Hereditary angioedema (HAE) is a rare,autosomal dominant inherited disorder with an incidence of approximately 1 in 50,000.Among its various tapes,HAE with normal C1 inhibitor levels (HAE-nC1-INH)is exceptionally rare...Hereditary angioedema (HAE) is a rare,autosomal dominant inherited disorder with an incidence of approximately 1 in 50,000.Among its various tapes,HAE with normal C1 inhibitor levels (HAE-nC1-INH)is exceptionally rare.^([1]) HAE symptoms include recurrent episodes of skin and mucosal edema that can occur anywhere in the body.^([1-4]) Laryngeal edema is life-threatening,as it can lead to airway obstruction and potentially fatal suffocation.^([1-3])Edema of the gastrointestinal mucosa may cause abdominal pain,vomiting,and symptoms that are often misdiagnosed as acute abdomen.^([1-4]) This study included four patients,including one with HAE-nC1-INH (genetic testing revealed a heterozygous mutation in the KNG1 gene (c.1404G>C:p.Q468H)) and three with HAE due to C1 inhibitor deficiency (HAE-C1-INH).This case series aims to increase knowledge of HAE by illustrating its diverse clinical presentations and emphasizing features that may prompt clinical suspicion and facilitate timely diagnosis.展开更多
Objective To determine the safety and efficacy of fresh frozen plasma （FFP） iniusion for the treat- ment of hereditary angioedema （FIAE）. Methods The medical records of patients with HAE admitted to Peking Union ...Objective To determine the safety and efficacy of fresh frozen plasma （FFP） iniusion for the treat- ment of hereditary angioedema （FIAE）. Methods The medical records of patients with HAE admitted to Peking Union Medical College Fiospital who had received FFP infusion during 2004 and 2010 were reviewed and PubMed database iFom 1966 to the present were searched using the following key words： hereditary angioedema and fresh frozen plasma. The patient＇s age, sex, body location of HAE attacks, the dose of FFP infusion, time of beginning to improvenaent, time to complete remission, complication, C 1 inhibitor activity, and outcome were analyzed. Results A total of 13 enrolled patients （7 male and 6 female） received 16 times of FFP infusion, in- cluding 2 patients undergoing FFP infusion in Peking Union Medical College Hospital and 11 patients re- ported in the literature. The mean dosage of FFP infusion was 586±337 mL. Two cases suffered from wors- ening abdominal pain and one case experienced skin rash. Only I patient had no improvement in symptom owing to transfusion related reaction. There was a definite improvement in symptom 49± 19 minutes after beginning FFP infusion. The remission time decreased from 61.7±27.0 hours to 3.3 （2.0, 12.0） hours after FFP infusion. FFP infusion was effective for both type I and type Ⅱ HAE. Conclusion FFP seems to be safe and effective for acute attacks of HAE.展开更多
Three cases of transient proximal small bowel angioedema induced by intravenous administration of nonionic iodinated contrast media (CM) are presented.Computed tomography (CT) images in the venous phase displayed the ...Three cases of transient proximal small bowel angioedema induced by intravenous administration of nonionic iodinated contrast media (CM) are presented.Computed tomography (CT) images in the venous phase displayed the proximal small bowel with circumferential thickening of the wall including the duodenum and proximal segment of the jejunum.The bowel wall was normal in non-enhanced images,and normal or inconspicuous in arterial phase enhanced images.In one of the three cases,the bowel wall was thickened in venous phase but disappeared in the 40 s delayed phase images.No filling defect was seen in the lumen of the superior mesenteric artery and vein.No peritoneal effusion or mesentery abnormality was found.Each of these patients reported only mild abdominal discomfort and recovered without specific treatment within a short time.Only one patient suffered mild diarrhea after scanning which had resolved by the following day.The transient anaphylactic small bowel angioedema due to intravenous iodinated contrast media was easily diagnosed based on its characteristic CT findings and clinical symptoms.Differential diagnosis may include inflammatory and ischemic bowel disease,as well as neoplasms.A three-phase CT protocol and good under-standing of this disorder are fundamentally important in the diagnosis of this condition.The supposed etiology behind the transient anaphylactic reaction to intravenous administration of iodinated CM in small bowel is similar to other CM-induced hypersensitive immediate reactions.The predilection location of transient anaphylactic bowel angioedema is the small intestine,particularly the proximal segment.A speculated cause may be the richer supply of vessels in the small intestine,ample mucous folds and loose connective tissue in the duodenum and the jejunum.展开更多
