Subglottic Schwannoma: a report of a rare case that was treated with medial thyrotomy.

We present a rare case of primary subglottic schwannoma in a 51-year-old man. He presented with subacute dyspnea and progressively worsening stridor. Videoendoscopy revealed the presence of a submucosal mass in the subglottic area, which measured 2 cm in diameter and had occluded approximately 80% of the lumen. Computed tomography (CT) and magnetic resonance imaging (MRI) showed a tumor arising from the left posterior wall of the subglottis and extending from the level of the cricoid cartilage to the first tracheal ring. After tracheostomy for airway management, subglottic schwannoma was diagnosed via fiberscopy-assisted punch biopsy. We removed the tumor via a medial thyrotomy (laryngofissure), and the post-excisional raw surface of the cricoid and tracheal cartilage was covered with a free buccal mucosal flap, which was attached using absorbable sutures and fibrin glue. No complications including recurrent nerve palsy developed after the procedure, and early postoperative ambulation was successfully performed. The patient is currently well, and the subglottic wound is clear. Although long-term follow-up is required, medial thyrotomy and coverage of the exposed cartilage with a free buccal mucosal flap is more effective for large subglottic schwannomas than laryngeal or tracheal resection with permanent tracheal tunnel formation from the viewpoint of phonation disability and the risk of complications.

Angiotensin II receptor blocker-induced angioedema in the oral floor and epiglottis.

We report the rare case of angioedema (also known as Quincke edema), which was induced by valsartan, an angiotensin II receptor blocker (ARB). ARBs are a new class of antihypertensive agent that is developed to exclude the adverse effects of angiotensin-converting enzyme inhibitors. In theory, ARBs do not contribute to the occurrence of angioedema because they do not increase the serum level of bradykinin, the responsible substance for angioedema. However, some reports of ARB-induced angioedema have recently been published. In this study, we present the forth case and the first Asian case of angioedema due to valsartan, which is one of the ARBs. Otolaryngologist should be wary of the prescribing ARB and discontinue ARBs treatment soon, if angioedema is recognized.

Two Different Tracts and Origin of Pyriform Sinus Fistula.

OBJECTIVE: Suppurative acute thyroiditis is caused by pyriform sinus fistula (PSF), and PSF frequently elicits deep neck abscess. However, complete fistulectomy is the ideal management of PSF, and studies on surgical findings of PSF are exceedingly rare. This study aimed to reveal the origins of PSF, each route, and clinical presentation. METHODS: This is a multicenter study. We have conducted 19 complete fistulectomies of PSF in Japan, analyzed routes of the fistulas, estimated the origins, and investigated their histological and clinical findings. RESULTS: No recurrence was observed in all cases. Five of 12 cases showed thymic and/or parathyroid tissues around the fistulas, passing inside the inferior horn of thyroid cartilage, were regarded as having 3rd pouch origin, and tended to have low frequency of severe deep neck abscess. The remaining 7 cases originated from the 4th pouch running outside of the horn and showed frequent severe infection. CONCLUSION: PSF have 2 different routes depending on their generation and may present different clinical manifestations.

Mucocele in an Onodi cell with simultaneous bilateral visual disturbance.

The Onodi cell is a large pneumatized posterior ethmoid cell and closely related to optic nerve. We present an extremely rare case of retrobulbar optic neuropathy caused by mucocele in an Onodi cell. A 79-year-old man complained of headaches and simultaneous bilateral visual disturbance. A computed tomography (CT) scan demonstrated a mucocele in an Onodi cell, which involved bilateral optic nerves. The surgical treatment with a transnasal endoscopic approach was performed, resulting in the improving of visual acuity. The bilateral optic nerves were identified along each lateral wall into an Onodi cell accompanied with bone defect. In an Onodi cell, even if the lesion is isolated and/or small, it may be closely related to ocular symptoms. Imaging studies should be considered for the differential diagnosis because early diagnosis and prompt surgical treatment for mucocele are needed for recovery of visual impairment.

Congenital arhinia: a case report and functional evaluation.

