Epstein-Barr Virus-Positive Mucocutaneous Ulcer on the Gingiva of a Patient in Whom Immunosuppressive Drugs Could Not Be Withdrawn: A Case Report and Review of the Literature.

Epstein-Barr virus-positive mucocutaneous ulcer (EBV-MCU) is characterized by ulcers confined to the skin and mucus membranes. EBV-MCU is an EBV-positive lymphoproliferative disorder that occurs during the use of immunosuppressive drugs such as methotrexate. We herein report a case of EBV-MCU in the maxillary gingiva. A 73-year-old woman was referred to our department in March 2021. During the initial examination, bone exposure and ulceration were observed in the extraction socket of the maxillary bilateral central incisors. The patient was taking methotrexate for rheumatoid arthritis and was unable to stop due to disease progression. In March 2021, curettage of the extraction socket of the maxillary anterior teeth and extraction of the maxillary right lateral incisor, which was difficult to preserve due to severe tooth mobility, was performed under local anesthesia. The extraction site epithelialized and healed well. Three months later, inflammation flared, and ulceration was observed. Extraction of the unsalvageable maxillary teeth and an excisional biopsy of the palatal gingiva were performed. The histopathological diagnosis was EBV-MCU. The postoperative course was uneventful, and no evidence of recurrence was found two years postoperatively; follow-up will be continued. There are many reports of EBV-MCU remission with the cessation of methotrexate treatment. In our patient, withdrawal was difficult because of the progression of rheumatoid arthritis, but remission was achieved by improving the oral cavity environment through an excisional biopsy and tooth extraction.

Factors affecting the quality of life of patients with medication-related osteonecrosis of the jaw during treatment: A quality-of-life survey and causal analysis.

This prospective cohort study aimed to investigate the impact of medication-related osteonecrosis of the jaw (MRONJ) on health-related quality of life (HRQOL) and oral health-related QOL (OHRQOL) and the association between the downstaging of MRONJ and OHRQOL. The HRQOL and OHRQOL of 44 patients with MRONJ were assessed using the SF-36v2 and the General Oral Health Assessment Index (GOHAI), respectively. Treatment was performed in accordance with the AAOMS position paper (2014). The SF-36v2 and GOHAI scores at the beginning of the survey were used to evaluate the impact of MRONJ on QOL. Potential confounders affecting the association between downstaging and QOL improvement were selected using directed acyclic graphs. Multiple regression analysis was performed to evaluate causal inferences. HRQOL scale scores declined below the national average. The three-component summary score (3CS), comprising the physical component summary (PCS), mental component summary (MCS), and role/social component summary (RCS), revealed that performance status and primary disease significantly affected the PCS and RCS (P = 0.005 and P < 0.001, respectively) and PCS and MCS (P = 0.024 and P = 0.003, respectively). The MRONJ stage did not influence the 3CS; however, OHRQOL declined in a stage-dependent manner (P = 0.005). Downstaging of MRONJ was independently associated with the improvement rate of the total GOHAI scores after adjusting for variables (P = 0.045). The HRQOL of patients with MRONJ declined; however, this may depend on the underlying disease status rather than the MRONJ stage. Improvement of the disease status can potentially predict an improvement in OHRQOL, regardless of the treatment modality.

Etiology of Oral Potentially Malignant Disorders and Squamous Cell Carcinoma Based on Cellular Stress Regulation and Matrix Stiffness.

The oral cavity serves as the initial segment of the digestive system and is responsible for both nutritional supplementation and the mechanical breakdown of food. It comprises distinct hard and soft tissues; the oral mucosa is subject to mechanical stress and interaction with microbiota. In oral cancer, tumors exhibit abnormal cellular networks and aberrant cell-cell interactions arising from complex interplays between environmental and genetic factors. This presents a challenge for clinicians and researchers, impeding the understanding of mechanisms driving oral cancer development and treatment strategies. Lesions with dysplastic features are categorized under oral potentially malignant disorders, including oral leukoplakia, erythroplakia, oral submucous fibrosis, and proliferative verrucous leukoplakia, carrying a high malignancy risk. In this review, we discuss oral cancer cell characteristics and the stiffness of the surrounding matrix. We also discuss the significance of stiffness equilibrium in oral potentially malignant disorders, particularly oral submucous fibrosis, possibly triggered by mechanical stress such as betel quid chewing.

