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Complication of benign tracheobronchial strictures by self-expanding metal stents.

OBJECTIVES: Self-expanding metal stents are used to palliate benign strictures. We examined the complications of this approach. METHODS: Between 1997 and 2002, we observed recurrent airway obstruction and extension of benign inflammatory strictures after the placement of tracheobronchial Microvasive Ultraflex stents and Wallstents (Boston Scientific Corp, Natick, Mass), in 10 patients with postintubation strictures and 5 with other indications; all but 1 patient were referred to us. Patients with tracheal (9), subglottic (1), combined tracheal and subglottic (3), and bronchial (2) strictures had been treated with covered and uncovered Wallstents (6) and Microvasive Ultraflex stents (9). RESULTS: After stent insertion, stricture and granulations within previously normal airway were seen in all patients. New subglottic strictures resulting from the stent caused hoarseness in 4 patients. A bronchoesophageal fistula was found in 1 patient at presentation and a tracheoesophageal fistula in another during extraction of a Wallstent. Primary surgical reconstruction, judged to have been feasible before wire stent insertion in 10 patients, was possible after stenting in only 7 and failed in 2. Palliative tubes were placed in 60% (9/15). Self-expanding metal stents may lengthen luminal damage, incite subglottic strictures, and cause esophagorespiratory fistula in inflammatory airway strictures. The injury is severe, occurs after a short duration of stenting, and precludes definitive surgical treatment or requires more extensive tracheal resection. CONCLUSION: The current generation of self-expanding metal stents should be avoided in benign strictures of trachea and bronchi.

Adult↗

The utility of the pectoralis myocutaneous flap in the management of select cervical esophageal anastomotic complications.

OBJECTIVE: The majority of cervical esophageal anastomotic complications can be successfully managed nonoperatively. A small group of patients may have anastomotic strictures or leakage and fistula formation that are chronic and resistant to nonoperative therapy. The purpose of this study was to review our experience with the use of the pectoralis myocutaneous flap to treat these patients. METHODS: Since April 1992, four patients have undergone pectoralis myocutaneous flap repair of cervical esophageal anastomotic complications at our institution. Two patients had chronic strictures, one patient underwent prophylactic repair with a pectoralis myocutaneous flap to prevent stricture formation, and one patient had a chronic anastomotic fistula. The pectoralis myocutaneous flap was harvested in the standard fashion. The technique of anastomotic repair is described. The medical records were retrospectively reviewed to determine patient characteristics and our results. RESULTS: Two suture line leaks developed: one small, contained leak required no intervention, and the other resolved with cervical drainage. Pneumonia, seroma at the site of the pectoralis myocutaneous flap donor, transient hoarseness, and partial skin graft loss occurred in one case each. There were no deaths. Hospital stay ranged from 12 to 22 days. A good functional result was obtained in three patients. CONCLUSION: Our results show that pectoralis myocutaneous flap repair of select cervical anastomotic complications is safe and well tolerated even in patients with complicated problems.

Adult↗

Results of radiotherapy for chemodectomas.

Between 1974 and 1988, 10 Mayo Clinic patients had unresectable, locally recurrent, or partially resected chemodectomas. Of these 10 tumors, 9 were confirmed pathologically, and 1 was diagnosed clinically. The chemodectoma was located in the jugular bulb in five patients, the middle ear in three, and the carotid body in two. The following symptoms were noted: tinnitus (in eight patients), loss of hearing (in six), hoarseness (in six), dysphagia (in four), pain (in three), and alteration of mental status (in one). Many patients had more than one symptom. Treatment was delivered with megavoltage photons and electrons; total doses ranged from 16.2 to 52 Gy (median, 46 Gy), and the daily doses ranged from 1.6 to 2.4 Gy. Follow-up among the nine survivors ranged from 3 1/2 to 16 years (median, 7 1/2 years). In one patient, the response could not be assessed because the patient died of renal failure 4 months after treatment. All nine assessable patients had decreased symptoms and objective control of the tumor (no evidence of progression of disease). Of the nine assessable patients, four had complete responses, one had a partial response, and four had stable disease. No patient experienced progression of disease after radiotherapy. We conclude that radiotherapy for chemodectomas yields successful results--namely, decreased symptoms and objective control of the tumor.

