Primary Branchiogenic Carcinoma.

BACKGROUND: Primary branchiogenic carcinoma (BC) is an exceptional diagnostic entity. In most cases, suspicion of BC is confirmed to be a cystic lymph node metastasis of an occult primary tumor. Martin and Khafif therefore established diagnostic criteria, emphasizing above all the importance of a histologically assessed transformation of cystic lining epithelium into an invasive squamous cell carcinoma in one pathological lesion after the exclusion of a primary tumor. CASE REPORT: We present a case report of an 80-year-old patient who presented for a follow-up for an 8-year history of a benign lateral cervical cyst, which was not surgically treated due to the patients high risk of complications from general anesthesia. In spite of the risk, the surgery was performed after a sudden clinical progression, and surprisingly, post-operative histology revealed BC. Conslusion: BC is a very rare entity, strictly defined by Martins and Khafifs criteria. Recently, BCs have been presented in the literature only as unique case reports responding as in our patient to the above mentioned criteria, with particular emphasis on the histological aspect, imaging the cystic lining epithelium transforming into an invasive squamous cell carcinoma in one pathological lesion and a long history without evidence of primary tumor. Key words: branchiogenic carcinoma - lateral branchial neck cyst - occult carcinoma - metastasis of unknown primary origin.

Cervical metastasis of gingival carcinoma misdiagnosed as branchiogenic carcinoma, a rare entity - report of a case and review of literature.

BACKGROUND: A cervical cystic mass is associated with a number of pathologies that present with similar symptoms. These conditions are difficult to differentiate using fine-needle aspiration (FNA), ultrasound (US), computed tomography (CT) and magnetic resonance imaging (MRI). Another dilemma in the differential diagnosis of cervical cystic masses is due to the controversies associated with the existence of branchiogenic carcinoma (BC). BC is an extremely rare disease that must be differentiated from other conditions presenting with cervical cystic masses, especially cystic metastasis from occult primary lesions. CASE PRESENTATION: We present a case report of a right cervical cystic metastasis from a significantly small squamous cell carcinoma primary gingival lesion misdiagnosed as BC by histopathology. A 62-year-old female presented with a painless progressively enlarging cervical mass at the anterior edge of the sternocleidomastoid muscle in the right submandibular region. Preoperative MRI and US revealed a well-defined cystic round mass. Postoperative histological examination indicated BC. Positron emission tomography/computed tomography (PET/CT) revealed high 18F-FDG (18F 2-fluoro-2-deoxy-D-glucose) uptake in surgical regions with a SUV (standard uptake value) max 4.0 and ipsilateral nasopharynx with a SUVmax 4.4, without any distant metastasis. Pathologic results revealed nasopharyngeal lymphadenosis. Considering the low incidence of BC and the limitation of diagnosis in one institution, the patient was referred to another hospital. Physical examination detected a significantly small neoplasm (~3 mm diameter) in the right lower gingiva. Histopathological examination of the neoplasm revealed a well-differentiated squamous cell carcinoma. Surgery, including a partial mandibulectomy and modified neck dissection (neck level I-V and submental lymph nodes) were undertaken. Postoperative histopathological results revealed a well-differentiated squamous cell carcinoma of right lower gingiva and two metastatic lymph nodes in the 18 lymph nodes of level II. A month later, recurrence occurred in the right cervical level II. The patient was placed on postoperative concurrent chemo-radiotherapy and supportive care. The patient suffered from cachexia and survived for only six months after surgery. CONCLUSIONS: In cases of cervical cystic masses that appear after the age of 40, clinicians should bear in mind that occult primary lesions should be excluded and examination of the gingiva should be undertaken. PET/CT has a limited role in identifying small occult primary lesions and a comprehensive physical examination must be carefully performed.

Diagnosis and treatment of bilateral primary branchiogenic carcinoma: A case of malignant transformation of branchial cleft cyst.

