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Shintaku M. Esophageal intramural pseudodiverticulosis. World J Gastroenterol 2024; 30:137-145. [PMID: 38312118 PMCID: PMC10835521 DOI: 10.3748/wjg.v30.i2.137] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/19/2023] [Revised: 12/21/2023] [Accepted: 12/28/2023] [Indexed: 01/12/2024] Open
Abstract
Esophageal intramural pseudodiverticulosis (EIPD) is a disease of unknown pathogenesis characterized by usually systemic, cystic dilatation of the excretory ducts of esophageal submucosal glands. In this article, I review the epidemiology, clinical manifestations, endoscopic findings, esophagographic findings, and histopathology of EIPD. I also discuss the etiology and possible pathogenesis of EIPD based on my experiences with this disease and a review of the literature. EIPD usually presents with dysphagia in middle-aged individuals. It is often complicated with secondary infections, most commonly candidiasis. On esophagography, EIPD is delineated as small, multiple, flask-shaped outward projections within the esophageal wall. In recent years, EIPD has been mainly diagnosed by endoscopic findings of multiple, localized, small mucosal depressions. The orifices of the "pseudodiverticula" periodically open and close, and excrete mucus onto the mucosal surface. On histopathological examination, the luminal surface of dilated ducts in EIPD is covered by multilayered, hyperplastic epithelial cells, but myoepithelial cells in the glandular acini are well preserved. Treatment of EIPD is usually symptomatic therapy, and prevention of the infectious complications is important. The etiology and pathogenesis of EIPD are largely unknown, but functional abnormalities of autonomic nerve fibers innervating the esophageal glands likely play an important role, since the structures of the glands are basically preserved in this disease.
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Affiliation(s)
- Masako Shintaku
- Department of Gastroenterology, Japan Community Healthcare Organization Hoshigaoka Medical Center, Hirakata 573-8511, Osaka, Japan
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Raza D, Mubashir M, Zia HA. Outpouching in the Esophagus: An Uncommon Endoscopic Finding of Esophageal Intramural Pseudodiverticulosis in the Absence of Esophageal Candidiasis. Cureus 2023; 15:e39805. [PMID: 37398731 PMCID: PMC10313527 DOI: 10.7759/cureus.39805] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 05/31/2023] [Indexed: 07/04/2023] Open
Abstract
We present a case of a 51-year-old female with a history of acquired immunodeficiency syndrome (AIDS) and medication non-compliance who experienced progressively worsening dysphagia to both solids and liquids over a three-month period. The patient underwent an esophagogastroduodenoscopy (EGD), which revealed multiple small pseudodiverticula without any other notable abnormalities. Subsequently, a barium esophagogram was performed, confirming the presence of multiple esophageal pseudodiverticula. Biopsies taken during the procedure showed chronic inflammatory changes, with no evidence of viral or fungal elements. In light of the patient's HIV history and the absence of esophageal candidiasis, the diagnosis of esophageal intramural pseudodiverticulosis (EIP) was made. The patient was initiated on highly active antiretroviral therapy (HAART) and received high-dose proton pump inhibitors (PPIs). Remarkably, the patient reported a complete resolution of her dysphagia symptoms during the follow-up visit. Risk factors associated with EIP include HIV infection, diabetes mellitus (DM), and esophageal candidiasis. To confirm the diagnosis, a barium esophagogram is considered the preferred imaging study. The management of EIP focuses on PPI therapy, the dilation of strictures if present, and addressing the underlying etiology. Given the association between EIP and esophageal malignancies, surveillance endoscopy may be recommended in these patients. This case highlights the importance of considering EIP as a potential cause of dysphagia, particularly in individuals with HIV/AIDS, even in the absence of esophageal candidiasis. Prompt diagnosis and appropriate management can lead to symptom resolution and improved quality of life for affected patients.
