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Drulovic J.,University of Belgrade | Andrejevic S.,Institute of Allergy and Clinical Immunology | Bonaci-Nikolic B.,Institute of Allergy and Clinical Immunology | Mijailovic V.,Institute for Thyroid Gland and Metabolism Cigota
Vojnosanitetski Pregled

Background. Hashimoto's encephalopathy (HE) is a rare autoimmune syndrome characterized by various neuropsychiatric manifestations, responsive to steroid treatment and associated with Hashimoto's thyroiditis. There are only a few reports suggesting that intravenous immunoglobulins (IVIG) might represent an efficacious treatment modality for the severe steroid-resistant HE cases. We presented a patient with HE who developed a complete recovery after the IVIG therapy followed by a long-lasting remission. Case report. We described herien a female patient with the one-year history of autoimmune thyroiditis before the development of neuropsychiatric manifestations. In May 1999, a 38-year-old woman presented at the Institute of Neurology, Clinical Center of Serbia, Belgrade, with the brain-stem syndrome which responded well to steroid treatment. After detailed examinations, the diagnosis of Hashimoto's encephalopathy was established. Two years later, in June 2001, new manifestations (unsteadiness in gait, personality changes, seizures, and persistent headache) gradually developed during a 6 month period. Response to steroids was unsatisfactory and partial, since headaches and personality changes had continuously worsened. In January 2002, the patient received IVIG (0.4 g/kg body weight daily for 5 days). Gradual improvement was noticed and a complete recovery developed over the following weeks. Up to March 2009 during a 7-year follow-up period, remission persisted. Conclusion. To our best knowledge, this is the first report of a long-lasting remission of Hashimoto's encephalopathy after IVIG therapy. Therefore, this case further supports administration of IVIG, as a potentially beneficial treatment modality, in severe cases of Hashimoto's encephalopathy which are completely or partially resistant to steroids. Source

Kang H.-R.,Hanyang University | Kang H.-R.,Institute of Allergy and Clinical Immunology | Jee Y.K.,Dankook University | Kim Y.-S.,Hanyang University | And 16 more authors.
Pharmacogenetics and Genomics

Recent investigations suggest genetic susceptibility of allopurinol-induced severe cutaneous adverse reactions (SCARs). However, the strength of association was variable according to phenotypes and ethnic backgrounds. To explore genetic markers for allopurinol-induced SCARs in Koreans, we genotyped human leukocyte antigen (HLA) class I alleles of 25 cases of allopurinol-induced SCARs (20 cases of drug-induced hypersensitivity syndrome and five cases of Stevens-Johnson syndrome/toxic epidermal necrolysis) and 57 patients tolerant to allopurinol. Frequencies of B*5801 [92.0 vs. 10.5%, Pc=2. 45×10-11, odds ratio (OR)=97.8], Cw*0302 (92.0 vs. 12.3%, Pc=9.39×10-11, OR=82.1), and A*3303 (88.0 vs. 26.3%, Pc=3.31×10-11, OR=20.5) were significantly higher in SCARs compared with tolerant controls. In contrast, A*0201 was not found in SCARs patients despite relatively high frequency in tolerant controls (29.8%). We found strong positive association of HLA-B*5801 and negative association of HLA-A*0201 with the development of allopurinol-induced SCARs in the Korean population. © 2011 Wolters Kluwer Health | Lippincott Williams and Wilkins. Source

Andrejevic S.,Institute of Allergy and Clinical Immunology | Zlatanovic M.,Institute of Rheumatology | Damjanov N.,Institute of Rheumatology | Ivanovic B.,Institute for Cardiovascular Diseases | Tarabar D.,Military Medical Academy
Vojnosanitetski Pregled