Adult↗

Tracheopathia osteoplastica: familial occurrence.

Tracheopathia osteoplastica is an unusual disease characterized by cartilaginous or bony projections into the tracheobronchial lumen, with sparing of the posterior membranous portion of the tracheobronchial tree. The cause of this disorder is unknown. The diagnosis is seldom made because of the chronic and asymptomatic nature of the condition. More than 90% of the cases are diagnosed at postmortem examination. Symptoms may include dyspnea, coughing, hemoptysis, hoarseness, and wheezing. Tomography of the trachea may reveal beaded calcification of the tracheobronchial cartilages. Bronchoscopy is diagnostic. Histologically, the abnormal growths show heterotopic bone formation. No known treatment is available. We describe two female patients, one with hemoptysis and another with asthmalike symptoms, in both of whom bronchoscopy established the diagnosis of tracheopathia osteoplastica. The first patient had recurrent episodes of pneumonia attributable to bronchial obstruction by bony projections. In the second patient, removal of large lesions that obstructed the upper part of the trachea relieved the dyspnea. Of interest is the fact that the first patient was the biologic mother of the second. To our knowledge, this constitutes the first report of familial occurrence of tracheopathia osteoplastica.

Aged↗

Rupture of the larynx in a newborn.

BACKGROUND: Lesions of the laryngotrachea due to repeated intubation trauma after birth are reported. CASE: A macrosomic neonate was born at term after uneventful pregnancy. On the basis of diminished uterine contractions and arrest of descent, the head was delivered by using obstetric forceps. No intubation was attempted after birth. Ten hours later, the infant developed acute life-threatening respiratory distress and subcutaneous emphysema. Intubation was impossible. Open surgical exploration of the neck revealed a rupture of the cricothyroid ligament within the larynx. The defect was closed immediately, and after artificial ventilation for 7 days, the infant was extubated. One year later, the child is developing satisfactorily, without stridor or hoarseness. CONCLUSION: In macrosomic neonates after traumatic delivery, a laryngotracheal lesion should be considered when respiratory distress syndrome combined with subcutaneous emphysema occurs.

Birth Injuries↗

Respiratory distress in a pregnant woman with congenital vocal cord paralysis.

BACKGROUND: Vocal cord paralysis is one of the most common congenital laryngeal lesions. Hoarseness and dysphonia are common in unilateral cases. Stridor and respiratory distress are seen in bilateral cases. CASE: A 33-year-old primigravida with bilateral congenital vocal cord paralysis presented in the third trimester with a 2-week history of bronchitis and progressive shortness of breath. She developed stridor, her respiratory status deteriorated, and she was intubated with difficulty. She went into cardiac arrest, was resuscitated and placed on a ventilator, and a tracheotomy was performed. Her condition improved, she remained stable until 38 weeks, and was delivered by cesarean. CONCLUSION: Congenital vocal cord paralysis may result in life-threatening respiratory distress during pregnancy. Tracheotomy placement may be useful in the management of these patients.

Adult↗

Sjögren's disease.

In summary, Sjögren's syndrome is a chronic autoimmune disorder characterized by lymphocytic infiltration of the exocrine glands, particularly the salivary and lacrimal glands. The sicca complex of xerophthalmia and xerostomia are the hallmark features of Sjögren's symptomatology. In addition to these hallmark symptoms of oral and ocular dryness, one third of patients with Sjögren's syndrome present with extraglandular manifestations. Many osf these extraglandular manifestations, as well as the sicca symptoms, have otolaryngologic relevance. Because patients with Sjögren's syndrome may present with vague or nonspecific head and neck complaints, ranging from oral dryness to hoarseness to hearing loss, an otolaryngologist's high index of suspicion for this disorder may prevent delay in diagnosis, allow appropriate diagnostic evaluation, and optimize therapeutic intervention.