Periodontal therapy for localized severe periodontitis in a patient receiving fixed orthodontic treatment: a case report.

BACKGROUND: Orthodontic treatment involves movement of teeth by compression and resorption of the alveolar bone using orthodontic forces. These movements are closely linked to the interactions between the teeth and the periodontal tissues that support them. Owing to an increase in adults seeking orthodontic treatment, orthodontists increasingly encounter patients with periodontal diseases, in whom orthodontic treatment is contraindicated. In rare cases, periodontitis may develop after treatment initiation. However, no approach for treating periodontitis after the initiation of orthodontic treatment has been established. Here, we present an approach for managing localized severe periodontitis manifesting after initiating orthodontic treatment. CASE PRESENTATION: A 32-year-old Japanese woman was referred to the Department of Dentistry and Oral Surgery by an orthodontist who observed symptoms of acute periodontitis in the maxillary molars that required periodontal examination and treatment. A detailed periodontal examination, including oral bacteriological examination, revealed localized severe periodontitis (stage III, grade B) in the maxillary left first and second molars and in the mandibular right second molar. After consultation with the orthodontist, the orthodontic treatment was suspended based on the results of the bacteriological examination to allow for periodontal treatment. Full-mouth disinfection was performed with adjunctive oral sitafloxacin. Periodontal and bacteriological examinations after treatment revealed regression of the localized periodontitis with bone regeneration. Thereafter, orthodontic treatment was resumed, and good progress was achieved. CONCLUSIONS: Orthodontists should recognize the risk of acute severe periodontitis in young adults. Asymptomatic patients with localized severe periodontitis may clear a screening test before orthodontic treatment but develop acute symptoms with bone resorption during orthodontic treatment. Therefore, patients requiring orthodontic treatment should be examined by their family dentist or a periodontist to rule out periodontal issues that may impede orthodontic treatment. The patients should also be informed of age-related risks. Further, periodontists, family dentists, and orthodontists who treat adults should be informed about periodontitis and the need for interdisciplinary collaboration. In patients who develop periodontitis after orthodontic treatment initiation, temporary interruption of orthodontic treatment and aggressive periodontal intervention may facilitate recovery.

Multicenter retrospective study of nivolumab for recurrent/metastatic oral squamous cell carcinoma.

OBJECTIVES: Immunotherapy with nivolumab for patients with recurrent/metastatic oral squamous cell carcinoma has not been evaluated. Here, we aimed to examine the efficacy, safety, and prognostic factors of nivolumab in these patients. MATERIALS AND METHODS: This multicenter retrospective observational study involved patients who received nivolumab between April 2017 and June 2019. The patient characteristics were evaluated for association with progression-free and overall survival. Progression-free and overall survival rates were calculated; parameters that were significant in the univariate analysis were used as explanatory variables. Independent factors for progression-free and overall survival were identified using multivariate analysis. RESULTS: Totally, 143 patients were included. The overall response and disease control rates were 27.3% and 46.2%, respectively. The median, 1- and 2-year progression-free survival rates were 2.7 months, 25.4%, and 19.2%, respectively; those for overall survival were 11.2 months, 47.3%, and 33.6%, respectively. The independent factors affecting progression-free survival were performance status and immune-related adverse event occurrence, whereas those affecting overall survival were performance status, target disease, and number of previous lines of systemic cancer therapy. Eight patients reported grade ≥3 immune-related adverse events. CONCLUSION: Nivolumab was effective for recurrent/metastatic oral squamous cell carcinoma treatment and was well tolerated by patients.

Protocol for a multicentre, prospective observational study of elective neck dissection for clinically node-negative oral tongue squamous cell carcinoma (END-TC study).

INTRODUCTION: In early-stage oral tongue squamous cell carcinoma (OTSCC), elective neck dissection (END) is recommended when occult lymph node metastasis is suspected; however, there is no unanimous consensus on the risks and benefits of END in such cases. The management of clinically node-negative (cN0) OTSCC remains controversial. This study, therefore, aimed to evaluate the efficacy of END and its impact on the quality of life (QoL) of patients with cN0 OTSCC. METHODS AND ANALYSIS: This is a prospective, multicentre, nonrandomised observational study. The choice of whether to perform END at the same time as resection of the primary tumour is based on institutional policy and patient preference. The primary endpoint of this study is 3-year overall survival. The secondary endpoints are 3-year disease-specific survival, 3-year relapse-free survival and the impact on patient QoL. Propensity score-matching analysis will be performed to reduce selection bias. ETHICS AND DISSEMINATION: This study was approved by the Clinical Research Review Board of the Nagasaki University. The protocol of this study was registered at the University Hospital Medical Information Network Clinical Trials Registry. The datasets generated during the current study will be available from the corresponding author on reasonable request. The results will be disseminated internationally, through scientific and professional conferences and in peer-reviewed medical journals. TRIAL REGISTRATION NUMBER: UMIN000027875.

