Clear cell adenocarcinoma of the base of the tongue: a case report and review of the literature

Clear cell adenocarcinoma is an extremely rare tumor of the head and neck region. We report a case of a 75-year-old Caucasian woman with a 10-day history of hemoptysis but with no pain or other significant symptoms. A head and neck computed tomography scan with contrast medium showed an irregular, soft-tissue-like, irregularly enhanced lesion of the base of the tongue extending to its posterolateral portion. The tumor reached the lateral wall of the oropharynx, which showed a nonhomogeneous aspect. The patient underwent resection of the tumor via a conservative transmandibular approach. A clear cell adenocarcinoma of the base of the tongue is rarely a primary malignant lesion; it is more frequently a secondary lesion from a metastatic renal tumor. Because of this neoplasm's relatively slow growth rate and low incidence of metastasis or local recurrence, the gold standard of treatment is complete excision of the tumor with a sufficient tumor-free margin.     

舌根、咽旁肿瘤外科手术径路比较研究

目的：比较舌根、咽旁肿瘤几种主要手术径路的优缺点，重点分析正中-颌舌沟径路。方法：对41例住院且行外科切除术的舌根、咽旁肿瘤病人的手术方法、手术体会、并发症等进行回顾性研究。结果：口内直接入路、口底舌骨上入路、旁侧入路、正中-颌舌沟入路等4种手术径路皆能满足该区域外科手术的需要，达到完全切除肿瘤和即刻修复的目的。4种径路各具不同的特点。结论：合适的外科径路是舌根和咽旁肿瘤外科手术的关键，正中-颌舌沟径路是一种损伤小、组织保护好的外科径路。     

Occult primary tumors. The management of isolated submandibular lymph node metastases

The management of patients with cervical metastases from unknown primary tumors presents a therapeutic challenge to both the head and neck surgeon and radiotherapist. If after careful search the primary tumor remains truly occult, traditional methods of radiotherapy encompassing fields from the base of skull to clavicles are often employed, with significant attendant morbidity. To determine if more limited therapy would be effective in cases of isolated regional lymph node metastases, the patterns of tumor spread to nodes in the submandibular region were studied. A retrospective analysis of 472 radical neck specimens obtained from 1975 to 1985 revealed 19 cases (4.0%) of cervical metastases limited to the submandibular triangle. Sources of these tumors included lip (3), buccal mucosa (4), nasal vestibule (1), floor of mouth (4), alveolar ridge (3), oral tongue (1), and unknown (3). It appears that solitary submandibular nodal metastases predominantly arise from sites in the oral or nasal cavity. This suggests that in patients with isolated submandibular lymph node metastases from occult primary sites, a more conservative therapeutic approach to potential primary sites is indicated after treatment of the metastatic focus.     

Isolated plexiform neurofibroma: treatment with three-dimensional conformal radiotherapy

OBJECTIVES: To present a case of an unusual benign tumor of the tongue treated successfully with radiotherapy. STUDY DESIGN: Case report. METHODS: Retrospective chart review. RESULTS: A 60-year-old man presented with a painful submucosal lesion of the tongue base. Computed tomography showed an infiltrative soft-tissue mass involving the left base of the tongue. Operative biopsy revealed plexiform neurofibroma. Because of the patient's operative risk and the potential morbidity of surgical resection, he was treated with three-dimensional conformal radiotherapy (3DCRT). His treatment was accomplished using a five-field arrangement treating exclusively the mass lesion to a total tumor dose of 60 Gy. After treatment, the patient's tongue pain resolved, and he noted minimal transient xerostomia. Serial follow-up radiographic examinations showed the base of tongue mass to be slightly smaller 4 months after treatment. The most recent follow-up magnetic resonance image reveals a further decrease in size of the mass. The patient is now over 3 years out from treatment. CONCLUSIONS: Solitary plexiform neurofibroma of the tongue base is a rare tumor. These benign neoplasms are usually treated with either observation or surgical excision. This case demonstrates that, when significant symptoms necessitate active management, these lesions may be successfully treated with minimal morbidity using 3DCRT. The ability of this technique to deliver a conformal radiation dose to the tumor volume while sparing the surrounding normal tissues may expand the application of radiotherapy in the treatment of these benign lesions of the head and neck.     