Digestive System Diseases↗

Recurrent laryngeal nerve paralysis: anatomy and etiology.

Etiologies of adult vocal paralysis are varied by the site of the lesion as well as the extent and cause of the damage. Most large series point to surgery and neoplastic causes for recurrent nerve paralysis. A detailed history is important when working up a patient with this voice disorder. Knowledge of the anatomy of the head, neck, and chest as well as the mechanisms behind vocal fold paralysis is essential in the evaluation and treatment of recurrent nerve paralysis. Many of the surgical and traumatic causes of hoarseness are from compression type injuries. Recovery is dependent on the type, extent, and site of nerve lesion. Familiarity with this data allows the otolaryngologist to tailor management to suit each patient with vocal fold paralysis.

Cervical Vertebrae↗

Congenital anomalies of the larynx and trachea.

A variety of congenital anomalies arise within the laryngeal or tracheal airway. Symptoms primarily include airway obstruction, hoarseness, and difficulty feeding. The diagnosis is typically made by a combination of clinical presentation, physical examination, and endoscopic evaluation. Definitive intervention may be necessary requiring endoscopic or open laryngeal surgery. Some of the more common congenital laryngeal and tracheal anomalies are discussed with respect to their diagnostic evaluation, clinical presentation, and management.

Glottis↗

Medical and surgical treatment of pediatric dysphonia.

The production of sound for communication is the last of the prime functions of the larynx, yet it is its most salient feature. Pediatric dysphonia may be caused by a variety of general causes. These causes may be classified as infectious, anatomic, congenital, inflammatory, neoplastic, neurologic, and iatrogenic. When a child presents with hoarseness, a complete assessment is necessary. Fine points during the history and physical may help distinguish between those pathologic conditions found in these general categories.

Child↗

Disorders of phonation in children.

Vocal disturbances in children are surprisingly common. Typical pathologic conditions differ, however, from those that are seen commonly in adults. Disturbances in the vibratory characteristic of the vocal folds cause dysphonia. The most common causes for dysphonia are infectious, anatomic, congenital, inflammatory, neoplastic, neurologic, or iatrogenic in nature. A child who presents with hoarseness demands a rapid and thorough assessment. Fine points during the history may provide the clinician with clues as to the correct etiologic category. Proper treatment requires an accurate diagnosis. Although some conditions are similar in the adult larynx, treatment of the pediatric larynx often differs. An understanding of the changing and growing pediatric larynx is necessary for treatment and satisfactory results.

Child↗

Management of vagus nerve injury afer carotid endarterectomy.

BACKGROUND: Inadvertent injury to the vagus nerve or its branches during carotid endarterectomy can result in adductor vocal cord paralysis (hoarseness) and cricopharyngeal dysfunction (dysphagia) with aspiration, known as "double trouble." This study describes our experience in the management of this complication in cases where conservative treatment failed. METHODS: All patients were examined by a vascular surgeon, a head and neck surgeon, and a speech therapist. Their examinations included comprehensive speech evaluation, video stroboscopy, video fluoroscopy, and methylene blue testing for aspiration. All patients underwent Teflon injections to medialize the paralyzed vocal cord and a cricopharyngeal myotomy to restore swallowing and alleviate aspiration. RESULTS: Fourteen patients, eight men and six women, were treated. The duration of dysfunction was 24 weeks in two patients, 6 weeks in four patients, 4 weeks in three patients, and 1 week in five patients. Five patients had severe dysfunction (defined as difficulty in swallowing both solid and liquid foods with more than 20% aspiration), seven patients had moderate dysfunction (defined as difficulty swallowing solid food with aspiration of less than 20%), and two patients had mild dysfunction (defined as difficulty in swallowing solids but with no aspirations). After the Teflon injections and myotomy, 13 of 14 patients had satisfactory outcomes, including normal voice and swallowing. CONCLUSIONS: Vagus nerve injury from a carotid endarterectomy can be a debilitating complication. Prevention, early recognition, and prompt correction of these injuries are important in the management of this complication.

