Pigmented villonodular synovitis occurring in the temporomandibular joint

ObjectivePigmented villonodular synovitis occurring in the region of the temporomandibular joint is a rare disease, requiring a review of the treatment method, follow-up period.MethodRefer to the past literature, along with a retrospective search.ResultsAn excision, including the skull base bone, was performed in all cases; however, recurrence was found in one case on which fractional excision was performed. Past reports have also indicated that en bloc resection was considered desirable.ConclusionIt is necessary to perform en bloc resection on patients with pigmented villonodular synovitis occurring in the region of the temporomandibular joint. Furthermore, due to reported cases of recurrence after a long period of time, follow-up observations of about 10 years are considered necessary.     

Foreign bodies in the ear,nose, and throat in Japan: association with sociocultural and geographical conditions

ObjectiveForeign bodies (FBs) in ear, nose, and throat (ENT) are common ENT emergencies but are sometimes life-threatening. However, FBs could be avoidable by the efficient announcement about the risk of these occurrence to the public. Fish bones are commonly found as throat FBs, and small toys are commonly found as pediatric ear and nose FBs. We hypothesized that there were relationships between the occurrence of FBs and sociocultural/geographical conditions. The purpose of this study is to clarify the risk factors of FBs in ENT regions related to eating customs and weather conditions.MethodsFrom April 2009 to March 2014, 94,479 patients visited the Chuo Emergency Clinic (CEC) in Osaka, which is a local emergency center for Osaka prefecture in Japan. Among them, 3229 patients with throat FBs, 577 children (0–15 years of age) with ear FBs, and 1999 children (0–15 years of age) with nose FBs were enrolled into the present study. Monthly trends in the number of throat FBs were examined in relation to fish eating customs. The monthly average of the daily ratio of pediatric patients with ear or nose FBs to the total number of patients were examined in relation to weather parameters using a database of the Japan Meteorological Agency.ResultsThe incidence of throat FBs was significantly higher in July and January (p < 0.05, analyzed by ANOVA and Tukey–Kramer test), presumably because Japanese people have more chances to eat fish in these months due to the traditional fish-eating customs. There was also a significant correlation between the number of pediatric patients with ear and nose FBs and the bad weather parameters including daily rainfall (r = 0.76, p = 0.0043; r = 0.57, p = 0.050, respectively, analyzed by the Pearson product-moment correlation coefficient). This is because children would spend longer time inside on rainy days, which increases the chance of putting a small toy part in the ear and nose.ConclusionsFBs in throat and ear/nose occurred more frequently in the specific periods to eat fish and rainy days, respectively. Therefore, public announcement on the risk of occurrence of FBs based on sociocultural and geographical data is helpful to prevent FBs.     

Development of a rat model of patulous eustachian tube by mandibular nerve resection

ObjectivePatulous eustachian tube (PET) is currently treated using a variety of conservative or surgical approaches. To further elucidate the pathology of PET and to establish new therapies, the development of an animal model is necessary. The objective of this study was to develop a highly reproducible and sustainable rat model of PET by mandibular nerve resection.MethodsSixteen rats underwent mandibular nerve resection. Following an external incision, the main trunk of the mandibular nerve at the foramen ovale was identified in the pterygoid fossa, and its branches were resected. This surgery was performed on the right side, with the unoperated left side used as the control. To determine eustachian tube (ET) function, passive opening pressure (POP) was measured using inflation method up to Week 16 post-surgery. Changes in POP were statistically compared to the preoperative level on the operated and control sides. In addition, specimens of the ET and its surrounding tissue at Week 16 post-surgery were prepared for morphological evaluation in eight rats.ResultsOn the control side, POP did not significantly decrease across the 16 weeks post-surgery compared to the preoperative level. In contrast, on the operated side, POP was significantly decreased at Week 2 and continued to be lower than the preoperative level until at least Week 16 post-surgery. POP dropped 10% or more on the operated side examined in thirteen rats. Histologically, the medial pterygoid muscle was atrophied on the operated side mainly.ConclusionMandibular nerve resection in rats may be an effective method for generating an animal model of PET. It was suggested that this rat model may be useful for verifying new treatments for PET.     