F-18 FDG PET for detection of occult primary tumor in patients with lymphatic metastases of the neck region]

BACKGROUND: The occult primary tumor remains an important diagnostic problem in patients with lymph node metastases despite a large variety of imaging modalities. Therefore, the aim of our study was to evaluate the impact of F-18-FDG-positron-emission-tomography (PET) in these patients. METHODS: The study group included a total of 28 patients aged 39 to 84 years with cervical lymph node metastases of a histologically confirmed squamous cell carcinoma (n = 24) or an undifferentiated carcinoma (n = 4). All patients received 370 MBq F-18-FDG intravenously, and whole-body images were acquired at 60 min p.i. with an ECAT EXACT 47 (921) (Siemens, CTI). All lesions were evaluated either by histology or by CT/MRI. RESULTS: In 12 patients PET did not reveal suspected tumorous lesions. In contrast in 16/28 patients PET showed focal tracer accumulations corresponding to potential primary tumor sites located in the lungs (n = 7), in the region of the tonsilla palatina (n = 5), in the submandibular gland (n = 1), in the nasopharynx (n = 1), in the larynx (n = 1), or at the base of the tongue (n = 1). In nine out of these 16 patients, the primary was confirmed in the lungs in 5 patients, in the larynx, at the base of the tongue, in the nasopharynx, and the tonsilla palatina in one patient each, respectively. Moreover, in 6 out of 16 patients, PET was false-positive. One patient refused further evaluation of PET findings. CONCLUSIONS: In approximately one third of all patients, PET detected the primary tumor site, which significantly influenced further therapeutic procedures. Thus, F-18-FDG-PET may be a valuable diagnostic tool in the detection of the primary tumor in patients with cervical lymph node metastases.     

Modified set back tongue flap: an alternative surgical approach in the reconstruction of defects of the base of the tongue

The base of the tongue after tumor resection can be reconstructed in several ways. In addition to direct suturing of small defects, a variety of local or microvascularly anastomosed flaps have been described. The present paper is aimed at introducing a local tongue flap for the reconstruction of medium-sized defects. Six patients were treated at the hospital between 1999 and 2002 with the above technique to cover defects affecting one-half to three-fourths of the base of the tongue. One patient had secondary bleeding and the other one experienced no complications. Nutrition could be started after 7.5 days (4-12) postoperatively on average. The tracheostomy was closed at 16.2 days (12-22) on average after the initial operation. The hospitalization of the patients was 20.3 days (15-27) on average. The modified set back tongue flap is an alternative in the reconstruction of subtotal defects of the base of the tongue. Easy handling, good functional outcome in swallowing, and the low complication rate were advantageous in the present series.     

Schwannoma (neurilemmoma) of the tongue

CONCLUSION: Schwannoma of the tongue always presents as a painless mass on the lingual surface and exhibits no gender preference. A schwannoma on the base of tongue is usually larger at presentation than one on the tongue itself, since it is asymptomatic initially. Complete resection is achievable and recurrence is uncommon. OBJECTIVES: To describe the patient demographics, presenting signs and symptoms, tumor location, imaging studies, management, and therapeutic outcomes of schwannoma of the tongue, an uncommon tumor of neuroectodermal origin that may arise from any myelinated nerve fiber. PATIENTS AND METHODS: In this retrospective study, records of 10 patients who underwent surgical removal of schwannomas of the tongue or base of the tongue between January 1988 and June 2005 were reviewed and summarized. RESULTS: The records of five men and five women with ages ranging from 9 to 45 years (mean 27.4 years) were reviewed. The locations of the schwannomas were as follows: four on the lateral border of the tongue, two on the tongue tip, one on the ventral side of the tongue, and three on the base of the tongue. The major clinical sign was a painless mass on the lingual surface. For all patients, no tumor recurrence or complication was noted after tumor extirpation.     

