scholarly journals Anti-OJ (Anti-isoleucyl-tRNA Synthetase) Autoantibody- Positive Anti-synthetase Syndrome: a Case Report

Author(s):  
Ji-Na Gu ◽  
Wang Yan ◽  
Qiao-Ling Gao ◽  
Lin Chen

Abstract Background: Anti-OJ (anti-isoleucyl-tRNA synthetase) autoantibody-positive anti-synthetase syndrome(ASS) is a rare systemic autoimmune diseases that manifest as an inflammatory myopathy and interstitial lung diseas.Case presentation: We present a case of anti-OJ antibody-positive ASS with recurrent joint pain, fever, significantly elevated inflammatory markers, occult myositis but no interstitial pneumonia in a 75-year-old male patient. The patient was misdiagnosed for more than one year. Recurrent hepatocellular carcinoma (HCC) was confirmed after 1 year of the diagnosis of ASS, and the clinical symptoms were relieved after surgical resection. Conclusion: We report this rare case of anti-OJ antibody-positive ASS with atypical manifestations to raise awareness of the disease for clinicians.

2021 ◽  
Vol 19 (1) ◽  
Author(s):  
Cristina Morreale ◽  
Dario Bleidl ◽  
Angela Rita Sementa ◽  
Clara Malattia

Abstract Background Primary cutaneous mucinosis are a heterogeneous group of diseases characterized by the deposition of glycosaminoglycans in the dermis and the follicles. These diseases are rare in children therefore their diagnosis and management are still challenging. Joint involvement has been reported in patients with secondary cutaneous mucinosis and, rarely, in primary mucinosis. We describe a case of Cutaneous Mucinosis of Infancy with joint involvement. Case presentation An healthy 5-year-old boy showed acute arthritis of the left knee and left elbow confirmed by ultrasound. Laboratory tests were within normal range. Symptoms disappeared after a course of nonsteroid anti-inflammatory drugs. One year later, the knee swelling reappeared; juvenile idiopathic arthritis was diagnosed and intra-articular steroid injection was performed. Due to persistence of arthritis of the knee he was admitted to our hospital. On physical examination variable skin-colored lesions were observed, which had been in existence for over 2 years. We performed a skin biopsy that showed an interstitial mucine deposition in the reticular dermis. Cutaneous Mucinosis of Infancy was diagnosed. Discussion and conclusions Cutaneous Mucinosis of Infancy is a persistent dermatosis with benign prognosis and no treatment is generally required. Our case report is particularly interesting because it is the first in which joint involvement has been reported in CMI, a disorder that has so far been described as limited to skin involvement. Further studies will be necessary in order to clarify the pathogenesis of joint involvement in primary mucinosis.


2020 ◽  
Author(s):  
Jianing Tong ◽  
Jianmin Niu ◽  
Qiaoyun Li ◽  
Li Hu ◽  
Hui Zhang

Abstract Background: Malignant ovarian Brenner tumors are extremely rare worldwide, accounting for only 1% of malignant ovarian tumors. Their clinical symptoms and pathology are complex and erratic, and the images are mostly non-specific. This poses difficulties in preoperative diagnosis and distinguishing them from other cystic solid ovarian tumors. Here this study has reported on a case of bilateral malignant ovarian Brenner tumor with peritoneal effusion. Case presentation: A 54-year-old woman presented with intermittent vaginal bleeding for more than two months and abdominal pain for one month. Two hard mass of five centimeters with poor movement could be touched at each side of the ovarian areas. The CT findings indicated the presence of two large cystic and solid masses in both adnexal regions. The operation had watched the size of the masses in the bilateral ovarian was both 6cmx6cmx5cm and their surface ulceration showed rotten fleshy tissue. After the operation, combined with morphological and immunohistochemical features, the ovarian specimens were consistent with bilateral ovarian malignant Brenner tumors.Conclusions: Although the incidence of bilateral malignant OBT is extremely low and this disease is extremely rare clinically, the gynecologists should be more informed of its diagnosis and treatment.


