- Email: [email protected]
Postendoscopic intraoral prolapse of a giant esophageal fibrovascular polyp
Formosan Journal of Surgery (2016) 49, 165e168
Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.e-fjs.com
CASE REPORT
Postendoscopic intraoral prolapse of a giant esophageal fibrovascular polyp Ashok Kumar a,*, Anu Behari a, V.K. Kapoor a, Arpit Verma a, Kiran Preet Malhotra b a Department of Surgical Gastroenterology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India b Department of Pathology, Dr. RML Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
Received 11 October 2015; received in revised form 10 January 2016; accepted 9 April 2016 Available online 2 August 2016
KEYWORDS esophagus; fibrovascular polyp; left transcervical approach; nasotracheal intubation
Abstract Fibrovascular polyp of the esophagus is a benign, slow-growing, intraluminal, submucosal tumor. When present in the upper esophagus, it may rarely prolapse through the mouth and may cause asphyxia because of acute airway obstruction. Here, we report a case of a giant fibrovascular esophageal tumor that prolapsed through the mouth during the removal of an upper gastrointestinal endoscope, inducing respiratory distress. Urgent nasotracheal intubation was performed, and the tumor was successfully removed surgically through a left transcervical approach under general anesthesia, thus saving the patient. Copyright ª 2016, Taiwan Surgical Association. Published by Elsevier Taiwan LLC. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/ by-nc-nd/4.0/).
1. Introduction Fibrovascular polyp of the esophagus is a rare, slowgrowing, intraluminal, benign submucosal tumor and represents < 2% of all benign esophageal tumors.1 It usually occurs in middle-aged men and mostly arises from the upper one-third of the esophagus, adjacent to the
Conflicts of interest: All authors declare no conflicts of interest. * Corresponding author. E-mail address: [email protected] (A. Kumar).
cricopharynx. Most patients remain undiagnosed; however, symptomatic patients generally present with dysphagia, especially for solid food. Rarely, these giant polypoidal tumors may prolapse through the mouth and become stuck. The resultant acute airway obstruction may cause lethal asphyxia.2 The elongated sausage-shaped polyp is covered by normal squamous epithelium, because of which these lesions are usually not diagnosed in upper gastrointestinal (GI) endoscopies. In our case, a giant fibrovascular polyp prolapsed through the mouth and stuck during endoscope retrieval, inducing breathing difficulty. Subsequently, the tumor was removed through a left transcervical approach.
http://dx.doi.org/10.1016/j.fjs.2016.04.004 1682-606X/Copyright ª 2016, Taiwan Surgical Association. Published by Elsevier Taiwan LLC. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
166
A. Kumar et al.
Tumor recurrence is rare, and few cases have been reported.1
1.1. Case Report A 37-year-old man without any remarkable medical history presented with complaints of progressive dysphagia, especially for solid food, weight loss, and constant substernal dull aching pain for 7 months. The patient had no history of upper GI bleeding, melena, or any respiratory symptoms. The patient was of average build, was well-nourished, and had stable vital signs. Physical examination was unremarkable. Hematological and biochemical profiles were within normal limits. Upper GI endoscopy revealed a smooth bulge in the proximal and mid esophagus, and the final impression was extra luminal extrinsic compression; the lower esophagus and stomach were normal. Contrast-enhanced computed tomography (CT) revealed a submucosal, nonenhancing, homogenous 15.7 cm 2.7 cm 3.6 cm mass in the esophagus, extending from the thoracic inlet to 4 cm proximal to the gastroesophageal (GE) junction. No significant mediastinal or hilar lymphadenopathy was observed (Figure 1). The patient was referred for a repeat upper GI endoscopy for a second opinion. The repeat upper GI endoscopy revealed a tubular mass originating from the upper esophagus, with intact overlying mucosa, that reached down toward the GE junction, stopping 4 cm above the GE junction. During withdrawal of the endoscope, the polypoidal mass was sucked into the scope and exited through the mouth, and the patient presented to us with a giant polypoidal mass prolapsing through the mouth. The mass could not be pushed back into the esophagus and was causing respiratory difficulty (Figure 2). After successful nasotracheal intubation, a left transcervical neck incision was made to expose the cervical esophagus. The exact origin of the tumor was unclear at that time; slight traction on the tumor through the mouth created a slight inversion of the lateral wall of the dissected cervical esophagus. A 4-cm longitudinal esophagotomy just inferior to the point of inversion was
Figure 2
Giant polyp exiting the mouth.
