[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.

Laryngeal edema as a symptom of local cytokine release syndrome after BCMA-targeting CAR-T therapy for relapsed and refractory multiple myeloma.

Cytokine release syndrome (CRS) can be a major side effect of chimeric antigen receptor T-cell (CAR-T) therapy, and may occasionally become life-threatening in patients with factors such as high tumor burden or poor performance status. Among the many CRS events observed in B-cell maturation antigen (BCMA)-targeting CAR-T therapy, local symptoms (also called local CRS) are poorly understood due to their low frequency. Here, we present the case of a 54-year-old woman with refractory multiple myeloma exhibiting laryngeal edema as a local CRS. Before CAR-T therapy, she was diagnosed with progressive disease indicated by a left thyroid mass. After local irradiation, she received the BCMA-targeting CAR-T agent idecabtagene vicleucel (ide-cel). On day 2, the patient developed CRS, which resolved on treatment with tocilizumab. However, on day 4, laryngeal edema worsened, and was judged to be a local CRS. Intravenous dexamethasone rapidly reduced this edema. In conclusion, laryngeal edema rarely occurs as a local CRS, and to the best of our knowledge, has never been reported after ide-cel infusion. Dexamethasone was effective for reducing the local reaction that persisted after treatment of systemic symptoms with tocilizumab.

Laryngeal edema following remimazolam-induced anaphylaxis: a rare clinical manifestation.

BACKGROUND: Remimazolam is an ultra-short-acting intravenous benzodiazepine, which has been used as sedative/anesthetic in procedural sedation and anesthesia. Although peri-operative anaphylaxis due to remimazolam has been reported recently, the spectrum of the allergic reactions is still not fully known. CASE PRESENTATION: We describe a case of anaphylaxis following remimazolam administration in a male patient undergoing colonoscopy under procedural sedation. The patient presented complex clinical signs including airway changes, skin symptoms, gastrointestinal manifestations and hemodynamic fluctuations. Different from other reported cases, laryngeal edema was the initial and main clinical feature of remimiazolam-induced anaphylaxis. CONCLUSIONS: Remimazolam-induced anaphylaxis has a rapid onset and complex clinical features. This case reminds anesthesiologists should be particularly alert to the unknown adverse reactions of new anesthetics.

The Mortality from Hereditary Angioedema Worldwide: a Review of the Real-World Data Literature.

This study aims to review the global mortality secondary to laryngeal edema in patients diagnosed with hereditary angioedema and their relatives over the years, as well as to describe epidemiological and clinical findings associated with this outcome. An extensive search of the literature was made in PubMed, Scopus, and Embase to identify mortality rates secondary to laryngeal edema in patients with hereditary angioedema. The search was carried out in September of 2020 and in April of 2021, and keywords based on the MeSH terms were searched in three databases. The filter of language was used for finding only articles in English, and there was no limit to the year of publication. A total of twenty-three articles fulfilled the inclusion criteria for review and data extraction. The analyzed studies included 3292 patients and 411 deaths from asphyxia due to laryngeal edema. One hundred and three deaths in close relatives were described as secondary to the same cause. The main findings were summarized in tables: year and place of publication, the number of patients and deaths from laryngeal edema, patients previously diagnosed, and death age. Death rates from laryngeal edema had an average of one death for every 20 patients. Eight studies reported deaths in relatives. For every 7.4 patients in these studies, one relative died. The percentage among deaths in general associated with laryngeal edema was evaluated in three studies (32.7%, 44.4%, and 56%). The high frequency of this outcome suggests that deaths still occur, and improvement of hereditary angioedema treatment still needs to be met.

A case of late-onset larynx angioedema after ranibizumab intravitreal injection: Ranibizumab-related angioedema.

This case report describes an unusual case of late-onset larynx angioedema after ranibizumab intravitreal injection. A 72-year-old female patient presented to our clinic for decreased vision; right eye (RE) fundoscopy and optical coherence tomography (OCT) revealed mild chorioretinal atrophy and choroidal neovascularization with subretinal fluid. A ranibizumab injection was planned in the RE, with standard pretreatment with daily oral administration of betamethasone, cetirizine, and ranitidine because her medical history revealed two adverse drug reactions (ADRs) to contrast media (CM). Despite the premedication, 2 h after injection, the patient referred throat closing sensation and dyspnea that resolved within few hours by betamethasone 4 mg intramuscular injection, without further reoccurrence. In occasion of the second intravitreal injection, video rhinofibrolaryngoscopy revealed subglottic edema that resolved within few hours by betamethasone 4 mg intramuscular injection. This report suggests that, even in cases of intravitreal injection, patients with history of allergy, despite the anti-allergic treatment, should be hospitalized to detect late onset of such a life-threatening complication.

