scholarly journals Propylthiouracil-induced antineutrophil cytoplasmic antibody-associated vasculitis and agranulocytosis in a patient with Graves’ disease

Author(s):  
Maria Tomkins ◽  
Roxana Maria Tudor ◽  
Diarmuid Smith ◽  
Amar Agha

Summary This case is the first to describe a patient who experienced concomitant agranulocytosis and anti-neutrophil cytoplasmic antibody (ANCA)-associated vasculitis as an adverse effect of propylthiouracil treatment for Graves’ disease. A 42-year-old female with Graves’ disease presented to the emergency department (ED) with a 2-week history of fevers, night sweats, transient lower limb rash, arthralgia, myalgia and fatigue. She had been taking propylthiouracil for 18 months prior to presentation. On admission, agranulocytosis was evident with a neutrophil count of 0.36 × 109/L and immediately propylthiouracil was stopped. There was no evidence of active infection and the patient was treated with broad-spectrum antibodies and one dose of granulocyte colony-stimulation factor, resulting in a satisfactory response. On further investigation, ANCAs were positive with dual positivity for proteinase 3 and myeloperoxidase. There was no evidence of end-organ damage secondary to vasculitis, and the patient’s constitutional symptoms resolved completely on discontinuation of the drug precluding the need for immunosuppressive therapy. Learning points: Continued vigilance and patient education regarding the risk of antithyroid drug-induced agranulocytosis is vital throughout the course of treatment. ANCA-associated vasculitis is a rare adverse effect of antithyroid drug use. Timely discontinuation of the offending drug is vital in reducing end-organ damage and the need for immunosuppressive therapy in drug-induced ANCA-associated vasculitis. Similarities in the pathogenesis of agranulocytosis and drug-induced ANCA-associated vasculitis may offer insight into an improved understanding of vasculitis and agranulocytosis.

Author(s):  
Niamh O’Donnell ◽  
Aisling McCarthy ◽  
Ken Thong

Summary Carbimazole is a commonly used antithyroid drug (ATD), which is associated with several well-established side effects. However, Carbimazole-induced rhabdomyolysis is rarely reported in the literature. We report a 27-year-old male who presented with upper limb myalgia and significantly raised creatine kinase elevation, 1-month post commencement of Carbimazole for Graves’ disease. Carbimazole was ceased with subsequent clinical and biochemical improvement. Though the pathophysiology remains unclear, we hope to raise awareness regarding this rare adverse effect with a view to promote early recognition and prompt discontinuation of the offending medication caused by a commonly used medication in endocrinology. Learning points Musculoskeletal complaints can relate to unidentified and untreated hyperthyroidism. However one must be mindful that the treatment for these disorders can too induce myopathies. ATD-induced myopathy should be considered when there is a temporal relationship between introduction of ATDs and the onset of symptoms. If ATD-induced myopathy is being considered, other causes of myopathy should still be outruled. Prompt discontinuation of potentially offending medications may provide resolution of symptoms and avoid significant consequences.


2012 ◽  
Vol 97 (1) ◽  
pp. E49-E53 ◽  
Author(s):  
Natsuko Watanabe ◽  
Hiroto Narimatsu ◽  
Jaeduk Yoshimura Noh ◽  
Takuhiro Yamaguchi ◽  
Kazuhiko Kobayashi ◽  
...  

2018 ◽  
Vol 2018 ◽  
pp. 1-4 ◽  
Author(s):  
Yusho Ishii ◽  
Tsuyoshi Shirai ◽  
Yousuke Hoshi ◽  
Yoko Fujita ◽  
Yuko Shirota ◽  
...  

