A Rare Case of Sinonasal Seromucinous Hamartoma Developing from the Nasal Septum.

A 45-year-old man presented with a history of chronic left nasal congestion. Nasal endoscopy revealed a pedunculated polypoid mass with glandular epithelium surface on the posterior nasal septum. Computed tomography revealed a 25-mm mass-like growth in the left posterior nasal cavity attached to the nasal septum with a stalk. The patient underwent transnasal endoscopic surgery, and the tumor was removed under a block with safety margin. The final pathological diagnosis was sinonasal seromucinous hamartoma (SH). Sinonasal SH is a rare tumor with only 31 reported cases. Transnasal endoscopic surgery is currently the first-line treatment for sinonasal SH. Differential diagnoses of this lesion include inflammatory polyps, respiratory epithelial adenomatoid hamartoma, and adenocarcinoma. Although SH is a benign tumor, its progression to adenocarcinoma has been reported. Therefore, unilateral posterior nasal tumors must be diagnosed precisely.

A Rare Case of Scalp Metastasis from Encapsulated Angioinvasive Follicular Thyroid Carcinoma.

Cutaneous metastases from thyroid carcinomas are extremely rare; however, the scalp is a common site for cutaneous metastases from follicular thyroid carcinomas (FTCs). We report the case of a 77-year-old male patient with a blood-rich scalp lesion. Histopathological tests of punch biopsy specimens revealed subcutaneous well-formed follicular structures that were similar to those found in the thyroid gland. Immunohistochemistry using thyroid transcription factor-1 (TTF-1) and paired-box gene family 8 (PAX8) revealed an FTC metastasis. We performed total thyroidectomy and resection of the scalp lesion at the same time and administered postoperative radioactive iodine treatment. The primary thyroid lesion was diagnosed as an FTC based on extracapsular extension and vessel invasion. The patient has not experienced disease recurrence since the treatment. When scalp metastasis of thyroid carcinoma is suspected, we recommend total extirpation, including the primary tumor and scalp metastasis, for an improved prognosis.

Primary diffuse large B-cell lymphoma of the Larynx.

The larynx is a rare site of extranodal non-Hodgkin lymphoma (NHL), accounting for less than 1% of all primary laryngeal neoplasms. We report a rare case of laryngeal diffuse large B-cell lymphoma (DLBCL) in an 85-year-old female patient, which was difficult to diagnose even after several biopsies from the primary laryngeal lesion, both under local and general anesthesia, and the diagnosis of DLBCL was obtained from the lymph node biopsy, which appeared in the proximity of the larynx 2 months after the first biopsy from the larynx. Since the diagnosis of laryngeal NHL is sometimes difficult when sufficient samples cannot be obtained, repeated biopsies may be required. Due to the small number of cases, there is no definite consensus regarding the best management of laryngeal NHL. Thus, a standard treatment option for DLBCL, such as 3 courses of R-CHOP (rituximab + cyclophosphamide, doxorubicin, vincristine and prednisone) followed by Involved-field radiation therapy, or 6∼8 course of R-CHOP, are also applied for the treatment of laryngeal DLBCL. For this case, doxorubicin was not adopted and 8 courses of R-COP (rituximab + cyclophosphamide, vincristine and prednisone) at a decreased dose were chosen because of her age (85-year-old) and cardiac hypofunction.

Impact of radiotherapy for head and neck cancer on obstructive sleep apnea: a prospective study.

