals with this issue from the perspective of adults; and lack of the long-term follow-up of patients with pharyngeal perforation (PP); all these prompt the authors to report on their experience in management of this type of injury in child, and to review the existing literature pertinent to this unique injury. 2. Case report A four and half-year-old boy while climbing out of bed stumbled on a duvet cover and sustained a blunt injury to the neck and face with gradual development of dysphagia and odynophagia without any respiratory problems or hoarseness. The patient was examined by traumatologist at a district hospital. Only excoriations on the right chin and right hip were revealed. Subcutaneous emphysema, weakness, sensory deficit or absent deep tendon reflexes were not observed as well as no stridor was auscultated on the neck or chest. An initial computed tomography (CT) investigation of the neck showed air strip in the retropharyngeal soft tissue without fracture of bone structures or irregularity of the pharyngeal wall (Fig. 1). The patient was transferred by ground ambulance to intensive care unit of our pediatric department to rule out suspected rupture of trachea. Upon arrival, the child was cardiopulmonary stabilized, vital signs were unremarkable, breathing of room air did not cause respiratory problems. A rigid cervical collar was put on. Indirect laryngoscopy revealed only a small hematoma in the region of left palatopharyngeal arch. Additionally, chest X-ray and bronchoscopy did not show pneumomediastinum or rupture of the trachea and bronchi, respectively, therefore carrying on with conservative management was recommended. Since a history of the child indicated atrial and ventricular septal defects the administration of antibiotics was started (amoxicillin and clavulan acid 30 mg/kg every 8 h four days intravenously and subsequently ten days per os) and oral feeding was withheld (nil per os) for 48 h. Esophagogram recommended by surgeon could not be performed due to absence of sufficient cooperation from the child. On the 2nd postinjury day a follow-up lateral cervical X-ray confirmed the extension of prevertebral space along with the presence of air reaching from the C1 to the PE borderline, however a vague brightening was spreading to the C6-7 level (Fig. 2). The diagnosis of a small PP was established. Because of the favorable clinical course, oral administration of liquid supplements was commenced the next day. On the 4th postinjury day a follow-up X-ray showed
M/4.5 Present case
Excoriation on the chin and hip, gradual development of dysphagia and odynophagia
Broad-spectrum intravenous antibiotics 5 cm laceration of the posterior pharyngeal wall Yes Cervical and chest X-ray, duplex ultrasound of the neck, flexible laryngoscopy, direct laryngoscopy and esophagoscopy, gastrografin swallow at ten days post-injury Fall from motorcycle at low speed, impacting the left side of neck on the end of his handlebar Christey M/16 2005
Haemoptysis, contusion to the left side of his neck, hoarse voice, minor subcutaneous emphysema on the left side of the neck, vomiting, coughing
No No 2 A small hematoma in the region of left palatopharyngeal arch
Excellent No Yes Yes 9
No
Tracheostomy Surgery Complications Diagnostic methods Clinical presentation Pathophysiological mechanism Gender/age at diagnosis (years) Case
Table 1 Pharyngeal perforation following blunt neck injury in children reported in the literature.
Intubation Lesion
Nil per Nasogastric Antibiotics os (days) tube
Outcome
6
Fig. 1. Initial cervical CT scan demonstrating air strip in the retropharyngeal area.
M. Svetlikova et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 5–9
7
and finally due to penetrating or BCT [2–7,12–15]. Neonatal perforations are associated with utilization of a rigid suction catheter [16], nasogastric [17] or endotracheal tube placement [18], and digital trauma during resuscitation [19]. Iatrogenic perforation has more favorable prognosis than injury caused by blunt mechanism as the later is often associated with more extensive ruptures, often intervening intrathoracic part of esophagus [7,20,21]. 3.3. Pathophysiological mechanism
Fig. 2. A cervical side X-ray showing the air strip in the prevertebral cervical region (2nd postinjury day).
