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Sialadenoma papilliferum of the esophagus
Vol. 93, No. 3, 1998 ISSN 0002-9270/98/$19.00 PI1 sOOO2-927q97)001234
THE AMERICAN JOURNAL OF G~s~~~wwxxoc~ Copyright 0 1998 by Am. Cdl. of Gastmenterology published by Elsevier Science Inc.
Sialadenoma Jang-Ming Department
Papilliferum
of the Esophagus
Su, Hon-Ki Hsu, Ping-I Hsu, Chung-Yang
of Surgery and Internal Medicine,
Wang, and Huang-Chou
Veterans General Hospital-Kaohsiung,
Chang
Kaohsiung,
Taiwan
with somewhitish exudative patches(Fig. 1). There was no evidence of Barrett’s metaplasticepithe:.ium.The pathological examination of the biopsy specimen from the tumor showed a well differentiated adenocarcinoma. Computed tomography of the chest and abdomen was nondiagnostic. Under the impressionof esophagealadenocarcinoma,transhiatal esophagectomyand left colon interposition were performed. The postoperative course was uneventful and the patient remainedwell without evidence of recurrence during 12 months of follow-up. Grossly, a broad-based,well circumscribed polypoid tumor with finely granular surface, measuring 10 X 5 X 5 mm, projected into the lumen in the distal esophagus.Microscopically, the tumor disclosedboth exophytic papillary and submucosalcomponents. The papillary fronds extending above the level of adjacent esophagealmucosa were supported by the inflamed fibrovascular cores and were covered by stratified squamousepithelium (Fig. 2). The glandular epithelium delineatedthe cytologically bland double layers of cells, including high columnar cells with eosinophilic cytoplasm and basal cells with clear cytoplasm. The scattered mucus-secretingcells wer’: stainedpositively by alcian blue, pH = 2.5, and diastasa-digestedperiodic acid Schiff. These features were simikr to sialadenoma papilliferum, a rare neoplasmof the minor salivary glands. The pathological analysis suggestedthat the adenomaoriginated in the submucosalgland ducts.
Sialadenoma papilliferum is an extremely rare benign tumor of the esophagus. We report a 70-yr-old woman who was first thought to have adenocarcinoma in the distal esophagus. Transhiatal esophagectomy and left colon interposition were performed. The pathological diagnosis of sialadenoma papilliferum of the esophagus arising in the submucosal gland ducts was confirmed after surgery. (Am J Gastroenterol1998;93:461-462.0 1998 by Am. Coll. of Gastroenterology)
INTRODUCTION Esophagealadenomaswithout Barrett’s metaplastic epithelium are very rare. Fewer than 20 caseshave been reported in the world literature (l-l 1). Although not all of them can be identified in their origins, seven adenomasare histologically confirmed to arise in the submucosalgland of the esophagus(1, 6-l 1). Rouseet al. describedthree, possibly four, submucosaladenomasthat bear a resemblanceto sialadenomapapilliferum, a rare neoplasm of the minor salivary glands (11). We recently encountered a case of sialadenomapapilliferum of the esophagusarising in the submucosalgland ducts. CASE REPORT A 70-yr-old woman was admitted to our hospital in June 1994 with abdominal distention and tarry stools in the previous 2 days. Endoscopy revealed an active bleeder on the high body of the stomach and it also incidentally discloseda 4-mm-diameterpolypoid tumor located 35 cm from the incisor teeth. No biopsy was made because of vital instability. She underwent gastrotomy and suture-ligation for the active bleeder on the gastric high body becauseof uncontrolled upper gastrointestinal bleeding. The postoperative course was complicated with intraabdominal abscess. She was discharged after l-month of hospitalization. However, she was lost to follow-up after discharge. In late February 1996, she was referred to our hospital complaining of abdominal fullness. Endoscopy revealed a growing, broad-basedlo-mm-diameter polypoid tumor located at the samesite as shown in the previous endoscopy. The finely granular surface of the tumor was partly covered Received Apr. 14, 1997; accepted Nov. 10, 1997. 461
DISCUSSION Benign tumors of the esophagusare rare and leiomyomas are the mostcommon (5,12). The n0rma.lesophagealglands include cardiac-type glands in the lamina propria and submucosal glands. Histologically, the cardiac-type glands resemblethe cardiac glands of the stomachand are composed of cells secretingneutral mucins. The submucosalglandsare consideredto be a continuation of the minor salivary glands of the oropharynx and produce acid mucins (13). Esophageal adenomasmay be mucosal or submucosalin origin. There have been fewer than 20 adenomaswithout Barrett’s metaplastic epithelium reported in the a,orld literature. Although most reports could not provide a clearly microscopic description to identify their origins, seven adenomasare confirmed histologically to arise in the submucosalgland of the esophagus(1, 6-l 1). Moreover, Rouse et al. described three, possibly four submucosaladenomasof the esophagus
462
su er al.
