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Rhabdomyosarcoma of the Oral Cavity: a Case Report

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Rhabdomyosarcoma of the Oral Cavity: a Case Report Published online: 2019-09-30 Rhabdomyosarcoma of the Oral Cavity: A Case Report Ozkan Miloglua Sare Sipal Altasb Mustafa Cemil Buyukkurtc Burak Erdemcid Oguzhan Altune ABSTRACT Rhabdomyosarcoma (RMS), a tumor of skeletal muscle origin, is the most common soft tissue sarcoma encountered in childhood and adolescence. The common sites of occurrence are the head and neck region, genitourinary tract, retroperitonium, and, to a lesser extent, the extremities. In the head and neck region, the most commonly affected sites are the orbit, paranasal sinuses, soft tissues of the cheek, and the neck. RMS is relatively uncommon in the oral cavity, and the involve- ment of the jaws is extremely rare. Here, we report a case of oral RMS in a 13-year-old child and describe the clinical, radiological, histopathological, and immunohistochemical findings. (Eur J Dent 2011;5:340-343) Key words: Mouth neoplasm; Oral pathology; Alveolar rhabdomyosarcoma; Radiotherapy; Che- motherapy. INTRODUCTION Rhabdomyosarcoma (RMS), which was first tissue neoplasm of skeletal muscle origin. It ac- described by Weber in 1854, is a malignant soft counts for 6% of all malignancies in children un- der 15 years of age.1 The most commonly affected a Department of Oral Diagnosis and Radiology, Faculty of areas are the head and neck region, genitourinary Dentistry, Ataturk University, Erzurum, Turkey. tract, retroperitonium, and, to a lesser extent, the b Department of Pathology, Faculty of Medicine, Ataturk extremities.2 The head and neck RMSs are ana- University, Erzurum, Turkey. c Department of Oral and Maxillofacial Surgery, Faculty tomically divided into 2 categories: parameningeal of Dentistry, Sifa University, Izmir, Turkey. (including RMS of the nose, nasopharynx, parana- d Department of Radiation Oncology, Faculty of Medicine, sal sinuses, middle ear, mastoid, infratemporal Ataturk University, Erzurum, Turkey. e Department of Oral Diagnosis and Radiology, Faculty of fossa, and pterygopalatine fossa) and nonparame- Dentistry, Inonu University, Malatya, Turkey. ningeal (including RMS of the scalp, orbit, parotid gland, oral cavity, oropharynx, and larynx). RMS of Corresponding author: Ozkan Miloglu Department of Oral Diagnosis and Radiology, the oral cavity accounts for 10–12% of all the head Faculty of Dentistry, and neck RMS cases.3 Ataturk University, Erzurum, Turkey. Clinically, the manifestations of RMS may vary Phone: +90 442 2311805 Fax: +90 442 2360945 from a small cutaneous nodule on the face to an E-mail: [email protected] extensive fast-growing facial swelling, which may European Journal of Dentistry 340 Miloglu, Altas, Buyukkurt, Erdemci, Altun be painless or occasionally associated with pain, for CD99, chromogranin, S 100, HMB 45, EMA, and trismus, paresthesia, facial palsy, and nasal dis- Pan CK. After performing the standard diagnos- charge.1,4 tic work-up, the tumor was diagnosed as alveolar On the basis of the histological findings, 4 broad RMS. She was referred to the Pediatric Oncology subtypes of RMS have been identified: botryoid and Department, and the proposed treatment plan spindle cell RMS, embryonal RMS, alveolar RMS, was a combination of chemotherapy, including ifo and undifferentiated RMS.5,6 The histogenesis of sfamid+vincristine+actinomycine-D (IVA regimen) RMS is still unclear, but the most widely accepted and Mesna, and radiotherapy. Although the tumor hypothesis is that RMS arises due to proliferation showed dramatic regression after the initial 12 of embryonic mesenchymal tissue.7 weeks of chemotherapy, the patient’s parents re- In this report, we present a case of oral RMS fused external beam radiation therapy. During the arising within the left cheek mucosa and involv- post-chemotherapy radiographic evaluation, the ing the mandibular bone in a 13-year-old girl, and tumor was found to have regressed. However, af- we describe the clinical, radiological, histopatho- ter 6 months, she experienced an increase in pain logical, and immunohistochemical features of this and mass localization, limitations in temporoman- RMS. dibular articulation, and loss in weight; therefore, she reconsulted the Oncology Clinic. Surprisingly, CASE REPORT the tumor was found to have rapidly increased in A 13-year old girl was referred to our institu- size, almost reaching the pretreatment size, indi- tion in August 2006 for the investigation of pain- cating the reversal of the effects of chemotherapy. ful swelling in her mouth. On history taking, her Owing to this development, she was administered parents reported that the disease had started as chemotherapy with carboplatin+epirubisin+vincris a painless swelling in May 2006, and that she had tine for 8 weeks. Following the completion of che- received oral and parenteral antibiotics for the motherapy, she was treated with external beam past 4 months; however, the treatments were in- radiation therapy (5,400 cGY) for 8 weeks. Despite effective, and she was therefore referred to us. the treatment, the tumor continued to increase in Written