Glossodynia is defined as a burning sensation on the tongue without any systemic or local cause. Although its pathophysiology remains unclear, it has been associated with neuropathic, nociceptive, and psychogenic pains. In Japan, difficulties are encountered in the treatment of glossodynia as prescribed medications are not covered by health insurance. Recently, a traditional Chinese medicine treatment (kampo), rikko-san (TJ-110), has been suggested to be useful for glossodynia. The objective of this retrospective study was to evaluate the efficacy of rikko-san gargling in patients with glossodynia. In total, 221 patients with glossodynia who were treated by rikko-san gargling between 2012 and 2018 were examined. Of these, 90 patients (seven men, 83 women; mean age, 67 years) who were treated by gargling with rikko-san dissolved in water for >1 month and no other medications were included. Tongue pain was assessed using a visual analog scale (VAS). Improvement was defined as at least a 50% reduction in the VAS score after treatment compared with that before treatment. The associations between the efficacy of rikko-san gargling and factors such as age, disease duration, pain area, and psychiatric disorders were evaluated. Improvement was noted in 60% of the patients (54/90) ; the mean administration period to the end of treatment in the improvement group was approximately 5 months. There was no significant correlation between the aforementioned factors and the efficacy of rikko-san gargling. Treatment in 41 patients was completed with rikko-san gargling alone, whereas 40 patients required additional treatment such as another kampo medicine or ethyl loflazepate. No side effect was observed in any patient, although stomatitis was noted in one excluded case.
The mechanism underlying rikko-san’s delivery pathway is distinct from that of selective serotonin reuptake inhibitors, benzodiazepines, and nonsteroidal anti-inflammatory drugs. We speculated that saishin (Asiasarum root), one of the major components of rikko-san, may reduce peripheral stimulation to the tongue surface through its anesthetic effect. In addition, the transmucosal actions of other components in rikko-san may influence various receptor-related neuropathic pains. In conclusion, rikko-san gargling is efficacious and safe for the treatment of glossodynia.
Congenital afibrinogenemia is a hemorrhagic disease caused by synthesis disorder of fibrinogen (Fbg). We report the perioperative management of tooth extraction in a patient with congenital afibrinogenemia. The patient was a 13-year-old boy. He was given a diagnosis of congenital afibrinogenemia based on abnormal umbilical cord bleeding at birth. He was referred to our hospital because of right cheek swelling. There was swelling accompanied by redness and warmth from the right cheek to the submandibular region. In the oral cavity, the crown of the right mandibular first molar had collapsed, and an abscess was palpated at the gingivobuccal fold. We diagnosed a subperiosteal abscess and phlegmon due to apical periodontitis of the right mandibular first molar. Because the blood Fbg concentration was below the detection limit in the clinical examination, incisional drainage was performed after infusion of Fbg preparation. After the inflammation resolved, he was admitted for tooth extraction and bleeding management. Fbg preparation was infused before surgery, and the blood Fbg concentration was 117 mg/dL. Local hemostatic treatments were carefully executed after tooth extraction, and Fbg preparation was infused so that the blood Fbg concentration was maintained at 100 mg/dL or higher. Thereafter, he has never had hemorrhage, and the postoperative course was favorable.
Sjjögren’s syndrome (SS) is a chronic autoimmune disease characterized by a progressive lymphocytic infection of the exocrine gland with varying degrees of systemic involvement. SS is thought to be rare in childhood. However, a recent epidemiological study revealed that the incidence of childhood SS per 100,000 children was more than 0.5. Here we report a case of SS in childhood. A 14-year old girl presented with a rash on her forearm and cheek, but did not complain of dry mouth. Salivary secretion was decreased, and moisture value was measured at the dry mouth level. Ultrasonography revealed multiple hypoechoic areas, hyperechoic bands, and a reticular pattern on salivary glands. On laboratory examinations, antinuclear antibody, anti Ro/SS-A antibody, and anti-La/SS-B antibody were positive. The histopathological finding in the minor salivary gland was chronic inflammation. A diagnosis of SS was made from these findings. We summarized previous reports on child patients with SS and compared it with clinical features in adult patients. Children with SS complain frequently of rash and fever, but rarely of a dry mouth.
Porphyria is a rare disease caused by genetic abnormalities of heme synthesis enzymes. Patients with porphyria may have lethal acute attacks induced by various stresses and drugs. We describe our experience in performing multiple tooth extraction safely in a 30-year-old woman. We carefully examined the drugs used and operated with the patient under sedation with intravenous anesthesia to reduce stress and were able to treat without inducing an acute attack.
Non-sebaceous lymphadenoma (NSL) is a rare benign salivary gland tumor that is difficult to diagnose preoperatively on the basis of clinical features. We report a case of NSL arising in the left submandibular region in a 66-year-old woman. The patient had complained of a painless mass in the left submandibular region. Her medical history included a left parotidectomy for adenoid cystic carcinoma (ACC) 22 years ago. Computed tomography showed an enhanced mass with the inside low-density at the distal part of the submandibular gland and no residual parotid gland. On the basis of these findings, lymph node metastasis from ACC or a submandibular gland tumor was suspected. The mass was removed with the left submandibular gland under general anesthesia. Histopathologically, the mass was diagnosed as a NSL. There has been no sign of recurrence as of 2 years after the operation.
