Ann Thorac Surg 2006;82:1119-1121
© 2006 The Society of Thoracic Surgeons
Case report
Esophageal Intramural Pseudodiverticulosis With Esophageal Strictures Successfully Treated With Dilation Therapy
Fuminori Teraishi, MDa,*,
Toshiyoshi Fujiwara, MDa,
Atsushi Jikuhara, MDb,
Shingo Kamitani, MDb,
Yasuo Morino, MDc,
Katsuaki Sato, MDb,
Noriaki Tanaka, MDa
a Division of Surgical Oncology, Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine and Dentistry, Okayama, Japan
b Department of Gastrointestinal Surgery, Sato Hospital, Okayama, Japan
c Department of Radiology, Mitsubishi Mizushima Hospital, Okayama, Japan
Accepted for publication January 16, 2006.
* Address correspondence to Dr Teraishi, Department of Gastroenterological Surgery, Okayama University Graduate School of Medicine and Dentistry, Okayama, 700-8558 Japan (Email: f-tera{at}md.okayama-u.ac.jp).
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Abstract
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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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Introduction
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Esophageal intramural pseudodiverticulosis is a rare condition with only 220 reported cases. In patients presenting with dysphagia or esophageal stricture, or both, the diagnosis of esophageal intramural pseudodiverticulosis may be initially missed, but once correctly identified, empiric therapy generally results in successful management of symptoms. Esophageal intramural pseudodiverticulosis is a benign condition and severe complications are rarely seen. In this report, we describe the use of findings from an esophagogram, a chest computed tomographic scan, and an endoscopy to diagnosis esophageal intramural pseudodiverticulosis, which was effectively treated by medication for esophagitis and endoscopic dilation.
The patient, a 67-year-old man, presented with a 1-year history of progressive dysphagia for solid foods. He also had a history of intermittent dysphagia and heartburn for 10 years with no prior roentgenogram or endoscopic examinations. He had no other significant past medical history. Physical and laboratory evaluation were unremarkable.
A barium esophagogram revealed multiple, tiny, flask-shaped outpouchings measuring from 2 to 4 mm in length with mild strictures in the upper thoracic esophagus (Fig 1). Chest computed tomographic scan demonstrated marked thickening of the esophageal wall and small intramural gas collections at several levels (Fig 2). Endoscopy revealed a fibrotic, benign stenosis 25 cm from the incisors, approximately 8 mm in diameter. Esophageal biopsies stained with hematoxylin and eosin demonstrated diffuse hypertrophy and a small number of neutrophils in the squamous epithelium characteristics of active chronic esophagitis. Microscopic examination of endoscopic brushings confirmed Candida albicans as the causing agent. Endoscopy after dilation therapy revealed multiple small dimples representing the diverticular orifices (Fig 3). The stomach and duodenum were normal. On the basis of these clinical findings, we made a diagnosis of esophageal intramural pseudodiverticulosis with chronic esophagitis.
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Fig 1. Esophagogram shows multiple collar button-shaped outpouchings of various sizes in longitudinal rows parallel to the long axis of the esophagus and associated strictures in the upper thoracic esophagus.
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Fig 2. Chest computed tomographic scan demonstrates circumferential thickening of the esophageal wall and intramural gas collection.
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Medical therapy for esophagitis was initiated using omeprazole (20 mg twice a day) with an antifungal drug for esophageal candidiasis. In addition, a 12 x 13.5 x 15 mm Controlled Radial Expansion balloon dilator (Boston Scientific Japan, Tokyo) was placed in the stricture and was gradually dilated at the recommended pressure. The procedure was well tolerated by the patient without complications, and he subsequently required esophageal dilation therapy 6 times in total during a 4-month period. A follow-up endoscopy after 4 months of treatment revealed no severe stricture; however, on esophagogram the pseudodiverticula persisted and were similar in size and number. At a 1-year follow up, the patient was asymptomatic and medication with the proton pump inhibitor was maintained.
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Comment
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Esophageal intramural pseudodiverticulosis was first reported by Mendl and coworkers [1] in 1960; since then only 220 cases have been reported worldwide [2]. The condition is a little more common in males than females (ratio, 3 males to 2 females), and the mean age of the patients at the time of diagnosis is 53.5 years (range, 0.75 to 86 years) [3]. Esophageal intramural pseudodiverticulosis commonly presents with dysphagia, such that 47% of the patients have constant symptoms, 24% intermittent, and 9% progressive symptoms [4]. Dysphagia can occur independent of stricture formation in esophageal intramural pseudodiverticulosis [5]. Esophageal intramural pseudodiverticulosis is often associated with gastroesophageal reflux and other concomitant esophageal diseases including esophagitis, Crohn's disease, tuberculosis, Mallory-Weiss syndrome, and achalasia [4, 5]. A higher occurrence is seen in patients with diabetes mellitus and chronic alcoholism [4]. In this case, esophageal intramural pseudodiverticulosis may be related to a combination of gastroesophageal reflux and Candida esophagitis.
Esophageal intramural pseudodiverticulosis is characterized histologically by distinct excretory ducts with dilatation of the submucosal esophageal mucus glands; however, the cause and pathogenesis of esophageal intramural pseudodiverticulosis remains unclear. Previous reports showed that ductal dilatation in esophageal intramural pseudodiverticulosis was caused by an obstruction of the ducts due to inflammatory cells, desquamated epithelium, or submucosal fibrosis, or a combination thereof [4, 6]. Other reports demonstrated that the cause of esophageal intramural pseudodiverticulosis may be due to abnormal motor activity of the esophagus, more specifically, the incidence of esophageal intramural pseudodiverticulosis associated with achalasia or the esophageal web has been increasingly cited [6].
Radiographic evaluation is vital in the diagnosis of esophageal intramural pseudodiverticulosis. Barium esophagogram will show numerous tiny flask or collar button-shaped outpouchings consistent with pseudodiverticula, whereas computed tomography in esophageal intramural pseudodiverticulosis is marked by thickening of the esophageal wall, diffuse irregularity of the esophageal lumen, and intramural gas collection [7]. Endoscopy allows direct visualization of the diverticular orifices; however, it has been reported that this is seen in only 20% of patients [5]. The results in our case are well correlated with these findings, and led us to distinctly confirm the diagnosis of esophageal intramural pseudodiverticulosis. Esophageal intramural pseudodiverticulosis has a benign course with a good response to empiric therapy, including anti-inflammatory medication and endoscopic dilation therapy. However, some reports have shown severe complications with esophageal intramural pseudodiverticulosis, including mediastinitis from a fistula or esophageal perforation [2]. In addition, iatrogenic injury from endoscopic manipulation can lead to small localized perforations that affect the anterior mediastinum [8]. Although endoscopic dilation therapy may be necessary in patients with esophageal stricture, the risk of causing lacerations from forced dilation is high and should only be performed by highly experienced endoscopists. On the basis of the previously mentioned findings and due to significant symptoms, our patient underwent dilation carefully using a balloon dilator. Dysphagia improved dramatically without any complications; however, the tiny diverticula did not resolve after treatment.
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Acknowledgments
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We thank Dr Jonathan Daniel for the scientific editing of this article.
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References
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- Mendl K, McKay JM, Tanner CH. Intramural diverticulosis of the esophagus and Rokitanski-Aschoff sinuses in the gallbladder Br J Radiol 1960;33:496-501.
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- Evans PR. Oesophageal intramural pseudodiverticulosis-always benign? Aust NZ J Med 1991;21:58-61.
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Abstract
Introduction