BACKGROUND: In recent years, a relatively high prevalence of obstructive sleep apnea (OSA) in patients following radiotherapy (RT) for head and neck cancer (HNC) has been reported; however, little is known regarding the impact of RT on sleep disorders and the underlying mechanisms. This aim of this study was to elucidate the pathogenesis of OSA by comparing the clinical and sleep test parameters and magnetic resonance imaging (MRI) findings before and after HNC treatment with radiation. METHODS: This prospective study included patients scheduled for RT with or without chemotherapy or bioradiotherapy for HNC. Patients diagnosed with HNC between May 2017 and August 2020 were consecutively recruited. The results of the sleep tests were analyzed both before and after treatment. The clinical characteristics of the patients and cephalometric and MRI parameters were also measured. RESULTS: First, a total of 32 patients (64.8±11.8 years old; BMI, 22.7±3.6 kg/m2) underwent pre-treatment sleep tests. The prevalence of OSA [apnea hypopnea index (AHI) ≥5] in these patients was 81.3% (26 patients) before treatment, and the mean AHI was 20.8±19.0 events/hr. Next, 21 patients performed a sleep test both before and after treatment. Regarding subjective symptoms, there were no significant differences in the Epworth Sleepiness Scale (ESS) (P=0.142) or Pittsburgh Sleep Quality Index (PSQI) (P=0.935) after treatment; however, the BMI and neck circumference significantly decreased after treatment (P<0.0001 and P=0.0001, respectively). The incidence of OSA in these patients was 81.0% (17 patients) before treatment and 85.7% (19 patients) after treatment (P=1.0). Overall, the AHI was not significantly different, changing only from 14.5 to 14.9 after treatment (P=0.147). The MRI parameters showed that the retroglossal pharyngeal area increased significantly after treatment (P=0.007). CONCLUSIONS: This study found that the prevalence of OSA before and after RT for HNC was higher than that in the normal population, despite a significant decrease in BMI and increase in the retroglossal pharyngeal area after treatment. We suggest that physicians who manage patients with HNC should consider the occurrence of OSA before and after treatment.

Basal cell adenocarcinoma of the maxillary sinus: A case report.

Basal cell adenocarcinoma is a low-grade malignancy of the salivary glands. Basal cell adenocarcinoma of the minor salivary gland is an extremely rare disease that originates from the maxillary sinus. The histopathological characteristics of basal cell adenocarcinomas are similar to those of basal cell adenomas. However, basal cell adenocarcinomas can be differentiated from basal cell adenomas based on their tendency to invade surrounding tissues. Surgical resection is the first-line treatment for basal cell adenocarcinomas. An 86-year-old man underwent operations for a maxillary sinus tumor twice in our department. The pathological results of the tumor at both times revealed basal cell adenoma. After 4 and 5 years since the last operation, the tumor recurred, and the patient was treated with partial maxillectomy using Weber-Ferguson incision. We observed invasions to the surrounding tissue, and based on immunohistochemical findings, the patient was diagnosed with basal cell adenocarcinoma. Herein, we present an extremely rare case of basal cell adenocarcinoma arising from the maxillary sinus, in detail.

Adenoid Cystic Carcinoma Developed from the Parotid Gland to the Ear Lobe of a Young Woman.

Adenoid cystic carcinoma (ACC) is a relatively rare malignant tumor. It is more common in women than in men and typically develops in the lacrimal, salivary, and breast glands. ACC of the external auditory canal (EAC) is exceedingly rare, and its invasion into the ear lobe is even more unusual. In this report, we present a case of ACC that presented as a mass on the surface of the ear lobe in a 28-year-old woman and was initially diagnosed as infected atheroma. For wide resection of the tumor, half of the entire auricula was resected and superficial parotidectomy was performed. After confirming no tumor cells on the surface of the facial nerve, the defect was reconstructed by the combination of platysma muscle flap to prevent Frey syndrome and free forearm flap for the ear lobe form. There was no recurrence or metastasis of the tumor, and Frey syndrome did not occur at 2 years and 8 months after surgery. The patient was satisfied with the result, oncologically and cosmetically. Even in young patients, comprehensive treatments (including diagnosis, resection, and reconstruction) are important in painful ear lobe masses.

A case of ectopic salivary gland of the larynx.

A 46-year-old man presented with sore throat. Laryngoscopic findings revealed a smooth yellow mass occupying the anterior portion of the false vocal fold on the left side. The authors performed biopsy under general anesthesia. The histopathological diagnosis was ectopic salivary gland. Because salivary glands are usually not found under the false vocal fold mucosa, ectopic salivary gland of the larynx was diagnosed. It is necessary to consider the possibility of ectopic salivary gland for mass lesions if swelling of the provisional vocal cord is found.

