[TWO CASES OF LARYNGEAL EDEMA CAUSED BY SUBLINGUAL ALLERGEN IMMUNOTHERAPY].

Sublingual immunotherapy is a widely used treatment, and serious adverse reactions such as anaphylaxis are rare. We report two cases of laryngeal edema as adverse reactions to sublingual immunotherapy, which could be continued due to a change in the administration method. Case 1 presents a 15-year-old male suspected to have had anaphylaxis due to the dust at the age of 6 years. He started treatment with Miticure® and developed laryngeal edema 30 minutes after taking the 10000JAU dose on the 10th day. laryngeal edema was treated with intravenous infusion. Case 2 presents a 48-year-old woman. She started treatment with Cidacure® and developed respiratory distress and laryngeal edema 1 hour after taking the 5000JAU dose on the 5th day. she had resolved mildly without therapeutic intervention. In both cases, the patients were switched to sublingual spitting, resumed with the initial dose cautiously, and were able to continue. Sublingual immunotherapy is a safe treatment, but sudden adverse reactions may occur. Laryngeal symptoms may be treated by changing to the sublingual spitting method, but laryngeal findings should be examined, and the dosage should be carefully increased.

Pilot Study: Pneumatic Compression Garment Therapy for Postradiotherapy Laryngopharyngeal Edema.

OBJECTIVES: Pneumatic compression garment therapy (PCGT) has been established as treatment for postradiotherapy lymphedema, and its use in head and neck patients is becoming more common. Although effects on interstitial edema of the cervical soft tissues have been studied, effects on internal laryngopharyngeal edema, as well as associated symptoms of dysphagia and dysphonia, have yet to be published. METHODS: We surveyed 7 patients treated with radiation for head and neck cancer (HNC) who had also been prescribed PCGT for cervical lymphedema. Patients were asked about subjective experience with the device, and also administered the Eating Assessment Tool-10 (EAT-10) and Voice Handicap Index-10 (VHI-10) surveys regarding their symptoms after using PCGT. Laryngoscopy videos from these same periods were also reviewed and scored using a validated tool for assessing laryngopharyngeal edema. RESULTS: 85% of patients reported at least some improvement in dysphagia and dysphonia following PCGT. Average EAT-10 score after PCGT was 11.4 and average VHI-10 score after PCGT was 8.7. These compare more favorably to historical scores for the same questionnaires in similar patient populations. Laryngeal edema scores on endoscopic examination were not significantly different after at least 3 months of therapy (pre: 20.15, post: 20.21, P = .975); however, the utility of this result is limited by a low inter-rater reliability (Krippendorff α = .513). CONCLUSIONS: While we are unable to show any difference in objective assessment of laryngopharyngeal edema on endoscopic examination in this small pilot study, patients report substantial subjective improvement in postradiotherapy dysphagia and dysphonia following cervical PCGT that warrants more formal investigation.

Current status of the management of hereditary angioedema in China: a patient-based, cross-sectional survey.

BACKGROUND: Hereditary angioedema (HAE) is a rare, inherited disorder characterized by unpredictable and recurrent cutaneous and mucosal oedema. OBJECTIVES: This study aimed to identify the current status of HAE management in China. MATERIALS AND METHODS: An internet-based survey was sent to 129 patients with type 1 and 2 HAE diagnosed between 1983 and 2017; a total of 107 patients provided complete disease management information. RESULTS: The survey response rate was 82.9% (107 responders). Ten patients reported 18 fresh frozen plasma (FFP) transfusions for the main purpose of treating lethal laryngeal oedema. Two patients reported adverse events. Eighty-nine (83.2%) patients had taken danazol for long-term prophylaxis, and 74 (69.2%) were on long-term danazol prophylaxis. Among patients on long-term prophylaxis, 56 (75.7%) patients reported a decrease in their annual attack frequency after taking danazol. Twenty-five (33.8%) patients had an attack frequency of no more than once per year. The most common side effects were menstrual disorders, weight gain, osteoarticular pain, acne, and sebaceous hypersecretion. Patients with higher education levels, positive family histories, and laryngeal oedema attacks before medication tended to show better adherence. Patients maintaining high or medium adherence showed better control of laryngeal and gastrointestinal involvement than patients with low adherence. Four (3.7%) patients reported current or past use of tranexamic acid. CONCLUSION: Attenuated androgen and FFP transfusions remain the mainstay in China, where specifically targeted drugs are currently lacking. Maintaining good medication adherence should be recommended to achieve improved disease control.

