Recently, distraction osteogenesis （ DOG） has been used to various deformities of oral and maxillofacial area, because it is effective for not only increasing bone length but also the volume of surrounding soft tissues, including skins, muscles, vessels, and nerves. In patients who have skeletal reversed occlusion caused by maxillary hypoplasia with cleft lip and palate, we perform 2-stage surgery, which is combining maxillary advancement by distraction technique before mandibular setback surgery, in order to extend the palatal scar tissue at the same time, and they get stable occlusion. This technique can be applied to treatment for hemifacial microsomia（ HFM） patients with soft tissue turgor in adult as combining mandibular advancement by distraction technique before maxillary translation surgery. Additionally, DOG is useful option to secondary lengthening of the reconstructed mandible for obtaining improvement of maxillamandibular relation, and soft tissue expansion for making the space of mandibular reconstruction in the patients with contracture of oral tissue after tumor resection. However, especially in patients treated with radiation, they have encountered several complications such as infection, failure of bone formation, and skin breakdown, therefore, we should consider the therapeutic indication of DOG on patients carefully.
Purpose: Obstructive sleep apnea syndrome (OSAS) is a disease that can affect life prognosis, against a background of respiratory disorder during sleep and decreased quality of sleep. The first-choice treatment for OSAS is continuous positive airway pressure (CPAP), but dislodgment of the CPAP device has been described in a number of cases. Oral appliances (OA) work well in mild to moderate OSAS, even in cases where CPAP devices are displaced. Respiratory impairment has been evaluated in many institutions to assess therapeutic effectiveness. The present study evaluated and compared the quality of sleep before and after use of OA. We focused on changes in the quality of sleep. Methods: The study group comprised 73 patients who underwent OA treatment at Tokyo Dental College Ichikawa General Hospital between July 2003 and December 2008. The patients were divided into an improvement group and non-improvement group and studied. The improvement group comprised 42 patients in whom the apnea hypopnea index on overnight polysomnography improved to >50% or ≦5/h. The other 31 patients were in the non-improvement group. Sleep parameters (i.e., sleep stage, sleep latency, rapid eye movement［REM］ sleep latency, sleep efficiency, and total arousal index) before and after treatment were compared, and the effect of OA on the quality of sleep was investigated, adjusting for disease severity. Results: In patients with moderate and severe OSAS in the improvement group, the proportion of Stage REM significantly increased and the proportion of Stage Ⅰ as well as the total arousal index significantly decreased. In addition, the proportion of Stage II in patients with moderate OSAS in the improvement group decreased significantly. Discussion: The use of OA improved breathing disorders such as apnea and hypopnea in patients with moderate and severe OSAS and reduced the total arousal index, resulting in normalization of the sleep stage cycle. The present study demonstrates that treatment of OSAS requires not only improvements in respiratory disorders, but also adequate consideration of sleep quality.
Objective: To analyze the outcomes of the secondary correction of unilateral cleft lip nose deformities in childhood, the preoperative and postoperative nasal forms were assessed clinically and three-dimensionally. Patients and methods: Fifteen Japanese children with unilateral complete cleft lip with/without cleft palate (UCLP) who had undergone follow-up secondary cleft lip nose correction in childhood and were followedup for 2 to 4 years at the Department of Oral and Maxillofacial Surgery, Kagoshima University Hospital were studied. All patients were treated by rhinoplasty through a unilateral reverse-U incision combined with the medial-upward advancement of nasolabial components and vestibular expansion using a free mucosal graft. Three-dimensional (3D) nasal forms were longitudinally measured using a 3D noncontact laser scanner preoperatively and 3 months and more than 2 year postoperatively in 6 patients. Analyzed variables included angular and linear measurements, deviation of the nasal midline, and curvature of the nasal alar. Results: There were no serious complications in any patients. Comparison of the preoperative and postoperative 3D nasal forms revealed that the nasal height was significantly increased (p<0.05) and the deviation of the nasal midline was improved in the lower half of the nose (p<0.05) postoperatively. The significant difference in the curvature of the nasal alar grooves between the cleft and noncleft sides (p<0.05) disappeared postoperatively. However, longitudinal observation of the nasal forms demonstrated slight relapse of the nasal height and nostril shape more than 2 years postoperatively. Conclusions: Our procedure for secondary correction of nasal deformities in childhood improves the nasal height and symmetry of the alar forms of patients with UCLP. Long-term follow-up is necessary to clarify effects on the stability and growth of nasal tissues reconstructed in childhood.
The patient was a 68-year-old woman with limited mouth opening. The patient had a medical history of urinary bladder cancer, which recurred with bilateral metastases to the tibia. She presented with marked swelling around the left temporomandibular joint region. Panoramic radiography and magnetic resonance imaging (MRI) revealed extensive osteolysis of the left condylar process. MRI also showed a soft tissue mass around the distracted condylar process. Needle biopsy was performed. The histopathological diagnosis was transitional epithelial cancer. The final diagnosis was metastatic urinary bladder cancer in the temporomandibular joint region. Radiation therapy was performed for bilateral bone metastases of the tibia, pelvis minor, and the left temporomandibular joint. She died 10 months presentation.