Branchial cleft cysts are common congenital lateral neck masses, with 95 % originating from the second branchial cleft. Although most cysts are benign, there are rare instances of malignancy. Here, we present a rare case of primary branchiogenic carcinoma originating from both sides of the neck in a 68-year-old male. Through a detailed analysis of this rare bilateral primary branchiogenic carcinoma, we present the complexity of diagnosing such rare phenomena and the limitations of existing diagnostic methods, emphasizing the need to improve diagnostic methods and the importance of further research for understanding and dealing with similar cases.

Carcinoma within a lymphoepithelial cyst: a case report.

Malignant transformation arising in benign lymphoepithelial cysts is a complex and rare occurrence, and related research is limited. This study presents a case of the malignant degeneration of lymphoepithelial cyst in parapharyngeal space. Clinicopathological features and differential diagnosis are discussed with literature review to provide reference for clinical diagnosis and treatment management.

[Malignant transformation of a branchial cleft cyst]. / Rosszindulatúan elfajult nyaki branchiogen cysta.

Branchial cleft cyst is the most common birth defect involving the neck. Malignant transformation is known, however, differentiating from a neck metastasis of a squamous cell carcinoma of unknown primary is challenging. Even though there are strict criterias, the diagnosis of this entity remains controversial. We present the case of a 69-year-old woman, who presented with a swelling under the left side of the mandible. After diagnostic workup, fine-needle aspiration biopsy raised the suspicion of a cystic squamous cell carcinoma metastasis, therefore we performed panendoscopy and modified radical neck dissection. The pathological examination confirmed branchial cleft cyst carcinoma. After surgery, the patient received adjuvant radiation and chemotherapy. During the case workup, we present the difficulties of the diagnostic process, differential diagnostic problems, and the review of the international literature. In the case of a solitary, cystic mass on the neck without a primary tumor, we should consider the possibility of a branchiogenic carcinoma. Orv Hetil. 2023; 164(10): 388-392.

Primary branchiogenic carcinoma: malignant degeneration of a branchial cyst, a case report.

BACKGROUND: Primary branchiogenic carcinoma (PBC) is an extremely rare and poorly documented disease developed from a brachial cleft cyst. CASE: A 51-year-old patient was referred to our unit for an upper neck mass. PBC was confirmed in accordance with Kahfif's diagnostic criteria. Prophylactic selective neck dissection was performed in a second-stage surgery to ensure the complete removal of the neoplasm. Branchiogenic origin with lymphoid tissue was confirmed in the "host cyst" after histological examination and no other tumors were found elsewhere. Regular follow up documented no relapse 12 months after surgery. CONCLUSIONS: Although rare, PBC must be suspected in presence of cervical masses, especially in patients older than 40 years. A standardized treatment algorithm still lacks, but prophylactic selective neck dissection could be considered as the first line choice after the diagnosis has been confirmed.

Carcinoma within a lymphoepithelial cyst: a case report / 华西口腔医学杂志

Malignant transformation arising in benign lymphoepithelial cysts is a complex and rare occurrence, and related research is limited. This study presents a case of the malignant degeneration of lymphoepithelial cyst in parapharyngeal space. Clinicopathological features and differential diagnosis are discussed with literature review to provide reference for clinical diagnosis and treatment management.

Branchiogenic carcinoma with high-risk-type human papillomavirus infection: A case report.

Branchiogenic carcinoma (BC) usually appears as a mass lesion with a predominant cystic component. Since lymph node metastasis from oropharyngeal carcinoma (OPC) has a cystic appearance, it is occasionally difficult to distinguish between BC and nodal metastases from clinically silent OPC. Factors associated with the malignant transformation process in BC remain obscure. The present study reports the case of a 56-year-old man with a right cystic cervical mass that was diagnosed as squamous cell carcinoma based on examination by fine-needle aspiration biopsy. The primary tumor could not be detected despite several imaging examinations, a pan-endoscopy of the head and neck, esophagus and stomach, biopsies of the head and neck regions, and bilateral tonsillectomies. The pathological findings of the surgical specimens from a radical neck dissection were consistent with the histological characteristics of BC, with evidence of transition from dysplasia through intraepithelial carcinoma to invasive carcinoma. Normal squamous epithelium and dysplastic and cancerous portions in the BC showed strong p16INK4a immunoreactivity. The expression of p16INK4a was also observed in all 9 nodal metastases in the neck dissection specimens. The cystic formation observed in the BC was not observed in the nodal metastases. As the presence of human papillomavirus-16 in the tumor was confirmed by polymerase chain reaction, quantitative polymerase chain reaction was employed for the measurement of human papillomavirus-16 viral load and integration. The results showed that the viral load of human papillomavirus-16 was 3.01×107/50 ng genomic DNA, and the E2/E6 ratio was 0.13, so the integration state was judged to be the mixed type. To the best of our knowledge, this is the first report of BC associated with high-risk-type human papillomavirus infection. The study indicates that a human papillomavirus-positive neck mass may not necessarily be OPC, but that it could be BC with a poor prognosis. This report lends support to the existence of BC and proposes that the etiology is human papillomavirus infection.