regression of the air strip at the prevertebral cervical region (Fig. 3) therefore a soft oral feeding was started and the patient was transferred to the ward. During the entire hospitalization the patient was stable, without elevated body temperature and his oxygen saturation remained 98% on room air. Dysphagia and odynophagia gradually subsided and eventually disappeared. The patient was discharged from the hospital well tolerating oral feeds on the 8th postinjury day. Three months later an outpatient followup demonstrated favorable clinical state of the child without development of any complications. 3. Discussion 3.1. Incidence Overall, the PE perforation following BCT constitutes less than 2% of all perforations of this region [10]. Over the past nearly 40 years (1970–2009) there were reported only 22 such cases in English literature [11] with one case observed in sixteen years old teenager [1]. 3.2. Etiology PP may be caused iatrogenically following intubation, endoscopy, transesophageal echocardiography or surgery. The rupture may also occur after radiotherapy, ingestion of corrosive substances or as a postemetic rupture as seen in Boerhaave syndrome,
Several theories try to explain the mechanism by which PP after BCT arises. The most frequently mentioned mechanism is perforation of the pharynx due to barotraumas which encounter in traffic accident. There is a whiplash mechanism of cervical spine along with closure of airways due to an impact of the upper region of the neck against the edge of the steering wheel and forced expiration caused by the concurrent compression of the chest [6]. The frequency of this type of injury decreased after the introduction of seat belts [10]. Another theory supposes that the PP arises due to anterior cervical spurs, posterior edge of the thyroid lamina or hyoid fracture while hyperextended vertebral bodies of the cervical spine [2–4,6]. In the sole reported pediatric case [1], the barotraumas mechanism seems to be less likely. In the setting of hyperextended cervical spine while falling from motorcycle at low speed, actual direct external blunt impact neck on the end of its handlebar compressing the pharynx directly against cervical spine could participate in the injury more likely. Predisposing factor for rupture of the pharynx may also be the absence of longitudinal fibers between mucosal and serous layer of the inferior pharyngeal constrictor muscle of hypopharyngoesophageal transition zone, the region commonly perforated during instrumentation, or more rarely secondary to blast injuries or in the postemetic setting [22]. The transition zone is well known as Killian‘s dehiscence [6,7]. In the present case it seems to be a reasonable assumption to explain the PP just by barotraumas due to suspected blow to the chest as indirectly indicated excoriation on his right hip as well. Due to ligamentous laxity together with a high head to body weight ratio, immaturity of paraspinal muscles and the underdeveloped uncinate processes underlie cervical spine of child to be prone to greater hyperextension [23] which can cause significant strain and tension PE structures over cervical spine ultimately resulting in their thinning and thereby weakening and subsequent forced expiration could easily cause their rupture. In addition, anterior edge of conical pattern of pediatric cervical vertebral bodies may work as stiffer subject and finally may cause PP in extremely hyperextension setting of cervical spine along with current push of pharynx against the cervical spine caused by external blunt impact to the neck region. Clinical course of patients with whiplash-associated disorder may range from no complication to weakness, sensory deficit or absent deep tendon reflexes [24,25] even spinal cord injury without radiographic abnormality (SCIWORA). The age range of children with SCIWORA is 1.5–16 years while much higher incidence is in younger than 9 years [26]. Since our patient was without any neurological deficit, magnetic resonance imaging through the cervical region was not indicated neither initially nor 3 months later. 3.4. Clinical presentation
Fig. 3. A follow-up cervical side X-ray demonstrating regression of the emphysema in the retropharynfgeal region (4th postinjury day).