Cardiac metastasis in a living patient with oral cancer

Cardiac metastasis from head and neck cancers are very rare. Metastases to heart are mostly diagnosed at autopsy, and seldom found while patients are alive. Patients with cardiac metastasis do not present with specific symptoms in the early stages, and diagnosis is often delayed until the disease has advanced significantly. Here, we report a 66-year-old lady who was diagnosed with cardiac metastasis 10 months after surgical resection of oral cancer. She died one month following the discovery of cardiac metastasis. Cardiac metastasis should be considered when unexplained and progressive decline of general health is observed, even in the absence of abnormalities on the electrocardiogram. Early diagnosis may be made by analyzing the chronological changes in the cardiac accumulation of fluorodeoxyglucose during positron emission tomography–computed tomography scan.     

Metastasis of adenoid cystic carcinoma from a submandibular gland to the larynx

The present article discusses the first reported case of adenoid cystic carcinoma (ACC) metastasis from a submandibular gland to the larynx. Both treatments of distant metastasis of ACC and secondary laryngeal tumor are challenging. Despite its slow progression, ACC is associated with high rates of local recurrence, distant metastasis, and poor prognosis. Patients with secondary laryngeal cancer often have other concurrent metastatic lesions. Therefore, treatment selection should consider the biological behavior of the tumor and characteristics of the laryngeal lesion, along with the general condition and quality of life of the patient. The patient (55-year-old female) had a history of ACC of the right submandibular gland, removed surgically 9 years prior to the present consultation. Follow-up showed multiple pulmonary metastases. The patient complained of dysphonia lasting 3 months. Following the diagnosis of ACC metastasis to the larynx (supraglottic) and a neck lymph node via biopsy, we performed partial laryngectomy, left neck dissection, and tracheotomy. Histopathological examination showed an increase in the tumor grade over time. Two months after discharge, there was no obvious local recurrence or increase in lung metastasis.     

Direct approach to the anterior and lateral part of the maxillary sinus with an endoscope

ObjectiveThe Caldwell-Luc (CL) approach with a sublabial incision is used to manipulate the anterior wall of the maxillary sinus. Paresthesia is one of the major complications associated with the CL approach. We developed a new method, “direct approach to the anterior and lateral part of the maxillary sinus with an endoscope” (DALMA), that negates the need for a sublabial incision and minimizes dental paresthesia by reducing the risk of anterior superior alveolar nerve injury. This study aimed to describe how to perform the DALMA technique, and to review its effectiveness and associated complications.MethodsWe retrospectively reviewed 10 patients who had received DALMA.ResultsTen patients underwent DALMA. The anterior superior alveolar nerve was identified in all patients. Access to the lateral side beyond the infraorbital canal and anterior wall of the maxillary sinus was achieved.ConclusionsDALMA is a simple, effective, and minimally invasive technique that can be used as an alternative to CL with sublabial incision.     

Familial congenital choanal atresia with GATA3 associated hypoparathyroidism-deafness-renal dysplasia syndrome unidentified on auditory brainstem response

Hypoparathyroidism-deafness-renal dysplasia (HDR) syndrome is a rare autosomal dominant disorder primarily caused by GATA3 haploinsufficiency and is challenging to diagnose in early childhood. We report a Japanese family with HDR syndrome and congenital choanal atresia. The 6-year-old female proband was diagnosed with epilepsy at the age of three. Under carbamazepine monotherapy, the patient presented hypoparathyroidism accompanied by severe hypocalcemia. Subsequently, renal ultrasound analysis revealed bilateral multicystic dysplastic kidneys. Because she had difficulty hearing, we sequenced GATA3 and determined that she had a c.708_709insC (p.Ser237Glnfs*66) allelic variant in exon 3. As a result, we found a family of this disease. Each family member, including her grandfather, mother, and two siblings, had HDR syndrome of varying clinical penetrance. We found a craniofacial anomaly, congenital choanal atresia, which was inherited as an autosomal dominant trait. Hypocalcemia coupled with vitamin D deficiency, triggered by carbamazepine treatment, ultimately revealed the proband’s childhood- onset HDR syndrome. Pure-tone audiometry revealed different severities of deafness as well as the progression of sensory hearing loss. However, auditory brainstem response for hearing screening is probably insufficient for ascertaining HDR syndrome in the early stages of life. We presented new clinical clues to diagnose the HDR syndrome.     