Arteriovenous malformation (AVM) of the jaw is considered a rare lesion. We report a case of AVM of the mandible. A 51-year-old man was referred to our department for further evaluation of a mandibular lesion. Panoramic radiography showed a multilocular radiolucency extending from the lower retromolar region to the right ramus of the mandible. A biopsy was performed for differential diagnosis between an ameloblastoma and keratocystic odontogenic tumor. However, severe hemorrhage occurred during the biopsy. The patient was admitted to our hospital urgently. An intraosseous AVM supplied by the maxillary artery was suspected on angiography. Embolization of the right maxillary artery was performed to decrease the hemorrhage during operation. One hour after the embolization, a segmental mandibulectomy was perfomed with the patient under general anesthesia. Histopathologically, the lesion was diagnosed as an AVM. The patient was followed up for 2 years without recurrence.
We describe a rare case of a varix with old thrombosis in a 71-year-old woman. A round, elastic hard, mobile mass (10 mm in a diameter) was detected in the right buccal region. The surface of the mass was smooth, and the overlying buccal mucosa was normal. Radiographic examinations showed a clearly bordered round radiolucent mass at the anterior edge of the ramus. The mass was completely enucleated through an intraoral approach with the patient under local anesthesia. Histopathologically, the mass was diagnosed as a varix with old thrombosis. The postoperative course has been uneventful.
In this super-aging society, treatment of patients with a variety of underlying diseases has become more complex. This case report describes a 78-year-old woman who was receiving long-term steroid therapy for rheumatoid arthritis. The patient was referred to our hospital for swelling of the right cheek and neck, associated with pain and trismus. Computed tomography (CT) revealed osteomyelitis of the mandible, which led to an abscess extending to the masticator space, facial cellulitis, and progressive internal jugular vein thrombosis. Clinical examination revealed decreased renal function and hypokalemia resulting from dehydration. We performed a surgical incision and drainage of the abscess to reduce inflammation. Steroid treatment, electrolyte abnormalities, and thrombosis were managed in coordination with a physician. Our experience shows that a multi-disciplinary team approach is required for the treatment of patients with underlying diseases.
Solitary neurofibroma is a very rare type of benign oral tumor. This report describes one such case arising in the maxilla. A 12-year-old girl, symptomless except for disturbed canine tooth eruption, was referred to our department. The tumor was surgically excised with the patient under general anesthesia. The tumor had a smooth surface and measured approximately 45 × 35 × 25 mm. Internally, the mass was solid, milky white. Histopathologically, the mass showed notable proliferation of spindle cells and reacted positively to immunohistochemical staining for S-100 protein. The histopathological diagnosis was a neurofibroma. Four years after operation, there has been no evidence of recurrence.
Tumor thrombus in the internal jugular vein is extremely rare in oral carcinoma. We report a case of buccal mucosal carcinoma accompanied by tumor thrombus in the internal jugular vein. A 74-year-old man was referred to our center in March 2008 because of ulceration of the right buccal mucosa. Both computed tomography and ultrasonography revealed multiple cervical lymph node metastases and thrombus formation in the internal jugular vein. The clinical diagnosis was a squamous cell carcinoma of the buccal mucosa (T3N2bM0). Surgical resection of the primary lesion and right neck dissection were performed. Metastatic lymph nodes were connected to the internal jugular vein by small fruit-tree-like vessels, and tumor thrombus extended from the metastatic lymph nodes to the internal jugular vein. Multiple lung and bone metastases occurred, and the patient died 13 months after the operation.
The development of methotrexate (MTX)-associated lymphoproliferative disorder (MTX-LPD) in the oral cavity is rare, despite numerous recent reports on the treatment of rheumatoid arthritis (RA) with MTX. An 84-year-old woman with RA, who was receiving MTX therapy since November 2008, presented to our hospital in April 2010. She complained of pain, and a nodule, with deep ulceration and induration, was seen extending from the right lower gingiva to the buccal mucosa. Histopathological examination of a biopsy specimen showed a diffuse proliferation of mostly large atypical lymphocytes. The tumor cells were positive for CD20, CD79a, MUM1, and LMP1, accompanied by EBER-ISH positive reaction. The condition was diagnosed as Epstein-Barr virus (EBV)-positive diffuse large B-cell lymphoma, which indicated MTX-LPD. MTX was discontinued. The tumor disappeared after 2 months. Within 8 months, the bone sequestrum spontaneously separated, and the affected area was covered with normal mucosa. Examination after 2 years 4 months did not show any evidence of recurrence.
