An Unusual Cause of Airway Obstruction in a Patient in the Endoscopy Suite Taizoon Q. Dhoon, MD,* Jennifer L. Gerber, MD,* Thuy B. Tran, MD,† Ali Mahtabifard, MD,‡ and Karen S. Sibert, MD* A 74-year-old man presented for outpatient endoscopy because of dysphagia and the sensation of a mass in the back of his throat. Esophagogastroduodenoscopy demonstrated a soft tissue mass in the proximal esophagus that prolapsed into the hypopharynx on withdrawal of the endoscope. Complete airway obstruction ensued, requiring emergent tracheotomy. The patient was transferred to the hospital for further treatment. Surgical resection revealed a rare giant fibrovascular polyp, which may be associated with asphyxiation and sudden death. (A&A Case Reports. 2014;3:15–9.)
F
ibrovascular polyps (FVPs) are rare benign tumors of the esophagus. Due to an indolent growth pattern, FVPs may come to medical attention only when their size progresses to the point that patients become symptomatic and incur risk for progressive dysphagia, intermittent bleeding, asphyxiation, and death. We present the case of a patient who required emergent tracheotomy due to sudden airway obstruction by a giant FVP at the conclusion of esophagogastroduodenoscopy (EGD). The patient reviewed the case report and provided verbal and written consent for publication.
Case Description
A 74-year-old man was referred by his primary physician for EGD because of progressive dysphagia when attempting to swallow solid foods, 1 episode of hemoptysis, and the sensation of a foreign body in his throat. Once, when lying in the prone position, he experienced the sensation of a mass coming up into the back of his throat, which resolved when he swallowed. On another occasion, he felt a similar sensation of a mass coming up into his throat during an episode of coughing; this was associated with a small amount of bleeding. A barium swallow with video fluoroscopy 2 years earlier had been read as normal except for postnasal drip and a 5-mm Zenker’s diverticulum. The patient was referred to an otolaryngologist for further evaluation 1 month before admission and underwent direct laryngoscopy, which was reported as normal. Repeat video swallow 4 days before admission was interpreted as normal except for diminished motility in the lower esophagus; the radiologist recommended further evaluation via esophagram. The patient’s medical history was significant for human immunodeficiency virus with a stable CD4 count higher than 500 cells/mm3, asthma, and gastroesophageal reflux disease. The patient denied recent From the Departments of *Anesthesiology, †Surgery, and ‡Cardiothoracic Surgery, Cedars-Sinai Medical Center, Los Angeles, California. Accepted for publication March 18, 2014. Funding: None. The authors declare no conflicts of interest. Address correspondence to Karen S. Sibert, MD, Department of Anesthesiology, Cedars-Sinai Medical Center, 8700 Beverly Blvd., Room 8211, Los Angeles, CA 90048. Address e-mail to [email protected]. Copyright © 2014 International Anesthesia Research Society DOI: 10.1213/XAA.0000000000000040
July 15, 2014 • Volume 3 • Number 2
weight loss; his body mass index was normal at 23 kg/m2. Physical examination, including the external appearance of the patient’s head and neck, vital signs, and laboratory values, were within normal limits. EGD was scheduled at an ambulatory surgery center. An anesthesiologist administered sedation with IV propofol, fentanyl, and midazolam. With the patient breathing spontaneously in the left lateral decubitus position, endoscopy demonstrated a smooth, elongated polypoid mass, several centimeters in length, oriented longitudinally in the cervical esophagus. No photographs were taken. The gastroenterologist opted not to excise or biopsy the lesion and began to withdraw the endoscope, whereupon the mass prolapsed into the hypopharynx. An otolaryngologist was called into the procedure room to evaluate the mass and described it in his report as “somewhat bulging and pedunculated.” Neither the gastroenterologist nor the otolaryngologist had encountered a similar lesion before, and no diagnosis was made. No Zenker diverticulum was appreciated. While the otolaryngologist was examining the mass, the patient took a deep breath. The free distal portion of the mass, though its base was still attached in the proximal esophagus, was forcefully aspirated into the laryngeal opening, obstructing respiration. The otolaryngologist attempted to grasp the mass and dislodge it from the airway, without success. Sedation was discontinued and the patient was returned to the supine position. The patient’s airway continued to be completely obstructed despite his vigorous attempts to breathe and cough. The oxyhemoglobin saturation began to decrease and the anesthesiologist was unable to ventilate the lungs effectively via mask. Succinylcholine 100 mg was administered. On direct laryngoscopy, the vocal cords could not be seen; only soft tissue was visible in the pharynx. Tracheal intubation could not be accomplished, and the tissue in the posterior pharynx began to bleed. Emergent tracheotomy was successfully performed by the otolaryngologist and an endotracheal tube was inserted. The patient was transferred to a nearby hospital and admitted to the intensive care unit for further evaluation and treatment. Consultant physicians from the pulmonary, gastroenterology, and otolaryngology services concurred that the esophageal mass had prolapsed into the patient’s posterior pharynx, producing complete airway obstruction. On the first day after hospital admission, the patient was taken to surgery for revision of the tracheotomy and insertion of a cases-anesthesia-analgesia.org
