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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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Naqvi H, Yousaf MN, Sandhu G, Bhansali D, Farooqi R. Recurrent Dysphagia Associated with Esophageal Intramural Pseudodiverticulosis. Case Rep Gastroenterol 2021; 15:551-556. [PMID: 34616255 PMCID: PMC8454220 DOI: 10.1159/000517093] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/23/2021] [Accepted: 05/03/2021] [Indexed: 11/19/2022] Open
Abstract
Esophageal intramural pseudodiverticulosis (EIPD) is a rare disorder of the esophagus characterized by the presence of outpouching flask-shaped lesions. These lesions represent false lumens that may be scattered throughout the esophageal wall. We present a rare case of EIPD complicated with esophageal strictures. The case is unique because the small lesions of EIPD remained undiagnosed for many years on prior esophagogastroduodenoscopy examinations until complicated with worsening symptoms of dysphagia due to esophageal stricture. The patient was managed with serial dilations of esophageal stricture. Diagnosing these lesions may be missed in early stages as the lesions are small and may require other radiological modalities to confirm the diagnosis. EIPD lesions itself can be managed conservatively with anti-inflammatory therapy and treatment of underlying risk factors. Those with severe disease and coexisting complications are managed with endoscopic or surgical interventions.
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Affiliation(s)
- Haider Naqvi
- Department of Medicine, Medstar Union Memorial Hospital, Baltimore, Maryland, USA.,Department of Medicine, MedStar Good Samaritan Hospital, Baltimore, Maryland, USA.,Department of Medicine, Medstar Franklin Square Medical Center, Baltimore, Maryland, USA.,Department of Medicine, MedStar Harbor Hospital, Baltimore, Maryland, USA
| | - Muhammad Nadeem Yousaf
- Department of Medicine, Medstar Union Memorial Hospital, Baltimore, Maryland, USA.,Department of Medicine, MedStar Good Samaritan Hospital, Baltimore, Maryland, USA.,Department of Medicine, Medstar Franklin Square Medical Center, Baltimore, Maryland, USA.,Department of Medicine, MedStar Harbor Hospital, Baltimore, Maryland, USA
| | - Gavneet Sandhu
- Department of Medicine, Medstar Union Memorial Hospital, Baltimore, Maryland, USA.,Department of Medicine, MedStar Good Samaritan Hospital, Baltimore, Maryland, USA.,Department of Medicine, Medstar Franklin Square Medical Center, Baltimore, Maryland, USA.,Department of Medicine, MedStar Harbor Hospital, Baltimore, Maryland, USA
| | - Deepty Bhansali
- Department of Pathology, Medstar Franklin Square Medical Center, Baltimore, Maryland, USA
| | - Rehan Farooqi
- Department of Medicine, MedStar Good Samaritan Hospital, Baltimore, Maryland, USA
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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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Bamidele OF, Olokoba AB, Bojuwoye MO, Barde AA. Oesophageal intramural pseudodiverticulosis: a rare endoscopic finding. Ghana Med J 2019; 53:184-186. [PMID: 31481816 PMCID: PMC6697774 DOI: 10.4314/gmj.v53i2.15] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022] Open
Abstract
Oesophageal intramural pseudodiverticulosis is an uncommon condition, characterized by multiple small rounded cavities seen in the oesophageal wall during oesophagogastroduodenoscopy. It is often associated with gastro-oesophageal reflux disease, achalasia, oesophageal candidiasis and diabetes mellitus. We report a 40 year old Nigerian man who presented with recurrent dysphagia and endoscopic findings typical of oesophageal intramural pseudodiverticulosis. The patient was managed medically with resolution of the dysphagia. This report highlights the occurrence of this rare and benign cause of dysphagia in Nigeria.
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Affiliation(s)
- Opeyemi F Bamidele
- Department of Medicine, Dalhatu Araf Specialist Hospital, Lafia. Nigeria
| | | | - Matthew O Bojuwoye
- Department of Medicine, University of Ilorin Teaching Hospital, Ilorin. Nigeria
| | - Amanda A Barde
- Department of Medicine, Dalhatu Araf Specialist Hospital, Lafia. Nigeria
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Akkari I, Jazia EB, Mrabet S, Jemni I. Candida albicans: a cause or a consequence of esophageal intramural pseudo-diverticulosis. Pan Afr Med J 2019; 33:280. [PMID: 31692736 PMCID: PMC6815468 DOI: 10.11604/pamj.2019.33.280.19601] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/29/2019] [Accepted: 07/02/2019] [Indexed: 11/11/2022] Open
Abstract
Esophageal intramural pseudo-diverticulosis is a rare disease of unknown etiology. It is characterized by multiple pseudodiverticula with segmental or diffuse involvement of the esophagus. We report, the case of a 78-year-old male who suffered from severe dysphagia. Diagnosis of esophageal intramural pseudo-diverticulosis was based on endoscopic and radiologic explorations. Histological analysis of esophageal mucosal biopsies has shown the presence of candida albicans. Antifungal treatment leads to spectacular improvement of dysphasia. Subsequently, the patient presented a cardio-respiratory failure and died despite adequate treatment.
