An uncommon etiological factor for aspiration pneumonitis caused by spontaneous sphenoid sinus meningoencephalocele with cerebrospinal fluid rhinorrhea: a case report.

BACKGROUND: Aspiration pneumonitis is an inflammatory disease of the lungs which is difficult to diagnose accurately. Large-volume aspiration of oropharyngeal or gastric contents is essential for the development of aspiration pneumonitis. The role of cerebrospinal fluid (CSF) rhinorrhea is often underestimated as a rare etiological factor for aspiration in the diagnosis process of aspiration pneumonitis. CASE PRESENTATION: We present a case of a patient with 4 weeks of right-sided watery rhinorrhea accompanied by intermittent postnasal drip and dry cough as the main symptoms. Combined with clinical symptoms, imaging examination of the sinuses, and laboratory examination of nasal secretions, she was initially diagnosed as spontaneous sphenoid sinus meningoencephalocele with CSF rhinorrhea, and intraoperative endoscopic findings and postoperative pathology also confirmed this diagnosis. Her chest computed tomography showed multiple flocculent ground glass density shadows in both lungs on admission. The patient underwent endoscopic resection of meningoencephalocele and repair of skull base defect after she was ruled out of viral pneumonitis. Symptoms of rhinorrhea and dry cough disappeared, and pneumonitis was improved 1 week after surgery and cured 2 months after surgery. Persistent CSF rhinorrhea caused by spontaneous sphenoid sinus meningoencephalocele was eventually found to be a major etiology for aspiration pneumonitis although the absence of typical symptoms and well-defined risk factors for aspiration, such as dysphagia, impaired cough reflex and reflux diseases. CONCLUSIONS: We report a rare case of aspiration pneumonitis caused by spontaneous sphenoid sinus meningoencephalocele with CSF rhinorrhea, which can bring more attention and understanding to the uncommon etiology for aspiration, so as to make more accurate diagnosis of the disease and early surgical treatment.

Total laryngectomy for management of chronic aspiration pneumonia in a myopathic dog.

A 5-month-old female pit bull terrier dog evaluated for ataxia, progressive regurgitation, and recurrent aspiration pneumonia had markedly elevated creatine kinase activity, non-inflammatory generalized myopathy, and severe esophageal dysmotility. A narrow-field total laryngectomy was performed. The dog is doing well 30 months after surgery, and no longer has episodes of aspiration pneumonia, despite intermittent regurgitation. This case represents the first application of total laryngectomy for the prevention of chronic recurrent aspiration pneumonia in the dog.

Laryngectomie totale pour la gestion d'une pneumonie par aspiration chronique chez un chien myopathique. Une chienne Pit Bull Terrier âgée de 5 mois évaluée pour de l'ataxie, de la régurgitation progressive et une pneumonie par aspiration récurrente présentait une activité de la créatine kinase particulièrement élevée, une myopathie généralisée non inflammatoire et un trouble de motilité de l'œsophage grave. Une laryngectomie totale à champ étroit a été réalisée. La chienne se porte bien 30 mois après la chirurgie et n'a plus d'épisodes de pneumonie par aspiration, malgré une régurgitation intermittente. Ce cas représente la première application d'une laryngectomie totale pour la prévention d'une pneumonie par aspiration chronique récurrente chez un chien.(Traduit par Isabelle Vallières).

[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.

Anterior mediastinal tracheostomy with a median mandibular splitting approach in a Larsen syndrome patient with posterior cervical arthrodesis.

Larsen syndrome is a rare congenital connective tissue disorder characterized by multiple joint dislocations. A novel anterior mediastinal tracheostomy with a median mandibular splitting approach is presented for the treatment of airway obstruction in a Larsen syndrome patient with posterior cervical arthrodesis.

Tracheoesophageal Diversion and Laryngotracheal Separation Procedures for Radiotherapy-Related Intractable Aspiration Pneumonia in Nasopharyngeal Carcinoma.

