TEMPORAL BONE INVOLVEMENT BY PROSTATIC ADENOCARCINOMA: REPORT OF TWO CASES AND REVIEW OF THE LITERATURE Aysegul A. Sahin, MD, Jae Y. Ro, MD, Nelson G. Ordonez, MD, Mario A. Luna, MD, Randal S. Weber, MD, and Alberto G. Ayala, MD

Although axial bone and cranial metastases are common in patients with prostatic carcinoma, temporal bone involvement is rare. We report 2 cases of prostatic adenocarcinoma with temporal bone metastasis and review the relevant literature. One case had a 3-year history of a prostatic carcinoma, whereas in the second case the temporal bone metastasis was the initial presentation. Awareness of the possibility of temporal bone involvement by prostate carcinoma and application of immunohistochemical studies will help to arrive at the correct diagnosis. HEAD & NECK 1991;13:349-354

carcinoma. The purposes of this article are to reemphasize the possibility of temporal bone involvement by metastatic tumors even in patients without such prior history and to stress the importance of complete clinical workup and use of immunohistochemical studies. CASE REPORTS

Care 1. A 69-year-old white man with a history

involvement of the middle ear or temporal bone by metastatic carcinoma is ~ a r e . l Kobayashi -~ et al.’s 1986 review of the world literature revealed 134 cases of temporal bone metastasis.* Origin in the prostate was cited in only 9 of these cases. We report 2 additional cases of metastasis of prostatic adenocarcinoma to the temporal bone. In 1 of these cases, the temporal bone metastasis was the initial presentation of prostatic adeno-

From the Department of Pathology, M. D. Anderson Cancer Center, Houston, Texas. Acknowledgment: The authors thank Mrs. Bobbie Coleman for her secretarial assistance. Address reprint requests to Dr. Sahin at the Department of Pathology, Box 85, M. D. Anderson Cancer Center. 1515 Holcombe Blvd., Houston, TX 77030. Accepted for publication September 20, 1990. CCC 0148-6403/91/040349-06 $04.00 0 1991 John Wiley 8, Sons, Inc.

Temporal Bone Metastases

of prostatic adenocarcinoma was initially seen in March 1989 with pain in the right temporal region, dizziness, and hearing loss, all of gradual onset. The prostatic cancer (stage B) had been diagnosed in 1986. Radiotherapy had yielded good clinical response, and there had been no evidence of disease progression until this representation. Physical examination at M. D. Anderson Cancer Center disclosed right-sided 7th and 8th cranial nerve palsies. On bone scan, there was increased uptake of radionuclide in the area of the right temporal bone and in the right posterior ribs, right patella, and dorsal spine. Audiometric studies showed no response to speech and pure tone stimuli at maximum output. Electromyelography and cranial nerve conduction studies were suggestive of a peripheral lesion involving the right 7th and 8th cranial nerves. Computed tomography and magnetic resonance imaging of the head revealed right temporal bone lesions with involvement of middle ear. The

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A 73-year-old white man was initially seen by his local physician in 1984 because of gradual-onset, right-sided ear pain, hearing loss, and tinnitus. A diagnosis of serous otitis media was made. The symptoms continued despite treatment, and the patient was referred to a local otolaryngology clinic, where physical examination revealed a polypoid, friable mass partially occluding the right external auditory canal. Computed tomography of the head showed expansile, osteolytic destruction of the right petrous temporal bone and associated soft tissue densities in the right middle ear. Biopsy revealed adenocarcinoma (Figure 3). A subsequent general physical examination was unremarkable except for the discovery of a hard prostatic nodule on digital rectal examination. Needle biopsy of this nodule showed prostatic adenocarcinoma (Figure 4) that histologically resembled that of the ear tumor. The patient was referred to M. D. Anderson Cancer Center for treatment. Bone scan at the M. D. Anderson Cancer Center showed osteoblastic densities in the spine and pelvis consistent with metastatic prostatic carcinoma. Immunohistochemical studies on the ear mass biopsy demonstrated positive staining for PAP and PSA in the tumor cells (Figure 5). The patient underwent bilateral orchiectomy and Case 2.