AJG - Vol. 93, No. 3, 1998 adenocarcinoma is characterized by single-layered irregular neoplastic glands and pleomorphic adenocarcinomatous cells. Careful attention to the architectural and cytological features of the constituent glands and an awareness of this entity should prevent misdiagnosis and resultant overtreatment (11). Two previously reported cases of sialadenoma papilliferum of the esophagus had been p::esent for at least 2 and 8 yr, respectively, before endoscopic removal with no recurrence in 12 months and 2 yr, respectively, of individual follow-up (9, 11). Our patient’s ade:loma had been present for at least 17 months before resection and did not recur in 12 months of follow-up. It is observed that the distinctive adenomas are slow growing, with no evidence of recurrence after complete resection. Esophagotomy or endoscopic snare cautery may be optional for tumor removal. ACKNOWLEDGMENTS
FIG. 1. A broad-based, lo-mm-diameter polypoid tumor is seen in the distal esophagus. Its finely granular surface is partly covered with some whitish exudative patches.
We acknowledge the help given by Han Chang, M.D., Department of Pathology, Show-Chwan Memorial Hospital, Changhua, Taiwan, in reviewing the manuscript. Reprint requests and correspondence: Jang-Ming of Surgery, Veterans General Hospital-KaohEiung, Kaohsiung, Taiwan.
Su, M.D., Department 386 Ta-Chung 1st Rd,
REFERENCES
FIG. 2. In high magnification, some exophytic papillary fronds are supported by the inflamed flbrovascular cores and covered by stratified squamous epithelium. Scattered mucin-secreting cells are seen (hematoxylin and eosin, X 100).
and these adenomas had similar histological features to sialadenoma papilliferum, a rare neoplasm of the minor salivary glands. They suggested that these adenomas derive from the submucosal gland ducts (11). In our case, the pathological features also showed resemblances to sialadenoma papilliferum (14) and conformed to the recent second edition of the WHO’s histological typing of salivary gland tumors ( 15). When an esophageal submucosal gland duct adenoma is encountered, the exclusion of adenocarcinoma is most important in an endoscopic biopsy specimen (11). The presence of the exophytic papillomatous epithelium and ductlike glands lined by cytologically bland double layers of cells is indicative of sialadenoma papilliferum. In contrast,
1. Weigert C. Ein Fall von Adenoma Polypcsum Oesophagi. Arch Path01 Anat 1876;67:516-7. 2. Hicquet Cl, Jourdain V. Un cas d’adenoma de oesophage. Vir Arch Path01 Anat 1933;86:231-4. 3. Moersch HJ, Harrington SW. Benign tuners of tbe esophagus. Ann Otol Rhino1 Laryngol 1944;53:800-17. 4. Schmidt HW, Clagett OT, Harrison EG, J-. Benign tumors and cysts of the esophagus. J Thorac Cardiovasc Surf: 1961;41:717-32. 5. Plachta A. Benign tumors of the esophagus: Review of literature and report of 99 cases. Am J Gastroenterol 1962;38:639-52. 6. Spin FP. Adenomas of the esophagus: A case report and review of the literature. Gastrointest Endosc 1973;20:26-7. 7. Lesbros F, Berger G, Mallet-Guy Y, et al. Adenome des glandes oesophagiennes ressemblant a un adenome mucipare bronchique (a propos d’un cas avec etude histologique et ultrastructurale). Arch Anat Cytol Path01 1981;29:55-8. 8. Banducci D, Rees R, Bluett MK, et al. Pleomorphic adenoma of the cervical esophagus: A rare tumor. Ann ‘Ihorac Surg 1987;44:653-5. 9. Tsutusmi M, Mizumoto K, Tsuiiuchi T, et al. Serous cvstadenoma of the esophagus. Acta Path01 Jpu 1990;40:153-5. ’ 10. Takubo K, Esaki Y, Watanabe A, et al. Adenoma accompanied by superficial squamous cell carcinoma of the esophagus. Cancer 1993; 71:2435-8. 11. Rouse RV, Soetikno RM, Baker RJ, et al. Esophageal submucosal gland duct adenoma. Am J Surg Path01 1995;19:1191-6. 12. Postlethwait RW. Benign tumors and cyst j of the esophagus. Surg Clin North Am 1983;63:925-3 1. 13. DeNardi FD, Riddell RH. Esophagus. In: Stemberg SS, ed. Histology for pathologists. New York: Raven Press, 1992:521-6. 14. Ellis GL, Auclair PL. Tumors of the salivary glands. In: Rosai J, Sobin LH, eds. Atlas of tumor pathology. WasTington, D.C.: Armed Force Institute of Pathology, 1995:126-30. 15. Seifert G, Sobin LH. Histological typing of salivary gland tumours. World Health Organization international histological classification of tumours, 2nd ed. New York: Springer-Verlag, 1991.