Symptoms indicating this rare injury include pain in cervical or thoracic region, contusion and excoriation of anterior part of neck or odynophagia and dysphagia as observed in the present case. The patient may experience only sore throat, dyspnea, stridor or hoarseness [1,6,8,14,15,27,28]. Furthermore, expectoration of bloody saliva or feeling of pressure in throat was reported as well
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[1,27]. Palpation can reveal submandibular hematoma, crepitating [8], fluctuation or inflammatory swelling, sometimes laryngeal dislocation [6,14,15]. On the other hand, the clinical course may be asymptomatic in the first 24 [27] to 48 h [7]. Interestingly, isolated PE ruptures were observed in approximately half of all cases with traumatic subcutaneous emphysema [22]. 3.5. Diagnostic method consideration The lateral cervical X-ray demonstrating air strip in retropharyngeal space or subcutaneous emphysema raises the suspicion of PP significantly [6,14,15,20,29] as well as chest X-ray showing mediastinal or pericardial emphysema [14,15] in the presence of the symptoms described above [28]. A few cases documented that air strip in retropharyngeal space along with clinical picture are satisfactory for making of definitive diagnosis and calling for appropriate treatment [7,20,30]. A direct/indirect laryngoscopy and esophagoscopy represent additional diagnostic possibilities. Fiberoptic examination is diagnostic method of choice in order to obtain information about the presence, site, and extent of pharyngeal tear. Rigid endoscopy gives a superior view but requires a general anesthesia and can be impractical in the setting of an immobilized cervical spine [28] as well as in pediatric patients. On the base of our experience we believe that in child with favorable clinical course after BCT indirect laryngoscopy is all that should be done especially when CT investigation was performed initially. Axial CT scan of cervical region may also show pharyngeal emphysema, crack aryepiglotic hematoma or fracture of bone structures or cartilaginous lesions, pathological processes in parapharyngeal and laryngeal space [8,22,27]. Irregular walls of the pharynx on CT scans should elicit a strong suspicion of PP as well [8]. Although these investigations may even fail to detect the rupture of the pharynx wall, especially when the defect is small [27], laryngoscopy may reveal indirect signs such as reduced mobility of voice ligaments and edema or hematoma of pharyngo-laryngeal area [4–6,20,27,30] as observed in the present case. If diagnostic uncertainty persists or clinical deterioration develops throughout the course one should consider an esophagogram using barium paste [27] or gastrografin [31] in the lateral/ supine decubitus position which may increase its sensitivity [27]. Esophagogram may reveal leakage of contrast medium into the retropharyngeal region in the upper cervical region [27]. Sensitivity of this method is 90% for esophageal rupture while for lesions localized in the pharynx it decreases to 60–75%, however its specificity in this location amounts to 100% [12,14,15,32,33]. Overall, there is little consensus about the best choice of diagnostic approach in the literature, therefore a combination of several diagnostic methods (e.g. X-ray, direct/indirect laryngoscopy, flexible fiberoptic nasopharyngolaryngoscopy, flexible bronchoscopy and esophagoscopy, CT investigation and esophagogram) may be necessary in peculiar cases [22,27]. These diagnostic measures are especially indicated in patients revealing clinical deterioration [1] when suspicion of (a) esophagus involvement or (b) larger PP demands surgical intervention. 3.6. Treatment Non-surgical strategy is reserved for cases where the lesion is smaller than 2 cm and only affects the pharynx while the airway is felt to be secure. Broad-spectrum antibiotics are administered with respect to the normal microbial flora of the oropharynx and esophagus (penicillin series) [7,21]. In the case of gram-negative oropharyngeal colonization addition of a gentamicin is desirable [21]. The success of treatment was recorded even with combination of 2nd generation cephalosporin and metronidazole [11]. The