Abdominal involvement in angioedema is often a challenge to diagnose.Acute onset abdominal pain is its most common presenting symptom,and misdiagnosis may lead to unnecessary surgical intervention.Familiarity with the...Abdominal involvement in angioedema is often a challenge to diagnose.Acute onset abdominal pain is its most common presenting symptom,and misdiagnosis may lead to unnecessary surgical intervention.Familiarity with the types and presentations of angioedema can be invaluable to clinicians as they consider the differential diagnoses of a patient presenting with abdominal pain. Detailed personal and family histories,careful physical examination of the patient,combined with knowledge of angioedema types,can help clinicians perform their diagnostic evaluation.An accurate diagnosis is essential in order to provide appropriate treatment to patients with angioedema.Depending upon the diagnosis,treatment may be the avoidance of provoking factors(such as allergens or medications),inhibiting histamine-provoked reactions,or treating C1 esterase inhibitor deficiency.展开更多
HEREDITARY angioedema （HAE） is an autosomal dominant inherited condition which was initially described by Osier in 1888.1 Patients with HAE can develop rapid subcutaneous or submucosal edema involving the hands, fee...HEREDITARY angioedema （HAE） is an autosomal dominant inherited condition which was initially described by Osier in 1888.1 Patients with HAE can develop rapid subcutaneous or submucosal edema involving the hands, feet, limbs, face, intestinal tract, even larynx and trachea. The mortality of an acute attack of HAE without treatment was reported as high as 30%.2 HAE is caused by the deficiency of Cl esterase inhibitor （CIINH） which results in episodes of edema in parts of the human body,展开更多
BACKGROUND: In recent years, thrombolytic therapy has become the main treatment of ischemic stroke. But the increasing use of alteplase in ischemic stroke has made some complications more evident. Angioedema is a rare...BACKGROUND: In recent years, thrombolytic therapy has become the main treatment of ischemic stroke. But the increasing use of alteplase in ischemic stroke has made some complications more evident. Angioedema is a rare but potentially life-threatening complication of alteplase treatment. Only a few studies have examined the incidence of angioedema after treatment with alteplase for stroke.METHODS: A 75-year-old man complaining of right hemiparesis was admitted to our emergency department. He was diagnosed as having acute ischemic stroke, and alteplase infusion was given two hours after the onset of stroke symptoms. Immediately after the completion of infusion he was noted to have a large swollen tongue.RESULTS: His neurological symptoms resolved gradually within 4 hours, whereas his upper extremity strength improved to 4/5 and lower extremity 5/5. Lingual edema resolved within 16 hours without any complication. He died from presumed nosocomial infection 5 days later.CONCLUSIONS: Lingual angioedema may appear as a possible complication in patients who were treated with alteplase. The management of these patients should be very careful.展开更多
A 46-year-old female patient with terminal ileum Crohn’s disease and ankylosing spondylitis presented with recurrent angioedema and urticaria. Investigations ruled out hereditary angioedema, and environmental or food...A 46-year-old female patient with terminal ileum Crohn’s disease and ankylosing spondylitis presented with recurrent angioedema and urticaria. Investigations ruled out hereditary angioedema, and environmental or food allergen triggers. She was diagnosed with chronic idiopathic urticaria with angioedema, and was treated with a trial of intravenous immunoglobulin immunotherapy, danazol, prednisone and hydroxyzine. Due to ongoing bowel and arthritic complaints, she was started on infliximab infusions and within 2 treatments, she had complete resolution of the angioedema and urticaria, as well as of the bowel and arthritic symptoms. Unfortunately she developed allergic reactions to the infliximab and was switched to another anti-tumor necrosis factor （TNF）-a agent, adalimumab. Since then, she has had no further angioedema or urticaria, and her Crohn’s disease has been quiescent. This is the first known case report of chronic idiopathic urticaria with angioedema coexistent with Crohn’s disease that was successfully treated with anti-TNF-α agents.展开更多
BACKGROUND Paliperidone palmitate is a once-monthly injectable,atypical antipsychotic.To our knowledge,there has been only one report of paliperidone palmitate-induced angioedema presenting with acute laryngeal edema ...BACKGROUND Paliperidone palmitate is a once-monthly injectable,atypical antipsychotic.To our knowledge,there has been only one report of paliperidone palmitate-induced angioedema presenting with acute laryngeal edema with subsequent respiratory arrest.Here,we present a case report of paliperidone palmitate-induced angioedema with a relatively mild clinical presentation compared with the previously reported case,and the patient's condition was not complicated by lifethreatening anaphylaxis.CASE SUMMARY A 79-year-old female,who had a major neurocognitive disorder due to Alzheimer's disease with behavioral disturbances.Paliperidone palmitate was offlabel used to control her aggression,irritability,and psychosis.After induction doses(150 mg and 100 mg intramuscularly,given 1 wk apart),she developed intermittent swelling of the face,eyelids,and lips on day 17 after the initial dose,and the edema was explicitly seen on day 20.The diagnosis was paliperidone palmitate-induced angioedema.The monthly injection dose was discontinued on day 33 after the initial dose.The angioedema was subsequently alleviated,and it had completely resolved by day 40 after the initial dose.CONCLUSION Paliperidone palmitate-induced angioedema is a rare condition and can present with a mild,intermittent facial edema,which may be overlooked in clinical practice.展开更多
