Rare Presentation of Self-Limiting Kikuchi-Fujimoto Disease in Relapsing Nature.

BACKGROUND: Kikuchi-Fujimoto disease (KFD) is a rare, benign, and self-limiting disease that is commonly associated with cervical lymphadenopathy and fever. The disease has a wide spectrum of clinical manifestations, and definitive diagnosis is based on the histological appearance in the excision biopsy of the lymph nodes. Recurrence of KFD is reported rarely. Case Presentation. A 56-year-old Iranian woman with a background history of thrombocytopenia presented with fever, malaise, loss of appetite, and weight loss with cervical lymphadenopathy. The excision biopsy of the cervical lymph nodes confirmed the diagnosis of KFD, and she made a full recovery with improvement of symptoms, regression of cervical lymph nodes, and normalization inflammatory markers. One year after remission, she presented with similar clinico-biochemical profile, and repeat biopsy confirmed KFD. CONCLUSION: Although the rate of recurrence of the disease is very low, the treating physician should consider the possibility and confirm it histologically.

Young Male Presenting with an Acute Diarrheal Illness with Unexplained Transudative Ascites: An Atypical Presentation of Appendicular Tuberculosis.

INTRODUCTION: Appendicular tuberculosis is a rare form of extrapulmonary tuberculosis involving the gastrointestinal tract. Diagnosis of appendicular tuberculosis is difficult due to its atypical presentation. Histological confirmation remains the gold standard in diagnosis. Case Presentation. We report a 37-year-old Sri Lankan male presenting with a diarrheal illness with high fever for 8 days in the background of constitutional symptoms for 1-month duration. He was pale and had moderate amount of free fluid in the abdomen. Inflammatory markers were elevated, and CT abdomen revealed a thickened elongated appendix. Diagnostic paracentesis revealed a lymphocytic transudative ascites. A macroscopically minimally inflammed appendix removed at laparotomy and histology confirmed presence of tuberculous granulomata with caseation. He made an uneventful recovery by the anti-tuberculous therapy. CONCLUSION: High degree of suspicion is needed in diagnosis of appendicular tuberculosis due to its nonspecific presentation, and we emphasize the need of histological assessment of the appendix resected for the case of clinical appendicitis, as it may prompt the diagnosis of a rare but treatable case of tuberculosis.

Atypical case of hantavirus infection in Sri Lanka mimicking leptospirosis: a case report.

BACKGROUND: Hantavirus infection is an emerging zoonotic infection which has two characteristic patterns of presentation: hantavirus pulmonary syndrome and hemorrhagic fever with renal syndrome. The clinical presentation of hantavirus infection closely mimics leptospirosis. CASE PRESENTATION: This case report describes a previously apparently well 36-year-old Sri Lankan Sinhalese man who presented with an acute febrile illness with myalgia, with liver involvement in the form of transaminitis, cardiac involvement in the form of myocarditis, acute kidney injury, and pulmonary involvement. He was initially managed as severe leptospirosis with multiorgan dysfunction with antibiotics, steroids, and N-acetyl cysteine. A diagnosis of acute hantavirus infection was made subsequently. He made an uneventful recovery. CONCLUSION: Hantavirus infections need to considered in the differential diagnosis of patients presenting with acute febrile illness with multiorgan involvement. Larger studies are needed to evaluate the seroprevalence of hantavirus in Sri Lanka because it could be an emerging serious public health problem.

Infective myositis, an uncommon presentation of melioidosis: a case report and review of the literature.

