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Benign Lymphoepithelial Cysts of the Parotid: Long-Term Surgical Results

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Benign Lymphoepithelial Cysts of the Parotid: Long-Term Surgical Results HIV/AIDS - Research and Palliative Care Dovepress open access to scientific and medical research Open Access Full Text Article CommentarY Benign lymphoepithelial cysts of the parotid: long-term surgical results Matthew K Steehler1 Abstract: Benign lymphoepithelial cysts are a widely recognized cause of parotid gland Mark W Steehler2–4 swelling in patients infected with the human immunodeficiency virus (HIV). These cysts are Steven P Davison1,5 pathognomonic for HIV. The cysts frequently grow to be exceptionally large, causing physical deformity and gross asymmetry of facial contour. This clinical commentary analyzes this 1Georgetown University Hospital Department of Otolaryngology cosmetically deforming disease entity and the many treatments that accompany it. The patient Head and Neck surgery, Washington, presented in this paper is a surgical case-control. The case is a microcosm for our findings upon 2 DC, USA; Lake Erie Consortium review of the literature. Treatment options for benign lymphoepithelial cysts include repeated for Osteopathic Medical Training, 3Millcreek Community Hospital, fine-needle aspiration and drainage, surgery, radiotherapy, sclerotherapy, and conservative 4Ear Nose and Throat Surgeons therapy, with institution of highly active antiretroviral therapy medication. Based on this surgical of Northwest Pennsylvania, Erie, PA, USA; 5DAVinci Plastic Surgery, case-control and our review of the literature, it is concluded that surgical intervention offers the Washington, DC, USA best cosmetic result for these patients. Keywords: parotid, benign lymphoepithelial cyst, parotidectomy, human immunodeficiency virus, head and neck surgery, cosmetic surgery Introduction A 32-year-old female with human immunodeficiency virus (HIV) infection presented to our office six years after we had performed a right superficial parotidectomy for a 10 cm × 8 cm benign lymphoepithelial cyst (BLEC). This was cured surgically with excellent cosmetic outcome. The patient presented at this time with a similarly large lesion on the contralateral side which was removed in a similar fashion (see Figure 1). The patient tolerated the procedure well with no postoperative morbidity, normal facial nerve function, and a well healed scar. The 6-year follow-up on this patient represents a case control study demonstrating the efficacy of parotidectomy in the treatment of BLEC. The only indication for removal of BLEC is when there is an expanding lesion distorting the patient’s facial appearance. In order to delve further into the best treatment for this cosmetic problem, a Medline search and review of the literature on BLEC was performed. To the best of the authors’ knowledge, this paper is the first to review the etiology, diagnosis, and Correspondence: Matthew K Steehler Department of Otolaryngology, Head and treatment options for BLEC. Neck Surgery, Georgetown University Hospital, 1st Floor Gorman Building, 3800 Reservoir Drive, Washington, Literature review DC 20007, USA Benign lymphoepithelial cysts (BLECs) are a widely recognized cause of parotid Tel +1 814 450 0742 gland swelling in HIV-infected patients. Although they are neither invasive nor Fax +1 202 444 1312 Email [email protected] associated with malignant degeneration, BLECs can become large and disfiguring. submit your manuscript | www.dovepress.com HIV/AIDS - Research and Palliative Care 2012:4 81–86 81 Dovepress © 2012 Steehler et al, publisher and licensee Dove Medical Press Ltd. This is an Open Access article http://dx.doi.org/10.2147/HIV.S27755 which permits unrestricted noncommercial use, provided the original work is properly cited. Steehler et al Dovepress of BLEC is the “obstructive theory” which states that lymphoid proliferation in the parotid gland leads to ductal obstruction and salivary dilation that mimics a true cyst.1,3,4,7–9 The second hypothesized theory is that HIV-related reactive lymphoproliferation occurs in the lymph nodes of the parotid gland. The parotid glandular epithelium becomes trapped in normal intraparotid lymph nodes, resulting in cystic enlargement.1,7,8 Diagnosis of BLEC is made based on history, physical examination, and fine-needle aspiration biopsy. BLEC is nearly diagnostic for HIV.1,2 Physical examination will show obvious fluctuant cystic facial deformity. Fine-needle Figure 1 Patient with HIV BLEC of the left parotid gland. aspiration biopsy of BLEC results in clear, proteinaceous Notes: Patient is status postsuperficial parotidectomy on the right side 6 years ago (complete resolution). Patient routinely wears multiple accessories daily to distract (straw-colored) fluid, with a mixture of epithelial (squamous) others from her