A case of eosinophilic esophagitis with autoimmune polyendocrine syndrome type 2, including autoimmune gastritis

2021 ◽  
Author(s):  
Maki Ayaki ◽  
Noriaki Manabe ◽  
Minoru Fujita ◽  
Jun Nakamura ◽  
Aya Sunago ◽  
...  
Keyword(s):  
Eosinophilic Esophagitis ◽  
Autoimmune Gastritis ◽  
Syndrome Type ◽  
Autoimmune Polyendocrine Syndrome

scholarly journals Bilateral Tibial Proximal Fractures Caused by Secondary Osteomalacia due to Autoimmune Polyendocrine Syndrome Type 2: A Case Report

2021 ◽  
Vol 11 (4) ◽  
Author(s):  
Daisuke Nakagawa ◽  
Keisuke Oe ◽  
Tomoaki Fukui ◽  
Ryosuke Kuroda ◽  
Takahiro Niikura
Keyword(s):  
Case Report ◽  
Pharmacological Treatment ◽  
Plate Osteosynthesis ◽  
Deformity Correction ◽  
Serum Phosphate ◽  
Tibial Osteotomy ◽  
Syndrome Type ◽  
Radiographic Findings ◽  
Autoimmune Polyendocrine Syndrome

Introduction: Hypophosphatemic osteomalacia can be overlooked or confused with other musculoskeletal disorders due to the variety of associated clinical, laboratory, and radiographic findings. If osteomalacia is diagnosed early and the fractures are not displaced, they often heal with nutritional supplements, but, if they progress to displaced fractures, they may require surgical intervention. Case Report: We present a case of secondary osteomalacia due to autoimmune polyendocrine syndrome Type 2 due to this condition, the patient developed bilateral tibial proximal fractures and her varus deformity had progressed. No clear indication of the timing for surgery for adults with osteomalacia has been reported. However, medical treatment improves the symptoms of osteomalacia and it is reported that in children, appropriate level of the serum phosphate (P) should be attained and maintained for the successful bone healing after osteotomy. Therefore, we prioritized pharmacological treatment and prescribed surgery after confirming that the value of serum phosphate P had been improved to recommended levels (2.5-3.5 mg/dl). We performed high tibial osteotomy for the right side and gradual correction by an external fixation for the left tibia, because of more severe deformation, and converted to an internal fixation to shorten the treatment period. During conversion, we performed the operation with a locking plate by the minimal invasive plate osteosynthesis method (MIPO). Conclusion: We conclude that the use of different deformity correction methods, depending on the degree of deformity, and the pharmacological treatment of osteomalacia may lead to favorable results. Keywords: Osteomalacia, autoimmune polyendocrine syndrome type 2, deformity correction method.

scholarly journals Petrified pinna and pericarditis in autoimmune polyendocrine syndrome

2019 ◽  
Vol 12 (6) ◽  
pp. e229369
Author(s):  
Yub Raj Sedhai ◽  
Soney Basnyat
Keyword(s):  
Diabetes Mellitus ◽  
Adrenal Insufficiency ◽  
Clinical Entity ◽  
Syndrome Type ◽  
Auricular Cartilage ◽  
Acute Pericarditis ◽  
Autoimmune Polyendocrine Syndrome ◽  
Local Trauma

Petrified pinna refers to the calcification or ossification of the external auricular cartilage. It is an uncommon clinical entity and is most often associated with local trauma, frostbite or inflammation. Auricular calcification may be the exclusive cutaneous marker of underlying endocrinopathy. It has been most commonly associated with adrenal insufficiency and other endocrine conditions like diabetes mellitus, hypothyroidism and acromegaly. We present a 47-year-old Caucasian manwho presented with acute pericarditis with tamponade physiology, who was found to have petrified pinnae as a telltale sign of the underlying autoimmune polyendocrine syndrome type 2.