Partial neurolysis of the hypoglossal nerve for selective lingual atrophy in a porcine model

OBJECTIVES: Obstructive sleep apnea in children is most commonly treated with adenotonsillectomy; however, in cases of significant tongue base prolapse, this may prove inadequate. Surgical procedures used to increase the retroglossal airway have significant morbidities and low patient acceptability. We theorized that effective reduction of the tongue base can be achieved through partial denervation of the hypoglossal nerve, which is easily accessed in the submandibular space with minimal morbidity. METHODS: We performed a prospective, experimental study in which topographic innervation maps of porcine tongue were generated by stimulating the hypoglossal main trunk and peripheral branches. The effects of complete unilateral nerve sectioning on tongue base volume and linear dimensions were measured and compared to the contralateral control side. In the final stage, only the peripheral nerve that was determined as the main supply to the tongue base was sectioned, and the results were compared to those in the matched controls. RESULTS: A medial branch of the hypoglossal nerve was consistently identified as the main motor supply to the tongue base. Complete denervation resulted in a measurable decrease in the volume of the tongue base as compared to that of controls. Partial neurolysis produced inconsistent changes in 2 subjects, with decreases in linear dimensions that were not proportional to the decrease in volume. Histologically, complete denervation was followed by a significantly greater replacement of muscle with fat and connective tissue as compared with partial neurolysis. CONCLUSIONS: There were inconsistent changes in volume and linear dimensions of the tongue base following partial neurolysis of the hypoglossal nerve over the 3-month experimental period.     

Squamous-cell carcinoma of the tongue: treatment results and prognosis

OBJECTIVE: The aim of the study was to assess the results of curative treatment of patients with squamous cell carcinoma of the tongue and to evaluate survival and predictive factors of recurrence. PATIENTS AND METHODS: A series of 309 patients with squamous cell carcinoma of the tongue treated with curative intent was studied from January 1988 to December 1999. The percentage of oral tongue cancer was 82.2 and the percentage of cancer of base of the tongue was 17.8. Most patients underwent surgical procedure alone or combined with radiotherapy (92%). We performed 252 neck dissections. Bilateral dissections were performed for cancer of the apex linguae, cancer of the base of the tongue, for patients with N2c neck disease and whenever the primary tumor site crossed the median line. Twenty-five patients (8%) were treated with radiation therapy alone. Mean follow-up was 55 months. The functional results were assessed within a minimum of 10 months postoperative follow-up. RESULTS: In 45.2%, there was histological evidence of node invasion with 53.5% of extracapsular node spread in the neck specimens. Extracapsular node spread did not influence survival or recurrences. Occult cervical metastasis in an elective neck dissection in clinically negative necks was found in about 20% of patients with 47% of extracapsular node spread (41% for cancer of mobile tongue and 80% for those of base of the tongue). About 23% of patients with cancer of base of the tongue staged N0 had histological node invasion in controlateral neck nodes. The postoperative mortality rate was 0.9%. The rate of complications was 17%. The cancer recurred in 41.7% of all cases. Twelve percent of all patients had second primary cancers of the upper aerodigestive tract. The overall survival and non-recurrence rates at 2 and 5 years were higher in cancer of oral tongue than in cancer of base of the tongue. Survival rates were better when neck nodes were clinically or histologically negatives and in early-stage carcinomas. Non-recurrence rates were better when nodes were clinically or histologically negatives and when margins of exeresis were not involved. The functional results were better in oral tongue cancer than in base of the tongue cancer. DISCUSSION: Prognosis (survival and non-recurrence rates and functional results) of squamous cell carcinomas of oral tongue was better than prognosis of those of base of tongue. We recommend an aggressive surgical procedure even in patients with neck classed N0 (with reservations for T1 lesions with small depth of invasion): an ipsilateral supraomohyoid neck dissection for cancer of oral tongue and a bilateral supraomohyoid neck dissection for cancer of base of the tongue, cancer of oral tongue which crosses the median line of the oral cavity and cancer of the apex linguae. Postoperative radiotherapy must be performed when margins are positives and/or when nodes are involved with or without extracapsular spread.     

Endoscopic surgery for Warthin's tumor of the nasopharynx

Warthin's tumor almost exclusively occurs in the parotid gland. It is the second most common tumor after the pleomorphic adenoma. The extraparotid or ectopic Warthin's tumor is rare and is normally situated in the submandibular gland, cervical lymph node, lip, cheek, tongue, hard palate, lacrimal gland and larynx. In this paper we present a case of the Warthin's tumor with ectopic localization in the nasopharynx with asymptomatic outcome. A 56-year-old man has been diagnosed for 10-year history of headache. He had no past history of any otolaryngologic disease. The axial and coronal NMR scans showed a round-shaped, cyst-like tumor of approximately 12 mm in diameter. The tumor was in the middle line of the nasopharynx without compression of the Eustachian tubes. The histopathologic diagnosis was consisted with Warthin's tumor. The patient underwent tumor removal using an endoscopic transnasal approach. The Warthin's tumor is a benign lesion with extremely rare occurrence in the nasopharynx. The symptoms are varied and sometimes it could be recognized accidentally. In a very rare cases it may transform to malignant neoplasm. The optional approach to the nasopharynx must be selected after careful consideration of the individual case. An endoscopic power instrumentation surgery are recommended for small lesions.     