patient is nourished by nasogastric tube and administering liquid supplements per os should begin at 4th–6th posttraumatic day. However, as demonstrated our experience the placement of nasogastric tube can be avoided and a regime ‘‘nil per os’’ should be instituted. In generally, the beginning of oral food intake depends on the type, size, location of the perforation and the overall clinical condition of the patient [7,11,21]. Any conditions which increases airway pressure (coughing, vomiting, or sneezing) should be eliminated [34,35]. Niezgoda et al. [7] reported that four of the five patients treated conservatively recovered and perforation was healed without complications. Prompt surgical strategy should be applied in case the PP exceeds 2 cm or the lesion extends into the esophagus [7]. Niezgoda et al. [7] observed that delay of surgical treatment extending 12 h lead to development of paraesophageal abscess which required surgical management. However, some authors recommend more conservative approach [20,36] as documented in the case of teenager with 5 cm laceration of the posterior pharyngeal wall [1]. Surgical approach is commonly performed along the anterior sternocleidomastoid muscle for optimal exposure of the retropharyngeal space [6] and includes surgical exploration, debridement and suture closure of perforation [37]. A placement of adequate retropharyngoesophageal drain is necessary [7]. If sepsis, pyrexia or local swelling occurs and clinical picture deteriorates during conservative management, it is necessary to search for the possible development of cervical or mediastinal abscess, esophageal fistula, mediastinitis, pyo/pneumothorax, pneumoperitoneum, pneumopericard or pseudoaneurysm of the carotid artery. These complications need to be surgically treated without delay [1,4–7,14,21,28,34,35,38]. Any suspicion of impending respiratory difficulty due to airway trauma with resultant edema should lead to early airway management considering orotracheal intubation or less risky tracheostomy as airway edema and distorted neck anatomy secondary to blunt injury may lead to further airway injury during orotracheal intubation [1,22]. 3.7. Follow-up Follow-up cervical X-ray, esophagogram or CT scan depends on the type, size and location of PP, and on general clinical picture [21,27]. If the child is in good clinical condition, a cervical X-ray performed on the 4th postinjury day is all what should be done, especially when the procedure demonstrated disappearance of the air strip at the prevertebral cervical region or at least its substantial regression. 4. Conclusion Due to the low incidence of the traumatic PP in children, limited available data and the heterogeneous diagnostic and treatment modalities used, it makes difficult to draw any meaningful conclusions relating to diagnosis, treatment and prognosis of these patients. A greater number of patients are essential for selection of optimal therapeutical strategy. On the base of our experience and the reviewed literature, conservative approach seems to be appropriate in the setting of this type of injury in children. Conflict of interest The authors declare that they have no conflict of interest. Funding None.
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Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Review Article
Pharyngeal perforation after blunt cervical trauma in child Mariana Svetlikova a, Ivo Starek b, Michaela Spenerova a, Jan Potesil a, Igor Sulla c, Csaba Hucko b, Vladimir Mihal a, Vladimir Balik d,* a
Department of Pediatrics, University Hospital Olomouc and Faculty of Medicine and Dentistry, Palacky University, I. P. Pavlova 6, 775 20 Olomouc, Czech Republic b Department of Otorhinolaryngology, University Hospital Olomouc and Faculty of Medicine and Dentistry, Palacky University, I. P. Pavlova 6, 775 20 Olomouc, Czech Republic c Department of Surgery, Hospital of Slovak Railways and Institute of Neurobiology, Slovak Academy of Sciences, Soltesovej 4, 040 01 Kosice, Slovak Republic d Department of Neurosurgery, University Hospital Olomouc and Faculty of Medicine and Dentistry, Palacky University, I. P. Pavlova 6, 775 20 Olomouc, Czech Republic
A R T I C L E I N F O
A B S T R A C T
Article history: Received 2 September 2013 Accepted 6 October 2013 Available online 15 November 2013
Pharyngeal perforation caused by non-penetrating cervical trauma is an extremely rare clinical entity both in adults and children. Data concerning management of this type of injury are quite rare in surgical and even scarcer in pediatric literature. Since delay in treatment may be associated with life-threatening complications, prompt diagnosis coupled with appropriate therapy is essential for achieving favorable clinical outcome. To the best of authors’ knowledge, the present study illustrates for the first time the experience with successful treatment of pharyngeal perforation caused by a blunt cervical trauma in a child. ß 2013 Elsevier Ireland Ltd. All rights reserved.
Keywords: Pharyngeal perforation Blunt non-penetrating cervical trauma Pediatrics
Contents 1. 2. 3.
4.