OBJECTIVES: Congenital arhinia is rare clinical entity. An unusual case of congenital arhinia with no surgical treatment is presented. STUDY DESIGN: Case report. METHODS: A 9-year-old with congenital arhinia was referred for treatment of otitis media with effusion. In addition to his undergoing imaging studies since birth, various functional evaluations regarding the nose were performed. Furthermore, the pathogenesis of arhinia is discussed. RESULTS: The imaging workup revealed the absence of nasal bones, the nasal septum, and turbinates, hypoxia of the maxilla, and a high-arched palate. Furthermore, magnetic resonance imaging clearly demonstrated the absence of olfactory bulbs and tracts, resulting in no response with olfactometry. Among the examinations performed, chromosomal analysis, polysomnography, and an intelligence test produced normal results. In the hearing assessment, he had a slight conductive hearing loss. A phonetic examination revealed the typical pattern of rhinophonia clausa. CONCLUSIONS: This study presents congenital arhinia associated with facial anomalies. Because there was no life-threatening complications and normal psychomotor development, the patient could not only be followed up long-term but could also have his nose functionally evaluated. It is highly likely that this case might have been the result of the failure of medial and lateral nasal processes to grow, which was the underlying pathogenesis.

[Inflammatory pseudotumor arising in the ethmoid sinus: a case report].

Inflammatory pseudotumors are rare and are characterized by solitary, demarcated lesions with numerous inflammatory cells, mesenchymal cells, and collagenous fibers. They usually occur in the lung, but have also been reported in the thyroid gland, pleura, liver, kidney, bile duct, spinal cord, testis, and soft tissues. Since inflammatory pseudotumors of the head and neck are very rare, their clinical and radiological features, prognosis, and therapeutic management have never been clearly described. We report a case of inflammatory pserdotumor arising in the ethmoid sinus and the clinical, radiological, and histopathological findings.

A case of pyriform sinus fistula infection with double tracts.

Pyriform sinus fistula is a rare clinical entity and the precise origin remains controversial. The fistula is discovered among patients with acute suppurative thyroiditis or deep neck infection of the left side of the neck and is usually located in the left pyriform sinus. To the best of our knowledge, only a single tract has been reported to be responsible for pyriform sinus fistula infection. We present a case of a 13-year-old female patient with a pyriform sinus fistula that caused a deep infection of the left side of the neck and showed double-tract involvement discovered during surgical resection of the entire fistula. Both tracts arose around the pyriform sinus and terminated at the upper portion of the left lobe of the thyroid.

Improvement of tracheal flap method for laryngotracheal separation.

OBJECTIVES/HYPOTHESIS: Recurrent pneumonia due to intractable aspiration is a life-threatening disease. A tracheal flap method for children without previous tracheostomy has been previously reported. This study reports that improvements of this method and its three subtypes are widely applicable to patients with various conditions. STUDY DESIGN: Surgical technique study. METHODS: The tracheal flap method does not involve transection of the trachea but achieves laryngotracheal separation using the tracheal, mucoperichondrial, and sternohyoid muscle, along with anterior cervical skin flaps. This method can be divided into three subtypes as follows: A-type, utilizing the tracheal flap (for patients without previous tracheostomy); B-type, utilizing the mucoperichondrial and sternohyoid muscle flaps (for patients lacking an anterior tracheal wall); and C-type, utilizing the esophageal flap (for patients with severe hypersalivation). In all three subtypes, the anterior cervical skin flap is employed. RESULTS: The tracheal flap method was performed in 30 patients (24 children and six adults) at risk of developing intractable aspiration pneumonia. In all 30 cases, aspiration pneumonia was prevented without severe complications. No fistula formation was observed. CONCLUSIONS: All three subtypes (A-, B-, and C-type) of the tracheal flap method are effective in preventing the recurrence of aspiration pneumonia. This method is applicable to diverse patient backgrounds regardless of age or previous tracheostomy. It is less invasive than Lindeman procedure. Furthermore, this method is acceptable to patients' families and improves the QOL of both patients and caregivers.

A new and less invasive procedure for arytenoid adduction surgery: endoscopic-assisted arytenoid adduction surgery.

