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Multiple oral leiomyomas in an infant: a rare case.
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MedLine Citation:
PMID:  22701189     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Oral leiomyoma is a benign smooth muscle tumor that occurs most frequently in the uterine myometrium, gastrointestinal tract, and skin. Incidence in the oral cavity is considered uncommon. Most cases are reported in adults, with very few cases described in children. A rare case of multiple leiomyomas localized on the tongue, cheek, and floor of the mouth of an 8-month-old baby is reported. The diagnosis of leiomyoma in the oral cavity is mainly determined by histological studies; however, immunohistochemical tests are recommended in order to differentiate from other tumors. Surgical excision of the lesion appears to be the best treatment option. A review of the literature did not reveal any previously reported case of multiple oral leiomyomas.
Authors:
Efraín Alvarez; María P Laberry; Carlos M Ardila
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Publication Detail:
Type:  Journal Article     Date:  2012-06-03
Journal Detail:
Title:  Case reports in dentistry     Volume:  2012     ISSN:  2090-6455     ISO Abbreviation:  Case Rep Dent     Publication Date:  2012  
Date Detail:
Created Date:  2012-06-15     Completed Date:  2012-08-23     Revised Date:  2013-03-01    
Medline Journal Info:
Nlm Unique ID:  101573242     Medline TA:  Case Rep Dent     Country:  Egypt    
Other Details:
Languages:  eng     Pagination:  804305     Citation Subset:  -    
Affiliation:
Department of Oral and Maxillofacial Surgery, School of Dentistry, University of Antioquia, Calle 64 No. 52-59, Medellín, Colombia.
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Journal Information
Journal ID (nlm-ta): Case Rep Dent
Journal ID (iso-abbrev): Case Rep Dent
Journal ID (publisher-id): CRIM.DENTISTRY
ISSN: 2090-6447
ISSN: 2090-6455
Publisher: Hindawi Publishing Corporation
Article Information
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Copyright © 2012 Efraín Alvarez et al.
open-access:
Received Day: 3 Month: 3 Year: 2012
Accepted Day: 12 Month: 4 Year: 2012
Print publication date: Year: 2012
Electronic publication date: Day: 3 Month: 6 Year: 2012
Volume: 2012E-location ID: 804305
ID: 3371667
PubMed Id: 22701189
DOI: 10.1155/2012/804305

Multiple Oral Leiomyomas in an Infant: A Rare Case
Efraín Alvarez1
María P. Laberry2
Carlos M. Ardila3*
1Department of Oral and Maxillofacial Surgery, School of Dentistry, University of Antioquia, Calle 64 No. 52–59, Medellín, Colombia
2Department of Maxillofacial Surgery, University of Antioquia, Calle 64 No. 52–59, Medellín, Colombia
3Biomedica Stomatology Group, School of Dentistry, University of Antioquia, Calle 64 No. 52–59, Medellín, Colombia
Correspondence: *Carlos M. Ardila: martinardila@gmail.com
[other] Academic Editors: G. Colella, S. S. De Rossi, M. W. Roberts, N. Shah, and S. R. Watt-Smith

1. Introduction

Leiomyomas are benign mesenchymal tumors arising from smooth muscles. They frequently occur in the uterus and gastrointestinal tract, but they can initiate wherever smooth muscle cells subsist. Histologically, there are three types of leiomyomas: leiomyoma (solid), angiomyoma (vascular leiomyoma), and the rare form of epithelioid leiomyoma (leioblastoma). Oral leiomyoma is a rare tumor, with most of the cases noted in adults and few cases reported in children. The first case of a mandibular intraosseous leiomyoma in an 8-month-old baby was recorded by Bertolini et al. [1]. The tumor generally manifests as a slow-growing painless lesion, often of a purplish color. Since leiomyomas cannot be clinically distinguished from fibromas, the diagnosis is exclusively based on the histological findings. In order to accomplish more detailed analysis and more exact differential diagnosis, immunohistochemical studies are recommended. A case of multiple leiomyomas localized on the tongue, cheek, and floor of the mouth with their histologic and immunohistochemical profile is presented. A review of the literature did not reveal any previously reported case of multiple oral leiomyomas.


2. Case Report

An 8-month-old girl was referred to the Department of Oral and Maxillofacial Surgery of the University Hospital San Vicente in Medellín, Colombia, for evaluation of multiple erythematous lesions on the tongue, cheek and floor of the mouth (Figure 1). The lesions had been present for the previous four months. Physical examination revealed well-demarcated nodules measuring around 1 cm in diameter. The overlying mucosa appeared normal and nonulcerated. In order to complete a histological analysis, excisional biopsies were performed. The surgical samples were fixed in 10% buffer formalin for a minimum of 48 hours, embedded in paraffin, and cut at 5 μm to be stained with Hematoxylin-Eosin technique. Microscopically, fusiform cell proliferation with elongated nuclei and eosinophilic cytoplasm was observed. Several blood vessels lined by a thin layer of endothelial cells were observed intercalated in the fascicules (Figure 2). Immunohistochemical techniques were also applied, involving monoclonal antibodies against actin, vimentin, cytokeratin, and the S-100 protein (Figure 3). The immunohistochemical study revealed the expression of vimentin, desmin, muscle specific actin (MSA), and smooth muscle actin (SMA) within the tumor cells. The other markers studied (cytokeratin, AE1/AE3, EMA, S100, and CD34) were negative. Diagnosis of vascular leiomyomas was confirmed. Computerized tomography ruled out bone association, corroborating the completely mucosal allocation of the leiomyomas. Finally, the lesions were removed under general anaesthesia (Figure 4). The patient had a correct evolution without any postoperative incident. The patient returned after a 6-month period, at which time there was no evidence of recurrence.


