Lingual Tonsil Research Articles

Transoral robotic lingual tonsillectomy in adults: preliminary results

Conclusion: Hypertrophy of lingual tonsils can be safely and effectively managed by transoral robotic surgery via a minimally invasive approach. The operating costs can be relatively high although the advantages to patients seem to justify the procedure. Transoral robotic surgery can represent the definitive treatment in hypertrophy of the lingual tonsils. Objective: The purpose of this work was to assess the feasibility, safety, and outcomes of transoral robotic surgery for lingual tonsillar hypertrophy in adults with obstructive sleep apnea. Methods: Retrospective medical records of six patients who underwent tongue base resection using the da Vinci Surgical Robotic System from September, 2012 through December, 2013 in a tertiary care hospital were evaluated. Demographic data, preoperative and postoperative polysomnography and Epworth Sleepiness Scale (ESS) scores, robot docking time, operative and postoperative aspects including surgical time, amount of bleeding, and early and late complications were collected and analyzed. Results: All patients successfully underwent lingual tonsillectomy using the da Vinci Surgical system. Transnasal endotracheal entubation was performed in all patients and they were all extubated before leaving the operation room. No tracheotomies were performed perioperatively for airway management. The patients were discharged on the fifth postoperative day. Operative time, estimated blood loss, and postoperative complication profiles were within the expected and acceptable limits for lingual tonsillectomy. There was a statistically significant decrease in apnea hypopnea index (AHI) (mean AHI from 27.5 to 6.3) and ESS scores (from 14.1 to 7.1).

Evaluation of coblation lingual tonsil removal technique for obstructive sleep apnea in Asians: preliminary results of surgical morbidity and prognosticators

Retroglossal obstruction is one of the etiologies causing obstructive sleep apnea (OSA) and can be addressed by removing some tissues of the tongue base. However, because of its deep-seated location, its surgical removal is still challenging. Although coblation technique has been introduced, its efficacy and morbidity need further evaluation, particularly in Asians. This study aimed to assess its safety and effectiveness and to identify outcome prognosticators. Forty-seven OSA patients who underwent coblation lingual tonsil removal were included. Retroglossal obstruction was confirmed by drug-induced sleep videofluoroscopy. Attended full-night polysomnography was performed twice; before and 6 months after surgery in 27 patients. The tongue base was fully exposed with three deep-seated traction sutures, visualized with a 30° or 70° endoscope, and ablated using a coblator. Surgical success was defined with postoperative apnea hypopnea index (AHI) <20 and reduction >50 %. Postoperative morbidities were evaluated. Demographic and polysomnographic parameters between success and failure groups were compared. None of the patients had immediate postoperative hemorrhage. Postoperatively, one patient had delayed hemorrhage and one patient severe respiratory difficulty. Taste loss, tongue dysmotility, dental injury or severe oropharyngeal stricture were absent. A mean AHI decreased from 37.7 ± 18.6 to 18.7 ± 14.8/h (P < 0.001). The success rate was 55.6 %. Their mean minimal oxygen saturation was significantly lower (P = 0.004) in the failure group. Coblation lingual tonsil removal technique showed minimal morbidity and favorable outcome in Koreans. The surgical outcome might be associated with the severity of single respiratory events.

Early events in the pathogenesis of foot-and-mouth disease in pigs; identification of oropharyngeal tonsils as sites of primary and sustained viral replication.