Here, we present the case of a 29-year-old woman with nasal septal perforation and positive myeloperoxidase- (MPO-) anti-neutrophil cytoplasmic antibody (ANCA). She had been diagnosed with Graves’ disease and had been treated with propylthiouracil (PTU) for 14 months. A biopsy of the nasal septum revealed an infiltration of inflammatory cells, with no evidence of malignancy or granulomatous change. Because of the use of PTU, destructive nasal lesion, and positive MPO-ANCA, she was diagnosed with drug-induced ANCA-associated vasculitis (AAV) and was treated with prednisolone and methotrexate after the cessation of PTU. Although PTU is known to be the medicine that induces drug-induced AAV, the manifestation of nasal septal perforation in drug-induced AAV is poorly identified. This is the rare case of drug-induced AAV which manifested only nasal septal perforation.


2020 ◽  
Vol 11 ◽  
pp. 204062232093063
Author(s):  
Chunjia Li ◽  
Yu Zou ◽  
Xin Lu ◽  
Guochun Wang ◽  
Xiaoming Shu

Background: The antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV) are rare multisystem autoimmune diseases characterized by inflammatory cell infiltration causing necrosis of small blood vessels. Pituitary involvement in AAV is poorly described. This study aimed to describe the prevalence, clinical characteristics, and outcomes of pituitary involvement in patients with AAV. Methods: A total of 150 patients diagnosed with AAV and hospitalized in the China–Japan Friendship Hospital between 2009 and 2019 were enrolled in this retrospective study. Patients diagnosed with pituitary involvement in AAV were selected for inclusion. Results: Three patients (2%) were identified with pituitary involvement. Two patients had positive ANCA titers, one with proteinase 3 positive and one with myeloperoxidase positive antibodies. Pituitary dysfunction presented as an initial symptom in one patient and developed over the course of the diseases in the other two patients. All three patients had abnormal hormones. Among them, two patients had an enlarged pituitary, shown by magnetic resonance images (MRIs), and one patient had a normal sized pituitary, shown by MRI, but presented with increased linear radioactivity uptake in the pituitary fossa by positron emission tomography-computed tomography. All patients were treated with corticosteroid and immunosuppressive therapy. Both pituitary dysfunction and vasculitis were in remission. Conclusion: Pituitary involvement is uncommon in AAV and it can occur at any point during AAV. The main clinical manifestations are central diabetes insipidus and panhypopituitarism. Immunosuppressive therapy could significantly alleviate clinical symptoms as well as pituitary imaging.


Thyroid ◽  
2019 ◽  
Vol 29 (10) ◽  
pp. 1390-1398 ◽  
Author(s):  
Nami Suzuki ◽  
Jaeduk Yoshimura Noh ◽  
Marino Hiruma ◽  
Akiko Kawaguchi ◽  
Mitsuha Morisaki ◽  
...  

2020 ◽  
pp. 1-4
Author(s):  
MosabNouraldein Mohammed Hamad

Agranulocytosis is an infrequent and serious side effect of antithyroid drugs characterized by a noticeable reduction in granulocyte and neutrophil count, it usually occurs within the first 2-3 months of treatment. There is a variety of mechanisms by which ATD can induce agranulocytosis, direct drug effects, and immunological mechanisms. We present 33 years old female attended Atbara teaching hospital who has developed agranulocytosis 2 weeks after starting ATD to treat relapsed Graves' disease. What was unusual about this patient is that symptoms have occurred in a period less than 15 days of starting treatment and with a dose of 45 mg /day. The physician must educate the patient about the possibility of early onset of serious side effects of ATD and to seek medical advice as soon as possible.


Author(s):  
Aiswarya Mohan ◽  
Aravind H ◽  
Chakravarthy S Maddipati ◽  
Roshni P R

Antithyroid drugs (ATD’s) are widely used as the first line treatment option for the management of hyperthyroidism, especially for patients with Graves’ disease. They are classified into thionamide (Methimazole, Carbimazole and Propylthiouracil) and Non-thionamide (Iodine containing compounds) ATD’s. These drugs are associated with various types of adverse effects ranging from mild to potentially life threatening. Antithyroid arthritis syndome (AAS) is one of the major and uncommon side effects of ATD therapy requiring immediate drug discontinuation and hospitalization presents itself with myalgia, arthralgia and arthritis along with fever and rash of varying severity and non-specific laboratory findings, making its diagnosis and management clinically challenging. Here we report the case of 32 year old female with Graves’ disease who experienced severe migratory polyarthritis after the initiation of methimazole therapy. Her symptoms started to disappear after the prompt withdrawal of methimazole. We also concluded that this adverse effect of ATD’s might not be dose dependent by comparing our case with 6 other case reports of AAS. Here our objective is to raise awareness among the clinicians regarding the differential diagnosis and management of this major, uncommon and potentially life threatening adverse effect of ATD therapy.