performed, and the tumor was pulled and delivered through the esophagotomy after dividing the stalk (Figure 3A). The esophagostomy was closed using two layers of interrupted absorbable suture. The patient had an uneventful postoperative course. Oral sips were allowed after a radiological dye study excluded any leak. The patient was discharged on postoperative Day 10 in satisfactory general condition and has remained asymptomatic for 2 years. Gross examination revealed a giant elongated (sausageshaped) 15 cm 4.5 cm 3.5 cm tumor with central umbilication (Figure 3B). Histopathology revealed that the polypoidal mass was covered with normal squamous mucosa and had a microscopic composition of lymphocytes and plasma cells interspersed with fibroblasts and blood vessels (Figure 4).
1.2. Consent Written informed consent was obtained from the patient for publication of this case report and all accompanying images.
2. Discussion
Figure 1 Computed tomography of the thorax showing the nonenhancing, homogenous nature of the polyp.
Fibrovascular polyps are very rare intraluminal benign tumors of the esophagus. To date, only w100 cases have been reported, with the largest single series reporting 16 cases.3 Most patients were men in their 60s and 70s, but a few cases have been reported in infants as well.4 The male-to-female ratio for fibrovascular polyp incidence is approximately 3:1.5 The most common occurrence site is the upper one-third of the esophagus (the inferior part of cricopharynx and rarely from the hypopharynx), with tumor size ranging from a few
Prolapse of an esophageal fibrovascular polyp
Figure 3
167
(A) Polyp delivered though left lateral esophagostomy. (B) Sausage-shaped polypoidal mass after surgery.
Figure 4 Stratified squamous epithelium with underlying scattered thin-walled vessels and lymphoid aggregates (hematoxylin and eosin 50).
centimeters to large tumors that reach up to the stomach. These tumors are usually pedunculated because of a combination of the propulsive act of swallowing and propulsive peristalsis of the esophagus toward the caudal end. Clinically, patients present with nonspecific symptoms, and these tumors mostly remain undiagnosed until they become huge. The most common symptoms are dysphagia (87%), respiratory difficulty (25%), and regurgitation through the mouth (10%).6 Rarely, they cause asphyxia because of obstruction of the larynx and can thus be lethal. Tumors usually show a uniform, fibromyxoid appearance on the cut surface. Histological examinations typically show
benign squamous epithelium on the underlying lamina propria, which is expanded because of a mixture of highly vascularized loose and dense fibrous tissue with intervening lobules of adipose tissue and small clusters of mature adipocytes. Focal areas of spindle cell differentiation may also be present.2 Initial diagnosis is usually made after barium studies and a review of patient history; the most common finding is a smooth intraluminal sausage-shaped mass with a bulbous tip with varying degrees of lobulation. During an upper GI endoscopy, the esophageal lumen and the surface of the polyp can resemble normal esophageal mucosa; therefore, it is frequently missed during endoscopies, as in our case.7 Endoscopic ultrasound is useful in revealing the site of origin of the stalk and the size and vascularity of the polyp. It also enables guided fine-needle aspiration for cytology.8 CT and magnetic resonance scans are valuable tools for determining the appropriate surgical treatment. Multiplanar CT scanning provides information on the location, size, and pedicle attachment of the tumor, which is critical for deciding the surgical treatment. Magnetic resonance scans with axial, coronal, and sagittal views can help in precise identification of the stalk. Because of the risk of a lethal complication (e.g., asphyxia), removal of these polyps is strongly recommended. These tumors may be removed through transoral, transcervical, transthoracic, and endoscopic approaches, depending on the location and size of the polyp. Polyps smaller than 2 cm can be removed endoscopically and those larger than 8 cm are usually treated through a left cervical approach. In very large polyps, the stalk may be divided through cervical vertical esophagostomy, following which the head of the polyp can be removed through gastrostomy. Successful endoscopic removal of a giant fibrovascular polyp in a 63-year-old man with dwarfism and obstructive sleep apnea has been reported. A 10-cm giant fibrovascular polyp arising just below the cricoid cartilage was removed endoscopically. The base of the lesion was exposed using a
168 Weerda bivalved laryngoscope, and a bipolar cautery combined with a snare was used to transect the base with excellent hemostasis.9 Polyp removal is curative, and recurrence and malignant transformation are rare. In conclusion, giant fibrovascular polyps may rarely prolapse through the mouth after endoscopy and may lead to fatal asphyxia. Immediate surgical removal through an appropriate surgical approach can save the patient.