15
tracheostomy tube. Direct laryngoscopy and esophagoscopy revealed polypoid tissue in the posterior hypopharynx, with attachment in the cervical esophagus at the level of the cricopharyngeus muscle. Due to extensive swelling and edema, no resection or manipulation of the mass was attempted. Subsequent computed tomography of the neck identified a 9-cm polypoid-like lesion originating in the cervical esophagus at the level of C4 and extending distally through T2, suggestive of a giant FVP (Figs. 1 and 2). A thoracic surgeon performed repeat EGD on hospital day 4 under general anesthesia. A large, mobile polyp with a bifid tip was observed in the proximal esophagus with its stalk originating at the level of the cricopharyngeus muscle and upper esophageal sphincter. The tip of the mass was grasped and the mass was pulled cephalad into the hypopharynx for inspection. At the end of the procedure, the mass was pushed back into the esophagus under direct vision with laryngoscopy. On hospital day 8, the thoracic surgeon performed left lateral transcervical esophageal exploration and complete excision of the polyp, which was a bifid mass originating from a single stalk (Figs. 3 and 4). The pathologic diagnosis was esophageal FVP with overlying ulceration and reactive changes, and no histologic evidence of malignancy. The patient’s hospital course was complicated by aspiration pneumonia and neck-wound infection requiring surgical debridement. A gastrostomy tube was inserted for enteral feeding. The tracheostomy was removed on hospital day 21. The patient’s recovery was uneventful, and there were no apparent neurologic sequelae. He was discharged to a rehabilitation facility on hospital day 25. Two months after discharge, the patient required esophageal dilation due to anastamotic stenosis. His oral intake improved, allowing removal of the gastrostomy tube. He has returned to full-time employment.
DISCUSSION
FVPs are rare, benign, pedunculated, submucosal tumors that project into the lumen of the esophagus; they represent
Figure 1. Sagittal computed tomography scan shows a pedunculated soft tissue mass arising at the origin of the cricopharyngeus muscle at the level of C4 (arrow). The mass extends to the level of the clavicle at T2 (arrow).
16 cases-anesthesia-analgesia.org
1% to 2% of esophageal tumors. FVPs as long as 26cm in length have been reported.1,2 Giant FVPs are defined as polyps larger than 5 cm in diameter. The overall incidence of FVP is unknown because there are slightly >120 reported cases in the literature.1,3–6 Two studies indicate that the occurrence rate is approximately equal in men and women; two-thirds of patients are older than 50 years.1,7 FVPs are composed of a mixture of fibrous tissue, adipose tissue, and vascular components1,3,4,6,8 Rarely, FVPs may undergo malignant transformation.2,6,8 They are covered by normal squamous epithelium. As in our case, the normal appearance of the FVP mucosa may render it difficult to differentiate the mass from the mucosa of the pharynx and esophagus. This epithelium has the potential to ulcerate and bleed, especially in larger tumors.4,9 The risk of bleeding may increase with distal esophageal extension if the distal tip of the polyp is exposed to gastric acid. As a rule, FVPs originate within the proximal one-third of the esophagus. The most common presentation is progressive dysphagia, cited in 50% to 62% of cases.10 Other manifestations include respiratory symptoms (25%), sore throat (5%), odynophagia (7%), nonexertional substernal chest pain (8%), persistent cough (8%), foreign body sensation (25%), and weight loss (13%–19%). Hematemesis, epigastric pain, and emesis have been described, although the incidence is variable.1,2,5,8,11,12 Regurgitation of the mass has been described in 12% to 38% of cases as a result of loose, redundant tissue. This tissue stretches distally with swallowing and leads to elongation of the mass. A large FVP has the potential to prolapse into the oropharynx, causing a “regurgitation of a fleshy mass” sensation, as reported by our patient.6 Symptomatic patients report a mass protruding into the mouth, requiring digital maneuvers to move the mass downward out of the airway. Without a history of apparent regurgitation of the polyp, the initial diagnosis of a FVP can be difficult.1 Since the polyp’s normal squamous epithelium has the appearance of normal mucosa, up to 25% of cases are missed on endoscopy.2,13 Asphyxiation and sudden death can occur if a polyp regurgitates and occludes the larynx.2,5,8,11,12,14–16 Complete excision of FVPs is recommended due to the risk of progressively debilitating symptoms, malignant transformation, airway obstruction, and sudden death. Endoscopic resection is recommended for small polyps
F
ibrovascular polyps (FVPs) are rare benign tumors of the esophagus. Due to an indolent growth pattern, FVPs may come to medical attention only when their size progresses to the point that patients become symptomatic and incur risk for progressive dysphagia, intermittent bleeding, asphyxiation, and death. We present the case of a patient who required emergent tracheotomy due to sudden airway obstruction by a giant FVP at the conclusion of esophagogastroduodenoscopy (EGD). The patient reviewed the case report and provided verbal and written consent for publication.