Giant peripheral ossifying fibroma with coincidental squamous cell carcinoma: a case report.

BACKGROUND: Peripheral ossifying fibroma is an inflammatory or reactive hyperplasia of the gingiva that is usually small. It is formed by hard tissue in fibrous tissue, and the name "neoplastic lesion" has tended to be used frequently in Europe and America. Clinically, peripheral ossifying fibromas are painless, solitary, exophytic, sessile, or pedunculated and more frequently found in females than in males. To the best of our knowledge, there have been no reports of malignant cases. We herein report the case of giant peripheral ossifying fibroma with squamous cell carcinoma. CASE PRESENTATION: The patient was an 83-year-old Japanese woman who visited our hospital with a gingival massive mass. She was referred to us for an examination and treatment because it was difficult to perform tracheal intubation for surgery of sigmoid colon cancer at another hospital. The mass measured 83 × 58 × 35 mm, and it protruded to the extra-oral region from the right maxillary premolar alveolar region. Panoramic X-ray revealed the shadow of the mass in the right maxillary premolar region, which included some hard tissue. Computed tomography showed scattering calcified images in the mass. Magnetic resonance imaging was not performed because she had vertebral artery clips and screws in her forehead. Given the above findings, we performed a biopsy under local anesthesia. However, we were unable to diagnose absolutely whether the dysplastic squamous epithelia were pseudocarcinomatous hyperplasia of the gingiva or well-differentiated squamous cell carcinoma. Therefore, tumor resection was performed under general anesthesia. The histopathological diagnosis was peripheral ossifying fibroma with coincidental squamous cell carcinoma. There have been no signs of recurrence during follow-up as of 2 years after surgery. CONCLUSIONS: The etiology of giant peripheral ossifying fibroma with squamous cell carcinoma is still not definite. Therefore, careful observation is necessary. It needs to be examined by accumulation of more cases in the future. We herein report the case of giant peripheral ossifying fibroma coincidental squamous cell carcinoma.

Clinical value of entire-circumferential intraoperative frozen section analysis for the complete resection of superficial squamous cell carcinoma of the tongue.

OBJECTIVES: We aimed to evaluate the clinical value of an entire-circumferential intraoperative frozen section analysis (e-IFSA) for the complete resection of superficial squamous cell carcinoma (SCC) of the tongue. MATERIALS AND METHODS: A total 276 specimens from 51 patients with pT1-2, N0, mucosal or submucosal invasion SCC were analyzed to evaluate the diagnostic accuracy of the e-IFSA and the added value of the e-IFSA to iodine staining. The e-IFSA results were compared with the final histologic results obtained using permanent sections. All specimens for the e-IFSA were taken over the entire circumference 5 mm outside from the iodine unstained areas. The outline of the main resected specimen after taking these outer mucosal specimens were defined as the surgical margins determined by iodine staining alone. RESULTS: The e-IFSA results were in excellent agreement with final histological results (Cohen's kappa value: 0.85) and the e-IFSA showed high sensitivity (100%) and high negative predictive value (100%). The actual complete resection rate with an e-IFSA was 100% (51/51), and no patient required additional resection after surgery. In contrast, 10/51 patients (20%) patients showed residual atypical mucosal epithelium at or beyond the margin determined by iodine staining alone; this difference was statistically significant (P = 0.002). The 5-year local control rate and 5-year overall survival rate after this procedure were both 100%. CONCLUSION: An e-IFSA has additional value when performed in conjunction with iodine staining. An e-IFSA would be useful for achieving complete resection of superficial SCC of the tongue.

Influence of prostate cancer status on the prevalence of medication-related osteonecrosis of the jaw.