OBJECTIVES/HYPOTHESIS: Arytenoid adduction (AA) is the most effective procedure for improving voice function in patients affected by unilateral vocal fold paralysis (UVFP), but it is often associated with severe complications following airway obstruction. The aim of this study is to describe a new and less invasive AA surgical procedure termed endoscopic-assisted AA surgery (EAAS) and to evaluate its voice outcomes. STUDY DESIGN: We demonstrated this method using extirpated larynges from three laryngeal cancer patients. Ten patients with severe UVFP underwent EAAS alone or combined with type I thyroplasty or lipoinjection laryngoplasty. METHODS: EAAS involves the placement of permanent adducting sutures around the muscular process (MP) of the arytenoid cartilage using two needles, a penetration needle and a loop needle, under endoscopic guidance. To define the anatomic position of the MP for safer needle insertion, the location of the MP was measured relative to three landmarks on computed tomography/X-ray images of the larynges and in resected larynges. For all patients with UVFP, the maximum phonation time, mean airflow rate, and three acoustic analysis parameters were measured before and after EAAS. RESULTS: The values of the three variables were similar in all cases. Most patients achieved a maximum phonation time of more than 10 seconds and a mean airflow rate of less than 200 mL/second. All three acoustic analysis parameters were significantly improved after surgery. CONCLUSIONS: EAAS is a simple and effective arytenoid rotation procedure.

Congenital laryngeal anomalies presenting as chronic stridor: a retrospective study of 55 patients.

OBJECTIVES: Congenital laryngeal anomalies are less frequent, but their causes are surprisingly variable. In addition, a variety of synchronous airway lesions as well as comorbidities are accompanied. The objective of this study was to review of patients with congenital laryngeal anomalies presenting as chronic stridor in our experiences. METHODS: Fifty-five patients, 30 male (54.5%) and 25 female (45.4%), were enrolled in this study, and their hospital records were retrospectively reviewed. RESULTS: The most frequent diagnosis was laryngomalacia (36.4%), followed by subglottic stenosis (30.9%) and vocal cord paralysis (29.1%). Twenty-six (47.3%) of the 55 patients had synchronous airway lesions, whereas thirty-one (56.4%) had various comorbidities. Further analysis was performed in patients diagnosed with laryngomalacia, subglottic stenosis, or vocal cord paralysis, which are major causes of congenital laryngeal stridor. The frequency of synchronous airway lesions was not different significantly in these three groups. On the other hand, the frequency of establishment of airway in patients with laryngomalacia was significantly lower compared to those with subglottic stenosis or vocal cord paralysis. Moreover, median duration of the symptoms and the proportion of patients with poor outcome and decease in laryngomalacia were shorter and lower than that in subglottic stenosis or vocal cord paralysis. CONCLUSIONS: While a variety of congenital airway anomalies were causes of chronic stridor, laryngomalacia was the most frequent diagnosis. Severe condition and progression of symptoms should increase suspicion of the synchronous airway lesions and/or comorbidities, which may be important factors for outcome as well as indication of surgical intervention.

Simple and new surgical procedure for laryngotracheal separation in pediatrics.

OBJECTIVES: In children with severe physical and mental disabilities who repeatedly develop aspiration pneumonia due to intractable aspiration, laryngotracheal separation/tracheoesophageal anastomosis or laryngotracheal separation has been performed in many institutions for the prevention of aspiration, and good results have been reported. However, families sometimes show a marked reluctance to give consent to these surgical techniques because of tracheal transection. A purpose of this study is to evaluate a new surgical procedure for laryngotracheal separation without tracheal transection. STUDY DESIGN: Case-series study. METHODS: As a new, simple, less invasive surgical technique for the prevention of aspiration without tracheal transection, we performed tracheal closure (tracheal flap method) in six children. A U-shaped flap of the tracheal anterior wall from the 2nd to the 4th/5th tracheal ring was produced, bent toward the tracheal lumen, and sutured to the tracheal posterior/lateral walls by mattress stitches for tracheal closure. In addition, the closure was covered with a cutaneous U-shaped flap for reinforcement and a permanent tracheal stoma was constructed. RESULTS: In all six patients, aspiration pneumonia could be prevented without severe complications. CONCLUSIONS: Tracheal closure (tracheal flap method) has effects comparable to those of other surgical techniques for the prevention of aspiration, and may be useful for aspiration prevention in children with severe physical and mental disabilities.

Successful video-assisted mediastinoscopic drainage of descending necrotizing mediastinitis.

Descending necrotizing mediastinitis is an uncommon form of mediastinitis that can rapidly progress to septacemia. To date, the optimal surgical approach has remained controversial. We report a case of descending necrotizing mediastinitis that was treated successfully through a transcervical approach with video-assisted mediastinoscopy. In our case, because the abscess was separated into small compartments, especially in the paratracheal space, the abscess was drained using video-assisted mediastinoscopy. This less-invasive approach may be an option in the treatment of descending necrotizing mediastinitis, especially when the abscess in the paratracheal space is separated into small compartments.