3. Discussion

To the best of our knowledge, this is the first case of multiple leiomyomas in an infant. Oral leiomyomas are considered uncommon neoplasms, representing only 0.016% to 0.065% of all the leiomyomas [2]. The vascular variant is the most frequent in the oral cavity with 75% of all cases corresponding to this histological type [24]. The greater occurrence of this variation is related to the most frequent source of smooth muscle in the oral cavity, represented by the wall of blood vessels [5]. The highest prevalence of head and neck leiomyoma is observed between 40 and 49 years of age [24] and the lips are the most common site followed by the tongue, cheeks, palate, and gingivae [3]. In the present case, the patient was 8 months old at the moment of her diagnosis and several leiomyomas were localized in the floor of the mouth, representing an unusual case. Similarly, Bertolini et al. [1] reported a leiomyoma in an 8-month-old, but the diagnosis was a mandibular intraosseous lesion. The gender preference for females is in agreement with the literature [15].

Most of the vascular leiomyomas are nodular, painless, slow growth lesions, less than 2 cm in diameter, and of a color that can vary from white to blue [6]. This case corroborates with what has been published previously. From the clinical appearance, it is very difficult to differentiate a leiomyoma from other mesenchymal tumors such as fibromas, neurofibromas, lipomas, mucoceles or the leiomyosarcoma, the malignant counterpart [15, 7], and therefore the final diagnosis of oral leiomyoma is mainly determined by a histological and immunohistochemical study.

The vascular leiomyoma is characterized by a well-defined proliferation of mesenchymal tapered cells with eosinophilic cytoplasm and elongated basophilic nuclei that show tapered endings [4]. Immunohistochemically, leiomyomas are reactive with vimentin, desmin, α-smooth muscle actin, and muscle specific actin. In this context, the immunohistochemical technique is an important aid, in which these tumors express immunoreactivity for SMA and negativity for the S-100 protein. In this case, fusiform and epithelial-like cell areas were observed and the immunohistochemical study revealed the expression of vimentin, desmin, MSA, and SMA within the tumour cells. Immunohistochemical findings similar to those noted in the case presented here have also been reported [4, 6].

Wide surgical resection is the most reported treatment in reviewed literature with successful results. However, to remove the tumor, a carbon dioxide laser has been used [7]. Recurrence rate is very low if complete resection is achieved [8].


Conflict of Interests

The authors declare that thier is no conflict of interests.


Ethical Approval

Ethical approval was not required.


References
1. Bertolini F,Bianchi B,Corradi D,Caradonna L,Sesenna E. Mandibular intraosseous leiomyoma in a child: report of a caseJournal of Clinical Pediatric DentistryYear: 200327438538712924741
2. Brooks JK,Nikitakis NG,Goodman NJ,Levy BA. Clinicopathologic characterization of oral angioleiomyomasOral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and EndodonticsYear: 2002942221227
3. Bhattacharyya I,Summerlin DJ,Cohen DM,Ellis GL,Bavitz JB,Gillham LL. Granular cell leiomyoma of the oral cavityOral Surgery, Oral Medicine, Oral Pathology, Oral Radiology and EndodontologyYear: 20061023353359
4. Gaitan-Cepeda LA,Quezada-Rivera D,Tenorio-Rocha F,Leyva-Huerta ER,Mendez-Sánchez ER. Vascular leiomyoma of the oral cavity. Clinical, histopathological and immunohistochemical characteristics. Presentation of five cases and review of the literatureMedicina Oral, Patologia Oral y Cirugia BucalYear: 2008138E483E488 Article ID 1111111504..
5. Hachisuga T,Hashimoto H,Enjoji M. Angioleiomyoma: a clinicopathologic reappraisal of 562 casesCancerYear: 19845411261306722737
6. Nonaka CFW,Pereira KMA,da Costa Miguel MC. Oral vascular leiomyoma with extensive calcification areasBrazilian Journal of OtorhinolaryngologyYear: 2010764, article 539
7. Janas A,Grzesiak-Janas G,Sporny S. Removal of oral cavity leiomyoma with carbon dioxide laserQuintessence InternationalYear: 2008391e1e418551205
8. Wertheimer-Hatch L,Hatch GF III,Hatch KF,et al. Tumors of the oral cavity and pharynxWorld Journal of SurgeryYear: 200024439540010706911

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