Presentation of an extraordinary colic: abdominal pain as the first and only utterance of an acquired C1-inhibitor deficiency.

C1-inhibitor deficiency is a rare disease which incorporates acute self-limiting intermittent swelling of the subcutaneous tissue and mucous membranes. Attacks most frequently affect the face and/or the upper airway. Isolated angioedema of the small bowel is an uncommon manifestation and often accompanied by diagnostic delay. In the present case, abdominal pain turned out to be the first and only utterance of an acquired C1-inhibitor deficiency, secondary to a splenic marginal zone lymphoma. Imaging showed wall thickening of the small intestine, ascites and splenomegaly. The abdominal pain and intestinal wall thickening with surrounding ascites on imaging spontaneously resolved each episode within 2-3 days. Gastrointestinal manifestations of angioedema may mimic an acute abdomen, and subsequently one-third of these patients undergo unnecessary surgery prior to a definite diagnosis. This emphasises the importance of considering the diagnosis in case of an 'extraordinary colic'.

A Gut Feeling: Isolated Small Bowel Angioedema due to Angiotensin-Converting Enzyme Inhibitor.

Isolated angioedema of the small intestine is a rare adverse event in patients taking angiotensin-converting enzyme inhibitors. Here, we present a case of visceral angioedema in a 32-year-old woman who presented with left upper quadrant pain, nausea, vomiting, diarrhea, and characteristic radiographic signs of small bowel angio-edema, six months after starting lisinopril. Her symptoms improved within 48 hours of withholding the offending agent and with supportive care. We discuss the epidemiology, pathophysiology, diagnosis, and management of angiotensin-converting enzyme inhibitor- induced angioedema.

Bowel angioedema induced by iodinated contrast media.

We present the case of a patient with small bowel angioedema induced by iodinated contrast media during computed tomography. It is important to know this entity and to differentiate it from other intestinal diseases in order to avoid inappropriate treatment.

Point-of-Care Ultrasound on Management of Cellulitis Versus Local Angioedema in the Pediatric Emergency Department.

OBJECTIVES: To evaluate whether ultrasound can differentiate between cellulitis and angioedema from insect bites in pediatric patients. METHODS: A prospective, pre-post study in an urban pediatric emergency department of patients younger than 21 years with soft tissue swelling from insect bites without abscesses were enrolled. Treating physician's pretest opinions regarding the diagnosis and need for antibiotics were determined. Ultrasound of the affected areas was performed, and effects on management were recorded. Further imaging, medications, and disposition were at the discretion of the enrolling physician. Phone call follow-ups were made within a week of presentation. RESULTS: Among 103 patients enrolled with soft tissue swelling secondary to insect bites, ultrasound changed the management in 27 (26%) patients (95% confidence interval [CI], 18-35%). Of the patients who were indeterminate or believed to require antibiotics, ultrasound changed management in 6 (23%) of 26 patients (95% CI, 6%-40%). In those patients who were believed not to require antibiotics, ultrasound changed management in 12 (16%) 77 patients (95% CI, 7%-24%). Patients with diagnosis of local angioedema achieved symptom resolution 1.4 days sooner than patients diagnosed with cellulitis (mean, -1.389; 95% CI, -2.087 to -0.690; P < 0.001). No patient who was initially diagnosed as local angioedema received antibiotics upon patient follow-up. CONCLUSIONS: Point-of-care ultrasound changed physician management in 1 of 4 patients in the pediatric emergency department with soft tissue swelling secondary to insect bites. Ultrasound may guide the management in these patients and lead to improved antibiotic stewardship in conjunction with history and physical examination.

Angioedema late in the course of adjuvant nivolumab therapy for melanoma.

Although immune checkpoint inhibitors improve survival in cancer patients, they have also been linked with unusual side effects. The most common side effects of these agents are immune-mediated phenomena such as itching, skin rash, arthralgias, mild transaminitis and asymptomatic thyroid dysfunction. We describe herein a case of facial angioedema occurring 20 weeks after initiating adjuvant nivolumab therapy for melanoma. The patient had full resolution of symptoms with cessation of nivolumab and a short steroid course. As the causality of an association between nivolumab and angioedema seems legitimate, we expect further similar cases to surface in patients treated with immune checkpoint inhibitors. Prompt drug withdrawal and steroids are crucial to ensure favorable clinical outcomes in these patients.

An unusual presentation of ACE inhibitor-induced visceral angioedema.

ACE inhibitors (ACEi) are common anti-hypertensive drugs that can cause angioedema. Though classic, or facial angioedema is rare, visceral angioedema is even less common. When angioedema occurs, it typically presents early, within 30 days of initiating therapy. Visceral angioedema most commonly presents with nausea, emesis, abdominal pain and diarrhoea, and thus is often mistaken for an episode of gastroenteritis. When a CT scan is obtained, it typically shows characteristic findings, including ascetic fluid, mild mesenteric oedema and thickening of the small bowel. In this case report, we present a patient who did not experience her first episode of visceral angioedema until after she had been on ACEi therapy for 5-7 years. In addition, she experienced recurrent episodes of visceral angioedema that were separated by approximately 4 years at a time. Both of these features make for a particularly unique presentation.

Angioedema in Stroke Patients With Thrombolysis.