informed consent was obtained from her size, and she died 2 years after the initial treat- parent for further investigations. ment. Clinical examination showed severe facial asymmetry. A large, firm, and tender swelling DISCUSSION was noted on the left cheek and it caused pain. After neuroblastoma and Wilms tumor, RMS The skin had appeared stretched and inflamed. is the third most common extracranial malignant The opening of the mouth was partly restricted. tumor that affects children. Of all soft tissue sar- Intraoral examination revealed an extensive mass comas, RMS accounts for only 2–5% tumors in involving the buccal mucosa (Figure 1). Magnetic adults but approximately 60% tumors in children.8 resonance imaging (MRI) showed an infiltrative The incidence of RMS is highest in children aged large soft tissue lesion in the maxillary sinus, in- 1–4 years, lower in children aged 10–14 years, and fraorbitary space, pterygopalatine fossa, pterygoid lowest in those aged 15–19 years.5,6 Our patient plates, and the ramus, which caused displace- was 13 years old and did not fall in the very high- ment of the adjacent structures (Figure 2). An risk category. incisional biopsy was made. Histological exami- The head and neck region is the most common nation of the specimen showed clusters of small site for RMS, with the orbit being the most fre- round cells with hyper chromatic nuclei and eo- quent primary site. The most common site of in- sinophilic cytoplasm separated by fibrovascular volvement in the oral cavity is the tongue followed septae (Figure 3). The morphologic features of the by the soft palate, hard palate, and buccal muco- cells indicated a differential diagnosis compris- sa.9 In our patient, the buccal mucosa, mandibular ing RMS, Ewing’s sarcoma, malignant melanoma, alveolar ridges, and hard palate were all involved. and an epithelial tumor. The neoplastic cells were The soft tissue was more damaged than the bone, strongly positive for vimentin, desmin, myoglobin, suggesting that the tumor primarily originated in and muscle specific-actin (Figure 4) and negative the buccal mucosa. July 2011 - Vol.5 European Journal of Dentistry 341 Oral rhabdomyosarcoma In general, most patients have an advanced phism noted was critical for differentiating RMS disease even at the stage of initial presentation from Ewing’s sarcoma. The presence of an alveo- because RMSs are known to show rapid growth lar pattern, pleomorphism, cohesive nature of the and the patients generally tend to delay medical cells, and the absence of lymphadenopathy ruled consultation.5,6 In our case, when the patient pre- out the diagnosis of lymphoma. In this respect, the sented at our clinic, she had severe facial asym- most critical differential diagnosis is neuroblas- metry and experienced difficulty in opening the toma. A diffuse pattern of small round cells and mouth. In cases of RMS, radiographic examination the presence of rosettes/pseudorosettes with pale reveals the size of the lesion, space relation, and eosinophilic material seen in cases of neuroblas- the extent of bone destruction, while MRI reveals toma may suggest alveolar RMS. High level of uri- the affected areas such as the mandible, condyle, nary catecholamines observed in neuroblastoma maxillary sinus, and infra-orbitary space. is critical to rule out this tumor.8,9 A careful histological examination is required Prognosis of RMS is relatively poor compared to to differentiate such lesions from other more fre- that of other oral soft tissue malignant lesions10,11 quent and aggressive lesions affecting the con- and depends on the clinical staging and the ana- cerned site. In our case, the marked pleomor- tomic site of the tumor.10 An early and accurate Figure 1. Intraoral aspect showing extensive mass involving the buccal mucosa. Figure 2. Magnetic Resonance Imaging (MRI) scan showing an infiltrative large soft tissue lesion in the maxillary sinus, infra-orbitary space, pterygopalatine fossa, pterygoid plates, and the ramus causing displacement of the adjacent structures. Figure 3. Photomicrograph showing a sheet of mesenchymal cells in a myxoid stro- Figure 4. Positive immunohistochemical staining for muscle specific-actin (strep- ma (Hemotoxylin/Eosin (H/E) x200). tavidin biotin x200). European Journal of Dentistry 342 Miloglu, Altas, Buyukkurt, Erdemci, Altun diagnosis of the tumor and a combined therapeu- 2. Durães GV, Jham BC, Mesquita ATM, dos Santos CRR, Mi- tic approach involving surgery, chemotherapy, randa JL. Oral embryonal rhabdomyosarcoma in a Child: A and radiation therapy are known to dramatically case report with immunohistochemical analysis. Oral On- improve the survival rates, as seen in cases re- col Extra 2006;42:105-108. corded over the past 20 years.8 Effective surgical 3. Al-Khateeb T, Bataineh AB. Rhabdomyosarcoma of the excision is challenging in cases of RMS of the head oral and maxillofacial region in Jordanians: a retrospective or neck region owing to the involvement of other analysis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod crucial structures in these locations. Therefore, 2002;93:580-585. we planned a multidisciplinary approach with in- 4.
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