Hereditary angioedema （HAE） is an autosomal dominant condition that affects one in about 50 000 persons, characterized by recurrent episodes of subcutaneous or submucosal swelling involving the hands, feet, limbs, f...Hereditary angioedema （HAE） is an autosomal dominant condition that affects one in about 50 000 persons, characterized by recurrent episodes of subcutaneous or submucosal swelling involving the hands, feet, limbs, face, intestinal tract, even larynx and trachea.展开更多
BACKGROUND Angioedema is a disorder characterized by edema of the face,lips,tongue,and extremities due to increased vascular permeability.Angioedema of the tongue usually occurs bilaterally,and the incidence of unilat...BACKGROUND Angioedema is a disorder characterized by edema of the face,lips,tongue,and extremities due to increased vascular permeability.Angioedema of the tongue usually occurs bilaterally,and the incidence of unilateral angioedema of the tongue is rare.This study reports a rare case of unilateral angioedema of the tongue with no identifiable cause and repeated recurrence even after discontinuation of an angiotensin-converting enzyme inhibitor.CASE SUMMARY The patient was a 65-year-old woman with pre-existing hypertension and hyperlipidemia and had been receiving 20 mg/d of lisinopril.She was diagnosed with angioedema due to unilateral swelling of the tongue.No airway obstruction was observed,and the symptoms improved following the administration of 0.3 mg of epinephrine intramuscularly and 125 mg of methylprednisolone intravenously.Although lisinopril was discontinued,unilateral angioedema of the tongue continued to recur every 2-5 mo,with the symptoms improving following the administration of prednisolone and an antihistamine.Daily oral administration of 500 mg of tranexamic acid after dinner prevented the recurrence of angioedema.CONCLUSION Careful monitoring and identification of the underlying mechanism play a crucial role in the treatment of angioedema.展开更多
Hereditary angioedema secondary to C1-inhibitor deficiency is a rare autosomal dominant disorder characterized by a deficiency of C1 esterase inhibitor.An eight-year-old girl showed periorbital painless swelling, diag...Hereditary angioedema secondary to C1-inhibitor deficiency is a rare autosomal dominant disorder characterized by a deficiency of C1 esterase inhibitor.An eight-year-old girl showed periorbital painless swelling, diagnosed as ethmoiditis. A craniofacial scan did not evidence a paranasal sinus involvement, C1INH levels were undetectable, with low C4 levels: 7.6 mg/dl and C1INH: <8.46 mg/dl. The genetic study identified a rare mutation of the C1INH gene. This clinical report is of relieve because paediatric cases described in literature are rare, did not presented a positive family history, and received a diagnosis after many attacks. Furthermore our girl received a prompt diagnosis of HAE at the first attack of angioedema.展开更多
Angiotensin-converting enzyme inhibitor induced angioedema (AIIA) can vary from mild to life-threatening. A vast majority of cases of AIIA occur within a month of starting an angiotensin-converting enzyme-inhibitor (A...Angiotensin-converting enzyme inhibitor induced angioedema (AIIA) can vary from mild to life-threatening. A vast majority of cases of AIIA occur within a month of starting an angiotensin-converting enzyme-inhibitor (ACE-I). We present a 48-year-old male who presented with respiratory failure secondary to AIIA, after being on lisinopril for over 8 years. He had no previous complications secondary to lisinopril and aside from smoking, carried no risk factors for AIIA. Despite conventional treatment for angioedema, he had a prolonged stay in the Medical Intensive Care Unit (MICU). Following discharge, there hasn’t been a recurrence of AIIA since the discontinuation of lisinopril. The case is intended to caution that AIIA remains possible even late into a chronic regimen of ACE-I. This is a risk that shouldn’t be neglected, even with sparse risk factors or longer duration of ACE-I use. Conventional treatment is not currently in line with proposed etiologies of AIIA. We advocate for more clinical trials involving pharmaceutical agents targeting bradykinin accumulation.展开更多