Background: Intractable aspiration and aspiration pneumonia are complications after radiotherapy for nasopharyngeal carcinoma (NPC), and they may be life-threatening in severe cases. In the past, the efficacy of controlling aspiration and aspiration pneumonia in such patients was not ideal. Objectives: We aimed to evaluate the effect of tracheoesophageal diversion and laryngotracheal separation (TED-LTS) procedures for these patients. Material and Methods. We retrospectively analyzed the medical data of five patients with intractable aspiration and recurrent aspiration pneumonia caused by NPC radiotherapy who underwent TED-LTS surgery. The patients were evaluated in terms of aspiration pneumonia control, body weight improvement, removal of tube feeding, oral feeding, and complications. Results: Intractable aspiration and aspiration pneumonia were completely controlled in all cases, and the patients' body weight increased from 46.46 ± 4.6 (38.9-50.3) kg to 55.32 ± 2.7 (51.4-56.7) kg. Four patients were able to consume an oral semisolid diet, and one patient maintained an oral liquid diet. Tube feeding was not required in 4 patients. One patient developed postoperative esophageal fistula, which improved after conservative treatment. Conclusion: TED-LTS is effective for intractable aspiration and aspiration pneumonia caused by NPC radiotherapy and can be used to restore partial oral feeding. However, strict surgical indications should be followed.

A Giant Esophageal Epiphrenic Diverticulum Causing Dysphagia, Fungal Infection and Aspiration Pneumonia in an Elderly Patient.

Esophageal epiphrenic diverticulum is a rare condition. We present a case of a 70-year male with no known comorbidities who presented with dysphagia, cough, fever, and weight loss for 3 months. CT chest with contrast showed an out-pouching arising from the distal dorsal oesophagus, 15.6 cm long with 0.9 cm neck, containing food particles causing compression and consolidation of adjacent lung segments. His upper gastrointestinal endoscopy showed a large diverticulum arising at 30 cm from incisors with overlying ulcerated and necrotic mucosa. A biopsy of diverticular mucosa showed fungal hyphae and spores and was negative for malignancy. The patient was given antifungals and showed significant improvement of symptoms but considering the huge size of the diverticulum, he was referred to a thoracic surgeon. Key Words: Epiphrenic diverticulum, Dysphagia, Aspiration pneumonia, Fungal infection.

Two cases of glottic closure for refractory aspiration pneumonia after vertical partial laryngectomy.

Vertical partial laryngectomy is a well-established surgical procedure for early glottic cancers with acceptable functional and oncological outcomes. However, on a long-term basis, aspiration might be a serious problem with aging. Here we presented two cases of refractory aspiration pneumonia after vertical laryngectomy. Case 1: A 76-year old gentleman with a past history of malignant lymphoma treated by chemotherapy and radiotherapy had glottic cancer, which was treated by repeated vertical partial laryngectomies. Although glottic caner had been well controlled, he started to suffer from refractory aspiration pneumonia. Since his cervical skin was very thin and hard and his general condition was poor, we employed modified Kano's method for glottic closure. Case 2: A 87-year old Japanese male had a past history of glottic cancer treated by radiotherapy and vertical partial laryngectomy. He was repeatedly hospitalized for severe aspiration pneumonia. At the age of 87, he had second primary oropharyngeal cancer. Kano's method was simultaneously performed at the time of resection of oropharyngeal cancer. Postoperative courses were uneventful without sign of leakage in both cases. The patients started oral intake 2 weeks after the surgery. They have been alive without aspiration pneumonia and takes normal diet.

Chronic Aspiration Pneumonitis Caused by Spontaneous Cerebrospinal Fluid Fistulae of the Skull Base.

