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Metastatic malignancy in the submandibular gland
Metastatic malignancy in the submandibular gland
Metastatic disease in the major salivary glands has lwn obscrvc~d und reported in the literature. Often the site of origin of the primary tumor is the skin or mucosal lining of the head and nwk structures, and the primary tumors most commonly are mclanomxs or squamous-cell carcinomas. The most frequently involved salivary gland is the parotid. The suhnandilmlar and su)dingu:tl glands are rarely involved 1)~ nietastatic diseaw. Secondary lesions arising from distant primary tumors may occasionally involw the parotid gland, but thry are ~stwnwly rare in the other major salivary glands. The second reported c:w of nwtastatic carcinoma in the submandib ular gland is presented here. Our case ant1 tllc, prwiously wportcd case are similar in that both primary tumors originntc>d in the breast. The clinical and morphologic features are rcriewcd.
M
alignant neoplasms arising from sites out&c the head and neck region glands are 1111c0rI1Ir1011. Most reportccl and metastasizing to the major salivary cases have involved the parotitl gland.‘-+ Only one c’asc of metastatic spread to the submandibular gland from a tumor outsitlc the head and neck has been previously recorded in the literature.” \Vo present in this article the second case of this type. CASE REPORT A GO-year-old white woman was admitted to Kings County Hospital in September, 1973, complaining of a m:ws in the right subnu~ndibular region, accomp:n~ic~d by au wrache on the right side. In 1959, she had undergone a left radical mastectomy and oophorectomy for duct carciThe patient did well until noma of the brcnst. This was followed by 4,250 I‘ of radiation. 1965, wlwn shot developed a mass in the right breast. This was biopsied and diagnosed as a lobular carcinoma, and a right radical mnstcctomy was performed. r”lhe tolerated the procedure weI1 and was sulwquently followed in thr henst clinic.
469
470
Solomon,
Rosen,
Fig. 1. Hemisected
and
Gwrdner
submandibular
gland. Arrows
point to ill-defined
focus of white indura-
tion. In 1970, a “coin lesion” in the lower lobe of the right lung was observed on routine chest films. The lesion was excised via lobectomy and diagnosed as a papillary adenocarcinoma of terminal bronchiolar origin. After recovery she remained well until the following year, when she developed paresthesia of the left thumb and forefinger. These symptoms were accompanied by a loss of flexor movement of the left arm. In 1972, an enlarged left supraclavicular lymph Diagnosis of the aspirate was “consistent with node was biopsied via needle aspiration. adcnocarcinoma.” This region was radiated (5,000 r). During the current hospitalization, positive physical findings included the aforementioned right submandibular mass, diminished flexor and extensor movement of the left arm, and paresthesia of the lrft fingertips. No axillary lymph nodes were palpated and all scars from previous operations were unremarkable. The submandibular mass measured 4.0 by 2.0 cm. and was described as being movable and not attached to the overlying skin In addition, a mass t,hat was 1.0 cm. in diameter was palpated in the lower pole of the parotid gland. This mass \VXS on palpation. The clinical impression of the subdescribed as being fixed, firm, and painful mandibular swelling was that of a chronic sialadenitis; however, in view of this patient’s history, the possibility of some neoplastic lesion was considered. The findings of thr chest studies were unremarkable. x-ray examination were within normal limits, and all laboratory Excision of the submandibular gland was recommended and this was accomplished, with the patient under general anesthesia, without complication. The surgical wound healed in a normal fashion. Pathology On cut A submandibular gland, 3.0 by 1.5 em. in size, was submitted for examination. section it demonstrated the usual lobulations, gray-pink coloration, and an irregularly shaped, ill-defined focus of white induration 0.5 cm. in diameter (Fig. 1). Microscopic examination of the white zone of induration revealed a large focus of infiltrating adenocarcinoma (Figs. 2 and 3). The remainder of the gland showed changes consistent with chronic sialadenitis. At this point in the evaluation of the disease process all available histologic material from previous surgical procedures was reviewed. The solitary right lower lobe lesion that was removed in 1970 produced a spectrum of opinion when seen by several pathologists. The majority believed this lesion to be of metastatic nature; however, the possibilty of its representing an unrelated primary terminal bronchiolar
Pm. 8. Section of suhmxndihular gland containing ill-defined lesion points to nerve. (Hematoxylin and rosin stain. Magnification, x15.)