Introduction . . . . . . . . . . . . . . . . . . . . . . . Case report . . . . . . . . . . . . . . . . . . . . . . . . Discussion . . . . . . . . . . . . . . . . . . . . . . . . Incidence . . . . . . . . . . . . . . . . . . . . 3.1. 3.2. Etiology . . . . . . . . . . . . . . . . . . . . . Pathophysiological mechanism . . 3.3. Clinical presentation . . . . . . . . . . . 3.4. Diagnostic method consideration. 3.5. 3.6. Treatment . . . . . . . . . . . . . . . . . . . Follow-up . . . . . . . . . . . . . . . . . . . 3.7. Conclusion . . . . . . . . . . . . . . . . . . . . . . . . References . . . . . . . . . . . . . . . . . . . . . . . .
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1. Introduction
* Corresponding author. Tel.: +420 775 031 127/421 903 890 652; fax: +420 58 844 2539. E-mail addresses: [email protected] (M. Svetlikova), [email protected] (I. Starek), [email protected] (M. Spenerova), [email protected] (J. Potesil), [email protected] (I. Sulla), [email protected] (C. Hucko), [email protected] (V. Mihal), [email protected] (V. Balik). 0165-5876/$ – see front matter ß 2013 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijporl.2013.10.060
Rupture of the pharyngo-esophageal (PE) region following nonpenetrating blunt cervical trauma (BCT) is a rare phenomenon in adults and extremely rare in pediatric practice - only two pediatric cases including the one present here have been reported, so far (Table 1) [1]. The injury may lead to serious consequences in terms of morbidity and mortality, especially if not diagnosed and treated timely [2–8] with reported mortality rate for unrecognized injury as high as 92% [9]. The size and location of the perforation of
M. Svetlikova et al. / International Journal of Pediatric Otorhinolaryngology 78 (2014) 5–9 Excellent No No Amoxicillin and clavulan acid 30 mg/kg every 8 hours four days intravenously and subsequently ten days per os No Computed tomography of the neck, chest X-ray, bronchoscopy, indirect laryngoscopy Fall out of bed
pharynx is crucial for determining appropriate treatment strategy. The infrequent occurrence of this high-risk injury in children; diagnostic embarrassment arising from the recognition of especially small defect; a fact that the vast majority of published work deals with this issue from the perspective of adults; and lack of the long-term follow-up of patients with pharyngeal perforation (PP); all these prompt the authors to report on their experience in management of this type of injury in child, and to review the existing literature pertinent to this unique injury. 2. Case report A four and half-year-old boy while climbing out of bed stumbled on a duvet cover and sustained a blunt injury to the neck and face with gradual development of dysphagia and odynophagia without any respiratory problems or hoarseness. The patient was examined by traumatologist at a district hospital. Only excoriations on the right chin and right hip were revealed. Subcutaneous emphysema, weakness, sensory deficit or absent deep tendon reflexes were not observed as well as no stridor was auscultated on the neck or chest. An initial computed tomography (CT) investigation of the neck showed air strip in the retropharyngeal soft tissue without fracture of bone structures or irregularity of the pharyngeal wall (Fig. 1). The patient was transferred by ground ambulance to intensive care unit of our pediatric department to rule out suspected rupture of trachea. Upon arrival, the child was cardiopulmonary stabilized, vital signs were unremarkable, breathing of room air did not cause respiratory problems. A rigid cervical collar was put on. Indirect laryngoscopy revealed only a small hematoma in the region of left palatopharyngeal arch. Additionally, chest X-ray and bronchoscopy did not show pneumomediastinum or rupture of the trachea and bronchi, respectively, therefore carrying on with conservative management was recommended. Since a history of the child indicated atrial and ventricular septal defects the administration of antibiotics was started (amoxicillin and clavulan acid 30 mg/kg every 8 h four days intravenously and subsequently ten days per os) and oral feeding was withheld (nil per os) for 48 h. Esophagogram recommended by surgeon could not be performed due to absence of sufficient cooperation from the child. On the 2nd postinjury day a follow-up lateral cervical X-ray confirmed the extension of prevertebral space along with the presence of air reaching from the C1 to the PE borderline, however a vague brightening was spreading to the C6-7 level (Fig. 2). The diagnosis of a small PP was established. Because of the favorable clinical course, oral administration of liquid supplements was commenced the next day. On the 4th postinjury day a follow-up X-ray showed
M/4.5 Present case
Excoriation on the chin and hip, gradual development of dysphagia and odynophagia
Broad-spectrum intravenous antibiotics 5 cm laceration of the posterior pharyngeal wall Yes Cervical and chest X-ray, duplex ultrasound of the neck, flexible laryngoscopy, direct laryngoscopy and esophagoscopy, gastrografin swallow at ten days post-injury Fall from motorcycle at low speed, impacting the left side of neck on the end of his handlebar Christey M/16 2005
Haemoptysis, contusion to the left side of his neck, hoarse voice, minor subcutaneous emphysema on the left side of the neck, vomiting, coughing
No No 2 A small hematoma in the region of left palatopharyngeal arch
Excellent No Yes Yes 9
No
Tracheostomy Surgery Complications Diagnostic methods Clinical presentation Pathophysiological mechanism Gender/age at diagnosis (years) Case
Table 1 Pharyngeal perforation following blunt neck injury in children reported in the literature.