Neurotic angioedema may be secondary to ACE (Angiotensin-converting enzyme) inhibitor. From a case of neurotic angioedema in a hypertensive man under ACE (Angiotensin-converting enzyme) inhibitor, we expose the clinic...Neurotic angioedema may be secondary to ACE (Angiotensin-converting enzyme) inhibitor. From a case of neurotic angioedema in a hypertensive man under ACE (Angiotensin-converting enzyme) inhibitor, we expose the clinical characteristics typical of the edema and its evolution. A 73-year-old man has consulted for edema of the face and redness of the palmar surface of the hand not pruriginous for 2 days. He has already had conventional anti-edematous treatments but no changes. In the absence of a technical platform, no paraclinical examination was carried out. The cessation of ACE inhibitor and the administration of tranexamic acid were made immediately. Signs regressed within 24 hours. Neurotic angioedema is a diagnostic problem in countries where biological examination is still limited and if practitioners are unaware of the possibility of its occurrence during IEC treatment. While the patient’s prognosis depends on the precocity of management.展开更多
A 23 years old female presented to Lincoln Medical and Mental Health center with skin rash and angioedema after she received 20 pills of lamotrigine 25 mg, and the patient used to take this medication before two table...A 23 years old female presented to Lincoln Medical and Mental Health center with skin rash and angioedema after she received 20 pills of lamotrigine 25 mg, and the patient used to take this medication before two tables a day for many months, after she received the appropriate management she improved. After reviewing MEDLINE we found a few cases reported life threatening complications related to lamotrigine intoxication and sudden increase the dose, so we should be aware about these complications before prescribing this medication.展开更多
A pregnant woman diagnosed with type 1 angioedema seeks care at a public hospital for planning the delivery. This report presents ways to prevent and manage an acute HAE crisis during childbirth and early postpartum w...A pregnant woman diagnosed with type 1 angioedema seeks care at a public hospital for planning the delivery. This report presents ways to prevent and manage an acute HAE crisis during childbirth and early postpartum without the availability of first-line medications, such as plasma-derived human C1-INH concentrate.展开更多
Angioedema is a known side effect of angiotensin-converting enzyme inhibitors (ACE-I). However, trauma precipitating angioedema is a rare event. We detail a case of trauma-induced angioedema in a patient taking an ACE...Angioedema is a known side effect of angiotensin-converting enzyme inhibitors (ACE-I). However, trauma precipitating angioedema is a rare event. We detail a case of trauma-induced angioedema in a patient taking an ACE-I. Specifically, a patient presented to the emergency department (ED) having suffered a seizure from symptomatic hyponatremia;later, the patient precipitously developed angioedema requiring nasotracheal intubation. Herein, the mechanisms and treatments for angioedema are discussed. Acute angioedema is important to the emergency medicine physician because quick recognition, regardless of its precipitant can stave off untoward complications, possible respiratory failure and airway emergencies.展开更多
Angiotensin Receptor Blockers (ARB) are used as an alternative of Angiotensin Converting Enzyme Inhibitors (ACEI) in patients where ACEIs cannot be used because of their known adverse effects, cough and angioedema. Th...Angiotensin Receptor Blockers (ARB) are used as an alternative of Angiotensin Converting Enzyme Inhibitors (ACEI) in patients where ACEIs cannot be used because of their known adverse effects, cough and angioedema. Thus ARB induced angioedema is considered to be a rare phenomenon and it is continued to be used as an alternatives of ACEIs. In this case report, we reported a case of 78-year-old gentleman who presented to emergency department with losartan, an ARB induced angioedema, who did not have history of any previous use of ACEIs. He was given steroids and antihistamine as a treatment. His angioedema resolved rapidly and he was discharged after six hours of emergency department (ED) observation with stable hemodynamically. We, the authors by reporting this case, wants to make clinicians aware ARB, however rarely, can cause angioedema, which can be life threatening if clinicians are not aware of it and diagnose and stop the offending drug promptly and treat it early.展开更多
Background: Hereditary angioedema (HAE) is a primary immunodeficiency disorder characterized by C1 complement inhibitor deficiency and unregulated activation of complement. Aspirin hypersensitivity is related to an in...Background: Hereditary angioedema (HAE) is a primary immunodeficiency disorder characterized by C1 complement inhibitor deficiency and unregulated activation of complement. Aspirin hypersensitivity is related to an increase in the amount of leukotrienes with eosinophil and mast cell activation and increased levels of glandular kallikrein with upregulated local conversion of bradykinin. Both conditions can be present in the same patient. Objectives: We present five patients with HAE;they were all being treated in similar ways according to the therapeuthic options available in our institute (danazol). However, three of them had recurrent episodes of angioedema;in these cases, it was identified aspirin hypersensitivity as a cause of poor disease control. A review of the literature is included. Case