2015 ◽  
Vol 2015 ◽  
pp. 1-4
Author(s):  
Leena Wadhwa ◽  
Sanjana N. Wadhwa ◽  
Sunita Jindal

Case Presentation. Mrs. X, 35 years old, case of primary infertility, was diagnosed to have genital tuberculosis on the basis of PCR positive and hysterolaparoscopy findings and received category I ATT for 6 months. Following ATT completion, her USG revealed no evidence of tuboovarian mass or hydrosalpinx. Since her tubes were patent, she underwent 3 cycles of ovulation induction and 2 cycles of IUI. The women presented with acute PID, five days after IUI, and was conservatively managed. She again presented 24 days after IUI with persistent low grade fever and abdominal pain. Suspecting relapse of genital tuberculosis, she was started on category II ATT. She had acute episodes of high grade fever with chills 2 weeks after starting ATT and MRI revealed bilateral TO masses suggestive of pyosalpinx. Emergency laparotomy was done, pus was drained, and cyst wall was removed and HPE was suggestive of chronic inflammation with few granulation tissues. ATT was continued for one year and the woman improved.Conclusion. The possibility of flare-up of PID (pelvic inflammatory disease) in treated case of tuberculosis undergoing infertility management should be kept in mind and aggressive management should be done.


2014 ◽  
Vol 2014 ◽  
pp. 1-3
Author(s):  
Osman Zikrullah Sahin ◽  
Teslime Ayaz ◽  
Suleyman Yuce ◽  
Fatih Sumer ◽  
Serap Baydur Sahin

Introduction. Acute renal failure (ARF) develops in 33% of the patients with rhabdomyolysis. The main etiologic factors are alcoholism, trauma, exercise overexertion, and drugs. In this report we present a rare case of ARF secondary to probably donepezil-induced rhabdomyolysis.Case Presentation. An 84-year-old male patient was admitted to the emergency department with a complaint of generalized weakness and reduced consciousness for two days. He had a history of Alzheimer’s disease for one year and he had taken donepezil 5 mg daily for two months. The patient’s physical examination revealed apathy, loss of cooperation, and decreased muscle strength. Laboratory studies revealed the following: urea: 128 mg/dL; Creatinine 6.06 mg/dL; creatine kinase: 3613 mg/dL. Donepezil was discontinued and the patient’s renal function tests improved gradually.Conclusion. Rhabdomyolysis-induced acute renal failure may develop secondary to donepezil therapy.


2019 ◽  
Vol 2019 ◽  
pp. 1-5
Author(s):  
Iqbal Multani ◽  
Snezana Popovic ◽  
Naveen Parasu ◽  
Michelle Ghert

Introduction. Mazabraud’s Syndrome is a rare condition that is defined by the presence of fibrous dysplasia lesions in the bone and intramuscular myxomas in the soft tissue. Malignant transformation, in the setting of Mazabraud’s Syndrome, of the fibrous dysplasia lesions into a sarcomatous neoplasm occurs in less than 1% of cases—with only six previously reported cases. Case Presentation. Here, we present a 62-year-old Caucasian female who developed an osteosarcoma in a fibrous dysplasia lesion of the proximal femur in the background of Mazabraud’s Syndrome. The patient was treated with wide excision and endoprosthetic reconstruction. She declined adjuvant chemotherapy. She is alive without evidence of disease one-year postoperatively. Conclusion. Patients with Mazabraud’s Syndrome remain at low risk for malignant transformation. However, close monitoring of asymptomatic patients with this condition for radiographic changes in their lesions and/or clinical symptoms is recommended.


2020 ◽  
Vol 8 (1) ◽  
pp. 18-18
Author(s):  
Somaye Rezaei ◽  
Fariba Zarzanalivan ◽  
Pouya Pirouti ◽  
Mohammad Reza Amiri Nikpour ◽  
Abdolreza Javadi ◽  
...  

Background: Isolated intracranial Rosai-Dorfman disease (RDD) is an extremely rare, idiopathic histo-proliferative disorder. RDD is associated with the proliferation of histiocytes and emperipolesis. Case Presentation: we report a case with isolated intracranial RDD. A 47-year- old man presented with a dizziness, falling, and then secondary generalized seizure, hemiparesis and right hemisensory deficit. This case preoperatively was misdiagnosed with meningioma. Histopathological examination revealed pale histiocytes displaying emperipolesis which were positive for S-100 and CD68 proteins and negative for CD1a marker. BRAF V600E mutation was negative. Conclusion: In this case, total resection was performed and clinical symptoms were regressed completely.