A time-course study was performed to elucidate the early events of foot-and-mouth disease virus (FMDV) infection in pigs subsequent to simulated natural, intra-oropharyngeal, inoculation. The earliest detectable event was primary infection in the lingual and paraepiglottic tonsils at 6 hours post inoculation (hpi) characterized by regional localization of viral RNA, viral antigen, and infectious virus. At this time FMDV antigen was localized in cytokeratin-positive epithelial cells and CD172a-expressing leukocytes of the crypt epithelium of the paraepiglottic tonsils. De novo replication of FMDV was first detected in oropharyngeal swab samples at 12 hpi and viremia occurred at 18–24 hpi, approximately 24 hours prior to the appearance of vesicular lesions. From 12 through 78 hpi, microscopic detection of FMDV was consistently localized to cytokeratin-positive cells within morphologically characteristic segments of oropharyngeal tonsil crypt epithelium. During this period, leukocyte populations expressing CD172a, SLA-DQ class II and/or CD8 were found in close proximity to infected epithelial cells, but with little or no co-localization with viral proteins. Similarly, M-cells expressing cytokeratin-18 did not co-localize with FMDV proteins. Intra-epithelial micro-vesicles composed of acantholytic epithelial cells expressing large amounts of structural and non-structural FMDV proteins were present within crypts of the tonsil of the soft palate during peak clinical infection. These findings inculpate the paraepiglottic tonsils as the primary site of FMDV infection in pigs exposed via the gastrointestinal tract. Furthermore, the continuing replication of FMDV in the oropharyngeal tonsils during viremia and peak clinical infection with no concurrent amplification of virus occurring in the lower respiratory tract indicates that these sites are the major source of shedding of FMDV from pigs.

The Association of Reflux and Lingual Tonsil Hypertrophy: A Systematic Review

Objectives: Evaluate the association between lingual tonsil hypertrophy (LTH), gastroesophageal reflux disease (GERD), and laryngopharyngeal reflux (LPR). Methods: A systematic literature search was performed using MEDLINE and the Cochrane Library databases through July 2013 to identify original research articles examining the effects of GERD or LPR on LTH in both adults and children. Only original research articles in English were retrieved using the keywords “reflux and lingual tonsil,” “reflux and base of tongue hypertrophy,” “laryngopharyngeal reflux and lingual tonsil,” “LPR and lingual tonsil,” and extraesophageal reflux and lingual tonsil.” Results: Out of 33 studies that matched the search criteria, 5 studies in adults and 3 studies in children met the inclusion criteria. In adults, one study noted a significantly increased prevalence of LTH in patients with signs and/or symptoms of LPR (62.4%) versus patients without signs and/or symptoms of LPR (29.3%). Four studies noted an increase in the severity of LTH in the presence of signs or symptoms of LPR. In the pediatric population, all 3 studies noted a positive association between the presence of LPR and GERD and the presence of LTH (30%-70%). Conclusions: The results of this systematic review suggest an association between LTH and reflux in both adults and children. This study is limited by heterogeneity in methodology and definitions of LTH and reflux between included studies. Further investigation is needed to characterize the clinical entity of LTH as well as the nature of the relationship between LTH and reflux disease.

Factors Associated with Hypertrophy of the Lingual Tonsils

Objectives: Identify factors that may be associated with lingual tonsil hypertrophy (LTH). Methods: Retrospective chart review identified 380 patients from August 2013 to January 2014 with graded lingual tonsils, documented during routine flexible laryngoscopy. Lingual tonsils were graded using a 0 to 4 scale: 0 = complete absence of lymphoid tissue, 1 = lymphoid tissue scattered over tongue base, 2 = lymphoid tissue covers entirety of tongue base with limited thickness, 3 = lymphoid tissue 5 to 10 mm in thickness, 4 = lymphoid tissue &gt;1 cm in thickness (above tip of epiglottis). Reflux symptom index (Reflux Symptom Index (RSI)–collected during patient intake), presence of obstructive sleep apnea (OSA; confirmed by polysomnogram), smoking habits, and basic demographics were gathered. Ï•2 and linear multi-variate regression analyses were used to identify significant correlating demographics with LTH. Results: Overall, 59.5% were male with a mean age of 50.2 ± 16.5 years and body mass index (BMI) of 30.1 ± 18.0. Ï•2 analysis revealed no significant relationship between OSA and LTH ( P = .059). When RSI was stratified to ≥10 or &lt;10, a Cochran-Armitage test supported the trend hypothesis that as RSI increases lingual tonsil grading increases. Significant univariate correlates included age ( r = –0.307, P &lt; .001) and smoking ( r = 0.186, P = .002). Multivariate regression revealed the combination of age and smoking ( r = –0.297, P &lt; .001) to be a significant correlate. Conclusions: LTH does not seem to be associated with OSA in this group of patients. Abnormal RSI may be a factor associated with increased lingual tonsil thickness. Younger patients who smoke are more likely to have LTH.

