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Cleft lip, with or without a cleft palate, is the most common craniofacial birth defect. Speech disorders, particularly impairment in articulation, are severe problems resulting from the velopharyngeal incompetence associated with a cleft palate. In cases requiring prolonged tracheal cannulation further delays speech and language development, especially during the pre-verbal period of an infant. In this case report, we describe the treatment course of an infant with bilateral cleft lip and palate requiring long-term cannulation. The patient underwent tracheotomy due to laryngotracheal stenosis after cheiloplasty at the age of 1 year. The tracheal cannula was in situ for a period of 6 years (to the age of 6 years). An intensive program of articulation and vocalization practice was performed during the period of tracheal cannulation. Following removal, the patient acquired almost normal speech and language functions. Coordination between several disciplines was critical for treatment to benefit the patient.

Prader-Willi syndrome (PWS) is characterized by infantile hypotonia, hypogonadism, hyperphagia, developmental delay and mild mental retardation and characteristic facial features caused by the lack of genes on chromosome 15q11-q13. Frequency of PWS is estimated to be 1/10,000–1/15,000 and cleft lip and/or palate case is rare. Most PWS babies suffer from hypotonia with feeding problems and speech articulation defects, even though they have normal anatomical structure in oral region. We report a case of PWS with cleft palate (CP). Case We report on a three-month-old male who was referred to our department from pediatrician of our hospital to treat a cleft in the soft palate. He had hypotonia, feeding problems, almond-shaped eyes and bilateral cryptorchidism at birth. The chromosome test was performed and he was diagnosed as PWS. The volume of milk feeding was improved by feeding training with age. However, when he started feeding with solid foods at 12 months old, a nasal emission was observed, so he disliked eating. After palatoplasty performed at 18 months old, a nasal emission was disappeared and food intake was increased markedly. One year after palatoplasty, his swallowing function was progressed and the velopharyngeal function improved to slight insufficiency. Conclusion In this case, it is possible that palatoplasty had an effect on not only velopharyngeal functions but also his swallowing functions.

Objective Cytokeratin (CK) 19 is a member of the acidic type I CK family. Recently, CK19 expression has been found in various tumor tissues; however, the significance of this remains unknown. The purpose of this study is to clarify the roles of CK19 in the progression of oral squamous cell carcinoma (OSCC). Methods A total of 100 patients who had been diagnosed with OSCC at our department between January 2011 and December 2016 was included. The patients were divided into three groups based on an optimal cut-off points (5% and 77%) of the labeling index (LI) as follows: group A; LI Results Histologically high-grade tumors were significantly more common in group C than in groups A and B. Furthermore, the incidence of nodal metastasis was significantly higher in group C than in other groups. Intense CK19 immunoreactivity was detected in metastatic lymph nodes of groups B and C, but not from group A. Moreover, patients with advanced pN stage and extranodal extension were more common in groups B and C than group A. Disease-specific survival curves revealed poorer prognoses in group C. Conclusions These results suggest that CK19 is involved in OSCC invasion and metastasis and could be a novel biomarker of highly invasive OSCC with metastatic potential.

Osteoid osteoma is a benign bone-forming tumor and characterized by its limited growth potential, not exceeding 2 cm. The radiological hallmark of this tumor is a nidus, which is a small round area of relative radiolucency. Osteoid osteoma can involve any bone but is most commonly found in long bones and is extremely rare in the head and neck region. This disease characteristically presents with dull pain, worse at night, and sometimes relieved with NSAIDs. A 24-year-old Japanese woman presented with spontaneous pain and tenderness on the lingual side of her mandibular second molar on the right side. The patient reported that her pain had gradually increased, becoming more continuous and severe and no longer responding to NSAIDs. An initial panoramic radiograph revealed an oval, internally non-uniform, somewhat obscure boundaries in the right mandible. Computed tomography (CT) scan revealed a sclerotic lesion with a delineated central calcified nidus surrounded by a radiolucent band. The interior of the nidus was a non-uniform, irregularly shaped area of high absorption. The nidus was removed with intralesional curettage under general anesthesia. The histopathology of the specimen consisted of actively proliferating osteoblasts mixed with an interlacing network of immature bone and osteoid trabeculae. Immunohistochemistry revealed that hardly detected osteoblasts or fibrous stromal cells with intense nuclear immunoreactivity for p16 and/or murine double minute 2 (mdm2). We thus distinguished the tumor from Low-grade central osteosarcoma (LGCO) with immunohistochemical findings. The histopathological diagnosis was thus osteoid osteoma.

