Anti-neutrophil cytoplasmic antibody (ANCA) associated small-vessel vasculitis in a patient with diabetic nephropathy and autoimmune polyendocrinopathy syndrome (APS) Type 2: a case report

Murray, Jonathan; Baines, Laura; Pearce, Simon H. S.; Ball, Steve; Leech, Nicola; Wood, Katrina; Kanagasundaram, Nigel S.

doi:10.5414/cn107142

Cited by 3 publications

(2 citation statements)

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“…On the other hand, similar reports of vascular complications in PAS are much less common. A few case reports have recently found an association between PAS and specific vascular complications such as cutaneous vasculitis, small-vessel vasculitis, serositis with effusive pericarditis leading to cardiac tamponade and stroke, but these are rare and the mechanism by which these events occur has not been studied ( 11 , 12 , 13 , 14 ). To our knowledge, there are no specific reports of DVTs in patients with PAS.…”

Section: Discussionmentioning

confidence: 99%

Deep vein thrombosis, an unreported first manifestation of polyglandular autoimmune syndrome type III

Horsey

Hogan

Oliver

2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryA 71-year-old woman with severe right lower leg pain, edema and erythema was presented to the Emergency Department and was found to have an extensive deep vein thrombosis (DVT) confirmed by ultrasound. She underwent an extensive evaluation due to her prior history of malignancy and new hypercoagulable state, but no evidence of recurrent disease was detected. Further investigation revealed pernicious anemia (PA), confirmed by the presence of a macrocytic anemia (MCV=115.8fL/red cell, Hgb=9.0g/dL), decreased serum B12 levels (56pg/mL), with resultant increased methylmalonic acid (5303nmol/L) and hyperhomocysteinemia (131μmol/L), the presumed etiology of the DVT. The patient also suffered from autoimmune thyroid disease (AITD), and both antithyroglobulin and anti-intrinsic factor antibodies were detected. She responded briskly to anticoagulation with heparin and coumadin and treatment of PA with intramuscular vitamin B12 injections. Our case suggests that a DVT secondary to hyperhomocystenemia may represent the first sign of polyglandular autoimmune syndrome III-B (PAS III-B), defined as the coexistent autoimmune conditions AITD and PA. It is important to recognize this clinical entity, as patients may not only require acute treatment with vitamin B12 supplementation and prolonged anticoagulation, as in this patient, but may also harbor other autoimmune diseases.Learning pointsA DVT can be the first physical manifestation of a polyglandular autoimmune syndrome.Hyperhomocysteinemia secondary to pernicious anemia should be considered as an etiology of an unprovoked DVT in a euthyroid patient with autoimmune thyroid disease.Patients with DVT secondary to hyperhomocysteinemia should undergo screening for the presence of co-existent autoimmune diseases in addition to treatment with B12 supplementation and anticoagulation to prevent recurrent thromboembolism.

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Section: Discussionmentioning

confidence: 99%

Deep vein thrombosis, an unreported first manifestation of polyglandular autoimmune syndrome type III

Horsey

Hogan

Oliver

2016

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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“…Associations between autoimmune vasculitis and both isolated Hashimoto thyroiditis and APS have rarely been reported (6–8). Evaluation for diseases known to be associated with vasculitis, such as Sjogrens syndrome, sarcoidosis and systemic lupus erythematosis, was negative in our patient.…”

Section: Discussionmentioning

confidence: 99%

Acute mental status change as the presenting feature of adrenal insufficiency in a patient with autoimmune polyglandular syndrome type II and stroke

Watson

Raj

Eugster

et al. 2014

Journal of Pediatric Endocrinology and Metabolism

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Primary adrenal insufficiency (AI) in children usually presents with non-specific symptoms such as fatigue, nausea, vomiting, and anorexia. Here, we report an unusual case of a 15 year old girl who presented with acute mental status change and was ultimately diagnosed with AI due to autoimmune polyglandular syndrome type II (APS2). Central nervous system imaging revealed a cerebral infarction. To our knowledge, the constellation of APS2, stroke and acute mental status change has not been previously reported. We review the literature with regard to the presentation of AI as well as the association between vasculitis and APS2.

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MPO-ANCA-associated vasculitis in the context of autoimmune polyglandular syndrome type 3: case report and literature review

Dikovec

Wolters

Vossen

et al. 2022

Rheumatology Advances in Practice

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Anti-neutrophil cytoplasmic antibody (ANCA) associated small-vessel vasculitis in a patient with diabetic nephropathy and autoimmune polyendocrinopathy syndrome (APS) Type 2: a case report

Cited by 3 publications

References 0 publications

Deep vein thrombosis, an unreported first manifestation of polyglandular autoimmune syndrome type III

Deep vein thrombosis, an unreported first manifestation of polyglandular autoimmune syndrome type III

Acute mental status change as the presenting feature of adrenal insufficiency in a patient with autoimmune polyglandular syndrome type II and stroke

MPO-ANCA-associated vasculitis in the context of autoimmune polyglandular syndrome type 3: case report and literature review

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