A Standardized Lingual Tonsil Grading System: Interexaminer Agreement

Objectives: Comparisons between studies involving the tongue base are limited by the lack of a universal system for grading lingual tonsils. The authors propose a new standardized grading system for lingual tonsil hypertrophy (LTH). Validation was assessed via an interexaminer agreement study. Methods: Video assessment: The proposed grading system consists of a 0 to 4 scale with: 0 = complete absence of lymphoid tissue, 1 = lymphoid tissue scattered over tongue base, 2 = lymphoid tissue covering entirety of the tongue base with limited vertical thickness, 3 = lymphoid tissue 5 to 10 mm in thickness, 4 = lymphoid tissue &gt;1 cm in thickness (above tip of the epiglottis). A teaching video was created to demonstrate identification of this grading system. After viewing the teaching video, 8 trained otolaryngologists graded 25 video clips of the tongue base, recorded during flexible laryngoscopy. Live assessment: A second study was performed by 2 examiners directly examining the tongue base of 23 patients using flexible laryngoscopy during a routine clinic visit. Each examiner viewed and recorded the grade of the lingual tonsils independently. Results: Video assessment: The overall Fleiss kappa statistic was found to be κ = 0.449 ( P &lt; .0001). This value denotes moderate agreement. Live assessment: The overall kappa for nominal data were found to be κ = 0.8665 ( P &lt; .0001). This value denotes perfect agreement. Conclusions: The moderate interexaminer correlation demonstrated during video-assessment, and perfect interexaminer correlation demonstrated during live assessment, indicate that this proposed grading system may be a valuable and useful tool in creating a common language to describe lingual tonsils.

Lingual Tonsil Abscess with Parapharyngeal Extension: A Case Report

Lingual tonsil abscess is a rare disorder previously reported only once in the English literature. Because of their similar structure to that of the palatine tonsils, the lingual tonsils have the propensity to develop infection in the same way. The progression of infection, however, is different in that the lingual tonsils lack a capsule, thus preventing the formation of a peritonsillar abscess. Therefore, the only place for infection to spread is either into the tongue or into the parapharyngeal space. Here we present our experience with the latter, and we provide radiographic evidence of the disease. Lingual tonsil abscess, although rare, is an important potential cause of airway obstruction and must be considered in the case of a sore throat with a normal oropharyngeal exam.

Waldeyer ring lymphoma: a case series.

We encountered a patient with a tongue base lymphoma that we initially diagnosed as a lingual tonsil in view of its benign appearance. We established the correct diagnosis of Waldeyer ring lymphoma by histology. This case led us to conduct a study of all cases of Waldeyer ring lymphoma that had been treated at our center during a 10-year period. We retrospectively examined our case records and found 35 such cases. From this group, we excluded 5 cases because of incomplete data. Thus our final study group was made up of 30 patients-14 males and 16 females, aged 14 to 76 years (mean: 51.6; median 54). The primary presenting signs and symptoms were dysphagia (n = 17 [57%]), a neck mass (n = 7 [23%]), nasal symptoms (n = 5 [17%]), and pain (n = 1 [3%]). Only 4 patients (13%) had B symptoms. A total of 20 patients (67%) presented with tonsillar involvement, 8 (27%) with nasopharyngeal involvement, 1 (3%) with tongue base lymphoma, and 1 with anterior tongue involvement. Most patients (77%) presented at an early stage. Histologically, 25 patients (83%) had high-grade diffuse large B-cell lymphoma, 4 (13%) had T-cell lymphoblastic lymphoma, and 1 (3%) had follicular lymphoma. Twenty-one patients (70%) were treated with chemotherapy, 4 (13%) received adjuvant chemotherapy with either radiotherapy or surgery, 3 (10%) resorted to other forms of treatment (primarily traditional remedies), and 2 (7%) declined treatment altogether. There were 14 patients (47%) alive at the end of the study period.