Acknowledgments We thank Dr Prashant Verma, Gastroenterologist, Department of Gastroenterology, RML Institute of Medical Sciences, Lucknow, India, for the endoscopic evaluation of the patient. We thank Dr Rajnikant Yadav, Radiologist, Department of Radiology, RML Institute of Medical Sciences, for interpretation of the radiological studies.
References 1. Drenth J, Wobbes T, Bonenkamp JJ, Nagengast FM. Recurrent esophageal fibrovascular polyps: case history and review of the literature. Dig Dis Sci. 2002;47:2598e2604.
A. Kumar et al. 2. Sargent RL, Hood IC. Asphyxiation caused by giant fibrovascular polyp of the esophagus. Arch Pathol Lab Med. 2006;130: 725e727. 3. Levine MS, Buck JL, Pantongrag-Brown L, Buetow PC, Hallman JR, Sobin LH. Fibrovascular polyps of the esophagus: clinical, radiographic, and pathologic findings in 16 patients. AJR Am J Roentgenol. 1996;166:781e787. 4. Paik HC, Han JW, Jung EK, Bae KM, Lee YH. Fibrovascular polyp of the esophagus in infant. Yonsei Med J. 2001;42: 264e266. 5. Avezzano EA, Fleischer DE, Merida MA, Anderson DL. Giant fibrovascular polyps of the esophagus. Am J Gastroenterol. 1990;85:299e302. 6. Lee SY, Chan WH, Sivanandan R, Lim DT, Wong WK. Recurrent giant fibrovascular polyp of the esophagus. World J Gastroentrol. 2009;15:3697e3700. 7. Hoseok I, Kim JS, Shim YM. Giant fibrovascular polyp of the hypopharynx: surgical treatment with the biappoach. J Korean Med Sci. 2006;21:749e751. 8. Solerio D, Gasparri G, Ruffini E, et al. Giant fibrovascular polyp of the esophagus. Dis Esophagus. 2005;18:410e412. 9. Pham AM, Rees CJ, Belafsky PC. Endoscopic removal of a giant fibrovascular polyp of the esophagus. Ann Otol Rhinol Laryngol. 2008;117:587e590.
Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.e-fjs.com
CASE REPORT
Postendoscopic intraoral prolapse of a giant esophageal fibrovascular polyp Ashok Kumar a,*, Anu Behari a, V.K. Kapoor a, Arpit Verma a, Kiran Preet Malhotra b a Department of Surgical Gastroenterology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India b Department of Pathology, Dr. RML Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
Received 11 October 2015; received in revised form 10 January 2016; accepted 9 April 2016 Available online 2 August 2016
KEYWORDS esophagus; fibrovascular polyp; left transcervical approach; nasotracheal intubation
Abstract Fibrovascular polyp of the esophagus is a benign, slow-growing, intraluminal, submucosal tumor. When present in the upper esophagus, it may rarely prolapse through the mouth and may cause asphyxia because of acute airway obstruction. Here, we report a case of a giant fibrovascular esophageal tumor that prolapsed through the mouth during the removal of an upper gastrointestinal endoscope, inducing respiratory distress. Urgent nasotracheal intubation was performed, and the tumor was successfully removed surgically through a left transcervical approach under general anesthesia, thus saving the patient. Copyright ª 2016, Taiwan Surgical Association. Published by Elsevier Taiwan LLC. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/ by-nc-nd/4.0/).
1. Introduction Fibrovascular polyp of the esophagus is a rare, slowgrowing, intraluminal, benign submucosal tumor and represents < 2% of all benign esophageal tumors.1 It usually occurs in middle-aged men and mostly arises from the upper one-third of the esophagus, adjacent to the
Conflicts of interest: All authors declare no conflicts of interest. * Corresponding author. E-mail address: [email protected] (A. Kumar).
cricopharynx. Most patients remain undiagnosed; however, symptomatic patients generally present with dysphagia, especially for solid food. Rarely, these giant polypoidal tumors may prolapse through the mouth and become stuck. The resultant acute airway obstruction may cause lethal asphyxia.2 The elongated sausage-shaped polyp is covered by normal squamous epithelium, because of which these lesions are usually not diagnosed in upper gastrointestinal (GI) endoscopies. In our case, a giant fibrovascular polyp prolapsed through the mouth and stuck during endoscope retrieval, inducing breathing difficulty. Subsequently, the tumor was removed through a left transcervical approach.