Background and Purpose- Oral angioedema (OA) is a rare but life-threatening complication in patients with ischemic stroke receiving intravenous thrombolysis with r-tPA (recombinant tissue-type plasminogen activator). This study intended to determine associations between thrombolysis-related OA and ischemic stroke lesion sites using a voxel-wise lesion analysis. Methods- Prospective registry data were used to identify ischemic stroke patients with thrombolysis-related OA between 2002 and 2018. For the study registry, ethics approval was obtained by the Ethics Committee of the Friedrich-Alexander Universität (FAU) Erlangen-Nürnberg (clinical registry registration: 377_17Bc). Ischemic stroke patients with thrombolysis treatment but without OA admitted in the years 2011 and 2012 comprised the control group. Ischemic lesions were manually outlined on magnetic resonance imaging (1.5T or 3T) or computed tomographic scans and transformed into stereotaxic space. We determined the lesion overlap and compared the absence or presence of OA voxel-wise between patients with and without lesions in a given voxel using the Liebermeister test. Stroke severity was rated using the National Institutes of Health Stroke Scale score, and blood pressure, heart rate, blood glucose levels, and body temperature were determined on admission. Results- Fifteen ischemic stroke patients with thrombolysis-related OA were identified. The voxel-wise analysis yielded associations between OA and ischemic lesions in the insulo-opercular region with a right hemispheric dominance. Mean blood pressure was significantly lower in patients with OA than in controls. Age, National Institutes of Health Stroke Scale scores, infarct volumes, heart rate, and blood glucose levels did not differ between patients with and without OA. Conclusions- The voxel-wise analysis linked thrombolysis-related OA to right insulo-opercular lesions. The lower blood pressure in patients with thrombolysis-related OA may reflect bradykinin effects causing vasodilatation and increasing vascular permeability.

[A case of angioedema with dysarthria mimicking transient ischemic attack].

An 85-year-old woman was transported to our emergency room by ambulance with a complaint of slurred speech. Neurological examination revealed dysarthria only. We considered that lingual edema identified on physical examination might have influenced dysarthria. However, we were unable to perform sufficient evaluation, since she could not open her mouth widely or push the tongue out beyond the lips. We considered the incidence of acute cerebrovascular disease because of the acute onset, and performed emergency brain MRI. Imaging revealed that although no abnormality was present in the brain parenchyma, edema of the tongue and soft palate was evident on T2-weighted sagittal imaging. We confirmed the dysarthria was caused by tongue edema due to angioedema. In addition, we diagnosed angiotensin-converting enzyme inhibitor (ACEI)-induced angioedema, because ACEI had been started 2 months earlier as pharmacotherapy for hypertension. Tongue swelling due to angioedema should be considered when examining patients with dysarthria.

Angioedema of the small bowel caused by lisinopril.

In conjunction with BMJ Case Reports, DTB will feature occasional drug-related cases that are likely to be of interest to readers. These will include cases that involve recently marketed drugs for which there is limited knowledge of adverse effects and cases that highlight unusual reactions to drugs that have been marketed for several years.

[A Rare Cause of an Angioedema-Like Habitus]. / Nicht jede Gesichtsschwellung ist ein Angioödem.

A Rare Cause of an Angioedema-Like Habitus Abstract. We report about a 69-year-old patient arriving at the emergency room with suspected allergic reaction grade III due to a pronounced facial swelling, dyspnea and dizziness. However, the cause was a bilateral pneumothorax with massive soft-tissue emphysema. On the one side it was due to a spontaneous secondary genesis in the context of a chronic pulmonary disease, on the other side it was due to a traumatic serial rib fracture.

Angioedema intestinal aislado inducido por enalapril / Isolated intestinal angioedema induced by enalapril

El angioedema inducido por inhibidores de la enzima convertidora de angiotensina es una entidad poco frecuente caracterizada por edema en piel y mucosas, debido al aumento de la permeabilidad vascular provocada por la inhibición de la enzima convertidora y el subsiguiente aumento de la bradiquinina. De manera frecuente cursa con compromiso facial y de mucosas, siendo infrecuente el compromiso intestinal o de vía aérea. El angioedema intestinal puede presentarse asociado a angioedema facial o aislado, siendo este último excepcional. Cursa con episodios recurrentes de dolor, distensión abdominal y diarrea acuosa con recuperación completa en dos o tres días. Si bien es una entidad poco frecuente, el hecho de que esté asociada a fármacos utilizados con frecuencia nos hace incluirla en el diagnóstico diferencial del dolor abdominal recurrente. Presentamos un caso de angioedema intestinal aislado, asociado al uso de enalapril.

Angioedema induced by angiotensin converting enzyme inhibitors is a rare entity characterized by skin and mucosal edema, due to increased vascular permeability caused by inhibition of the converting enzyme and subsequent increase in bradykinin. It frequently presents with facial and mucosal involvement, being uncommon the intestinal or airway compromise. Intestinal angioedema may be associated with facial or isolated angioedema, the latter being exceptional. It is associated with recurrent episodes of pain, abdominal distention and watery diarrhea which complete recovery in two or three days. Although it is a rare entity, the fact that it is associated with frequently used drugs makes us include it in the differential diagnosis of recurrent abdominal pain. We report a case of isolated intestinal angioedema associated with the use of enalapril.

[Isolated intestinal angioedema induced by enalapril]. / Angioedema intestinal aislado inducido por enalapril.

Angioedema induced by angiotensin converting enzyme inhibitors is a rare entity characterized by skin and mucosal edema, due to increased vascular permeability caused by inhibition of the converting enzyme and subsequent increase in bradykinin. It frequently presents with facial and mucosal involvement, being uncommon the intestinal or airway compromise. Intestinal angioedema may be associated with facial or isolated angioedema, the latter being exceptional. It is associated with recurrent episodes of pain, abdominal distention and watery diarrhea which complete recovery in two or three days. Although it is a rare entity, the fact that it is associated with frequently used drugs makes us include it in the differential diagnosis of recurrent abdominal pain. We report a case of isolated intestinal angioedema associated with the use of enalapril.

Angioedema orofaríngeo secundario a captopril sublingual: un caso inusual con afectación glandular / Oropharyngeal angioedema induced by sublingual captopril: an unusual case with glandular involvement

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