A case of multiple empyema caused by Streptococcus intermedius.

We report the case of a patient with multiple empyema present throughout his body, including chronic sinusitis and chronic suppurative otitis media, as well as subsequent epidural empyema, all caused by Streptococcus intermedius. A 38-year-old man presented with chief complaints of headache, left ear discharge, and nasal congestion. Imaging studies revealed pansinusitis, soft tissue signs in the mastoid cells, and otitis media. The patient was treated with meropenem hydrate, 6g/day. While clinical findings indicated improvement of the sinusitis, his headache did not improve. Further examination with contrast computerized tomography (CT) 'a chest radiography' blood cultures were performed, and the patient was diagnosed with multiple empyema (with an epidural empyema, pulmonary suppuration) caused by S. intermedius. Subsequent burr hole drainage was implemented to drain the epidural empyema. Long-term administration was required to treat pulmonary suppuration. While they remain rare, there has been a recent upward trend in the frequency of cases in which a young, previously healthy patient has developed multiple empyema throughout their body despite the absence of complicating diseases that pose an immune deficiency risk, such as diabetes or infection with the human immunodeficiency virus (HIV). In order to properly diagnose and treat patients presenting with multiple empyema infection with S. intermedius should be included in the differential diagnosis.

Manometric assessment of pharyngeal swallowing pressure after mandibular reconstruction.

Oral cancer patients who undergo mandibular bone partial resection often exhibit swallowing disturbance after surgery. We examined the usefulness of manometry in obtaining quantitative data on swallowing function in patients after mandibular bone partial resection. Postsurgical swallowing function was investigated, using a combination of videofluorography (VF) and manometry, in five patients with oral cancer, who underwent mandibular bone partial resection. Three patients underwent reconstruction of soft tissue defects only, while the remaining two patients underwent reconstruction of both soft tissue defects and a bony segment using a reconstruction plate or vascularized fibula with a skin paddle. Oropharyngeal swallowing pressure ranged from 61 to 82 mmHg compared to the normal range of 109 ± 31 mmHg (mean ± SD). Partial resection of the mandible without reconstruction of the bony segment defect had a negative effect upon oropharyngeal swallowing pressure. Four of the five patients exhibited normal function in the oral stage. Elevation of the larynx was disturbed in 80% of the patients. None of the patients in this study experienced aspiration after surgery. In conclusion, a combination of VF and manometry revealed that mandibular bone partial resection resulted in disturbed elevation of the larynx while pharyngeal swallowing pressure decreases in those patients who do not undergo bony segment reconstruction. Our results suggested that the tongue, including the base of the tongue, should be set in a closed space so that pharyngeal swallowing pressure does not release.

Leiomyosarcoma of the hypopharynx: a case report.

Cases of leiomyosarcoma in the head and neck are very rare with only four reports of a leiomyosarcoma originating in the hypopharynx. A rare case of leiomyosarcoma of the hypopharynx is described. A 62-year-old woman, who had experienced slight difficulty of swallowing and hoarseness, visited our hospital. A smooth-surfaced tumor was found extending from the postcricoid to an area in the left pyriform sinus, and the vocal cord on the left side was fixed. Imaging studies, including FDG-PET CT imaging showed the tumor in the postcricoid, but no distinguishable distant metastases. A total laryngectomy with a left-lobe thyroidectomy was performed. Since no metastases of the regional lymph nodes were found by imaging, a neck dissection was not performed. Dense infiltration by spindle-shaped and/or round-like cells was observed histologically as the predominant growth pattern. In immunohistochemical staining, the tumor was positive for desmin and muscle actin, while it was negative for myoglobin, myogenin, cytokeratin, and S-100 protein. Electron microscopic examination did not show Z banding and sarcomeres. Those features lead to the diagnosis of a leiomyosarcoma. No recurrence has been found at 1 year after the surgery. Immunohistochemical staining and electron microscopic examination are essential for a definite diagnosis of leiomyosarcoma.

Gel chromatographic characterization of proteins in mucous and serous middle ear effusions of patients with otitis media in comparison to serum proteins.