Adenosquamous carcinoma of unknown primary origin: a case report and literature review.

BACKGROUND: Adenosquamous carcinoma is a rare variant of semicircular canal that can affect various regions, including the head and neck. Adenosquamous carcinoma is characterised pathologically by the simultaneous presence of distinct areas of semicircular canal and adenocarcinoma, and usually takes an aggressive course with local recurrences, early lymph node metastases and distant disseminations. CASE: We report a rare case of neck adenosquamous carcinoma of unknown primary origin, which was well-controlled by thorough resection without any other additional therapy. CONCLUSION: We discuss the diagnosis and treatment of adenosquamous carcinoma along with a review of pertinent literature. We also consider the potential differential diagnosis of branchiogenic carcinoma.

Cystic squamous cell carcinoma of the neck: could a second metastatic focus help?

Squamous cell carcinoma of the neck presenting clinically as predominantly cystic lesion has often been considered as branchiogenic carcinoma in the past. However, such cystic lesions presently constitute a distinct form of head-neck metastasis, and a co-existent second metastatic focus could supposedly help distinguish them from branchiogenic carcinoma. We here present a case where, although the primary remained elusive, the associated spinal metastasis precluded us from diagnosing a cystic squamous cell carcinoma as branchiogenic carcinoma. Thus a second metastatic focus, if found, could add to the existing knowledge to distinguish between a cystic squamous cell carcinoma and the so-called branchiogenic carcinoma, especially when the primary could not be found.

A Case of Squamous Cell Carcinoma Arising from Branchial Cleft Cyst / Oral Science International

Carcinoma arising from the remnant of branchial epithelium or branchial cleft cyst is known as branchiogenic carcinoma. It is very rare, and its existence is a matter of controversy. We report a case of cystic carcinoma of the upper neck that fully met Martin's criteria for branchiogenic carcinoma. A 53-year-old male visited Tsurumi University Dental Hospital with a swelling on the left side of the neck. Three tumors were excised from the neck, and histopathology revealed squamous cell carcinoma in a cystic lesion. As metastatic carcinoma of the cervical lymph nodes was suspected, the appropriate clinical tests and imaging were performed to determine the possible presence of a primary tumor. However, no primary carcinoma was found. These findings suggest that this was a case of branchiogenic carcinoma. The patient was treated with radiotherapy and followed up over an 8-year period. No evidence of recurrence was found.

Fine Needle Aspiration Cytology of the Branchiogenic Carcinoma: Report of A Case / 대한세포병리학회지

Development of an invasive squamous cell carcinoma within a branchial cleft cyst (branchiogenic carcinoma) is very rare and this case is the first description of cytologic findings in Korea. A 62-year-old man presented with a 5-month history of an enlarged right neck mass. Fine needle aspiration cytology of the mass showed low cellularity and the background containing proteinaceous debris, inflammatory cells, and histiocytes suggested a cystic nature. Abundant lymphocytes, a few anucleated squames, and rare atypical squamous cells with pyknotic nuclei and abnormal keratinization were also noted. After excision of the mass, histologic findings were consistent with a branchiogenic carcinoma. We recommend to raise the possibility of carcinoma (primary or metastatic) when we observe a few dyskeratotic cells in a cystic mass within the neck area, especially in an old male.