A peculiar site of leiomyosarcoma: the tongue tip--report of a case

Primary leiomyosarcoma of the tongue is an exceedingly rare malignant mesenchymal tumour, which is hardly ever addressed in the differential diagnosis of a tongue lesion. Only 7 cases of this tumour have been documented in English-language medical literature. This study reports an additional case of primary leiomyosarcoma of the tongue in a 54-year-old woman, presenting with a foreign body sensation at the tip of the tongue with a rapidly and progressively enlarging ulcerative nodule. The patient received tumour excision with a 1-year follow-up without evidence of local recurrence or distant metastasis. Leiomyosarcoma in this unusual site seems to have better prognosis than elsewhere, although the number of cases are too small to draw a conclusion. This study further reviews the literature review and discusses the clinico-pathological presentation of this peculiar rare tongue lesion.     

Leiomyosarcomas of the oral cavity: report of a radiation-associated and a metastatic case

Background

Leiomyosarcoma is rare in the oral cavity, where it may arise as primary, radiation-associated, or metastatic tumor. This article reports two cases of oral leiomyosarcoma, discussing the range of clinicopathological features and the significance of these presentations.

Case report

One case is a radiation-associated leiomyosarcoma arising in the tongue of a 71-year-old male occurring 22?years after radiation therapy for tonsil squamous cell carcinoma that was surgically treated. The other one is a mandible metastasis from a retroperitoneal widespread leiomyosarcoma in a 69-year-old man, who was treated by surgery and chemotherapy but died from the disease.

Discussion

Post-radiotherapy sarcomas of the oral cavity and oral metastasis from soft tissue sarcomas are very uncommon, but based on patient??s clinical history, they should be considered by oral health care providers in order to allow an early diagnosis and proper and timely management. Finally, to the best of our knowledge, this seems to be the first reported case of tongue leiomyosarcoma arising in a previously irradiated field.     

Leiomyosarcomas of the oral tissues: clinicopathologic analysis of 50 cases.

PURPOSE: Primary oral leiomyosarcomas are rare tumors. Information regarding the biological behavior, prognosis, and appropriate management of this neoplasm is lacking in the literature. The purpose of this report was to summarize the data of isolated case reports of primary oral leiomyosarcoma that have been published in the English literature during the past 25 years. The cases of 4 additional new patients who have been treated in our department during the past 10 years are also presented. PATIENTS AND METHODS: The data for 46 patients obtained from 32 individual articles retrieved from the English literature were added to our 4 cases and produced a total number of 50 cases of primary leiomyosarcomas of the oral tissues. Patients were analyzed according to demographic data, anatomic location, type of treatment, and survival. RESULTS: Primary oral leiomyosarcoma may affect any age with peaks of occurrence in the third, sixth, and seventh decades of life. There is no gender predilection. Female patients presented the higher incidence in the third decade, whereas males had an even age distribution. The tumor arises in approximately 70% of the cases in the maxillary and mandibular bones. Radical surgery was the treatment of choice. Radiotherapy and chemotherapy when applied in recurrent tumors had little effect. The most adverse prognostic factor was the positive surgical margins. The 5-year survival was 62% (62.9% for females and 52.6% for males, P > .1968). CONCLUSIONS: Cases of oral leiomyosarcoma appear to be associated with major neurovascular structures of the facial skeleton, as evidenced from the imaging studies of our 4 patients. Primary oral leiomyosarcoma is a rare tumor that should be managed with aggressive surgical resection in order to safeguard curability. Histopathologic diagnosis is greatly facilitated with positive immunohistochemical staining for smooth muscle antigenic markers.     

Effect of tongue reduction on the orthodontic and surgical treatment of dysgnathia.

The results of reduction of tongue size in connection with orthodontic and surgical correction of prognathic anomalies of the jaws are presented. It is shown that reduction of the tongue size combined with orthodontic treatment arrests of tendency to prognathic growth when it is performed early. The results further indicate that the effect of prophylactic reduction of tongue size to check relapse following surgical correction of prognathism or open bite can only be confirmed in cases of genuine macroglossia. Since there was only one relapse in our cases without reduction of tongue size, the indication for preoperative prophylactic reduction of the tongue is limited. Personal results and experience have been compared with the findings published in the current literature.     

Hemangiopericytoma of the tongue: report of case

A case of hemangiopericytoma of the tongue is reported. A comprehensive literature review regarding hemangiopericytoma of the tongue has been presented. Only ten cases of hemangiopericytoma of the tongue have been reported in the literature, including the present case, a 29-year-old male.     

