Azathioprine hypersensitivity syndrome: report of two cases

Azathioprine hypersensitivity syndrome is a rare but potentially severe side effect of azathioprine use. It has a variable and non-specific presentation making it difficult to distinguish from sepsis or disease relapse. High clinical suspicion is therefore required for recognition and prompt cessation of azathioprine for symptom resolution. Herewith two cases of severe azathioprine hypersensitivity syndrome are described, one in association with Sweet syndrome. Both presented with vague symptoms 2 weeks after commencing azathioprine for antineutrophil cytoplasmic antibody vasculitis. The differentials of sepsis and disease relapse were considered prior to cessation of azathioprine which resulted in a dramatic improvement in both cases. These cases highlight the diagnostic challenge azathioprine hypersensitivity syndrome presents. It should be suspected when there is a temporal relationship to drug initiation, with absence of infection or serological evidence of disease relapse.

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Emergency Department and Radiological Cost of Delayed Diagnosis of Cannabinoid Hyperemesis

Journal of Addiction ◽

10.1155/2019/1307345 ◽

2019 ◽

Vol 2019 ◽

pp. 1-4 ◽

Cited By ~ 8

Author(s):

David I. Zimmer ◽

Ross McCauley ◽

Varun Konanki ◽

Joseph Dynako ◽

Nuha Zackariya ◽

...

Keyword(s):

Emergency Department ◽

Delayed Diagnosis ◽

The United States ◽

Clinical Syndrome ◽

Cannabis Use ◽

Diagnostic Challenge ◽

High Clinical Suspicion ◽

Ed Visits ◽

Inappropriate Utilization ◽

Utilization Of Healthcare

Background. Chronic cannabis use has become prevalent with decriminalization, medical prescription, and recreational legalization in numerous US states. With this increasing incidence of chronic cannabis use a new clinical syndrome has become apparent in emergency departments and hospitals across the country, termed Cannabinoid Hyperemesis (CH). CH has been described as cyclical vomiting and abdominal pain in the setting of chronic cannabis use, which is often temporarily relieved by hot showers. CH presents a diagnostic challenge to clinicians who do not have a high clinical suspicion for the syndrome and can result in high costs and resource utilization for hospitals and patients. This study investigates the expenditures associated with delayed CH evaluation and delayed diagnosis. Methods. This is a retrospective observational study of 17 patients diagnosed with CH at three medical centers in the United States from 2010 to 2015, consisting of two academic centers and a community hospital. Emergency department (ED) costs were calculated and analyzed for patients eventually diagnosed with CH. Results. For the 17 patients treated, the total cost for combined ED visits and radiologic evaluations was an average of $76,920.92 per patient. On average these patients had 17.9 ED visits before the diagnosis of CH was made. Conclusion. CH provides a diagnostic challenge to clinicians without a high suspicion of the syndrome and may become increasingly prevalent with current trends toward cannabis legalization. The diagnosis of CH can be made primarily through a thorough history and physical examination. Awareness of this syndrome can save institutions money, prevent inappropriate utilization of healthcare resources, and save patients from unnecessary diagnostic tests.

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Bullous hemorrhagic Sweet syndrome with cryptococcoid neutrophils in patients positive for antineutrophil cytoplasmic antibody without primary vasculitis

JAAD Case Reports ◽

10.1016/j.jdcr.2020.10.006 ◽

2020 ◽

Vol 6 (12) ◽

pp. 1196-1200

Author(s):

Alex Sherban ◽

Collin Fuller ◽

Mansha Sethi ◽

Eleni McGeehin ◽

Dawn Hirokawa ◽

...

Keyword(s):

Antineutrophil Cytoplasmic Antibody ◽

Sweet Syndrome ◽

Primary Vasculitis

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Association of Serum Calprotectin (S100A8/A9) Level With Disease Relapse in Proteinase 3–Antineutrophil Cytoplasmic Antibody–Associated Vasculitis

Arthritis & Rheumatology ◽

10.1002/art.39814 ◽

2016 ◽

Vol 69 (1) ◽

pp. 185-193 ◽

Cited By ~ 23

Author(s):

Ruth J. Pepper ◽

Juliana B. Draibe ◽

Ben Caplin ◽

Fernando C. Fervenza ◽

Gary S. Hoffman ◽

...

Keyword(s):

Antineutrophil Cytoplasmic Antibody ◽

Proteinase 3 ◽

Disease Relapse

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Azathioprine Hypersensitivity Syndrome in a Cohort of Antineutrophil Cytoplasmic Antibody-Associated Vasculitis Patients

The Journal of Allergy and Clinical Immunology In Practice ◽

10.1016/j.jaip.2018.10.018 ◽

2019 ◽

Vol 7 (3) ◽

pp. 1004-1009 ◽

Cited By ~ 2

Author(s):

Arno C. Hessels ◽

Jan Stephan F. Sanders ◽

Annick A.J.M. van de Ven ◽

Bart-Jan Kroesen ◽

Annechien J.A. Lambeck ◽

...

