- Email: [email protected]
Esophageal Intramural Diverticula: Multiple and Single
Ann Thorac Surg 2016;101:769–71
CASE REPORT HUBBARD AND DEMOS ESOPHAGEAL INTRAMURAL DIVERTICULA
769
4. Richardson JD. Outcome of tracheobronchial injuries: a longterm perspective. J Trauma 2004;56:30–6. 5. Huh j, Milliken JC, Chen JC. . Management of tracheobronchial injuries following blunt and penetrating trauma. Am Surg 1997;63:896–9. 6. Hood MR. Injury to the trachea and major bronchi. In: Hood MR, Arthur BD, Culliford AT (eds). Thoracic trauma. Philadelphia: WB Saunders; 1989. 7. Chu CP, Chen PP. Tracheobronchial injury secondary to blunt chest trauma: diagnosis and management. Anaesth Intensive Care 2002;30:145–52. 8. Kara H, Bayir A, Ak A, Tufekci N, Degirmenci S, Akinci M. Tracheal rupture developing after blunt thoracic trauma. Case Rep Clin Med 2013;2:502–4.
Esophageal Intramural Diverticula: Multiple and Single Jennifer Hubbard, BS, and Nicholas J. Demos, MD Ross University School of Medicine, Miramar, Florida, and Hoboken University Medical Center, Hoboken, New Jersey
D
iverticula in the human body usually produce symptoms caused by pressure on the surrounding organs or by the deterioration of their mucosal lining. However, intramural esophageal diverticula are associated with symptoms of gastroesophageal reflux or related symptoms and signs not quite related to their pathogenesis or pathophysiology. We review the cases of three patients, which may somewhat illuminate the nature of these unusual and rare diverticula.
Fig 1. Esophagram of patient 1 showing multiple intramural diverticula, a moderate lower esophageal stricture, and a hiatal hernia.
the wall, and areas of Barrett’s esophagus (Fig 3). Motility and scan studies showed esophagogastroparesis. Through a transabdominal approach, a partial fundoplication, a loop gastroenterostomy and, dilation of the esophageal stricture were performed. At the sixthmonth follow-up visit, his strength and nutritional status were restored. At the 15-year follow-up visit, the man described no symptoms.
Patient 2 A 49-year-old woman was admitted with massive bloody vomiting. Her hemoglobin level was restored with two
Case Reports Patient 1 A 57-year-old man with a history of gastroesophageal reflux disease and diabetes mellitus had stopped eating. He was malnourished, with electrolyte imbalance. An esophagram revealed multiple intramural pseudodiverticula, a moderate lower esophageal stricture, and a hiatal hernia (Fig 1). Endoscopy showed food impaction, a mild stricture, and a hiatal hernia (Fig 2). Biopsy revealed pseudodiverticula of the esophagus, hypertrophy of Accepted for publication March 10, 2015. Address correspondence to Dr Demos, 4 Cambridge Dr, Short Hills, NJ 07078; email: [email protected].
Ó 2016 by The Society of Thoracic Surgeons Published by Elsevier
Fig 2. Endoscopic view of patient 1 showing multiple small pits throughout the esophagus were seen. In addition, severe lower esophagitis, a mild stricture, and sliding hiatal hernia were identified. The arrow points to the opening of the intramural diverticulum. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2015.03.045
FEATURE ARTICLES
More than 50 years ago, two investigators described a rare case of multiple esophageal diverticula, which they named esophageal intramural pseudodiverticulosis (EIPD). To this day, the pathogenesis of this condition remains obscure. Several coexisting conditions, such as strictures, esophageal dysmotility, or infection, require medical treatment. We present two typical cases of EIPD and one case of a rare single esophageal intramural diverticulum. (Ann Thorac Surg 2016;101:769–71) Ó 2016 by The Society of Thoracic Surgeons
770
CASE REPORT HUBBARD AND DEMOS ESOPHAGEAL INTRAMURAL DIVERTICULA
Ann Thorac Surg 2016;101:769–71
FEATURE ARTICLES
Fig 3. Biopsy specimen from patient 1 showing goblet metaplasia of Barrett’s (left) and hyperplasia of the wall of the pseudodiverticulum (right). (HNE, 100x.)
blood transfusions. A barium swallow revealed extensive intramural pseudodiverticulosis and hiatal hernia. On endoscopy, bloody orifices were seen of numerous small diverticula similar to those in patient 1 (Fig 4). Suspected of having opioid dependency, she signed out of the hospital against medical advice.