OBJECTIVES/HYPOTHESIS: Spontaneous cerebrospinal fluid (CSF) leaks of the skull base are associated with obesity, multiparity, and elevated intracranial pressure. Although spontaneous CSF leaks often present with rhinorrhea, they can be an underdiagnosed cause of chronic aspiration pneumonitis, a complication that has not been previously reported in detail. STUDY DESIGN: Retrospective case series. METHODS: The authors retrospectively reviewed all patients undergoing surgical repair of CSF fistulae at the University of Southern California between 2011 and 2018 to identify those presenting with pulmonary symptoms including dyspnea, aspiration, chronic cough, and shortness of breath caused by chronic noniatrogenic CSF pneumonitis. RESULTS: Symptomatic chronic pneumonitis was evident in six of 20 patients with spontaneous CSF rhinorrhea. Five women (mean body mass index = 36) had CSF leaks arising from the fovea ethmoidalis (n = 4) and lateral sphenoid region (n = 1). One man had a middle fossa floor dehiscence draining through the eustachian tube. All patients had bilateral ground-glass opacities in their lungs on computed tomography imaging that were attributed to spontaneous CSF fistulae arising from noniatrogenic skull base defects, and one patient underwent a biopsy of a lung lesion at another hospital showing chronic bronchiolitis and adjacent peribronchiolar metaplasia. Five patients underwent endoscopic endonasal repair using an autologous fascial graft and pedicled nasoseptal flap, and one underwent craniotomy for repair. All patients underwent successful repair with no complications or evidence of recurrence. Upon repair of the spontaneous CSF leaks, both pneumonitis symptoms and ground-glass opacities on imaging resolved in all six cases. CONCLUSIONS: Skull base CSF fistulae should be considered as a reversible cause of chronic pneumonitis that is not alleviated or worsens with standard treatment. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:462-466, 2021.

Drainage of the tracheal blind pouch created by laryngotracheal separation.

Laryngotracheal separation is a simple and reliable operation for the treatment of patients with repetitive and intractable aspiration; however, it is apprehended that pooling in the tracheal blind pouch may cause postoperative complications. In the present study, we examined drainage of the blind pouch created by laryngotracheal separation. Fourteen patients aged 3-63 years with repetitive aspiration pneumonia underwent laryngotracheal separation by the modified Lindeman procedure. A barium swallow was performed 10-30 days after surgery. X-rays of the lateral view of the neck were taken at 6 and 24 h after the swallow, and then every 24 h until the contrast medium cleared. The contrast medium in the blind pouch cleared within 24 h in nine patients. In the remaining five, the clearance time was < or =48 and < or =72 h in two patients each, and 96 h in one patient. The clearance time in patients aged under 20 years was < or =24 h, while middle-aged to elderly patients showed prolonged clearance time. No late complications of the blind pouch, such as infections, were observed. The potential risk of complications caused by pooling in the tracheal blind pouch in laryngotracheal separation is prevented presumably due to the slow but continuous turnover of pooling material. This result supports the validity and usefulness of laryngotracheal separation for the treatment of intractable aspiration.

[Laryngotracheal separation as treatment for severe bronchopulmonary aspiration]. / Separación laringotraqueal como tratamiento de la aspiración broncopulmonar grave.

We present the case of a patient with brain stem tumour and severe chronic aspiration. The bilateral dysfunction of lower cranial nerves and the severe gastroesophageal reflux contributed to the aspirations. Despite medical treatment and cuffed tracheotomy tube, she required almost constant hospitalization for a year and a half due to respiratory infections. Laryngotracheal separation dramatically reduced the infections and improved her quality of life.

[Consecutive aspiration]. / Konsekutive Aspiration.

We report on a 40 year-old man who presented with recurrent pneumonia with limited effect of antibiotics. A chest CT-scan revealed a tumor in the right hilar region leading to a partial atelectasis of the middle lobe. We removed a rather uncommon foreign body from the right main stem bronchus.

[Bronchoscopic findings in children with prolonged respiratory symptoms]. / Indication de la bronchoscopie chez des enfants avec une symptomatologie respiratoire trainante traitement.

OBJECTIVE: This retrospective study aims to establish an algorithm indicating a bronchoscopy, based on clinical and radiological criteria predictive of the presence of a foreign body (FB), in children with prolonged respiratory symptoms and no history of foreign body aspiration (FBA); to establish a study of these criteria and to compare the clinical, radiological and broncoscopic findings in such cases. METHODS: We chose to review the records of 73 children (age < or = 15 years) with prolonged respiratory symptoms (for at least 15 days) and no history of FBA who underwent bronchoscopy at our institution between 1996 and 2005. RESULTS: The mean age was 3 years and 2 months, the majority of the patients were between 1 and 3 years of age (56%), 59% of the patients were boys and the mean of evolution of symptoms before the broncoscopy was 3 months and 3 weeks. We found a foreign body (FB) in 17 cases (23.2%). The FBs were in 88.2% of the cases of vegetable origin. A granulation tissue was associated in 47% of positive bronchoscopies. There were no complications related to bronchoscopy. The clinical and the radiological findings were no specific of FBs. CONCLUSION: Bronchoscopy must be indicated in suspected cases of FBA to prevent delayed diagnosis and pulmonary sequelae. The clinical and radiological findings may help the clinician to indicate it.

