a rare case of retroauricular warthin's tumor in a 46-year ...a rare case of retroauricular...

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CORRESPONDENCE A rare case of retroauricular Warthins tumor in a 46-year-old man Dear Editor, A 46-year-old Taiwanese man presented with a 4-month history of a slow-growing mass in the right retroauricular area (Figure 1). The asymptomatic, ill-dened, movable, soft, skin-colored subcu- taneous nodule had a mild central depression. Apart from a 20- year habit of smoking 20 cigarettes/day, his medical history was un- remarkable. An incisional biopsy under the clinical impression of an epidermal cyst was performed to conrm tumor diagnosis because of the large size of the mass. Results of a histopathological examination revealed hyperkera- tosis, acanthosis with marked dermal and subcutaneous brosis with a small upper portion of a cystic tumor at the deep surgical margin. The cystic tumor was lined by oncocytic epithelial cells with lymphoid and plasma cells in the stroma (Figure 2). The epithelial wall was composed partly of squamous epithelial cells and partly of columnar epithelial cells with mucinous cells. The patient was then referred to the Otolaryngology department where a computed tomography scan without contrast was ar- ranged. A 28-mm solitary mass lesion with interior cystic changes and mild heterogeneous enhancement was found in the inferior, supercial portion of the right parotid gland (Figure 3, arrow). A right partial parotidectomy with facial nerve dissection was per- formed. The cut surface of the 2.5-cm well-circumscribed nodule showed white rm cystic change. No recurrence was noted at 3- year follow-up. The histopathological ndings conrmed the diagnosis of a Warthins tumor (WT, which is descriptively referred to as papillary cystadenoma lymphomatosum). The WT was rst described by Aldred Scott Warthin in 1929. 1 The descriptive term for WT is papillary cystadenoma lympho- matosum. The WT is the second most common benign salivary gland tumor representing approximately 215% of all parotid tu- mors after pleomorphic adenomas. A WT is usually unilateral and almost exclusively originates in the parotid gland, which is wrapped around the mandibular ramus. Only approximately 10% of such tumors are bilateral. 2 Although cases of WT arising outside the ordinary region of parotid gland are rare, there have been reports of WT originating in the submandibular gland, larynx, lower lip, palate, and even in the buccal fold. 3 Most of these tumors occur in patients between the 5 th and 7 th decades of life, and there appears to be a predilection in males. Smoking has been strongly associated with the tumor with smokers hav- ing eight times more risk of developing the tumor than non- smokers. Although its pathogenesis remains unknown, many scholars favor the hypothesis that WT arises from salivary gland tissue entrapped within regional lymph nodes during embryogenesis. 1 Clinically, WT usually presents as a solitary well-dened, slow growing, spherical to ovoid painless mass in the mandibular area, although some patients experience mild to severe pain. The mass usually measures 24 cm in diameter, but a giant one has been pre- viously reported with a diameter of 10 cm. 4 While most cases were described as lateral neck swelling or masses, 1 periauricular intra- parotid masses have also been reported. 5 In our case, WT originated from the lower pole of supercial lobe in the retroauricular area. Three cases of WT associated with skin have been reported in English literature, and they were all presented as ulcerated tumors. 68 A histopathological examination of WT may show cysts that are lined by epithelial cells with oncocytic features extending as papillary structures. The presence of prominent lymphocytic inl- trates in addition to the papillary-proliferating, bilayered epithe- lial lining epithelium is the most distinguishing feature. The characteristic epithelium can help clinicians differentiate WT from mucoepidermoid carcinoma, acinic cell adenocarcinoma, and cystadenocarcinoma. Prior to the operation, complete bilat- eral screening of the head and neck is suggested because the tu- mor can be bilateral. Excision remains the best way to cure the tumor, but recurrence has been reported. Long-term follow-up is recommended in view of possible further occurrences of sali- vary and extrasalivary tumors. 9 This reinforces the importance Figure 1 A esh-colored subcutaneous nodule in the right retroauricular area. Contents lists available at SciVerse ScienceDirect Dermatologica Sinica journal homepage: http://www.derm-sinica.com DERMATOLOGICA SINICA 32 (2014) 124125 1027-8117/$ see front matter Copyright Ó 2013, Taiwanese Dermatological Association. Published by Elsevier Taiwan LLC. All rights reserved. http://dx.doi.org/10.1016/j.dsi.2013.05.007