Aged↗

Teflonomas of the larynx and neck.

Intracordal fluorocarbon (Teflon, Mentor O and O Inc, Norwell, MA) injection has been used for decades to correct paralytic dysphonia, a result of unilateral laryngeal paralysis. Infrequently, the Teflon extravasates and infiltrates into the soft tissues of the neck and larynx producing a mass that clinically simulates a malignant lesion. We report eight cases of so-called "Telonomas" of the larynx and neck that have been identified in the files of the Armed Forces Institute of Pathology Otolaryngic Tumor Registry and from the Cytopathology Service at the George Washington University Medical Center. Patients ranged in age from 31 to 72 years. Vocal cord paralysis, treated by Teflon injection, was caused by primary laryngeal carcinoma or metastatic carcinoma (breast, lung) involving the recurrent laryngeal nerve, surgical trauma to the recurrent laryngeal nerve, or postviral neuritis. Subsequent symptomatology, related to extravasation of the Teflon with a resulting "Teflonoma", included a neck mass or persistent hoarseness. Infrequently, there was associated airway obstruction or voice changes. Diagnosis was made by fine needle aspiration or by excision of the suspicious mass and subsequent identification of a foreign body granulomatous reaction with associated birefringent material. Infrared absorption spectrophotometry identified the foreign material as a fluorocarbon which was further substantiated by scanning electron microscopy and energy dispersive x-ray analysis. Surgical removal of the mass alleviated all symptoms.

Adult↗

Jugular bulb diverticulum associated with lower cranial nerve palsy and multiple aneurysms.

BACKGROUND: Jugular bulb diverticulum is a rare condition that has never been reported in association with lower cranial nerve palsy. CASE DESCRIPTION: A 54-year-old woman developed acute swallowing disturbance and hoarseness. Neuroradiological examinations demonstrated a jugular bulb diverticulum as well as unruptured internal carotid and basilar tip aneurysms on the same side. Laboratory data for both serum and cerebrospinal fluid were normal. Steroids were given, and the cranial nerve palsy disappeared within 1 month. The aneurysms were successfully clipped. During the follow-up period of more than 1 year, the patient has done well and has no neurological deficit. CONCLUSION: This is the first reported case of jugular bulb diverticulum associated with a lower cranial nerve palsy and multiple aneurysms. We recommend close observation of patients with this condition regardless of its clinical presentation.

Anti-Inflammatory Agents↗

Primary epithelioid hemangioendothelioma originating in the lower petroclival region: case report.

BACKGROUND: We report the first case of primary epithelioid hemangioendothelioma (EH) originating in the lower petroclival region. CASE DESCRIPTION: A 55-year-old female presented with a 45-year history of subclinical atrophy on the right side of her tongue and a 15-year history of hoarseness. Neuroimaging revealed an expansile, homogeneously enhanced intraosseous mass with bony shell and honeycomb configuration in the petroclival region. A right far lateral transcondylar approach was utilized for subtotal removal of the tumor, which demonstrated high vascularity in the petroclival bone. Histopathological and immunohistochemical examination confirmed the diagnosis of EH. Because of the intermediate malignancy, adjuvant gamma knife radiotherapy was performed for the residual mass 5 months after surgery. CONCLUSION: EH rarely occurs in the skull base region. The appearance of surrounding bony structure and rich vascularity are important findings for the differential diagnosis. A far lateral transcondylar approach provides sufficient exposure of the jugular tubercle and lower clivus. Additional radiotherapy may be recommended for residual tumors with a high MIB-1 labeling index.

Diagnosis, Differential↗

Choroid plexus papilloma and meningioma: coincidental posterior fossa tumors: case report and review of the literature.

