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      Oral Angioleiomyoma in Early Childhood Patient: A Case Report of an Uncommon Lesion and Literature Review

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          A bstract

          Leiomyoma is a benign soft tissue tumor originating from smooth muscle, rarely seen in the oral cavity, due to the scarcity of this tissue in the mouth. The tumor may occur at any age, without sex predilection. Rare reports of this lesion have been in pediatric patients. The lesion is typically asymptomatic and has slow growth, the final diagnosis is defined by histopathological and immunohistochemical examination. Treatment involves resection of the lesion. The lesion is classified histologically as solid leiomyomas, angioleiomyomas (AL), or epithelioid leiomyomas. We report the case of a 1-year-old female child with a painless papule on the hard palate. Histopathological examination revealed biphasic LA showing proliferation of spindle-shaped nuclear cells in straw-shaped bundles or a concentric design. This is the fourth documented case of AL in early childhood.

          How to cite this article

          Bezerra TMM, Chaves FN, Carvalho FSR, et al. Oral Angioleiomyoma in Early Childhood Patient: A Case Report of an Uncommon Lesion and Literature Review. Int J Clin Pediatr Dent 2021;14(6):828–832.

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          Angioleiomyoma: a clinical, pathological and radiological review.

          Nausheen F Khan, Shreyansh Sutaria, S R Annapureddy … (2004)
          Angioleiomyoma is a benign tumour arising from the vascular smooth muscle (tunica media) and presents commonly between third and fifth decades of life. Although there are sporadic reports about this tumour in the literature, none describes all the information in detail. This review is an attempt to collate all the facts in one concise article. Angioleiomyoma presents as a painful mass in approximately 60% of the cases. One of the distinct clinical feature noted is the increase in size of the swelling with physical activity of the involved part, especially in the hand. It should be considered in the differential diagnosis of painful nodular lesions of the extremity. Pre-operative diagnosis is difficult, but with a high index of suspicion and awareness, it is possible. The use of ultrasound and magnetic resonance imaging should be considered. It causes minimal morbidity and excision is usually curative. Histological examination using smooth muscle Actin stain portraits the smooth muscle bundles clearly.
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            Clinicopathologic characterization of oral angioleiomyomas.

            Steve Goodman, W. K. Brooks, Gary A. Levy … (2002)
            The purpose of this study was to better define the clinicopathologic features of oral angioleiomyomas. 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. 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. 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.
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              Recurrent oral angioleiomyoma

              V. G. Mahima, Karthikeya Patil, H. S. Srikanth (2011)
              Angioleiomyomas are vascular variant of leiomyomas which are benign tumors of smooth muscle. They are exceedingly rare in the oral cavity. Malignant transformation of these tumors has also been reported occasionally which warrants knowledge of this soft tissue tumor. A 57 year old male patient reported with a 15 day history of an asymptomatic growth that had started insidiously in his lower left back tooth region. Clinical examination revealed a solitary, oval, sessile growth in the mandibular left retro molar region. Excisional biopsy was suggestive of Angioleiomyoma. A recurrence of the same was noted two months later which was also histopathologically reported as Angioleiomyoma. The same was confirmed using special stains. This case reports an unusual presentation of Angioleiomyoma with regards to both recurrence as well as rapid growth. It is important to be well aware of this uncommon entity as these tumors often can mimic or transform into malignancy. Precise clinicopathological examinations are therefore invaluable in establishing an accurate diagnosis and delivering suitable treatment.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Int J Clin Pediatr Dent
                Journal ID (iso-abbrev): Int J Clin Pediatr Dent
                Journal ID (publisher-id): IJCPD
                Title: International Journal of Clinical Pediatric Dentistry
                Publisher: Jaypee Brothers Medical Publishers
                ISSN (Print): 0974-7052
                ISSN (Electronic): 0975-1904
                Publication date (Print): Nov-Dec 2021
                Volume: 14
                Issue: 6
                Pages: 828-832
                Affiliations
                [1 ] Dental Clinic Department, Postgraduate Program in Dentistry, Federal University of Ceará, Fortaleza, Ceará, Brazil
                [2,3 ] School of Dentistry, Postgraduate Program in Health Sciences, Federal University of Ceará, Sobral, Ceará, Brazil
                [4 ] Division of Oral and Maxillofacial Surgery, Postgraduate Program in Dentistry, Federal University of Ceará, Fortaleza, Ceará, Brazil
                [5,6 ] Division of Oral Pathology, Postgraduate Program in Dentistry, Federal University of Ceará, Fortaleza, Ceará, Brazil
                [7 ] Department of Morphology, Postgraduate Program in Dentistry, Federal University of Ceará, Fortaleza, Ceará, Brazil
                Author notes
                Filipe N Chaves. Rua Conselheiro José Júlio, S/N, Centro, Sobral, Ceará – CEP: 62.010-820. Phone/Fax: (55) (88) 3695-4622, e-mail: filipenobrechaves@ 123456gmail.com
                Article
                DOI: 10.5005/jp-journals-10005-2082
                PMC ID: 8783224
                SO-VID: 28f64c01-574b-4c88-8a04-f4a28aa27119
                Copyright © Copyright © 2021; The Author(s).
                License:

                © The Author(s). 2021 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( https://creativecommons.org/licenses/by-nc/4.0/), which permits unrestricted use, distribution, and non-commercial reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Categories
                Subject: Case Report

                Keywords: angioleiomyoma,early childhood,palate,pediatric patient,smooth muscle tumor
                Data availability:
                Keywords: angioleiomyoma, early childhood, palate, pediatric patient, smooth muscle tumor

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