A Case of Unilateral Recurrent Nerve Palsy with Chronic Inflammatory Demyelinating Polyradiculoneuropathy.

Chronic inflammatory demyelinating polyradiculoneuropathy (CIDP) is a disease in which peripheral sensory and motor nerves of the four limbs are impaired due to autoimmune mechanism-induced demyelinating changes through a 2-month or longer chronic course. The incidence of complication by cranial neuropathy has been reported to be 15%, but there have been very few reports on disorder of the vagus nerve and its branch, the recurrent nerve. We report a patient who developed left recurrent nerve palsy with CIDP. The patient was a 48-year-old male. The disease developed as progressive muscle weakness and numbness of the four limbs 3 years before and was diagnosed as CIDP. The symptoms had been improved by high-dose intravenous gamma-globulin therapy. However, from 2 months before he became aware of breathy hoarseness, and bilateral decreased grip strength and sensory disturbance of the upper and lower limbs recurred and progressed. On laryngoscopy disorder of left vocal fold movement and glottal closure incompetence during phonation were observed, and neurogenic changes were detected in the left thyroarytenoid muscle by needle electromyography for the intrinsic laryngeal muscles. High-dose intravenous gamma-globulin therapy was performed and left vocal fold movement recovered with recovery of bilateral grip strength and sensory disturbance of the upper and lower limbs, and phonation was also normalized.

Nerve fiber analysis of the lingual branch of the glossopharyngeal nerve in human adult subjects.

PURPOSE: Fibers of the glossopharyngeal part of the superior constrictor muscle are connected with fibers of the transverse lingual muscle, forming a ring of muscle at the base of the tongue. This group of muscles constrict the midpharyngeal cavity during retrusive movement of the tongue. The purpose of this study is to identify the contribution of the lingual branch of the glossopharyngeal nerve to the neuro-motor control of three muscles: the glossopharyngeal part of the superior pharyngeal constrictor muscle, the palatopharyngeal and the palatoglossus muscles. METHODS: Six en bloc samples (9 sides), including the tissue from the skull base to the hyoid bone were obtained from adult human cadavers. Nerve fiber of the lingual branch of the glossopharyngeal nerve (main root of the glossopharyngeal nerve) was examined by the use of a binocular stereomicroscope. RESULTS: We observed that, after branching to the stylopharyngeal muscle, the lingual branch of the glossopharyngeal nerve branched to the glossopharyngeal part of the superior pharyngeal constrictor muscle, the palatopharyngeal and the palatoglossus muscles before inserting into the space between the muscle layers of the superior and middle pharyngeal constrictors. CONCLUSION: These neuromuscular arrangements may suggest the presence of specialized constrictive movements of the midpharygeal cavity at the level of the base of the tongue with the retrusive movement of the tongue. The simultaneous contraction of the palatopharyngeal and palatoglossus muscles on the pharyngeal stage of deglutition may aid in the passage of bolus from the oral cavity to the midpharyngeal cavity by increasing pharyngeal pressure.

[Effect of laryngeal closure on recurrent aspiration pneumonia in patients with neurodegenerative disease].

Three patients with neurodegenerative diseases who had developed repeated aspiration pneumonia underwent laryngeal closure, a surgical procedure at the larynx to prevent aspiration. None of these patients have developed aspiration pneumonia since the procedure. One patient needed endoscopic suction and cough assist machine to clear thick sputum, because tracheostomy bypassed the upper airway and so prevented moisturization of inhaled air. While two patients achieved freedom from tracheal cannulation, one needed continued cannulation because of narrowing of the stoma due to improvements in the nutritional condition. One patient was able to resume oral intake. Although the right timing to perform the procedure and optimal care along with long-term observation are important, laryngeal closure is an effective option for patients with neurodegenerative diseases to prevent recurrent aspiration pneumonia.

Thyroid-like low-grade nasopharyngeal papillary adenocarcinoma with squamous differentiation: a novel histological finding.

