Causation of metastasis of adenoid cystic carcinoma of the mandible to the Gasserian ganglion in a 47-year-old woman was determined through the dental history. The patient’s experience of multiple tooth extraction during a single visit three years before suggested that the carcinoma existed in the mandible at that time, and that curettage after extraction pushed the tumor cells through the perineural space of the inferior alveolar nerve toward the ovoid foramen, thus causing a metastatic lesion of the Gasserian ganglion to develop. Radiotherapy was instituted; neither recurrence nor metastasis of the carcinoma has occurred.
Metastasis of adenoid cystic carcinoma of the mandible to the Gasserian ganglion Y . Yosh im ura, DDS K. H asegaw a, DDS T . W ada, DDS K. Fujita, DDS K. Kaw akatsu, DDS, Osaka, Japan
A denoid cystic carcinom a, w hich originally was term ed “ cylindrom a” by B illroth,1 is a com m on tum or o f the salivary and m ucous glands in and around the oral cavity.2"6 T h e clinical features of the disease are th at the tum or grows slowly; there is a high incidence o f local recurrence and m etas tasis; neurologic pain is caused by the tu m o r’s infiltration into the nerves; and it prim arily affects w om en.3,5 T he following case report m erits consideration because o f the rarity o f location and the m ode of m etastasis o f adenoid cystic carcinom a.
Report of case A 47-year-old w om an w as referred by her neurosurgeon in M ay 1972 to the departm ent of oral and maxillofacial surgery, O saka (Japan) U niversity D ental School. She was operated on
by a m em ber of the departm ent o f neurosurgery after clinical diagnosis confirm ed neurinom a of the right G asserian ganglion; histopathologic exam ination o f the specim en show ed it to be adenoid cystic carcinom a. T he neurosurgeon suspected that the prim ary lesion existed in the rhino-otological o r the oral region, o r both. A n exam ination conducted by the otolaryngological departm ent show ed no pathological findings. T he p atien t’s dental history show ed that three years previously several right m olars w ere ex tracted in one visit at a dental clinic because of severe m ovem ent and abnorm al sensation of that region. T he w ound seem ed to heal w ithout com plications, bu t paresthesia of the right m en tal region rem ained. T he m edical history was noncontributory. ■ Oral exam ination: O n the right side of the floor o f the m outh a scar about 2.5 cm in length was JADA, V ol. 96, M arch 1978 ■ 469
observed along the dental arch. Tongue m ove m ent also was restricted slightly, especially any m ovem ent to the left side. P aresthesia o f the right m ental and infraorbital regions was experienced. A com prehensive exam ination o f the m ajor saliv ary glands show ed no abnorm alities. Subm an dibular, subm ental, and cervical regional lymph nodes w ere not palpable except for a very small, m ovable one on the right side. Inspection and palpation in and around the oral cavity did not show any o th er pathological findings. ■ Laboratory findings: R esults o f all laboratory tests w ere within norm al limits, including blood cell count and analysis, urinanalysis, prothrom bin test o f liver function, and serum electrolytes. W asserm an reaction was negative. A radiograph o f the chest show ed no abnorm alities. ■ Radiographic exam ination: A t the right mental foram en, a pea-sized, ovoid cavity with defined margins caused by bone resorption was observed and was indicative of a cystic lesion (Fig 1). Subm entovertex projection o f the skull showed a larger ovoid foram en on the right side than on the left. (Fig 2). M any radiopaque areas also were observed on angiogram s taken by the departm ent o f neurosurgery.
Fig 1 ■ Ovoid-shaped bone resorption at m ental form en.
fully and subm itted for biopsy. R esults o f the bi opsy o f the m andibular lesions and G asserian ganglion showed tum or parenchym a in a glandu lar architecture, characterized by small and large islands in fibrous connective tissue (Fig 3,4). The characteristic cribriform pattern was not clearly evident in every place (Fig 5); there w ere abun dant areas o f solid growth. T um or cells stained
■ B iopsy: Reflection o f the m ucoperiosteal flap on the lingual side was perform ed after inducing anesthesia to the inferior alveolar nerve. T he bone defect was filled with whitish-gray tissue in the m andible, originally noticeable on the radio graph. Portions o f the tissue w ere rem oved care
TH E A UTHO RS
YO SH IM U R A
HASEGAW A
W ADA
Dr. Yoshim ura is an assistant, Dr. Fujita is an associate professor,
FUJITA
KAWAKATSU
oral radiology; and Dr. H asegaw a is an instructor, departm ent of
and Dr. Kawakatsu is a professor, departm ent of oral and m axillo-
oral pathology. Osaka University D ental School, Joan-cho, Kita-ku,
facial surgery; Dr. W ada is an assistant professor, departm ent of
Osaka, Japan. Address requests fo r reprints to Dr. Yoshim ura.