OBJECTIVE: The aim of this study was to evaluate the risk of osteonecrosis of the jaw (ONJ) in patients with prostate cancer, particularly the relationship between prostate cancer progression and ONJ development. STUDY DESIGN: This single-center, retrospective, observational study included 113 patients who received zoledronic acid or denosumab for prostate cancer with bone metastasis between January 2012 and March 2020. The risk of ONJ was evaluated regarding age; antiresorptive drugs; duration of antiresorptive treatment; prostate cancer status, including castration-resistant prostate cancer (CRPC) and prostate-specific antigen level; chemotherapy; radium-223 treatment; corticosteroid treatment; diabetes mellitus; and dental extractions. RESULTS: Overall, 28 patients had ONJ; 10 patients received zoledronic acid and 18 patients received denosumab. Multiple logistic regression analysis demonstrated that CRPC (odds ratio = 6.01; 95% confidence interval, 1.76-20.05; P = .004) and dental extractions (odds ratio = 12.40; 95% confidence interval, 3.42-44.70; P < .001) were significantly associated with ONJ. In addition, antiresorptive treatment lasting more than 1 year partially mediated between CRPC and development of ONJ. CONCLUSION: CRPC and dental extraction are risk factors for developing ONJ, and antiresorptive treatment lasting more than 1 year is a partial mediator between CRPC and ONJ.

Development of a microsurgery-assisted robot for high-precision thread traction and tension control, and confirmation of its applicability.

BACKGROUND: Microsurgery requires high skills for suturing using fragile threads, often within narrow surgical fields. Precise tension is required for good healing and to avoid the risk of thread breakage. METHODS: To meet the demands, we developed a novel assist robot utilizing high-precision sensorless haptic technology. The robot adopts a cable-driven mechanism to maintain a distance from the surgical area and enhances compatibility with surgical equipment such as microscopes. The robot performance was verified through in vitro and in vivo experiments using a rat model. RESULTS: The realization of precise tension control was confirmed in both experiments. In particular, in the in vivo experiments, the developed robot succeeded to produce a knot with an accurate tension of 0.66% error. CONCLUSIONS: The developed robot can realize to control traction force precisely. This technology might open up the window for a full assist robot for microsurgery with haptic feeling.

Tooth extraction in mice administered zoledronate increases inflammatory cytokine levels and promotes osteonecrosis of the jaw.

INTRODUCTION: Osteonecrosis of the jaw (ONJ) occurring after invasive dental treatment often adversely affects patients' activities of daily living. Long-term administration of strong anti-bone resorptive agents such as bisphosphonates prior to invasive dental treatment is considered an ONJ risk factor; however, pathological mechanisms underlying ONJ development remain unclear. MATERIALS AND METHODS: We developed an ONJ mouse model in which a tooth is extracted during treatment with the bisphosphonate zoledronate. RESULTS: We observed induction of apoptosis in osteocytes, resulting in formation of empty lacunae in jaw bones at sites of tooth extraction but not in other bones of the same mice. We also observed elevated levels of inflammatory cytokines such as TNFα, IL-6 and IL-1 in jaw bone at the extraction site relative to other sites in zoledronate-treated mice. We also report that treatment in vitro with either zoledronate or an extract from Porphyromonas gingivalis, an oral bacteria, promotes expression of inflammatory cytokines in osteoclast progenitor cells. We demonstrate that gene-targeting of either TNFα, IL-6 or IL-1 or treatment with etanercept, a TNFα inhibitor, or a neutralizing antibody against IL-6 can antagonize ONJ development caused by combined tooth extraction and zoledronate treatment. CONCLUSIONS: Taken together, the cytokine storm induced by invasive dental treatment under bisphosphonate treatment promotes ONJ development due to elevated levels of inflammatory cytokine-producing cells. Our work identifies novel targets potentially useful to prevent ONJ.

Acute odontogenic infection combined with crowned dens syndrome: a case report.

