SURGICAL ONCOLOGY AND RECONSTRUCTION

A Case of Squamous Cell Carcinoma Arising From Branchial Cleft Cyst Asuka Nakano, DDS,* Katsuaki Mishima, DDS, PhD,y Naoki Katase, DDS, PhD,z and Yoshiya Ueyama, DDS, PhDx Squamous cell carcinoma throughout the epithelium of a lateral cervical cyst is considered extremely rare. This report describes an additional case of this very rare clinical condition. A 70-year-old man presented with a well-defined, immobile, painless mass in the left neck that was excised with a diagnosis of branchial cyst. Histologic findings of the excised specimen were lateral branchial cyst with high-grade dysplasia and carcinoma of the squamous epithelial lining. The patient was followed for more than 2 years 10 months and no evidence of recurrence or other cancer has been found. Ó 2015 American Association of Oral and Maxillofacial Surgeons J Oral Maxillofac Surg 73:781-785, 2015

Report of Case

Branchiogenic carcinoma in which lymphoepithelial tissues undergo malignant transformation was first reported by Volkman in 1882.1 Branchiogenic carcinoma is rare and the existence of primary branchiogenic carcinomas remains controversial.2-4 The most common cause of cervical cystic squamous cell carcinoma is secondary cancer metastasis from malignant lesions arising in the oral cavity or other areas.2-4 Many reports have failed to provide sufficient evidence to distinguish this lesion from nodal metastasis arising from unrecognized primary tumors. The most important criteria for histopathologic diagnosis of a primary branchiogenic carcinoma would be proof of the occurrence of lesions within a lateral branchial cyst, transition from a normal to a malignant epithelium, and evidence of premalignant epithelial changes within the lateral cervical cyst.2,5 The present case also satisfied these histologic conditions in that histologic progression from chronic inflammation to dysplasia to carcinoma was found in a branchial cyst.

A 70-year-old man was referred to the authors’ department in August 2010 with a left neck mass of 6-month duration. A 450-mm mass was palpable at the anterior margin of the sternocleidomastoid muscle in the left neck. The overlying skin was normal, and the mass was elastic, hard, immobile, and painless (Fig 1). Magnetic resonance imaging showed high signal intensity accompanied by a low signal intensity septum on T2-weighted images at the level II zone, at the anterior margin of the sternocleidomastoid muscle, posterior to the submandibular gland, and lateral to the great vessels (Fig 2). This finding strongly suggested a cervical cystic lesion. General hematologic and biochemical tests showed no abnormalities. The patient also was diagnosed with prostate cancer at approximately the same time and received hormone therapy. Fine-needle aspiration cytology was performed and a pale-yellow, semitransparent, cloudy liquid was confirmed. Cytodiagnosis was Class I.

*Senior Resident, Department of Oral and Maxillofacial Surgery,

Address correspondence and reprint requests to Dr Mishima:

Yamaguchi University Graduate School of Medicine, Yamaguchi,

Department of Oral and Maxillofacial Surgery, Yamaguchi University

Japan.

Graduate School of Medicine, 1-1-1 Minamikogushi, Ube, Yamaguchi

yAssociate Professor, Department of Oral and Maxillofacial

755-8505, Japan; e-mail: [email protected]

Surgery, Yamaguchi University Graduate School of Medicine, Yamaguchi, Japan. zAssistant

Professor,

Department

of

Molecular

Received August 26 2014 Accepted October 29 2014 Ó 2015 American Association of Oral and Maxillofacial Surgeons

and

Developmental Biology, Kawasaki Medical School, Okayama, Japan.

0278-2391/14/01709-1

xProfessor and Chair, Department of Oral and Maxillofacial

http://dx.doi.org/10.1016/j.joms.2014.10.036

Surgery, Yamaguchi University Graduate School of Medicine, Yamaguchi, Japan.

781

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FIGURE 1. Extraoral findings at first examination. Clinical examination showed a 450-mm, well-defined, immobile, painless mass at the anterior border of the sternomastoid muscle and at the level II zone. Nakano et al. Squamous Cell Carcinoma From Branchial Cyst. J Oral Maxillofac Surg 2015.

SQUAMOUS CELL CARCINOMA FROM BRANCHIAL CYST

These clinical findings suggested a lateral cervical cyst. Surgery was performed under general anesthesia in December 2010. A horizontal incision 5 to 6 cm below the horizontal portion of the mandible was made through the superficial fascia and the platysma. The cyst was carefully separated from the anterior and inner aspects of the sternocleidomastoid muscle. The cystic legion was easily dissected from the surrounding tissue. The spinal accessory nerve, facial vein, and hypoglossus nerve were preserved. The existence of a duct was not clear. The excised specimen was an elastic cystic structure (Fig 3A). Maximum length and transverse diameter were 48 and 30 mm, respectively (Fig 3B). The cut surfaces showed a cystic structure that contained a yellowish-brown fluid. This case showed the features of a branchial cyst. Thus, the surgical specimen was a cystic lesion composed of a fibrous connective tissue wall that

FIGURE 2. Computed tomogram of mass. Computed tomography indicated a hypodense, homogeneous cyst located lateral to the great vessels, anterior to the sternocleidomastoid muscle, and posterior to the submandibular gland. Nakano et al. Squamous Cell Carcinoma From Branchial Cyst. J Oral Maxillofac Surg 2015.