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Affiliation(s)
- Daniyal Raza
- Internal Medicine, Louisiana State University (LSU) Health Shreveport, Shreveport, USA
| | - Maryam Mubashir
- Gastroenterology and Hepatology, Louisiana State University (LSU) Health Shreveport, Shreveport, USA
| | - Hassaan A Zia
- Gastroenterology and Hepatology, Louisiana State University (LSU) Health Shreveport, Shreveport, USA
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Spontaneous Rupture of Esophageal Diverticulum-A Case Report and Literature Review. Diagnostics (Basel) 2022; 13:diagnostics13010019. [PMID: 36611314 PMCID: PMC9818745 DOI: 10.3390/diagnostics13010019] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/09/2022] [Revised: 12/11/2022] [Accepted: 12/19/2022] [Indexed: 12/24/2022] Open
Abstract
The spontaneous rupture of the esophageal diverticulum is a rare condition that occurs without any warning signs. Its incidence is low, but the mortality rate is high. This paper reports a case of spontaneous esophageal diverticulum rupture and analyzes it along with 13 other cases to explore its prevention and treatment measures. When patients suffer from chronic swallowing difficulties and chest pain or vomiting that cannot be explained after meals, they should be suspected to have a possible spontaneous rupture of the esophageal diverticulum, which is critical to the patient's prognosis.
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Frieling T, Kreysel C, Blank M, Mülle D, Euler P, Melchior I. Not always eosinophilic esophagitis – intramural pseudodiverticulosis of the esophagus – a case report and literature review. ZEITSCHRIFT FUR GASTROENTEROLOGIE 2020; 58:1201-1207. [DOI: 10.1055/a-1288-1521] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/27/2022]
Abstract
Abstract
Background Intramural pseudodiverticulosis of the esophagus (EIPD) is a rare disease leading to dysphagia, chest pain, and weight loss. The diagnosis is difficult, and the disease can be confounded with eosinophilic esophagitis (EoE). We present a patient with esophageal intramural pseudodiverticulosis and a literature review.
Case report The 45-year-old white caucasian woman with a history of nicotine and alcohol abuse had progressive hoarseness and severe dysphagia for solid food. Esophagogastroduodenoscopy (EGD) showed proximal esophageal stenosis, thrush esophagitis, and mucosal alteration with trachealization suspicious of EoE. However, repeated bouginage EGD and barium swallow revealed typical signs of esophageal intramural pseudodiverticulosis (EIPD). The patient was treated successfully by bougingage, acid suppression, and antifungal therapy. The literature analysis revealed the characteristics of EIPD according to age, sex, risk factors, and therapy modalities.
Conclusion The case report and the literature overview suggest that EIPD can be confounded with EoE.
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Affiliation(s)
- Thomas Frieling
- Department of Internal Medicine and Gastroenterology, HELIOS Clinic Krefeld, Germany
| | - Christian Kreysel
- Department of Internal Medicine and Gastroenterology, HELIOS Clinic Krefeld, Germany
| | - Michael Blank
- Department of Internal Medicine and Gastroenterology, HELIOS Clinic Krefeld, Germany
| | - Dorothee Mülle
- Department of Internal Medicine and Gastroenterology, HELIOS Clinic Krefeld, Germany
| | - Philipp Euler
- Department of Internal Medicine and Gastroenterology, HELIOS Clinic Krefeld, Germany
| | - Ilka Melchior
- Department of Internal Medicine and Gastroenterology, HELIOS Clinic Krefeld, Germany
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Ali O, Asumu H, Kaur T, Mathew A, Kim R. A rare cause of dysphagia due to esophageal intramural pseudodiverticulosis: a case report and review of literature. BMC Gastroenterol 2020; 20:72. [PMID: 32178627 PMCID: PMC7074979 DOI: 10.1186/s12876-020-01209-y] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/08/2019] [Accepted: 02/28/2020] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Esophageal intramural pseudodiverticulosis is an uncommon, idiopathic disorder characterized by multiple small outpouchings protruding from the esophageal lumen. Esophageal intramural pseudodiverticulosis is associated with conditions such as gastroesophageal reflux disease and diabetes mellitus, as well as emergent complications including pneumomediastinum. The most common presenting symptom is dysphagia with associated esophageal stricture formation. While the pathogenesis of EIP has yet to be determined, it is important to bring awareness to this unique disease with distinctive diagnostic findings and treatment options. CASE PRESENTATION In this case, we present a 62-year-old woman who suffered from dysphagia, an inability to tolerate a regular diet, and unintentional weight loss for several years prior to her diagnoses. She was diagnosed by esophagram and esophagogastroduodenoscopy to have esophageal intramural pseudodiverticulosis, complicated by severe stricture formation. Following treatment with sequential dilatation and maintenance H2-blocker therapy, she achieved significant symptomatic improvement. CONCLUSIONS This case highlights the importance of accurate identification and treatment of an uncommon cause of dysphagia, esophageal intramural pseudodiverticulosis. Treatment includes dilatational therapy, as successfully demonstrated in our patient. Furthermore, treatment is focused on optimizing medical management, as demonstrated in our patient with the addition of an H2-blocker for GERD, or addressing potentially serious underlying causes, such as carcinoma, with surgery.
