Path. Res. Pract. 187,514-519 (1991 )

Teaching Case

Adenosquamous Carcinoma of the Esophagus A Case Report J. A. Bombl, A. Riverola, J. M. Bordas 1 and A. Cardesa Department of Pathology and 1 Endoscopy Section, Hospital Clinic, Faculty of Medicine, University of Barcelona, Spain

SUMMARY

A case of adenosquamous carcinoma of the esophagus is presented. This carcinoma is a typical neoplasm of the upper aerodigestive tract almost exclusively originating in squamous epithelium in continuity with minor salivary glands. It is a very rare tumor in the esophagus, and is often diagnosed as mucoepidermoid carcinoma. The differential diagnosis between them is important due to the better prognosis of the last entity. The histology and electron microscopy is described and the literature is reviewed.

Introduction The most frequent malignant neoplasm of the esophagus is the squamous carcinoma followed in frequency by adenocarcinoma 18. Other types of carcinoma of the esophagus are very uncommon and constitute less than 2% of the tumors from this 10cation24. The adenosquamous carcinoma is a rare neoplasm of the upper aerodigestive tract mainly developing at the nasal, oral and laryngeal cavities 8 • The differential diagnosis of adenosquamous carcinoma from mucoepidermoid carcinoma represents a not yet entirely resolved matter of controversy2,9, however, their distinction seems to be important due to the worse prognosis adscribed to the first of these two entities 17• Both types of carcinomas are extremely rare at the esophagus and less than thirty cases have been published altogether 1,3,5,6, 10-16, 19,20,22,23,25-29 (Table 1). We are reporting here one additional case of what we consider to represent an example of adenosquamous carcinoma of the esophagus, and at the same time we are reviewing the literature on this subject.

associated one month later with a chronic cough during ingestion, a change in the tone of his voice, and a loss of weight of 30 kg. During hospitalization, he showed cachexia and dehydration. The radiology of the esophagus demonstrated a complete stenosis. The endoscopy revealed this stenosis with alimentary retention. The esophagic epithelium showed inflammatory signs. At the distal third of the esophagus (37 em from the teeth) there was a complete stenosis forming a blind, irregular sac covered by a mamillated and red tissue which bled easily on contact with the endoscope. This image was highly suspicious of a neoformative process, suggesting a propagated tumor from the stomach. A biopsy for histopathological study was performed. The TAC and ecography showed a transparietal infiltration involving the diaphragm with extension in the regional lymph nodes. The analytical studies revealed neither cholestasis nor alteration of the bone fraction of alkaline phosphatase. Given the extent of the neoplasia, a palliative treatment by laser endoscopic photoreduction was done. This treatment allowed the oral feeding and the patient was discharged from the hospital days thereafter. He died 5 weeks later. No autopsy permit was granted.

Case Report A 59-year-old male without a relevant previous clinical history, with the exception of heavy smoking habits. Three months before his hospitalization he developed a progressive dysphagia to solid foods, astenia and anorexia, 0.144·0338191/0187-0514$3.50/0

Pathology Report The biopsy material of the tumoral mass consisted of 3 fragments, each 2 X 2 X 2 mm in size, fixed in neutral © 1991 by Gustav Fischer Verlag, Srungan

Adenosquamous Carcinoma of th e Esophagus · SIS Table 1. Adenosquamous and mucoepidermoid carcinomas arising from the esophagus

Author McPeak and Arens J6 Stout and Lattcs 23 DodgeS Azzopardi and Menzies J

1947 1957 1961 1962

Kasai and Shimamura 10 Lonat-Jacoh et aL" Kay et aL'I

1967 1968 1968

Weitzner et af.21l Turnbull et al." Toriie et aL26 Osamura et al. 19 Woodward et al." Bell-Thomson et al. 3

1970 1973 1976 1978 1978 1980

Kormano and Yrjiina l4 Emoto et a1. 8 Smith er al. 22 Marsufuji et al. ls Kuwano et al. B Pascal and Clearfield 20 Takubo et .1. 25 Present Case

1981 1982 1984 1985 1985 1987 1987 1989

Age

Sex

Location

Treatment

Surviva l

46 64 68 54 47 58 61 47 66 67 63 67 53 49

M M

Lower third Lower Lower Lower Middle Upper Lower Lower Lower Lower Upper Middle Lower Lower Middle Middle Middle Upper Middle Lower Middle

Not resected

2 rno

Resection Resection Resection Resection Resection Resection Resection Resection Resection Resection Radiation only Resection Rad. chernoth. Resection Resection Resection Resection Resection Resection

Alive/well 15 rno postop. Alive/well 14 years postop. 12 mo

Lower Middle Lower

Resection Radiat. Resection Laser photoreduct.