Management of Laryngotracheal trauma: A review of current trends and future Directions.

Laryngotracheal trauma is rare but can pose serious threats to one's life. Presenting symptoms vary according to the severity of injury. Immediate Airway control is first step in the management, intubation should be considered by a senior member of the trauma team if the injury is minor while tracheostomy should be reserved for more severe injuries. Evaluation by a fibre-optic laryngoscopy and CT scan should be done whenever possible. Reconstruction is done according to the site involved using suture, titanium miniplates and stents. Tissue engineering has added a new horizon in this management but up till now complete laryngotracheal regeneration is very far-fetched, but tissue regeneration at individual sites have shown some positive results. More work needs to be done in this less explored field including laryngeal transplantation.

Inhalational injury and the larynx: A review.

OBJECTIVE: To review and discuss the existing research on the pathophysiology, impact and management of inhalational injury on the larynx and lower respiratory tract. DATA SOURCES: A literature search was conducted on the PubMed, MedLine, Embase, Web of Science and Google Scholar databases based on the keywords "airway burn", "inhalational injury" and "larynx". REVIEW METHODS: Inclusion criteria included English language studies containing original and review data on airway injury. Data was reported using the Preferred Reporting Items for Systematic Reviews and Meta-Analysis guidelines. CONCLUSIONS: Abnormal laryngeal and lower airway findings are common in burns patients and the incidence tends to increase with severity of the burns. Most patients with abnormal findings remain dysphonic decades after the initial injury. Larynx, the inlet to the airway, is exposed to the most intense thermal damage and highest concentration of chemical in inhalational injury. Airway injury is common and may result in long term morbidity. Healing of this tissue architecture is prolonged and different from cutaneous burn. Many patients receive prolonged intubation for medical complications that arise due to the burn injury. The degree of subglottic damage, however, is more extensive and occurs sooner compared with those without inhalational injuries. IMPLICATIONS FOR PRACTICE: With advances in acute medical and surgical management of burn and inhalational injury, airway injury is an important secondary outcome with lasting impact. Awareness of these potential complications and early involvement of medical and allied health team are important steps in improving patient care. A multi-disciplinary approach to management will optimise the short and long-term morbidity management and ultimately our patients' quality of life.

Emergency percutaneous transtracheal jet ventilation in a hypoxic cardiopulmonary resuscitation setting: a life-saving rescue technique.

(Un)anticipated difficult airway remains a challenge in anaesthesia. Percutaneous transtracheal jet ventilation has been shown to be an adequate technique for temporary oxygenation and ventilation and has been described as an acknowledged method in emergency settings of an unanticipated difficult airway. These emergency settings can be considered as low incidence high-risk situations. Both technical and non-technical skills should be trained regularly as education and simulation continues to play an important factor in patient safety. Furthermore, postoperative laryngeal oedema due to altered lymphatic drainage patterns must be considered as a possible mechanism of an upper airway obstruction in combination with a history of neck dissection and radiotherapy.

Hereditary angio-oedema with C1 inhibitor deficiency: Characteristics and diagnostic delay of Czech patients from one centre

BACKGROUND: Hereditary angio-oedema (HAE) is manifested by repeated episodes of localised subcutaneous or sub-mucosal oedema. Symptoms are extremely variable in frequency, localisation, and severity. Atypical or mild clinical symptoms of the disease may lead to erroneous diagnosis, causing diagnostic delay. The goal of this study was to assess how diagnostic delay has changed over 33 years at a single referral centre. METHODS: We analysed diagnostic delay and first symptoms of HAE in patients who were diagnosed at an immunology department between 1980 and 2013. Patient's records were analysed. RESULTS: The median diagnostic delay in 77 HAE type 1 and 2 patients was seven (range, 0-42) years. The difference observed in diagnostic delay between probands (18 [0-42] years) and others (1 [0-37] year) was significant (p < 0.001). Our data show a significant negative correlation between the length of diagnostic delay and the year of diagnosis in our group of patients (p = 0.024). The median age of first symptoms among all HAE patients (N = 64) was 17 (1-40) years. The first symptoms of HAE in 64 patients were analysed. Twenty-six patients had abdominal, seventeen peripheral, five facial, two urogenital, and three had laryngeal oedema as the first manifestation of the disease. The last death that was attributed to HAE was in 1977. CONCLUSIONS: Our observations demonstrate improved awareness of HAE among physicians, as documented by the significant decrease in diagnostic delay. It is believed that earlier treatment will improve patient quality of life and life expectancy

