Teng-Chin Wang1*, Yi-Tsung Yang2, and Ming-Tsun Tsai3
1Department of Otolaryngology, Tainan Municipal Hospital, Tainan City, Taiwan
2Division of Hematology-Oncology, Department of Internal Medicine, National Taiwan University Hospital Hsin-Chu Branch, Taiwan
3Institute of Clinical Medicine, National Yang-Ming University, Taipei, Taiwan
Received: 11 May, 2016; Accepted: 13 June, 2016; Published: 14 June, 2016
Teng-chin Wang, MD, Department of Otolaryngology, Tainan Municipal Hospital, Tainan City, Taiwan; No.670, Chongde Rd., East Dist., Tainan City 701, Taiwan; Tel: +886-6-2609926; Fax: +886-6-2606351; E-mail:
Wang TC, Yang YT, Tsai MT (2016) Polymorphous Low-Grade Adenocarcinoma: A Rare Cause of Long-term Epistaxis. Arch Otolaryngol Rhinol 2(1): 027-029. DOI: 10.17352/2455-1759.000018
© 2015 Wang TC, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Polymorphous low grade adenocarcinoma; Minor salivary glands tumor; Periorbita; Lamina papyracea
Polymorphous low-grade adenocarcinoma (PLGA) of the paranasal sinus is an extremely rare disease. PLGA is a minor salivary gland neoplasm that occurs mostly in the junction of the soft and hard palate, followed by the buccal mucosa and upper lip. Only a few cases have been reported in the sinonasal area. PLGA generally follows a benign clinical course with frequent perineural invasion but low metastatic potential.
We report on a patient with PLGA in the ethmoid sinus, which presented as a nasal polyp with epistaxis for more than 2 years. The patient received surgical excision with adjuvant radiotherapy and chemotherapy because of perineural and lymphavascular invasion. One year after treatment, the patient was free of disease. PLGA of the paranasal sinus is an extremely rare disease. Epistaxis as the initial presentation is also uncommon. Local recurrences and distant metastasis are rare if complete excision is performed. Long-term follow-up is required.
Polymorphous low-grade adenocarcinoma (PLGA) of the paranasal sinus is an extremely rare malignancy. Being a malignant minor salivary gland neoplasm, PLGA has a predilection for intraoral sites . An extraoral tumor is rare, and nasal tumors constitute less than 1% of cases . Although the neoplasm exhibits an infiltrative grown pattern, which frequently results in perineural invasion, the prognosis is favorable. Local recurrences are uncommon, as are distant metastases . Surgical intervention is the primary option for treating PLGA. Radiotherapy and adjuvant chemotherapy have been used for treating the forms of the disease that are more advanced; however, there is no definite evidence of their advantages . We present the case of a patient with PLGA of the ethmoid sinus, manifesting as a long-term epistaxis.
A 52-year-old man presented with a 30-month history of intermittent left epistaxis combined with purulent nasal discharge. He had a history of coronary artery disease and took aspirin regularly. In addition, he had undergone the bilateral Caldwell–Luc surgery for benign maxillary sinus disease previously. He visited different clinics several times for his condition, but it was incorrectly diagnosed as chronic paranasal sinusitis or the side effect of an anticoagulant therapy. Because the treatments did not improve his condition, he visited our department seeking medical help. The physical examination was unremarkable, and no obvious lesions or bleeding over the common meatus, septum, or nasopharynx was observed. However, fiberoptic examination revealed scant crust accumulation inside the middle meatus, and an easily bleeding lesion was identified after removing the crust (Figure 1a). Biopsy was performed, and a histological analysis revealed a basal cell adenoma. However, malignant carcinoma was not ruled out. The patient underwent computed tomography (CT), which revealed enhanced soft tissue in the left ethmoid sinus and nasal cavity. No skull base invasion was observed. However, the left lamina papyracea was unremarkable, and there was no intraorbital destruction at diagnosis (Figure 1b).
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