Patient 3 A 6-year-old boy was admitted to the hospital because of dysphagia. An esophagram revealed one medium-size intramural diverticulum (Fig 5). Excision resulted in cure.
Fig 4. Endoscopic view of patient 2 showing bleeding intramural diverticula.
Fig 5. Single intramural diverticulum in patient 3, anteroposterior view of esophogram (Operation performed at Jersey City Medical Center, Jersey City, NJ by Dr Ralph Gualtieri).
Comment In 1975, only 11 cases of esophageal intramural pseudodiverticulosis (EPID) had been reported. As of 2006, only 220 had been reported [1]. EIPD can occur at any age, although it is much more common in older adults; the average age is 53.3 years, and the higher incidence occurs in men [1]. As of 2000, 13 cases had been reported in children [2, 3]. Esophageal pseudodiverticula were first reported in 1960 [4]. The pathophysiology of EIPD is poorly understood. It was hypothesized that EIPD was the result of asynchronous contraction producing increased pressure and formation of diverticula in weakened points of the esophageal wall. No single dysmotility pattern was detected [4]. Inflammatory obstruction of deep mucosal glands was thought to play a role in the etiology [5]. With continued analysis, it is thought today that a chemical, mechanical, or infectious process causes obstruction of the outlet of excretory ducts. The obstruction causes reactive hypertrophy with increased intraluminal pressure resulting in a pseudodiverticulum [1, 3]. EPID is regularly associated with severe esophagitis [1], as in our first patient. A higher incidence of esophageal intramural diverticulosis is seen in patients with diabetes mellitus, hiatal hernias, and chronic alcoholism [1, 6]. Patient 1 had two of these three conditions. Although dysphagia is the most common presentation, odynophagia is more common in patients with hiatal hernia. Hemorrhage is associated with EIPD, as seen in patient 2, whose case was first published in 2001 [6]. A strong association with esophageal webs was first seen by Hahne and colleagues [6].
Ann Thorac Surg 2016;101:771–3
CASE REPORT NANDATE ET AL GUNSHOT INDUCED AAF DIAGNOSED BY INTRAOPERATIVE TEE
The authors wish to thank Tara Jane Becker and Ioana Agams for editorial assistance.
References 1. Teraishi F, Fujiwara T, Jikuhara A, et al. Esophageal intermural pseudodiverticulosis with esophageal strictures successfully treated with dilation therapy. Ann Thorac Surg 2006;82:1119–21. 2. Demos NJ. The stapled, uncut gastroplasty for hiatal hernia: 24 years’ follow-up. Dis Esophagus 1999;12:14–21. Ó 2016 by The Society of Thoracic Surgeons Published by Elsevier
3. Lingaraj K, Prabhakaran K, Quak SH. Esophageal intramural pseudodiverticulosis associated with a web in a 12-year old boy. J Pediatr Surg 1999;33:1573–4. 4. Mendl K, McKay JM, Tanner CH. Intramural diverticulosis of the oesophagus and Rokitansky-Aschoff sinuses in the gallbladder. Br J Radiol 1960;33:496–501. 5. O’Connor OJ, Brady A, Shanahan F, Quigley E, O’Riordain M, Maher M. Esophageal intramural pseudodiverticulosis characterized by barium esophagography: a case report. J Med Case Rep 2010;4:145. 6. Hahne M, Schilling D, Arnold J, Riemann JF. Esophageal intramural pseudodiverticulosis: review of symptoms including upper gastrointestinal bleeding. J Clin Gastroenterol 2001;33:378–82. 7. Solomon NS, Berseth CL, Braverman RM, Munded MM, Ferry GD. Infantile esophageal intramural pseudodiverticulosis. J Pediatr Gastroenterol Nutr 2000;31:76–9. 8. Freud E, Golinsky D, Ziv N, Mor C, Zahavi I, Zer M. Esophageal intramural pseudodiverticulosis: a congenital or acquired condition. J Pediatr Gastroenterol Nutr 1997;24:602–7.