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Affiliation(s)
- Imen Akkari
- Gastroenterology Department, Farhat Hached Hospital, Sousse, Tunisia
| | - Elhem Ben Jazia
- Gastroenterology Department, Farhat Hached Hospital, Sousse, Tunisia
| | - Soumaya Mrabet
- Gastroenterology Department, Farhat Hached Hospital, Sousse, Tunisia
| | - Imen Jemni
- Gastroenterology Department, Farhat Hached Hospital, Sousse, Tunisia
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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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Sawada R, Oshikiri T, Nakamura T, Yamamoto M, Matsuda T, Sumi Y, Itoh T, Zen Y, Suzuki S, Kakeji Y. Mass-Forming Deep Pseudodiverticulosis of the Esophagus With 18F-Fluorodeoxyglucose Uptake. Ann Thorac Surg 2018; 106:e309-e311. [PMID: 29859150 DOI: 10.1016/j.athoracsur.2018.04.082] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/18/2018] [Revised: 04/24/2018] [Accepted: 04/28/2018] [Indexed: 11/29/2022]
Abstract
This report describes a case of esophageal intramural pseudodiverticulosis associated with an inflammatory esophageal mass with high 18F-fluorodeoxyglucose (FDG) uptake. A 48-year-old man presented with dysphagia caused by an esophageal stricture. Simultaneous positron emission tomography and magnetic resonance imaging showed an FDG-avid lesion in the lower esophagus. Because of suspected malignancy, the patient underwent subtotal esophagectomy. Histologic examination confirmed the diagnosis of an inflammatory tumor associated with esophageal pseudodiverticulosis. Unlike typical cases, this patient's pseudodiverticula involved both the submucosa and the muscularis propria. This case suggests that esophageal intramural pseudodiverticulosis can manifest with a tumorlike mass that may be FDG positive in response to associated inflammation.
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Affiliation(s)
- Ryuichiro Sawada
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan.
| | - Taro Oshikiri
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Tetsu Nakamura
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Masashi Yamamoto
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Takeru Matsuda
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Yasuo Sumi
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Tomoo Itoh
- Department of Diagnostic Pathology, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Yoh Zen
- Department of Diagnostic Pathology, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Satoshi Suzuki
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan
| | - Yoshihiro Kakeji
- Department of Gastroenterological Surgery, Graduate School of Medicine, Kobe University, Kobe, Japan
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Esophageal intramural pseudodiverticulosis as a diagnostic and therapeutic problem. POLISH JOURNAL OF THORACIC AND CARDIOVASCULAR SURGERY 2016; 13:265-268. [PMID: 27785146 PMCID: PMC5071599 DOI: 10.5114/kitp.2016.62621] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 12/23/2014] [Accepted: 08/18/2016] [Indexed: 11/17/2022]
Abstract
The article presents the case of a 68-year-old patient with alcohol dependence syndrome, who was admitted, in serious condition, to the Department of Surgery due to esophageal intramural lesions of unclear etiology. The imaging studies showed no signs of transmural perforation of the esophageal wall. Esophagogastroscopy revealed intramural fluid reservoirs and small oval cavities with smooth edges in the esophageal mucosa. The patient was treated conservatively with parenteral nutrition and rehabilitation. Subsequently, the patient was transferred to the intensive care unit because of cardiorespiratory failure. Despite adequate pharmacological treatment, the patient died.
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A Treatment Option for Esophageal Intramural Pseudodiverticulosis. ACG Case Rep J 2014; 1:134-6. [PMID: 26157852 PMCID: PMC4435313 DOI: 10.14309/crj.2014.28] [Citation(s) in RCA: 7] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/20/2013] [Accepted: 03/18/2014] [Indexed: 12/15/2022] Open
Abstract
Esophageal intramural pseudodiverticulosis (EIPD) is a rare condition often presenting with esophageal strictures. Treatment is often limited to endoscopic dilatation and treatment of the underlying esophageal pathology. We present a case of a patient with longstanding GERD on famotidine (she experienced anaphylaxis with proton pump inhibitors [PPIs]) who presented with dysphagia and weight loss. Work-up revealed a diagnosis of EIPD with a 5-mm mid-esophageal stricture. Therapy with dilatation was unsuccessful until the addition of sucralfate, after which dilatation was successful and symptoms resolved. In patients who are unable to take PPIs, the addition of sucralfate may enhance the success of dilatations of esophageal strictures and EIPD.
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Esophageal intramural pseudodiverticulosis: a rare endoscopic finding. Case Rep Med 2013; 2013:154767. [PMID: 24348570 PMCID: PMC3852083 DOI: 10.1155/2013/154767] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/25/2013] [Revised: 09/16/2013] [Accepted: 10/10/2013] [Indexed: 11/18/2022] Open
Abstract
A 76-year-old woman, presenting with a 4-year history of progressive dysphagia, was submitted to endoscopic examination. The upper endoscopy revealed a proximal esophageal stricture and inflammatory mucosa associated with multiples small orifices in the esophageal wall, some of them fulfilled with white spots suggestive of fungal infection. This was a typical endoscopic finding of esophageal intramural pseudodiverticulosis, a benign and rare condition, related to chronic esophagitis and others comorbid states, such as gastroesophageal reflux disease or infectious esophagitis, diabetes mellitus, alcohol consumption, and achalasia. Dysphagia is the predominant symptom and can be accompanied by esophageal stricture in 80% to 90% of patients. The pathogenesis is unknown, and as the pseudodiverticulosis is an intramural finding, endoscopy biopsies are inconclusive. The main histological finding is dilation of the submucosal glands excretory ducts, probably obstructed by inflammatory cells. The treatment consists in management of the underlying diseases and symptoms relief. In this particular case, the patient was submitted to antifungal drugs followed by endoscopic dilation with thermoplastic bougies, with satisfactory improvement of dysphagia.
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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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