Presentation: We present the cases of four females and one male (age range 21 - 58 years) with type I HAE. Subjects were all ISSSTE beneficiaries (state workers) treated at the National Medical Center “20 de Noviembre”. Aspirin hypersensitivity was identified in three patients. Elimination of NSAIDs along with dietary elimination of high salicylate-containing foods improved control of angioedema crisis (severe and/ or recurrent episodes). Discussion: Aspirin hypersensitivity was identified as a factor for poor control in our patients with HAE. Such cases improved with dietary elimination of high salicylate-containing foods and avoidance of NSAIDs. Conclusions: This is the first report of patients with HAE and aspirin hypersensitivity as a cause of poor control. We recommend a deliberate search of these comorbidities, especially in cases of poor disease control. Further studies are needed to continue the investigation on this topic.展开更多
Hereditary angioedema is a rare but life-threatening disease, usually resulting from upper respiratory tract traumas and stress. In this case report, we present the management of a 14-year-old female patient who was d...Hereditary angioedema is a rare but life-threatening disease, usually resulting from upper respiratory tract traumas and stress. In this case report, we present the management of a 14-year-old female patient who was diagnosed with hereditary angioedema and scheduled to undergo transurethral resection of bladder (TURB) procedure for bladder tumor. She was on prophylactic danazol treatment and prior to the operation the dose of danazol was increased. On the day of the operation, patient was given C1-IHN concentrate and was sedated. In conclusion, hereditary angioedema is a rare disease in which multidisciplinary and aggressive approach during anesthesia would yield successful results.展开更多
Liquid injectable silicone has been used for soft tissue augmentation for five decades. Many complications following liquid silicone injection have been reported. To diagnose and manage silicone granuloma remains diff...Liquid injectable silicone has been used for soft tissue augmentation for five decades. Many complications following liquid silicone injection have been reported. To diagnose and manage silicone granuloma remains difficult. Silicone granuloma must be diagnosed with the history of liquid silicone injection and the histology of tissue biopsy. We presented a case of granulomatous reaction after the injection of liquid silicone for chin augmentation forty years ago, causing total facial swelling, which mimicking angioedema initially. We administered methylprednisolone to the patient. Initial response to methylprednisolone was favorable.展开更多
基金supported by the National Social Science Fund of China (19VJX168)。
文摘Hereditary angioedema (HAE) is a rare,autosomal dominant inherited disorder with an incidence of approximately 1 in 50,000.Among its various tapes,HAE with normal C1 inhibitor levels (HAE-nC1-INH)is exceptionally rare.^([1]) HAE symptoms include recurrent episodes of skin and mucosal edema that can occur anywhere in the body.^([1-4]) Laryngeal edema is life-threatening,as it can lead to airway obstruction and potentially fatal suffocation.^([1-3])Edema of the gastrointestinal mucosa may cause abdominal pain,vomiting,and symptoms that are often misdiagnosed as acute abdomen.^([1-4]) This study included four patients,including one with HAE-nC1-INH (genetic testing revealed a heterozygous mutation in the KNG1 gene (c.1404G>C:p.Q468H)) and three with HAE due to C1 inhibitor deficiency (HAE-C1-INH).This case series aims to increase knowledge of HAE by illustrating its diverse clinical presentations and emphasizing features that may prompt clinical suspicion and facilitate timely diagnosis.
文摘Objective To determine the safety and efficacy of fresh frozen plasma （FFP） iniusion for the treat- ment of hereditary angioedema （FIAE）. Methods The medical records of patients with HAE admitted to Peking Union Medical College Fiospital who had received FFP infusion during 2004 and 2010 were reviewed and PubMed database iFom 1966 to the present were searched using the following key words： hereditary angioedema and fresh frozen plasma. The patient＇s age, sex, body location of HAE attacks, the dose of FFP infusion, time of beginning to improvenaent, time to complete remission, complication, C 1 inhibitor activity, and outcome were analyzed. Results A total of 13 enrolled patients （7 male and 6 female） received 16 times of FFP infusion, in- cluding 2 patients undergoing FFP infusion in Peking Union Medical College Hospital and 11 patients re- ported in the literature. The mean dosage of FFP infusion was 586±337 mL. Two cases suffered from wors- ening abdominal pain and one case experienced skin rash. Only I patient had no improvement in symptom owing to transfusion related reaction. There was a definite improvement in symptom 49± 19 minutes after beginning FFP infusion. The remission time decreased from 61.7±27.0 hours to 3.3 （2.0, 12.0） hours after FFP infusion. FFP infusion was effective for both type I and type Ⅱ HAE. Conclusion FFP seems to be safe and effective for acute attacks of HAE.