Influencia de las anomalías faringolaríngeas diagnosticadas mediante laringoscopia indirecta en la predicción de la dificultad de intubación

ObjectiveTo determine the pharyngolaryngeal anomalies not usually included in the evaluation of difficult airway, in order to investigate the influence of these anomalies in the prediction of difficult intubation. To do this, indirect laryngoscopy with a 70° rigid laryngoscope was performed on all patients during the preoperative period. MethodsThis is an observational, prospective study on 300 consecutive patients who were scheduled for endotracheal intubation under general anesthesia. In addition to assessing the airway in the preoperative period by demographic and clinical predictors of difficult airway, rigid indirect laryngoscopy was performed to diagnose pharyngolaryngeal anomalies. Later, under general anesthesia and direct laryngoscopy it was checked to see if there was difficulty in intubating the larynx, and its association with all previous variables was investigated. A logistic regression model for prediction purposes was developed, and its power of discrimination was achieved by assessing the area under the curve. ResultsDuring the examination by indirect laryngoscopy 46 anomalies were found, which were as follows: 31 abnormalities of the epiglottis (22 omega epiglottis, 9 flaccid or hypertrophic epiglottis); 6 findings of hypertrophic lingual tonsils, 3 cases of upper airway tumors, and 6 patients with tongue disorders. Intubation difficulty was found in 14 cases (4.66%). The regression model found, and its coefficients to develop it were: f(x)=1.322+(2.173 thyromental distance <6.5cm)+(1.813 omega epiglottis)–(1.310*cm opening mouth). Global power of discrimination was 0.83, with a 95% confidence interval from 0.709 to 0.952). ConclusionIndirect laryngoscopy allowed pharyngolaryngeal anomalies to be diagnosed, including omega epiglottis, which was one of the variables included in the logistic regression model.

Reduction of immunity in HIV-infected individuals: Can fibrosis induce hypoplasia in palatine and lingual tonsils of individuals with HIV infection?

The role of tonsils in oral immunity has been described. However, the pathogenesis of HIV infection in these organs is still unclear. The aim of this study is to perform histological and immunohistochemical analysis of the palatine and lingual tonsils of autopsied individuals with or without HIV infection. Twenty-six autopsied individuals with HIV infection (HI) (n=13) and without HIV infection (CO) (n=13) were selected. Palatine and lingual tonsil fragments were collected for histological and immunohistochemical analysis. We found in the HI group a higher frequency of hyaline degeneration in both palatine and lingual tonsils; smaller follicle areas, and a higher percentage of collagen in comparison with the CO group. In the HI group, there was higher density of blood vessels in palatine tonsils than in the CO group. In the HI group, there were significant positive correlations between palatine and lingual tonsils and the area of lymphoid follicles, and between the percentage of blood vessels and collagen in palatine tonsils. In addition, there was a significant negative correlation between the percentage of collagen and lymphoid follicle area in both palatine and lingual tonsils in the HI group. These findings suggest that the immune functions of these tonsils are prejudiced by fibrosis. Therapies to reduce the neoformation of collagen are required to improve immune function of organs against pathogens.

Lingual Tonsillar Metastasis From Rectal Carcinoma

A 66-year-old man presenting with sacral pain 18 months after radical transabdominal resection of rectal mucinous adenocarcinoma underwent F-FDG PET/CT cancer surveillance. Detection of multiple nodules in lingual tonsil and left neck by imaging initially suggested tonsillar squamous carcinoma (as a second primary) with spread to cervical nodes, given the rarity with which rectal carcinoma metastasizes to the head and neck. Ultimately, the tonsillar neoplasm proved to be adenocarcinoma of colorectal origin based on its shared histologic features and compatible immunostaining profile.