71

M

F

M

M M M M M

F F F M M M M F M M F

64 60 81 73 50 66 7 cases 63 M 55 M M 59

formalin. The matcrial was included in paraffin and stained with HE, PAS, Aleian Blue and mucicarmin. Another fragment of tissue was fixed in glutaraldehyde and osmium tetroxide for ultrastructural study. It was included in Araldit (Durcupan ACM), and examined under a Zeiss-M-109-Turbo electron microscope. The optical microscopic study showed an infiltrating carcinoma constituted by a predominantly squamous pattern alternating with glandular lumens. The predominating squamous cell carcinoma areas showed several foci with squamous cell carcinoma on the surface, which appeared .in continuity with cords and nests of poorly differentiated squamous epithelium with little keratinization that invaded the surrounding stroma. In addition there was the accompanying prominent feature of gland formation and mucous production, showing positive reac-

tion with PAS, Aleian blue and mllcicarmin. Both patterns, the squamous and the glandular one appeared well intermingled with each other, nevertheless there was a tendency for predominance of the squamous cell pattern on the surface and of the glandular pattern in deeper regions. The overall cell differentiation deserved a grade of malignancy of 1II on a scale of IV. Considering all the findings the diagnosis was of an adcnosquamous carcinoma of the esophagus (Figs. 1,2).

Fig. 1. Microscopic picture showing squamous carcinoma in the surface and nests of poorly differentiated squamous carcinoma associated with adcnocarcinomarous components (HE, X 100).

.I , , .

'\; ..:t)-""I il' ...

""

'..

1 IIlO

40 rna 6 mo 17 rn a Unknown 1 rno 20 rno 7 rno I rno 5010

Alive/well 24 rno postop. Ali vdwdl 8 mo pOStop. 9 rno Ali vdwdl 17 rno postop. 1 rno

4 mo

2 rno

516 .

J.

A. Bombi



518 .

J. A. Bombi ct al.

to the continuity of the excretory duct cells with the surface epithelium the adenosquamous carcinoma has the property of invading the covering mucosal surface giving rhe pattern similar to either in situ squamous cell carcinoma or to Paget disease. These findings are never seen in mucoepidermoid carcinoma in which intramucosal growth is nor a characteristic feature. The adenosquamous carcinoma of the esophagus develops more frequently in men than in women (311) and is usually found in the lower third, less often in the middle and very rarely in the upper third. Perhaps this distribution is related to the carcinogenetic or promotional effect due to the gastroesophagic reflux, illustrated in some recent cases of Barrett's esophagus20 This association between Barrett's esophagus and carcinoma with squamous and glandular areas is infrequent and has only been reported occasionally29. For the diagnosis of this neoplasia the collision tumor as well as the adenoacanthoma with areas of benign squamous mctaplasia in an adenocarcinoma must be excludcd from this group. This type of rumor must also be differentiated from the intraductal infiltration of minor salivary glands of the esophagus by squamous carcinoma, rccently reported by Takubo et aJ. '\ who found thcm in 19% of the cases of esophageal carcinoma. These authors occasionally described arcas of ring-celled mucous carcinoma. In our own experimental studies we have also found rhis type of changes in rats treated by esophagojejunostomy and subcuraneous injection of 2.6-Dimerhylnitrosomorpholine' l, promoting tumoral induction wirh reflux esophagitis. Biologically, adenosquamous carcinoma behaves much worse than high grade mucoepidermoid carcinoma, showing a high propensity to regional lymph node metastases and a very short average survival I ? In general, the treatment for these tumors is a surgical resection. One case documents a survival for 14 years, but this is most likely due to the small size of the tumorS because after diagnosis patients rarely survive longer than 12 months, occasionally they live more than 2 years.

Acknowledgements The authors are grateful ro Miss Angustias Gonzalez for her secretarial work and Miss Elena Rull for the technical assistance.

