Tongue

Amyloidosis: A rare case with nodular lesions on the tongue and buccal mucosa

October 23, 2013     Evren Erkul, MD; Hakan Cincik, MD
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The pathogenesis of amyloidosis appears to be multifactorial, but the common final pathway is identical in all forms of the disease; i.e., the production of amyloid fibrils in the extracellular matrix.

Oral hairy leukoplakia in an HIV- negative patient

June 11, 2013     Evan M. Graboyes, MD; Clint T. Allen, MD; Rebecca D. Chernock, MD; and Jason A. Diaz, MD
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Abstract

We report the case of a 31-year-old woman with systemic lupus erythematosus who was found to have oral hairy leukoplakia (OHL). She was on immunosuppressive therapy but was human immunodeficiency virus (HIV)-negative. OHL has been previously reported in HIV-negative patients who were immunosuppressed for other reasons, such as solid organ or hematopoietic stem cell transplantation, hematologic malignancies, or systemic diseases. To the best of our knowledge, this is the first case of OHL in an HIV-negative patient reported in the otolaryngology literature. It adds to the growing list of cases of OHL in HIV-negative patients and serves as a reminder to physicians to include OHL in the differential diagnosis for oral cavity lesions in all immunosuppressed patients. The article also summarizes the current state of knowledge about the pathogenesis of OHL, its relation to the Epstein-Barr virus, and the treatment options.

Intramuscular lipoma of the tongue masquerading as angioedema

January 24, 2013     Ali Amirzadeh, MD; William Klaustermeyer, MD
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Abstract

In most cases, the diagnostic evaluation of angioedema is challenging, as there are many possible etiologies. We report a case of an infiltrating lipoma of the tongue that masqueraded as angioedema. The patient, a 68-year-old man, presented with tongue swelling that had followed a waxing and waning course over a 6-month period. Physical examination showed a diffusely enlarged tongue with no discrete mass. A laboratory evaluation for angioedema was unremarkable. After the patient's condition did not respond to treatment with antihistamines and oral prednisone, a further workup was initiated. Magnetic resonance imaging of the neck and computed tomography of the oral cavity revealed only diffuse enlargement of the tongue. The patient underwent a tongue biopsy, which identified the cause of the swelling to be an infiltrating lipoma of the tongue. Clinicians should be aware that other causes of tongue swelling may mimic angioedema.

Bipolar radiofrequency-induced interstitial thermoablation for oral cavity vascular malformations: Preliminary results in a series of 5 children

October 31, 2012     Senol Civelek, MD; Ibrahim Sayin, MD; Ibrahim Ercan, MD; Burak Omur Cakir, MD; Suat Turgut, MD
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Abstract

We report our assessment of the effectiveness of bipolar radiofrequency-induced interstitial thermoablation (BRIT) for the treatment of certain oral cavity vascular malformations in 5 children. Two of these patients had lymphangiomatous macroglossia (LM), 1 had lymphangioma circumscriptum (LC), and 2 had a venous malformation (VM). Each patient underwent BRIT at least twice; treatment was delivered at 4- to 8-week intervals according to each patient's circumstances. The 2 patients with LM required three treatment sessions; although their tongue volume decreased after each session, both still required a partial glossectomy to achieve a satisfactory reduction in volume. The patient with LC underwent two BRIT treatments, which reduced the size of the lesion by half; the remainder was excised. The 2 patients with a VM (1 buccal and 1 lingual) responded well to BRIT, and their malformations almost completely disappeared. Our early results with BRIT suggest that it is an effective treatment for oral cavity vascular malformations-more so for patients with venous rather than lymphangiomatous lesions.