Transoral laser microsurgery for the unknown primary: Role for lingual tonsillectomy

We conducted a retrospective review of patients with unknown primary head and neck cancer who underwent a transoral laser microsurgery (TLM)-assisted search for an occult tumor. Fifty-two patients at a single center with unknown primaries of squamous cell carcinoma (SCC) were treated with a surgical algorithm. The overall rate of primary tumor identification was 75% (39 of 52 patients). When selecting those cases in which a TLM algorithm with lingual tonsillectomy was utilized, the detection rate was 86% (31 of 36). Tumors were most commonly found in the lingual (65.0%) and palatine tonsils (27.5%). When directed biopsies did not locate a tumor, a higher yield was obtained with the addition of a lingual tonsillectomy in 8 patients. A surgical algorithm for the unknown primary that includes TLM-assisted techniques, including a lingual tonsillectomy, offers the greatest likelihood of successfully detecting the location of occult primary tumors.

The prevalence of human papilloma virus (HPV) infections in oral squamous cell carcinomas: A retrospective analysis of 88 patients and literature overview

In addition to tobacco and alcohol consumption, the two main risk factors for oral squamous cell carcinoma (OSCC), recent studies have revealed infections with human papilloma virus (HPV) as an additional risk factor for OSCC development. In the field of head and neck malignancies, the prevalence of HPV infections in oropharyngeal cancer (OC) ranges in different studies up to 84%. While HPV infection is discussed as an independent risk factor in this region, its distinguished role in carcinogenesis of tumours localized to the oral cavity remains still uncertain.In this study, we analysed the HPV status in 88 consecutive patients with OSCCs localized anterior of the palatoglossal arch who were treated in the Department of Oral and Maxillofacial Surgery at the University Medical Center Mainz. The HPV status analysis was performed using DNA-PCR and immunostaining of p16 protein.The prevalence of HPV-positive OSCCs was about 6% (5 patients). In 3 patients the HPV subtypes 16/18 were found. No significant differences between the HPV positive and negative patients regarding age, gender, smoking and alcohol consumption, localization and TNM level could be detected. Contrary to other studies focussing on cancers of the lingual and palatine tonsil, the prevalence of HPV infections was much lower in the oral cavity. Therefore HPV infection might play a less important role in oral carcinogenesis.

Difficult Laryngeal Mask Airway Placement.