Cepharanthine and Z-100 have been used for radiation leukopenia. But there is no report that they are applied in preoperative chemoradiotherapy. The purpose of this study is to evaluate the efficacy of cepharanthine and Z-100 against complications such as myelosuppression, oral mucositis and xerostomia during preoperative chemoradiotherapy for oral squamous cell carcinoma (OSCC) retrospectively. Between April 2008 and March 2013, 38 OSCC patients (27 males and 11 females) received preoperative chemoradiotherapy. One male and one female stopped on the way due to severe oral mucositis. As we started to use cepharanthine and Z-100 simultaneously since January 2010, 18 patients received neither cepharanthine nor Z-100 between April 2008 and December 2009 (Group A). Between January 2010 and March 2013, 18 patients received both cepharanthine (6 mg per day) and Z-100 (20 μg, twice a week) during radiotherapy (Group B). There is no significant difference of maintenance rate of blood cell counts. But patients of Group B had milder oral mucositis ( p = 0.0045) and milder xerostomia ( p = 0.010) than those of Group A. These results indicate that it is difficult against myelosuppression with low dose and short period of cepharantine and Z-100 in preoperative chemoradiotherapy. However, they lead mild mucositis and xerostomia. This is a great help to accomplish preoperative chemoradiotherapy.

The complications of orthognathic surgery may include but are not limited to postoperative unexpected bleeding, abnormal fractures, infections, and nerve damages. Peripheral facial palsy is rare. We report a case of 23-year-old woman with signs of malocclusion, including pectus excavatum, wrist sign, and thumb sign for Marfan syndrome (MFS) underwent peripheral facial palsy after orthognathic surgery with mandibular advancement. Because cardiovascular, thoracic, and ocular abnormalities were absent, she was suspected of having MFS but not definitively diagnosed. On the other hand, she was diagnosed with a maxillary protrusion with mandibular retrognathism. After presurgical orthodontic treatment, we performed Le Fort I osteotomy and 8 mm advancement by bilateral sagittal split ramus osteotomy. Because the periosteum is vulnerable, we checked the bleeding more are fully and frequently during the operation than usual. The day after the operation, peripheral facial palsy with inability to wrinkle the forehead, blink, and grimace were found. The patient was administered mecobalamin, linear polarized infrared irradiation, and stellate ganglion blocks. The patient was also administered valacyclovir for 6 days and steroid pulse therapy for 7 days. Six months after the operation, the facial palsy had disappeared completely. In 58 previous reports, the facial palsy was induced by the reasons, including the operative procedure, postoperative hematoma, edema, perioperative stress, and tissue vulnerability. In the present case, because of the suspected MFS, tissue vulnerability was considered to be one of the major cause of facial palsy, which should be paid attention more carefully in addition to general causes.

Objective Oral lichen planus (OLP) displays various and complicated clinical presentations which often make their differential diagnosis challenging, and thus helpful clinical practice guidelines for the diagnosis and treatment of OLP have been long awaited. Methods The Japanese OLP Working Group (OLP-WG) has collected and analyzed a total of 393 OLP cases provided by 48 institutions nationwide from 2009 to 2011 toward the establishment of valuable clinical practice guidelines for OLP. Collected samples were classified according to their original diagnoses into three groups: bilateral reticular (Group 1); bilateral atrophic/erosive (Group 2); and unilateral (Group 3) buccal lesions. Ten OLP-WG members used intraoral pictures to categorize the cases into Andreasen’s six types, and then examined the biopsy specimens to make pathological and comprehensive diagnoses. Results When Andreasen’s reticular, plaque, and papular types were sorted into white (W) type, and when atrophic, erosive, and bullous types were sorted into red (R) type, they formed six clusters based on the number of the members’ judgments: W1 (W dominant), W/R (W-R competing), R1 (R dominant), UD (undeterminable dominant), W2 (boundary), and R2 (boundary). Both in the bilateral and unilateral lesion groups, proportions of cases which were comprehensively diagnosed as OLP were significantly higher in cluster W1 than those in cluster R1 (p Conclusions These results indicated that the simple W or R classification would be a better substitute for Andreasen’s in the clinical categorization of OLP.