BACKGROUND: Melioidosis is considered endemic in certain areas of the world. Musculoskeletal and soft tissue involvement are relatively uncommon presentations in melioidosis. We present a case of infective myositis in a patient with melioidosis in Sri Lanka, which is not considered an endemic country. Even though multiple cases of melioidosis have been reported with an increasing number in Sri Lanka, infective myositis secondary to melioidosis was not reported previously. CASE PRESENTATION: A 60-year-old Sinhalese man with diabetes presented with fever of 4 months' duration and a limp with a painful lump on the right side of the upper thigh of 2 months' duration. He had been treated in a local hospital for community-acquired pneumonia 3 weeks prior to this admission, for which he had received intravenous meropenem and teicoplanin with intensive care unit admission. He had a 0.5-cm × 0.5-cm tender lump over the right vastus lateralis muscle, and contrast-enhanced computed tomography of the area showed an ill-defined, heterogeneously enhancing, hypodense area involving the vastus lateralis, vastus intermedius, and quadratus femoris, suggestive of infective myositis but without abscess formation. Histopathology of the muscle biopsied from the vastus lateralis showed suppurative inflammation of subcutaneous fat with connective tissue necrosis and muscle infiltrated by lymphocytes. These features are suggestive of infective myositis possibly due to melioidosis. Although the result of a culture taken from the muscle biopsy was negative, the patient's antibody titer was strongly positive for melioidosis. He did not show any other areas with infected foci. He was treated with intravenous meropenem for 2 weeks and responded well. He was discharged with trimethoprim-sulfamethoxazole for 6 months as a maintenance therapy. CONCLUSION: Melioidosis is commonly an undiagnosed disease that has a wide variety of clinical presentations. Myositis in melioidosis is uncommon, and careful evaluation is mandatory to avoid misdiagnosis of this treatable but fatal disease. The clinician should have a high index of clinical suspicion, and further clinical and epidemiological studies are needed to determine the true burden of the disease.

Bartter syndrome-like phenotype in a patient with diabetes: a case report.

BACKGROUND: Bartter's syndrome is a rare genetic tubulopathy affecting the loop of Henle leading to salt wasting. It is commonly seen in utero or in early neonatal period. Rare cases of acquired Bartter's syndrome are reported in association with infections like tuberculosis, granulomatous conditions like sarcoidosis, autoimmune diseases, and drugs. The mainstay of management includes potassium, calcium, and magnesium supplementation. CASE PRESENTATION: We report the case of a 62-year-old Sri Lankan Sinhalese man with diabetes and hypertension presenting with generalized weakness with clinical evidence of proximal myopathy. He was severely hypokalemic with high urinary potassium excretion and hypochloremic metabolic alkalosis. He poorly responded to intravenously administered potassium supplements. A diagnosis of idiopathic Bartter-like phenotype was made. He responded well to spironolactone and indomethacin. CONCLUSIONS: Patients presenting with body weakness need serum potassium estimation. Acquired Bartter's syndrome although rare, should be ruled out in those with hypokalemia and metabolic alkalosis with increased urinary potassium loss with poor response to potassium replacement.

Lessons learnt from managing a case of dengue hemorrhagic fever complicated with acute liver failure and acute kidney injury: a case report.

BACKGROUND: Dengue is a common arboviral infection with a diverse spectrum of clinical manifestations. Dengue hemorrhagic fever is a more severe form of infection characterized by plasma leak and hemoconcentration. Although hepatic dysfunction is common in dengue illness, massive liver necrosis is rarely reported. Lactic acidosis is a poor prognostic marker in liver failure related to dengue. Management of acute renal injury in dengue hemorrhagic fever due to prolonged shock is challenging as the fluid reabsorption during the recovery phase expands the intravascular volume and precipitates heart failure and pulmonary edema. CASE PRESENTATION: We report the case of a 43-year-old Sri Lankan Sinhalese woman with serologically confirmed dengue fever presenting with evidence of plasma leakage developing acute liver failure evidenced by deranged liver functions, coagulopathy, and altered sensorium and acute kidney injury with anuria. She had elevated serum lactate levels. In addition to the "standard care," she was managed with intravenously administered N-acetyl cysteine and blood transfusions, even in the absence of bleeding or dropping packed cell volume, targeting a higher packed cell volume anticipating a better oxygenation at tissue level. Continuous veno-venous hemodialysis was employed and continued for 138 hours removing the fluids reabsorbed during the recovery phase to prevent her from developing heart failure and pulmonary edema. She made full recovery with no sequelae. CONCLUSIONS: N-acetyl cysteine and packed cell transfusion aiming at a higher packed cell volume to maintain adequate tissue perfusion during shock may be beneficial in acute liver failure due to dengue virus. The use of a continuous form of renal replacement such as continuous veno-venous hemodialysis is of paramount importance in managing fluid states in the recovery phase of dengue hemorrhagic fever in those with renal impairment. Interesting observations made in the fluid dynamics during the reabsorption phase need further studies preferably with an animal model.

Delayed presentation of severe rhabdomyolysis leading to acute kidney injury following atorvastatin-gemfibrozil combination therapy: a case report.

