Caso Clínico

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Synchronous salivary gland tumors with regard to two cases

Tumores sincrónicos de glándula salivar. A propósito de dos casos

 

J. Azúa-Romeo¹, E. Saura Fillat², T. Usón Bouthelier², M. Tovar Lázaro³, J. Azúa Blanco⁴

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Abstract: We present two new cases of synchronous tumors of the salivary gland. The first one refers to two benign tumors in the same parotid gland, while second one shows a benign lesion and a malignant tumor in two ipsilateral salivary glands. The appearance of synchronous multiple unilateral parotid tumors of different histologic types is extremely rare, accounting for less than 0.3%; this is usually a Warthin´s Tumor associated to other tumors. The current literature has been reviewed and 42 cases have been found entailing two different parotid tumors coexisting. Of these 11 referred to adenolymphoma associated with pleomorphic adenoma. Preoperative cytological diagnosis, as well as an exhaustive intraoperative inspection are the clues to discover nonidentified second tumors. Key words: Synchronous; Salivary gland; Tumor; Cytology.

Resumen: Presentamos dos casos de pacientes con tumores sincrónicos de glándula salivar. El primero se refiere a dos tumores benignos en la misma glándula parótida, mientras que el segundo caso se trata de una lesión maligna y otra benigna en glándulas parótida y submaxilar ipsilaterales. Los dobles tumores sincrónicos homolaterales de glándula salivar son muy infrecuentes, suponiendo menos del 0,3% de los tumores de esta localización. La bibliografía revisada refiere 42 casos entre los que sólo 9 presentan un tumor maligno y otro benigno. El diagnóstico citológico preoperatorio, junto con una exhaustiva inspección macroscópica intraoperatoria son las claves para detectar los segundos tumores, habitualmente más pequeños y no detectados en estudios previos, y así adecuar el procedimiento terapéutico. Palabras clave: Sincrónico; Glándula salivar; Tumor; Citología.

Recibido: 13 de septiembre 2004

Aceptado: 11 de noviembre 2004

 

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1 Patólogo. Instituto Aragonés de Ciencias de la Salud.
Profesor Facultad de Medicina. Zaragoza, España.
2 Servicio de Cirugía Oral y Maxilofacial. Hospital Universitario Miguel Servet. Zaragoza, España.
3 Atención Primaria. Zaragoza, España
4 Patólogo. Profesor Facultad de Medicina. Zaragoza, España

Correspondencia:
Dr. Javier Azúa-Romeo
Sº Anatomía Patológica (Banco de Tumores)
Hospital Universitario Miguel Servet
Pº Isabel la Católica 1 y 3
50009 Zaragoza, España.

 

Introduction

The appearance of two ipsilateral salivary gland tumors of different histologic types is extremely rare making up less than 0.3% of all salivary gland tumors.¹ The current literature describes only 42 cases, of which only 9 refer to the coexistence of a benign tumor with a malignant one.²

This is normally a Warthin´s tumor associated with another type of lesion. We present two cases that are quite exceptional. One cases documents the association of a Warthin’s tumor with a pleomorphic adenoma in the same parotid gland, and the other documents the association of an acinic cell carcinoma with an oncocytoma of the parotid and submaxillary glands on the left side respectively. The initial diagnostic approach is usually made by means of fine needle aspiration puncture (FNA), through which high sensibility and specificity is obtained, allowing an appropriate therapeutic approach.⁵

Case Report

Case 1

A 44 year-old patient was sent by her General Practitioner to the Oral and Maxillofacial Unit due to the appearance of a tumor-like mass in the parotid gland, which to palpation appeared uninodular. A benign tumor was suspected and fine needle aspiration was carried out with a 23 G needle in the Pathological Anatomy Unit. The material obtained was fixed immediately in 95% alcohol while one aspirate was reserved and air-dried. The samples that were fixed in alcohol were stained by the Papanicolaou technique, while those that were air-dried were stained using the Diff- Quick technique.

Cytologically a lesion was observed consisting of ductal epithelial cells with a basophilic fibrillar and myxomatous background. In addition chondroid material and fragments of fibrillar eosinophilic stroma could be appreciated. The cytological diagnosis was of pleomorphic adenoma (PA) and, as a result, a total parotidectomy was carried out while preserving the facial nerve.

The surgical specimen was fixed in 10% formaldehyde solution and a tumor that was 2 x 2 cm was observed macroscopically. It was whitish in color with a soft elastic consistency, and it was partially encapsulated. A polylobulated nodular lesion measuring 1.5 cm, lymphoid in appearance and separated from the main tumor by a fine strand of stroma (Fig. 1A) was also observed. Following fixation, the specimen was completely sectioned and it was routinely processed in order to obtain paraffin blocks. The stain used for the microscopical observation was the classical hematoxylineosin.