BACKGROUND: Calcium pyrophosphate dihydrate crystal deposition disease is a condition in which calcium pyrophosphate dihydrate crystal is deposited in joint cartilage and ligaments. Calcium pyrophosphate dihydrate crystal deposition disease that involves calcification around the odontoid process of the second cervical vertebra is called crowned dens syndrome. Crowned dens syndrome is accompanied by fever in addition to acute and intense neck, posterior head, and temporal pain; thus, distinguishing crowned dens syndrome may be difficult in the presence of odontogenic infection. To the best of our knowledge, this is the first report describing a patient with crowned dens syndrome with coexisting odontogenic infection. CASE PRESENTATION: A 75-year-old Japanese woman was examined in the Emergency Department of this hospital due to a chief complaint of worsened buccal swelling on the left side. An odontogenic infection was considered, and she underwent her first examination. She presented with a body temperature of 37.4 °C, marked swelling and tenderness of her left lower eyelid through to her left cheek, and pain on the left temporal area. Blood tests revealed a leukocyte count of 6700/µL and a C-reactive protein level of 7.15 mg/dL. There was swelling and pain around the gingiva and acute purulent apical periodontitis of left maxillary second premolar. Cellulitis of the left cheek was diagnosed. After performing drainage of the pus, antibiotic treatment was initiated. Although her clinical symptoms improved, blood tests on day 9 of hospitalization revealed a leukocyte count of 6500/µL and a C-reactive protein level of 25.62 mg/dL, which were indicative of worsening symptoms. Computed tomography was performed to evaluate remote infection and images revealed a calcification around the odontoid process of her second cervical vertebra. When she was referred to the Orthopedic Surgery Department, pseudogout of the cervical spine was diagnosed. Subsequently, oral acetaminophen was initiated, and both her leukocyte count and C-reactive protein improved markedly. CONCLUSIONS: In the presence of persistent fever and abnormally high leukocyte and C-reactive protein indicative of an inflammatory reaction, coexistence of pseudogout should be considered. In particular, when symptoms of temporal pain are present, the possibility of pseudogout of the cervical spine must be considered in the differential diagnosis.

Clinical value of 3D SPECT/CT imaging for assessing jaw bone invasion in oral cancer patients.

PURPOSE: This study compared the diagnostic accuracy of jaw bone invasion (JBI) of oral cancer observed with three-dimensional (3D) SPECT/CT, CT, and MRI, and evaluated the clinical advantages of 3D SPECT/CT compared to conventional two-dimensional (2D) SPECT/CT. MATERIALS AND METHODS: From April 2014 to January 2018, consecutive 16 oral cancer patients with suspected JBI, who had preoperatively undergone the imaging tests, were retrospectively enrolled. The likelihood of JBI was independently scored by a radiologist and oral surgeon. Using 2D or 3D SPECT/CT images, 20 oral surgeons delineated virtual surgical areas on 3D-printed jaws for 3 cases in which the extent of JBI was fully pathologically confirmed. The surgeons completed questionnaires regarding surgical planning and explanations for patients using Likert scales. RESULTS: JBI was found in 9 patients including 5 (56%) with initial bone invasion. 3D SPECT/CT showed very high negative predictive value (100%) and inter-observer agreement (kappa = 0.917). 3D SPECT/CT was more sensitive than CT and MRI when inconclusive findings for JBI were considered negative. Compared to 2D SPECT/CT, 3D SPECT/CT had greater clinical advantages such as surgical planning and explanation to patients (p < 0.005). CONCLUSION: 3D SPECT/CT is useful not only for detecting JBI but also for surgical planning.

The Effect of Improving Oral Hygiene through Professional Oral Care to Reduce the Incidence of Pneumonia Post-esophagectomy in Esophageal Cancer.

Radical esophagectomy for thoracic esophageal cancer is invasive and frequently results in postoperative pulmonary complications. Postoperative pneumonia is the most common such complication and affects hospital mortality and survival rates. Oral care has been very effective in reducing pneumonia. In Japan, preoperative professional oral care is highly recommended. However, there are few studies on the effect of preoperative improvements in oral hygiene as a result of intervention on the incidence of postoperative pneumonia. The primary end-point of this retrospective study was the incidence of postoperative pneumonia after radical esophagectomy. The oral health levels of 46 patients were individually categorized, and then patients were grouped according to whether they maintained or improved their oral hygiene. At the first dental examination, oral health levels were classified as good in 22 patients and bad in 24. Of the 46 patients studied, 39 patients maintained or improved their oral hygiene (good control group), whereas 7 showed no improvement (bad control group). Postoperative pneumonia occurred in eight patients: four in the good control group and four in the bad control group. Statistical analysis with postoperative pneumonia as a dependent variable showed a significant effect of oral hygiene improvement on the incidence of pneumonia. Logistic regression analysis with this factor as an independent variable demonstrated that the risk of postoperative pneumonia was reduced in the good control group (OR 0.086, 95% CI 0.014-0.529). Therefore, preoperative professional oral care may improve oral hygiene and oral health, which may in turn reduce the incidence of postoperative pneumonia.