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.

Life-threatening laryngeal attacks in hereditary angioedema patients.

BACKGROUND: Hereditary angioedema due to C1-inhibitor deficiency (HAE-C1INH) is a rare genetic disease that runs in the family. As a result of the disease, acute swellings of the subcutaneous tissue and mucous membranes of the digestive and respiratory systems, including the larynx, occur. Any attack of the disease involving the throat and larynx is particularly dangerous and requires knowledge of clinical determinants of the disease and its proper management. MATERIALS AND METHODS: The study included adult consecutive HAE-C1INH patients having follow-up visits in our centre. The group was examined with a structured clinical questionnaire, concerning the last 6 months and focusing particularly on laryngeal swelling attacks. RESULTS: 55 subjects (F/M - 35/20, age range - 18-76) were included in the study. Laryngeal attacks occurred in 19 individuals (34.5%): 1-3, 4-6, and ≥7 attacks in 9, 8 and 2 patients, respectively, two of whom required intubation. In comparison to other patients, subjects with laryngeal attacks were characterised by significantly more frequent: (1) facial attacks, (2) severe disease activity, (3) the occurrence of female patients, (4) mental stress as a trigger of attacks. All patients with laryngeal attacks had a rescue medication at home and 15/19 (78%) patients could use it at home. Most of them used plasma-derived C1-inhibitor 17/19 (89.5%) and icatibant, 8/19 (42.1%). DISCUSSION: HAE-C1INH patients with laryngeal attacks require particular attention. Proper training regarding the identification of these patients, adequate management, access to emergency services and emergency drugs are essential to ensure the safety of subjects with this localization of HAE-C1INH attacks.

[Effectiveness of spirolactone on the treatment of laryngeal edema and complications after H-uvulopalatopharyngoplasty for patients with obstructive sleep apnea].

Objective:To determine whether taking spirolactone orally after H-UPPP may relieve laryngeal edema and complications for patients with obstructive sleep apnea（OSA）. Method:Fifty patients with OSA to undergo H-UPPP operation were randomly divided equally to the intervention group（taking spirolactone 20 mg orally twice a day for 7 days after H-UPPP） or the control group, all patients received conventional therapy after H-UPPP including anti-infection, hemostatic treatment, fluid replacement and expectorant by fogged absorption. The pharyngeal wound, diet, sleep and speaking pronunciation of all patients in each group were evaluated every other day in 7 days postoperation. The minimum oxygen saturation of blood（SaO2） during sleep at night each day and the period needed for staphyledema resolution of all patients were recorded and compared between each group. Result:Postoperatively, the intervention group had significantly slighter bleeding at wound site, better sleep and more legible speaking pronunciation than the control group after 3 days to 5 days（P<0.05）. The wound dehiscence of the intervention group was significantly slighter than the control group within 7 days after operation（P<0.05）. During 3 days to 7days after operation, the intervention group had a significantly better diet than the control group（P<0.05）. The average minimum SaO2during sleep at night in the intervention group was significantly higher than that in the control group from 3 days to 5 days post operation（P<0.05）. Period needed for staphyledema resolution in the intervention group（4.1±1.5） days was significantly shorter than that in the control group（5.9±1.8） days （P<0.05）. Conclusion:Taking spirolactone orally after H-UPPP may relieve laryngeal edema and complications for OSA patients, and it will also shorten the period needed for staphyledema resolution.

Use of erbium laser in the treatment of persistent post-radiotherapy laryngeal edema: a case report and review of the literature.

BACKGROUND: Post-radiotherapy laryngeal edema may affect the patients' quality of life, leading to repeated treatment attempts, which include massage/physical therapy, inhalations, and/or tracheostomy. CASE PRESENTATION: We report the surgical treatment approach of a 69-year-old patient with severe persistent post-radiotherapy laryngeal edema. After multiple inpatient admissions and failed conservative therapy, we used the erbium laser to treat the arytenoid edema. After repeated procedures, no complications were observed. The patient remained free of symptoms after 30 months of follow-up. CONCLUSIONS: The authors provide an easy-to-perform, safe, and quick surgical technique without non-severe or severe complications. Using this technique repeatedly, complications from excessive thermal damage with CO2 laser or unpleasant solutions such as tracheostomy can be avoided.