Thyroid-like low-grade nasopharyngeal papillary adenocarcinoma (TL-LGNPPA) is an extremely rare neoplasm originating from the nasopharyngeal surface epithelium. Histopathologically, TL-LGNPPA is characterized by cuboidal/columnar tumor cells forming papillary fronds and thyroid transcription factor-1 (TTF-1) expression resembling papillary thyroid carcinoma. To date, the recorded histological features of TL-LGNPPA have been almost uniform, and the range of histological variations in this tumor type has not been sufficiently understood. Here, we report on a 68-year-old man with TL-LGNPPA. Microscopic examination of the resected tumor revealed findings typical of papillary adenocarcinoma of this type, and moreover, this case showed scattered squamous cell foci as a hitherto unreported finding. The squamous cells showed no obvious nuclear atypia or proliferating activity, and their presence was similar to the "squamous metaplasia" of papillary thyroid carcinoma. Immunohistochemically, p40 and TTF-1 coexpression was observed in the squamous cell nuclei, indicating their origin from the glandular tumor cells of TL-LGNPPA.

[Safety and efficacy of surgical closure of the larynx for recurrent aspiration pneumonia in persons with severe motor and intellectual disabilities: a comparative study with tracheoesophageal diversion].

OBJECTIVE: We retrospectively investigated the efficacy and complications of surgical closure of the larynx (SCL) for recurrent aspiration pneumonia in comparison with tracheoesophageal diversion. METHODS: The subjects were persons with severe motor and intellectual disabilities (SMID) who had undergone surgery for recurrent aspiration pneumonia between 1994 and 2011: A 8 SCL patients group and a 16 tracheoesophageal diversion patients group. We investigated two groups the lower respiratory infection incidence, length of hospital stay for the surgery, postoperative complications, and rate of cannula withdrawal, by reviewing medical records. RESULTS: Both the SCL and the tracheoesophageal diversion group showed a reduction in the incidence of infection after surgery, indicating that the efficacy of SCL was equivalent to that of tracheoesophageal diversion in preventing aspiration pneumonea. The SCL group showed a reduction in the length of hospital stay and an increased rate of cannula withdrawal as compared with the tracheoesophageal diversion group. CONCLUSION: The efficacy of SCL was equivalent to that of tracheoesophageal diversion in preventing aspiration for SMID. We consider SLC to have potential for reducing the burden on patients.

Observation of vocal fold and pharyngeal paralysis after carotid endarterectomy using a magnifying laryngoscope.

BACKGROUND: Injury to the vagus nerve or one of its branches during carotid endarterectomy (CEA) can result in vocal fold paralysis (VFP), but the exact mechanisms and site of injury responsible for VFP after CEA are unclear. The aim of this study was to identify the site of nerve injury in patients with VFP after CEA using magnifying laryngoscopy. METHODS: We performed 96 consecutive CEA procedures in 87 patients over 5 years. After 56 CEA procedures, we examined vocal fold movements with a flexible nasolaryngoscope and detected VFP in 5 of 40 cases (9 %). At 6-8 weeks after CEA, these five patients also underwent magnifying laryngoscopy at another institution by a specialist in vocalization. RESULTS: We confirmed ipsilateral VFP and pharyngeal paralysis in three patients. The other two patients recovered from their nerve injuries spontaneously before the magnifying examination. CONCLUSIONS: VFP and pharyngeal paralysis were caused by damage to the recurrent laryngeal and pharyngeal nerves. Therefore, the probable site of nerve injury during CEA was near the inferior vagal ganglion of the vagus nerve trunk in our three patients.

Vernet's syndrome after carotid endarterectomy.

Unilateral paresis of cranial nerves IX to XI is defined as Vernet's syndrome. We retrospectively assessed cranial nerve symptoms from the clinical records of 143 carotid endarterectomy patients. A flexible nasolaryngoscope was used to examine vocal fold movements in 73 patients. If vocal fold paresis (VFP) was confirmed, the patient also underwent magnifying laryngoscopy (for correct diagnosis of injury to the glossopharyngeal and vagus nerves). It was found from clinical records that 8 patients (6%) were confirmed to have cranial nerve symptoms corresponding to Vernet's syndrome; 7 patients (9 %) had VFP on nasolaryngoscopy. In 2 patients, magnifying laryngoscopy confirmed ipsilateral VFP, pharyngeal paresis, pharyngeal wall hypesthesia, and ipsilateral pharyngeal wall swelling. These 2 patients also had symptoms of injury to the accessory nerve. Damage to cranial nerves IX to XI probably occurred in the parapharyngeal space, based on the existence of posterior pharyngeal wall edema or swelling after carotid endarterectomy.