Gunshot-Induced Aorto-Left Atrial Fistula Diagnosed by Intraoperative Transesophageal Echocardiography Koichiro Nandate, MD, PhD, Vijay Krishnamoorthy, MD, Lisa K. McIntyre, MD, Edward D. Verrier, MD, and G. Burkhard Mackensen, MD, PhD Department of Anesthesiology and Pain Medicine; and Divisions of Trauma Surgery, and Cardiothoracic Surgery, Department of Surgery, University of Washington, Seattle, Washington
Aorto-left atrial fistula (AAF) is rarely encountered in clinical practice, and the early diagnosis can be very challenging. This report describes a unique case of AAF caused by a gunshot injury and the pivotal role of transesophageal echocardiography for diagnosis and assessment. (Ann Thorac Surg 2016;101:771–3) Ó 2016 by The Society of Thoracic Surgeons
F
istulae between the aorta and the left atrium are rarely observed, and previous case reports have been associated with complications of cardiovascular disease, surgery, or intervention [1, 2]. Here we present a unique case of a gunshot-induced aorto-left atrial fistula (AAF) diagnosed by intraoperative transesophageal echocardiography. A 19-year-old male suffered a gunshot wound to the thorax, and he was resuscitated at the scene by emergency personnel prior to being transported to the emergency department (ED) at our institution. Initial physical examination in the ED revealed a very narrow pulse
Accepted for publication Feb 6, 2015. Address correspondence to Dr Nandate, Department of Anesthesiology & Pain Medicine, University of Washington, Box 356171, Seattle, WA 98195; email: [email protected].
0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2015.02.129
FEATURE ARTICLES
Inflammation of the submucosal glands and excretory duct dilatation have been identified [1]. Inflammatory cells and submucosal fibrosis are also seen [1]. It is unknown what leads to duct outlet obstruction, and this gap in knowledge raises the question of compound causes and secondary or primary esophageal infections. A barium swallow will show structure, hypertonicity, hypotonicity, or a combination in almost every case [5]. Some literature reports cite more than 90% of patients with EIPD having strictures on endoscopy. A barium esophagram shows flasks or button-shaped outpouchings consistent with dilatation of the submucosal glands [1]. The literature suggests that as little as 20% to 25% of EIPD are found on endoscopy [1, 5, 3]. Esophageal testing may reveal esophageal dysmotility. This is a classic example of the inflammatory cause versus dysmotility cause that has been debated for more than 50 years. The main goal of treatment is correction of the underlying pathologic disorder. Strictures respond to dilation, proton pump inhibitors, and antifungal medication [6]. Fundoplication stops the acid reflux and the mucosal injury, which, if allowed to continue, result in enlargement of the diverticula after only months and may lead to eventually carcinoma. Single diverticula are rare. As of 2000, 13 cases had been reported in children [3]. The youngest patient reported was 37 days old, the mean age being 11 years. The presenting symptom is dysphagia. Diagnosis is made by esophagram [1, 7]. Although it is a benign condition, fistulas to the mediastinum may be fatal [7]. It is still unknown whether pediatric esophageal intramural diverticulum is acquired or congenital [7, 8]. The congenital origin is supported by the association of other congenital anomalies such as tracheoesophageal fistula and esophageal webs [8]. The acquired origin is supported by development from a normal esophagus to one with esophageal intramural diverticulum in as short a time as 7 months [7]. Association with gastroesophageal reflux, strictures, corrosive acid ingestion, and dysmotility have been reported [8]. Asthma, producing increased negative-positive intrathoracic pressures, may play a role [7]. Reflux has been known to provoke asthmatic attacks [2]. The child (patient 3) had a single intramural diverticulum without other identifiable cause. Excision cured the child.