BACKGROUND: Rhabdomyolysis is a rare but serious complication of lipid-lowering therapy. Statin and fibrate combination increases the risk of rhabdomyolysis possibly by pharmacodynamic interactions. Advanced age, diabetes, hypothyroidism, polypharmacy, and renal impairment are known to increase the risk of rhabdomyolysis. Management strategies include fluid resuscitation and urine alkalinization. Renal indications such as refractory hyperkalemia, acidosis, fluid overload, or uremic complications mandate renal replacement therapy in rhabdomyolysis. CASE PRESENTATION: We report the case of a 62-year-old Sri Lankan Sinhalese man with dyslipidemia, type 2 diabetes mellitus with renal impairment, and hypothyroidism who was on atorvastatin; he was started on gemfibrozil and developed muscle symptoms. Although gemfibrozil was discontinued soon after, he presented with rhabdomyolysis with acute kidney injury 1 month later. He needed hemodialysis due to refractory hyperkalemia, metabolic acidosis, and fluid overload. CONCLUSIONS: Rhabdomyolysis is a rare but serious complication due to lipid-lowering therapy with statins and fibrates. Treating physicians should be aware and patients should be warned to report about muscle symptoms after starting statins or fibrates. Rhabdomyolysis may occur with mild symptoms and signs and may occur later, even after discontinuation of the drug.

Fever with pancytopenia: unusual presentation of extrapulmonary tuberculosis: a case report.

BACKGROUND: Tuberculosis is a major health problem in the developing world. Diagnosis of extrapulmonary tuberculosis is delayed because the presentation is nonspecific. Extrapulmonary tuberculosis can present with various hematological manifestations, including pancytopenia. Pancytopenia could be due to hypersplenism, maturation arrest, hemophagocytic lymphohistiocytosis, or infiltration of the bone marrow by caseating or noncaseating granulomas causing reversible or irreversible fibrosis. CASE PRESENTATION: We report a case of a 56-year-old Sri Lankan Sinhalese man who presented with pyrexia of known origin with significant loss of weight and loss of appetite. He had mild pallor with mild hepatosplenomegaly. He had high inflammatory markers with pancytopenia in a peripheral blood smear. His chest radiograph was unremarkable, and he had a negative Mantoux test result. A diagnosis of disseminated tuberculosis was made on the basis of caseating tuberculous granulomas in the bone marrow. CONCLUSIONS: Disseminated tuberculosis remains a diagnostic challenge because the presentation is vague and nonspecific. In case of pyrexia of unknown origin with peripheral cytopenia, the possibility of disseminated tuberculosis should be considered, particularly in endemic areas. Simultaneous culture and histopathological examination of the bone marrow is important in such instances, because results of common tests such as chest radiography or Mantoux tests can be negative.

Asymptomatic thyroiditis presenting as pyrexia of unknown origin: a case report.

BACKGROUND: Pyrexia of unknown origin is a difficult and challenging problem for the physician. Endocrine disorders, such as subacute thyroiditis, rarely present with pyrexia of unknown origin. Subacute thyroiditis can have a broad spectrum of clinical presentations including fever and biochemical thyrotoxicosis without overt signs or symptoms. CASE PRESENTATION: A previously healthy 42-year-old Sri Lankan Sinhalese man was extensively investigated for a prolonged fever of 3 weeks with high inflammatory markers. He had mild tenderness over his neck with cervical lymphadenopathy with no thyrotoxic symptoms or signs. An ultrasound scan revealed an enlarged thyroid with increased vascularity and he had suppressed thyroid-stimulating hormone with elevated free thyroxine and free triiodothyronine hormone levels. Fine-needle aspiration cytology confirmed thyroiditis. He responded well to low-dose steroids. CONCLUSION: Subacute thyroiditis should be considered in the diagnostic workup of pyrexia of unknown origin even in the absence of overt toxic symptoms of thyroid hormone excess.

Fever with lymphadenopathy - Kikuchi Fujimoto disease, a great masquerader: a case report.