Otitis media with effusion (OME) is an inflammatory disease of the middle ear cavity that is associated with middle ear effusions (MEEs), which are frequently mucous and serous for pediatric and adult patients exhibiting low and high responsiveness to medical treatment, respectively. To assess the pathological outcomes in mucous and serous MEEs, their protein compositions were determined by sodium dodecyl sulfate-polyacrylamide gel electrophoresis and Western blotting in comparison with those in the same patients' sera. A mucin, which is immunochemically identical with nasal mucin, was a characteristic consituent of mucous MEEs (n = 25), being present at the concentration of 59.4 mg/ml and comprising about 60% of the total proteins, but it was not detected in serous MEEs (n = 30) or sera. Serum proteins with molecular weights of less than 260 kDa were detected in serous and mucous MEEs, in which albumin was the major protein. Albumin, IgM and alpha1-acid glycoprotein, and lysozyme, IgA and IgG in MEEs were present at lower and higher concentrations than in sera, respectively. The ratios of IgA, IgG, IgM and alpha1-acid glycoprotein to albumin in mucous MEEs were 4-, 3-, 1.4- and 1.0-times higher than those in the respective pediatric sera, and those in serous MEEs were 1.7-, 1.7-, 0.6- and 0.3-times higher than those in adult sera. Also, the concentrations of lysozyme in mucous and serous MEEs were 19 and 3 microg/ml, but those in pediatric and adult sera were negligible. These results indicate that the contents of these proteins, in comparison to albumin, might be useful criteria for assessing the inflammation level in MEEs.

An effective management regimen for laryngeal granuloma caused by gastro-esophageal reflux: combination therapy with suggestions for lifestyle modifications.

According to our previous study, lifestyle modification in combination with drug therapy is much more effective than drug therapy alone in cases of laryngopharyngeal reflux (LPR). Most patients with LPR will have chronic gastro-esophageal reflux diseases (GERD) and require long-term medical therapy for control, resulting in high total expenditure on pharmacologic agents. We combined pharmacologic management with lifestyle modifications for the management of GERD with successful outcomes in patients with GERD-related laryngeal granulomas. Although further studies are needed, guidance concerning lifestyle modifications in combination with PPI therapy may be not only a clinically effective but also a cost-effective method for the management of laryngeal granulomas caused by gastro-esophageal reflux.

Salivary duct carcinoma in the sinonasal tract.

Salivary duct carcinoma (SDC) is an uncommon malignant tumor, characterized by aggressive behavior and poor prognosis. SDC usually arises from ductal epithelium of the major salivary glands, and it is quite infrequent elsewhere. We present a rare case of a 73-year-old man with SDC, which is possibly originated from the paranasal sinuses or the lacrimal system. Microscopic evaluation revealed that the tumor cells, with pleomorphic nuclei and abundant eosinophilic cytoplasm, formed cell nests and duct-like structure. A cribriform growth pattern was also seen. Immunohistochemical staining was positive for cytokeratins (CAM 5.2 and 34betaE12), gross cystic disease fluid protein 15 (GCDFP-15), and androgen receptor protein, while p63 and involucrin were negative. The patient already had multiple metastasis of the tumor in the lung at diagnosis, and he could not undergo definitive surgical procedures, because of severe restrictive lung disease. Although SDC in the sinonasal tract is quite rare, SDC should be in the differential diagnosis in these regions, due to its aggressive behavior and poor prognosis.

Videofluoroscopic assessment of swallowing function in patients with myasthenia gravis.