TI,is work is supported in part by a grant from CIRlT-89.

adenocarcinoma. Confirmation of its existence by the finding of

mucous gland tumours. Br J Surg 49: 497-506 2 Barnes L, Peel RL (1990) Head and Neck Pathology: A Text

Atlas of Differential Diagnosis. Igaku·Shoin, New York, , Bell Thom:ion

cinoma of rhe esophagus squamous cell carcinoma and mucoepidermoid carcinoma. Jpn J Cancer Clin 28: 1754-1757 7 Evans HL (1984) We have Illet the enemy, but it's another

neoplasm (lerrer). Am J Clin Pathol 82: 512-513 8 Gerughey RM, Hennigar GR, Brown FM (1968) Adenosqua-

mous carcinoma of the nasal, oral and laryngeal cavities. Cancer

22: 1140-1154 , Hyans VJ, Batsakis JG, Michaels L (1988) Tumors of the

upper respiratory tract and ear. Atlas of Tumor Pathology.

Second Series. Fascicle 25. AFlI'. Washington, 104-107 10

Kasai M, Shimamura N (1967) Primary adenocarcinoma of

the middle thoracic esophagus. Jpn J Cancer Clin 13: 700- 704 II Kay S (1968) Mucoepidermoid carcinoma of the esophagus. Report of two cases. Cancer 22: 1053-1059 12 Kormano MJ, Yrjana J (1981 ) Radiology of uncommon esophageal neoplasms. Europ J Radiol 1: 51~56

13 Kuwano H, Ueo H, Sugimachi K, Inokuchi K, Toyoshima S, Enjoi M (1985 ) Glandular or mucus-secreting components in squamous cell carcinoma of the esophagus. Cancer 56:

514- 518 I. Lortat-Jacob JL, Maillard IN, Richard CA, Fekete F, Huguier M, Conte-MartiJ (1968) Primaey esophageal adenocarcinoma: Report of 16 cases. Surgery 64: 535-543 15 Matsufuji H, Kuwano H, Ueo H, Sugimachi K, lnokuchi K (1985) Mucoepidermoid carcinomas of the esophagus. A case report. Jpn J Surg 15: 55-59 16 McPeak E, Arens WL (1947) Adenoacal1thoma of esopha-

gus. Report of one case with consideration of tumor's resem-

blance to so-called salivary gland tumor. Arch Pathol Lab Med 44: 385-390 17 Mills SR. Discussion of case 7 (1989) Spring Anatomic Pathology Slide Seminar. An Soc of Clinical Pathologists 18 Ming S (1973) Tumors of the esophagus and stomach.

Fascicle 7, Second Series. Atlas of Tumor Pathology. Washington.

AFII'

]9 Osamura PY, Sato 5, Masahiko M, Miwa T (1978) Mucoepidermoid carcinoma of the esophagus. Am JGastroenter01 69: 467-470

20

Pascal R, Clearfield H (1987) Mucoepidermoid (adeno-

squamous) carcinoma arising in Barretr's esophagus. Dig Dis Sci

32:428-432 11

Pera M, Cardesa A, Bambi JA, Ernst H, Pera C, Mohr U

(1989) Influence of esophagojejunostomy on the induerion of

adenocarcinoma of the distal esophagus in Sprague-Dawley Rats by subcutaneous injection of 2.6-Dimethylnitrosomorpholine. The spectrum of carcinoma arising in Barrett's esophagus. Am

Azzopardi JG, Menzies T (1962) Primary oesophageal

26-27

In: Pathol. Annual Part I 1987. Rosen PP and Fecher RE (Eds) 1- 53 5 Dodge OG (1961 ) Gamo-esophageal carcinoma of mixed histological eype. J Pathol Baer 81: 459-471 6 Emoto 1, Chihara T, Tarnai M, Yoshida H, Kobayashi M, Okana M, Gotoh, Okuda S, Tani S. A patient with double car-

Cancer Res 49: 6803-6808 22 Smith RRL, Hamilton SR, Boitnott JK, Rogers EL (1984)

References I

• Dardick I, Van Nostrand AWP (1987) Morphogenesis of

salivary gland tumors. A prerequisite to improving classifications.

J.