May-Hegglin anomaly is a rare autosomal dominant inherited disorder characterized by giant platelets, thrombocytopenia, and inclusion bodies in granulocytes. Clinically, because of thrombocytopenia, a mild hemorrhagic diathesis may occur. We extracted the mandibular third molar in a patient with May-Hegglin anomaly whose platelet count was visually reduced to 1.6 × 104/μL. The patient was a 27-year-old woman, and the right mandibular third molar to be removed had completely erupted. In this case, good results were obtained with no postoperative bleeding only with local hemostasis (use of an absorptive hemostatic agent, gingival suture). In conclusion, in patients with May-Hegglin anomaly, even if the platelet count on visual observation is as low as 1.6 × 104/μL, tooth extraction is considered possible when an appropriate local hemostatic method is applied.
Hangeshashinto is an oriental medicine, usually prescribed for stomatitis in dentistry. Although drug-induced lung injury is a serious side effect of Oriental medicine, reports of injuries caused by hangeshasihnto are rare. We report a case of lung injury probably caused by hangeshashinto in a patient with chronic stomatitis. A 67-year-old woman visited a dental clinic with complaints of right buccal mucosa pain. Red lesions were present in the posterior region of the bilateral mandibular buccal gingiva and the surrounding buccal mucosa. Because gargling was ineffective, she was referred to our department. The lesions were diagnosed as chronic stomatitis by biopsy. We administered hangeshashinto, but the patient began to exhibit cough with small amounts of phlegm after 2 weeks. Additionally, exertional dyspnea gradually developed, and she visited a pulmonary medical clinic. Based on clinical examinations, blood tests, and chest CT findings, and the patient’s medical history, the diagnosis was lung injury probably caused by hangeshashinto. After hangeshashinto treatment was stopped, the patient’s symptoms improved after 1 week. The intraoral symptoms showed a tendency towards improvement, and the chest symptoms did not recur during 1 year 2 months of follow up. When hangeshashinto is prescribed, care must be taken regarding the possibility of serious side effects.
“ Kissing molars (KMs)” is a rare condition in which the root apices of two impacted molars face opposite directions so that the occlusal surfaces come into contact and the crowns are located within a single follicle.
We encountered a 29-year-old man in whom panoramic radiographs and computed tomography scans revealed impaction of the left mandibular third and fourth molars. The occlusal surfaces of the impacted molars were in contact with each other with the root apices facing opposite directions, and clearly delineated, unilocular, round and cystic permeation was observable surrounding the same crown. KMs are classified from Class I toⅢ at the site of impaction. In our patient, the third molar and the fourth molar (supernumerary tooth) were classified as Class Ⅲ. Tooth extraction and extirpation were performed with the patient under general anesthesia and the histopathological finding was a dentigerous cyst. The postoperative course has been good with no recurrence of the lesion for 18 months.
In our department, we have performed mandibular reconstruction using autogenous particulate cancellous bone and marrow (PCBM) harvested from the posterior iliac crest and a custom-made titanium mesh tray for extended mandibular defects caused by tumor resection and so on. However, harvesting PCBM from the posterior iliac crest has a disadvantage of postural changes during surgery. On the other hand, we can safely and easily harvest a sufficient quantity of PCBM from the tibia.
The patient was a 56-year-old man who underwent immediate mandibular reconstruction using PCBM harvested from the tibia and a custom-made titanium mesh tray after segmental mandibular resection for an extended odontogenic keratocyst in the right mandible. We could harvest approximately 50 g (approx. 49 mL) of PCBM from the unilateral tibia and achieve successful mandibular reconstruction with a natural configuration. Finally, the bone quality of the reconstructed mandible become good. Eleven months later, dental implant placement was performed, and the final occlusal reconstruction with a bone-anchored bridge was completed. The postoperative course has been functionally and aesthetically uneventful for 5 years after mandibular reconstruction. In addition, there were no complications, such as gait disturbance associated with PCBM harvested from the tibia.
Hemophilia A is a bleeding disorder caused by a deficiency of clotting factor VIII. Approximately 20% of the patients develop inhibitors in response to replacement therapy with factor VIII concentrates. Once inhibitors develop, hemostatic management becomes very difficult because replacement therapy is generally ineffective. We report a case of persistent post-extraction bleeding in a hemophilia A patient with high responding inhibitors. A 24-year-old man was referred to our department for dental treatment. He had a history level of treatment with factor VIII inhibitor (21 Bethesda units/ml). Clinical and radiological examinations revealed that the upper right second molar and left second premolar required dental extraction. We consulted his hematologist and performed dental extraction during factor VIII-bypassing therapy with recombinant activated factor VIIa (rFVIIa) according to the hemostatic treatment guidelines for inhibitors in patients with congenital hemophilia, 2013 revised edition. However, the hemorrhagic oozing out of the extraction sockets repeatedly occurred despite additional curettage of the sockets and application of a mouth splint. Then, we changed the bypass agent to activated prothrombin complex concentrates (aPCC), and successful hemostasis was achieved without serious complications. On the 18th postoperative day, complete epithelial closure of the sockets was observed.
Vascular malformations occur due to errors during vasculogenesis. The incidence of vascular malformations in the jawbone is reportedly rare. A diagnosis of vascular malformations in the jawbone is often difficult, and treatment requires precise identification of the location and extension of the tumor. We describe our experience with a case of mandibular capillary malformation in a 12-year-old girl. The patient was experiencing right side mental nerve desensitization and referred to our hospital. The clinical diagnosis at the first visit was a right mandible tumor. Although a biopsy was performed with the patient under general anesthesia, there was pulsatile bleeding in the mandible, and sampling was difficult. Arteriovenous malformation was suspected from the angiographic findings, and surgery after embolization was planned. Embolization of the facial artery, maxillary artery, and transverse facial artery was performed. Mandibular segmental resection and a vascularized iliac bone graft were performed after 2 days of embolization. A histopathologic diagnosis of capillary malformation was made. As of 1 year and 9 months after surgery, there is no recurrence with good aesthetics.