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Affiliation(s)
- Osman Ali
- Department of Internal Medicine, University of Maryland Medical Center Midtown Campus, Baltimore, MD, USA
| | - Hazel Asumu
- Department of Internal Medicine, University of Maryland Medical Center Midtown Campus, Baltimore, MD, USA
| | - Tanisha Kaur
- Department of Internal Medicine, University of Maryland Medical Center Midtown Campus, Baltimore, MD, USA
| | - Angelina Mathew
- Department of Internal Medicine, University of Maryland Medical Center Midtown Campus, Baltimore, MD, USA
| | - Raymond Kim
- Department of Internal Medicine, University of Maryland Medical Center Midtown Campus, Baltimore, MD, USA.
- Department of Gastroenterology and Hepatology, University of Maryland School of Medicine, Baltimore, MD, 21201, USA.
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Esophageal intramural pseudodiverticulosis complicated with severe stricture requiring surgical resection. Clin J Gastroenterol 2019; 12:292-295. [PMID: 30697669 DOI: 10.1007/s12328-019-00940-8] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/14/2018] [Accepted: 01/24/2019] [Indexed: 10/27/2022]
Abstract
Esophageal intramural pseudodiverticulosis (EIPD) is an uncommon benign disorder leading to esophageal strictures. The etiology remains unknown; however, anti-fungal treatments or endoscopic balloon dilation can improve early esophageal strictures and these rarely require surgical treatment. We report a case of a 46-year-old male with a 6 cm-long esophageal stricture due to EIPD, which did not improve following treatment with an anti-fungal agent, eventually causing aspiration pneumonia. Therefore, we performed a thoraco-laparoscopic esophagectomy, and his symptoms were improved after surgery. This case suggests that a surgical treatment should be considered in patients with extensive, severe strictures attributable to EIPD.
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Takeshita N, Kanda N, Fukunaga T, Kimura M, Sugamoto Y, Tasaki K, Uesato M, Sazuka T, Maruyama T, Aida N, Tamachi T, Hosokawa T, Asai Y, Matsubara H. Esophageal intramural pseudodiverticulosis of the residual esophagus after esophagectomy for esophageal cancer. World J Gastroenterol 2015; 21:9223-9227. [PMID: 26290650 PMCID: PMC4533055 DOI: 10.3748/wjg.v21.i30.9223] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/09/2015] [Revised: 04/07/2015] [Accepted: 06/15/2015] [Indexed: 02/06/2023] Open
Abstract
A 91-year-old man was referred to our hospital with intermittent dysphagia. He had undergone esophagectomy for esophageal cancer (T3N2M0 Stage III) 11 years earlier. Endoscopic examination revealed an anastomotic stricture; signs of inflammation, including redness, erosion, edema, bleeding, friability, and exudate with white plaques; and multiple depressions in the residual esophagus. Radiographical examination revealed numerous fine, gastrografin-filled projections and an anastomotic stricture. Biopsy specimens from the area of the anastomotic stricture revealed inflammatory changes without signs of malignancy. Candida glabrata was detected with a culture test of the biopsy specimens. The stricture was diagnosed as a benign stricture that was caused by esophageal intramural pseudodiverticulosis. Accordingly, endoscopic balloon dilatation was performed and anti-fungal therapy was started in the hospital. Seven weeks later, endoscopic examination revealed improvement in the mucosal inflammation; only the pseudodiverticulosis remained. Consequently, the patient was discharged. At the latest follow-up, the patient was symptom-free and the pseudodiverticulosis remained in the residual esophagus without any signs of stricture or inflammation.