文摘Three cases of transient proximal small bowel angioedema induced by intravenous administration of nonionic iodinated contrast media (CM) are presented.Computed tomography (CT) images in the venous phase displayed the proximal small bowel with circumferential thickening of the wall including the duodenum and proximal segment of the jejunum.The bowel wall was normal in non-enhanced images,and normal or inconspicuous in arterial phase enhanced images.In one of the three cases,the bowel wall was thickened in venous phase but disappeared in the 40 s delayed phase images.No filling defect was seen in the lumen of the superior mesenteric artery and vein.No peritoneal effusion or mesentery abnormality was found.Each of these patients reported only mild abdominal discomfort and recovered without specific treatment within a short time.Only one patient suffered mild diarrhea after scanning which had resolved by the following day.The transient anaphylactic small bowel angioedema due to intravenous iodinated contrast media was easily diagnosed based on its characteristic CT findings and clinical symptoms.Differential diagnosis may include inflammatory and ischemic bowel disease,as well as neoplasms.A three-phase CT protocol and good under-standing of this disorder are fundamentally important in the diagnosis of this condition.The supposed etiology behind the transient anaphylactic reaction to intravenous administration of iodinated CM in small bowel is similar to other CM-induced hypersensitive immediate reactions.The predilection location of transient anaphylactic bowel angioedema is the small intestine,particularly the proximal segment.A speculated cause may be the richer supply of vessels in the small intestine,ample mucous folds and loose connective tissue in the duodenum and the jejunum.
文摘Abdominal involvement in angioedema is often a challenge to diagnose.Acute onset abdominal pain is its most common presenting symptom,and misdiagnosis may lead to unnecessary surgical intervention.Familiarity with the types and presentations of angioedema can be invaluable to clinicians as they consider the differential diagnoses of a patient presenting with abdominal pain. Detailed personal and family histories,careful physical examination of the patient,combined with knowledge of angioedema types,can help clinicians perform their diagnostic evaluation.An accurate diagnosis is essential in order to provide appropriate treatment to patients with angioedema.Depending upon the diagnosis,treatment may be the avoidance of provoking factors(such as allergens or medications),inhibiting histamine-provoked reactions,or treating C1 esterase inhibitor deficiency.
文摘HEREDITARY angioedema （HAE） is an autosomal dominant inherited condition which was initially described by Osier in 1888.1 Patients with HAE can develop rapid subcutaneous or submucosal edema involving the hands, feet, limbs, face, intestinal tract, even larynx and trachea. The mortality of an acute attack of HAE without treatment was reported as high as 30%.2 HAE is caused by the deficiency of Cl esterase inhibitor （CIINH） which results in episodes of edema in parts of the human body,
文摘BACKGROUND: In recent years, thrombolytic therapy has become the main treatment of ischemic stroke. But the increasing use of alteplase in ischemic stroke has made some complications more evident. Angioedema is a rare but potentially life-threatening complication of alteplase treatment. Only a few studies have examined the incidence of angioedema after treatment with alteplase for stroke.METHODS: A 75-year-old man complaining of right hemiparesis was admitted to our emergency department. He was diagnosed as having acute ischemic stroke, and alteplase infusion was given two hours after the onset of stroke symptoms. Immediately after the completion of infusion he was noted to have a large swollen tongue.RESULTS: His neurological symptoms resolved gradually within 4 hours, whereas his upper extremity strength improved to 4/5 and lower extremity 5/5. Lingual edema resolved within 16 hours without any complication. He died from presumed nosocomial infection 5 days later.CONCLUSIONS: Lingual angioedema may appear as a possible complication in patients who were treated with alteplase. The management of these patients should be very careful.
文摘A 46-year-old female patient with terminal ileum Crohn’s disease and ankylosing spondylitis presented with recurrent angioedema and urticaria. Investigations ruled out hereditary angioedema, and environmental or food allergen triggers. She was diagnosed with chronic idiopathic urticaria with angioedema, and was treated with a trial of intravenous immunoglobulin immunotherapy, danazol, prednisone and hydroxyzine. Due to ongoing bowel and arthritic complaints, she was started on infliximab infusions and within 2 treatments, she had complete resolution of the angioedema and urticaria, as well as of the bowel and arthritic symptoms. Unfortunately she developed allergic reactions to the infliximab and was switched to another anti-tumor necrosis factor （TNF）-a agent, adalimumab. Since then, she has had no further angioedema or urticaria, and her Crohn’s disease has been quiescent. This is the first known case report of chronic idiopathic urticaria with angioedema coexistent with Crohn’s disease that was successfully treated with anti-TNF-α agents.