(within
b&x). Arrow
wrc~il~oma ww seriously vonxidcrrtl by otllcrs. l’llis spectrum of interpretation is not at all unusual it1 caws of lung lesions of tllis type’. TtlcL histologic pattern of the tumor seen in our sul~m:~ndilmlar gland sptximrrr +s that of an :Idl~IIoCar~:i~lo~tla with productive fibrosis and perineural space infiltration. The tumor ww lwated within tile glandular p:w~wcl~ym:~. No definite evidcncr of sulnnandil~ular gland origin \VRS nottd (Figs. 2-4). Because tlltk appearance of tlw suhmandilmlar gland tumor w:w almost identical to that of tile tumor prewnt in the, left breast rcserted in 1959, IV(~consitler tllcl left breast to Iw tllca source of tllct submandibular lesion. Moreowr, the morpllologic pattern of tile sulm~:n~dihular tumor seen in this ease is not cllaracteristic of primary ailtllo(‘ar(‘inorIla arising in saliwry glands, althouyh it may occur. We Imw studi one c:xw of primwy adcnocarcinoma of tlw parotitl gland in wllic.11 tilt, microscopic appearaucc of tllc, tumor was quite similar to tll:l t in tile prwvut c:lse.
DISCUSSION
C’onlep ant1 Arella’ rcviewrtl cight,x=ont~ ~ascs of parotid glantl metastasis. Tli(: majority (83 per writ) of the primary lesions in their study wcrc melanomas of the skin of the tcmplc, scalp, or car, and squamous-w11 eareinomas from the oral cavity, accessory nasal sinuses, the pliarps, ant1 ear. The rc~rnaintler of the primary lesions arose in other head and nwk strudurcs. I’atc,v anti associates’ rcportctl six additional eases of nietastatic lesions in the parotid gland. b’ivc of thcsc were from distant primary sites-lniig (two vases), liicliic~p, pancrcas, ant1 stonmc.h. In two of thcsc ceasesthe metastatic natuw of the tumor was not irnmctliately aypreciatcd and thci wrrwt tliapiosis was matlc only after revitx of riecropsy findings. anti J~o~wr,~iii their rcviw of m:ilignant, salivary glaild Ciriigt'
472
Solomon, Rosen, and Gardner
Oral Surg. March, 1975
Ffo. 3. Section showing detail of lesion seen in Fig. 2, an adenocarcinoma with productive fibrosis and perineural space infiltration. (Hematoxylin and eosin stain. Magnification, x125.)
Fig. 4. Section from left breast mandibular gland lesion. (Hematoxylin
showing detail of adenocnrcinoma that and eosin stain. Magnification, x125.)
resembles
sub-
tumors, presented another nine cases of metastatic tumor. Eight of these cases were parotid gland lesions. The primary sites were facial melanoma (two cases), squamous-ccl1 carcinoma arising in the same head and neck structures mentioned in the report of Conely and Arena (four cases), squamous-cell carcinoma of the renal pelvis, and an adenocarcinoma arising in a meibomian gland of the eyelid. Only one of these primary tumors that metastasized to the parotid gland did not arise in the region of the head and neck. The ninth case in this study involved the submandibular gland, and the site of the primary lesion was the breast. To our knowledge, this is the only case mentioned in the literature of a metastatic lesion in the submandibular glantl arising from a distant primary site. It is of interest that the submandibular gland tumor in our case was also of breast origin.