Developmental delays assessed using the Enjoji Scale in children with cochlear implants who have intellectual disability with or without autism spectrum disorder

ObjectiveIntellectual disability (ID) and autism spectrum disorder (ASD) are common among children who are candidates for cochlear implants. However, the implications of these comorbidities for cochlear implant placement have been not fully established. This study sought to identify these implications by comparing developmental delays among children with these conditions.MethodsParticipants were children who were followed up at least every 6 months for 24 months after cochlear implant surgery. Developmental delays were assessed using the Enjoji Scale of Infant Analytical Development (Enjoji Scale) and compared in three groups with hearing loss: those with ID (ID group, n = 4); those with ASD and ID (ASD + ID group, n = 4); and those with typical development (control group, n = 5). Developmental delay was evaluated longitudinally before and after cochlear implant placement for 18 months.ResultsAmong the six subscales that make up the Enjoji Scale, language development and intelligence development were significantly delayed in all three groups and were exacerbated over time except for language development in the control group. Emotional development and social behavior were significantly delayed only in the ASD + ID group. Comparison of intergroup differences revealed delays in language development in the ID and ASD + ID groups compared with the control group.ConclusionThe Enjoji Scale successfully demonstrated developmental delays characteristic to the underlying comorbidities of ID with or without ASD in children with cochlear implants. The Enjoji Scale can be a useful diagnostic tool for screening children with cochlear implants for ID with or without ASD.     

Endocytosis of CF in marginal cells of stria vascularis regulated by ROCK and MLCK signaling cascade,but not G-proteins

Objective The endocytosis of cationized feritin (CF) via a clathrin-mediated pathway is regulated by a signaling network. Marginal cells showed the active endocytosis of CF via a clathrin-mediated pathway. The internalization of receptors through this clathrin-mediated pathway is an important regulatory event in signal transduction. Numerous kinases are involved in endocytosis, and each endocytic route is subjected to high-order regulation by cellular signaling mechanisms. In this study, we investigated whether ROCK and MLCK signaling cascades and G-proteins regulate the endocytosis of CF in marginal cells of the stria vascularis.Methods CF was infused into the cochlear duct with pertussis toxin (PTX),Clostridium botulinum C3 toxin (BTX), guanosine(g-thio)-triphosphate (GTP-γS), ML-7, Y-27632. Endocytic activity was measured at 30 min after the start of infusion under an electron microscope.Results In marginal cells, CF was internalized via a clathrin-mediated pathway that depends on F-actin and microtubules (MT). Its processes were controlled by myosin light chain kinase (MLCK) and Rho-associated kinase (ROCK), but not affected by G-protein-coupled receptor (GPCR) or the RhoA signaling cascade.Conclusion Our previous study showed that the main endocytotic pathway of microperoxidase (MPO) did not depend on the Rho/ROCK molecular switch or actin/myosin motor system, but was mainly regulated by the RhoA signaling cascade. The present study results indicate that these signaling cascades regulating CF internalization completely differ from the cascades for MPO internalization.     

Oral administration of an herbal medicine to prevent progressive hearing loss in a mouse model of diabetes

ObjectiveTsumura Suzuki Obese Diabetes (TSOD) mice exhibit early age-associated hearing loss. Histopathological analysis of these mice shows narrowing of capillaries in the stria vascularis and chronic reduction of blood flow in the cochlea. In this study, we investigated the effect of oral administration of a herbal medicine or calorie restriction on hearing in TSOD mice.MethodsTSOD mice were divided into 4 groups: CR (calorie restriction), BF and DS (treated with the herbal medicines, Bofutsushosan and Daisaikoto, respectively), and the control group. Body weight, blood glucose levels, and auditory brainstem responses (ABRs) were measured. The cochleae were excised and evaluated histopathologically.ResultsBlood glucose levels were suppressed in the CR, BF, and DS groups. In addition, the elevation of ABR thresholds was inhibited in the CR, BF, and DS groups. Cochlear blood vessels remained wide in the three treatment groups compared with the control group. These results suggested that the administration of these herbal medicines improved glucose tolerance and yielded results similar to those on calorie restriction.ConclusionOral administration of 2 herbal medicines can prevent hearing function disorder in a model mouse of diabetes. The results may clarify the possibility of clinical application.     