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Page 1: A rare case of retroauricular Warthin's tumor in a 46-year ...A rare case of retroauricular Warthin’s tumor in a 46-year-old man Dear Editor, A46-year-oldTaiwanesemanpresentedwitha4-monthhistory

at SciVerse ScienceDirect

DERMATOLOGICA SINICA 32 (2014) 124–125

Contents lists available

Dermatologica Sinica

journal homepage: http: / /www.derm-sinica.com

CORRESPONDENCE

A rare case of retroauricular Warthin’s tumor in a 46-year-old man

Figure 1 A flesh-colored subcutaneous nodule in the right retroauricular area.

Dear Editor,

A 46-year-old Taiwanese man presented with a 4-month historyof a slow-growing mass in the right retroauricular area (Figure 1).The asymptomatic, ill-defined, movable, soft, skin-colored subcu-taneous nodule had a mild central depression. Apart from a 20-year habit of smoking 20 cigarettes/day, his medical historywas un-remarkable. An incisional biopsy under the clinical impression of anepidermal cyst was performed to confirm tumor diagnosis becauseof the large size of the mass.

Results of a histopathological examination revealed hyperkera-tosis, acanthosis with marked dermal and subcutaneous fibrosiswith a small upper portion of a cystic tumor at the deep surgicalmargin. The cystic tumor was lined by oncocytic epithelial cellswith lymphoid and plasma cells in the stroma (Figure 2). Theepithelial wall was composed partly of squamous epithelial cellsand partly of columnar epithelial cells with mucinous cells. Thepatient was then referred to the Otolaryngology departmentwhere a computed tomography scan without contrast was ar-ranged. A 28-mm solitary mass lesion with interior cystic changesand mild heterogeneous enhancement was found in the inferior,superficial portion of the right parotid gland (Figure 3, arrow). Aright partial parotidectomy with facial nerve dissection was per-formed. The cut surface of the 2.5-cm well-circumscribed noduleshowed white firm cystic change. No recurrence was noted at 3-year follow-up.

The histopathological findings confirmed the diagnosis of aWarthin’s tumor (WT, which is descriptively referred to as papillarycystadenoma lymphomatosum).

The WT was first described by Aldred Scott Warthin in 1929.1

The descriptive term for WT is papillary cystadenoma lympho-matosum. The WT is the second most common benign salivarygland tumor representing approximately 2–15% of all parotid tu-mors after pleomorphic adenomas. A WT is usually unilateraland almost exclusively originates in the parotid gland, which iswrapped around the mandibular ramus. Only approximately10% of such tumors are bilateral.2 Although cases of WT arisingoutside the ordinary region of parotid gland are rare, therehave been reports of WT originating in the submandibular gland,larynx, lower lip, palate, and even in the buccal fold.3 Most ofthese tumors occur in patients between the 5th and 7th decadesof life, and there appears to be a predilection in males. Smokinghas been strongly associated with the tumor with smokers hav-ing eight times more risk of developing the tumor than non-smokers. Although its pathogenesis remains unknown, many

1027-8117/$ – see front matter Copyright � 2013, Taiwanese Dermatological Associatiohttp://dx.doi.org/10.1016/j.dsi.2013.05.007

scholars favor the hypothesis that WT arises from salivary glandtissue entrapped within regional lymph nodes duringembryogenesis.1

Clinically, WT usually presents as a solitary well-defined, slowgrowing, spherical to ovoid painless mass in the mandibular area,although some patients experience mild to severe pain. The massusually measures 2–4 cm in diameter, but a giant one has been pre-viously reported with a diameter of 10 cm.4 While most cases weredescribed as lateral neck swelling or masses,1 periauricular intra-parotidmasses have also been reported.5 In our case,WToriginatedfrom the lower pole of superficial lobe in the retroauricular area.Three cases of WT associated with skin have been reported inEnglish literature, and they were all presented as ulceratedtumors.6–8

A histopathological examination of WT may show cysts thatare lined by epithelial cells with oncocytic features extending aspapillary structures. The presence of prominent lymphocytic infil-trates in addition to the papillary-proliferating, bilayered epithe-lial lining epithelium is the most distinguishing feature. Thecharacteristic epithelium can help clinicians differentiate WTfrom mucoepidermoid carcinoma, acinic cell adenocarcinoma,and cystadenocarcinoma. Prior to the operation, complete bilat-eral screening of the head and neck is suggested because the tu-mor can be bilateral. Excision remains the best way to cure thetumor, but recurrence has been reported. Long-term follow-upis recommended in view of possible further occurrences of sali-vary and extrasalivary tumors.9 This reinforces the importance

n. Published by Elsevier Taiwan LLC. All rights reserved.