Usefulness of C1 esterase inhibitor protein concentrate in the management of hereditary angioedema of oropharyngeal tissue.

Hereditary angioedema is an autosomal-dominant disorder caused by mutation of the gene encoding the C1 esterase inhibitor (C1-INH). It manifests as painless, nonpruritic, nonpitting episodic swelling of the subcutaneous tissues, gastrointestinal, and upper respiratory tracts in the absence of urticaria. An attack typically takes 24 h to peak and resolves over 48-72 h. The most serious manifestation is a laryngeal attack associated with upper airway swelling. The aim of this case report is to describe the lifesaving use of a novel C1-INH protein concentrate in a patient with mild-to-moderate dyspnea caused by swelling of the upper airway (larynx) and tongue.

Hyaluronidase Injection in the Vocal Folds for Vocal Hemorrhage, Reinke Edema, and Hyaluronic Acid Overinjection: A Novel Application in the Larynx.

Hyaluronidase (HAase) injection into the vocal folds is an off-label use of an enzyme for liquefaction of hyaluronic acid (HA). HAase injection was performed in 14 cases in 13 patients. Office and operative injections of HAase were performed. The indications were five cases of overinjection of HA, six cases of Reinke edema and polypoid corditis, and three cases of acute vocal hemorrhage with early fusiform polyp formation. All the patients tolerated the HAase without complications. Significant voice improvements were noted between pre- and postinjection procedures as evaluated by a self-rating of voice by the Voice Handicap Index-10. A marked decrement in the mass on the side of the injection of HAase was noted in all groups successfully injected upon viewing by videostroboscopy. HAase prevented an additional operative phonosurgery in 10 patients. In conclusion, HAase injection can be used in the vocal folds with good effect in patients. The indications are polypoid corditis with overproduction of HA, acute vocal hemorrhage, and correction of iatrogenic HA overinjection.

Manejo del fracaso de la extubación en la unidad de terapia intensiva pediátrica / Management of extubation failure in the pediatric intensive care unit / Gestão de falha de extubação na Unidade de Terapia Intensiva Pediátrica

El fracaso de la extubación es la incapacidad de tolerar el retiro del tubo endotraqueal con necesidad de reintubación. Puede ser causada por la obstrucción de la vía aérea superior y por la aspiración o la incapacidad de manejar las secreciones, factores que se evidencian al retirar el tubo. La reintubación por fracaso respiratorio post-extubación debe ser evitada en lo posible, ya que aumenta el riesgo de neumonía asociada al respirador, la estancia hospitalaria y en terapia intensiva, y la morbimortalidad. Describimos el manejo del fracaso de la extubación de causa laríngea en la Unidad de Terapia Intensiva de un hospital pediátrico.

Extubation failure is the inability to tolerate removal of the endotracheal tube with subsequent reintubation. It can be caused by obstruction of the upper airway and aspiration or inadequate clearance of airway secretion, factors that become evident when removing the tube. Reintubation due to postextubation respiratory failure should be avoided if possible because it increases the risk of ventilator associated pneumonia, hospital and intensive care unit stay, and morbidity and mortality. We describe the management of failed extubation due to laryngeal cause in the Intensive Care Unit of a pediatric hospital.

Falha de extubação é a incapacidade de tolerar a remoção do tubo endotraqueal na necessidade de reintubação. Ela pode ser causada por obstrução das vias aéreas superiores e aspiração ou incapacidade para lidar com secreções, factores que são evidentes para remover o tubo. Reintubação devido a insuficiência respiratória pós-extubação deve ser evitada, se possível, porque aumenta o risco de pneumonia associada à ventilação mecânica, permanência hospitalar e terapia, morbidade e mortalidade. Nós descrevemos a gestão de extubação falhou devido à causa da laringe na Unidade de Terapia Intensiva de um hospital pediátrico.

Ultrasound-guided laryngeal air column width difference and the cuff leak volume in predicting the effectiveness of steroid therapy on postextubation stridor in adult. Are they useful?