470 ■ JADA, Vol. 96, M arch 1978
Fig 3 ■ Nest of tum or cells shows solid form in scanty strom a (hem atoxylin-eosin stain. Original m agnification, x100).
Fig 4 ■ S pecim en from m andible shows nest of tum or cells that form small cell islands and ductal structures (hem atoxylin-eosin stain. O riginal m agnification, x200).
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Fig 2 ■ M any radiopaque areas seen on angiogram of cerebral vessels. N otice ovoid foram en is larger on right side than on left.
with hem atoxylin-eosin showed small bodies with relatively small am ounts o f cytoplasm . M itotic di vision did not occur in both solid and cribriform patterns. N ucleoli were distinct in some areas. T he specim en taken from the G asserian ganglion showed a histopathologic architecture similar to that observed in the m andible. T he pathological diagnosis was adenoid cystic carcinom a. ■ Treatm ent: R adiotherapy was initiated in June 1972. D osages o f radiation adm inistered were 1,000 rads per w eek; the patient was scheduled to receive a total dose o f 8,000 rads. D osages were adm inistered at three-w eek intervals because se vere stom atitis developed after the patient re ceived 6,000 rads. T reatm ent was com plete after adm inistration of 8,000 rads. Follow -up exam inations have been continued. In F ebruary 1975, the p atien t’s general health was good; neither recurrence nor m etastasis of adenoid cystic carcinom a was detected. She said that the abnorm al feeling she experienced in the right infraorbital and m ental regions had di minished slightly.
r
Fig 5 ■ S pecim en from m andible shows typical cribriform struc ture of adenoid cystic carcinom a (hem atoxylin-eosin stain. O rig inal m agnification, x200).
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Y oshim ura— others: A DE N O ID C YSTIC C A R C IN O M A OF M A ND IB LE ■ 471
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Discussion
-= to
A denoid cystic carcinom a rarely affects the m an dible.7-9 It often is m isdiagnosed as an odon togenic tum or, including an am eloblastom a or a cyst; the m andibular prem olars, m olars, and angle are the areas m ost affected (Table 1). O ther saliv ary gland tum ors also arise in these regions of the m andible.9' 11 It is considered that adenoid cystic carcinom a is derived from either epithelial com ponents o f odontogenic cyst that underw ent the m ucous m etaplasia, aberrant salivary gland tis sues such as subm andibular and sublingual saliv ary g lands,12,13 or a retrom olar m ucous gland that becam e part o f the m andible during develop m en t.14 Although m etastasis o f a carcinom a is lym phogenous or hem atogenous in nature, or both, the characteristic m ode of spread in adenoid cystic carcinom a is infiltration o f the neural sheath or perineural spaces as well as the previ ously m entioned ro u te.15 It is considered that the tum or invades into the inferior alveolar nerve to w ard the G asserian ganglion if a prim ary lesion exists in the mandible. A nother possibility is that an aberrant m ucous gland located at the ganglion changes into malignant tissue that is not as sociated with m andibular adenoid cystic car cinom a. H ow ever, the dental history of our p a tient, which indicated the experience o f multiple tooth extraction during a single visit three years before, suggests that adenoid cystic carcinom a may have existed in the mandible at that tim e, and that curettage after extraction pushed the tum or cells into a nerve canal tow ard the ovoid foram en, thus causing a m etastatic lesion of the trigeminal gangion to develop. M eyer and Shklar6,16 established the following four criteria to confirm the presence of m etas tasis: — a proven prim ary tum or with histologic con firm ation and, w henever possible, with radiographically supportive evidence; — maxillary or m andibular m etastasis with his tologic confirm ation and with radiographic evi dence o f bone involvem ent; — histologic correlation o f the m etastatic oral lesion with the prim ary lesion; and — direct extension ruled out by a wide, clear margin around the prim ary lesion w hen the pri m ary site is anatom ically near the m etastasis, with no tum or tissue present betw een the two foci. O ur case satisfied all criteria. H ow ever, tum or cells may exist along the nerve sheath, but their 4 7 2 ■ JADA, V ol. 96, M arch 1978
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Metastasis of adenoid cystic carcinoma of the mandible to the Gasserian ganglion Y . Yosh im ura, DDS K. H asegaw a, DDS T . W ada, DDS K. Fujita, DDS K. Kaw akatsu, DDS, Osaka, Japan
A denoid cystic carcinom a, w hich originally was term ed “ cylindrom a” by B illroth,1 is a com m on tum or o f the salivary and m ucous glands in and around the oral cavity.2"6 T h e clinical features of the disease are th at the tum or grows slowly; there is a high incidence o f local recurrence and m etas tasis; neurologic pain is caused by the tu m o r’s infiltration into the nerves; and it prim arily affects w om en.3,5 T he following case report m erits consideration because o f the rarity o f location and the m ode of m etastasis o f adenoid cystic carcinom a.