Introduction. Watermelon stomach (WS) or gastric antral vascular ectasia (GAVE) is a rare cause of upper gastrointestinal bleeding described in a variety of autoimmune disorders. Association of watermelon stomach with Sjögren's syndrome is extremely rare. Case report. We presented a 67-year old female with primary Sjögren's syndrome (SS) who had developed a persistent severe iron-deficiency anemia. An upper gastric endoscopy revealed the presence of gastric antral vascular ectasia (GAVE) as a cause of occult gastrointestinal bleeding. The treatment with argon-plasma coagulation was postponed as the conservative therapy with iron substitution and proton pump inhibitor led to improvement of anemia and hemoglobin levels normalization. Conclusion. This is the first report of WS in a patient with primary SS without the presence of coexisting autoimmune disorder. Recognition of this rare, but clinically important, cause of gastrointestinal bleeding may decrease comorbidity in patients with autoimmune disorders including primary Sjögren's syndrome. Source

Moon I.J.,Sungkyunkwan University | Kim S.-W.,Seoul National University | Han D.H.,Seoul National University | Shin J.M.,Seoul National University | And 4 more authors.
American Journal of Rhinology and Allergy

Background: Although mucosal cysts in the paranasal sinuses (PSMCs) are commonly detected, the long-term follow-up studies of PSMCs are sparse. This study evaluated the natural course of PSMCs and identified risk factors for the disease progression. Methods: A total of 133 subjects with PSMCs who underwent health checkup including brain magnetic resonance imaging more than two times with an interval of ≥24 months between January 2000 and December 2009 were included. The characteristics of PSMCs were analyzed on the initial and follow-up images. Nasal symptoms, smoking status, and comorbid medical conditions were evaluated using structured questionnaires and medical records. Results: The mean follow-up duration was 40.38 months (range, 24.0-109.8 months). The mean size of PSMCs decreased from 15.07 to 12.73 mm. Only 8.3% of subjects showed an increase in size, whereas the size of cysts was decreased or unchanged in the remaining 91.7% of subjects. Six (4.5%) subjects complained of nasal symptoms during follow-up and subsequent sinusitis was developed in 3% of subjects. An increase in cyst size was associated with development of sinusitis (odds ratio = 45.375). Initial size of cysts >20 mm and bilateral location were significant risk factors for progression (p = 0.019 and p = 0.039, respectively). Conclusion: The majority of PSMCs in this follow-up study were decreased or unchanged and most subjects were asymptomatic. Just observation is enough for most PSMCs. However, those who have a large cyst (>20 mm) or bilateral cysts at initial diagnosis were at risk for disease progression and should be regularly followed. Copyright © 2011, OceanSide Publications, Inc. Source

Moon I.J.,Sungkyunkwan University | Lee J.-E.,Seoul National University | Kim S.-T.,Seoul National University | Han D.H.,Seoul National University | And 4 more authors.

Background: Though mucosal cysts in the paranasal sinuses (PSMCs) are common findings on radiographic images, the nature of PSMCs and risk factors for the development of PSMCs have not yet been determined. The aim of this study was to evaluate the characteristics of PSMCs using brain magnetic resonance (MR) imaging. Methodology/Principal: A total of 6831 subjects who underwent health checkup including brain MR imaging were included in this study. The characteristics of PSMCs, including their location, number and size, as well as the presence of obstruction of the sinus ostium and sinusitis, were analysed using brain MR images. Structured questionnaires and medical records were reviewed to evaluate the smoking status and comorbid medical conditions. Results: The overall prevalence of PSMCs was 7.4% and was significantly higher in females than in males. PSMCs were most commonly found in the maxillary sinus, most of which were located unilaterally as a solitary cyst. Large cysts were associated with obstruction of the sinus ostium and subsequent sinusitis. Smoking was a single important risk factor for developing PSMCs. No significant associations were found between symptoms (nasal/respiratory) and the presence of PSMCs. Conclusions: The prevalence of PSMCs was 7.4% and decreased with age. Large cysts may lead to obstruction of the sinus and subsequent sinusitis. Smoking was an important risk factor for PSMCs, and the total amount of smoking correlated with cyst size. Most subjects were asymptomatic, and specific treatment was not performed. Source

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