Intubation Lesion
Nil per Nasogastric Antibiotics os (days) tube
Outcome
6
Fig. 1. Initial cervical CT scan demonstrating air strip in the retropharyngeal area.
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7
and finally due to penetrating or BCT [2–7,12–15]. Neonatal perforations are associated with utilization of a rigid suction catheter [16], nasogastric [17] or endotracheal tube placement [18], and digital trauma during resuscitation [19]. Iatrogenic perforation has more favorable prognosis than injury caused by blunt mechanism as the later is often associated with more extensive ruptures, often intervening intrathoracic part of esophagus [7,20,21]. 3.3. Pathophysiological mechanism
Fig. 2. A cervical side X-ray showing the air strip in the prevertebral cervical region (2nd postinjury day).
regression of the air strip at the prevertebral cervical region (Fig. 3) therefore a soft oral feeding was started and the patient was transferred to the ward. During the entire hospitalization the patient was stable, without elevated body temperature and his oxygen saturation remained 98% on room air. Dysphagia and odynophagia gradually subsided and eventually disappeared. The patient was discharged from the hospital well tolerating oral feeds on the 8th postinjury day. Three months later an outpatient followup demonstrated favorable clinical state of the child without development of any complications. 3. Discussion 3.1. Incidence Overall, the PE perforation following BCT constitutes less than 2% of all perforations of this region [10]. Over the past nearly 40 years (1970–2009) there were reported only 22 such cases in English literature [11] with one case observed in sixteen years old teenager [1]. 3.2. Etiology PP may be caused iatrogenically following intubation, endoscopy, transesophageal echocardiography or surgery. The rupture may also occur after radiotherapy, ingestion of corrosive substances or as a postemetic rupture as seen in Boerhaave syndrome,
Several theories try to explain the mechanism by which PP after BCT arises. The most frequently mentioned mechanism is perforation of the pharynx due to barotraumas which encounter in traffic accident. There is a whiplash mechanism of cervical spine along with closure of airways due to an impact of the upper region of the neck against the edge of the steering wheel and forced expiration caused by the concurrent compression of the chest [6]. The frequency of this type of injury decreased after the introduction of seat belts [10]. Another theory supposes that the PP arises due to anterior cervical spurs, posterior edge of the thyroid lamina or hyoid fracture while hyperextended vertebral bodies of the cervical spine [2–4,6]. In the sole reported pediatric case [1], the barotraumas mechanism seems to be less likely. In the setting of hyperextended cervical spine while falling from motorcycle at low speed, actual direct external blunt impact neck on the end of its handlebar compressing the pharynx directly against cervical spine could participate in the injury more likely. Predisposing factor for rupture of the pharynx may also be the absence of longitudinal fibers between mucosal and serous layer of the inferior pharyngeal constrictor muscle of hypopharyngoesophageal transition zone, the region commonly perforated during instrumentation, or more rarely secondary to blast injuries or in the postemetic setting [22]. The transition zone is well known as Killian‘s dehiscence [6,7]. In the present case it seems to be a reasonable assumption to explain the PP just by barotraumas due to suspected blow to the chest as indirectly indicated excoriation on his right hip as well. Due to ligamentous laxity together with a high head to body weight ratio, immaturity of paraspinal muscles and the underdeveloped uncinate processes underlie cervical spine of child to be prone to greater hyperextension [23] which can cause significant strain and tension PE structures over cervical spine ultimately resulting in their thinning and thereby weakening and subsequent forced expiration could easily cause their rupture. In addition, anterior edge of conical pattern of pediatric cervical vertebral bodies may