Report of case A 47-year-old w om an w as referred by her neurosurgeon in M ay 1972 to the departm ent of oral and maxillofacial surgery, O saka (Japan) U niversity D ental School. She was operated on
by a m em ber of the departm ent o f neurosurgery after clinical diagnosis confirm ed neurinom a of the right G asserian ganglion; histopathologic exam ination o f the specim en show ed it to be adenoid cystic carcinom a. T he neurosurgeon suspected that the prim ary lesion existed in the rhino-otological o r the oral region, o r both. A n exam ination conducted by the otolaryngological departm ent show ed no pathological findings. T he p atien t’s dental history show ed that three years previously several right m olars w ere ex tracted in one visit at a dental clinic because of severe m ovem ent and abnorm al sensation of that region. T he w ound seem ed to heal w ithout com plications, bu t paresthesia of the right m en tal region rem ained. T he m edical history was noncontributory. ■ Oral exam ination: O n the right side of the floor o f the m outh a scar about 2.5 cm in length was JADA, V ol. 96, M arch 1978 ■ 469
observed along the dental arch. Tongue m ove m ent also was restricted slightly, especially any m ovem ent to the left side. P aresthesia o f the right m ental and infraorbital regions was experienced. A com prehensive exam ination o f the m ajor saliv ary glands show ed no abnorm alities. Subm an dibular, subm ental, and cervical regional lymph nodes w ere not palpable except for a very small, m ovable one on the right side. Inspection and palpation in and around the oral cavity did not show any o th er pathological findings. ■ Laboratory findings: R esults o f all laboratory tests w ere within norm al limits, including blood cell count and analysis, urinanalysis, prothrom bin test o f liver function, and serum electrolytes. W asserm an reaction was negative. A radiograph o f the chest show ed no abnorm alities. ■ Radiographic exam ination: A t the right mental foram en, a pea-sized, ovoid cavity with defined margins caused by bone resorption was observed and was indicative of a cystic lesion (Fig 1). Subm entovertex projection o f the skull showed a larger ovoid foram en on the right side than on the left. (Fig 2). M any radiopaque areas also were observed on angiogram s taken by the departm ent o f neurosurgery.
Fig 1 ■ Ovoid-shaped bone resorption at m ental form en.
fully and subm itted for biopsy. R esults o f the bi opsy o f the m andibular lesions and G asserian ganglion showed tum or parenchym a in a glandu lar architecture, characterized by small and large islands in fibrous connective tissue (Fig 3,4). The characteristic cribriform pattern was not clearly evident in every place (Fig 5); there w ere abun dant areas o f solid growth. T um or cells stained
■ B iopsy: Reflection o f the m ucoperiosteal flap on the lingual side was perform ed after inducing anesthesia to the inferior alveolar nerve. T he bone defect was filled with whitish-gray tissue in the m andible, originally noticeable on the radio graph. Portions o f the tissue w ere rem oved care
TH E A UTHO RS
YO SH IM U R A
HASEGAW A
W ADA
Dr. Yoshim ura is an assistant, Dr. Fujita is an associate professor,
FUJITA
KAWAKATSU
oral radiology; and Dr. H asegaw a is an instructor, departm ent of
and Dr. Kawakatsu is a professor, departm ent of oral and m axillo-
oral pathology. Osaka University D ental School, Joan-cho, Kita-ku,
facial surgery; Dr. W ada is an assistant professor, departm ent of
Osaka, Japan. Address requests fo r reprints to Dr. Yoshim ura.