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FIGURE 3. Perioperative findings and appearance of excised specimen. A, A horizontal incision 5 to 6 cm below the horizontal portion of the mandible was made through the superficial fascia and the platysma. The cystic legion was easily dissected from the surrounding tissue. The spinal accessory nerve, facial vein, and hypoglossus nerve were preserved. The existence of a duct was not clear. B, The excised specimen was an elastic cystic structure with a smooth surface and measured 3 cm in diameter. Nakano et al. Squamous Cell Carcinoma From Branchial Cyst. J Oral Maxillofac Surg 2015.

included lymphatic tissue with lymphoid follicles. The cyst wall was lined by nonkeratinizing stratified epithelium that showed varying degrees of cellular atypia. Increases in cellular atypia and the Ki-67 index that are features of a transition from bronchial cyst to carcinoma were observed. In some areas, the lining epithelium was completely replaced by atypical cell proliferation, and invasion into the connective tissue was observed (Fig 4). The lesion was diagnosed as branchiogenic carcinoma. Positron emission tomography was performed in March 2011. No evidence of recurrence was found. The patient was followed for over 2 years 10 months; no evidence of recurrence or another cancer has been found.

Discussion Although there are various theories regarding the development of a branchial cleft cyst, it is usually believed to be derived from the vestigial remnants of a branchial cleft or pouch during fetal life. Branchial cysts are usually localized in the lateral anterior neck and can be found, although their presence is rare, in the parapharyngeal space or nasopharynx.6 Conversely, the development of an invasive squamous cell carcinoma within a cervical cyst as a result of malignant transformation of the epithelium, which is known as branchiogenic carcinoma, is considered extremely rare.2-4 The existence of branchiogenic carcinoma remains controversial and some investigators have claimed that most branchiogenic carcinomas are in fact cystic metastases from oropharyngeal carcinoma (most commonly tonsillar carcinoma) and not true carcinomas arising in a branchial cleft cyst.7,8

However, other investigators have argued that primary branchiogenic carcinomas do exist.9,10 It is very important to distinguish branchiogenic carcinoma from secondary cancer that has metastasized from a malignant lesion arising in the oral cavity or another location. In 1950, Martin et al11 proposed diagnostic criteria for branchiogenic carcinoma to help differentiate this entity from cervical lymph node metastases of unknown primary origin. These criteria were as follows: 1. Location of the tumor along a line anterior to the sternocleidomastoid muscle between the tragus and the clavicle 2. Histologic appearance of the tumor is consistent with tissue present in the branchial vestige 3. The clinical course of the disease is that no primary tumor occurs within a 5-year follow-up period after diagnosis 4. Histologic evidence of a cancer developing in the wall of an epithelial-lined cyst situated in the lateral aspect of the neck Although many researchers have used these criteria to diagnose and describe branchiogenic carcinoma, these criteria have been criticized by several investigators. The criticism of Zimmermann et al10 was that the criterion of a 5-year follow-up without manifestation of a primary lesion was too theoretical and was of limited clinical value because the diagnosis and adequate treatment of an area of a possible primary tumor cannot be postponed for a 5-year period. Furthermore, to show premalignant changes in the epithelium of the cyst, the criteria of Martin et al need to be extended to describe the histologic findings in greater detail. Black and Maran12 stated that a fifth criterion, namely

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SQUAMOUS CELL CARCINOMA FROM BRANCHIAL CYST

FIGURE 4. Microscopic analysis of surgical specimen. A, Gross findings of the surgical specimen (magnification, 1). B, At higher magnification (200), the surgical specimen was determined to be a cystic lesion that exhibited features of a branchial cyst composed of a fibrous connective tissue wall that included lymphatic tissue with lymphoid follicles. C, D, The cyst wall was lined by nonkeratinizing stratified epithelium that showed cellular atypia in varying degrees (magnification, 200). Transition from a bronchial cyst into carcinoma was observed, showing an increase C, in cellular atypia and D, in the Ki-67 index. In some areas, E, the lining epithelium was completely replaced by atypical cell proliferation (magnification, 200), and F, invasion into the connective tissue was observed (magnification, 100). Nakano et al. Squamous Cell Carcinoma From Branchial Cyst. J Oral Maxillofac Surg 2015.