Haggict Re, Ellis FH (1980) Mu coepider-

moid and adenoid cystic carcinomas of the esophagus. JThorae

Cardiovas Surg 79: 438-446

Surg Pathol 8: 563-573 >J Stout AP, Lattes R (1957) Tumors of the esophagus. Fascicle 20. Atlas of Tumor Pathology. Washington. AFIP 24 Suzuki H, Nagayo T (1980) Primary tumors of the esophagus other rhan squamous cell carcinoma: histologic classification and statistics of the surgical and autopsied materials in Japan. Inst

Adv Surg Oncol 3: 73-109 25

Takubo K, Takai A, Takayailla S, Sasajirna K, Yamashita

K,

FujitaK (1987) Intraductal spread of esophageal squamous cell carcinoma. Cancer 59: 1751-1757

Lefter to the Case 26 Toriic S, Takeda S, Kohli Y, Tanaka T, Kodama M, Kawai K. (1976) A case of mucoepidermoid carcinoma of esophagus. Jpn J Cancer Clin 22: 606- 610 27 Turnbull AD, Rosen P, Goodner JT, Beattie E (1973) Primary malignant tumors of (he esophagus other than typical epidermoid carcinoma. Ann Thorac Surg 15: 463-473

519

28 Weitzner S (1970) Mucoepidermoid carcinoma of esophagus. Report of a case. Arch Pathol Lab Med 90: 271-273 29 Woodward BH, Shelburne JD, Vollmer RT, Posrlethwalt RW (1978) Mucoepidermoid carcinoma of the esophagus: A case report. Hum Parhol 9: 352-354

Received June 22, 1990· Accepted Augusr 29,1990

Key words: Adenosquamous carcinoma - Esophagus Prof. Dr. A. Cardesa, Dept. of Pathology, Hospital Clinic, Faculty of Medicine, University Barcelona, Villarroel, 170, E-08036-Barcelona, Spain

Letters to the Case

c.

Brocheriou

Paris, France Bambi et al. 2 report one case of adenosquamous carcinoma of the esophagus and describe the main histopathologic features of this tumoral entity in the upper aerodigestive tract, one of its localizationsl . Firstly it is important to distinguish the adenosquamous carcinoma from other morphologically close tumoral entities: adenoacanthoma and mucoepidermoid carcinoma. Adenoacanthoma is a glandular carcinoma with foci of epidermoid metaplasia. The squamous elements are benign and absent in the metastases. On the contrary the metastases from adenosquamous carcinoma usually show a double epidermoid and glandular proliferation, the latter being predominant. Distinction between adenosquamous and mucoepidermoid carcinoma is more difficult, often unknown by the surgical pathologist: in both cases there are epidermoid and glandular components. However, in the mucoepidermoid carcinomas dlese two components are inrermixed and mucosecretion is constant. On the other hand in the adenosquamous carcinomas the areas of purely epidermoid component with intercellular bridges and keratin are clearly separated from glandular areas, the latter being usually deeply located. In contrast to the opinion of Gerughty4 mucosecretion is no more considered as a necessary condition to make the diagnosis of adenosquamous carcinoma l . An important condition emphasized by Bambi' is the quasiconstanr presence, in the adenosquamous carcinoma, of in situ or superficial epidermoid carcinomatous changes. In mucoepidermoid carcinomas this condition has never been met in the oral, nasal and laryngeal cavities. Electronic microscopy affords no mor-

phological arguments for distinguishing mucoepidermoid carcinoma from adenosquamous carcinoma, because in both cases it shows glandular and squamous elements. The latter are not clearly seen on Fig. 4 of Bombi's report. Another point of discussion is the frequency of adenosquamous carcinoma in rhe upper aerodigestive tract, especially in the esophagus. The lack of understanding of this lesion could explain the paucity of reported cases of adenosquamous carcinoma. For example the coexistence of epidermoid carcinoma and adenoid cystic carcinoma is easy to recognize because of the typical morphological aspect of the adenoid cystic carcinoma; but this is not the case in the presence of a common glandular malignant component. Some high-grade mucoepidermoid carcinomas described in the esophagus or elsewhere in the body are in fact real adenosquamous carcinomas. Besides, as the co-existence of glandular and epidermoid proliferation is far more frequent in the lower third than in the upper third of the esophagus, it is important to eliminate a collision rumor, association of a gastric adenocarcinoma and an esophageal epidermoid carcinoma. Finally the malignant nature of the glandular component should be ensured: an epidermoid carcinoma can spread into the underlying glands and constitute ambiguous histologic features. Nevertheless frequency of the adenosquamous carcinoma is certainly low. In fact no case of adenosquamous carcinoma has been mentioned by Bogomoletz 1 in a recent review of 76 cases of superficial carcinoma of the esophagus though this review described 5 cases of superficial

Adenosquamous carcinoma of the esophagus. A case report.

A case of adenosquamous carcinoma of the esophagus is presented. This carcinoma is a typical neoplasm of the upper aerodigestive tract almost exclusiv...
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