Role of phosphorylcholine in Streptococcus pneumoniae and nontypeable Haemophilus influenzae adherence to epithelial cells

ObjectivePhosphorylcholine (PC) is a structural component of Streptococcus pneumoniae (Spn) and nontypeable Haemophilus influenzae (NTHi), and is known to be associated with adherence through the platelet activating factor receptor (PAF-R). Furthermore, high PC expression is considered to be involved in Spn and NTHi virulence. In this study, we examined the influence of PC expression on the adherence of Spn and NTHi to epithelial cells in order to clarify the potential effectiveness of a vaccine targeting PC.MethodsTwenty-seven strains of Spn and twenty-two strains of NTHi were used, cultured overnight, and PC expression was evaluated by fluorescence activated cell sorting; the strains were divided into two groups: PC low expression (PC-low) and PC high expression (PC-high) groups. Bacterial adherence was then examined using Detroit 562 cells and BALB/c mice. Bacterial invasion was then examined in Detroit 562 cells.ResultsThe adherence of Spn and NTHi and invasion of NTHi in the PC-high group was significantly reduced by pretreatment with a monoclonal anti-PC antibody (TEPC-15), PAF-R antagonist (ABT-491), and PC-keyhole limpet hemocyanin (PC-KLH). However, such findings were not observed in the PC-low group.ConclusionThe present study suggests that PC is involved in the mucosal adhesion of Spn and NTHi, and the mucosal invasion of NTHi with PC-high strains, but not PC-low strains. These results suggest that a PC-targeting mucosal vaccine only affects PC-high Spn and NTHi strains and does not disturb commensal bacterial flora in the upper respiratory tract, which comprises nonpathogenic PC-low bacteria.     

Long-term efficacy and toxicity of concurrent chemoradiotherapy with nedaplatin and S-1 for head and neck squamous cell carcinoma

ObjectiveThe present study aimed to retrospectively analyze the long-term efficacy and toxicity of concurrent chemoradiotherapy with nedaplatin and S-1 for head and neck squamous cell carcinoma.MethodsThe study enrolled 53 patients (23 with stage II disease, 13 with stage III disease, and 17 with stage IV disease). S-1 was administered orally twice a day for 14 days, followed by a two-week rest period. Nedaplatin was intravenously administered on day 4. Where possible, two courses of chemotherapy were performed. Radiotherapy was started with the administration of S-1. We analyzed the clinical response, survival rate, acute adverse events, and late swallowing toxicity.ResultsThe complete response rates for the primary tumor and neck lymph node metastases were 94.3% and 79.3%, respectively. The five-year overall survival rate was 79.5%, the five-year disease-specific survival rate was 84.8%, and the five-year relapse-free survival rate was 73.7%. The main acute adverse events were leukopenia, neutropenia, mucositis, and dermatitis. No patient had severe nephrotoxicity. Late swallowing toxicity was observed in 13 patients.ConclusionsThe low toxicity, and low nephrotoxicity of chemoradiotherapy with nedaplatin and S-1 have a positive impact on long-term survival. The combination of nedaplatin and S-1 can be used instead of cisplatin and 5-?uorouracil as a safer regimen, especially in patients with some complications and those requiring treatment in an outpatient setting.     

Effectiveness of fibrin coating in the management of web formation after laryngomicrosurgery

Purpose

To explore the effectiveness of fibrin coating in reducing web formation after endoscopic management of the anterior commissure of the larynx.

Surgical treatments for a case of superior canal dehiscence syndrome associated with patulous Eustachian tube