OBJECTIVE: To evaluate the effectiveness of steroids therapy on postextubation stridor (PES) depending on the clinical response, the ultrasound guided laryngeal air column width difference (LACWD) and the cuff leak volume (CLV). DESIGN: Prospective, observer-blinded study. SETTING: Carried out in intensive care unit in Tanta university hospital. PATIENTS: 432 patients of both sexes received mechanical ventilation for more than 24 hours and met defined criteria for a weaning trial. MEASUREMENTS: Ultrasound guided LACWD and CLV were conducted before extubation. Patients developing postextubation stridor were intravenously given 8 mg of dexamethasone every 8 hours for 3 days. The clinical response, ultrasounds guided LACWD and CLV before and after steroid therapy were analyzed. Primary outcome and secondary outcomes of our study were reported. RESULTS: 387 patients (89.5%) had no PES and 45 patients (10.5%) had PES. Risk factors for PES were longer duration of intubation, younger age and female gender. Both CLV and LACWD showed significant decrease (P< .05) in patients with PES in comparison with no PES patients. 45 patients with PES received dexamethasone treatment; 18 were completely recovered while 27 patients needed reintubation after 1 h. of these 27 patients; 19 patients had successful extubation while 8 patients had tracheostomy. In patients with PES, CLV and LACWD showed significant increase (P< .05) in comparison with before administration. Level of CLV <200 ml and LACWD <0.9 mm carry high sensitivity with high positive predictive value and high accuracy for presence of PES. CONCLUSION: Steroids therapy improves postextubation stridor. Both LACWD and CLV are non-invasive and simple methods for monitoring of laryngeal edema regression after steroid therapy. We recommend administration of corticosteroids to patients with a lower level of leak volume and LACWD before extubation.

Potentially lethal pharyngolaryngeal edema with dyspnea in adult patients with mumps: A series of 5 cases.

In this article we describe 5 rare cases of mumps-associated pharyngolaryngeal edema. To the best of our knowledge, this report includes the first case of mumps-associated pharyngolaryngeal edema in a patient who had previously received mumps vaccination, and these cases represent the sixth report of mumps-associated pharyngolaryngeal edema in the English literature. All 5 of our patients with mumps infection were adults and manifested airway stenosis due to pharyngolaryngeal edema. This edema responded favorably to steroid treatment without tracheotomy. We conclude that a pharyngolaryngeal examination is recommended for patients with mumps infection. Steroid treatment is usually effective against pharyngolaryngeal edema; however, in certain cases tracheotomy may be inevitable.

The role of icatibant-the B2 bradykinin receptor antagonist-in life-threatening laryngeal angioedema in the ED.

Angioedema is a localized, sudden, transient, and often recurrent swelling of the deeper layers of the skin or mucosa with no epidermal component. It is caused by vasoactive substances that produce a transient increase in endothelial permeability. Angioedema involving the laryngeal components is a life-threatening situation for the patient,and it is a challenge for the emergency medicine physician to rapidly achieve a safety airway. Most cases of laryngeal angioedema are induced by histamine release; but 10% are bradykinin induced, which does not respond to the conventional algorithm of treating allergic induced angioedema. We present a case report of an angiotensin converting enzyme (ACE) inhibitor­induced laryngeal angioedema alleviated only after treatment with the new bradykinin receptor inhibitor medication icatibant which was licensed only for use in hereditary angioedema. We reviewed the literature for the use of icatibant in acquired drug-induced angioedema; and because of the similar pathogenesis between the hereditary angioedema and the ACE inhibitor­induced angioedema,we propose an algorithm for careful use of icatibantin life-threatening angioedema in the emergency department.

[Prophylactic use of icatibant before tracheal intubation of a patient with hereditary angioedema type III. (A literature review of perioperative management of patients with hereditary angioedema type III)]. / Uso profiláctico de icatibant en un caso de angioedema hereditario tipo III. (Revisión de la literatura sobre manejo perioperatorio en pacientes con angioedema hereditario tipo III).

Type III hereditary angioedema is a rare familial disorder that has recently been described as a separate condition. Triggers for episodes of angioedema include surgery, dental procedures, and tracheal intubation maneuvers. Since episodes affecting the upper airway are potentially life-threatening, prophylactic treatment is recommended in these situations. The use of icatibant (Firazyr(®)), for prevention of angioedema prior to tracheal intubation, is reported in a patient with type iii hereditary angioedema. A literature review on the anesthetic management of this condition was conducted.