Hypoglossal nerve tumor: A rare primary extracranial meningioma of the neck

October 31, 2012     Abu Bakar Zulkiflee, MS; Narayanan Prepageran, FRCS; Omar Rahmat, MS; Pailoor Jayalaskhmi, MPath, FRCPath; Tengku Sharizal, MS
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Abstract

We report a case of primary extracranial meningioma arising from the hypoglossal nerve in a 54-year-old man who presented with a 9-month history of hoarseness and progressive dysphagia. He had also noticed that his tongue was deviated to the left and, as a result, he was having difficulty pronouncing words. Examination revealed fasciculation and muscle wasting on the left side of the tongue. Other cranial nerve functions were normal. Contrast-enhanced computed tomography detected a heterogeneous mass that had arisen above the bifurcation of the left common carotid artery and had extended to near the skull base. Transcervical excision of the tumor was performed, and histopathology identified it as a meningioma of the hypoglossal nerve. The patient recovered uneventfully, and he was without recurrence at more than 2 years of follow-up. A primary extracranial meningioma is extremely rare, and its presentation may be subtle. A thorough investigation is necessary to avoid fatal compressive symptoms.

Development of hemangioma in a tongue harboring long-standing angiokeratoma circumscriptum

October 31, 2012     J. Madana, MS, DNB; Deeke Yolmo, MS; S. Gopalakrishnan, MS; Sunil Kumar Saxena, MS
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Abstract

Angiokeratoma is a very rare vascular lesion of the papillary dermis. It is characterized by vascular ectasia with overlying epidermal hyperkeratosis. The systemic form of angiokeratoma is associated with Fabry disease and fucosidosis. There are several localized forms. Tongue involvement is uncommon. Hemangiomas are tumors made up of capillaries; they grow by active endothelial proliferation as opposed to expansion of vascular spaces in vascular malformations. Lingual hemangiomas are usually indolent, but they can cause cosmetic deformities, recurrent hemorrhage, and functional problems with speaking, mastication, and deglutition. We report a case of angiokeratoma of the tongue with an underlying hemangioma in a 30-year-old woman. Angiokeratomas have been reported to develop over arteriovenous malformations and in the area of lymphangioma circumscriptum following repeated local trauma. To the best of our knowledge, the development of a lingual hemangioma in a patient with long-standing angiokeratomatous lesions has not been previously reported in the literature.

A comparison of unilevel and multilevel surgery in obstructive sleep apnea syndrome

August 10, 2012     Ümit Tunçel, MD; Hasan Mete İnançlı, MD; Şefik Sinan Kürkçüoğlu, MD; Murat Enoz, MD
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Abstract

We conducted a retrospective study of 35 adults who had undergone tongue base suspension in addition to other surgery for the treatment of obstructive sleep apnea syndrome (OSAS). Our goal was to determine the most effective surgical approach by comparing these patients’ preoperative apnea-hypopnea index (AHI) with their postoperative scores at 6 months. The 35 patients were divided into three groups based on a preoperative assessment of their level of airway obstruction; the assessment included a determination of AHI and endoscopic findings during the Müller maneuver. Patients in group 1 (n = 9) exhibited unilevel obstruction, and they were treated with lateral pharyngoplasty; patients in group 2 (n = 13) also had unilevel obstruction, and they underwent Z-palatoplasty; patients in group 3 (n = 13) had multilevel obstruction, and they underwent combined surgery. Postoperatively, all three groups exhibited statistically significant improvements in AHI, but we determined that of the three approaches, multilevel surgery was significantly more effective (p < 0.001).

Primary solitary extramedullary plasmacytoma of the tongue

July 5, 2012     Cem Onal, MD; Ezgi Oymak, MD; Aysun Uguz, MD; Melek Ergin, MD
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Abstract

Extramedullary plasmacytomas are rare malignancies. Most cases (80%) are seen in the head and neck region, where they represent 1% of all head and neck malignancies. We report a case of an extramedullary plasmacytoma of the tongue that was treated successfully with three-dimensional conformal radiotherapy. The patient was a 50-year-old woman who was admitted to our hospital with bilateral pain and ulceration on the sides of her tongue. Findings on magnetic resonance imaging and positron-emission tomography did not demonstrate any mass within the tongue or any lymphatic or distant metastasis. A tissue biopsy identified a plasma cell neoplasm. The patient was treated with a total dose of 50 Gy delivered in 2-Gy daily fractions. After 54 months of post-treatment follow-up, she exhibited no sign of systemic myeloma or local recurrence. This case is presented not only for the rarity of the tumor type, but also for its unusual location.