FIGURE 1. Case 1. Middle ear biopsy shows a glandular neoplasm infiltrating the dense stroma. The glands are lined by a single to pseudostratified layer of cuboidal cells (hematoxylineosin, x 100, original magnification).

serum prostate-specific antigen (PSA) level was 62.8 ng/mL (normal, 4.0-10.0 ng/mL). Biopsy of the middle ear mass revealed irregularly shaped and variably sized glands embedded in subepithelial connective tissue (Figure 1). The glands were composed of cuboidal to columnar cells with hyperchromatic nuclei and occasionally prominent nucleoli. Immunoperoxidase staining showed strong reactivity in the tumor cells for both prostatic acid phosphatase (PAP) and PSA (Figure 2),confirming the prostatic origin of this adenocarcinoma. The patient underwent systemic chemotherapy and irradiation of the temporal bone area. The temporal bone lesions were stabilized and remained stable as of latest follow-up in January 1990.

FIGURE 2. Case 1. lmmunoperoxidase staining for PSA on the ear tumor shows diffuse strong reactivity in the tumor cells. Note the negative staining of the overlying squamous epithelium (x 100, original magnification).

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FIGURE 3. Case 2. Ear biopsy shows small, irregular nests and clusters of tumor cells infiltrating into connective tissue and bone (hematoxylin-eosin, x250, original magnification). The insert is a higher magnificationof the tumor glands (hematoxylineosin, x400, original magnification).

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A

B FIGURE 4. Case 2. Focus of moderately differentiated adenocarcinoma in needle biopsy of prostate (hematoxylin-eosin; A, x250; B, x400, original magnifications).

was prescribed estrogens. He did well for 3 years, until metastases developed in several long bones. The long bone metastases progressed despite irradiation, and therapy with ketoconazole was begun. He was alive with stable disease as of the last follow-up in March 1990. DISCUSSION

Although direct invasion of the middle ear or temporal bone by neoplasms arising in the nasopharynx or parotid gland is not uncommon, distant metastasis to this region is rare and has been the subject of case reports since its first description by Hutchison in 1907.6-10 Some authors believe that the incidence is likely higher than the number of reported cases Since the symptoms of midwould dle ear or temporal bone metastasis may be less

FIGURE 5. Case 2. Diffuse cytoplasmic positivity in the tumor cells for PSA (immunoperoxidase, x400, original magnification).

Temporal Bone Metastases

debilitating than the other clinical problems of patients with multiorgan involvement, they may be clinically overlooked. Further, histologic examination of the temporal bone is not part of a routine postmortem examination, so autopsy studies might not reflect an accurate estimate of frequency. Berlinger et al. described several different patterns of metastatic involvement of the temporal bone by solid tumors: metastasis from a distant site through hematogenous or lymphatic dissemination, meningeal carcinomatosis, and leptomeningeal extension of an intracranial primary tumor.12 The middle ear and hence the temporal bone could conceivably be involved by any tumor that has spread t o the cervical nodes, The setting for this type of metastasis would typically be widespread tumor. However, isolated temporal bone metastasis is thought to represent hematogenous spread, since the sluggish blood flow in the bone-marrow-containing areas of the temporal bone (petrous portion) would provide a suitable environment for the deposit of tumor cells. Not surprisingly, isolated temporal bone metastasis has most frequently been found to arise from primary tumors that are known to have proclivity to spread t o bone: more than half of the cases have been referable to breast, lung, kidney, or prostate cancer, in that order. Jahn et al. examined 19 temporal bones from 11 patients who died of cancer that had metastasized, and they found the petrous apex, mastoid, middle ear, and external canal, in descending order, the most common sites for hematogenous spread in that area."