Purpose The purpose of this study was to clarify the usefulness of noninvasive examination items such as sialometry and Visual Analog Scale (VAS) in distinguishing Sjogren's syndrome (SS) in dry mouth patients from neurogenic/neuropsychiatric disorders and drugs (DND). Patients and methods The study cohort comprised 50 patients with SS and 28 patients with DND. The gum test and Saxon test for stimulated salivary flow rate (SSFR), the spitting test for unstimulated salivary flow rate (USFR) and VAS were performed in all the patients with dry mouth. Results In SS patients, the SSFR (mean: gum test, 6.34 mL/10 min; Saxon test, 1.19 g/2 min) and USFR (0.61 mL/15 min) were decreased. In DND patients, the SSFR (gum test, 16.35 mL/10 min; Saxon test, 3.58 g/2 min) was within the normal range, but the USFR (0.90 mL/15 min) was decreased. In VAS, SS patients scored significantly higher in the items of “water-drinking at meals”, “difficulty in swallowing”, and “taste abnormality”, while significantly lower in the item of “oral pain”. Conclusion These results suggest that the SSFR, USFR and VAS could be useful in distinguishing DND from SS.

Purpose The purpose of this study was to evaluate the effect of infant orthopedic treatment using Hotz's plate and lip adhesion (LA) on maxillary arch of the patients with complete unilateral cleft lip and palate (UCLP) until 5 years of age. Patients and methods Fifty patients with complete UCLP who were treated in Cleft Lip and Palate Center of Harapan Kita Children and Maternity Hospital, Indonesia, were divided into three groups. Hotz (+) group: 22 patients treated with Hotz's plate until palatoplasty. Hotz–LA group: 14 patients treated with LA at one or two months of age and with Hotz plate until palatoplasty. Hotz (−) group: 14 patients treated without Hotz's plate and LA. Serial dental casts were obtained from each patient at following four time points: first visit, cheiloplasty, palatoplasty, and 5 years of age. Each maxillary dental cast was scanned, and the linear and angular dimensions were measured. Results Maxillary arch width and length of anterior part were larger in the Hotz (+) group than in the Hotz–LA and Hotz (−) groups at the palatoplasty and 5 years of age. The anterior part of the maxillary arch in the Hotz–LA and Hotz (−) groups was smaller and more flattened than that in the Hotz (+) group. Conclusions This study suggests that Hotz's plate prevents the maxillary arch from collapsing after cheiloplasty to the palatoplasty. This effect remained up to 5 years of age. However, the effects of Hotz's plate are negated in patients who underwent LA.

A surgical ciliated cyst of maxilla is well known as a delayed complication of radical maxillary sinus surgery for chronic sinusitis. Recent reports have suggested that orthognatic surgery, implant-related surgery, and fracture surgery also causes a development of surgical ciliated cyst. It is documented that small segments of sinus mucosa become entrapped between the bony edges of the maxilla, which resulting in cystic degeneration of the entrapped sinus mucosa many years later. However, few papers are available that fracture individual could lead to a development of a maxillary cyst with pseudostratified columnar ciliated epithelium. Here we report a unique case of the ciliated cyst in the maxilla, supposed to be derived from the zygomaticomaxillary fracture individual conservatively treated 30 years before. The diagnostic difficulty of this lesion is also discussed.

Myoepithelioma is a rare tumor, accounting for less than 1.5% of all salivary gland tumors. Generally, this tumor exhibits painless solid mass. We here report an extremely rare case of myoepithelioma with remarkable cystic formation in the parotid gland in an 80-year-old female. In this case, a malignant tumor with cystic formation of the parotid gland was clinically suspected following preoperative imaging examinations. Standard superficial parotidectomy was thus performed under general anesthesia. Histopathologic findings revealed that the tumor consisted of spindle cells, plasmacytoid cells, and epithelioid cells, and remarkable cystic formation was observed. Immunohistochemically, the neoplastic cells were positive for S-100, cytokeratin, vimentin, and calponin. The final histopathologic diagnosis was myoepithelioma. Four years have past after the surgery without recurrence of the tumor.