Kimura's disease was first described by Kimura and Sceto in China in 1937. Kimura disease is a chronic inflammatory disorder of idiopathic etiology. The clinical presentation of Kimura's disease is painless solitary or multiple subcutaneous nodules, asymmetric, mostly in the head and neck region with often association with lymphadenopathy. Typically, the nodules are found on preauricular, submandibular, and popliteal regions as well as oral cavity, larynx, and parotid glands. In the present report, we describe a case of a 27-year-old male presented to our hospital with history of right neck and lip swelling for 10 days. In the history, the patient mentioned that he had bloody diarrhea four to six times day and he lost 10 kg in 1 month. The specimen was sent for histopathological examination which showed the lymph node architecture is preserved with significant increase number of eosinophils which is consistent with Kimura's disease. There is no agreement on the management aspects in Kimura's disease so far. The primary treatment for Kimura's disease includes surgical resection. Additional medical therapy including regional or systemic steroid therapy, cytotoxic therapy, and radiation has also been utilized. Considered as an inflammatory process, the disease has an excellent prognosis, although it may recur locally and wax and wane over time.
During the past few decades, extensive researches were conducted to identify serological markers in patients with inflammatory bowel disease (IBD) that can reliably diagnose and monitor disease activity and help in predicting relapses. To date, several serological markers have been identified. This review will address the different serological markers and their clinical significance and applicability in medical practice. Serological markers include antibodies against microbial antigens, peptide antigens, autoantibodies, and basic inflammatory markers. Some serological markers such as anti-Saccharomyces cerevisiae antibodies (ASCA) and antibodies against exocrine pancreas (PAB) help the confirmation of the diagnosis of IBD to differentiate it from other non-IBD. Perinuclear anti-neutrophil cytoplasmic antibodies (pANCA) and ASCA can distinguish Chron’s disease and ulcerative colitis. Certain markers can aid stratification of Chron’s disease including antibodies to Pseudomonas fluorescens associated sequence I2 (Anti-I2), antibodies to bacterial flagellin (Anti-CBir1), ASCA, and antibodies to outer membrane porin C (Anti-OmpC). ASCA and pANCA can predict disease response to therapeutic agents (e.g. Infliximab). ASCA can also unaffected family members at risk of developing Chron’s disease.
Rationale:
Crohn's disease and ulcerative colitis are forms of inflammatory bowel disease (IBD) and are recognized causes of gastrointestinal diseases. Thromboembolism is known to be one of the extraintestinal complications of IBD. However, there have been no reports of a “thrombotic storm,” which is a rare form of thromboembolism, as the first manifestation of a case of previously unknown IBD. In this report, we describe a case of thrombotic storm as the initial presentation of IBD.
Patient concerns:
A 15-year-old girl with no known history of chronic medical illnesses presented to our clinic with complaints of infrequent loose bowel movements associated with occasional rectal bleeding 4 months prior.
Diagnoses:
Abdominal examination revealed tenderness upon superficial and deep palpation of the epigastrium and left lower quadrant of the abdomen. Digital rectal examination revealed no tenderness or masses in the rectal canal, but bloody stools were noted; imaging revealed extensive thrombosis. Sigmoidoscopy findings and histology were consistent with those of ulcerative colitis.
Interventions:
Anticoagulation therapy was initiated for the treatment of the thrombosis. Sigmoidoscopy and biopsy were also performed to ensure that an accurate diagnosis was made. The patient was placed on a therapy of methylprednisolone, mesalazine, and enoxaparin.
Outcomes: During follow-up, the patient's symptoms were found to have subsided, her test results had improved, and no further flare-ups of the ulcerative colitis were noted.
Lessons
: This case highlights the fact that thrombosis can precede IBD and can make the accurate diagnosis of such cases more challenging. The risk of thrombus propagation also poses a challenge in such cases.
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