ICPis-Induced Autoimmune Polyendocrine Syndrome Type 2: A Review of the Literature and a Protocol for Optimal Management

2020 ◽  
Vol 105 (12) ◽  
pp. e4208-e4218 ◽  
Author(s):  
Yun Shi ◽  
Min Shen ◽  
Xuqin Zheng ◽  
Yang Chen ◽  
RuiLing Zhao ◽  
...  
Keyword(s):  
Adverse Events ◽  
Checkpoint Inhibitors ◽  
Evidence Synthesis ◽  
European Medicines Agency ◽  
Adrenal Crisis ◽  
Close Monitoring ◽  
Syndrome Type ◽  
Autoimmune Polyendocrine Syndrome ◽  
Central Registry

Abstract Context Immune checkpoint inhibitors (ICPis) targeting cytotoxic T-lymphocyte antigen 4 (CTLA-4), programmed cell death protein 1 (PD-1), and its ligand (PD-L1) are now approved to treat a variety of cancers. However, ICPis therapy is associated with a risk of immune-related adverse events (irAEs). Autoimmune polyendocrine syndrome type 2 (APS-2) is a rare endocrine irAE. Evidence Acquisition Several databases (PubMed, Web of Science, Cochrane Central Registry of Controlled Trials, ClinicalTrials.gov, and Scopus) were searched up to February 18, 2020, for case reports on endocrine irAEs and ICPis. The reported side effects and adverse events of the ICPis therapy in the US Food and Drug Administration (FDA) and European Medicines Agency (EMA) adverse events pharmacovigilance registries are also included. Evidence Synthesis Here, we provide an overview of all published and reported cases (n = 30) of ICPis-induced APS-2. We summarize the clinical characteristics, autoantibodies, human leukocyte antigen (HLA) genotypes, and therapies and propose an APS-2 screening strategy. Conclusions Given the life-threatening risks of endocrine dysfunction if it is not promptly recognized (such as diabetic ketoacidosis and acute adrenal crisis), physicians (especially endocrinologists and oncologists) should be familiar with APS-2. After diagnosis of an autoimmune disease induced by ICPis (especially PD-1 inhibitors), patients with a high-risk HLA allele (HLA-DR4) require close monitoring for the development of APS-2.

scholarly journals Case report on peripartum cardiomyopathy in a patient with Schmidt syndrome with twin pregnancy for emergency lower segment cesarean section

2021 ◽  
Vol 10 (7) ◽  
pp. 2858
Author(s):  
Lakshmi Ramanathan ◽  
Shibli Khalilullah ◽  
Gisha V. Mathew ◽  
Lamtai Zahir ◽  
Korichi Noureddine
Keyword(s):  
Cesarean Section ◽  
Twin Pregnancy ◽  
Peripartum Cardiomyopathy ◽  
Thyroid Disorder ◽  
Syndrome Type ◽  
Lower Segment ◽  
Autoimmune Thyroid ◽  
Lower Segment Cesarean Section ◽  
Autoimmune Polyendocrine Syndrome

Peripartum and autoimmune cardiomyopathy is an uncommon rare disorder associated with pregnancy. When it occurs association with autoimmune thyroid disorder and autoimmune adrenal insufficiency, it is eponymously referred to as Schmidt syndrome or autoimmune polyendocrine syndrome type 2 (APS type 2). Peripartum cardiomyopathy (PPCM) can be difficult to diagnose as the symptoms can be masked or misinterpreted due to the normal physiological changes during pregnancy, as the symptoms of heart failure can mimic those of pregnancy. PPCM is associated with considerable morbidity and mortality and so should not be underestimated. In this report, we are discussing the management of 32-years-old female with hypothyroidism and Addison’s disease (polyglandular syndrome type 2- Schmidt syndrome) who came for emergency lower segment cesarean section (LSCS) due to twin pregnancy (abnormal doppler of the second twin) and during the period developed pulmonary edema and was diagnosed as peripartum cardiomyopathy. 

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