10.2196/16664 ◽  
2020 ◽  
Vol 9 (4) ◽  
pp. e16664 ◽  
Author(s):  
Peter A Merkel ◽  
David R Jayne ◽  
Chao Wang ◽  
Jan Hillson ◽  
Pirow Bekker

Background Antineutrophil cytoplasmic antibody (ANCA)–associated vasculitis is a serious, often life-threatening disease. In new-onset disease or a relapse, the standard treatment is immunosuppressive therapy with glucocorticoids; these therapies are associated with substantial short- and long-term toxicity. Complement component 5a (C5a) binding to C5a receptor (C5aR) may play a central role in the pathogenesis of ANCA-associated vasculitis. Avacopan is a novel, orally bioavailable, and highly selective antagonist of human C5aR. Avacopan does not interfere with the production of C5b or the membrane attack complex (ie, terminal complement complex) and does not block C5a binding to a second receptor, C5L2 (also called C5aR2), shown to be protective in antimyeloperoxidase glomerulonephritis. This trial will evaluate if avacopan replaces the need for chronic glucocorticoids in the treatment of ANCA-associated vasculitis. Objective The aim of this study is to determine the proportions of patients in remission at week 26 and with sustained remission at week 52, defined as Birmingham Vasculitis Activity Score=0, and not taking glucocorticoids within the 4 weeks before week 26 and week 52, respectively. Methods The Avacopan Development in Vasculitis to Obtain Corticosteroid elimination and Therapeutic Efficacy study is a randomized, double-blind, active-comparator (prednisone), 2-arm study evaluating the safety and efficacy of avacopan versus prednisone, administered in combination with other immunosuppressive therapy. Eligible subjects will have active disease requiring induction of remission. Subjects are stratified based on the type of immunosuppressive therapy, ANCA subtype, and new or relapsing disease. Target sample size is 300 patients, enrolled at over 200 sites globally. All authors and local ethics committees approved the study design. All patients will provide informed consent. Results Enrollment of patients was completed in Q4 2018. Topline results are anticipated to be published by Q3 2020. Conclusions Results will be released irrespective of whether the findings are positive or negative. Trial Registration ClinicalTrials.gov NCT02994927; https://clinicaltrials.gov/ct2/show/NCT02994927 International Registered Report Identifier (IRRID) DERR1-10.2196/16664


2020 ◽  
Vol 7 (4) ◽  
pp. 707
Author(s):  
Avirup Majumdar ◽  
Virendra Atam ◽  
Saurabh Pandey ◽  
Prashant Singh ◽  
Himanshu Chauhan

Vasculitis is a process caused by inflammation of blood vessel walls and results in a variety of disorders.  Small-vessel vasculitis (vasculitis involving arteries, venules and capillaries) should be suspected in any patient who presents with a multisystem disease that is not caused by an infectious or malignant process. Testing for Antineutrophil cytoplasmic antibody (ANCA) is the basis of classification of small vessel vasculitis into ANCA associated and non - ANCA associated vasculitis. Apart from cutaneous manifestations like palpable purpura and vasculitic urticaria, digital gangrene in a patient with evidence of mononeuritis multiplex is highly suggestive of ANCA associated vasculitis (AAV). Clinically most of these vasculitides have overlapping clinical presentations and similar treatment. Early diagnosis and rapid initiation of treatment of AAV is recommended rather than ordering for definitive tests (e.g. histopathology or angiograms) since delay in treatment can result in serious end organ damage (pulmonary or renal).


Sign in / Sign up

Export Citation Format

Share Document