Tocilizumab, a humanized anti-interleukin 6 receptor antibody, has been reported to be effective for chronic rheumatoid arthritis (RA) in clinical trials and was approved in Japan for the first time in the world in April 2008. We report a patient with methotrexate (MTX)-associated lymphoproliferative disorder in whom bisphosphonate-related osteonecrosis of the jaw (BRONJ) was exacerbated by concurrent treatment with tocilizumab. A 60-year-old woman was referred to our department by her dentist because of diffuse swelling of the right submandibular region and a right lower gingival ulcer. Computed tomography showed extensive swelling of the cervical, axillary, subscapular, and inguinal lymph nodes. Her medical history included RA. She had received MTX and sodium risedronate hydrate. Her past medical history and present illness strongly suggested BRONJ. Bisphosphonate administration was terminated, and BRONJ was treated conservatively with antibiotics and local irrigation. Because RA was getting worse, she was given tocilizumab. Subsequently, gingival necrosis and BRONJ was apparently exacerbated. Tocilizumab and MTX were withdrawn, and she received antibiotics and surgical treatment. There have been no signs of recurrence for 18 months.
Traumatic carotid-cavernous sinus fistula (traumatic CCF) is a rare disorder in which the internal carotid artery is torn and forms an abnormal interconnection with the cavernous sinus as a result of craniofacial injuries. We describe a case of traumatic CCF occurring during follow-up after open reduction and internal fixation of a mandibular fracture. The patient was a 56-year-old woman who was injured in a motorcycle accident. She had a mandibular fracture without skull base or midface fracture. Conjunctival hyperemia of the left eye, exophthalmos, and vascular bruits in the palpebral region developed 87 days after injury. Brain computed tomography revealed left exophthalmos and abnormalities in the cavernous sinus region, as well as superior ophthalmic vein distention. Left carotid angiography revealed shunt flow into the cavernous sinus, leading to a diagnosis of CCF. Left internal artery occlusion was performed using a Guglielmi detachable coil. The present case suggests that delayed cerebrovascular disorder can occur during postoperative follow-up, even in patients with mandibular fracture alone.
Rhabdomyosarcoma is histopathologically classified into fetal, alveolus nest, and pleomorphic types. Reportedly, the incidence is high in children, but rhabdomyosarcoma is relatively rare in the elderly. Treatment methods for this soft tissue tumor were investigated by the International Rhabdomyosarcoma Study, and therapeutic results have been improved, but outcomes remain poor. We encountered and treated an elderly patient with pleomorphic type rhabdomyosarcoma. We report the case, with a literature review. The patient was a 77-year-old man who presented with swelling of the left cheek. Diffuse swelling of the left cheek and an ulcer of the buccal mucosa were observed, and the patient was given a histopathological diagnosis of polymorphismtype rhabdomyosarcoma. Surgical treatment was performed without chemotherapy or radiotherapy in consideration of his past medical history. The clinical course has been favorable, with no recurrence or metastasis for 6 years 10 months since surgery. However, continued strict follow-up is considered necessary because there is no standard treatment for pleomorphic type rhabdomyosarcoma, and outcomes are generally poor.
Congenital factor X deficiency is a rare autosomal, recessive inherited bleeding disorder, with an incidence of 1 per 500,000 individuals. We diagnosed congenital factor X deficiency disease in a patient with persistent gingival bleeding before tooth extraction. The patient was a 47-year-old man who repeatedly had gingival bleeding after dental scaling. The medical history included excessive epistaxis when he was an infant and difficulty in hemostasis after tooth extraction. The details of his family history were unknown, except that his younger brother also appeared to have a bleeding problem. Intraoral examination showed persistent bleeding from the right, upper second molar, buccal gingival crevice and tooth mobility. A blood test showed a prolonged prothrombin time and activated partial thromboplastin time, which required appropriate consultation and careful examination. Factor X activity was found to be low (2.2%) and a definitive diagnosis of congenital factor X deficiency was made. Because of subsequent repeated gingival bleeding, severe marginal periodontitis in the region of the right upper second molar and periapical periodontitis in the region of the right lower first molar were diagnosed, and the teeth were extracted. Tooth extraction was performed after transfusion of fresh frozen plasma. The wound was filled with oxidized cellulose and sutured. Congenital factor X deficiency was diagnosed and treated in this patient with persistent gingival bleeding before tooth extraction.
Nevoid basal cell carcinoma syndrome (NBCCS), also known as Gorlin syndrome or Basal Cell Nevus Syndrome (BCNS), is an autosomal dominant disorder characterized by developmental defects, including palmar or plantar pits, bifid ribs, or calcification of the falx cerebri, and tumorigenesis, such as basal cell carcinoma (BCC), medulloblastoma, or keratocystic odontogenic tumor (KCOT). Human patched-1 (PTCH1) has been identified as the causative gene of NBCCS. A 7-year-old boy was referred to our hospital in July 2010 by a dental clinic because of radiolucent findings on the right side of the maxilla and the left side of the mandible. NBCCS was diagnosed on the basis of our clinical findings and the diagnostic criteria proposed by Kimonis et al. Fenestration surgery of 2 tumors in the maxilla and mandible was performed with the patient under general anesthesia. Histopathological findings indicated that the 2 tumors were consistent with a diagnosis of KCOTs. We subsequently identified a PTCH1 germline mutation of c.2250+2t>a, leading to abnormal splicing in PTCH1. This splicing mutation was novel, and premature termination of PTCH1 as demonstrated by reverse-transcriptase polymerase chain reaction apparently caused the NBCCS manifestations seen in this patient. There has been no complications after 1 year of postoperative follow-up.