http://dx.doi.org/10.1016/j.fjs.2016.04.004 1682-606X/Copyright ª 2016, Taiwan Surgical Association. Published by Elsevier Taiwan LLC. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
166
A. Kumar et al.
Tumor recurrence is rare, and few cases have been reported.1
1.1. Case Report A 37-year-old man without any remarkable medical history presented with complaints of progressive dysphagia, especially for solid food, weight loss, and constant substernal dull aching pain for 7 months. The patient had no history of upper GI bleeding, melena, or any respiratory symptoms. The patient was of average build, was well-nourished, and had stable vital signs. Physical examination was unremarkable. Hematological and biochemical profiles were within normal limits. Upper GI endoscopy revealed a smooth bulge in the proximal and mid esophagus, and the final impression was extra luminal extrinsic compression; the lower esophagus and stomach were normal. Contrast-enhanced computed tomography (CT) revealed a submucosal, nonenhancing, homogenous 15.7 cm 2.7 cm 3.6 cm mass in the esophagus, extending from the thoracic inlet to 4 cm proximal to the gastroesophageal (GE) junction. No significant mediastinal or hilar lymphadenopathy was observed (Figure 1). The patient was referred for a repeat upper GI endoscopy for a second opinion. The repeat upper GI endoscopy revealed a tubular mass originating from the upper esophagus, with intact overlying mucosa, that reached down toward the GE junction, stopping 4 cm above the GE junction. During withdrawal of the endoscope, the polypoidal mass was sucked into the scope and exited through the mouth, and the patient presented to us with a giant polypoidal mass prolapsing through the mouth. The mass could not be pushed back into the esophagus and was causing respiratory difficulty (Figure 2). After successful nasotracheal intubation, a left transcervical neck incision was made to expose the cervical esophagus. The exact origin of the tumor was unclear at that time; slight traction on the tumor through the mouth created a slight inversion of the lateral wall of the dissected cervical esophagus. A 4-cm longitudinal esophagotomy just inferior to the point of inversion was
Figure 2
Giant polyp exiting the mouth.
performed, and the tumor was pulled and delivered through the esophagotomy after dividing the stalk (Figure 3A). The esophagostomy was closed using two layers of interrupted absorbable suture. The patient had an uneventful postoperative course. Oral sips were allowed after a radiological dye study excluded any leak. The patient was discharged on postoperative Day 10 in satisfactory general condition and has remained asymptomatic for 2 years. Gross examination revealed a giant elongated (sausageshaped) 15 cm 4.5 cm 3.5 cm tumor with central umbilication (Figure 3B). Histopathology revealed that the polypoidal mass was covered with normal squamous mucosa and had a microscopic composition of lymphocytes and plasma cells interspersed with fibroblasts and blood vessels (Figure 4).
1.2. Consent Written informed consent was obtained from the patient for publication of this case report and all accompanying images.
2. Discussion
Figure 1 Computed tomography of the thorax showing the nonenhancing, homogenous nature of the polyp.
Fibrovascular polyps are very rare intraluminal benign tumors of the esophagus. To date, only w100 cases have been reported, with the largest single series reporting 16 cases.3 Most patients were men in their 60s and 70s, but a few cases have been reported in infants as well.4 The male-to-female ratio for fibrovascular polyp incidence is approximately 3:1.5 The most common occurrence site is the upper one-third of the esophagus (the inferior part of cricopharynx and rarely from the hypopharynx), with tumor size ranging from a few
Prolapse of an esophageal fibrovascular polyp
Figure 3
167
(A) Polyp delivered though left lateral esophagostomy. (B) Sausage-shaped polypoidal mass after surgery.