THE AMERICAN JOURNAL OF G~s~~~wwxxoc~ Copyright 0 1998 by Am. Cdl. of Gastmenterology published by Elsevier Science Inc.
Sialadenoma Jang-Ming Department
Papilliferum
of the Esophagus
Su, Hon-Ki Hsu, Ping-I Hsu, Chung-Yang
of Surgery and Internal Medicine,
Wang, and Huang-Chou
Veterans General Hospital-Kaohsiung,
Chang
Kaohsiung,
Taiwan
with somewhitish exudative patches(Fig. 1). There was no evidence of Barrett’s metaplasticepithe:.ium.The pathological examination of the biopsy specimen from the tumor showed a well differentiated adenocarcinoma. Computed tomography of the chest and abdomen was nondiagnostic. Under the impressionof esophagealadenocarcinoma,transhiatal esophagectomyand left colon interposition were performed. The postoperative course was uneventful and the patient remainedwell without evidence of recurrence during 12 months of follow-up. Grossly, a broad-based,well circumscribed polypoid tumor with finely granular surface, measuring 10 X 5 X 5 mm, projected into the lumen in the distal esophagus.Microscopically, the tumor disclosedboth exophytic papillary and submucosalcomponents. The papillary fronds extending above the level of adjacent esophagealmucosa were supported by the inflamed fibrovascular cores and were covered by stratified squamousepithelium (Fig. 2). The glandular epithelium delineatedthe cytologically bland double layers of cells, including high columnar cells with eosinophilic cytoplasm and basal cells with clear cytoplasm. The scattered mucus-secretingcells wer’: stainedpositively by alcian blue, pH = 2.5, and diastasa-digestedperiodic acid Schiff. These features were simikr to sialadenoma papilliferum, a rare neoplasmof the minor salivary glands. The pathological analysis suggestedthat the adenomaoriginated in the submucosalgland ducts.
Sialadenoma papilliferum is an extremely rare benign tumor of the esophagus. We report a 70-yr-old woman who was first thought to have adenocarcinoma in the distal esophagus. Transhiatal esophagectomy and left colon interposition were performed. The pathological diagnosis of sialadenoma papilliferum of the esophagus arising in the submucosal gland ducts was confirmed after surgery. (Am J Gastroenterol1998;93:461-462.0 1998 by Am. Coll. of Gastroenterology)
INTRODUCTION Esophagealadenomaswithout Barrett’s metaplastic epithelium are very rare. Fewer than 20 caseshave been reported in the world literature (l-l 1). Although not all of them can be identified in their origins, seven adenomasare histologically confirmed to arise in the submucosalgland of the esophagus(1, 6-l 1). Rouseet al. describedthree, possibly four, submucosaladenomasthat bear a resemblanceto sialadenomapapilliferum, a rare neoplasm of the minor salivary glands (11). We recently encountered a case of sialadenomapapilliferum of the esophagusarising in the submucosalgland ducts. CASE REPORT A 70-yr-old woman was admitted to our hospital in June 1994 with abdominal distention and tarry stools in the previous 2 days. Endoscopy revealed an active bleeder on the high body of the stomach and it also incidentally discloseda 4-mm-diameterpolypoid tumor located 35 cm from the incisor teeth. No biopsy was made because of vital instability. She underwent gastrotomy and suture-ligation for the active bleeder on the gastric high body becauseof uncontrolled upper gastrointestinal bleeding. The postoperative course was complicated with intraabdominal abscess. She was discharged after l-month of hospitalization. However, she was lost to follow-up after discharge. In late February 1996, she was referred to our hospital complaining of abdominal fullness. Endoscopy revealed a growing, broad-basedlo-mm-diameter polypoid tumor located at the samesite as shown in the previous endoscopy. The finely granular surface of the tumor was partly covered Received Apr. 14, 1997; accepted Nov. 10, 1997. 461
DISCUSSION Benign tumors of the esophagusare rare and leiomyomas are the mostcommon (5,12). The n0rma.lesophagealglands include cardiac-type glands in the lamina propria and submucosal glands. Histologically, the cardiac-type glands resemblethe cardiac glands of the stomachand are composed of cells secretingneutral mucins. The submucosalglandsare consideredto be a continuation of the minor salivary glands of the oropharynx and produce acid mucins (13). Esophageal adenomasmay be mucosal or submucosalin origin. There have been fewer than 20 adenomaswithout Barrett’s metaplastic epithelium reported in the a,orld literature. Although most reports could not provide a clearly microscopic description to identify their origins, seven adenomasare confirmed histologically to arise in the submucosalgland of the esophagus(1, 6-l 1). Moreover, Rouse et al. described three, possibly four submucosaladenomasof the esophagus
462
su er al.