771
CASE REPORT HUBBARD AND DEMOS ESOPHAGEAL INTRAMURAL DIVERTICULA
769
4. Richardson JD. Outcome of tracheobronchial injuries: a longterm perspective. J Trauma 2004;56:30–6. 5. Huh j, Milliken JC, Chen JC. . Management of tracheobronchial injuries following blunt and penetrating trauma. Am Surg 1997;63:896–9. 6. Hood MR. Injury to the trachea and major bronchi. In: Hood MR, Arthur BD, Culliford AT (eds). Thoracic trauma. Philadelphia: WB Saunders; 1989. 7. Chu CP, Chen PP. Tracheobronchial injury secondary to blunt chest trauma: diagnosis and management. Anaesth Intensive Care 2002;30:145–52. 8. Kara H, Bayir A, Ak A, Tufekci N, Degirmenci S, Akinci M. Tracheal rupture developing after blunt thoracic trauma. Case Rep Clin Med 2013;2:502–4.
Esophageal Intramural Diverticula: Multiple and Single Jennifer Hubbard, BS, and Nicholas J. Demos, MD Ross University School of Medicine, Miramar, Florida, and Hoboken University Medical Center, Hoboken, New Jersey
D
iverticula in the human body usually produce symptoms caused by pressure on the surrounding organs or by the deterioration of their mucosal lining. However, intramural esophageal diverticula are associated with symptoms of gastroesophageal reflux or related symptoms and signs not quite related to their pathogenesis or pathophysiology. We review the cases of three patients, which may somewhat illuminate the nature of these unusual and rare diverticula.
Fig 1. Esophagram of patient 1 showing multiple intramural diverticula, a moderate lower esophageal stricture, and a hiatal hernia.
the wall, and areas of Barrett’s esophagus (Fig 3). Motility and scan studies showed esophagogastroparesis. Through a transabdominal approach, a partial fundoplication, a loop gastroenterostomy and, dilation of the esophageal stricture were performed. At the sixthmonth follow-up visit, his strength and nutritional status were restored. At the 15-year follow-up visit, the man described no symptoms.
Patient 2 A 49-year-old woman was admitted with massive bloody vomiting. Her hemoglobin level was restored with two
Case Reports Patient 1 A 57-year-old man with a history of gastroesophageal reflux disease and diabetes mellitus had stopped eating. He was malnourished, with electrolyte imbalance. An esophagram revealed multiple intramural pseudodiverticula, a moderate lower esophageal stricture, and a hiatal hernia (Fig 1). Endoscopy showed food impaction, a mild stricture, and a hiatal hernia (Fig 2). Biopsy revealed pseudodiverticula of the esophagus, hypertrophy of Accepted for publication March 10, 2015. Address correspondence to Dr Demos, 4 Cambridge Dr, Short Hills, NJ 07078; email: [email protected].
Ó 2016 by The Society of Thoracic Surgeons Published by Elsevier
Fig 2. Endoscopic view of patient 1 showing multiple small pits throughout the esophagus were seen. In addition, severe lower esophagitis, a mild stricture, and sliding hiatal hernia were identified. The arrow points to the opening of the intramural diverticulum. 0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2015.03.045
FEATURE ARTICLES
More than 50 years ago, two investigators described a rare case of multiple esophageal diverticula, which they named esophageal intramural pseudodiverticulosis (EIPD). To this day, the pathogenesis of this condition remains obscure. Several coexisting conditions, such as strictures, esophageal dysmotility, or infection, require medical treatment. We present two typical cases of EIPD and one case of a rare single esophageal intramural diverticulum. (Ann Thorac Surg 2016;101:769–71) Ó 2016 by The Society of Thoracic Surgeons
770
CASE REPORT HUBBARD AND DEMOS ESOPHAGEAL INTRAMURAL DIVERTICULA
Ann Thorac Surg 2016;101:769–71
FEATURE ARTICLES
Fig 3. Biopsy specimen from patient 1 showing goblet metaplasia of Barrett’s (left) and hyperplasia of the wall of the pseudodiverticulum (right). (HNE, 100x.)
blood transfusions. A barium swallow revealed extensive intramural pseudodiverticulosis and hiatal hernia. On endoscopy, bloody orifices were seen of numerous small diverticula similar to those in patient 1 (Fig 4). Suspected of having opioid dependency, she signed out of the hospital against medical advice.