Subglottic thyroglossal duct cyst: A rare intralaryngeal presentation

July 5, 2012     Regi Kurien, MS; Rajiv Michael, MS, DLO
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Abstract

Thyroglossal duct cysts are common midline neck swellings that can present at any site along their migratory pathway. They are frequently situated just below the hyoid bone. Extension to the subglottic area is very rare; such an unusual presentation can complicate the diagnosis of a thyroglossal duct cyst. We report the case of a 30-year-old man who presented with a subglottic thyroglossal duct cyst and associated laryngeal symptoms. To the best of our knowledge, only 2 similar cases have been previously reported in the literature, both of which occurred in 2-year-old boys. We believe, therefore, that ours is the first reported case of a subglottic thyroglossal duct cyst in an adult. We discuss the clinical presentation, diagnosis, and treatment of our patient, and we summarize the literature on intralaryngeal thyroglossal duct cysts.

Orofaciodigital syndrome

January 25, 2012     Baris Naiboglu, MD, Cagatay Oysu, MD, and Tanju Gokceer, MD
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Abstract

Orofaciodigital syndrome is a very rare entity with X-linked dominant inheritance characterized by oral, facial, and digital anomalies. Thirteen different types have been described in the literature to date. Of these, orofaciodigital syndrome type I has the highest incidence. Renal and central nervous system malformations may accompany the oral, facial, and digital anomalies. We report a case of orofaciodigital syndrome type I in a 9-year-old girl. The patient was admitted with a complaint unrelated to the syndrome. The coexistence of an oral anomaly with a digital anomaly in this patient led us to search for other possible anomalies. Ultrasonography revealed a diagnosis of polycystic kidneys. Physicians must be mindful of the external appearance of patients with this syndrome and be aware of life-threatening anomalies possibly associated with it.

Primary extranodal T-cell non-Hodgkin lymphoma of the tongue

January 25, 2012     J. Madana, MS, DNB, Deeke Yolmo, MS, Sunil Kumar Saxena, MS, and S. Gopalakrishnan, MS
article

Abstract

The most common sites of extranodal non-Hodgkin lymphoma (NHL) are the gastrointestinal tract and the head and neck region. Head and neck involvement accounts for 8 to 13% of all extranodal lymphomas. Primary NHLs of the oral cavity, especially in the tongue, are extremely rare, which makes it difficult to understand their biologic behavior. Extranodal NHLs of the tongue usually present as a nodular lesion of the B-cell type. The T-cell variant of tongue NHL with an ulcerated exophytic presentation is extremely rare. We report such a case in a 37-year-old woman.

Glossal abscess as a complication of tongue-base suspension surgery

December 15, 2011     Bobby A. Tajudeen, MD, Biana G. Lanson, MD, and Pamela C. Roehm, MD, PhD
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Abstract

Approximately 60 cases of tongue abscess have been reported in the English-language literature over the past 30 years. We report what we believe is the first case of a glossal abscess that arose as a complication of tongue-base suspension surgery. The patient was a 31-year-old man who presented with a several-day history of odynophagia, tongue swelling, voice changes, and increased snoring. Two years earlier, he had undergone a tongue-base suspension procedure for the treatment of obstructive sleep apnea. Computed tomography (CT) revealed a tongue abscess. During peroral incision and drainage, a knotted 0 Prolene suture was discovered within the abscess cavity. The suture was removed, the area was thoroughly irrigated, the drain was placed in the abscess cavity, and the incision was loosely closed. On postoperative day 5, repeat CT revealed resolution of the abscess, and the patient was discharged on oral antibiotics. Although glossal abscess is very rare, physicians should consider it in the differential diagnosis of any patient who presents with lingual swelling following tongue-base suspension surgery.

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