Nasal Septum Adenoid Cystic Carcinoma: a Case Report

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provided by Elsevier - Publisher Connector NASAL SEPTUM ADENOID CYSTIC CARCINOMA: A CASE REPORT

Shu-Yu Tai,1 Chen-Yu Chien,2 Chih-Feng Tai,2,4 Wen-Rei Kuo,2,4 Wan-Ting Huang,3 and Ling-Feng Wang2,4 Departments of 1Family Medicine, 2Otolaryngology–Head and Neck Surgery, and 3Pathology, Kaohsiung Medical University Hospital, and 4Department of Otolaryngology–Head and Neck Surgery, Faculty of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan.

Adenoid cystic carcinoma of the nasal septum is extremely rare. We present the case of a 56-year- old male who complained of nasal bleeding and nasal obstruction for 1 month. A mass arising from nasal septum was found by endoscope. The tumor was removed under lateral rhinotomy and histopathologic examination revealed adenoid cystic carcinoma with cribriform pattern. He then had postoperative radiotherapy. No recurrence was noticed after 1 year of follow-up. Despite its rarity, adenoid cystic carcinoma should be taken into consideration in the differential diagnosis of nasal tumor.

Key Words: adenoid cystic carcinoma, nasal septum (Kaohsiung J Med Sci 2007;23:426–30)

Sinonasal cancers comprise approximately 3% of all nasal obstruction, but negative nasal discharge, nasal the upper aerodigestive tract malignancies and less pain, or facial numbness. Nasal endoscopy revealed than 1% of all cancers. The incidence of malignant a well-defined, smooth, non-ulcerative mass arising tumors of the nasal septum is very low, comprising from the nasal septum (Figure 1A and B). It had elastic 2.7–8.4% of nasal and paranasal malignant tumors [1]. consistency on palpation. It had no contact with sinus Of all malignant paranasal sinus tumors, 5–15% are or turbinate. Computed tomography scan revealed a adenoid cystic carcinomas [2]. In the literature to date nasal septum tumor with septum destruction (Figure only six cases of adenoid cystic carcinoma arising from 1C and D). No cervical lymphadenopathy was present. the nasal septum have been reported [1,3–5]. In this Punch biopsy was done and the pathology report paper, we present our experience of a case of nasal revealed adenoid cystic carcinoma. Chest X-ray, bone septal adenoid cystic carcinoma. scan, and abdominal echo revealed no lung, bone, or liver metastasis. We performed lateral rhinotomy to excise the nasal septum tumor mass under general CASE PRESENTATION anesthesia. The tumor was removed by through and through septum total excision without preserving A 56-year-old male who had suffered from epistaxis opposite mucoperichondrium and nasal mucosa. The for 1 month visited our clinic. He also complained of tumor measured 4.0 × 3.0 × 0.5 cm in size (Figure 2A and B). Histologically, the tumor was relatively well circumscribed beneath the respiratory epithelium and Received: January 17, 2007 Accepted: March 27, 2007 comprised epithelial and myoepithelial cells arranged Address correspondence and reprint requests to: Dr Ling-Feng in tubular, cribriform, and solid patterns (Figure 2C Wang, Department of Otolaryngology–Head and Neck Surgery, and D). The microcystic spaces of cribriform pattern Kaohsiung Medical University Hospital, 100 Tzyou 1st Road, Kaohsiung 807, Taiwan. were filled with hyaline mucoid material. Unfortu- E-mail: [email protected] nately, the pathology did not reveal free superior and

C D

Figure 1. Nasal endoscopy reveals a well-defined, smooth, non-ulcerative mass arising from the (A) right, and (B) left nasal septum. Computed tomography of the nose and paranasal sinuses reveals a nasal septum tumor with septum destruction: (C) axial view; (D) coronal view. anterior margins. Neither perivascular nor perineural obstruction, epistaxis and symptoms depending on permeation was noted. Postoperative radiotherapy which structure has been invaded. Nasal bleeding with a dose of 7,400 cGy was administered to enforce and obstruction were the only presenting symptoms locoregional control. The patient recovered unevent- in our patient. The smooth bulging appearance of fully after operation and radiotherapy. No signs of this septal tumor could be mistaken for a high septal local recurrence and distal metastasis were noted deviation (Figure 1A and B). Palpation of its consis- after 1 year of follow-up. tency might be helpful in differential diagnosis. The differential diagnosis of a nasal septum tumor includes a wide variety of pathology including squa- DISCUSSION mous cell carcinoma, malignant melanoma, adenoid cystic carcinoma, adenocarcinoma, chondroma, chon- Adenoid cystic carcinoma in the nasal cavity and drosarcoma, osteosarcoma, schwannoma, lymphoma, paranasal sinuses origin often has a worse prognosis and mucoepidermoid carcinoma. A biopsy for definite than in any other area of the head and neck region. It diagnosis is necessary. is reported to occur in any age group with a peak The slow-growing adenoid cystic carcinoma can incidence in the fourth to sixth decades. Its present- reach large dimensions within the hollow nasal ing symptoms are usually nonspecific, such as nasal and sinus cavities before becoming symptomatic.