Evans and Cruickshank* observed that,, “When block dissection of lymph nodes in the neck is carried out for carcinomas secondary to a primary growth in the lip, mouth or tongue, it is usual Cor the dissection to include the submandibular gland, but, even microscopic+ally, it is quite unusual for the submandibular gland to be involved despite the sometimes advanced secondary carcinoma that may be present in the adjacent submandibular lymph areas.” The same has been true in our experience. The association of salivary gland carcinoma with breast carcinoma has recently been demonstrated by Berg and associatcs.Z In their series of 396 patients with salivary gland cancer, they found an incidence of primary breast cancer which exceeded by eightfold the normal incidence of breast cancer in their geegraphic location. The breast carcinomas in their series did not have any histological resemblance to the salivary gland carcinomas. Therefore, the possibility of encountering both breast and salivary glantl cancer in the same patient is not remote. The previously noted parotid nodule was thought to represent another focus of mctastatic disease, but the possibility of another primary lesion was not ruled out. The patient was startetl on a chemotherapy regimen of fluorouracil, methotrexate, prednisonc, and Cytoxan. She has been followed for 1 pear, during which time the parotid nodule has disappeared. Some complications secondary to the chemotherapy were observed, but the patient has not had overt recurrence of any of the aforementioned tumor conditions. She is currently being follo~~tl in the breast clinic. The differences in therapeutic approaches to a patient with disseminated breast carcinoma ant1 to a patient with treated and nonrecurrent breast carcinom;1 who develops an inclepenclent primary salivary gland carcinoma are great. This underscores the need to recognize the occurrence, albeit rare, of metastatic tliscase from distant sites to the salivary glands.
REFERENCES
1. Conlcy, i64.
J., and Arena,
H.: Parotid
Gland as n Foeus of Metastxsis,
Arch.
Surg. 87: 757.
196X
2. Patky, 1): H., Thackray, A. C., and Keeling, I). H.: Malignant Disease of the Br. .J. (:ancer 19: 712-737, 1965. Tumors of the Major Salivary Glands, 3. Grage, T. Is., and Lober, P. H.: Malignant 52: 281-294. 1962. 1. Evans, R. iv., ant1 Cruickahank, A. H.: Carcinoma: Epithelial Tumors of the Glands, Philadelphia, 1970, W. H. Saunders Co., pp. 266, 276. 5. Berg, .J. I$‘., Hutter, R. V. P., ant1 Forte, F. W.: The Unique Association Salivary Glantl Cancer and Breast Cancer, J. A. M. A. 204: 771-774, 1968. Ecprint
requests
to :
1)r. Marshall P. Solomon I)epartment of Pathology St:lte University-Kings County Hospital 451 (Xarkson Ave. Brooklyn, N. Y. 11203
Ccntcr
Parotid, Surgery Aalivaq Between
Metastatic disease in the major salivary glands has lwn obscrvc~d und reported in the literature. Often the site of origin of the primary tumor is the skin or mucosal lining of the head and nwk structures, and the primary tumors most commonly are mclanomxs or squamous-cell carcinomas. The most frequently involved salivary gland is the parotid. The suhnandilmlar and su)dingu:tl glands are rarely involved 1)~ nietastatic diseaw. Secondary lesions arising from distant primary tumors may occasionally involw the parotid gland, but thry are ~stwnwly rare in the other major salivary glands. The second reported c:w of nwtastatic carcinoma in the submandib ular gland is presented here. Our case ant1 tllc, prwiously wportcd case are similar in that both primary tumors originntc>d in the breast. The clinical and morphologic features are rcriewcd.
M
alignant neoplasms arising from sites out&c the head and neck region glands are 1111c0rI1Ir1011. Most reportccl and metastasizing to the major salivary cases have involved the parotitl gland.‘-+ Only one c’asc of metastatic spread to the submandibular gland from a tumor outsitlc the head and neck has been previously recorded in the literature.” \Vo present in this article the second case of this type. CASE REPORT A GO-year-old white woman was admitted to Kings County Hospital in September, 1973, complaining of a m:ws in the right subnu~ndibular region, accomp:n~ic~d by au wrache on the right side. In 1959, she had undergone a left radical mastectomy and oophorectomy for duct carciThe patient did well until noma of the brcnst. This was followed by 4,250 I‘ of radiation. 1965, wlwn shot developed a mass in the right breast. This was biopsied and diagnosed as a lobular carcinoma, and a right radical mnstcctomy was performed. r”lhe tolerated the procedure weI1 and was sulwquently followed in thr henst clinic.