Hemimacroglossia caused by isolated plexiform neurofibroma: a case report

OBJECTIVES: The present case is a diffuse isolated plexiform neurofibroma of the tongue that was not associated with neurofibromatosis that we treated with intraoral surgery. STUDY DESIGN: A case report. METHODS: We present a 5-year-old girl with isolated plexiform neurofibroma of the tongue with infiltration of the tongue base and review treatment approaches for this very rare tumor. RESULTS: An intraoral approach appears to be a good alternative method that allows both an acceptable exposure for total excision and limited postoperative sequelae. CONCLUSIONS: Isolated plexiform neurofibroma of the tongue is very rare. It causes a massive macroglossia that is progressive with advance of years and disturbs the patient's speech, occlusion, and tongue movements. Involvement of the tongue may be associated with infiltration into the deep structures of the neck by the tumor, thereby presenting considerable operative difficulties.     

阻塞性睡眠呼吸暂停低通气综合征舌根等离子射频消融术的并发症

目的 探讨阻塞性睡眠呼吸暂停低通气综合征(OSAHS)患者舌根等离子射频消融术并发症的原因及防治措施.方法 2008年3月至2009年12月经多道睡眠监测确诊的OSAHS患者行舌根等离子射频消融术193例,观察并分析其术中及术后出血、感染、舌根血肿、脓肿、疼痛、味觉改变、舌麻木感、舌体运动障碍、言语及吞咽功能障碍发生情况及处理措施.结果 术中未见并发症.术后疼痛186例(96.4%),颌下水肿155例(80.3%),继发性出血1例(0.5%),夜间猝死1例(0.5%),无舌黏膜溃疡、味觉改变、舌麻木感、舌根血肿、舌根脓肿、伸舌偏斜、言语及吞咽功能障碍.患者术后疼痛为轻中度,双氯芬酸钠栓可以起到良好的镇痛效果.继发性出血患者的出血量少,对症处理后未再发出血.夜间猝死1例发生在术后37 h,可能是术后局部水肿、疼痛加重睡眠呼吸暂停及夜间低氧血症,诱发心律失常而导致猝死.结论 舌根等离子射频消融术并发症多表现为疼痛、水肿等轻微反应,通过术后使用抗生素、激素及止痛药物可以有效控制.但是对于合并心肺疾病的重度OSAHS患者而言,舌根等离子射频消融术可诱发心源性猝死,应引起高度警惕.     

阻塞性睡眠呼吸暂停低通气综合征舌根等离子射频消融术的并发症

目的 探讨阻塞性睡眠呼吸暂停低通气综合征(OSAHS)患者舌根等离子射频消融术并发症的原因及防治措施.方法 2008年3月至2009年12月经多道睡眠监测确诊的OSAHS患者行舌根等离子射频消融术193例,观察并分析其术中及术后出血、感染、舌根血肿、脓肿、疼痛、味觉改变、舌麻木感、舌体运动障碍、言语及吞咽功能障碍发生情况及处理措施.结果 术中未见并发症.术后疼痛186例(96.4%),颌下水肿155例(80.3%),继发性出血1例(0.5%),夜间猝死1例(0.5%),无舌黏膜溃疡、味觉改变、舌麻木感、舌根血肿、舌根脓肿、伸舌偏斜、言语及吞咽功能障碍.患者术后疼痛为轻中度,双氯芬酸钠栓可以起到良好的镇痛效果.继发性出血患者的出血量少,对症处理后未再发出血.夜间猝死1例发生在术后37 h,可能是术后局部水肿、疼痛加重睡眠呼吸暂停及夜间低氧血症,诱发心律失常而导致猝死.结论 舌根等离子射频消融术并发症多表现为疼痛、水肿等轻微反应,通过术后使用抗生素、激素及止痛药物可以有效控制.但是对于合并心肺疾病的重度OSAHS患者而言,舌根等离子射频消融术可诱发心源性猝死,应引起高度警惕.     