2020 ◽  
Vol 6 (3) ◽  
pp. 92-96
Author(s):  
Dodik Tugasworo ◽  
Aditya Kurnianto ◽  
Retnaningsih Retnaningsih ◽  
Yovita Andhitara ◽  
Rahmi Ardhini ◽  
...  

Background: Neuromyelitis optica (NMO) is a rare autoimmune disease that covers 20-30% of diseases related to autoimmune disorders and about 1% of demyelination diseases. NMO symptoms are vary between individuals, there are generally 2 main symptoms, transverse myelitis and optic neuritis. This article reported a child with NMO disease based on The Consensus of the International Panel for NMO Diagnosis (IPND) 2015.Case Presentation: An 8-year old boy with spastic tetraparesis, bilateral nervus opticus dysfunction, urinary retention, and allodynia et causa suspected NMO. Patient received therapy using high-dose intravenous methylprednisolone and showing a clinical improvement.Conclusion: This patient was diagnosed NMO based on IPND 2015 with an unknown AQP4-Ab status accompanied by supporting clinical symptoms. The management of NMO with high-dose intravenous methylprednisolone in this patient provides a meaningful response to the clinical improvement of the disease. 


2021 ◽  
Vol 21 (1) ◽  
Author(s):  
Weiping Tan ◽  
Bin Dong ◽  
Jincui Gu ◽  
Yang Peng ◽  
Ruicong Xue

Abstract Background Anti-Ku is a rare antibody which can be positive in some rheumatic diseases and it might be related to cardiac involvement. Polymyositis is an inflammatory myopathy, and its cardiac involvement seldom presents as myopericarditis and anti-Ku positive. Case presentation In this case, we report a mid-aged woman with chest pain, upper limbs weakness and fever unrelated with infection. The diagnosis of this case was unquestionably myopericarditis supported by ECG, cardiac MRI and negative findings in coronary arteries. Diagnosis of polymyositis was further clarified by the evidence of persistently increased CK, degeneration of proximal muscle in MRI, muscular dystrophy with lymphocytes infiltration in muscle biopsy. In the analysis of autoantibodies, we surprisingly discovered positive anti-Ku. Glucocorticoid and mycophenolate mofetil were then prescribed for polymyositis. Patient follow-up indicated remission of both myopericarditis and polymyositis. We finally clarified this rare case as a positive anti-Ku polymyositis with myopericarditis as cardiac involvement. Conclusion This report presents a rare case with anti-Ku positive polymyositis and the cardiac involvement of polymyositis was manifested as myopericarditis. Therefore, positive anti-Ku might explain the myopericarditis as cardiac involvement in polymyositis. More cases and longer duration of follow-up is required for the comprehensive understanding of the disease.


2019 ◽  
Vol 3 (3) ◽  

Introduction: Rabies is an invariably deadly zoonotic viral infection transmitted mostly from rabid dog’s bite. This infection is targeting CNS with no cure once clinical symptoms had occur. Typically, the incubation period range from 20 to 90 days. In this case, it was approximately 1 year, proven that atypically long period of rabies incubation period is possible. Case presentation: Presenting an atypically long incubation period of rabies, approximately one year post transmission. Conclusion: Possible long incubation period of rabies and suspected diverse viral behaviour affecting its incubation period. Thus, needs further investigation on how and what determinants underlie the condition.


2019 ◽  
Vol 12 (5) ◽  
pp. 349-352
Author(s):  
K Cheema ◽  
M Raad ◽  
R Sehjal ◽  
S Virani ◽  
J Relwani

We report a rare case of atypical Mycobacterium intracellulare infection encountered in a left sternoclavicular joint of an immunocompetent patient. The 34-year-old female patient presented with a one-year history of left sternoclavicular joint pain and swelling. The patient had multiple radiological investigations, which were suspicious for an infective cause. The patient had a biopsy of the joint, which returned showing acid-fast bacilli. The patient consequently received a prolonged course of medical treatment for M. intracellulare.


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