The microscopical study showed that the main tumor was well-circumscribed, and surrounded by a connective pseudo-capsule, composed of a biphasic population of glandular epithelial cells and myoepithelial cells that were surrounded by myxoid fibrillar material, and the diagnosis of pleomorphic adenoma was consequently confirmed. (Fig. 1C). The other nodules were lymphatic nodes, but a new lesion appeared, small in size, very close to the mixed tumor. It was cystic in appearance and papillary, and it was made up of a double strand of oncocytic cells that were lining the cystic spaces with the adjacent lymphoid stroma (Fig. 1B). The definitive histopathological diagnosis was of synchronous pleomorphic adenoma and Warthin’s tumor of the right parotid gland.

Case 2

This was a 77 year-old male, that had a consultation for a palpable preauricular tumor-like mass on the left side, that was 3 cm in diameter and painless. An echography of the lesion was carried out which revealed a lesion that was occupying the space of the left parotid gland. It was hypoechogenic and measured 23 x 12 x 19 mm. A nodular lesion was appreciated by chance that was hypoechogenic and which, with posterior enhancement, measured 12 x 9 mm in the left submaxillary gland.

With this data FNA was carried out on both nodules. In the larger one the cellularity that was observed was greater and of a large size. The nuclei were in the center and occasionally at the periphery, and they were round with inconspicuous nucleoli and finely granular chromatin. The cytoplasm was very characteristic, being abundant, pale and foamy with indistinct borders. It was vacuolated and contained an abundance of purplish granules. The cells were arranged in groups that were more or less compact and in large bi-dimensional sheets, on occasions adopting an architectural pattern that was acinic. Next to this cellularity, smaller sized cells could be appreciated, with scant cytoplasm with small central nuclei either rounded or oval. The cytological image was consistent with a tumor made up of acinic cells, probably carcinoma and its excision was therefore proposed.

The FNA of the second lesion produced less material, given its smaller size. Nevertheless much homogenous cellular material was observed with rounded borders together with cytoplasm that was slightly basophilic and granular, with large central nuclei and no atypical features. This suggested a diagnosis of oncocytoma and its excision was also recommended.

The patient had no known allergies and he had a pacemaker due to 2^nd degree AVB. There was no other data of interest to the present case and he was admitted for programmed surgery for the tumor-like mass of the left parotid gland and left submaxillary gland.

The definitive histological examination confirmed the origin in both of the diagnoses, and the complete resection of both tumors, with no infiltration of the surgical margins. Unfortunately no macroscopic image has been kept of these specimens, but these can be seen microscopically in figure 2.

Discussion

The existence of synchronous tumors with different histological types in ipsilateral salivary glands or in the same gland is very rare. They account for 0.3% of all tumors of the salivary glands, and the most common association is a Warthin’s tumor with other entities.¹ In a revision of the literature we have found a total of 42 cases, of which 9 referred to the concurrence of a benign tumor with a malignant one – more frequently a Warthin’s tumor with a mucoepidermoid carcinoma – and 11 to the coexistence of a Warthin’s tumor with a pleomorphic adenoma.^2-4,6

Our cases have the additional characteristic of having an initial diagnosis by FNA in three of the four lesions, with the fourth lesion being a casual finding, that is to say the Warthin’s tumor in Case A. In our center an echography and FNA is routinely carried out on those patients with tumor-like masses with a likely salivary origin. Three passes are carried out with a 23G needle, with the help of a Cameco^® (manual aspiration device) after which two slides are fixed in alcohol so that they can be stained with Papanicolaou’s technique and the rest (6 slides) are air-dried so that the Diff-Quick stain can be used.⁷

Thanks to this diagnostic approach surgical interventions, as well as follow-ups and check-ups, as is the case, can be more reliably programmed for those patients not due for surgery.⁵

The most complicated part of cases with two salivary tumors lies in establishing treatment that is efficient for both tumors as, depending on the tumor that has been diagnosed first, and sometimes there may be only one as in Case A, the surgical approach may be insufficient, and the patient may have to be reoperated following the definitive histopathological diagnosis. When cytological studies are routinely carried out, high levels of effectiveness are reached, and the number of more aggressive diagnostic procedures is reduced. Nevertheless the surgeon should be watchful, and he should remember that, although not very common, there are these cases of double lesions and when faced by a nodule that is minimally suspicious an intraoperative study should be requested. Many authors in fact question the «infrequency» of these lesions and postulate the possibility that, rather than rare, these are undetected tumors.¹

Conclusions

We recommend a thorough preoperative examination, including imaging techniques and FNA in order to reach a diagnosis that is as accurate as possible, remembering that there can be a possibility of finding other tumor by chance in the same gland or an adjacent one. During the surgical act an exhaustive macroscopic examination should be carried out, and if there is anything suspicious, a microscopical examination should be carried out, in order to detect any multifocal tumors before closing the site.

References

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