阻塞性睡眠呼吸暂停低通气综合征舌根等离子射频消融术的并发症

目的 探讨阻塞性睡眠呼吸暂停低通气综合征(OSAHS)患者舌根等离子射频消融术并发症的原因及防治措施.方法 2008年3月至2009年12月经多道睡眠监测确诊的OSAHS患者行舌根等离子射频消融术193例,观察并分析其术中及术后出血、感染、舌根血肿、脓肿、疼痛、味觉改变、舌麻木感、舌体运动障碍、言语及吞咽功能障碍发生情况及处理措施.结果 术中未见并发症.术后疼痛186例(96.4%),颌下水肿155例(80.3%),继发性出血1例(0.5%),夜间猝死1例(0.5%),无舌黏膜溃疡、味觉改变、舌麻木感、舌根血肿、舌根脓肿、伸舌偏斜、言语及吞咽功能障碍.患者术后疼痛为轻中度,双氯芬酸钠栓可以起到良好的镇痛效果.继发性出血患者的出血量少,对症处理后未再发出血.夜间猝死1例发生在术后37 h,可能是术后局部水肿、疼痛加重睡眠呼吸暂停及夜间低氧血症,诱发心律失常而导致猝死.结论 舌根等离子射频消融术并发症多表现为疼痛、水肿等轻微反应,通过术后使用抗生素、激素及止痛药物可以有效控制.但是对于合并心肺疾病的重度OSAHS患者而言,舌根等离子射频消融术可诱发心源性猝死,应引起高度警惕.     

Hyalinizing clear cell carcinoma arising from the base of the tongue

Hyalinizing clear cell carcinoma is a low-grade indolent and rare salivary gland tumor originally described by Milchgrub et al. in 1994. We herein report a case of this tumor of the base of the tongue. A 66-year-old Japanese woman presented with a large painless mass in the throat. Computed tomography and magnetic resonance imaging revealed a 40×30-mm well-defined ovoid tumor arising from the base of the tongue. She underwent tracheostomy followed by a resection of the tumor via the transmandibular approach combined with a right-sided supra-omohyoid neck dissection. Because the tumor invasion of the surrounding tissue was limited, the surgical defect at the base of the tongue was relatively small, and no reconstructive procedure needed to be performed. The tumor was histopathologically diagnosed as hyalinizing clear cell carcinoma of the minor salivary gland. Her postoperative clinical course was uneventful. No aspiration or difficulty upon deglutition was recognized when she started transoral ingestion on the eighth postoperative day. The patient is currently free from disease 21 months after surgery. The pathology, clinical characteristics, and treatment of hyalinizing clear cell carcinoma are bibliographically reviewed.     

Growth and spread of squamous cell carcinoma of the floor of the mouth

Summary Forty-eight specimens of carcinoma of the floor of the mouth were analyzed histologically by step serial sections. Five tumors were so-called superficial spreading carcinomas with large fields of cancerous mucosa, while 43 specimens showed advanced vertical growth into adjacent structures. Three different routes of invasion could be defined. The region of the sublingual gland was the main pathway in 23 cases. The space between the intrinsic muscles of the tongue and the genioglossus muscle was the main direction of infiltration for 14 tumors. Only 6 specimens showed direct invasion into the intrinsic tongue muscles. There was a good correlation between tumor thickness and the occurrence of metastases. Twenty-six patients (54%) showed metastases in regional lymph nodes and 13 patients with submandibular metastases already had developed metastases along the jugular vein. Unilateral or bilateral functional neck dissections remained the standard treatment procedure for all cases with depths of invasion over 5 mm. To avoid local tumor recurrences, patterns of invasion have to be considered. Soft tissue structures like the sublingual gland, intrinsic tongue muscles, genioglosssus muscle and geniohyoid muscle have to be resected routinely. Management of the mandible should be conservative if radiological and clinical investigations are negative. Correspondence to: H. Steinhart     

Successful transoral videolaryngoscopic surgery for leiomyoma in the base of the tongue