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[Analysis of respiratory complications in 922 severely burned patients].

OBJECTIVE: To discuss the distribution of the respiratory complications in severely burned patients and the prevention and treatment experience against them. METHODS: Medical records of 922 adult patients with severe or extremely severe burn hospitalized in our burn ICU from January 2005 to December 2012 were screened and retrospectively analyzed, including patients transferred from other hospitals, patients with total burn area above 50% TBSA, the distribution and treatment of respiratory complications, and the mortality. Data were processed with chi-square test. RESULTS: The constituent ratio of patients transferred to our hospital was 71.1% in 2007 and 40.2% in 2010, while it remained about 50.0% in the other years. The ratios of patients with total burn area larger than 50% TBSA and that of patients with respiratory complications (χ(2) = 2.637, P > 0.05) showed no significant changes each year. Among these 922 burn patients, 523 patients suffered respiratory complications, among which laryngeal edema (50.9%, 266 cases), pulmonary infection (21.6%, 113 cases), and ARDS (11.9%, 62 cases) were the main components, with no significant change each year (with χ(2) values respectively 6.132, 6.319, 0.016, P values above 0.05). Among the patients with respiratory complications, except for 36 were not treated actively, 487 were treated by ventilator among which 228 had undergone tracheostomy, and the constituent ratios in the 8 years were close. Fifteen patients died, with 2 died of laryngeal edema, 3 of ARDS, and 10 of sepsis or MODS as a result of sepsis. CONCLUSIONS: Patients with severe burns were at high risk of respiratory complications, among which laryngeal edema was common, followed by pulmonary infection and ARDS. Prophylactic tracheostomy, mechanical ventilation, wound therapy, and anti-infection were all effective measures of prevention and treatment against these complications.

Vocal fold pseudocyst: results of 46 cases undergoing a uniform treatment algorithm.

OBJECTIVES/HYPOTHESIS: To describe treatment results and identify predictors of the need for surgical intervention in patients with vocal fold pseudocyst. STUDY DESIGN: Retrospective cohort study with longitudinal followup via survey. METHODS: Clinical records were reviewed for demographic information, VHI-10 score, and degree of severity of dysphonia. Videostroboscopic examinations were evaluated for presence of vocal fold pseudocyst, along with additional clinical variables, including laterality, reactive lesion, paresis, varix, and hemorrhage. Follow-up surveys were sent to all participants to evaluate current VHI-10 score and degree of vocal limitation. Results were analyzed to determine predictors of surgery and recurrence of pathology. RESULTS: Forty-six patients (41F:5M) with pseudocyst (40 unilateral: 6 bilateral) were reviewed. Twenty-three (50%) had reactive lesions, nineteen (41%) had paresis by clinical criteria, 10 (22%) had varices, and 6 (13%) had hemorrhage on examination. All underwent initial behavioral management (2-12 sessions of voice therapy; mean of 8 sessions). Seventeen (37%) eventually required surgical intervention. No demographic or clinical variables proved predictive of surgical intervention. Follow-up surveys were completed by 63% of patients, and 79% agreed with the statement that they were not professionally limited by their voices. CONCLUSION: This experience supports behavioral management as an initial intervention in patients with pseudocyst, sufficient by itself to restore vocal function in approximately two out of three patients. Neither initial severity nor any of the studied clinical findings predicted the need for surgery. The large majority of patients with pseudocyst are able to be treated effectively without impact in their professional function.

Pediatric mumps with laryngeal edema.

