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Turkish Journal of Cancer
2005, Volume 35, Number 2, Page(s) 093-095
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Delayed pulmonary metastasis of adenoid cystic carcinoma originated from external earway: Case report
ŞEREF KÖMÜRCÜ1, AHMET ÖZET1, MÜKERREM SAFALI2, ORHAN TÜRKEN1, BEKİR ÖZTÜRK1, FİKRET ARPACI1, BÜLENT YALÇIN1
1Gülhane Military Medical Academy, Departments of Medical Oncology, Ankara-Turkey
2Gülhane Military Medical Academy, Departments of Medical Pathology, Ankara-Turkey
Keywords: External earway, adenoid cystic carcinoma, pulmonary metastases, delayed metastases
Summary
Pulmonary metastases in a 68-year old male was detected 20 years after the operation for adenoid cystic carcinoma originated from external earway. Adenoid cystic carcinoma metastases was confirmed by fine-needle aspiration biopsy from multiple mass lesions in right chest. The pulmonary metastasis of adenoid cystic carcinoma emerging from external earway after such a long period is a rare case in the literature.[Turk J Cancer 2005;35(2):93-95].
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  • Summary
  • Introduction
  • Case Presentation
  • Disscussion
  • References
  • Introduction
    Adenoid cystic carcinoma of external earway originating from sweat glands is a rare tumor. The tumor generally has a benign clinical trend and emerges as crust verrucose discs or deep seated nodules. Detecting of dermal nodules and consisting of cystic area with solid fields is the main histological pattern. It has been reported that the presence of excessive solid fields is correlated with poor prognosis [1]. Multiple organ metastasis may be detected in aggressive tumors [2]. Although there is no consensus on treatment, surgery and postoperative radiotherapy might be preferred according to the degree of tumor invasion. It has been reported that the five year survival rate with the treatment is 61%, however this rate can be modified by the factors like histological grade, degree of invasion, and tumor existence in surgical margins [3]. A rare case with delayed pulmonary metastasis who had been operated 20 years ago due to adenoid cystic carcinoma of external earway was reported here.
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  • Case Presentation
    Multiple metastatic nodules were seen in right hemithorax on the X-ray taken for determining the etiology of thoracal pain in a 68-year old male. The patient underwent radical external earway excision + partial parotidectomy + radical neck dissection (there was not malignant involvement in parotid gland or lymph nodes) for adenoid cystic carcinoma of external earway 20 years ago (Figure 1). He had also a history of anterior resection 10 years ago due to rectal cancer. In thoracal CT, multiple metastatic nodules were seen in right hemithorax at different size and localization (Figure 2). There was no mass lesion on bronchoscopy. Adenoid cystic carcinoma was confirmed by CT guided fine-needle aspiration biopsy (Figure 3). There was not another organ involvement in screening. Chemotherapy was started to the patient for metastatic adenoid cystic carcinoma. Disease remained stabilized after six cycles of gemcitabine (1000 mg/m2 on days 1,8,15) and 5-fluorouracil (325 mg/m2 for 5 days). The patient died because of acute myocardial infarction in his one year follow-up.

    Fig 1: Adenoid cystic carcinoma invading cartilage. Tumor composed of nests and columns of cells of uniform appearance, arranged concentrically around gland like spaces containing hyaline material (H&E, x50)

    Fig 2: Thoracal CT shows multiple metastatic nodules in right hemithorax

    Fig 3: Smear of aspirate from the lung lesion shows monotonous tumor cells making spherical clusters with hyaline globules in the center of some clusters, consistent with metastatic adenoid cystic carcinoma (H&E, x200)