文摘BACKGROUND Paliperidone palmitate is a once-monthly injectable,atypical antipsychotic.To our knowledge,there has been only one report of paliperidone palmitate-induced angioedema presenting with acute laryngeal edema with subsequent respiratory arrest.Here,we present a case report of paliperidone palmitate-induced angioedema with a relatively mild clinical presentation compared with the previously reported case,and the patient's condition was not complicated by lifethreatening anaphylaxis.CASE SUMMARY A 79-year-old female,who had a major neurocognitive disorder due to Alzheimer's disease with behavioral disturbances.Paliperidone palmitate was offlabel used to control her aggression,irritability,and psychosis.After induction doses(150 mg and 100 mg intramuscularly,given 1 wk apart),she developed intermittent swelling of the face,eyelids,and lips on day 17 after the initial dose,and the edema was explicitly seen on day 20.The diagnosis was paliperidone palmitate-induced angioedema.The monthly injection dose was discontinued on day 33 after the initial dose.The angioedema was subsequently alleviated,and it had completely resolved by day 40 after the initial dose.CONCLUSION Paliperidone palmitate-induced angioedema is a rare condition and can present with a mild,intermittent facial edema,which may be overlooked in clinical practice.
文摘Hereditary angioedema （HAE） is an autosomal dominant condition that affects one in about 50 000 persons, characterized by recurrent episodes of subcutaneous or submucosal swelling involving the hands, feet, limbs, face, intestinal tract, even larynx and trachea.
文摘BACKGROUND Angioedema is a disorder characterized by edema of the face,lips,tongue,and extremities due to increased vascular permeability.Angioedema of the tongue usually occurs bilaterally,and the incidence of unilateral angioedema of the tongue is rare.This study reports a rare case of unilateral angioedema of the tongue with no identifiable cause and repeated recurrence even after discontinuation of an angiotensin-converting enzyme inhibitor.CASE SUMMARY The patient was a 65-year-old woman with pre-existing hypertension and hyperlipidemia and had been receiving 20 mg/d of lisinopril.She was diagnosed with angioedema due to unilateral swelling of the tongue.No airway obstruction was observed,and the symptoms improved following the administration of 0.3 mg of epinephrine intramuscularly and 125 mg of methylprednisolone intravenously.Although lisinopril was discontinued,unilateral angioedema of the tongue continued to recur every 2-5 mo,with the symptoms improving following the administration of prednisolone and an antihistamine.Daily oral administration of 500 mg of tranexamic acid after dinner prevented the recurrence of angioedema.CONCLUSION Careful monitoring and identification of the underlying mechanism play a crucial role in the treatment of angioedema.
文摘Hereditary angioedema secondary to C1-inhibitor deficiency is a rare autosomal dominant disorder characterized by a deficiency of C1 esterase inhibitor.An eight-year-old girl showed periorbital painless swelling, diagnosed as ethmoiditis. A craniofacial scan did not evidence a paranasal sinus involvement, C1INH levels were undetectable, with low C4 levels: 7.6 mg/dl and C1INH: <8.46 mg/dl. The genetic study identified a rare mutation of the C1INH gene. This clinical report is of relieve because paediatric cases described in literature are rare, did not presented a positive family history, and received a diagnosis after many attacks. Furthermore our girl received a prompt diagnosis of HAE at the first attack of angioedema.
文摘Angiotensin-converting enzyme inhibitor induced angioedema (AIIA) can vary from mild to life-threatening. A vast majority of cases of AIIA occur within a month of starting an angiotensin-converting enzyme-inhibitor (ACE-I). We present a 48-year-old male who presented with respiratory failure secondary to AIIA, after being on lisinopril for over 8 years. He had no previous complications secondary to lisinopril and aside from smoking, carried no risk factors for AIIA. Despite conventional treatment for angioedema, he had a prolonged stay in the Medical Intensive Care Unit (MICU). Following discharge, there hasn’t been a recurrence of AIIA since the discontinuation of lisinopril. The case is intended to caution that AIIA remains possible even late into a chronic regimen of ACE-I. This is a risk that shouldn’t be neglected, even with sparse risk factors or longer duration of ACE-I use. Conventional treatment is not currently in line with proposed etiologies of AIIA. We advocate for more clinical trials involving pharmaceutical agents targeting bradykinin accumulation.
文摘Neurotic angioedema may be secondary to ACE (Angiotensin-converting enzyme) inhibitor. From a case of neurotic angioedema in a hypertensive man under ACE (Angiotensin-converting enzyme) inhibitor, we expose the clinical characteristics typical of the edema and its evolution. A 73-year-old man has consulted for edema of the face and redness of the palmar surface of the hand not pruriginous for 2 days. He has already had conventional anti-edematous treatments but no changes. In the absence of a technical platform, no paraclinical examination was carried out. The cessation of ACE inhibitor and the administration of tranexamic acid were made immediately. Signs regressed within 24 hours. Neurotic angioedema is a diagnostic problem in countries where biological examination is still limited and if practitioners are unaware of the possibility of its occurrence during IEC treatment. While the patient’s prognosis depends on the precocity of management.