Keyword(s):

Antineutrophil Cytoplasmic Antibody ◽

Hypersensitivity Syndrome

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Sweet Syndrome Associated With Myelodysplastic Syndrome: Report of a Case. Review of the Literature

Reumatología Clínica (English Edition) ◽

10.1016/j.reumae.2012.01.002 ◽

2013 ◽

Vol 9 (4) ◽

pp. 246-247

Author(s):

Delia Reina ◽

Dacia Cerdà ◽

Daniel Roig ◽

Ramon Fíguls ◽

M. Luz Villegas ◽

...

Keyword(s):

Myelodysplastic Syndrome ◽

Case Review ◽

Review Of The Literature ◽

Sweet Syndrome ◽

Syndrome Report

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Anticonvulsant Hypersensitivity Syndrome: Report of 2 Cases from Kuwait

Medical Principles and Practice ◽

10.1159/000070760 ◽

2003 ◽

Vol 12 (3) ◽

pp. 197-199 ◽

Cited By ~ 6

Author(s):

Hanan A. Bin-Nakhi ◽

Sameera Sadeq ◽

Raimond G. Pinto ◽

Yousef Habeeb

Keyword(s):

Hypersensitivity Syndrome ◽

Anticonvulsant Hypersensitivity Syndrome ◽

Syndrome Report

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Severe cutaneous presentation of hydralazine-induced ANCA vasculitis without renal or pulmonary involvement, complicated by DIC

BMJ Case Reports ◽

10.1136/bcr-2020-238609 ◽

2021 ◽

Vol 14 (5) ◽

pp. e238609

Author(s):

Mae Xintong Huo ◽

Alzira Rocheteau M Avelino ◽

Gurpreet Singh

Keyword(s):

Intensive Therapy ◽

Antineutrophil Cytoplasmic Antibody ◽

Pulmonary Involvement ◽

High Dose ◽

Diagnostic Challenge ◽

Drug Induced ◽

Mucosal Involvement ◽

Ulcerative Lesions ◽

Long Term Prognosis

Hydralazine is a common arterial vasodilator used in the management of congestive heart failure and hypertension. It can be associated with drug-induced lupus and less commonly antineutrophil cytoplasmic antibody (ANCA) associated vasculitis (AAV). Drug-induced AAV typically has a favourable long-term prognosis. It is not commonly associated with primary skin involvement, as most cases also have notable kidney and lung disease. Cases with isolated skin findings are rare. We present a rare case of a 60-year-old woman on long-term hydralazine who presented with AAV with primary skin and mucosal involvement, in the form of diffuse bullous and ulcerative lesions, which posed a diagnostic challenge. Her hospital course was marked by several complications including disseminated intravascular coagulation. She required intensive therapy with high-dose steroids, plasmapheresis and rituximab. She tolerated immunosuppression well and with multidisciplinary supportive care, she recovered well and was able to be discharged from the hospital.

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Risperidone Induce Recurrence of Severe Acute Respiratory Distress in a Patient with Psychotic Disorders

European Psychiatry ◽

10.1016/j.eurpsy.2017.01.1160 ◽

2017 ◽

Vol 41 (S1) ◽

pp. S674-S675

Author(s):

N. Amel ◽

M. Hadjadj ◽

W. Maoudh

Keyword(s):

Intensive Care Unit ◽

Intensive Care ◽

Respiratory Distress ◽

Psychotic Disorders ◽

Specific Treatment ◽

Symptomatic Treatment ◽

Severe Side Effect ◽

Dramatic Improvement ◽

Anti Psychotic ◽

Medical File

Mrs. R, 61-years-old was admitted into the department on July 2015 with a history of major paranoid psychosis. Notion of asthma was indicated in the medical file without any specific treatment. At the admission she received risperidone 4 mg/day and duloxetine since one year. Between October 2015 and June 2016, the patient presented 19 episodes of respiratory decompensation, six of them required transfer in the emergency unit, and 3 in the intensive care unit (ICU) with the assistance of mechanical ventilation. During the last episode, transfer in the intensive care unit was refused by the ICU physician because of the high frequency of recurrence and the difficulty to extube the patient. “End of life” was therefore considered. Nevertheless, medical physician in charge of the patient decided to continue symptomatic treatment and to stop all anti-psychotic drugs susceptible to deteriorate respiratory disease. On the first day we observed a dramatic improvement and no recurrence occurred since now 3 months without any treatment. Between the admission and the occurrence of respiratory decompensation, hypereosinophilia (1610/mL) was observed with recovery in normal value after interruption of risperidone. We also noted an improvement of functional respiratory test. In conclusion, risperidone is an anti-psychotic drug largely used. Severe side effect may endanger life-threatening as described in this case. The recurrence of severe acute respiratory distress without induced factors founded needs to discuss the potential role of this drug.Disclosure of interestThe authors have not supplied their declaration of competing interest.

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