Patient 3 A 6-year-old boy was admitted to the hospital because of dysphagia. An esophagram revealed one medium-size intramural diverticulum (Fig 5). Excision resulted in cure.
Fig 4. Endoscopic view of patient 2 showing bleeding intramural diverticula.
Fig 5. Single intramural diverticulum in patient 3, anteroposterior view of esophogram (Operation performed at Jersey City Medical Center, Jersey City, NJ by Dr Ralph Gualtieri).
Comment In 1975, only 11 cases of esophageal intramural pseudodiverticulosis (EPID) had been reported. As of 2006, only 220 had been reported [1]. EIPD can occur at any age, although it is much more common in older adults; the average age is 53.3 years, and the higher incidence occurs in men [1]. As of 2000, 13 cases had been reported in children [2, 3]. Esophageal pseudodiverticula were first reported in 1960 [4]. The pathophysiology of EIPD is poorly understood. It was hypothesized that EIPD was the result of asynchronous contraction producing increased pressure and formation of diverticula in weakened points of the esophageal wall. No single dysmotility pattern was detected [4]. Inflammatory obstruction of deep mucosal glands was thought to play a role in the etiology [5]. With continued analysis, it is thought today that a chemical, mechanical, or infectious process causes obstruction of the outlet of excretory ducts. The obstruction causes reactive hypertrophy with increased intraluminal pressure resulting in a pseudodiverticulum [1, 3]. EPID is regularly associated with severe esophagitis [1], as in our first patient. A higher incidence of esophageal intramural diverticulosis is seen in patients with diabetes mellitus, hiatal hernias, and chronic alcoholism [1, 6]. Patient 1 had two of these three conditions. Although dysphagia is the most common presentation, odynophagia is more common in patients with hiatal hernia. Hemorrhage is associated with EIPD, as seen in patient 2, whose case was first published in 2001 [6]. A strong association with esophageal webs was first seen by Hahne and colleagues [6].
Ann Thorac Surg 2016;101:771–3
CASE REPORT NANDATE ET AL GUNSHOT INDUCED AAF DIAGNOSED BY INTRAOPERATIVE TEE
The authors wish to thank Tara Jane Becker and Ioana Agams for editorial assistance.
References 1. Teraishi F, Fujiwara T, Jikuhara A, et al. Esophageal intermural pseudodiverticulosis with esophageal strictures successfully treated with dilation therapy. Ann Thorac Surg 2006;82:1119–21. 2. Demos NJ. The stapled, uncut gastroplasty for hiatal hernia: 24 years’ follow-up. Dis Esophagus 1999;12:14–21. Ó 2016 by The Society of Thoracic Surgeons Published by Elsevier
3. Lingaraj K, Prabhakaran K, Quak SH. Esophageal intramural pseudodiverticulosis associated with a web in a 12-year old boy. J Pediatr Surg 1999;33:1573–4. 4. Mendl K, McKay JM, Tanner CH. Intramural diverticulosis of the oesophagus and Rokitansky-Aschoff sinuses in the gallbladder. Br J Radiol 1960;33:496–501. 5. O’Connor OJ, Brady A, Shanahan F, Quigley E, O’Riordain M, Maher M. Esophageal intramural pseudodiverticulosis characterized by barium esophagography: a case report. J Med Case Rep 2010;4:145. 6. Hahne M, Schilling D, Arnold J, Riemann JF. Esophageal intramural pseudodiverticulosis: review of symptoms including upper gastrointestinal bleeding. J Clin Gastroenterol 2001;33:378–82. 7. Solomon NS, Berseth CL, Braverman RM, Munded MM, Ferry GD. Infantile esophageal intramural pseudodiverticulosis. J Pediatr Gastroenterol Nutr 2000;31:76–9. 8. Freud E, Golinsky D, Ziv N, Mor C, Zahavi I, Zer M. Esophageal intramural pseudodiverticulosis: a congenital or acquired condition. J Pediatr Gastroenterol Nutr 1997;24:602–7.