Mumps virus infection primarily affects the salivary glands and may incur various complications. Laryngeal edema is such a rare complication that few adult cases have been reported. We report the first known pediatric patient with mumps with laryngeal edema. An 8-year-old boy developed dyspnea after a rapidly progressive swelling of his face and neck. Laryngoscopy revealed edematous changes in the supraglottic and subglottic regions, and computed tomography confirmed significant laryngeal edema in addition to swelling of the cervical soft tissue and the salivary glands. Laboratory findings revealed a high serum amylase level and confirmed the diagnosis of mumps. Intravenous steroid administration alleviated the dyspnea, although the patient required temporary tracheal intubation to maintain airway patency. He did not need tracheotomy and did not experience any other complications. Laryngeal edema must be regarded as a rare, potentially life-threatening complication of mumps. When mumps is diagnosed with significant swelling of the neck, an emergency airway should be established to prevent airway obstruction.

[Emergency treatment and management of anaphylaxis]. / Akuttherapie und Management der Anaphylaxie.

Anaphylaxis, the maximal manifestation of an immediate allergic reaction, is a life-threatening systemic reaction. The immediate therapy is chosen according to the clinical manifestations and new German guidelines are in preparation. Required measures include immediate removal of allergen, adequate positioning, assessment of severity and organ involvement and activation of emergency medical services. In anaphylaxis with primarily cardiovascular involvement, epinephrine is the treatment of choice and given together with volume substitution, oxygen, H(1)-antihistamines and corticosteroids. Obstruction of the airways is treated with intramuscular and inhaled epinephrine, or alternatively ß(2)-sympathicomimetics, and oxygen. Abdominal or cutaneous involvement, such as generalized urticaria, usually can be treated with intravenous H(1)-antihistamines, glucocorticoids and surveillance. In patients with anaphylaxis, the elicitor of the reaction has to be diagnosed by allergy testing. Patients with sustained risk for anaphylaxis should receive a self-medication kit and should be educated about behavioral patterns needed for prophylaxis and therapy of anaphylactic reactions. Patient educational intervention increases knowledge about anaphylaxis, and practical competence and thus, is a basis of a successful management of anaphylaxis.

Treatment of laryngeal hereditary angioedema.

BACKGROUND: In the emergency department, patients with laryngeal swelling and an inconclusive patient history may receive treatment for allergy-mediated angioedema. Intubation may be necessary if the patient does not respond to treatment. Because angioedema subtypes respond to different interventions, a correct diagnosis is vital. OBJECTIVES: Review the differential diagnosis of angioedema and characteristics differentiating subtypes. Discuss therapies for angioedema subtypes. Introduce therapies for prevention and acute treatment of hereditary angioedema (HAE). CASE REPORT: A 10-year-old girl presented with laryngeal swelling unresponsive to diphenhydramine, methylprednisolone, and epinephrine. It was later revealed that she had a family history of HAE, was C1 inhibitor deficient, and enrolled in a clinical study of acute HAE treatment. She was given 1000 units of nanofiltered C1 inhibitor and was able to swallow within 30 min. She was prescribed routine prophylaxis with C1 inhibitor concentrate and has had no subsequent severe HAE swelling attacks. CONCLUSION: This case illustrates the need for providers to consider HAE in light of available diagnostic testing and recent Food and Drug Administration approval of specific therapies for HAE.

Hereditary angioedema: a case of near fatal laryngeal swelling in a 41-year-old woman.

Hereditary angioedema (HAE) is characterized by nonpitting, nonpruritic subcutaneous, or submucosal edema that may be accompanied by a nonpruritic serpentine erythematous rash. The swelling in this autosomal dominantly inherited disorder is mediated by uncontrolled localized bradykinin production. The extremities, face, gastrointestinal tract, genitalia, larynx, and trunk are commonly involved. Nearly one-third of patients who are not treated may suffer asphyxiation from a laryngeal attack. HAE symptoms overlap with other conditions that cause swelling. An accurate diagnosis is therefore crucial when considering therapy. Recently approved treatments for HAE swelling attacks include C1 esterase inhibitor concentrate and ecallantide. This case describes a 41-year-old woman who was treated ineffectively for more than 20 years, was switched to an effective therapy, but suffered near fatal laryngeal edema due to a missed dose of the effective therapy. The case underscores the need for increased awareness of HAE among clinicians and the importance of good compliance to therapy among patients with HAE.