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| Year : 2018 | Volume
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| Issue : 1 | Page : 145-146 |
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| Diffuse esophageal submucosal retention cysts: An autopsy happenstance |
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Mona Agnihotri, Pradeep Vaideeswar
Department of Pathology (Cardiovascular and Thoracic Division), Seth GS Medical College, Mumbai, Maharashtra, India
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| Date of Web Publication | 22-Mar-2018 |
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How to cite this article:
Agnihotri M, Vaideeswar P. Diffuse esophageal submucosal retention cysts: An autopsy happenstance. Indian J Pathol Microbiol 2018;61:145-6 |
The entire esophagus at autopsy, in a 68-year-old hypertensive woman with a fatal spontaneous ascending aortic rupture, showed a smooth cobble-stoned appearance of its lining. Serial transverse sections revealed multiple cysts of varying sizes (0.3 cm–1.2 cm), filled with mucoid material [Figure 1]. On histology, the cysts were produced by the dilated ducts of the submucosal glands. The overlying squamous epithelium was intact [Figure 2]a. Interspersed between the cysts was normal submucosal glands [Figure 2]b. The cysts were predominantly lined by double-layered cuboidal epithelium with focal stratification [Figure 2]c and [Figure 2]d or attenuation. There was mild lymphocytic infiltrate present in the lamina propria, submucosa and around the dilated ducts. A diagnosis of diffuse esophageal submucosal retention cysts was made. | Figure 1: Serial cross sections of the esophagus at all levels showed multiple mucin-filled cysts in the submucosa resembling vesicles
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The submucosal tubulo-alveolar glands of esophagus are analogous to minor salivary glands and drain into cuboidal/squamous-lined ducts. Following complete or partial obstruction, these ducts can progressively enlarge to form cysts and are included in the differential diagnoses of the rare acquired esophageal cysts. In literature, however, there have been different terminologies to denote these lesions and these include retention cysts, mucocele, esophagitis cystica, or cyst of esophageal submucosal gland duct (ESGD).[1],[2] The term “mucocele” is used for extravasation of mucus from a ruptured duct into the connective tissue (even when it occurs in the esophagus [3]) and should be avoided though it may occur in the late stages of the disease. The term “esophagitis cystica” is analogous to colitis cystica profunda, gastritis cystica profunda, and cystitis cystica, but is less appropriate as the pathogenesis is different and the inflammation is minimal.[1] We feel retention cyst is the most appropriate nomenclature, rather than cyst of ESGD. The cysts, first described by Kuhne in 1889,[1] are hardly reported in recent literature. They can be solitary or multiple, particularly involving the lower end of the esophagus, ranging in size up to 1.5 cm and seen as cobble stoned or polypoidal lesions.[4] The cystic dilatation is related to partial or total obstruction of the ducts, related to motility disturbances, inflammation, keratinization around the duct orifice, or even basal cell hyperplasia.[1],[4] Many have asymptomatic clinical course and are discovered incidentally, though some patients can have dysphagia.[5] Apart from inflammation, occasional cases have shown stromal chondromatous metaplasia.[6] In the present case, there was a cobble stoning of the entire esophagus; however, no cause was found. The patient apparently did not have any cyst-related symptoms, since the attention was drawn to her catastrophic cardiovascular manifestations. Since they are often seen at autopsy, therapeutic guidelines are available for symptomatic localized lesions and not diffuse lesions. The rarity and use of diverse terminologies prompted the authors to present this case for awareness, interest, and establishing a unified nomenclature.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
 References | |  |
| 1. | Farman J, Rosen Y, Dallemand S, Iyer SK, Kim DS. Esophagitis cystica: Lower esophageal retention cysts. AJR Am J Roentgenol 1977;128:495-6.  |
| 2. | Nie L, Wu HY, Shen YH, Fan XS, Sun Q, Huang Q, et al. Esophageal submucosal gland duct adenoma: A clinicopathological and immunohistochemical study with a review of the literature. Dis Esophagus 2016;29:1048-53. |
| 3. | Goodman P, Kalangi SS, Kumar D, Balachandran S. Spontaneous mucocele of the upper esophagus: Radiologic demonstration. Dysphagia 1994;9:199-201. |
| 4. | Hover AR, Brady CE 3 rd, Williams JR, Stewart DL, Christian C. Multiple retention cysts of the lower esophagus. J Clin Gastroenterol 1982;4:209-12. |
| 5. | Edgin R, Mekhjian HS. Esophageal retention cyst: Unusual cause for dysphagia. J Clin Gastroenterol 1981;3 Suppl 1:57-9. |
| 6. | Tano S, Tanaka K, Iguchi T, Nishikawa K, Takayama R, Ninomiya K, et al. Large retention cyst with chondromatous metaplasia in the esophagus. Endoscopy 2011;43 Suppl 2:E262-3. |
Correspondence Address:
Pradeep Vaideeswar
Department of Pathology (Cardiovascular and Thoracic Division), Seth GS Medical College, Parel, Mumbai - 400 012, Maharashtra
India
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/IJPM.IJPM_346_17
[Figure 1], [Figure 2] |
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