Leiomyosarcoma of the maxilla

A case of leiomyosarcoma in the maxilla is presented together with a review of the literature. The most commonly encountered symptom at the early stage of oral leiomyosarcoma is a slowly enlarging, nonulcerated and painless mass. This tumor shows high incidence of local recurrence or metastasis, and poor prognosis. The histological confirmation of leiomyosarcoma seems difficult in the routine examination using biopsy specimen. Special staining methods and immunohistochemical analysis are useful for diagnosis of this tumor. The tumor slightly responded to chemotherapy consisting of adriamycin and cyclophosphamide.     

Primary oral leiomyosarcoma: A systematic review and update

The aim of this systematic review was to address the clinicopathologic inconsistencies noted with primary oral leiomyosarcoma in the literature by amassing the available data published into a comprehensive analysis. Eligibility criteria included publications of cases with dedicated immunohistochemical work‐up along with radiographs to evaluate location. Based on these criteria, the systematic review compiled 29 cases. Four primary location sites were identified in the reported cases: soft tissue presentation only, soft tissue with bony involvement, bony involvement only, and bony involvement with a soft tissue component. The majority of primary oral leiomyosarcoma cases reviewed showed a soft tissue predilection, which is in contrast to prior reports of jawbones being the most common site. In addition, there was an improved 5‐year survival rate for primary oral leiomyosarcoma limited to the oral cavity and gnathic bones without extension into paranasal sinuses.     

地图舌危险因素的研究进展

地图舌（良性游走性舌炎）是一种常见的浅表性良性舌部炎症,临床表现为丝状乳头剥脱红斑区伴周围黄白条带状微凸边缘,病理为非特异性炎症表现,大多数患者无症状,病损可自愈,常有复发,一般不需药物治疗。当病损累及舌部以外口腔黏膜时称异位性地图舌（游走性口炎）,其临床表现及组织病理学特点均似地图舌。地图舌病因尚不明确,与年龄相关,儿童多发;危险因素复杂,包括免疫因素、遗传因素、过敏体质、精神压力、吸烟、内分泌因素以及缺锌等;可伴发其他疾病如沟纹舌、银屑病、糖尿病、胃肠紊乱、灼口综合征或唐氏综合征等。地图舌是否为银屑病的口腔表征尚存争议,其与沟纹舌关系尚待进一步研究。本文就地图舌危险因素的研究进展作一综述。     

Broken tooth fragments embedded in the tongue: A case report

Tooth fragments embedded in the lip are reasonably common and have been well reported in the literature. Tooth fragments in the tongue are less common with few reported cases (Jacowski & Colas, 1952; Snawder et al., 1979). A case report of multiple tooth fragments in the tongue is presented and a hypothesis suggested as to the cause and positioning of the fragments.     

Primary intestinal-type adenocarcinoma of the tongue

Primary intestinal-type adenocarcinoma (ITAC) of the tongue is a rare neoplasm. The case of a 59-year-old male with a painless mass in the dorsum of the oral tongue diagnosed as primary ITAC is reported here. The particular characteristic of the case was the patient’s long-term medical history of 40 years. The patient underwent resection of the tongue carcinoma through a midline approach and direct suture, with postoperative chemoradiotherapy. Immunohistochemical staining indicated that the tumour was positive for CK20 and negative for CK7. A review of the English-language literature was performed, which identified six cases of primary ITAC and three cases of metastatic ITAC of the tongue. The clinical characteristics and therapeutic methods used in these cases were analysed.     

Lymphangiomas of the tongue

Lymphangiomas of the tongue are relatively uncommon developmental anomalies, which may present as either localised or diffuse lesions. Two cases are reported, and the literature reviewed.     

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目的探讨舌肌内巨大颗粒细胞瘤的临床特点。方法对中国医科大学口腔医学院口腔颌面外科2003—2004年收治的2例舌肌内颗粒细胞瘤患者的临床资料进行回顾性分析,结合文献探讨其临床特征、组织病理学特点、诊断及治疗。结果2例患者均为年轻女性,肿物均位于舌肌内,直径约5cm,为无痛性,病程为渐进性。组织学特点为无明显被膜,瘤细胞体积较大,呈圆形,胞浆丰富,可见较多嗜伊红均质细颗粒,过碘酸-希夫(PAS)染色阳性,瘤细胞呈巢状或腺泡状生长。手术切除后均无复发。结论舌肌内颗粒细胞瘤为良性病变,切除后不易复发。     

Osteoma of the buccal mucosa

Intraoral soft tissue osteomas are uncommon. Thirty cases have been reported in the literature. All except one occurred in the posterior region of the tongue. An additional case involving the buccal mucosa is presented.     