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Po

0

rn

I D

2CD

m

59

81

Coppola and Salanga. 19802'

Schrimpf et al., 198223

73

Right-sided ear pain, tinnitus, hearing loss

Dizziness, right-sided temporal pain, hearing loss

No (found on search)

Yes (3 yr prior, stage B, irradiation given)

Moderately differentiated adenocarcinoma

Dense sclerosis of mastoid bone, a defect in petrous bone

Osteolytic destructive mass in petrous bone with soft tissue component

Poorly differentiated adenocarcinoma with immunoreactivity for PAP and PSA Moderately differentiated adenocarcinoma with immunoreactivity for PAP and PSA

Poorly differentiated adenocarcinoma

Erosion of left temporal bone

Osteoblastic lesion in temporal bone with epidural extension

Poorly differentiated adenocarcinoma

NR

Destruction of apex of right pyramidal bone

Destructive lesion in petrous apex

NR

NR

Yes (4 yr prior, hormonal therapy given) No (4 mo later vertebral lesions developed, primary site found on search) No (primary site found on search) Yes (4 yr prior, well-differentiated, treated by TURP) No (1 yr later prostate biopsy revealed well-differentiated carcinoma)

Histologic findings of TBM

Radiologic findings of TBM

Prior history of prostatic carcinoma

Irradiation, chemotherapy, alive with stable disease 6 mo after TBM Hormonal therapy, alive 4 yr after TBM

Hormonal therapy, alive with stable disease 1 yr after TBM

Irradiation, alive with stable disease 1 yr after TBM

Hormonal therapy, died of tumor after 4 mo

Died of extensive metastatic prostatic carcinoma Irradiation. hormonal therapy: NR

Treatment/ follow-up

NR, not reported; PAP, prostatic acid phosphatase; PSA. prostate-specificantigen; TBM, temporal bone metastasis; TURP, transurethra/resection of the prostate.

Case 2

69

64

Applebaum and Dolsky, 1977"

Present Case 1

Ear pain

57

Helcl and Malec, 197320

Left-sided ear pain, preauricular tenderness, hearing loss Dizziness, right-sided ear pain, hearing loss

Generalized bone pain, vertigo, ataxia, left 8th nerve paralysis Tinnitus, hearing loss, temporomandibular joint pain

77

Janczewski and Fujita, 1972''

Source

Presenting symptorn(s)

Age (Yr)

Table 1. Clinicopathologic features in temporal bone metastasis from prostate carcinoma.

Temporal bone metastasis has evidenced itself in a variety of otologic symptoms, including otalgia, otorrhea, hearing loss, facial nerve paralysis, tinnitus, and aural mass. Most authors have emphasized the high incidence of facial nerve paralysis. Maddox noted that facial nerve paralysis with otalgia and preauricular swelling in a patient whose tympanic membrane appears to be unremarkable should raise the possibility of temporal bone involvement by tumor.7 The great majority of the published examples of temporal bone involvement were found in a setting of widespread metastatic disease or a history of cancer facilitating a straightforward diagnosis. However, in a few cases, like our second case, the presentation anteceded recognition of the primary tumor.13-16 The osseous metastatic propensity of prostatic carcinoma is well known, and it is not unusual in this disease for bone metastases to become symptomatic before there is any recognition of the primary 1esi0n.l~The most common osseous metastatic sites of prostatic carcinoma are the spine, sacrum, and pelvis.18 Of the 9 previously reported cases of metastasis to the temporal bone,4 a detailed clinical history is available for 5 (Table l).19-23 In 3 of the 5 patients, the initial prostatic cancer presentation was the symp-

toms related to the temporal bone metastasis. Because in most cases prostatic adenocarcinoma can still be controlled with hormonal treatment even after distant spread, it is crucial to identify the prostatic origin of a metastasis. Serum PAP and PSA values are elevated in a majority of prostatic cancer patients who present with bony metastases. However, for the roughly 20% to 30% of the patients who will not have serum elevation of one of these markers, it is particularly important that immunohistochemical studies for PAP and PSA be used on tissue sections to establish prostatic origin of a tumor.24 The application is also very important for the patients in whom symptoms of metastatic carcinoma precede diagnosis of the primary tumor. In both of our cases, 1 with a positive history and the other with antecedent presentation of the temporal bone metastasis, immunohistochemical stains for PAP and PSA confirmed the prostatic origin. Awareness that temporal bone involvement by prostate carcinoma occurs albeit apparently rarely, and that it may occur as a solitary metastasis or present before the primary tumor, should help diagnosticians usefully apply radiologic examinations and immunohistochemical studies for prostatic tumor markers in appropriate cases.