Treatment of severe generalized chronic periodontitis in a patient with Behçet's disease: A case report.

Behçet's disease is a systemic disorder of unknown etiology. It involves multiple organ systems and is characterized by recurring episodes of oral ulcers as well as ocular, genital, and skin lesions. Oral ulcers can affect tooth brushing and impair proper oral hygiene. As a result, a dental biofilm accumulates, and the condition of the teeth and periodontal tissue deteriorates. The aim of this case report is to highlight the efficacy of periodontal treatment for patients with Behçet's disease. A 51-year-old man with Behçet's disease presented with generalized severe periodontitis. After basic treatment of the periodontal tissues, periodontal surgery was performed at several sites with bony defects. However, the patient developed severe stomatitis in the oral mucosa and gingiva after periodontal surgery. Administration of the antimicrobial agent cefdinir had little effect on recovery; however, subsequent administration of sitafloxacin resulted in significant improvement of the stomatitis. This case demonstrates that periodontal therapy is very useful for alleviating the oral signs and symptoms of Behçet's disease. Systemic antibiotic treatment with sitafloxacin (but not cefdinir) and mechanical debridement were effective in preventing the recurrence of aphthous ulcer outbreaks after periodontal surgery.

[Determination of the Effect of Aprepitant and Fosaprepitant for Nausea in Patients with Oral Cancer Receiving Combination Chemotherapy with TPF].

The objective of this study was to determine the effect of aprepitant(from days 1 to 3, po)and fosaprepitant(day 1, iv) for nausea in patients with oral cancer receiving combination chemotherapy with docetaxel, nedaplatin, or cisplatin(divided doses for 5 days), and 5-fluorouracil(TPF).The incidence rate of nausea in the aprepitant group was 60%(6/10), and that in the fosaprepitant group was 90%(9/10).The incidence rate of continuous nausea for more than 2 days was significantly lower in the aprepitant group than in the fosaprepitant group(40%[4/10]vs 90%[9/10], p=0.02; c 2 test).In addition, the mean area under the curve of the chronological changes in the grade of nausea tended to be lower in the aprepitant group than in the fosaprepitant group.In both groups, 3 cases(30%)of vomiting were observed.However, the incidence of continued daily vomiting tended to be lower in the aprepitant group than in the fosaprepitant group.These results suggest that aprepitant is more effective than fosaprepitant for nausea induced by TPF.

The onset risk of carcinoma in patients continuing tacrolimus topical treatment for oral lichen planus: a case report.

Oral lichen planus is a chronic inflammatory mucocutaneous disease. Topical use of steroids and other immuno-modulating therapies have been tried for this intractable condition. Nowadays, tacrolimus ointment is used more commonly as a choice for treatment. However, a number of discussions have taken place after tacrolimus was reported to be carcinogenic. This report describes a patient who applied tacrolimus ointment to the lower lip after being diagnosed with oral lichen planus in 2008, and whose lesion developed squamous cell carcinoma in 2010. Since the relationship between tacrolimus and cancer development has been reported in only a few cases, including this case report, the clinician must be careful selecting tacrolimus as a second-line treatment for oral lichen planus.

Desmoid-type fibromatosis of the head and neck in children: a case report and review of the literature.

BACKGROUND: Desmoid-type fibromatosis is defined as an intermediate tumor that rarely occurs in the head and neck of children. There is no doubt as to the value of complete surgical excision for desmoid-type fibromatosis. However, in pediatric patients, surgeons may often be concerned about making a wide excision because of the potential for functional morbidity. Some studies have reported a lack of correlation between margin status and recurrence. Therefore, we discussed our findings with a focus on the state of surgical margins. CASE PRESENTATION: We report an unusual case of a 9-month-old Japanese girl who prior to presenting at our hospital underwent debulking surgery twice with chemotherapy for desmoid-type fibromatosis of the tongue at another hospital. We performed a partial glossectomy and simultaneous reconstruction with local flap and achieved microscopic complete resection. We also reviewed available literature of pediatric desmoid-type fibromatosis in the head and neck. CONCLUSIONS: We described successful treatment for the refractory case of pediatric desmoid-type fibromatosis. The review results showed that some microscopic incomplete resections of tumors in pediatric patients with desmoid-type fibromatosis tended to be acceptable with surgical treatment.