469
470
Solomon,
Rosen,
Fig. 1. Hemisected
and
Gwrdner
submandibular
gland. Arrows
point to ill-defined
focus of white indura-
tion. In 1970, a “coin lesion” in the lower lobe of the right lung was observed on routine chest films. The lesion was excised via lobectomy and diagnosed as a papillary adenocarcinoma of terminal bronchiolar origin. After recovery she remained well until the following year, when she developed paresthesia of the left thumb and forefinger. These symptoms were accompanied by a loss of flexor movement of the left arm. In 1972, an enlarged left supraclavicular lymph Diagnosis of the aspirate was “consistent with node was biopsied via needle aspiration. adcnocarcinoma.” This region was radiated (5,000 r). During the current hospitalization, positive physical findings included the aforementioned right submandibular mass, diminished flexor and extensor movement of the left arm, and paresthesia of the lrft fingertips. No axillary lymph nodes were palpated and all scars from previous operations were unremarkable. The submandibular mass measured 4.0 by 2.0 cm. and was described as being movable and not attached to the overlying skin In addition, a mass t,hat was 1.0 cm. in diameter was palpated in the lower pole of the parotid gland. This mass \VXS on palpation. The clinical impression of the subdescribed as being fixed, firm, and painful mandibular swelling was that of a chronic sialadenitis; however, in view of this patient’s history, the possibility of some neoplastic lesion was considered. The findings of thr chest studies were unremarkable. x-ray examination were within normal limits, and all laboratory Excision of the submandibular gland was recommended and this was accomplished, with the patient under general anesthesia, without complication. The surgical wound healed in a normal fashion. Pathology On cut A submandibular gland, 3.0 by 1.5 em. in size, was submitted for examination. section it demonstrated the usual lobulations, gray-pink coloration, and an irregularly shaped, ill-defined focus of white induration 0.5 cm. in diameter (Fig. 1). Microscopic examination of the white zone of induration revealed a large focus of infiltrating adenocarcinoma (Figs. 2 and 3). The remainder of the gland showed changes consistent with chronic sialadenitis. At this point in the evaluation of the disease process all available histologic material from previous surgical procedures was reviewed. The solitary right lower lobe lesion that was removed in 1970 produced a spectrum of opinion when seen by several pathologists. The majority believed this lesion to be of metastatic nature; however, the possibilty of its representing an unrelated primary terminal bronchiolar
Pm. 8. Section of suhmxndihular gland containing ill-defined lesion points to nerve. (Hematoxylin and rosin stain. Magnification, x15.)
(within
b&x). Arrow
wrc~il~oma ww seriously vonxidcrrtl by otllcrs. l’llis spectrum of interpretation is not at all unusual it1 caws of lung lesions of tllis type’. TtlcL histologic pattern of the tumor seen in our sul~m:~ndilmlar gland sptximrrr +s that of an :Idl~IIoCar~:i~lo~tla with productive fibrosis and perineural space infiltration. The tumor ww lwated within tile glandular p:w~wcl~ym:~. No definite evidcncr of sulnnandil~ular gland origin \VRS nottd (Figs. 2-4). Because tlltk appearance of tlw suhmandilmlar gland tumor w:w almost identical to that of tile tumor prewnt in the, left breast rcserted in 1959, IV(~consitler tllcl left breast to Iw tllca source of tllct submandibular lesion. Moreowr, the morpllologic pattern of tile sulm~:n~dihular tumor seen in this ease is not cllaracteristic of primary ailtllo(‘ar(‘inorIla arising in saliwry glands, althouyh it may occur. We Imw studi one c:xw of primwy adcnocarcinoma of tlw parotitl gland in wllic.11 tilt, microscopic appearaucc of tllc, tumor was quite similar to tll:l t in tile prwvut c:lse.
DISCUSSION
C’onlep ant1 Arella’ rcviewrtl cight,x=ont~ ~ascs of parotid glantl metastasis. Tli(: majority (83 per writ) of the primary lesions in their study wcrc melanomas of the skin of the tcmplc, scalp, or car, and squamous-w11 eareinomas from the oral cavity, accessory nasal sinuses, the pliarps, ant1 ear. The rc~rnaintler of the primary lesions arose in other head and nwk strudurcs. I’atc,v anti associates’ rcportctl six additional eases of nietastatic lesions in the parotid gland. b’ivc of thcsc were from distant primary sites-lniig (two vases), liicliic~p, pancrcas, ant1 stonmc.h. In two of thcsc ceasesthe metastatic natuw of the tumor was not irnmctliately aypreciatcd and thci wrrwt tliapiosis was matlc only after revitx of riecropsy findings. anti J~o~wr,~iii their rcviw of m:ilignant, salivary glaild Ciriigt'
472
Solomon, Rosen, and Gardner
Oral Surg. March, 1975
Ffo. 3. Section showing detail of lesion seen in Fig. 2, an adenocarcinoma with productive fibrosis and perineural space infiltration. (Hematoxylin and eosin stain. Magnification, x125.)