ObjectivesThe patulous Eustachian tube (PET) and superior semicircular canal dehiscence syndrome (SCDS) have similarity in their symptoms and similar effects caused by positional changes, causing difficulty in the differentiation between the two disorders. This report describes a case of both SCDS and PET that was eventually successfully treated.MethodsA 68-year-old man presented with hyperacusis to his own footsteps and gait disturbance. He had been diagnosed as PET two years before and had been treated by insertion of a silicone plug (Kobayashi plug) at the other hospital. Clinical case records, audiological data, cervical vestibular-evoked myogenic potential (cVEMP), Eustachian tube function tests and computed tomography (CT) were taken in the sitting position.ResultsWhile the CT confirmed superior semicircular canal dehiscence, the results of cVEMP was not typical of SCD likely due to preexisting hearing impairment in the right ear with a history of middle ear surgeries for the treatment of PET. He received round window reinforcement (RWR) and achieved relief from his symptoms but six months after the surgery, he visited again with complaints of autophony of his own voice and breathing. The tympanic membrane was found to move synchronous with respiration, and Eustachian tube function tests and the sitting CT confirmed the recurrence of severe PET. He had his silicone plug exchanged (increase in size of the Kobayashi plug) and achieved relief from symptoms.ConclusionsThe present case was a rare instance showing that PET and SCDS can occur simultaneously in a patient. The patient achieved relief from symptoms after treatment with RWR and insertion of the Kobayashi plug.     

Antitumor miR-150-5p and miR-150-3p inhibit cancer cell aggressiveness by targeting SPOCK1 in head and neck squamous cell carcinoma

Objective

Our recent studies have revealed that both strands of pre-miRNAs, the guide strand and the passenger strand, are involved in cancer pathogenesis. Analyses of miRNA expression signatures by RNA sequencing in head and neck squamous cell carcinoma (HNSCC) showed that both of the strands of pre-miR-150 (miR-150-5p and miR-150-3p) were significantly downregulated, and that these miRNAs acted as antitumor miRNAs in HNSCC cells. The aim of this study was to identify oncogenic genes in HNSCC cells that were regulated by miR-150-5p and miR-150-3p.

Methotrexate-associated lymphoproliferative disorder with multiple pulmonary nodules and bilateral cervical lymphadenopathy

As has been well recognized, methotrexate (MTX) leads to a state of immunosuppression and can provide a basis for the development of lymphoproliferative disorders (LPDs). MTX-associated LPDs can affect nodal sites as well as extranodal sites, though the manifestation of an LPD in the form of multiple pulmonary nodules is rare. Here, we report two cases of MTX-associated LPD with multiple bilateral pulmonary nodules, which was a finding suggestive of lung cancer, and bilateral cervical lymphadenopathy. After withdrawal of MTX, the multiple bilateral pulmonary nodules and bilateral cervical lymphadenopathy disappeared without chemotherapy in both cases. From these results, patients with pulmonary nodules and cervical lymphadenopathy should be examined for head and neck malignant tumors. Also, physicians should carefully check the administration of MTX. In patients with an MTX-associated LPD, we need to make an early diagnosis and consider discontinuing the administration of MTX as soon as possible.     

Influence of pneumococcal conjugate vaccines on acute otitis media in Japan

Objective

This study investigated: (i) changes in the incidence of acute otitis media (AOM) following introduction of public funding for free inoculation with 7- and 13-valent pneumococcal conjugate vaccines (PCV7 and PCV13, respectively) and (ii) changes in the rate of myringotomies for AOM (MyfA) in children 1 year following the publication of the first edition of the clinical practice guidelines for the diagnosis and management of AOM in children in Japan.

Attachment-oriented endoscopic surgical management for inverted papillomas in the nasal cavity and paranasal sinuses

ObjectiveThe treatment of all forms of sinonasal inverted papilloma (IP) is a complete, wide, local resection. The main surgical purpose is to remove all diseased mucosa and mucoperiosteum, together with a cuff of normal-looking mucosa at the attachment site, followed by drilling and/or coagulation. Our aim is to present our experiences in endoscopic surgical management of IP by using attachment-oriented excision.MethodsWe present 20 cases of sinonasal IP. The data collected includes the histopathological diagnosis, staging, extension of the tumor, tumor attachment site, approach to surgery, serum squamous cell carcinoma antigen (SCCA) level, and recurrences.ResultsAll patients underwent endoscopic surgery. A Caldwell-Luc operation was required in addition to the endoscopic surgery in one case. There was one case of recurrence (5%). After the additional operation, there was no recurrence. The tumor attachment sites vary, and the case of recurrence had a wide attachment site at the primary surgery. No major intra- or post-operative complications were observed.ConclusionThe present study shows that attachment-oriented excision for IP is useful for complete resection of IP. Surgeons should choose the surgical approach according to the location of the tumor attachment site rather than the Krouse staging system.