Mandibular ameloblastoma with intracerebral and pulmonary metastasis

Mandibular ameloblastoma with intracerebral and pulmonary metastasis

Mandibular ameloblastoma with intracerebral and pulmonary metastasis William V. Harrer, Philadelphia, Pa. POST MORTEM THOMAS SERVICE JEFFERSON M...

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Mandibular ameloblastoma with intracerebral and pulmonary metastasis William V. Harrer, Philadelphia, Pa. POST

MORTEM

THOMAS

SERVICE

JEFFERSON

M.D.,* AND UNIVERSITY

and. Art&r SURGICAL

S. Patchefsky,

PATHOLOGY

M.D.,**

SERVICE,

HOSPITAL

A

meloblastomas reportedly represent 1 per cent of all tumors of the jaw.ll z While multiple local recurrences occur in 50 to 72 per cent of reported cases,?, :i only seven cases of metastatic ameloblastoma have been reported. The following case report concerns a patient who had multiple recurrences with local excisions of a mandibular ameloblastoma, underwent a left hemimandibulectomy 15 years prior to death, and developed intracranial extension of the tumor and pulmonaq metastases. CASE REPORT Patient A. M., a 52-year-old Caucasian woman, was admitted to Thomas Jefferson University Hospital in November, 1968, because of symptoms secondary to metastatic amelo. blastoma. In 1915, when the patient was 29 years of age, a diagnosis of left mandibululnr ameloblastoma arising in a clentigerous cyst was made. During the next several years, t,here were multiple local recurrences and local excisions of this lesion. In 1953, 15 years prior to her final admission, the patient underwent a hemimandibulectomy at another institution. She was first admitted to Thomas Jefferson University Hospital in June, 1967, because oL vertigo, deafness, incoordination, and cough. A chest roentgenogram showed multipIt* pulmonary nodules. A skull film showed evidence of previous surgical intervention but no other abnormalities. Other x-ray studies yielded normal findings. In January, 1968, the patient was readmitted because of nausea, dyspnea, and left neck and facial pain. At that time, there was no evidence of local recurrence. Pulmonary nodules were still evident radiographically. A roentgenogram of the skull revealed destruction of the left sphenoi4 sinus. A chest film suggested an increase in the number of nodular pulmonary lesions. The patient’s final admission was in November, 1968, because of left facial pain, inability to swallow, and frequent cough. She appeared acutely ill and cachectic. She was cyanotic and dyspneic at rest. There was ptosis and severe conjunctivitis of the left eye. *Associate “*Assistant

in Pathology. Professor.

893

894

Harrer

and Patchefsky

Oral June,

Fig. 1. Chest roentgenogram, final hospital ameloblastoma. met ;astases from mandibular

Fig.

3. Brain

at autopsy.

Note

large

tumor

admission.

mass

Note

at left

bilateral

cerebellar-pontine

nodular

Burg. 1970

pu lmonary

angle.

Volume Number

Mnndi6ulur

29 6

nmeloZ,lastoma

895

A left seventh nerve paralysis was present. There were no palpable cervical lymph nod A chest film showed partial collapse of the right middle lobe and an increase in size the pulmonary parenchymal nodules (Fig. 1). Therapy was begun with a combination imidazole and fluorouracil. The patient’s condition continued to deteriorate and pnem nitis developed. She died in December, 1968, 23 years after the initial diagnosis ameloblastoma. A complete postmortem examination was performed 12 hours after death. Pertint autopsy findings included recurrent tumor in the soft tissue of the left neck and base

Fig. 3. Ameloblastoma Magnification, x100.)

Fig. x100.)

4. Metastatic

infiltrating

ameloblastoma

cranial

in

lung.

nerve

ganglion.

(Hematoxplin

(Hcmattoxylin

and

eosin

and

stain.

eosin

Magnificntiorl

st:l

les. of of noof slit of

896

sion.

Hurrer

and

Patchefsky

Fig. 5. Soft-tissue recurrence (Hematoxylin and eosin stain.

Oral Burg. June, 1970

in neck. Ameloblastoma Magnification, x400.)

demonstrating

blood

vessel

inva-

the skull, infiltrating into the cranial cavity (Fig. 2), and metastatic ameloblastoma involving both lungs. The tumor involved the left sphenoid area and left cerebropontine angle, with compression of the fifth, sixth, seventh, eighth, ninth, and tenth cranial nerves on the left (Fig. 3). In addition to tumor metastasis, the lungs showed severe, acute bilateral bronchopneumonia. Histologically, this tumor had the classic pattern of ameloblastoma consisting of islands and strands of an odontogenic type of epithelium within a mature fibrous stroma (Fig. 4). Very few mitotic figures were seen. There were many areas of squamous metaplasia. Areas of central necrosis with microscopic cystic degeneration were noted in the local tumor recurrence and in the metastatic sites. The tumor was seen in small blood vessels and lymphatics (Fig. 5).