470 ■ JADA, Vol. 96, M arch 1978
Fig 3 ■ Nest of tum or cells shows solid form in scanty strom a (hem atoxylin-eosin stain. Original m agnification, x100).
Fig 4 ■ S pecim en from m andible shows nest of tum or cells that form small cell islands and ductal structures (hem atoxylin-eosin stain. O riginal m agnification, x200).
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Fig 2 ■ M any radiopaque areas seen on angiogram of cerebral vessels. N otice ovoid foram en is larger on right side than on left.
with hem atoxylin-eosin showed small bodies with relatively small am ounts o f cytoplasm . M itotic di vision did not occur in both solid and cribriform patterns. N ucleoli were distinct in some areas. T he specim en taken from the G asserian ganglion showed a histopathologic architecture similar to that observed in the m andible. T he pathological diagnosis was adenoid cystic carcinom a. ■ Treatm ent: R adiotherapy was initiated in June 1972. D osages o f radiation adm inistered were 1,000 rads per w eek; the patient was scheduled to receive a total dose o f 8,000 rads. D osages were adm inistered at three-w eek intervals because se vere stom atitis developed after the patient re ceived 6,000 rads. T reatm ent was com plete after adm inistration of 8,000 rads. Follow -up exam inations have been continued. In F ebruary 1975, the p atien t’s general health was good; neither recurrence nor m etastasis of adenoid cystic carcinom a was detected. She said that the abnorm al feeling she experienced in the right infraorbital and m ental regions had di minished slightly.
r
Fig 5 ■ S pecim en from m andible shows typical cribriform struc ture of adenoid cystic carcinom a (hem atoxylin-eosin stain. O rig inal m agnification, x200).
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xi
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Y oshim ura— others: A DE N O ID C YSTIC C A R C IN O M A OF M A ND IB LE ■ 471
.¿ Í
Discussion
-= to
A denoid cystic carcinom a rarely affects the m an dible.7-9 It often is m isdiagnosed as an odon togenic tum or, including an am eloblastom a or a cyst; the m andibular prem olars, m olars, and angle are the areas m ost affected (Table 1). O ther saliv ary gland tum ors also arise in these regions of the m andible.9' 11 It is considered that adenoid cystic carcinom a is derived from either epithelial com ponents o f odontogenic cyst that underw ent the m ucous m etaplasia, aberrant salivary gland tis sues such as subm andibular and sublingual saliv ary g lands,12,13 or a retrom olar m ucous gland that becam e part o f the m andible during develop m en t.14 Although m etastasis o f a carcinom a is lym phogenous or hem atogenous in nature, or both, the characteristic m ode of spread in adenoid cystic carcinom a is infiltration o f the neural sheath or perineural spaces as well as the previ ously m entioned ro u te.15 It is considered that the tum or invades into the inferior alveolar nerve to w ard the G asserian ganglion if a prim ary lesion exists in the mandible. A nother possibility is that an aberrant m ucous gland located at the ganglion changes into malignant tissue that is not as sociated with m andibular adenoid cystic car cinom a. H ow ever, the dental history of our p a tient, which indicated the experience o f multiple tooth extraction during a single visit three years before, suggests that adenoid cystic carcinom a may have existed in the mandible at that tim e, and that curettage after extraction pushed the tum or cells into a nerve canal tow ard the ovoid foram en, thus causing a m etastatic lesion of the trigeminal gangion to develop. M eyer and Shklar6,16 established the following four criteria to confirm the presence of m etas tasis: — a proven prim ary tum or with histologic con firm ation and, w henever possible, with radiographically supportive evidence; — maxillary or m andibular m etastasis with his tologic confirm ation and with radiographic evi dence o f bone involvem ent; — histologic correlation o f the m etastatic oral lesion with the prim ary lesion; and — direct extension ruled out by a wide, clear margin around the prim ary lesion w hen the pri m ary site is anatom ically near the m etastasis, with no tum or tissue present betw een the two foci. O ur case satisfied all criteria. H ow ever, tum or cells may exist along the nerve sheath, but their 4 7 2 ■ JADA, V ol. 96, M arch 1978
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