Case Description
A 74-year-old man was referred by his primary physician for EGD because of progressive dysphagia when attempting to swallow solid foods, 1 episode of hemoptysis, and the sensation of a foreign body in his throat. Once, when lying in the prone position, he experienced the sensation of a mass coming up into the back of his throat, which resolved when he swallowed. On another occasion, he felt a similar sensation of a mass coming up into his throat during an episode of coughing; this was associated with a small amount of bleeding. A barium swallow with video fluoroscopy 2 years earlier had been read as normal except for postnasal drip and a 5-mm Zenker’s diverticulum. The patient was referred to an otolaryngologist for further evaluation 1 month before admission and underwent direct laryngoscopy, which was reported as normal. Repeat video swallow 4 days before admission was interpreted as normal except for diminished motility in the lower esophagus; the radiologist recommended further evaluation via esophagram. The patient’s medical history was significant for human immunodeficiency virus with a stable CD4 count higher than 500 cells/mm3, asthma, and gastroesophageal reflux disease. The patient denied recent From the Departments of *Anesthesiology, †Surgery, and ‡Cardiothoracic Surgery, Cedars-Sinai Medical Center, Los Angeles, California. Accepted for publication March 18, 2014. Funding: None. The authors declare no conflicts of interest. Address correspondence to Karen S. Sibert, MD, Department of Anesthesiology, Cedars-Sinai Medical Center, 8700 Beverly Blvd., Room 8211, Los Angeles, CA 90048. Address e-mail to [email protected]. Copyright © 2014 International Anesthesia Research Society DOI: 10.1213/XAA.0000000000000040
July 15, 2014 • Volume 3 • Number 2
weight loss; his body mass index was normal at 23 kg/m2. Physical examination, including the external appearance of the patient’s head and neck, vital signs, and laboratory values, were within normal limits. EGD was scheduled at an ambulatory surgery center. An anesthesiologist administered sedation with IV propofol, fentanyl, and midazolam. With the patient breathing spontaneously in the left lateral decubitus position, endoscopy demonstrated a smooth, elongated polypoid mass, several centimeters in length, oriented longitudinally in the cervical esophagus. No photographs were taken. The gastroenterologist opted not to excise or biopsy the lesion and began to withdraw the endoscope, whereupon the mass prolapsed into the hypopharynx. An otolaryngologist was called into the procedure room to evaluate the mass and described it in his report as “somewhat bulging and pedunculated.” Neither the gastroenterologist nor the otolaryngologist had encountered a similar lesion before, and no diagnosis was made. No Zenker diverticulum was appreciated. While the otolaryngologist was examining the mass, the patient took a deep breath. The free distal portion of the mass, though its base was still attached in the proximal esophagus, was forcefully aspirated into the laryngeal opening, obstructing respiration. The otolaryngologist attempted to grasp the mass and dislodge it from the airway, without success. Sedation was discontinued and the patient was returned to the supine position. The patient’s airway continued to be completely obstructed despite his vigorous attempts to breathe and cough. The oxyhemoglobin saturation began to decrease and the anesthesiologist was unable to ventilate the lungs effectively via mask. Succinylcholine 100 mg was administered. On direct laryngoscopy, the vocal cords could not be seen; only soft tissue was visible in the pharynx. Tracheal intubation could not be accomplished, and the tissue in the posterior pharynx began to bleed. Emergent tracheotomy was successfully performed by the otolaryngologist and an endotracheal tube was inserted. The patient was transferred to a nearby hospital and admitted to the intensive care unit for further evaluation and treatment. Consultant physicians from the pulmonary, gastroenterology, and otolaryngology services concurred that the esophageal mass had prolapsed into the patient’s posterior pharynx, producing complete airway obstruction. On the first day after hospital admission, the patient was taken to surgery for revision of the tracheotomy and insertion of a cases-anesthesia-analgesia.org
15