OBJECTIVE: We report an unusual case of an extraventricular choroid plexus papilloma (CPP) occupying the right ventral foramen magnum and lower right cerebellopontine angle (CPA), occurring together with a petro-tentorial meningioma. The clinical presentation, preoperative imaging, differential diagnosis, surgical treatment and histologic features of the two tumors are discussed. CASE DESCRIPTION: The patient presented with a 2-month history of headache, altered facial sensation, dysphagia, and gait unsteadiness. Neurologic examination confirmed a wide-based, unsteady gait, hoarse voice, anisocoria, and partial right vocal cord paralysis. Diagnostic imaging demonstrated right petro-tentorial and right ventral foramen magnum lesions, both hyperintense on contrasted images and suggestive of meningiomas. A right suboccipital craniotomy and far lateral approach was used to resect both tumors. The petro-tentorial tumor was a histologically confirmed meningioma, but the ventral foramen magnum tumor was an extraventricular CPP. CONCLUSIONS: This is a rare occurrence of concomitant meningioma and CPP. There is no known link between these two tumors. An exophytic 4th ventricular CPP must be considered in the differential diagnosis of a CPA or foramen magnum tumor.

Choroid Plexus Neoplasms↗

Pontine glioma with osteoblastic skeletal metastases in a child.

BACKGROUND: The development of systemic metastases from primary intracranial gliomas is rare. We report here a rare case of pontine glioma with osteoblastic skeletal metastases. CASE: This 12-year-old boy presented with a 4-month history of hoarseness, dysphagia, and a progressively ataxic gait. Cranial computed tomography (CT) and magnetic resonance imaging (MRI) revealed a brain stem tumor that was diagnosed as a low grade glioma by stereotactic biopsy. Twelve months later following chemotherapy and radiotherapy, neurologic examination and neuroradiologic studies disclosed a recurrence of the pontine glioma. Skeletal roentgenograms revealed widespread osteoblastic metastases in the skull, vertebral bodies, pelvis, and long bones. A specimen from the iliac bone demonstrated cells that were immunoreactive glial fibrillary acidic protein (GFAP). DISCUSSION: The mechanism of how glioma cells determine their biologic behavior at bony metastatic sites is not known. Infratentorial gliomas, which occur frequently in young patients and demonstrate active bony metabolism, may stimulate osteoblastic cells, and induce osteoblastic changes.

Bone Neoplasms↗

Exercise-induced anaphylaxis: a serious form of physical allergy associated with mast cell degranulation.

Exercise-induced anaphylaxis (EIA) is a unique and an increasingly recognized syndrome consisting of premonitory symptoms and signs of generalized body warmth, pruritus, and erythema, which progresses on continued exertion to confluent urticaria, laryngeal edema with stridor or hoarseness, and gastrointestinal colic and frequently culminates in vascular collapse. Previous studies of five individuals with this condition have demonstrated significant elevations of serum histamine concurrent with the early clinical manifestations after experimental exercise. To assess relevant morphologic alterations in the skin of these patients, cutaneous mast cells were examined by light and transmission electron microscopy before and during the initial erythema elicited by exertion. The marked alterations observed in mast cells immediately after exercise consisted of (1) loss of electron density and internal substructure of granules, (2) fusion of granule membranes with those of adjacent granules and with mast cell membranes creating conduits to the extracellular space, and (3) an apparent decrease in the number of intact granules per cell. Biopsy specimens obtained before exercise from patients with EIA and from two normal individuals who served as control subjects were identical, and the control subjects had normal mast cell morphology after exercise. Serum histamine levels were significantly elevated in patients with EIA after exercise at the time of biopsy, whereas control subjects had normal levels. These observations provide evidence that EIA is a distinct form of physical allergy associated with mast cell degranulation similar in morphology to that of human pulmonary mast cell IgE-Fc-dependent activation secretion. Characterization of this disorder is important because its prevalence may be underestimated, and its clinical consequences, which may include some morbidity, are not fully known.

Adult↗