Leiomyosarcoma of gingiva: report of a case and review of the literature

Leiomyosarcoma of the oral region has rarely been reported. A review of the literature yield only 24 cases. The histopathology with special reference to ultrastructural findings and immunohistochemical examination of an additional case of leiomyosarcoma of mandibular gingiva has been presented. The manifestation and symptoms, treatment and prognosis, and the origin of this tumor are discussed. Because of the high rate of local recurrence and metastases, extensive surgical excision and long-term follow-up are mandatory.     

Clinicopathologic characterization of oral angioleiomyomas

OBJECTIVE: The purpose of this study was to better define the clinicopathologic features of oral angioleiomyomas. STUDY DESIGN: A retrospective search was performed for angioleiomyomas among all smooth muscle tumors accessioned from 1963 to 2001 in an oral pathology service. Twelve lesions met histopathologic criteria for inclusion in the study and were combined with 97 additional cases identified from the English language literature, yielding 109 cases for evaluation. RESULTS: The mean age of the patients with oral angioleiomyomas was 45.0 years, with a 1.43:1 male to female predilection. The most frequently reported site was the lip, in 48.6% of patients, followed by the palate (21.1%), buccal mucosa and tongue (each 9.2%), mandible (8.3%), and buccal sulcus, labial sulcus, floor of mouth, and gingiva (each 0.9%). Most mucosal lesions varied in size from a few millimeters to 2 cm, with most central lesions of the mandible measuring greater than 2 cm. Although angioleiomyomas are vascular lesions, only 55.9% of cases appeared red, blue, or purple; the remainder were gray, white, or the color of normal mucosa. Tumors were typically described as painless and manifested a low growth rate. All lesions were well circumscribed and composed of numerous vascular spaces surrounded by thick smooth muscle walls. CONCLUSION: Oral angioleiomyomas are benign smooth muscle tumors with a limited degree of morbidity. Careful histologic inspection is necessary to distinguish these lesions from their malignant counterpart, the leiomyosarcoma. Surgical excision is the treatment of choice, and recurrence is rarely encountered.     

Solitary congenital granular cell lesion of the tongue

Congenital granular cell lesion (CGCL) is well known to appear as the congenital epulis, predominantly on the median maxillary alveolar ridge. This lesion rarely occurs on the tongue, and only 7 cases, including 4 cases with simultaneous occurrence on the alveolar ridge, have been reported in the English literature. In this report, we report a case of congenital granular cell lesion seen on the tongue. The patient was a 7-day-old Japanese girl who presented with a polypoid mass on the anterior ventral surface near the tip of the tongue. The lesion was excised at 4 months, and its histological examination revealed large granular cells. Immunochemical staining by S-100 was negative, and it was diagnosed as congenital granular cell lesion.     

Clinical approach of ankyloglossia in babies: report of two cases

Ankyloglossia is a developmental anomaly of the tongue characterized by a short lingual frenum, resulting in restricted movement of the tongue. Its etiology is undefined and there is no gender preference. Few studies are available in the literature and the diagnosis and management of ankyloglossia in infants remains controversial. We report two cases of infants submitted to lingual frenectomy, emphasizing the management of ankyloglossia and its implications in breast-feeding.     

Primary aspergillosis affecting the tongue of a leukemic patient

We describe a case of primary aspergillosis involving the tongue of a patient with acute myeloid leukemia. Intraoral aspergillosis is very rare and we found only 23 cases reported in the English literature. Clinically it was a 2-cm, ulcerated, grayish lesion on the dorsum of the tongue. Microscopically there was invasion of the epithelium, connective tissue and muscle of the tongue by fungal hyphae branching at 45 degrees angle. The large hyphae were easily seen by H & E stain, and were strongly positive for periodic acid-Schiff and Grocott methenamine. The patient was successfully treated with intravenous amphotericin B. Based on clinical, microscopic and culture data, the diagnosis of primary aspergillosis of the tongue was established. Invasive oral aspergillosis is a potentially lethal disease and it should be considered in immunosuppressed patients.     

Imaging features of primary retropharyngeal leiomyosarcoma with rhabdomyosarcomatous differentiation

Leiomyosarcomas are uncommon malignant neoplasms that account for less than 1 % of all cancers. A primary retropharyngeal leiomyosarcoma is an extremely rare neoplasm that has not been reported in the English-language literature. Herein, we report a case of a 46-year-old male patient with leiomyosarcoma showing focal rhabdomyosarcomatous differentiation arising from the retropharynx and describe the radiological imaging findings and pathological features.     

Bilateral lipomas of the tongue.

Multiple lipomas of the tongue are quite rare and are seldom reported in the literature. Presented herein is the case of a 76-year-old man with four symmetrical asymptomatic bilateral double lipomas of long standing on the tongue.