Fig. 4. Section from left breast mandibular gland lesion. (Hematoxylin
showing detail of adenocnrcinoma that and eosin stain. Magnification, x125.)
resembles
sub-
tumors, presented another nine cases of metastatic tumor. Eight of these cases were parotid gland lesions. The primary sites were facial melanoma (two cases), squamous-ccl1 carcinoma arising in the same head and neck structures mentioned in the report of Conely and Arena (four cases), squamous-cell carcinoma of the renal pelvis, and an adenocarcinoma arising in a meibomian gland of the eyelid. Only one of these primary tumors that metastasized to the parotid gland did not arise in the region of the head and neck. The ninth case in this study involved the submandibular gland, and the site of the primary lesion was the breast. To our knowledge, this is the only case mentioned in the literature of a metastatic lesion in the submandibular glantl arising from a distant primary site. It is of interest that the submandibular gland tumor in our case was also of breast origin.
Evans and Cruickshank* observed that,, “When block dissection of lymph nodes in the neck is carried out for carcinomas secondary to a primary growth in the lip, mouth or tongue, it is usual Cor the dissection to include the submandibular gland, but, even microscopic+ally, it is quite unusual for the submandibular gland to be involved despite the sometimes advanced secondary carcinoma that may be present in the adjacent submandibular lymph areas.” The same has been true in our experience. The association of salivary gland carcinoma with breast carcinoma has recently been demonstrated by Berg and associatcs.Z In their series of 396 patients with salivary gland cancer, they found an incidence of primary breast cancer which exceeded by eightfold the normal incidence of breast cancer in their geegraphic location. The breast carcinomas in their series did not have any histological resemblance to the salivary gland carcinomas. Therefore, the possibility of encountering both breast and salivary glantl cancer in the same patient is not remote. The previously noted parotid nodule was thought to represent another focus of mctastatic disease, but the possibility of another primary lesion was not ruled out. The patient was startetl on a chemotherapy regimen of fluorouracil, methotrexate, prednisonc, and Cytoxan. She has been followed for 1 pear, during which time the parotid nodule has disappeared. Some complications secondary to the chemotherapy were observed, but the patient has not had overt recurrence of any of the aforementioned tumor conditions. She is currently being follo~~tl in the breast clinic. The differences in therapeutic approaches to a patient with disseminated breast carcinoma ant1 to a patient with treated and nonrecurrent breast carcinom;1 who develops an inclepenclent primary salivary gland carcinoma are great. This underscores the need to recognize the occurrence, albeit rare, of metastatic tliscase from distant sites to the salivary glands.
REFERENCES
1. Conlcy, i64.
J., and Arena,
H.: Parotid
Gland as n Foeus of Metastxsis,
Arch.
Surg. 87: 757.
196X
2. Patky, 1): H., Thackray, A. C., and Keeling, I). H.: Malignant Disease of the Br. .J. (:ancer 19: 712-737, 1965. Tumors of the Major Salivary Glands, 3. Grage, T. Is., and Lober, P. H.: Malignant 52: 281-294. 1962. 1. Evans, R. iv., ant1 Cruickahank, A. H.: Carcinoma: Epithelial Tumors of the Glands, Philadelphia, 1970, W. H. Saunders Co., pp. 266, 276. 5. Berg, .J. I$‘., Hutter, R. V. P., ant1 Forte, F. W.: The Unique Association Salivary Glantl Cancer and Breast Cancer, J. A. M. A. 204: 771-774, 1968. Ecprint
requests
to :
1)r. Marshall P. Solomon I)epartment of Pathology St:lte University-Kings County Hospital 451 (Xarkson Ave. Brooklyn, N. Y. 11203
Ccntcr
Parotid, Surgery Aalivaq Between