work as stiffer subject and finally may cause PP in extremely hyperextension setting of cervical spine along with current push of pharynx against the cervical spine caused by external blunt impact to the neck region. Clinical course of patients with whiplash-associated disorder may range from no complication to weakness, sensory deficit or absent deep tendon reflexes [24,25] even spinal cord injury without radiographic abnormality (SCIWORA). The age range of children with SCIWORA is 1.5–16 years while much higher incidence is in younger than 9 years [26]. Since our patient was without any neurological deficit, magnetic resonance imaging through the cervical region was not indicated neither initially nor 3 months later. 3.4. Clinical presentation
Fig. 3. A follow-up cervical side X-ray demonstrating regression of the emphysema in the retropharynfgeal region (4th postinjury day).
Symptoms indicating this rare injury include pain in cervical or thoracic region, contusion and excoriation of anterior part of neck or odynophagia and dysphagia as observed in the present case. The patient may experience only sore throat, dyspnea, stridor or hoarseness [1,6,8,14,15,27,28]. Furthermore, expectoration of bloody saliva or feeling of pressure in throat was reported as well
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[1,27]. Palpation can reveal submandibular hematoma, crepitating [8], fluctuation or inflammatory swelling, sometimes laryngeal dislocation [6,14,15]. On the other hand, the clinical course may be asymptomatic in the first 24 [27] to 48 h [7]. Interestingly, isolated PE ruptures were observed in approximately half of all cases with traumatic subcutaneous emphysema [22]. 3.5. Diagnostic method consideration The lateral cervical X-ray demonstrating air strip in retropharyngeal space or subcutaneous emphysema raises the suspicion of PP significantly [6,14,15,20,29] as well as chest X-ray showing mediastinal or pericardial emphysema [14,15] in the presence of the symptoms described above [28]. A few cases documented that air strip in retropharyngeal space along with clinical picture are satisfactory for making of definitive diagnosis and calling for appropriate treatment [7,20,30]. A direct/indirect laryngoscopy and esophagoscopy represent additional diagnostic possibilities. Fiberoptic examination is diagnostic method of choice in order to obtain information about the presence, site, and extent of pharyngeal tear. Rigid endoscopy gives a superior view but requires a general anesthesia and can be impractical in the setting of an immobilized cervical spine [28] as well as in pediatric patients. On the base of our experience we believe that in child with favorable clinical course after BCT indirect laryngoscopy is all that should be done especially when CT investigation was performed initially. Axial CT scan of cervical region may also show pharyngeal emphysema, crack aryepiglotic hematoma or fracture of bone structures or cartilaginous lesions, pathological processes in parapharyngeal and laryngeal space [8,22,27]. Irregular walls of the pharynx on CT scans should elicit a strong suspicion of PP as well [8]. Although these investigations may even fail to detect the rupture of the pharynx wall, especially when the defect is small [27], laryngoscopy may reveal indirect signs such as reduced mobility of voice ligaments and edema or hematoma of pharyngo-laryngeal area [4–6,20,27,30] as observed in the present case. If diagnostic uncertainty persists or clinical deterioration develops throughout the course one should consider an esophagogram using barium paste [27] or gastrografin [31] in the lateral/ supine decubitus position which may increase its sensitivity [27]. Esophagogram may reveal leakage of contrast medium into the retropharyngeal region in the upper cervical region [27]. Sensitivity of this method is 90% for esophageal rupture while for lesions localized in the pharynx it decreases to 60–75%, however its specificity in this location amounts to 100% [12,14,15,32,33]. Overall, there is little consensus about the best choice of diagnostic approach in the literature, therefore a combination of several diagnostic methods (e.g. X-ray, direct/indirect laryngoscopy, flexible fiberoptic nasopharyngolaryngoscopy, flexible bronchoscopy and esophagoscopy, CT investigation and esophagogram) may be necessary in peculiar cases [22,27]. These diagnostic measures are especially indicated in patients revealing clinical deterioration [1] when suspicion of (a) esophagus involvement or (b) larger PP demands surgical intervention. 