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  • Introduction
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  • Disscussion
  • References
  • Discussion
    Adenoid cystic carcinoma which is frequently localized on the scalp is a rare tumor in external earway [4]. This tumor usually grows slowly. Some factors including tumor positivity at surgical margins, parotid gland and adjacent bone involvement, perineural invasion and local recurrences are associated with aggressiveness and high mortality rate [5]. When relapse develops, it generally occurs within two years. Late relapses were rarely reported as much as 14 years [5]. In our case, pulmonary metastasis was detected 20 years after treatment, this is the longest period for metastases in the literature for adenoid cystic carcinoma. Rarely widespread visceral involvement may be seen [6]. The performance status of patient and metastasis are important factors for selecting the treatment modality. The survival with surgery and postoperative radiotherapy is better than that with surgery alone [7]. The degree of local invasiveness is important for planning surgical procedure. The modified temporal bone resection with radical excision of external earway has been reported as an effective treatment for these patients [4]. Similar results were reported with step-wise surgery instead of en-bloc temporal bone and skull base resection [8]. In tumors which were thought to be aggressive (invaded to the adjacent tissue, higher grade or impossible to get negative surgical margins) elective neck lymph node dissection could be added to surgery. Primary radical excision of tumor with lymph node dissection was performed in our case. The wide excisional surgery should be recommended for the presence of invasion to the adjacent tissues (e.g. inner ear, temporal bone). Chemotherapy with known limited effect on metastatic adenoid cystic carcinoma patients, can be used for treatment. About 20% partial response was reported with weekly vinorelbine (30mg/m2, iv) at salivary gland origin [9]. In a patient with lung and liver metastasis, more than 50% regression was reported with docetaxel (100 mg/m2, iv, per 21 days) [10]. Interestingly, one patient with tumor originated from parotid gland and relapsed after primary therapy (surgery + radiotherapy) a partial response was detected with tamoxifen alone [11]. We treated our patient with gemcitabine + 5-fluorouracil regimen. After six cycles of chemotherapy, stable disease was seen on control CT imaging.

    In conclusion, our case with adenoid cystic carcinoma is the one with pulmonary metastases in the longest period in English literature.

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  • References

    1) Aikawa H, Tomonari K, Okino Y, et al. Adenoid cystic carcinoma of the external auditory canal: Correlation between histological features and MRI appearances. Br J Radiol 1997;70;530-2.

    2) Wassef M, Thomas V, Deffrennes D, et al. Primary adenoid cystic carcinoma of the skin. Histologic and ultrastructural study of two cases localized in the external auditory canal. Ann Pathol 1995;15;150-5.

    3) Pfreundner L, Schwager K, Willner J, et al. Carcinoma of the external auditory canal and middle ear. Int J Radiat Oncol Biol Phys 1999;1;777-88.

    4) Fliss DM, Kraus M, Tovi F. Adenoid cystic carcinoma of the external auditory canal. Ear Nose Throat J 1990;69;635, 638-9.

    5) Perzin KH, Gullane P, Conley J. Adenoid cystic carcinoma involving the external auditory canal. A clinicopathologic study of 16 cases. Cancer 1982;15:2873-83.

    6) Matthew RM, Subhashini J, Ponnaiya J, et al. Adenoid cystic carcinoma of the external auditory canal with pulmonary, renal and liver metastases. Indian J Cancer 1997;34;139-42.

    7) Hahn SS, Kim JA, Goodchild N, et al. Carcinoma of the middle ear and external auditory canal. Int J Radiat Oncol Biol Phys 1983;9;1003-7.

    8) Kinney SE, Wood BG: Malignancies of the external ear canal and temporal bone: surgical techniques and results: Laryngoscope 1987;97;158-64.

    9) Airoldi M, Bumma C, Bertetto O, et al. Vinorelbine treatment of recurrent salivary gland carcinomas. Bull Cancer 1998;85;892-4.

    10) Belli F, Di Lauro L, Zappanico A, et al. Docetaxel in the treatment of metastatic carcinoma of the salivary glands: Report of a case. Clin Ther 1999;150;77-9.

    11) Shadaba A, Gaze MN, Grant HR. The response of adenoid cystic carcinoma to tamoxifen. J Laryngol Otol 1997;111;1186-9.

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