BACKGROUND: Kikuchi Fujimoto disease is an uncommon benign condition of necrotizing histiocytic lymphadenitis commonly seen in East Asian and Japanese populations. It commonly presents with fever, cervical lymphadenopathy, and elevated inflammatory markers. Diagnosis of Kikuchi Fujimoto disease is based on histopathological studies of the involved lymph nodes. The presentation of Kikuchi Fujimoto disease can mimic many sinister conditions including lymphoma. Treatment is mainly supportive provided that accurate diagnosis is made and sinister conditions like lymphoma ruled out. CASE PRESENTATION: We report the case of an 18-year-old Sri Lankan Moor woman who presented with fever and cervical lymphadenopathy for 1 month. She had elevated inflammatory markers with high lactate dehydrogenase and ferritin levels. She had an extensive work-up including an excision biopsy of an involved lymph node and bone marrow biopsy. Finally, a diagnosis of Kikuchi Fujimoto disease was based on histopathology of the lymph node and negative bone marrow biopsy. CONCLUSIONS: Although Kikuchi Fujimoto disease is a self-limiting condition, it is a great masquerader which mimics the clinical features of many sinister conditions including tuberculosis, lymphoma, and adult-onset Still's disease. Early recognition of the disease is of crucial importance in minimizing potentially harmful and unnecessary evaluations and treatments.

Dengue hemorrhagic fever complicated with acute liver failure: a case report.

BACKGROUND: Dengue is a common arboviral infection with a clinically diverse spectrum of presentations. Although hepatic dysfunction is commonly identified in patients will dengue illness, acute liver failure is rare. The etiopathogenesis of hepatic dysfunction is multifactorial and related to direct viral invasion of hepatocytes, immunological factors and hypoxia particularly in cases of shock in dengue hemorrhagic fever. Ideal management of dengue-related hepatic dysfunction and acute liver failure is still debated. CASE PRESENTATION: We report a 53-year-old Sri Lankan Sinhalese male with serologically confirmed dengue fever presenting with evidence of plasma leakage developing acute liver failure evidenced by deranged liver functions, coagulopathy and altered sensorium. In addition to the 'standard care', the patient was managed with intravenous N-acetyl cysteine and blood transfusions even in the absence of bleeding or dropping packed cell volume (PCV), targeting a higher PCV in anticipation of better oxygenation at tissue level. He made a full recovery with no sequential infections. CONCLUSION: N-acetyl cysteine and packed cell transfusion aiming at a higher PCV to maintain adequate tissue perfusion during shock may be beneficial in acute liver failure due to dengue virus. Large randomized trials should be carried out to establish the efficacy of these treatment strategies to support these observations and change the current practice.

Acromegaly presenting with low insulin-like growth factor-1 levels and diabetes: a case report.

INTRODUCTION: Acromegaly is an endocrine disorder arising from excessive serum growth hormone levels in adulthood and is characterized by progressive somatic enlargement. Biochemical confirmation is achieved by demonstration of elevated baseline serum growth hormone levels which are not suppressed during an oral glucose tolerance test, and by increased levels of serum insulin-like growth factor-1. The serum insulin-like growth factor-1 level provides an assessment of integrated growth hormone secretion and is recommended for diagnosis, monitoring, and screening of acromegaly. We report a case of a patient with acromegaly secondary to a pituitary microadenoma who presented with low insulin-like growth factor-1. CASE PRESENTATION: An 83-year-old Sinhalese woman presented to our hospital with an enlarging multinodular goiter. She was observed to have macroglossia, thickened coarse skin, acral enlargement, and newly detected, uncontrolled diabetes. A diagnosis of acromegaly was suspected. She did not complain of recent headaches, vomiting, visual difficulties, or galactorrhea and was clinically euthyroid. Her pulse rate was 84 beats/min, and her blood pressure was 150/90 mmHg. A visual field assessment did not reveal a defect. Her random growth hormone levels were 149 mU/L (<10 mU/L), and her oral glucose tolerance test was supportive of acromegaly with a paradoxical rise of growth hormone. Her serum age-specific insulin-like growth factor-1 level was below normal at 124.7 ng/ml (normal range 150-350 ng/ml). Her serum insulin-like growth factor-1 level, measured after glycemic control was achieved with metformin and insulin, was elevated, which is characteristic of acromegaly. Magnetic resonance imaging of her pituitary revealed a pituitary microadenoma. Acromegaly secondary to a growth hormone-secreting pituitary microadenoma was confirmed. CONCLUSIONS: Systemic illnesses, including catabolic states, hepatic or renal failure, malnutrition, and diabetes mellitus, are known to decrease insulin-like growth factor-1 levels and may result in false-negative values in patients with acromegaly A low insulin-like growth factor-1 level does not exclude acromegaly in a patient with supportive clinical features and poorly controlled diabetes.