Surgical improvement of speech disorder caused by amyotrophic lateral sclerosis.

Amyotrophic lateral sclerosis (ALS) is a progressive debilitating neurological disease. ALS disturbs the quality of life by affecting speech, swallowing and free mobility of the arms without affecting intellectual function. It is therefore of significance to improve intelligibility and quality of speech sounds, especially for ALS patients with slowly progressive courses. Currently, however, there is no effective or established approach to improve speech disorder caused by ALS. We investigated a surgical procedure to improve speech disorder for some patients with neuromuscular diseases with velopharyngeal closure incompetence. In this study, we performed the surgical procedure for two patients suffering from severe speech disorder caused by slowly progressing ALS. The patients suffered from speech disorder with hypernasality and imprecise and weak articulation during a 6-year course (patient 1) and a 3-year course (patient 2) of slowly progressing ALS. We narrowed bilateral lateral palatopharyngeal wall at velopharyngeal port, and performed this surgery under general anesthesia without muscle relaxant for the two patients. Postoperatively, intelligibility and quality of their speech sounds were greatly improved within one month without any speech therapy. The patients were also able to generate longer speech phrases after the surgery. Importantly, there was no serious complication during or after the surgery. In summary, we performed bilateral narrowing of lateral palatopharyngeal wall as a speech surgery for two patients suffering from severe speech disorder associated with ALS. With this technique, improved intelligibility and quality of speech can be maintained for longer duration for the patients with slowly progressing ALS.

Fiber arrangements of the vertical lingual muscle in human adult subjects

This study identified the anatomy of the vertical lingual muscle and functional relationships between the vertical lingual and the other lingual muscles in the human tongue. Three whole tongues were obtained from adult human cadavers and were used for histological study by the serial section method. At the tip of the tongue, the fibers of the vertical lingual muscle cross with the transverse lingual muscle, and extend inferiorly to the fibers of the inferior longitudinal lingual muscle. At the body of the tongue, the fibers of the vertical lingual muscle are located between the fibers of the superior longitudinal lingual and inferior longitudinal lingual muscle, crossing the fibers of the transverse lingual muscle, instead of crossing the fibers of the extrinsic lingual muscles. At the base of the tongue, the fibers of the vertical lingual muscle start by the fibers of the superior longitudinal lingual muscle, and connect with the fibers of the posterior muscle bundle of the styloglossus muscle. The average diameters of the vertical lingual muscle fibers increased gradually as they approached the base of the tongue. These findings suggest that posterosuperior movement of the tongue body may be accomplished with downward movement of the tip of the tongue by contractions of both the vertical lingual and the styloglossus muscle. The inferior longitudinal lingual muscle may also play a supporting role for the vertical lingual muscle at the tip of the tongue (AU)

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An 8-year-old girl with a recurrence of obstructive sleep apnea syndrome caused by hypertrophy of tubal tonsils 4 years after adenotonsillectomy.

Obstructive sleep apnea syndrome affects 1% to 2% of children. It is caused mainly by upper airway obstruction and manifests as snoring and sleep disturbance. Adenotonsillectomy can improve quality of life because airway obstruction occurs when both tonsils and adenoids are enlarged. We describe an 8-year-old girl with a recurrence of obstructive sleep apnea syndrome caused by hypertrophy of the tubal tonsils 4 years after adenotonsillectomy. The findings from this case highlight the importance of 1) identifying hypertrophy of the residual adenoid and compensatory hypertrophy of the tubal tonsils in patients with obstructive sleep apnea syndrome after adenotonsillectomy and 2) determining the optimal timing of adenotonsillectomy with respect to both the severity of obstructive sleep apnea and compensatory hypertrophy of other lymphoid tissue of Waldeyer's ring, as growth of such tissues is most active during the first several years of life.