Laryngotracheal separation in pediatric patients: 13-year experience in a reference service.

OBJECTIVE: To evaluate clinical stability of neurologically impaired children and adolescents with recurrent pneumonia submitted to laryngotracheal separation. METHODS: Between October 2002 and June 2015, 92 neurologically impaired children from a reference service, with median age of 68.5 months were submitted to laryngotracheal separation. Data were evaluated and statistical analysis was made by Student's t test and Pearson's χ2 test (significance level adopted of 95%). RESULTS: Fifty-three children were male (57.6%). Forty-six children required admission to intensive care, and 42.4% needed mechanical ventilation. We observed that 90.2% of patients were exclusively fed by gastrostomy and 72.4% of the gastrostomies were performed before the tracheal surgery. Thirteen (14.1%) children had postoperative complications as follows: fistulae (5.4%), bleeding (4.3%), granuloma (2.2%) and stenosis (3.2%). A total of 24 patients had pneumonia in the postoperative period (26.1%), but there was a significant drop in occurrence of this condition after surgery (100% versus 26.1%; p<0.001). Twenty-three patients (25%) died. Postoperative complications were similar when comparing patients who died and those that presented good outcome (16.7% versus 13.2%; p=0.73). CONCLUSION: When well-indicated, the laryngotracheal separation reduces the incidence of postoperative pulmonary infections, thus improving quality of life and reducing admissions to hospital. Laryngotracheal separation should be indicated as a primary procedure in patients with cerebral palsy and recurrent aspiration pneumonia.

Diagnosis and Management of Laryngotracheoesophageal Cleft.

Laryngotracheoesophageal cleft (LTEC) is a rare anatomic cause of aspiration in the newborn and infant. The clinical presentation of infants with LTEC is variable and will usually vary with the degree of anatomic communication between the airway and esophagus. The diagnosis of LTEC is often challenging, particularly in cases where the presentation and pathology are more subtle. Management of LTEC depends upon the clinical presentation, degree of aspiration, and anatomic depth of the cleft. The goal of this review is to present the clinician with a practical approach to diagnosis and management of this rare but challenging congenital airway anomaly.

Pulmonary disease due to aspiration of food and other particulate matter: a clinicopathologic study of 59 cases diagnosed on biopsy or resection specimens.

Aspiration of particulate matter is a well-recognized complication in debilitated patients at autopsy but is not widely recognized in surgical pathology material. We have encountered a surprising number of cases on biopsy or resection specimens, and most were unsuspected clinically and pathologically. This study was undertaken to clarify clinical and pathologic features that facilitate the diagnosis of food/particulate matter aspiration pneumonia. Fifty-nine patients were identified with an average age of 57 (range 26 to 85), and a male/female ratio of 2:1. Common presenting symptoms (information available in 36 cases) included dyspnea (14), fever (9), and cough (6). A history of recurrent pneumonia was present in 9. Radiographic data were available in 34 cases. Bilateral infiltrates or nodules were found in 17 cases, whereas the changes were unilateral in 17. Solitary nodules clinically suspicious for neoplasm were present in 13. Aspiration was suspected clinically in only 4 of the 45 cases in which the clinical impression or differential diagnosis was stated. Predisposing factors for aspiration were identified in 32 patients, including esophageal or gastric causes (19), drug use (10), and neurologic conditions (6). Histologically, bronchiolitis obliterans-organizing pneumonia was present in 52 (88%) cases, usually in combination with multinucleated giant cells, acute bronchopneumonia/bronchiolitis, and/or suppurative granulomas. Foreign material was identified in all cases, including most commonly vegetable or food remnants (54 cases), and less often talc or microcrystalline cellulose (7), crospovidone (4), and kayexalate (2). Particulate matter aspiration pneumonia is a more common cause of lung infiltrates and nodules than generally appreciated. The diagnosis should be suspected when multinucleated giant cells, acute bronchopneumonia/bronchiolitis, and/or suppurative granulomas are found in a background of bronchiolitis obliterans-organizing pneumonia. The presence of foreign material confirms the diagnosis.