3.6. Treatment Non-surgical strategy is reserved for cases where the lesion is smaller than 2 cm and only affects the pharynx while the airway is felt to be secure. Broad-spectrum antibiotics are administered with respect to the normal microbial flora of the oropharynx and esophagus (penicillin series) [7,21]. In the case of gram-negative oropharyngeal colonization addition of a gentamicin is desirable [21]. The success of treatment was recorded even with combination of 2nd generation cephalosporin and metronidazole [11]. The
patient is nourished by nasogastric tube and administering liquid supplements per os should begin at 4th–6th posttraumatic day. However, as demonstrated our experience the placement of nasogastric tube can be avoided and a regime ‘‘nil per os’’ should be instituted. In generally, the beginning of oral food intake depends on the type, size, location of the perforation and the overall clinical condition of the patient [7,11,21]. Any conditions which increases airway pressure (coughing, vomiting, or sneezing) should be eliminated [34,35]. Niezgoda et al. [7] reported that four of the five patients treated conservatively recovered and perforation was healed without complications. Prompt surgical strategy should be applied in case the PP exceeds 2 cm or the lesion extends into the esophagus [7]. Niezgoda et al. [7] observed that delay of surgical treatment extending 12 h lead to development of paraesophageal abscess which required surgical management. However, some authors recommend more conservative approach [20,36] as documented in the case of teenager with 5 cm laceration of the posterior pharyngeal wall [1]. Surgical approach is commonly performed along the anterior sternocleidomastoid muscle for optimal exposure of the retropharyngeal space [6] and includes surgical exploration, debridement and suture closure of perforation [37]. A placement of adequate retropharyngoesophageal drain is necessary [7]. If sepsis, pyrexia or local swelling occurs and clinical picture deteriorates during conservative management, it is necessary to search for the possible development of cervical or mediastinal abscess, esophageal fistula, mediastinitis, pyo/pneumothorax, pneumoperitoneum, pneumopericard or pseudoaneurysm of the carotid artery. These complications need to be surgically treated without delay [1,4–7,14,21,28,34,35,38]. Any suspicion of impending respiratory difficulty due to airway trauma with resultant edema should lead to early airway management considering orotracheal intubation or less risky tracheostomy as airway edema and distorted neck anatomy secondary to blunt injury may lead to further airway injury during orotracheal intubation [1,22]. 3.7. Follow-up Follow-up cervical X-ray, esophagogram or CT scan depends on the type, size and location of PP, and on general clinical picture [21,27]. If the child is in good clinical condition, a cervical X-ray performed on the 4th postinjury day is all what should be done, especially when the procedure demonstrated disappearance of the air strip at the prevertebral cervical region or at least its substantial regression. 4. Conclusion Due to the low incidence of the traumatic PP in children, limited available data and the heterogeneous diagnostic and treatment modalities used, it makes difficult to draw any meaningful conclusions relating to diagnosis, treatment and prognosis of these patients. A greater number of patients are essential for selection of optimal therapeutical strategy. On the base of our experience and the reviewed literature, conservative approach seems to be appropriate in the setting of this type of injury in children. Conflict of interest The authors declare that they have no conflict of interest. Funding None.
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