[Dysphagia and dysarthria surgery for advanced-age myasthenia gravis].

Myasthenia gravis is often difficult to diagnose and treat in older subjects due to complications, previous history and reciprocal interaction with drugs used to treat complications. An 84-year-old woman with slowly progressive 2-year dysphagia and dysarthria had reached critical condition with aspiration pneumonia. She was diagnosed with thymoma-free myasthenia gravis and her respirator removed after being administrated an anticholinesterase drug. Her dysphagia and dysarthria did not improve. Because of severe osteoporosis with two previous lumbar compression fractures and excessive thinness, she could not be given prednisolone or immunosuppressive drugs. Following cricopharyngeal myotomy and bilateral lateral palatopharyngeal wall narrowing, she could eat without misdeglutition and speak clearly.

A pathological study of bamboo nodule of the vocal fold.

We examined pathologically a bamboo nodule of the vocal fold by means of immunohistochemical studies and scanning electron microscopic examination. A 38-year-old female showed a high index of antinuclear antibodies without any systemic symptoms but had complained of progressive voice disorder for 9 months. She had used her voice excessively in her occupation and for singing. Hematoxylin and eosin staining showed submucosal edema with lymphocyte and neutrocyte infiltrations and hyaline degeneration. Periodic acid methenamine silver staining showed hypertrophy of the basal lamina of the blood vessels. Immunohistochemical study showed IgG-positive cells in the blood vessel walls. Scanning electron microscopic study demonstrated immune complexes deposited as fine granules of high electrodense materials in the hypertrophic walls of the micro-blood vessels. After surgical resection of the bamboo nodules and advising her to avoid using her voice excessively, her voice improved gradually and the levels of IgG, immune complexes, and antinuclear antibody decreased for 1 year. These findings suggest that the bamboo nodules were not induced by an organ-specific reaction to an autoimmune disease, but mechanical damage to the micro-blood vessels induced by phonation injury of the vocal fold might have caused the deposit of high-molecular weight immune complexes in the damaged micro-blood vessels at the midportion of the vocal folds, which induced secondary inflammatory change at the midportion of the vocal fold.

A case of Creutzfeldt-Jacob disease with bilateral vocal fold abductor paralysis.

Bilateral vocal fold abductor paralysis was seen in a patient with Creutzfeldt-Jacob disease. After tracheotomy, the patient showed disappearance of reduced oxygen saturation with high-pitched inspiratory stridor and pulling phenomenon of the supraclavicular region and larynx. Electromyographic examinations of the intrinsic laryngeal muscles, including the thyroarytenoid and posterior cricoarytenoid muscles, demonstrated that there was no apparent action potential in those muscles during spontaneous respiratory movements, and there was no abnormal potential for those muscles at rest. By pushing the infrasternal region of the patient on the expiration, normal motor unit action potential could be seen in the posterior cricoarytenoid muscle on the next inspiration. Based on those findings, we concluded that bilateral vocal fold abductor paralysis in this case of Creutzfeldt-Jacob disease was not induced by disorders of the degeneration of motor nucleus in the ambiguus as in multiple system atrophy, but by a disorder of the upper motor neuron.

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.

[Progressive dysphagia with peculiar laryngeal movement induced by tardive dystonia].

Recently, patients receiving the long-term administration of typical antipsychotics have been recognized to be at risk of developing intractable tardive dystonia. A 44-year-old man was referred to our hospital because of progressive dysphagia for about 5 years. He had received several typical antipsychotic medications since at age of 24 years for the treatment of chronic schizophrenia. The patient had been suffering from an abnormal sensation in his throat and progressive dysphagia for five years, and nasal escape on deglutition for one year. A videotape recorder esophago pharyngography revealed that his larynx was positioned low, at the level of the 6th cervical vertebra, before swallowing, and was not elevated but rather descended to the level of the 7th vertebra upon swallowing. When the larynx was in this lower position, a small amount of swallowed material was transported to the esophagus. The remaining material in the pyriform sinus overflowed into the laryngeal cavity and lower airway after swallowing. However, the patient was able to eat with ease when he was with a girl friend, eating in a restaurant, and was hungry. The peculiar downward movement of the larynx was not observed during speech production, only during deglutition. Based on these findings, we suspected that his peculiar swallowing disorder might have been induced by tardive dystonia arising from the long-term administration of typical antipsychotics.