To the Editor; Laryngeal mask airway (LMA), which consists of a silicone tube connected to a miniature silicone mask appropriate for the shape of hypopharynx and covers the larynx as a gasket, was developed in 1980s in the United Kingdom. It is frequently used both to avoid negative effects of endotracheal intubation by establishing direct connection with the airway of patient and to easily provide a more reliable airway as compared to the facial mask. It has particular importance in difficult airway management and cardiopulmonary resuscitation since it can be successfully placed even by inexperienced users. Gel is applied to the posterior surface of the mask and it is placed blindly into the airway by pushing towards hypopharynx while the head of the patient is in extension position. Herein, we present a case, in which LMA could not be placed despite several attempts and a mass tissue was detected in the hypopharynx during laryngoscopy performed subsequently. An 81-year-old, 70 kg male patient was scheduled for cystoscopy in the urology clinic. Preanaesthetic evaluation revealed no pathology or concomitant disease except for advanced age. He had previously undergone prostate surgery under spinal anaesthesia. His Mallampati score was 3 and he had a large tongue (Figure 1). As the procedure was anticipated to take a short time and the patient, who had a high Mallampati score, did not want to be awake during the procedure, LMA was preferred for airway management. Figure 1 Examination image for Mallampati score Induction of anaesthesia was made with propofol, fentanyl and rocuronium, the patient was easily ventilated with a face mask, and an attempt was made to place a size 5 standard LMA; but as the mask, which was pushed forward, came back, it was thought that the mask was large for the patient and the attempt was repeated using a size 4 mask. However, as this attempt was also unsuccessful, it was thought that silicone tip of the balloon was folded; thus, I-Gel (size 4), which is a supraglottic airway device without a cuff and stands more stable, was tried to be placed. However as size 4 I-Gel LMA was pushed forward and ventilation was initiated, a significant air leakage occurred, and considering that size 4 was small, we again tried using size 5. However, observing substantial amount of air leak at the end of all these attempts, LMA was planned to be placed under laryngoscopic guidance. Airway examination, which was performed by a Macintosh laryngoscope, revealed a 2×2 cm mass located at the base of the hypopharynx, covered by normal mucosa, and appeared to be neither cystic nor ulcerated (Figure 2). It was thought that the mass, which was located at the same place where LMA device was placed, caused the tip of standard LMAs to be folded or remain between the mass and pharyngeal tissue, and hindered I-Gel LMA to closely fit after being placed and caused leakage from the back of the device, and consequently the attempts were unsuccessful. The patient, of whom Cormack-Lehane score was 2, was then intubated by size 8 endotracheal tube. We encountered no problem during intraoperative period and the patient was extubated without problem at the end of the procedure that lasted for 45 minutes; he was recommended to refer to the ENT outpatient clinic. Meanwhile, consent of the patient was obtained for presenting his case as a case report. Figure 2 The appearance of the mass through the laryngoscope Unforeseen dangers might have been in question because of blindly placed LMA, in this patient who had a deeply localized asymptomatic lesion that could not be detected by routine airway examination. If this mass located in the hypopharynx, had been cystic, it would have been perforated during the attempts to place the LMA and would have led to serious consequences by the spread of cyst content into the lungs. Again, if it had been a vascular mass like a haemangioma, it would have been difficult to control the unexpected bleeding. Lingual tonsil hyperplasia, which is more common in paediatric cases, may lead to similar problems (1, 2). Although blind placement of LMA without using a laryngoscope is an advantage, it may turn into a disadvantage in complicated cases and may lead to various problems. In case of recurrent failure while placing a supraglottic device, an airway disorder, as well as common problems such as incompatibility between device and patient anatomy and folded balloon, should be considered and attempts should aim at performing non-traumatic interventions as much as possible without insisting on blind replacement.

Obesity and Laryngopharyngeal Reflux as Risk Factors of Lingual Tonsil Hypertrophy in Patients With Sleep-Disordered Breathing—Reply

Obesity and Laryngopharyngeal Reflux as Risk Factors of Lingual Tonsil Hypertrophy in Patients With Sleep-Disordered Breathing—Reply

Obesity and Laryngopharyngeal Reflux as Risk Factors of Lingual Tonsil Hypertrophy in Patients With Sleep-Disordered Breathing: Validity of the Statistical Approach

Obesity and Laryngopharyngeal Reflux as Risk Factors of Lingual Tonsil Hypertrophy in Patients With Sleep-Disordered Breathing: Validity of the Statistical Approach

Safety of cold ablation (coblation) in the treatment of tonsillar hypertrophy of the tongue base

The treatment of diseases of the lingual tonsils is still under debate, and surgical interventions are often associated with significant morbidity and complications. The aim of the present study was to evaluate the safety of lingual tonsillectomy using cold ablation (coblation) as a new treatment of lingual tonsil diseases. In this retrospective, bicentric study, we included all patients between 2005 and 2012 who underwent cold ablation (Coblation(®)) of the lingual tonsils. We assessed the frequency of postoperative complications based on the patients' charts. A total of 108 patients (47 ± 13, 6 years) underwent lingual tonsillectomy using coblation. All patients were operated on under general anesthesia as inpatients. Intraoperative complications did not occur. Three patients (2.8%) needed revision surgery due to postoperative hemorrhage, and in one of those cases, three revisions were necessary. There was no postoperative airway compromise and no need for tracheostomy. There was no hypoglossal nerve paralysis, but in the case needing multiple revisions, a weakness of the hypoglossal nerve persisted. In all the cases, oral intake was possible with adequate analgesia. Coblation of the tongue base is a safe procedure with a relatively low rate of postoperative complications. Postoperative hemorrhage is the most relevant complication that occurred in our series of patients. Future studies are needed to evaluate the efficacy of the procedure in the treatment of obstructive sleep apnea.