tracheostomy tube. Direct laryngoscopy and esophagoscopy revealed polypoid tissue in the posterior hypopharynx, with attachment in the cervical esophagus at the level of the cricopharyngeus muscle. Due to extensive swelling and edema, no resection or manipulation of the mass was attempted. Subsequent computed tomography of the neck identified a 9-cm polypoid-like lesion originating in the cervical esophagus at the level of C4 and extending distally through T2, suggestive of a giant FVP (Figs. 1 and 2). A thoracic surgeon performed repeat EGD on hospital day 4 under general anesthesia. A large, mobile polyp with a bifid tip was observed in the proximal esophagus with its stalk originating at the level of the cricopharyngeus muscle and upper esophageal sphincter. The tip of the mass was grasped and the mass was pulled cephalad into the hypopharynx for inspection. At the end of the procedure, the mass was pushed back into the esophagus under direct vision with laryngoscopy. On hospital day 8, the thoracic surgeon performed left lateral transcervical esophageal exploration and complete excision of the polyp, which was a bifid mass originating from a single stalk (Figs. 3 and 4). The pathologic diagnosis was esophageal FVP with overlying ulceration and reactive changes, and no histologic evidence of malignancy. The patient’s hospital course was complicated by aspiration pneumonia and neck-wound infection requiring surgical debridement. A gastrostomy tube was inserted for enteral feeding. The tracheostomy was removed on hospital day 21. The patient’s recovery was uneventful, and there were no apparent neurologic sequelae. He was discharged to a rehabilitation facility on hospital day 25. Two months after discharge, the patient required esophageal dilation due to anastamotic stenosis. His oral intake improved, allowing removal of the gastrostomy tube. He has returned to full-time employment.
DISCUSSION
FVPs are rare, benign, pedunculated, submucosal tumors that project into the lumen of the esophagus; they represent
Figure 1. Sagittal computed tomography scan shows a pedunculated soft tissue mass arising at the origin of the cricopharyngeus muscle at the level of C4 (arrow). The mass extends to the level of the clavicle at T2 (arrow).
16 cases-anesthesia-analgesia.org
1% to 2% of esophageal tumors. FVPs as long as 26cm in length have been reported.1,2 Giant FVPs are defined as polyps larger than 5 cm in diameter. The overall incidence of FVP is unknown because there are slightly >120 reported cases in the literature.1,3–6 Two studies indicate that the occurrence rate is approximately equal in men and women; two-thirds of patients are older than 50 years.1,7 FVPs are composed of a mixture of fibrous tissue, adipose tissue, and vascular components1,3,4,6,8 Rarely, FVPs may undergo malignant transformation.2,6,8 They are covered by normal squamous epithelium. As in our case, the normal appearance of the FVP mucosa may render it difficult to differentiate the mass from the mucosa of the pharynx and esophagus. This epithelium has the potential to ulcerate and bleed, especially in larger tumors.4,9 The risk of bleeding may increase with distal esophageal extension if the distal tip of the polyp is exposed to gastric acid. As a rule, FVPs originate within the proximal one-third of the esophagus. The most common presentation is progressive dysphagia, cited in 50% to 62% of cases.10 Other manifestations include respiratory symptoms (25%), sore throat (5%), odynophagia (7%), nonexertional substernal chest pain (8%), persistent cough (8%), foreign body sensation (25%), and weight loss (13%–19%). Hematemesis, epigastric pain, and emesis have been described, although the incidence is variable.1,2,5,8,11,12 Regurgitation of the mass has been described in 12% to 38% of cases as a result of loose, redundant tissue. This tissue stretches distally with swallowing and leads to elongation of the mass. A large FVP has the potential to prolapse into the oropharynx, causing a “regurgitation of a fleshy mass” sensation, as reported by our patient.6 Symptomatic patients report a mass protruding into the mouth, requiring digital maneuvers to move the mass downward out of the airway. Without a history of apparent regurgitation of the polyp, the initial diagnosis of a FVP can be difficult.1 Since the polyp’s normal squamous epithelium has the appearance of normal mucosa, up to 25% of cases are missed on endoscopy.2,13 Asphyxiation and sudden death can occur if a polyp regurgitates and occludes the larynx.2,5,8,11,12,14–16 Complete excision of FVPs is recommended due to the risk of progressively debilitating symptoms, malignant transformation, airway obstruction, and sudden death. Endoscopic resection is recommended for small polyps