DISCUSSION Ameloblastomas are thought to arise from the epithelial lining of dentigerous cysts (as in the present case), remnants of dental lamina, enamel organ, or the basal layer of the oral mucous membrane.4 These tumors are locally invasive and can reach large size without evidence of metastasis.5 Seven cases of metastatic ameloblastoma have been previously reported+l’ and are summarized in Table I. In all but one of these casesthere were multiple local recurrences before there was evidence of metastasis. The interval between histologic diagnosis and proved metastasis has been from 3 to 34 years. It is of interest that seven of the eight reported cases have developed in the left jaw. No explanation for this is apparent. The predominant site of metastasis is the lung (five cases). Tumor aspiration has been suggested as the probable metastatic route.7j 8~I2 Cases of lymphatic6 and blood vessel extension*’ 8slo have also been reported. The present case demonstrates vascular invasion, although possible pulmonary tumor aspiration cannot be excluded. Direct extension of the tumor into the cranial cavity has been

Volume Number

Mandibular

29 6

Table I. Metastatic

ameloblastoma

897

ameloblastoma Side

Year

of

Site of metastasis

primary

published

Author

1928

Simmonss

1932

Vorzimer Perla7

1943

Schweitzer Barnfields

1959

tumors

Duration until death (wars i More than “3 (El)”

Four recurrences, hemimandil~alpc~ton~\

21

Six

I4

Ttventy-four local escisions, hemimnndibulectomy, radiun,

14

Seven Ioc,aI excisions, hemimandibulectom~, radiation therapy

3‘i

Multiple recurrence:,, x-ray therapy. radium

More than 38 (a, b)”

Seven local recurrences, hemimandibulnctomy

Clinical

course

Left

Node at carotid bifurcation (Case 2)

Right

Right

Left

Lung, maxilla, base of skull

Lee, White, and Tottens

Left

Lung, pleura, orbit, brain

1965

Tsukada, De La Pava, and Pickrenlo

Left

Lung

19F7

Hoke and Harrelsonll

Left

Cervical

1969

Lash and McCoy=

Left

Lung

Marc than 3 (2 ) .;-

One local exeiaion, h~mimandihnl~cton~~

1969

Harrer and Patchefsky

Left

Lung/skull, intracranial

‘3

Multiple loc:~I clxc+ sions, hrtmimandit)ulcctomy, ctrc~motherapy

“a =

Present

and and

patient

status

unknown.

lung/pleura

liver,

vertebrae

b =

Nineteen

years

from

first

surgical procedures, x-ray therally

histologic

diagnoris.

reportedsp s and was seen in this case. Histologically, tumors which will m&stasize cannot be differentiated from nonmetastasizing ameloblastomas.“, *‘I Ameloblastomas have a high recurrence rate. Local excision with a wide margin free of tumor is the treatment of choice. This may require hemimandibulectomy. Most patients with metastases have had multiple recurrences prior to the development of metastatic disease. Therefore, after incomplete remoua,l, as evidenced by locally recurrent tumor, earlier ablative surgery is intlic~atetl to prevent metastasis. SUMMARY A case in which a 52-year-old woman with a 23-year history of amcloblastoma was unsuccessfully treated by multiple local surgical excisions, a hemimandibulectomy 15 years prior to death, and finally chemotherapy is reported. This patient had widespread soft-tissue recurrence in the left neck, intracranial extension, and multiple parenchymal metastases. Evidence of vascular invasion was present. This represents the eighth reported ease of metastatic ameloblastoma and the third case of combined pulmonary and intracerebral extension. The secretarial R. I,ang,

authors acknowledge assistance of Miss M.D.

the photographic Barbara MacTough.

assistance of The postmortem

Mr.

Ronald Picard and thr: was performed hy JV;1rreu

898

Harrer

ad

Patchefsky

Oral June,

Surg. 1970

REFERENCES 1. Gorlin,

2.

3. 4. 5. 6. 7. 8. 9. 10. 11. 12. 13.

R. J., Chaudhry, A. P., and Pindborg, J. J.: Odontogenic Tumors-Classification, Histopathology, and Clinical Behavior in Man and Domesticated Animals, Cancer 14: 73-101, 1961. Small, I. A., and Waldron, C. A.: Ameloblastoma of the Jaws, ORAL SURG. 8: 281-297, 1955. Masson, J. K., McDonald, J. R., and Figi, F. A.: Adamantinoma of the Jaws: A Clinicopathologic Study of 101 Histologically Proven Cases, Plast. Reconstr. Surg. 23: 510525, 1959. Willis, R. A.: Pathology of Turnours, ed. 4, London, 1967, Butterworth & Co., Ltd. Slavin, G., and Cameron, Mac D. H.: Ameloblastomas in Africans From Tanzania and Uganda, Brit. J. Cancer 23: 31, 1969. Simmons, C. C.: Adamantinoma, Ann. Burg. 88: 693-704, 1928, Vorzimer, J., and Perla, D.: An Instance of Adamantinoma of the Jaw With Metastasis to the Right Lung, Amer. J. Path. 8: 445-453, 1932. Schweitzer, F. C., and Barnfield, W. F.: Ameloblastoma of the Mandible With Metastasis to the Lungs: Relnort of a Case. J. Oral Sura. 1: 287-295. 1943. Lee, R. E., Whyte, \< L., and Totten, R. 8.: xmeloblastoma With Distant Metastasis, Arch. Path. Surg. 68: 23-29, 1959. Tsukada, Y., De La Pava, S., and Pickren, J. W.: Granular-Cell Ameloblastoma With Metastasis to the Lungs-Report of a Case and Review of the Literature, Cancer 18: 916.925. 1965. Hoke, H. F., Jr., and Harrelson, A. B.: Granular Cell Ameloblastoma With Metastasis to the Cervical Vertebrae, Cancer 20: 991-999. 1967. Lash, M., and McCoy, G.: Ameloblastoma of the Mandible With Pulmonary Metastasis, Ann. Otol. 78: 430, 1969. Goldwyn, R., Constable, J., and Murray, J. E.: Ameloblastoma of the Jaw-A Clinical Study, New Eng. J. Med. 269: 126-129, 1963.