Malignant first branchial cleft cysts presented as submandibular abscesses in fine‐needle aspiration: Report of three cases and review of literature

Abstract

Malignant branchial cleft cyst is a rare entity. To the best of our knowledge, there have been one case of malignant first branchial cleft cyst and 13 cases of malignant second branchial cleft cysts in the literature that fulfill Khafif's modified criteria since 1982. We have encountered three such cases, presenting as unilateral submandibular abscesses, refractory to antibiotics, from men of 52, 53, and 61 years of age. Fine-needle aspiration was reported as "abscess" in the first case, "atypical squamous cystic lesion" in the second case, and "atypical first branchial cleft cyst" in the third case. Histopathology showed Type II malignant first branchial cleft cysts with in situ squamous-cell carcinoma in one patient and in situ and invasive squamous-cell carcinoma in the other two patients. Patients are alive and disease-free 3-4.5 years post-surgery and -radiation. Awareness of this extremely rare entity can potentially avoid underdiagnosis by the cytopathologist and overtreatment by the surgeons.

Branchial cleft cyst: an unusual ultrasound diagnosis in an austere environment

“…However, we are confident that given the location of the mass and its benign ultrasonographic characteristics, in addition to the patient's previous fine-needle aspiration suggesting such, the most likely diagnosis was a branchial cleft cyst. Importantly, several case reports [8][9][10] would argue that without this prior knowledge of a benign fine-needle aspiration, it would be unwise to assume, based solely upon ultrasound findings, that this not a cancerous lesion.…”

Primary branchiogenic carcinoma in the first branchial cleft of a child

“…Due to their shared anatomic region, primary branchial cleft cyst carcinoma needs to be distinguished from cystic cervical metastases arising from occult primary tumors in the upper aerodigestive tract, which have a much higher incidence rate (1,2,5,7,8). The majority of cystic cervical metastases originate from Waldeyer's ring, including the nasopharynx, tonsil and the base of the tongue.…”

“…Since some of the criteria, particularly the third point, although important in theory, are not quite feasible in clinical practice, Khafif et al (4) modified the criteria to emphasize the significance of clinical and histopathological evidence. These criteria are widely acknowledged by the majority of scholars, including Martin et al Furthermore, they all agree that histopathological evidence is the most crucial criterion, i.e., histopathological proof of gradual transition of normal squamous epithelium into invasive carcinoma (1,11,12). Clinically, it is also important that there is an absence of any identifiable primary malignant tumors following a thorough evaluation of the patient with endoscopy (nasopharyngoscopy, laryngoscopy, bronchoscopy and esophagoscopy) and radiographic examinations.…”

“…To date, there are only approximately 40 cases of primary branchiogenic carcinoma reported in the literature, which were found mostly in men aged 40-60 with the majority of cases involving second branchial cleft cysts (6,7). In terms of primary carcinoma in the first branchial vestigium, to the best of our knowledge, there are only 5 reported cases, all of which occurred in adults (1,8,9). Herein, we report the first case of primary first branchial cleft carcinoma in a 10-year-old Chinese girl who had suffered from first branchial cleft sinus from childhood.…”

“…Since Von Volkmann originally described this unique entity in 1882 (1,2), there has been skepticism and controversy over issues concerning its existence and diagnosis. After reviewing 250 cases in 1950, Martin et al (3) revealed that the previous diagnostic criteria for this carcinoma were inaccurate and questionable, and thus proposed four stringent criteria to establish an accurate diagnosis of primary branchiogenic carcinoma, which were later modified by Khafif et al (4).…”