obvious facial lesion (32-year-old patient, May 2010). and benign lymphoid cells.7,10 This combination of history, Abbreviation: BLEC, benign lymphoepithelial cyst. physical examination, and fine-needle aspiration biopsy is diagnostic for BLEC.3,10 These lesions can be a cosmetically devastating physical Treatment of this particular pathology has been widely deformity, causing isolation and depression.1–6 They are debated in the literature. Previous treatments for BLEC easily recognized as a manifestation of HIV in the aware have included repeated fine-needle aspiration and drain- population. age, surgery, radiotherapy, sclerotherapy, and conservative BLEC of the parotid gland is the most common salivary therapy with institution of highly active antiretroviral gland pathology in HIV.1,2 BLEC has two hypothesized therapy (HAART) medications.2–6,10–22 Table 1 summarizes etiologic mechanisms. The first theorized pathogenesis the advantages and disadvantages, duration of procedures, Table 1 Advantages and disadvantages of treatment for benign lymphoepithelial cysts of the parotid gland as well as treatment duration and cost of treatment Advantages Disadvantages Length of Estimated cost of treatment treatment HAART therapy • Noninvasive • Incomplete response $3 months $1250/month • Standard of care for HIV • Partial response FNA drainage • Minimally invasive • 100% recurrence rate #2×/month $450/procedure • Avoids radiation • Multiple treatments Radiation therapy • Noninvasive • Therapeutic doses of radiation (24 Gray) 3 weeks $17,600 total • Avoids needle stick injuries • Side effects include xerostomia, skin necrosis, mucositis • Incomplete response Sclerotherapy • Minimally invasive • Multiple treatments 1–4 procedures $450/procedure • Avoids radiation • Varying degrees of drug toxicities ranging Bleomycin, $1450/ from rash to pulmonary fibrosis procedure • Incomplete response OK-432, orphan drug, • Fibrosis makes future surgery more difficult not approved by the FDA in the US Surgery • Complete response • General anesthesia One surgery $8000 total • Cosmetically superior • 23-hour observational stay in hospital • Avoids radiation • 2–6-hour invasive procedure • Surgical scar • Surgical complications hematoma (7%), facial nerve injury (2.3%–6%), Frey’s syndrome (5%), recurrence (2%)15,16,26,27 Abbreviations: FNA, fine-needle aspiration; HAART, highly active antiretroviral therapy; HIV, human immunodeficiency virus. 82 submit your manuscript | www.dovepress.com HIV/AIDS - Research and Palliative Care 2012:4 Dovepress Dove HIV/AIDS - Research and Palliative Care 2012:4 Care and Palliative HIV/AIDS -Research press Table 2 Treatment of benign lymphoepithelial cysts: review of the literature Resource Modality Amount/drug Design Sample Follow-up Results Syebele and HAART Stavidune Retrospective 10 patients 3 months 90% of patients with “significant response” Bütow2 Lamivudine Efavirenz Monama and Sclerotherapy Bleomycin Retrospective 3 patients 12–15 months 3/3 resolution Tshifularo6 (180–270 U) Heran and Sclerotherapy OK-432 (0.25 KE, 0.5 KE) Retrospective 1 patient 12 months 1/1 “No clinically significant recurrence” Legiehn21 Meyer et al12 Sclerotherapy Alcohol (1/2 volume of cyst) Unblinded Prospective 11 patients 5.7 months 10/11 patients “happy” with results Marcus and Sclerotherapy Sodium morrhuate Retrospective 4 patients 2.5 months 6/6 reduction in size Moore4 (50 mg/mL to 3 mL) (6 cysts) 1/6 complete response Berg and Moore22 Sclerotherapy Sodium morrhuate Retrospective 9 patients Not specified 5/9 additional cyst formation (50 mg/mL, 4 mL) 4/9 compete response Lustig et al17 Sclerotherapy Doxycycline Prospective 8 patients 12–17 months 7/9 reduction in size (1 mg/mL, 2 mL) (9 cysts) 2/9 complete response Echavez et al14 Sclerotherapy Tetracycline Retrospective 3 patients 14–36 months 3/3 resolution (250 mg/2 mL) Beitler et al3 Radiotherapy 24 Gray Retrospective 19 patients 23 months 13/19 with adequate cosmetic control Beitler et al18 Radiotherapy 8–10 Gray Retrospective 12 patients 9.5 months 1/12 with adequate cosmetic control Goldstein et al19 Radiotherapy 8–10 Gray Retrospective 8 patients One month 5/8 complete response 3/8 partial response submit your manuscript manuscript submit your Shaha et al15 Surgery Superficial parotidectomy Retrospective 35 patients Up to 6 years Complete response Ferraro et al16 Surgery Enucleation Retrospective 10 patients 3–36 months One recurrence Benign lymphoepithelial parotid cysts parotid Benign lymphoepithelial 20 cysts 19 complete response Note: Cost data based on clinical/hospital data pooled from respective authors’ places of practice based on unilateral parotid lesion. | www.dovepress.co Dove press m 83 Steehler et al Dovepress and the cost of treatment for BLEC. A literature review of treatment for their cysts.13 In two studies evaluating sodium the aforementioned
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