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MESH Headings
- Aged, 80 and over
- Antifungal Agents/therapeutic use
- Biopsy
- Candida glabrata/isolation & purification
- Candidiasis/microbiology
- Deglutition Disorders/etiology
- Dilatation
- Diverticulosis, Esophageal/diagnosis
- Diverticulosis, Esophageal/etiology
- Diverticulosis, Esophageal/microbiology
- Diverticulosis, Esophageal/therapy
- Diverticulum, Esophageal/diagnosis
- Diverticulum, Esophageal/etiology
- Diverticulum, Esophageal/microbiology
- Diverticulum, Esophageal/therapy
- Esophageal Neoplasms/pathology
- Esophageal Neoplasms/surgery
- Esophageal Stenosis/etiology
- Esophagectomy/adverse effects
- Esophagoscopy
- Humans
- Male
- Neoplasm Staging
- Risk Factors
- Tomography, X-Ray Computed
- Treatment Outcome
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Liechty J, Wood R. Operative management of pulmonary abscess due to spontaneous perforation of diffuse intramural esophageal pseudodiverticulosis. Proc (Bayl Univ Med Cent) 2011; 24:216-9. [PMID: 21738294 DOI: 10.1080/08998280.2011.11928718] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/18/2022] Open
Abstract
Diffuse intramural esophageal pseudodiverticulosis is a rare, benign esophageal condition of unclear cause, pathologically manifested by pseudodiverticula along a portion of the esophagus. It most commonly presents as dysphagia and is usually amenable to endoscopic dilation. It is most reliably diagnosed endoscopically or with a fluoroscopic swallow study. Surgical intervention is rarely indicated but can create a need for an extensive resection and reconstruction, as in the case presented here.
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Affiliation(s)
- Joseph Liechty
- Department of Surgery, Baylor University Medical Center at Dallas
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Liu SM, Wu HH, Chang KK, Tseng LJ, Han SC, Mo LR. Esophageal intramural pseudodiverticulosis complicated with stricture. J Formos Med Assoc 2010; 109:241-4. [PMID: 20434033 DOI: 10.1016/s0929-6646(10)60048-4] [Citation(s) in RCA: 11] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/13/2007] [Revised: 04/08/2008] [Accepted: 06/12/2008] [Indexed: 11/18/2022] Open
Abstract
We report a rare case of diffuse esophageal intramural pseudodiverticulosis in a 35-year-old man complaining of severe dysphagia and vomiting for several months. The advanced morphological change in the esophagus caused irregular track formation, mimicking an ulcerative lesion on esophagogram. Endoscopic examination revealed an esophageal stricture with intact mucosa. Endoscopic ultrasonography and chest computed tomography showed multiple hyperechoic lesions of unknown nature and multiple air collection sites in the esophageal wall, respectively, making diagnosis difficult. The patient finally received a subtotal esophagectomy because of severe symptoms. The lesion was pathologically proven to be intramural pseudodiverticulosis with marked submucosal fibrosis. Our experience suggests that awareness of this rare pathology and the related image changes will be helpful for early diagnosis and treatment in the future.
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Affiliation(s)
- Shian-Min Liu
- Department of Pathology, Tainan Municipal Hospital, Tainan, Taiwan.
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Engel MA, Raithel M, Amann K, Greess H, Hahn EG, Konturek PC. Rare coincidence of eosinophilic esophagitis with esophageal stenosis and intramural pseudodiverticulosis. Dig Liver Dis 2008; 40:700-6. [PMID: 17643358 DOI: 10.1016/j.dld.2007.04.011] [Citation(s) in RCA: 9] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/18/2006] [Revised: 04/10/2007] [Accepted: 04/23/2007] [Indexed: 12/11/2022]
Abstract
We describe the first detailed case of eosinophilic esophagitis associated with esophageal intramural pseudodiverticulosis and gastro-esophageal reflux disease in a 24-year-old man, who suffered from recurrent dysphagia since the age of 3 years. He presented with symptoms of dysphagia, food impaction and malnutrition. An esophagogram revealed a high-grade stenosis in the proximal part of the esophagus. Histological evaluation of esophageal mucosal biopsies demonstrated more than 20 eosinophil granulocytes per high power field, indicative of eosinophilic esophagitis. Additionally, esophago-gastro-duodenoscopy showed pseudodiverticulosis in the distal portion of the esophagus. A therapeutic regimen consisting of topical steroid intake, antihistamines, proton-pump-inhibition and specific food avoidance led to significant clinical improvement within 6 weeks.
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Affiliation(s)
- M A Engel
- First Department of Medicine, Friedrich-Alexander-University Erlangen-Nuremberg, Ulmenweg 18, D-91054 Erlangen, Germany.