頸部腫瘤摘出・生検後に中咽頭癌と診断された症例の検討

In the case of oropharyngeal carcinoma, patients may present with symptoms similar to cervical lymphadenopathy, and the primary lesion may only be diagnosed after cervical mass extirpation/biopsy. We retrospectively analyzed the clinical course in 11 oropharyngeal carcinoma patients that were diagnosed after cervical mass extirpation/biopsy between 1998 and 2013. Before the diagnosis was made of oropharyngeal carcinoma, a cervical lymph node biopsy was performed in six patients; the lymph node was extirpated due to an initial diagnosis of lateral cervical cyst in four patients; and neck dissection was performed due to an initial diagnosis of primary unknown carcinoma in one patient. The primary tumor site in the oropharynx was the palatine tonsil in six patients and the lingual tonsil in five patients. Five of six patients with palatine tonsil carcinoma and three of five patients with lingual tonsil carcinoma were found to be positive for human papillomavirus (HPV). The duration from cervical lymph node extirpation/biopsy to final diagnosis was 1 to 13 months. All patients finally underwent radiation therapy or chemoradiotherapy, and they had no recurrence or metastasis. As the incidence of HPV-related oropharyngeal carcinoma increases, the number of oropharyngeal carcinomas assumed to be cervical lymphadenopathy due to the presenting symptoms may increase. It is important to investigate the oropharynx thoroughly so as to adequately differentiate the possibility of oropharyngeal carcinoma from that of cervical lymphadenopathy. Metastatic lymph nodes might present as cysts in cases of oropharyngeal carcinoma, it is therefore necessary to take the potential for metastatic lymph nodes in the oropharyngeal cancer into consideration when differentiating this disease from cervical cyst-shaped lesions.

A Case of Branchiogenic Carcinoma

A case of branchiogenic carcinoma is reported herein. The patient was a 59-year-old female who noticed a mass on the left neck and visited our hospital. CT and echography revealed a cystic lesion, and PET disclosed multiple abnormal accumulations on the bilateral cervical lymph nodes and pharynx. First of all, we suspected the tumor as being a malignant lymphoma or metastatic carcinoma. We resected the tumor under general anesthesia for biopsy. The histopathological diagnosis was squamous cell carcinoma arising from a branchiogenic cyst. We examined the whole body and added the biopsies of the epipharynx, palatine and lingual tonsils which revealed no malignancy and no other primary lesions existed. The tumor was therefore diagnosed as a branchiogenic carcinoma. Bilateral radical neck dissection, and postoperative chemotherapy and radiotherapy were performed. The patient has survived until the time of writing for 30 months without recurrence.

Tumor flare reaction in a patient with mantle cell lymphoma.