Gunshot-Induced Aorto-Left Atrial Fistula Diagnosed by Intraoperative Transesophageal Echocardiography Koichiro Nandate, MD, PhD, Vijay Krishnamoorthy, MD, Lisa K. McIntyre, MD, Edward D. Verrier, MD, and G. Burkhard Mackensen, MD, PhD Department of Anesthesiology and Pain Medicine; and Divisions of Trauma Surgery, and Cardiothoracic Surgery, Department of Surgery, University of Washington, Seattle, Washington
Aorto-left atrial fistula (AAF) is rarely encountered in clinical practice, and the early diagnosis can be very challenging. This report describes a unique case of AAF caused by a gunshot injury and the pivotal role of transesophageal echocardiography for diagnosis and assessment. (Ann Thorac Surg 2016;101:771–3) Ó 2016 by The Society of Thoracic Surgeons
F
istulae between the aorta and the left atrium are rarely observed, and previous case reports have been associated with complications of cardiovascular disease, surgery, or intervention [1, 2]. Here we present a unique case of a gunshot-induced aorto-left atrial fistula (AAF) diagnosed by intraoperative transesophageal echocardiography. A 19-year-old male suffered a gunshot wound to the thorax, and he was resuscitated at the scene by emergency personnel prior to being transported to the emergency department (ED) at our institution. Initial physical examination in the ED revealed a very narrow pulse
Accepted for publication Feb 6, 2015. Address correspondence to Dr Nandate, Department of Anesthesiology & Pain Medicine, University of Washington, Box 356171, Seattle, WA 98195; email: [email protected].
0003-4975/$36.00 http://dx.doi.org/10.1016/j.athoracsur.2015.02.129
FEATURE ARTICLES
Inflammation of the submucosal glands and excretory duct dilatation have been identified [1]. Inflammatory cells and submucosal fibrosis are also seen [1]. It is unknown what leads to duct outlet obstruction, and this gap in knowledge raises the question of compound causes and secondary or primary esophageal infections. A barium swallow will show structure, hypertonicity, hypotonicity, or a combination in almost every case [5]. Some literature reports cite more than 90% of patients with EIPD having strictures on endoscopy. A barium esophagram shows flasks or button-shaped outpouchings consistent with dilatation of the submucosal glands [1]. The literature suggests that as little as 20% to 25% of EIPD are found on endoscopy [1, 5, 3]. Esophageal testing may reveal esophageal dysmotility. This is a classic example of the inflammatory cause versus dysmotility cause that has been debated for more than 50 years. The main goal of treatment is correction of the underlying pathologic disorder. Strictures respond to dilation, proton pump inhibitors, and antifungal medication [6]. Fundoplication stops the acid reflux and the mucosal injury, which, if allowed to continue, result in enlargement of the diverticula after only months and may lead to eventually carcinoma. Single diverticula are rare. As of 2000, 13 cases had been reported in children [3]. The youngest patient reported was 37 days old, the mean age being 11 years. The presenting symptom is dysphagia. Diagnosis is made by esophagram [1, 7]. Although it is a benign condition, fistulas to the mediastinum may be fatal [7]. It is still unknown whether pediatric esophageal intramural diverticulum is acquired or congenital [7, 8]. The congenital origin is supported by the association of other congenital anomalies such as tracheoesophageal fistula and esophageal webs [8]. The acquired origin is supported by development from a normal esophagus to one with esophageal intramural diverticulum in as short a time as 7 months [7]. Association with gastroesophageal reflux, strictures, corrosive acid ingestion, and dysmotility have been reported [8]. Asthma, producing increased negative-positive intrathoracic pressures, may play a role [7]. Reflux has been known to provoke asthmatic attacks [2]. The child (patient 3) had a single intramural diverticulum without other identifiable cause. Excision cured the child.
771