~

REFERENCES

1, Conley J. Cancer of the middle ear. Ann Otol 1965;74:555-572. 2. Friedman I, Osborn DA. Metastatic tumours in the ear, nose, and throat region. J Laryngol Otol 1965;79:576591. 3. Goodman M. Middle ear and mastoid neoplasms. Ann Otol Rhinol Laryngol 1971;80419-424. 4. Kobayashi K, Igarashi M, Ohashi K,McBride RA. Metastatic seminoma of the temporal bone. Arch Otolaryngol Head Neck Surg 1986;112102- 105. 5. Hutchison R. On suprarenal sarcoma in children with metastases in the skull. Q J Med 1907;1:33-38. 6. Adams GL, Paparella M, el-Fiky F. Primary and metastatic tumors of the temporal bone. Laryngoscope 1971;81:1273- 1285. 7. Maddox HE. Metastatic tumors of the temporal bone. Ann Otol Rhinol Laryngol 1967;76:149-165. 8. Schuknecht HF, Allam AF, Murakami Y. Pathology of secondary malignant tumors of the temporal bone. Ann Otol Rhinol Laryngol 1968;77:5-22. 9. Jorgensen M. Metastatic carcinoma of the temporal bone. J Laryngol Otol 1961;75:513-518. 10. Hill BA, Kohut RI. Metastatic adenocarcinoma of the temporal bone. Arch Otolaryngol 1976;102:568-571. 11. Jahn AF, Farkashidy J, Berman JM. Metastatic tumors in the temporal bone: a pathophysiologic study. J Otolaryngol 1979;8:85-95. 12. Berlinger NT, Koutroupas S, Adams G, Maisel R. Patterns of involvement of the temporal bone in metastatic

Temporal Bone Metastases

and systemic malignancy. Laryngoscope 1980;90:619627. 13. Bergstrom LV, Baker BB, Sando I. Sudden deafness and facial palsy from metastatic bronchogenic carcinoma. J Laryngol Otol 1977;91:787-793. 14. Igarashi M, Card GG, Johnson PE, Alfrod BR. Bilateral sudden hearing loss and metastatic pancreatic adenocarcinoma. Arch Otolaryngol 1979;105:196- 199. 15. Baruah AK. Secondary malignant tumours of the temporal bone. J Laryngol Otol 1970;84:1167-1168. 16. Zirul ED, Steinbaum FL, West G, Matthews AJ. Metastatic renal cell carcinoma presenting as a tumor of the external auditory canal. J A m Osteopath Assoc 1983;83:255-257. 17. Saitoh H, Hida M, Shimbo T, Nakamura K, Yamagata J, Satoh T. Metastatic patterns of prostatic cancer: correlation between sites and number of organs involved. Cancer 1984;543078-3084. 18. Sorbera RJ, Taylor RG. Carcinoma of the prostate metastatic to the oro-facial region and mandible. Br J Oral Surg 1966;4121-126. 19. Janczewski G, Fujita S. Przerzut gruczolakoraka gruczolu krokowego do kosci skroniowej. Otolaryngol Pol 1972;2681-85. 20. Helcl F, Malec R. Syndrome of the pontocerebellar angle caused by metastasis of prostate carcinoma into pyramidal part of petrosal bone. S b Ved Pr Lek Fak Karlovy Univenity Hradci Kralove 1973;16:315-318. 21. Applebaum EL, Dolsky RL. Metastatic adenocarcinoma

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to the temporal bone. Trans A m Acad Ophthalmol Otolaryngol 1977;84154- 158. 22. Coppola RJ, Salanga VD. Metastatic prostatic adenocarcinoma to the temporal bone. Neurology 1980;30:311315. 23. Schrimpf R,Karmody CS, Chasin WD, Carter B. Scleros-

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ing lesions of the temporal bone. Laryngoscope 1982;92:1116- 1119. 24. Tell DT, Khoury JM, Taylor HG, Veasey SP. Atypical metastasis from prostate cancer. Clinical utility of the immunoperoxidase technique for prostate specific antigen. JAMA 1985;253:3514-3575.

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Temporal bone involvement by prostatic adenocarcinoma: report of two cases and review of the literature.

Although axial bone and cranial metastases are common in patients with prostatic carcinoma, temporal bone involvement is rare. We report 2 cases of pr...
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