AJG - Vol. 93, No. 3, 1998 adenocarcinoma is characterized by single-layered irregular neoplastic glands and pleomorphic adenocarcinomatous cells. Careful attention to the architectural and cytological features of the constituent glands and an awareness of this entity should prevent misdiagnosis and resultant overtreatment (11). Two previously reported cases of sialadenoma papilliferum of the esophagus had been p::esent for at least 2 and 8 yr, respectively, before endoscopic removal with no recurrence in 12 months and 2 yr, respectively, of individual follow-up (9, 11). Our patient’s ade:loma had been present for at least 17 months before resection and did not recur in 12 months of follow-up. It is observed that the distinctive adenomas are slow growing, with no evidence of recurrence after complete resection. Esophagotomy or endoscopic snare cautery may be optional for tumor removal. ACKNOWLEDGMENTS
FIG. 1. A broad-based, lo-mm-diameter polypoid tumor is seen in the distal esophagus. Its finely granular surface is partly covered with some whitish exudative patches.
We acknowledge the help given by Han Chang, M.D., Department of Pathology, Show-Chwan Memorial Hospital, Changhua, Taiwan, in reviewing the manuscript. Reprint requests and correspondence: Jang-Ming of Surgery, Veterans General Hospital-KaohEiung, Kaohsiung, Taiwan.
Su, M.D., Department 386 Ta-Chung 1st Rd,
REFERENCES
FIG. 2. In high magnification, some exophytic papillary fronds are supported by the inflamed flbrovascular cores and covered by stratified squamous epithelium. Scattered mucin-secreting cells are seen (hematoxylin and eosin, X 100).
and these adenomas had similar histological features to sialadenoma papilliferum, a rare neoplasm of the minor salivary glands. They suggested that these adenomas derive from the submucosal gland ducts (11). In our case, the pathological features also showed resemblances to sialadenoma papilliferum (14) and conformed to the recent second edition of the WHO’s histological typing of salivary gland tumors ( 15). When an esophageal submucosal gland duct adenoma is encountered, the exclusion of adenocarcinoma is most important in an endoscopic biopsy specimen (11). The presence of the exophytic papillomatous epithelium and ductlike glands lined by cytologically bland double layers of cells is indicative of sialadenoma papilliferum. In contrast,
1. Weigert C. Ein Fall von Adenoma Polypcsum Oesophagi. Arch Path01 Anat 1876;67:516-7. 2. Hicquet Cl, Jourdain V. Un cas d’adenoma de oesophage. Vir Arch Path01 Anat 1933;86:231-4. 3. Moersch HJ, Harrington SW. Benign tuners of tbe esophagus. Ann Otol Rhino1 Laryngol 1944;53:800-17. 4. Schmidt HW, Clagett OT, Harrison EG, J-. Benign tumors and cysts of the esophagus. J Thorac Cardiovasc Surf: 1961;41:717-32. 5. Plachta A. Benign tumors of the esophagus: Review of literature and report of 99 cases. Am J Gastroenterol 1962;38:639-52. 6. Spin FP. Adenomas of the esophagus: A case report and review of the literature. Gastrointest Endosc 1973;20:26-7. 7. Lesbros F, Berger G, Mallet-Guy Y, et al. Adenome des glandes oesophagiennes ressemblant a un adenome mucipare bronchique (a propos d’un cas avec etude histologique et ultrastructurale). Arch Anat Cytol Path01 1981;29:55-8. 8. Banducci D, Rees R, Bluett MK, et al. Pleomorphic adenoma of the cervical esophagus: A rare tumor. Ann ‘Ihorac Surg 1987;44:653-5. 9. Tsutusmi M, Mizumoto K, Tsuiiuchi T, et al. Serous cvstadenoma of the esophagus. Acta Path01 Jpu 1990;40:153-5. ’ 10. Takubo K, Esaki Y, Watanabe A, et al. Adenoma accompanied by superficial squamous cell carcinoma of the esophagus. Cancer 1993; 71:2435-8. 11. Rouse RV, Soetikno RM, Baker RJ, et al. Esophageal submucosal gland duct adenoma. Am J Surg Path01 1995;19:1191-6. 12. Postlethwait RW. Benign tumors and cyst j of the esophagus. Surg Clin North Am 1983;63:925-3 1. 13. DeNardi FD, Riddell RH. Esophagus. In: Stemberg SS, ed. Histology for pathologists. New York: Raven Press, 1992:521-6. 14. Ellis GL, Auclair PL. Tumors of the salivary glands. In: Rosai J, Sobin LH, eds. Atlas of tumor pathology. WasTington, D.C.: Armed Force Institute of Pathology, 1995:126-30. 15. Seifert G, Sobin LH. Histological typing of salivary gland tumours. World Health Organization international histological classification of tumours, 2nd ed. New York: Springer-Verlag, 1991.