文摘A 23 years old female presented to Lincoln Medical and Mental Health center with skin rash and angioedema after she received 20 pills of lamotrigine 25 mg, and the patient used to take this medication before two tables a day for many months, after she received the appropriate management she improved. After reviewing MEDLINE we found a few cases reported life threatening complications related to lamotrigine intoxication and sudden increase the dose, so we should be aware about these complications before prescribing this medication.
文摘A pregnant woman diagnosed with type 1 angioedema seeks care at a public hospital for planning the delivery. This report presents ways to prevent and manage an acute HAE crisis during childbirth and early postpartum without the availability of first-line medications, such as plasma-derived human C1-INH concentrate.
文摘Angioedema is a known side effect of angiotensin-converting enzyme inhibitors (ACE-I). However, trauma precipitating angioedema is a rare event. We detail a case of trauma-induced angioedema in a patient taking an ACE-I. Specifically, a patient presented to the emergency department (ED) having suffered a seizure from symptomatic hyponatremia;later, the patient precipitously developed angioedema requiring nasotracheal intubation. Herein, the mechanisms and treatments for angioedema are discussed. Acute angioedema is important to the emergency medicine physician because quick recognition, regardless of its precipitant can stave off untoward complications, possible respiratory failure and airway emergencies.
文摘Angiotensin Receptor Blockers (ARB) are used as an alternative of Angiotensin Converting Enzyme Inhibitors (ACEI) in patients where ACEIs cannot be used because of their known adverse effects, cough and angioedema. Thus ARB induced angioedema is considered to be a rare phenomenon and it is continued to be used as an alternatives of ACEIs. In this case report, we reported a case of 78-year-old gentleman who presented to emergency department with losartan, an ARB induced angioedema, who did not have history of any previous use of ACEIs. He was given steroids and antihistamine as a treatment. His angioedema resolved rapidly and he was discharged after six hours of emergency department (ED) observation with stable hemodynamically. We, the authors by reporting this case, wants to make clinicians aware ARB, however rarely, can cause angioedema, which can be life threatening if clinicians are not aware of it and diagnose and stop the offending drug promptly and treat it early.
文摘Background: Hereditary angioedema (HAE) is a primary immunodeficiency disorder characterized by C1 complement inhibitor deficiency and unregulated activation of complement. Aspirin hypersensitivity is related to an increase in the amount of leukotrienes with eosinophil and mast cell activation and increased levels of glandular kallikrein with upregulated local conversion of bradykinin. Both conditions can be present in the same patient. Objectives: We present five patients with HAE;they were all being treated in similar ways according to the therapeuthic options available in our institute (danazol). However, three of them had recurrent episodes of angioedema;in these cases, it was identified aspirin hypersensitivity as a cause of poor disease control. A review of the literature is included. Case Presentation: We present the cases of four females and one male (age range 21 - 58 years) with type I HAE. Subjects were all ISSSTE beneficiaries (state workers) treated at the National Medical Center “20 de Noviembre”. Aspirin hypersensitivity was identified in three patients. Elimination of NSAIDs along with dietary elimination of high salicylate-containing foods improved control of angioedema crisis (severe and/ or recurrent episodes). Discussion: Aspirin hypersensitivity was identified as a factor for poor control in our patients with HAE. Such cases improved with dietary elimination of high salicylate-containing foods and avoidance of NSAIDs. Conclusions: This is the first report of patients with HAE and aspirin hypersensitivity as a cause of poor control. We recommend a deliberate search of these comorbidities, especially in cases of poor disease control. Further studies are needed to continue the investigation on this topic.
文摘Hereditary angioedema is a rare but life-threatening disease, usually resulting from upper respiratory tract traumas and stress. In this case report, we present the management of a 14-year-old female patient who was diagnosed with hereditary angioedema and scheduled to undergo transurethral resection of bladder (TURB) procedure for bladder tumor. She was on prophylactic danazol treatment and prior to the operation the dose of danazol was increased. On the day of the operation, patient was given C1-IHN concentrate and was sedated. In conclusion, hereditary angioedema is a rare disease in which multidisciplinary and aggressive approach during anesthesia would yield successful results.
文摘Liquid injectable silicone has been used for soft tissue augmentation for five decades. Many complications following liquid silicone injection have been reported. To diagnose and manage silicone granuloma remains difficult. Silicone granuloma must be diagnosed with the history of liquid silicone injection and the histology of tissue biopsy. We presented a case of granulomatous reaction after the injection of liquid silicone for chin augmentation forty years ago, causing total facial swelling, which mimicking angioedema initially. We administered methylprednisolone to the patient. Initial response to methylprednisolone was favorable.