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Matsushita M, Uchida K, Okazaki K. Dermatomyositis accompanied by bleeding esophageal diverticula or intramural pseudodiverticulosis? Dig Dis Sci 2008; 53:592-3. [PMID: 17597399 DOI: 10.1007/s10620-007-9889-y] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/07/2007] [Accepted: 05/21/2007] [Indexed: 12/09/2022]
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Thibodeau MP, Brigand C, Ferraro P, Martin J, Duranceau A. Esophagectomy for complications of esophageal intramural pseudodiverticulosis. Dis Esophagus 2007; 20:178-82. [PMID: 17439604 DOI: 10.1111/j.1442-2050.2007.00666.x] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/11/2022]
Abstract
This report describes the clinical course of a patient with complications of esophageal intramural pseudodiverticulosis. The condition led to fistulization and abscess formation in the mediastinum. The initial presentation was for the septic process and appropriate antibiotic therapy led to infection control while the abscess drained spontaneously back into the esophageal lumen. A long stricture affecting the distal half of the esophagus became evident after a few months and could not be managed by repeat dilatations. After appropriate preparation, subtotal esophagectomy was offered to the patient with an initial right thoracic approach followed by laparotomy and left cervical reconstruction. A total gastric tube was used for reconstruction and placed in a substernal position. An uneventful postoperative evolution led to normal swallowing comfort.
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Affiliation(s)
- M-P Thibodeau
- Department of Surgery, Université de Montréal, Division of Thoracic Surgery, Center hospitalier de l'Université de Montréal, Montréal, Québec, Canada
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Teraishi F, Fujiwara T, Jikuhara A, Kamitani S, Morino Y, Sato K, Tanaka N. Esophageal intramural pseudodiverticulosis with esophageal strictures successfully treated with dilation therapy. Ann Thorac Surg 2006; 82:1119-21. [PMID: 16928560 DOI: 10.1016/j.athoracsur.2006.01.062] [Citation(s) in RCA: 14] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/02/2006] [Revised: 01/02/2006] [Accepted: 01/16/2006] [Indexed: 11/26/2022]
Abstract
We report a rare case of esophageal intramural pseudodiverticulosis with esophageal strictures. Barium esophagogram demonstrated multiple flask-shaped diverticula out of the esophageal wall with comprehensive luminal stenosis involving the proximal 8 cm and distal 4 cm of the esophagus. Chest computed tomographic scan demonstrated round wall thickening and several intramural gas collections of the proximal esophagus. Endoscopy revealed a fibrotic stricture and multiple small orifices of pseudodiverticula with mild inflammatory changes. Biopsy specimens showed active chronic inflammatory changes of the mucosa with candidiasis. Dysphagia improved dramatically with esophageal dilation. However, the tiny diverticula did not resolve after treatment.
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Affiliation(s)
- Fuminori Teraishi
- Division of Surgical Oncology, Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine and Dentistry, Okayama, Japan.
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Chen L, Walser EM, Schnadig V. Fatal hemorrhage secondary to ulcerated epiphrenic pseudodiverticulum. Arch Pathol Lab Med 2006; 130:867-70. [PMID: 16740042 DOI: 10.5858/2006-130-867-fhstue] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Abstract
A 47-year-old man was found unconscious after experiencing severe hematemesis. He had a history of alcohol abuse, hepatic cirrhosis, and previous episodes of gastrointestinal bleeding. Imaging studies indicated that the left gastric artery was the probable source of bleeding. A severe coagulopathy was also noted clinically. Angiographic embolization of the left gastric artery failed to staunch bleeding, and the patient died shortly after admission. Autopsy demonstrated a large, ulcerated epiphrenic esophageal pseudodiverticulum, the base of which was in close proximity to an esophageal branch of the left gastric artery. Cystic dilatation of adjacent esophageal submucosal glands was also seen. Gelatin embolic material, without associated thrombus formation, was found within the left gastric artery. Esophageal pseudodiverticulosis is an uncommon disorder that may be associated with dysphagia, stricture, and odynophagia. Bleeding and perforation are very rare complications. This case may be the first fatality proven by autopsy to be secondary to esophageal pseudodiverticulosis.
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Affiliation(s)
- Lei Chen
- Department of Cytopathology, The University of Texas Medical Branch, Galveston, TX 77555-0548, USA
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