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evidence of premalignant changes in the epithelium of the cyst, was required to aid a conclusive diagnosis. In 1989, Khafif and Prichep13 proposed strict criteria in addition to those of Martin et al. Based on the criteria of Khafif and Prichep, only 10 of 67 reviewed cases from 1951 to 1988 were true branchiogenic carcinomas. Very few cases have been reported that satisfy these criteria. Nevertheless, the authors believe the present case also satisfies these criteria. Some investigators have recommended diagnostic tonsillectomy to rule out clinically inapparent microinvasive tonsillar carcinoma. However, no diagnostic tonsillectomy has been performed in cases of cervical cyst with dysplasia or carcinoma in situ without invasion or adhesion to the surrounding tissue.10,12,14 In addition, by using positron emission tomography, the authors could confirm that the tumor was not a metastasis from other organs. Fine-needle aspiration cytology, panendoscopy, and direct biopsy also are very useful.15 The etiology of malignant transition within a branchial cyst is even less clear. Some investigators have postulated head and neck irradiation6 or inflammatory reactions5 as causative factors,16 but neither of these causes applies to the present case. Because of its rarity, the optimal approach for treatment of branchiogenic carcinoma is not well delineated. However, the treatment of cervical cyst with dysplasia or carcinoma in situ and without signs of malignant infiltration is generally complete surgical excision and such treatment provides a good prognosis.10,12,14 When invasion of, or adhesion to, surrounding tissue is evident, wide surgical excision of the tumor or ipsilateral radical neck dissection17 and postoperative irradiation to the half neck18 have been recommended. The histologic condition of the present case exhibited a histologic progression from chronic inflammation to dysplasia to carcinoma in situ and to frank carcinoma in a branchial cyst. The present case was negative for invasion of atypical cell remains inside the cystic wall and invasion was not found in the surrounding tissue and resection

stump. Therefore, adjuvant radiotherapy was not performed. However, because recurrence of lateral cervical cysts, including recurrence of cysts with malignant transformation, has been reported, clinical follow-up is required.12,13

References 1. Volkman R: Das fiefe branchiogene Halks karcinom. Zentralbl Chir 9:49, 1882 2. Thompson LD, Heffner DK: The clinical importance of cystic squamous cell carcinoma in the neck: A study of 136 cases. Cancer 82:944, 1998 3. Compagno J, Hyams VJ, Safavian M: Does branchiogenic carcinoma really exist? Arch Pathol Lab Med 100:311, 1976 4. Jereczek-Fossa BA, Casadio C, Jassem J, et al: Branchiogenic carcinoma—Conceptual or true clinic-pathological entity? Cancer Treat Rev 31:106, 2005 5. Sing B, Balwally AN, Sundaram K, et al: Branchial cleft cyst carcinoma: Myth or reality? Ann Otol Rhinol Laryngol 107:519, 1998 6. Bilgen C, Ogut F, Celtiklioglu F: A new case of a branchial cyst of parapharyngeal space. Ear Nose Throat J 80:387, 2001 7. Maturo SC, Michaelson PG, Faulkner JA: Primary branchiogenic carcinoma: The confusion continues. Am J Otolaryngol 28:25, 2007 8. Mallet Y, Lallemant B, Robin YM, et al: Cystic lymphnode metastases of head and neck squamous cell carcinoma: Pitfalls and controversies. Oral Oncol 41:429, 2005 9. S€ oderstr€ om K-O: In situ carcinoma in branchial cysts. ORL J Otorhinolaryngol Relat Spec 49:149, 1987 10. Zimmermann CE, von Domarus H, Moubyed P: Carcinoma in situ in a lateral cervical cyst. Head Neck 24:965, 2002 11. Martin H, Morfit HM, Harry E: The case for branchiogenic cancer (malignant branchioma). Ann Surg 2:867, 1950 12. Black B, Maran AG: Branchiogenic carcinoma. Clin Otolaryngol 3:27, 1977 13. Khafif RA, Prichep R: Primary branchiogenic carcinoma. Head Neck 11:153, 1989 14. Glosser JW, Pires CA, Feinberg SE: Branchial cleft or cervical lymphoepithelial cyst: Aetiology and management. J Am Dent Assoc 134:81, 2003 15. Roche JP, Younes MN, Funkhouser WK, et al: Branchial carcinoma of first branchial cleft cyst. Otolaryngol Head Neck Surg 143:167, 2010 16. Visweswara RN, Patel MH: Branchiogenic carcinoma: A case for radiation induced carcinogenesis. Cent Afr J Med 41:296, 1995 17. Hong KH, Moon WS, Chung GH: Radiological appearance of primary branchial cleft cyst carcinoma. J Laryngol Otol 113: 1031, 1999 18. Bhanote M, Yang GC: Malignant first branchial cleft cysts presented as submandibular abscesses in fine-needle aspiration: Report of three cases and review of literature. Diagn Cytopathol 36:876, 2008

A case of squamous cell carcinoma arising from branchial cleft cyst.

Squamous cell carcinoma throughout the epithelium of a lateral cervical cyst is considered extremely rare. This report describes an additional case of...
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