Figure 4 Stratified squamous epithelium with underlying scattered thin-walled vessels and lymphoid aggregates (hematoxylin and eosin 50).
centimeters to large tumors that reach up to the stomach. These tumors are usually pedunculated because of a combination of the propulsive act of swallowing and propulsive peristalsis of the esophagus toward the caudal end. Clinically, patients present with nonspecific symptoms, and these tumors mostly remain undiagnosed until they become huge. The most common symptoms are dysphagia (87%), respiratory difficulty (25%), and regurgitation through the mouth (10%).6 Rarely, they cause asphyxia because of obstruction of the larynx and can thus be lethal. Tumors usually show a uniform, fibromyxoid appearance on the cut surface. Histological examinations typically show
benign squamous epithelium on the underlying lamina propria, which is expanded because of a mixture of highly vascularized loose and dense fibrous tissue with intervening lobules of adipose tissue and small clusters of mature adipocytes. Focal areas of spindle cell differentiation may also be present.2 Initial diagnosis is usually made after barium studies and a review of patient history; the most common finding is a smooth intraluminal sausage-shaped mass with a bulbous tip with varying degrees of lobulation. During an upper GI endoscopy, the esophageal lumen and the surface of the polyp can resemble normal esophageal mucosa; therefore, it is frequently missed during endoscopies, as in our case.7 Endoscopic ultrasound is useful in revealing the site of origin of the stalk and the size and vascularity of the polyp. It also enables guided fine-needle aspiration for cytology.8 CT and magnetic resonance scans are valuable tools for determining the appropriate surgical treatment. Multiplanar CT scanning provides information on the location, size, and pedicle attachment of the tumor, which is critical for deciding the surgical treatment. Magnetic resonance scans with axial, coronal, and sagittal views can help in precise identification of the stalk. Because of the risk of a lethal complication (e.g., asphyxia), removal of these polyps is strongly recommended. These tumors may be removed through transoral, transcervical, transthoracic, and endoscopic approaches, depending on the location and size of the polyp. Polyps smaller than 2 cm can be removed endoscopically and those larger than 8 cm are usually treated through a left cervical approach. In very large polyps, the stalk may be divided through cervical vertical esophagostomy, following which the head of the polyp can be removed through gastrostomy. Successful endoscopic removal of a giant fibrovascular polyp in a 63-year-old man with dwarfism and obstructive sleep apnea has been reported. A 10-cm giant fibrovascular polyp arising just below the cricoid cartilage was removed endoscopically. The base of the lesion was exposed using a
168 Weerda bivalved laryngoscope, and a bipolar cautery combined with a snare was used to transect the base with excellent hemostasis.9 Polyp removal is curative, and recurrence and malignant transformation are rare. In conclusion, giant fibrovascular polyps may rarely prolapse through the mouth after endoscopy and may lead to fatal asphyxia. Immediate surgical removal through an appropriate surgical approach can save the patient.
Acknowledgments We thank Dr Prashant Verma, Gastroenterologist, Department of Gastroenterology, RML Institute of Medical Sciences, Lucknow, India, for the endoscopic evaluation of the patient. We thank Dr Rajnikant Yadav, Radiologist, Department of Radiology, RML Institute of Medical Sciences, for interpretation of the radiological studies.
References 1. Drenth J, Wobbes T, Bonenkamp JJ, Nagengast FM. Recurrent esophageal fibrovascular polyps: case history and review of the literature. Dig Dis Sci. 2002;47:2598e2604.
A. Kumar et al. 2. Sargent RL, Hood IC. Asphyxiation caused by giant fibrovascular polyp of the esophagus. Arch Pathol Lab Med. 2006;130: 725e727. 3. Levine MS, Buck JL, Pantongrag-Brown L, Buetow PC, Hallman JR, Sobin LH. Fibrovascular polyps of the esophagus: clinical, radiographic, and pathologic findings in 16 patients. AJR Am J Roentgenol. 1996;166:781e787. 4. Paik HC, Han JW, Jung EK, Bae KM, Lee YH. Fibrovascular polyp of the esophagus in infant. Yonsei Med J. 2001;42: 264e266. 5. Avezzano EA, Fleischer DE, Merida MA, Anderson DL. Giant fibrovascular polyps of the esophagus. Am J Gastroenterol. 1990;85:299e302. 6. Lee SY, Chan WH, Sivanandan R, Lim DT, Wong WK. Recurrent giant fibrovascular polyp of the esophagus. World J Gastroentrol. 2009;15:3697e3700. 7. Hoseok I, Kim JS, Shim YM. Giant fibrovascular polyp of the hypopharynx: surgical treatment with the biappoach. J Korean Med Sci. 2006;21:749e751. 8. Solerio D, Gasparri G, Ruffini E, et al. Giant fibrovascular polyp of the esophagus. Dis Esophagus. 2005;18:410e412. 9. Pham AM, Rees CJ, Belafsky PC. Endoscopic removal of a giant fibrovascular polyp of the esophagus. Ann Otol Rhinol Laryngol. 2008;117:587e590.