Nerve fiber analysis for the lingual nerve of the human adult subjects.

The neuro-motor control of the human tongue musculature had not been investigated in detail. This study identified first that the lingual nerve should play the neuro-motor control of some lingual muscles. Six en bloc samples (12 sides), including the tissues from the skull base to the hyoid bone, and three whole tongues were obtained from adult human cadavers. The former samples were used for the study of nerve fiber analysis of the lingual nerve with the aid of binocular stereomicroscope, and the latter samples were used for histological study by serial section method. On nerve fiber analysis of the lingual nerve from the trigeminal ganglion to the tongue musculature, we found that the motor- root of the trigeminal nerve gave off its supply to the lingual nerve and traveled into the lingual nerve, and branched to the superior and the inferior longitudinal muscles. On histological study, it was revealed that in the anterior part of the tongue the superior and the inferior longitudinal muscles surrounded the other lingual musculature and combined with the sub-mucosal connective tissues closely like the cutaneous muscle, for example, the facial muscles. The lingual nerve entered the inner side of the space between the genioglossus and the inferior longitudinal muscles with the lingual artery. These findings suggested that the superior and the inferior longitudinal muscles should be innervated by the motor fibers traveled into the lingual nerve from the motor root of the trigeminal nerve, and do not originate from the myotome originating in occipital somites but branchial muscles.

M-mode color Doppler ultrasonic imaging of vertical tongue movement during articulatory movement.

To observe and estimate the movement of the tongue, ultrasonic investigation is the most harmless real-time monitoring procedure for analyzing articulatory movements. Color Doppler ultrasonic imaging is special in that it can only sample a moving target, and it can indicate the velocity and direction of the target by color and brightness in real time. This study assessed and demonstrated the validity of M-mode color Doppler ultrasonic imaging to observe the movements of the tongue during syllable repetition tasks performed by normal subjects and dysarthric patients, those affected by amyotrophic lateral sclerosis, cerebellar ataxia, Parkinsonism, and polymyopathy. When the transducer was set below the jaw, upward movement was indicated by a blue signal and downward movement was indicated by a red one on the screen of the ultrasound machine. We also measured the velocity of the tongue by contrast scale classified by 15 degrees. Thus, we could observe vertical tongue movement by a color-coded pattern after quantitative analysis. The Doppler signal patterns of normal subjects were verified by simultaneous video x-ray fluorography recordings. The findings for dysarthric patients corresponded well with previously reported features analyzed by other methods. Therefore, color Doppler ultrasonic imaging of the tongue is a useful procedure to researchers for clinical speech and voice studies.

Physiological studies of retrusive movements of the human tongue.

SUMMARY: This study identified that physiologically the superior pharyngeal constrictor muscle at the level of the base of the tongue contributes to retrusive movement of the tongue with constriction of the mid-pharyngeal cavity and possesses unique properties in terms of motor speech control along with the genioglossus muscle. From a kinematic study involving trans-nasal fiberscopy and lateral X-ray fluorography, retrusive movement of the tongue was highly correlated with constrictive movement of the mid-pharyngeal cavity. An electromyographic study revealed that the superior pharyngeal constrictor muscle at the level of the base of the tongue contributes to retrusive movement of the tongue and that the genioglossus muscle contributes to protrusive movement. We also noted that this relationship between the activities of these two muscles were in response to postural changes during vowel productions without changes in the acoustic features. These findings suggest that these two muscles act not only antagonistically to produce retrusive and protrusive movement of the tongue, but also they complement each other to conserve the shape of the vocal tract for speech production. The functional relationship between these two muscles could contribute the consecutive movement of human speech production under various conditions and might be useful when applying rehabilitation approaches for the patients with neurological speech and swallowing disorders.