TO THE EDITOR: Tumor flare reaction (TFR) is a syndrome that clinically presents with painful lymph nodes and/or spleen enlargement, and it can be accompanied by fever, rash, and clear lymphocytosis [1]. Medical literature frequently points out its association with chronic lymphocytic leukemia (CLL) and lenalidomide treatment. In a study by Chanan-Khan et al. [2], it was observed that of 26 (58% of 45 patients) CLL patients who were administered 25 mg/day oral lenalidomide treatment for 21 days in 28-day cycles developed TFR reactions (50% developed grade 1-2 reactions, and 8% developed grade 3-4 reactions). In another study by Ferrajoli et al. [3], it was observed that 10 (28% of 35 patients) CLL patients who were first treated with a dose of 10 mg/day of lenalidomide and then with an increased dose of 25 mg/day developed TFR. While TFR cases after lenalidomide treatment have been frequently reported, TFR has not been reported after Hyper-CVAD plus Rituximab (R-HCVAD) treatment. Herein, we report a patient diagnosed with mantle cell lymphoma who developed TFR after use of high dose cyclophosphamide, doxorubicin, adriamycin and dexamethasone plus rituximab. The patient, who was being followed up for relapse of mantle cell lymphoma, developed fever, throat swelling, pain, and difficulty in swallowing after R-HCVAD treatment. Magnetic resonance imaging (MRI) of the patient's throat showed a mass, sized approximately 65×29 mm at its broadest point, with its borders not clearly identified; the mass caused distinct asymmetry in the hypopharynx and oropharynx, displaced the uvula to the right side, and continued to the epiglottis level. Distinct hypertrophy of the lingual and left palatine tonsils as well as of multiple lymph nodes on both sides were observed in the left submandibular area of the cervical chain of the neck, the biggest of which was sized 20×12.5 mm (Fig. 1). These findings suggested TFR after chemotherapy, and dexamethasone (24 mg/day) was initiated. The patient's fever decreased, and his complaints regressed on the 10th day of the treatment; throat MRI revealed edema and inflammation regression when compared to that observed on the previous MRI scan. A thin area of loculation was observed in the left retropharyngeal and parapharyngeal space and a small mass residue, 2.5×8 mm in size, was observed in the parapharyngeal space, with no clear contrast involvement. The patient's complaints abated completely after dexamethasone treatment; therefore, medications were stopped and follow-up was initiated. Fig. 1 The patient's throat magnetic resonance imaging scan showing a heterogeneously dense mass, sized approximately 65×29 mm at its broadest point, with its borders not clearly identified; therefore, the mass caused distinct asymmetry in the hypopharynx ... Until recently, it was thought that TFR associated with lenalidomide was a condition specific only in CLL patients. However, in 2010, Corazzelli et al. [4], reported three patients with relapsing or refractory Hodgkin lymphoma who developed TFR, and thus, showed that it was not so. Similarly, Eve and Rule [5], showed that three of 25 patients with mantle cell lymphoma who were treated with lenalidomide developed TFR. It was observed that two of the mantle cell lymphoma patients who developed TFR had a mild condition and recovered without any treatment. However, the other patient died of cerebrovascular complications although he was hospitalized owing to the severity of his condition (he had a prolonged reaction and his pain could not be controlled) [5]. While the etiology of TFR is not clearly known, it is thought that some mechanisms may be because of secondary production of cytokines. Studies have shown that lenalidomide does not directly induce apoptosis of or cytokine production by tumor cells, but depending on the dose, it does increase CD40, CD80, CD86, HLA-DR, and CD95 expression by activating B cells [1]. It has been shown that there is distinctive CD40 and CD86 expression in CLL cells on ex vivo lenalidomide administration, and that the expression is associated with TFR [1]. Andritsos et al. [1] have shown that, on pre- and post-treatment evaluation of the lymph nodes, infiltration of Ki67- and CD3-positive, CD8-positive, granzyme B-positive T cells increases. It has been observed that, in patients with Hodgkin lymphoma who are treated with lenalidomide and who develop TFR, cytokines (particularly interleukin [IL]-6, IL7, APRIL, BAFF/BLyS) are released because of B cell activation, and levels of free light chains increase distinctively; these changes are reflected in the clinical properties. Likewise, it has been shown that when the TFR subsides, cytokines and light chain levels decrease to normal levels [5]. Therefore, for patients who develop TFR, it is suggested that dexamethasone be used, as it provides distinctive improvement by especially inhibiting CD40 regulation [1]. The mechanism through which the R-HCVAD treatment leads to TFR is not known, but one of the above-listed mechanisms may play a role.