Swallowing function in myasthenia gravis (MG) was investigated by videofluoroscopy (VF). A total of 23 VF examinations were performed on 11 MG patients at various time points over the exacerbation and remission stages of disease. The assessment parameters on VF examination are set as follows: bolus transport from the mouth to the pharynx, bolus holding in the oral cavity, velopharyngeal seal, tongue base movement, pharyngeal constriction, laryngeal elevation, upper esophageal sphincter (UES) opening, and bolus stasis at the pyriform sinus (PS). Aspiration was also assessed on VF examination. Aspiration was seen in 34.8%, and half of these cases involved silent aspiration. Three of four cases that showed silent aspiration went on to experience aspiration pneumonia during the follow-up term. By Spearman's rank correlation, disturbance of laryngeal elevation was significantly correlated with aspiration (p=0.001), and incomplete UES opening was not significantly, but tended to be, correlated with aspiration (p=0.067). Although other parameters in the oral and pharyngeal phase on VF examination, such as bolus transport from the oral cavity to the pharynx, pharyngeal constriction, or stasis at the PS, were remarkably disturbed (in more than 50% of the examinations), those parameters were not good indicators for aspiration. When a disturbance of laryngeal elevation is found at a bedside clinical test, we recommend performing precise swallowing evaluation, such as VF, Fiberoptic Endoscopic Evaluation of Swallowing (FEES), and/or scintigraphic assessment of swallowing, for MG patients, to detect silent aspiration.

Swallowing function in patients with multiple-system atrophy with a clinical predominance of cerebellar symptoms (MSA-C).

We investigated the swallowing function of 21 patients with multiple system atrophy with a clinical predominance of cerebellar symptoms (MSA-C) by videofluoroscopy (VF). Twenty-six VF examinations were performed at various time points, and they were divided into three groups according to the duration following disease onset: Group A had 1 to 3 years following disease onset (the early stage of the disease), group B had 4 to 6 years following disease onset (the middle stage of the disease), and group C had more than 7 years following disease onset (the late stage of the disease). Swallowing function in the oral phase became gradually disturbed over the progression of MSA. Delayed bolus transport from the oral cavity to the pharynx was already seen in 50% of the patients in group A, and it was seen in more than 85% of the patients in group C. Bolus holding in the oral cavity was slightly disturbed in group A, but it was seen in 57% of the patients in group C. Our study shows that parkinsonism is related to swallowing dysfunction in MSA, but cerebellar dysfunction also affects coordination of the tongue; bolus transport in the oral cavity was disturbed in the early stage of disease. Progression of cerebellar dysfunction and overlapped parkinsonism will worsen tongue movement, and in the late stage of the disease, swallowing function of the oral phase (bolus transport and bolus holding) was remarkably disturbed. Swallowing function in the pharyngeal phase was not significantly correlated to the duration of the disease; however, our study showed that swallowing function in the pharyngeal phase was not assessed fully by VF examination in MSA-C only. Combination with other examinations, such as manometry and electromyography, may be useful, especially in the late stage of the disease. In addition, an analysis concerning the relationship between aspiration seen on VF examination and a history of aspiration pneumonia in MSA-C patients suggested that the sensory system at the larynx and trachea should also be assessed in patients in the late stage of MSA-C.

Repeated postoperative laryngeal obstruction due to bilateral cricoarytenoid joint involvement in rheumatoid arthritis.

A 61-year-old woman with a 40-year history of severe rheumatoid arthritis developed postoperative laryngeal obstruction twice; the first episode occurred just after surgery for cervical myelopathy and the second episode occurred 3 weeks after surgery when physiotherapy activity increased. A flexible laryngeal endoscopy demonstrated that abduction of the bilateral vocal cords was severely disturbed at the paramedian position, suggesting a diagnosis of cricoarytenoid arthritis. Early administration of corticosteroids was successful.

Longitudinal analysis of progression of dysphagia in amyotrophic lateral sclerosis.