Histologic findings and comparison with sputum smears in pulmonary aspiration of vegetable particles in syringomyelia: a case report.

BACKGROUND: Pulmonary aspiration of vegetable matter may be a serious cause of lung damage, requiring, almost always, the help of a histopathologic examination. CASE: We report the case of a 55-year-old woman with a granulomatous pneumonitis due to repeated pulmonary aspiration of vegetable particles and affected by a syringomyelia. Histologic findings from a lobectomy section are shown and compared with those observed in different cytologic sputum samples. CONCLUSION: The recognition of uncommon morphologic structures, and their comparison with others already known, both in histologic sections and in cellular smears, may be useful when it comes to establishing a sure diagnosis.

[Analysis of 11 patients with profound multiple disabilities who underwent laryngotracheal separation].

We examined the clinical usefulness of laryngotracheal separation surgery for patients with profound multiple disabilities (PMD). The subjects were 11 severely retarded children who experienced repeated incidents of aspiration pneumonia or who were enough to have aspiration pneumonia easily. A retrospective investigation of their medical records was performed regarding pre- and post-operative data, including the number of times sputum suctionings were required, the number of times pneumonia developed, respiratory conditions, and nutrition methods. The guardians were interviewed regarding musle tone, spasm, sleep quality, internal medications, and changes in mood. After surgery, improvement was confirmed in the number of times sputum suctionings were required, the incidence of pneumonia and respiratory conditions, and oral intake of food in three children. The guardians were aware of improvements in their children's sleep quality and mood. Laryngotracheal separation surgery can reduce the burden of health care for patients with PMD by improving their respiratory conditions and methods of nutrition intake.

Vagotomy decreases the neuronal activities of medulla oblongata and alleviates neurogenic inflammation of airways induced by repeated intra-esophageal instillation of HCl in guinea pigs.

Neuronal activity in the medulla oblongata and neurogenic inflammation of airways were investigated in a guinea pig model induced by repeated intra-esophageal instillation of hydrochloric acid (HCl) after vagotomy. Unilateral vagotomy was performed in the vagotomy group, while a sham-operation was performed in the sham group. Operation was not conducted in sham control group. Airway inflammation was observed with hematoxylin and eosin (HE) staining. C-fos protein was measured by immunohistochemistry (IHC) and Western blot (WB). Substance P was examined by IHC and enzyme-linked immuno sorbent assay (ELISA). Airway microvascular permeability was detected by evans blue dye (EBD) fluorescence. Inflammation of airway was observed in the trachea and bronchi after chronic HCl perfusion into the lower esophagus, and was alleviated after unilateral vagotomy. C-fos expression in the medulla oblongata was lower in the vagotomy group compared to the sham control and sham groups. Substance P-like immunoreactivity (SP-li), concentration and microvascular leakage in airway were lower in the vagotomy group than that in the other groups. Our results suggest that vagotomy improved neurogenic inflammation of airways and decreased neuronal activities, the afferent nerves and neurons in medulla oblongata may be involved in neurogenic inflammation of airways mediated by esophageal-bronchial reflex.

Intractable pulmonary aspiration in children: which operation?

Intractable aspiration is a life-threatening medical problem. There are many surgical procedures for the treatment of intractable aspiration. Our experience with three children was reported here. After failing initial conservative measures, the first two patients had supraglottic laryngeal closure performed through an endoscopic approach with a small hole left superiorly for phonation. However, both of them developed dehiscence of the closure postoperatively, necessitating a more definitive procedure to stop the aspiration. Their symptoms of aspiration were only relieved after they underwent laryngotracheal separation. In the third child, laryngotracheal separation was performed as the initial procedure and the symptoms of recurrent aspiration were relieved. In conclusion, the treatment of intractable aspiration requires a definitive procedure from the outset and we would suggest, based on literature review and on our experience, that laryngotracheal separation is the most effective and reliable option, albeit lack of phonation is inevitable. A comprehensive review of all procedures used for treatment of this condition was also undertaken.