Rhabdomyosarcoma of the Tongue: Report of a Case and Review of the Literature

Kebudi, Rejin; Özdemir, Gül Nihal

doi:10.3109/08880011003749678

Cited by 12 publications

(19 citation statements)

References 19 publications

(39 reference statements)

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“…In the Intergroup Rhabdomyosarcoma Studies(IRS) I, II and III, only 7 rhabdomyosarcomas of the tongue were registered, representing 0.34% of IRS participants [10] . Rejin Kebudi et al summarized a total of 13 cases of lingual RMS reported in English literatures from 1973 to 2009, whose median age was 3.5 months, much younger than the median age of 5 years in children with RMS reported in literature [9] . Our 2 cases were initially considered as mucoceles and tongue root tumors of unknown nature in local hospital respectively.…”

Section: Discussionmentioning

confidence: 98%

“…In case 2, tongue mass was found because of the symptom of choking milk. The differential diagnosis of the mass of tongue includes hemangiomas, fibroma, rhabdomyoma, lymphangioma, and lingual thyroid [8,9] , and primary lingual RMS is rare, the definite diagnosis of which requires pathological examination. In the Intergroup Rhabdomyosarcoma Studies(IRS) I, II and III, only 7 rhabdomyosarcomas of the tongue were registered, representing 0.34% of IRS participants [10] .…”

Section: Discussionmentioning

confidence: 99%

“…The most recognized reason now is that taking into account the complications and long-term adverse effects caused by surgery and radiotherapy, the local treatment intensity of infants is often low, leading to poor treatment outcome [12] . Among the 13 cases of lingual RMS reviewed by Rejin Kebudi et al, one 10-month-old girl received surgery after chemotherapy, the surgical margin was positive, and relapsed after 30 months of follow-up [9] .…”

Section: Casementioning

confidence: 99%

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Clinical Features, Prognosis and Susceptibility Genes of 2 Infants With Rhabdomyosarcoma of the Tongue: Case Report

Yang¹,

Xu²,

Su³

et al. 2021

Preprint

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Background Rhabdomyosarcoma(RMS) is the most common soft tissue tumor in children. RMS of the oral cavity accounts for 10–12% of all head and neck RMS, and the tongue is a rare primary site. The most common pathological types of RMS include embryonal rhabdomyosarcoma and alveolar rhabdomyosarcoma, and spindle cell rhabdomyosarcoma(SRMS) is rare. Meanwhile, it has been reported in the literature that infant is a rare onset age in RMS. We review two cases of infant tongue SRMS, and analyze the clinical features, prognosis and susceptibility genes. Case presentation The clinical data of 2 infants with RMS of the tongue admitted to Beijing Children's Hospital were retrospectively collected. Both the children and their parents accepted exome-customized capture sequencing of single gene disease in peripheral venous blood, and the results were analyzed. Both cases were infants and diagnosed with lingual RMS at 3 and 5 months after birth, respectively. The pathological type were both spindle cell rhabdomyosarcoma. The maximum diameter of the tumor in case 1 was 8mm, and 23mm in case 2. The clinical TNM staging both were T1aN0M0. IRS staging of case 1 was II, and case 2 was I. They were both classified in the low risk group, and treated with surgery and chemotherapy. The regimen of chemotherapy including 4 courses of VAC(vincristine, actinomycin D, cyclophosphamide) regimen first, followed by 4 courses of VA(vincristine, actinomycin D) regimen. Case 1 was in complete remission at the latest follow-up, and case 2 suffered from relapse 10 months after stopping chemotherapy, and achieved complete remission after the multi-disciplinary treatment of chemotherapy, surgery and radiotherapy. The venous blood gene test of the two children did not find pathogenic mutation or possible pathogenic mutation related to RMS. In case 1, variants of CDK4 and BRCA1 gene, both unclear significance (possibly) related with RMS, were detected. For case 2, we found three variants of unknown significance that were (possibly) associated with RMS, including variants of TRIP13, APC, and RAD54L gene. Conclusions Infant tongue RMS is rare, the clinical manifestations of which lack specificity, and early recognition is difficult. The success and timing of local treatment are important prognostic factors. Genetic testing may be helpful for early detection of tumor susceptibility and prognosis estimation.

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Section: Discussionmentioning

confidence: 98%

Section: Discussionmentioning

confidence: 99%

Section: Casementioning

confidence: 99%

See 1 more Smart Citation

Clinical Features, Prognosis and Susceptibility Genes of 2 Infants With Rhabdomyosarcoma of the Tongue: Case Report

Yang¹,

Xu²,

Su³

et al. 2021

Preprint

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“…Although malignant tumors, such as lymphoma and rhabdomyosarcoma, were considered, these are exceedingly rare in this location. 4 Benign tumors, such as teratoma and epulis, are more likely in this location and can present as rapidly growing tongue lesions, but these entities are also rare. 5,6 Although, grossly, the lesion appeared to be neoplastic, a thorough evaluation with multiple tumorspecific stains did not demonstrate a neoplastic lesion.…”

Section: Discussionmentioning

confidence: 99%

Unusual Fungal Lesion Presenting as a Neoplastic Pediatric Tongue Mass

et al. 2017

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Tongue lesions in the pediatric population are rare. The differential diagnosis of these lesions is broad, and rapid growth of the lesion is concerning for a neoplastic process. We present a rare case of a fungal lesion mimicking a neoplastic growth in a 22-month-old girl. She underwent complete excision successfully. Full evaluation for benign and malignant neoplasms was negative. Tissue culture demonstrated growth of a rare Candida species to be the cause of the lesion. Postoperatively, she continues to do well, without regrowth 6 months later. This case reinforces the role of tissue culture when histology fails to demonstrate a diagnosis and emphasizes the need for efficient communication between the pediatrician, otolaryngologist, and pathologist for timely excision.

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“…Rhabdomyosarcoma (RMS) comprises a group of soft tissue neoplasms that shares the propensity to undergo myogenesis. 1 There is a bimodal distribution of presentation with an initial peak incidence between 2-5 years of age and a second surge at [10][11][12][13][14][15][16][17][18][19]…”

Section: Introductionmentioning

confidence: 99%

Spindle cell rhabdomyosarcoma of the tongue – A rare entity

Khadim

Asif

Ali

et al. 2012

J Coll Med Sci-Nepal

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Spindle cell variant of Rhabdomyosarcoma is a rare malignant tumor in adults but even more so in tongue. We report a case of a 17 year old boy who presented with a polypoidal swelling on the tip of the tongue. The mass was excised and the specimen was sent to the Histopathology Department of Armed Forces Institute of Pathology for Histomorphological diagnosis. The histological evaluation showed a malignant spindle cell tumor. On the basis of morphological features and immunohistochemistry findings a final diagnosis of Rhabdomyosarcoma, Spindle cell variant was made. Journal of College of Medical Sciences-Nepal,2011,Vol-7,No-4, 63-66 DOI: http://dx.doi.org/10.3126/jcmsn.v7i4.6814

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Rhabdomyosarcoma of the Tongue: Report of a Case and Review of the Literature

Cited by 12 publications

References 19 publications

Clinical Features, Prognosis and Susceptibility Genes of 2 Infants With Rhabdomyosarcoma of the Tongue: Case Report

Clinical Features, Prognosis and Susceptibility Genes of 2 Infants With Rhabdomyosarcoma of the Tongue: Case Report

Unusual Fungal Lesion Presenting as a Neoplastic Pediatric Tongue Mass

Spindle cell rhabdomyosarcoma of the tongue – A rare entity

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