Kaohsiung J Med Sci August 2007 • Vol 23 • No 8 427 S.Y. Tai, C.Y. Chien, C.F. Tai, et al

A B

C D

Figure 2. The nasal septum tumor is covered by intact nasal mucosa bilaterally. The tumor size is 4.0 × 3.0 × 0.5 cm in the (A) right and (B) left sides of the nasal septum. (C) Pathology reveals cribriform variant of adenoid cystic carcinoma (hematoxylin and eosin; original magnification, 40×). (D) Adenoid cystic carcinoma with cribriform subtype (hematoxylin and eosin; original magnification, 400×).

Furthermore, the close relationship of nose and sinus Three histologic growth patterns have been iden- with surrounding vital structures, including the dura, tified and described: solid, cribriform, and tubular. brain, orbit, carotid arteries, and cranial nerves may Cribriform is the most common histologic subtype. result in an inadequate or high morbidity surgical Assessment of the histologic grade is of significance resection. in predicting the likelihood of tumor recurrence and Perineural invasion along cranial nerves is a survival. In one series of studies, 5-year recurrence rates pathognomonic factor of adenoid cystic carcinoma of 100%, 89%, and 59% were reported for tumors with and is believed to be responsible for the high rate solid, cribriform, or tubular growth patterns, respec- of local recurrence. Neck lymph node metastases are tively [6]. Similarly, the presence of greater than 30% extremely rare with adenoid cystic carcinoma. The solid growth has been reported to have a significantly lung is the most common site of metastasis and the poorer 5-year survival (5%) when compared to tumors less common sites include the bone, liver, brain, and with a predominantly cribriform (26%) or tubular (39%) kidney. Adenoid cystic carcinoma has the tendency growth pattern [3]. to spread hematogenously and perineurally but not The surgical approach is modified for the individ- lymphatically. ual tumor according to the tumor size and location.

428 Kaohsiung J Med Sci August 2007 • Vol 23 • No 8 Nasal septum adenoid cystic carcinoma

Endoscopic approach can remove the small and local- REFERENCES ized nasal septum tumor. Lateral rhinotomy incision alone can excise anterior septal tumors. Midfacial 1. Handa T, Yamamoto H, Yamakawa J, et al. A case degloving can approach the lower nasal septum tumor report of adenoid cystic carcinoma of the nasal septum. Nippon Jibiinkoka Gakkai Kaiho 1992;95:505–9. without cosmetic deformity. Additional exposure like 2. Wiseman SM, Popat SR, Rigual NR, et al. Adenoid cys- lateral rhinotomy with sublabial incision may be tic carcinoma of the paranasal sinuses or nasal cavity: required to resect posterior nasal septal lesions. Lateral a 40-year review of 35 cases. Ear Nose Throat J 2002; rhinotomy with lip-splitting incision can resect lesions 81:510–4, 6–7. of the nasal septum and floor. 3. Sivaji N, Basavaraj S, Stewart W, et al. Adenoid cystic A combination of radical surgery and postoperative carcinoma of the nasal septum. Rhinology 2003;41: 253–4. radiotherapy was the main therapy for sinonasal ade- 4. Schneiderman TA, Chaudhury SI. Adenoid cystic car- noid cystic carcinoma compared to either surgery or cinoma of the nasal septum. Otolaryngol Head Neck radiotherapy alone. But despite aggressive surgery, Surg 2002;127:251–2. high incidence of positive margins was noted due to 5. Howard DJ, Lund VJ. Reflections on the management the anatomical complexity of the nose and paranasal of adenoid cystic carcinoma of the nasal cavity and sinuses. So adjuvant radiotherapy is necessary in such paranasal sinuses. Otolaryngol Head Neck Surg 1985;93: 338–41. cases [2]. Chemotherapy appears to be ineffective in 6. Perzin KH, Gullane P, Clairmont AC. Adenoid cystic the treatment of adenoid cystic carcinoma. Long-term carcinomas arising in salivary glands: a correlation of follow-up is necessary because of the high incidence histologic features and clinical course. Cancer 1978;42: of local recurrence and distal metastasis. 265–82.

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