CASO CLÍNICO

Lingual angioleiomyoma: A case report

Angioleiomioma lingual: A propósito de un caso

I. Peña González¹, L.M. Junquera Gutiérrez², C. Aguilar Lisset³, J.C. De Vicente Rodríguez⁴, P. Villarreal Renedo²

1 Médico Residente. Servicio de Cirugía Oral y Maxilofacial.
2 Médico Adjunto. Servicio de Cirugía Oral y Maxilofacial.
3 Médico Residente. Servicio Anatomía Patológica.
4 Jefe de Sección. Servicio de Cirugía Oral y Maxilofacial.
Hospital Universitario Central de Asturias, Oviedo, España

Correspondence

ABSTRACT

Introduction. Oral leiomyomas are uncommon smooth muscle tumors due to the scant presence of this tissue in the oral cavity. The most common type is angioleiomyoma. Angioleiomyomas usually are asymptomatic tumors that can become malignant, so surgery is necessary. These tumors have a histopathologic diagnosis and the differential diagnosis from their malignant counterparts is important. Surgery is the treatment of choice and recurrence is rare.
Material and method. We report a case of lingual angioleiomyoma, its histopathologic findings, and our literature review.
Discussion. Lingual angioleiomyomas are tumors that are uncommon, usually asymptomatic, and easy to access for surgery. Histopathologic findings show intense positive staining to smooth muscle actin. The differential diagnosis is with other lingual masses, such as abscesses, neuromas and minor salivary gland tumors. Surgery is the treatment of choice and recurrence is rare.
Conclusions. Lingual angioleiomyomas are uncommon benign tumors to consider in the diagnosis of lingual masses. Malignancy must be excluded by adequate histopathologic study.

Key words: Angioleiomyoma; Tongue.

RESUMEN

Introducción. Los leiomiomas orales son tumores del músculo liso poco frecuentes debido a la escasez de este tejido en la boca. La forma más frecuente es el angioleiomioma. Son tumoraciones generalmente asintomáticas, que pueden malignizar, por lo que está indicada su extirpación. Su diagnóstico es anatomopatológico siendo importante diferenciarlas de su forma maligna. El tratamiento es quirúrgico y la recidiva inusual.
Material y método. Se presenta un caso de angileiomioma lingual y sus características anatomopatológicas, relacionando los hallazgos con la literatura existente.
Discusión. Los angioleiomiomas linguales son tumoraciones poco frecuentes, generalmente asintomáticas y de fácil acceso quirúrgico. Los hallazgos anatomopatológicos consisten en una proliferación vascular rodeada de un estroma positivo a la actina músculo liso específica. Ha de tenerse en cuenta en el diagnóstico diferencial de masas linguales tales como abscesos, neuromas y tumores de glándulas salivales menores. El tratamiento de elección es quirúrgico y la recidiva inusual.
Conclusiones. Los angioleiomiomas linguales son tumores benignos poco frecuentes pero que deben ser tenidos en cuenta en el diagnóstico diferencial de masas linguales. Es importante diferenciarlos de su forma maligna a través de su estudio anatomopatológico.

Palabras clave: Angioleiomiomas; Lengua.

Introduction

Leiomyomas are benign tumors of the smooth muscle that are most typically located in the feminine genitourinary system and gastrointestinal tract. They are uncommon in the mouth due to the scarcity of smooth muscle in this area.¹ Stout proposed the most widely accepted hypothesis of the origin of angioleiomyomas in 1937.² According to this author, these tumors develop from the tunica media of oral blood vessels. However, another possible origin may arise be the smooth muscle of the lingual duct located in the circumvallate papilla, a hypothesis proposed by Glas.³

Oral leiomyomas may occur at any age, although they are more common around 30-59 years of age. There is no difference in the incidence in men and women.^4,5

Clinically, leiomyomas occur as small, asymptomatic, well-delimited, spherical masses.^4-7 Fine-needle aspiration biopsy (FNAB) and imaging studies yield non-specific results, so the diagnosis is obtained by excisional biopsy and histopathologic study.⁶

The treatment of choice is surgery and the tumor rarely recurs after surgery.⁶ In cases of large tumors of vascular origin, previous embolization can help to diminish the probability of bleeding during the intervention.⁸

We report a new case of oral angioleiomyoma located in the tongue and review the literature on this pathology.

Clinical case

A 67-year-old man without a personal history of interest was referred to the department for evaluation of a lingual tumor for the last 6 months that was asymptomatic but grew progressively. The mass was located in the left anterior third of the tongue. The largest diameter of the tumor was approximately 5 mm and the tumor was of hard consistency and not adhered to deep planes. The lingual mucosa located over the tumor had a normal appearance. Enlarged lymph nodes were not found on palpation.

Excisional biopsy was performed under local anesthesia due to the small size of the mass. On gross inspection, it was a well encapsulated, easily excisable, ivory-white-colored tumor (Fig. 1).