Leiomyomas are benign tumors with smooth muscle differentiation that occur most frequently in the uterine myometrium. They are uncommon in the head and neck region. We report a rare case of tongue base leiomyoma successfully resected with transoral endoscopic surgery.A 14-year-old male was found to have a tongue base tumor. The tumor located in the right tongue base. It had a smooth surface and no deep invasion. The tumor was resected with transoral videolaryngoscopic surgery. There were no serious adverse events requiring further intervention. Histologically, the tumor was composed of densely cellular fascicles of spindle-shaped cells with smooth muscle differentiation with diffuse and intense reactivity for α-smooth muscle actin, desmin, calponin, and anaplastic lymphoma kinase on immunohistochemistry. After careful consideration of the differential diagnosis, the tumor was diagnosed a smooth muscle tumor, mostly consistent with leiomyoma.This is the first report of leiomyoma arising from the tongue base that was completely resected by transoral videolaryngoscopic surgery without adverse events. For tongue base tumors, endoscopic transoral surgery can be considered as an option for complete resection without impairment of postoperative function.     

Hyalinizing clear cell carcinoma arising from the base of the tongue

Hyalinizing clear cell carcinoma is a low-grade indolent and rare salivary gland tumor originally described by Milchgrub et al. in 1994. We herein report a case of this tumor of the base of the tongue. A 66-year-old Japanese woman presented with a large painless mass in the throat. Computed tomography and magnetic resonance imaging revealed a 40x30-mm well-defined ovoid tumor arising from the base of the tongue. She underwent tracheostomy followed by a resection of the tumor via the transmandibular approach combined with a right-sided supra-omohyoid neck dissection. Because the tumor invasion of the surrounding tissue was limited, the surgical defect at the base of the tongue was relatively small, and no reconstructive procedure needed to be performed. The tumor was histopathologically diagnosed as hyalinizing clear cell carcinoma of the minor salivary gland. Her postoperative clinical course was uneventful. No aspiration or difficulty upon deglutition was recognized when she started transoral ingestion on the eighth postoperative day. The patient is currently free from disease 21 months after surgery. The pathology, clinical characteristics, and treatment of hyalinizing clear cell carcinoma are bibliographically reviewed.     

Pleomorphic adenoma of the tongue base

INTRODUCTION: Pleomorphic adenoma is the most common lesion of minor salivary glands but is exceptional in the tongue base. REPORT CASE: We report the case of a 70-year-old woman with pleomorphic adenoma of the tongue base. The patient consulted for difficult swallowing, which developed over 4 months. Physical examination and computed tomography revealed a cystic tumour located in the right side of tongue base. The tumor was removed by transoral resection. The histological examination demonstrated a pleomorphic adenoma. DISCUSSION: We will discuss the diagnostic and therapeutically modalities of pleomorphic adenoma of the tongue base with a review of the literature.     

Distribution pattern of the cellular oral immune system in the major and minor salivary glands. Immunocytochemical findings

The cellular distribution of lymphocytes and immunocytes in the major and minor salivary glands was analysed comparatively by a semiquantitative method on mastoids from 53 random autopsies. In a second step, the immunoglobulin producing immunocytes were cytochemically distinguished by their content of IgA, IgG and IgM. In addition to the major salivary glands (parotid, sublingual and submandibular), seven minor salivary gland regions (palate, floor of the mouth, upper lip, lower lip, cheek, retrolingual region and tip of the tongue) were studied. The immunocytochemical differentiation was performed by the avidin-biotin-system; the findings were evaluated morphometrically. The following results were obtained: The incidence of a marked or massive infiltration with lymphocytes and immunocytes, especially in the periductal area, showed the following distribution: floor of the mouth 36%, sublingual gland 27%, cheek 26%, palate 25%, lower lip 12%, other salivary glands less than 10% (tip of the tongue 9%, submandibular gland 8%, parotid gland 6%, retrolingual region 4%). 90% of the immunocytes contained IgA, whereas only 10% showed IgG or IgM. The highest density of IgA producing immunocytes was found in the upper lip, followed by the glands in the cheek and lower lip, the submandibular gland and the glands in the floor of the mouth. The lowest infiltration rate with IgA containing immunocytes was seen in the glands of the tip of the tongue, of the cheek and in the submandibular and parotid glands. The glands of the lips and the cheek predominated with respect to IgG and IgM.(ABSTRACT TRUNCATED AT 250 WORDS)