Page 2: A rare case of retroauricular Warthin's tumor in a 46-year ...A rare case of retroauricular Warthin’s tumor in a 46-year-old man Dear Editor, A46-year-oldTaiwanesemanpresentedwitha4-monthhistory

Figure 2 (A) A cystic tumor lined by oncocytic epithelial cells with lymphoid and plasma cells infiltrating the stroma. (B) Epithelial cells with oncocytic features extending as papil-lary structures.

Figure 3 A noncontrast computed tomography in transverse section revealed the sol-itary mass lesion in the inferior, superficial portion of the right parotid gland, withinterior cystic change and mildly heterogeneous enhancement.

Correspondence / Dermatologica Sinica 32 (2014) 124–125 125

of accurate diagnoses of WT, especially in the retroauricular areas.We herein presented a case of exceptional WT in a middle-agedman as a pitfall for the differential diagnosis of a retroauricularsubcutaneous mass.

Han-Chi TsengDepartment of Dermatology, Chang Gung Memorial

Hospital-Kaohsiung Medical Center, Chang Gung University,College of Medicine, Kaohsiung, Taiwan

Jui-Hung KoDepartment of Dermatology, Chang Gung Memorial

Hospital at Taipei, Chang Gung University, College of Medicine,Taoyuan, Taiwan

Rosaline Chung-Yee Hui*Department of Dermatology, Chang Gung Memorial Hospital at Taipei,

Chang Gung University, College of Medicine,Taoyuan, Taiwan

*Corresponding author. Department of Dermatology, Chang GungMemorial Hospital, 199, Tun-Hwa North Road, Taipei 105, Taiwan.

Tel.: þ886 2 27135211x339; fax: þ886 2 27191623.E-mail addresses: [email protected], [email protected]

Conflicts of interest: The authors declare that they have no financial or nonfinancialconflicts of interest related to the subject matter or materials discussed in this article.

References

1. Teymoortash A, Krasnewicz Y, Werner JA. Clinical features of cystadenolym-phoma (Warthin’s tumor) of the parotid gland: a retrospective comparativestudy of 96 cases. Oral Oncol 2006;42:569–73.

2. Ascani G, Pieramici T, Rubini C, Messi M, Balercia P. Synchronous bilateral War-thin’s tumours of the parotid glands: a case report. Acta Otorhinolaryngol Ital2010;30:310–2.

3. Shah N, Tighe JV, Barrett AW, Kumar S, Allen JP. Bilateral intraparotid and extrap-arotid Warthin’s tumours. Br J Oral Maxillofac Surg 2007;45:238–9.

4. Fernández-Barriales M, García-Reija MF, Saiz-Bustillo RC. Giant Warthin tumour.Acta Otorrinolaringol Esp 2011;62:477–8. [Article in Spanish].

5. Lamont JP, McCarty TM, Fisher TL, Kuhn JA. Prospective evaluation of office-based parotid ultrasound. Ann Surg Oncol 2001;8:720–2.

6. Stárek I, Tichý T, Skýpalová P, Skálová A. Ulcerating Warthin’s tumour. ActaOtolaryngol 2004;124:744–6.

7. Abraham Z, Rozenbaum M, Keren R. Skin ulcer at the blunt apex of a giant War-thin’s tumor. J Dermatol 2000;27:523–8.

8. Sadahira C, Matsuota Y, Numahara T, Miyabe K, Mori N, Kubota Y. A case ofulcerative Warthin’s tumor. J Dermatol 2004;31:779–81.

9. Maiorano E, Lo Muzio L, Favia G, Piattelli A. Warthin’s tumour: a study of 78cases with emphasis on bilaterality, multifocality and association with othermalignancies. Oral Oncol 2002;38:35–40.

Received: Nov 30, 2012Revised: May 26, 2013

Accepted: May 29, 2013