The study with hematoxylin-eosin revealed tissue characterized by proliferation of small-caliber blood vessels with round lumina and thick walls. Acidophilic spindle cells emanated from the vessels, constituting the rest of the stroma (Fig. 2). An immunohistochemical study was made to establish the type of cells that constituted the tumoral stroma. Firstly, common muscle actin yielded a positive result. Then, to identify the muscle strain, the tissue was stained with smooth muscle actin, which gave an intensely positive result (Fig. 3). This established the diagnosis of angioleiomyoma and ruled out malignancy in light of the absence of cellular atypias and the scant number of mitoses.

Discussion

Smooth muscle tumors of the oral cavity are infrequent. Farman,¹ in 1975, reviewed 7748 smooth muscle tumors from locations throughout the body and found only 0.064% in the oral cavity. More recently, Wang et al.⁶ reviewed 160 cases of leiomyoma, finding 21% in the head and neck but only one intraoral leiomyoma. In 1969, the World Health Organization divided leiomyomas into three histologic subtypes:⁹ solid leiomyomas (true leiomyomas), vascular leiomyomas (angioleiomyomas-angiomyomas), and epithelial leiomyomas (leiomyoblastoma). The case reported here pertained to the vascular leiomyomas group. Angiomyomas occur more frequently in the subcutaneous tissue of the limbs, where they manifested as painful nodes.⁸ Among the leiomyomas of the oral cavity, the most frequent subtype is angiomyoma.^5,10 However, angiomyomas continue to be uncommon tumors in the mouth. In a review by Brooks et al.⁷ in 2002, of 76412 biopsies of the oral cavity, only 12 were angiomyomas (0.016%). Hachisuga et al.¹¹ studied a total of 562 cases of angiomyoma, of which only 8.5% were located in the head and neck. Oral angiomyomas are located more frequently in the cheek, palate and tongue,⁴ with the tongue being the second most frequent site.⁵ The age range was 30 to 59 years and the incidence was similar in men and women.^4,5 We report a new case of oral angiomyoma that was located in the tongue of a 67-year-old male patient, which was described above.

The origin of these tumors may be the smooth muscle of the tunica media of oral blood vessels,^2,5 although their origin in the lingual circumvallate papilla cannot be ruled out.³ For some authors, many angiomyomas are not true tumors but vascular malformations with hyperplasia of the smooth muscle cells. They establish the following evolution of the lesions, beginning with: hemangioma, angioma, vascular leiomyoma, leiomyoma, and solid leiomyoma. Vascular leiomyoma is, therefore, an intermediate stage in this sequence.

Clinically, leiomyomas appear as small (less than 2 cm), asymptomatic masses.^4-6 In our case, the tumor did not present any symptom and measured approximately 5 mm.

The differential diagnosis has to be established with:^5,10 abscess, foreign body, lingual thyroid, hemangioma, lymphangioma, cystic hygroma, retention cysts, papilloma, and benign tumors, eg, fibroma, neurofibroma, fibrous histiocytoma, neurilemmoma, lipoma, granular cell tumor (myoblastoma), and glandular tumors (eg, pleomorphic adenoma).

The results of FNAB and imaging tests are non-specific, which is why excisional biopsy and histopathologic study have to be made for diagnosis.⁶ In the case of angiomyomas, we find vascular spaces surrounded by smooth muscle.^4,6-8 Hematoxylin-eosin staining reveals a proliferation of thickwalled vessels surrounded by stroma of spindle cells. We, like other authors, think that immunohistochemical study with muscle-specific actin is sufficient to establish the diagnosis of angiomyoma and that it is unnecessary to perform another type of stain.⁶ It sometimes can be difficult to distinguish well differentiated leiomyoma from low-grade malignant leiomyosarcoma.^{5, 13} If it is located in the tongue, it is more likely to be benign.⁵ However, the key is the number of mitoses per high-magnification field.^5,13,14 Therefore, the presence of more than 10 mitotic figures per high-magnification field (40x) would suggest malignant behavior.¹³ The presence of fewer than 2 mitotic figures per field is indicative of good prognosis.¹⁴ In our case, the malignancy of the lesion was ruled out based on these principles.

Conclusions

The rate of malignization of oral leiomyoma is high, about 20%, compared with the same lesion in the feminine genitourinary tract.¹ Therefore, although these are generally asymptomatic tumors, exeresis is advisable. As encapsulated, well-circumscribed tumors, surgery does not entail any problem and is the treatment of choice.^6,7 They are usually small tumors and surgery can be performed under local anesthesia, as in our case. In the case of large angiomyomas, previous embolization to reduce the risk of operative bleeding can be beneficial.⁸ The recurrence of angiomyomas is unusual⁶ and malignization has not been reported in the literature.⁷

Correspondence:
Ignacio Peña González
Servicio de Cirugía Oral y Maxilofacial.
Hospital Universitario Central de Asturias.
c/ Celestino Villamil s/n, 33006 Oviedo, España
Email: napego_maxilo@hotmail.com

Recibido: 25.09.07
Aceptado: 14.01.08

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