OBJECTIVE: To investigate the progression of dysphagia in amyotrophic lateral sclerosis (ALS) using videofluoroscopy (VF), according to two different scales: the duration following bulbar symptom onset, and the ALS severity scale (ALSSS). METHODS: Seventy-two VF examinations, on 50 ALS patients, were performed at various time points, and the change in VF findings in the oral and pharyngeal phases was analyzed over time following bulbar symptom onset. The change according to advancement of the ALS severity scale was also assessed. RESULTS: Delayed bolus transport from the oral cavity to the pharynx, and bolus stasis at the pyriform sinus (PS), were seen in about half of the patients with no bulbar complaints. In contrast, upper esophageal sphincter (UES) opening was relatively well maintained in the late stage of dysphagia. The other parameters, such as bolus holding in the oral cavity, constriction of the pharynx, and elevation of the larynx, became worse over time following bulbar symptom onset. These parameters on VF examination also became worse as the ALSSS advanced; however, even patients in the group with normal eating habits showed disturbed bolus transport from the mouth to the pharynx, weak constriction of the pharynx, and bolus stasis at the PS. Generally, dysfunction had progressed dramatically, in most of the parameters, in the patients that required modification of food; however, UES opening and elevation of the larynx were relatively well maintained in advanced-ALSSS patients. CONCLUSION: In this study, we clearly show the progression pattern of dysphagia in ALS, and we also emphasize that VF is a useful tool to follow swallowing function in ALS patients. From our results, it would be useful to perform VF by 6 months following bulbar symptom onset, because VF can contribute to rehabilitation or appropriate diet modification; and later, around 1 year following bulbar symptom onset, VF should again be performed, since ALS patients show wide-ranging nutrition intake patterns, and they need assessment to introduce intervention, such as tube feeding, according to their varying status of dysphagia, around at this time point.

Foreign bodies in the aerodigestive tract in pediatric patients.

OBJECTIVE: To investigate pediatric foreign body cases in the aerodigestive tract, and to elucidate the characteristic problems in Japan. METHODS: A total of 310 pediatric patients (age 15 or below), gathered from two medical university hospitals (University of Tokyo and Jichi Medical School), were included in this study. Data were collected by retrospective chart review and were statistically analyzed. RESULTS: Two-year-olds were the most common patients, and the range from age 1 to age 4 included 67.7% of all the patients. The most involved sites were the nose (39.4%) and the pharynx (38.4%), followed by the esophagus (12.9%) and the trachea-bronchi (6.5%). Fish bones and toys were the representative foreign bodies (30.7 and 13.6%, respectively). Other foreign bodies often encountered included coins, food, candy, peanuts and nuts, and batteries. The type of foreign body was closely related to the site in which foreign bodies were lodged: 77.3% of foreign bodies in the pharynx were fish bones, and toys were the most common impacted foreign body in the nose. In the esophagus, representative impacted foreign bodies were coins (35.7%), but disk-type battery ingestion has been increasing in recent years. Although most foreign bodies in the esophagus were safely removed, one case of a disk-type battery had a serious sequela. In the trachea-bronchi, peanuts, food, cotton, and coins were impacted. A rigid bronchoscope was basically used to remove foreign bodies, but in some cases, a fine flexible endoscope, with a channel for fiber forceps, was useful, because it could be inserted into narrow bronchi. Advance of a flexible endoscope will make it an excellent tool for diagnosis and management of the trachea-bronchial foreign body. CONCLUSIONS: Fish bones in the pharynx, which were closely related to Japanese eating habits, and toys in the nose, were the typical foreign bodies encountered in this study. In the esophagus, an increased incidence of disk-type battery ingestion has become a serious problem in recent years. Since prevention is the most essential way to manage foreign body cases, feedback from studies to public education should be encouraged.

Videofluoroscopic and manometric evaluation of swallowing function in patients with multiple system atrophy.

We investigated swallowing function of 29 patients with multiple system atrophy (MSA) by videofluoroscopy and manometry. Abnormal findings in videofluoroscopy were generally consistent with those in Parkinson's disease. Although findings of videofluoroscopy were not correlated with a history of aspiration pneumonia, severity of disease was significantly correlated with a history of aspiration pneumonia. Oropharyngeal and hypopharyngeal swallowing pressures of the patients were decreased to 73.9 +/- 48.4 mm Hg and 85.3 +/- 42.9 mm Hg, respectively, both of which were significantly different from the pressures of the control group. Incomplete relaxation of the upper esophageal sphincter was seen in 23.1% of the MSA patients, all of whom had had MSA for more than 5 years. In conclusion, patients with MSA are at risk for aspiration pneumonia as disease severity increases, and the swallowing function of patients with more than 5 years' duration of MSA should be routinely followed up with both videofluoroscopy and manometry.