Fatal hemolytic anemia associated with metformin: A case report

Introduction

Metformin is a widely prescribed biguanide antidiabetic drug that has been implicated as a cause of hemolytic anemia in three previous case reports. We report a case of rapidly fatal hemolysis that was temporally associated with the initiation of metformin treatment for diabetes. Clinicians need to be aware of this rare but potentially serious side effect of metformin.

Case presentation

A 56-year-old Caucasian man with type 2 diabetes mellitus was started on metformin to improve glycemic control. Shortly afterwards, he developed progressive fatigue, exertional dyspnea, cranberry-colored urine and jaundice. Laboratory studies showed severe hemolysis, with a drop in hemoglobin from 14.7 to 6.6 g/dl over 4 days, markedly elevated lactate dehydrogenase, bilirubin and reticulocyte counts, and a low haptoglobin level. A peripheral blood smear showed no schistocytes, and a direct Coombs test was positive for anti-IgG and negative for anti-C3. Despite corticosteroid treatment and transfusion of packed red blood cells, the patient developed increasing dyspnea, hypotension, further decline in hemoglobin to 3.3 g/dl, and fatal cardiorespiratory arrest 12 hours after admission.

Conclusion

The serologic findings in this case suggest an autoimmune hemolytic anemia, caused either by a drug-induced autoantibody or a warm autoantibody. Based on the temporal association with metformin and the lack of other clear precipitating causes, we propose that metformin-induced hemolysis with a drug-induced autoantibody is a strong possibility. This mechanism differs from a previously described case with a possible antibody to the erythrocyte-drug complex. It has been shown, however, that hemolysis may occur via multiple mechanisms from the same drug. Clinicians should consider the possibility of metformin-associated immune hemolytic anemia in patients with otherwise unexplained hemolysis.

     

Severe airway stenosis associated with Crohn's disease: Case report

Background

Although it is recognised that smoking is a major risk factor for subjects with chronic obstructive pulmonary disease and is associated with respiratory symptoms, there is less agreement concerning the relationship between asthma and smoking. This study aims to examine the relationship between cigarette smoking and asthma prevalence.

Method

Data were used from two postal questionnaire surveys (1999 and 2001) in two general practice populations, using a respiratory questionnaire based on the ECRHQ and a generic quality of life questionnaire (EQ-5D). Only subjects less than 45 years old were included in the survey. An empirical definition of likely asthma was used based on respiratory questionnaire responses. Smoking was examined according to three categories, current smoker, ex smoker and never smoker.

Results

Almost 3500 subjects were included in the analyses. Current smokers had a higher prevalence of likely asthma compared to never smokers, odds ratio (OR) 1.59 (95% confidence interval (CI) 1.24 to 2.04). and also compared to ex smokers OR 1.79 (CI 1.25 to 2.56), but there was no difference between ex smokers and never smokers (OR 1.00 (0.75–1.35)). Current smoking was also positively associated with all symptoms but not with a history of hayfever/eczema.

Conclusion

Although the positive association found between current smoking and obstructive airways disease is likely to be due to the effect of cigarettes on asthma, it could reflect an association with early COPD (GOLD stages 0 or 1). Smoking cessation has a beneficial effect on the prevalence of respiratory symptoms and is therefore of paramount importance among these young adults.     

Anaphylactic reaction associated with Ranitidine in a patient with acute pancreatitis: a case report

Sustained chronic tachyarrhythmias often cause a deterioration of cardiac function known as tachycardia-induced cardiomyopathy or tachycardiomyopathy. The incidence of tachycardia-induced cardiomyopathy is unknown, but in selected studies of patients with atrial fibrillation, approximately 25% to 50% of those with left ventricular dysfunction had some degree of tachycardia-induced cardiomyopathy. It is an important clinical entity due to the high incidence and potential reversibility of the disease process. This case describes a cardiomyopathy induced by excess caffeine consumption. Six months following withdrawal of caffeine from the subject's diet, full resolution of symptoms occurred.     

A classical phenotype of Anderson-Fabry disease in a female patient with intronic mutations of the GLA gene: a case report

Background

Infective Endocarditis (IE) is considered as a multifaceted problem in every aspect from etiology and presentation to diagnosis and management. Early recognition of this disease and especially its complications, remain a critical task for the cardiologist. Atrial endocarditis is a rare and sometimes unrecognized complication of mitral valve endocarditis.

Case presentation

We present a 48 year-old male patient who was admitted to our clinic because of recent onset of malaise, fever, jaundice and peripheral edema. Important physical findings were peripheral stigmata of IE in addition to holosystolic murmur over the left sternal border. Transthoracic and transesophageal echocardiophy revealed a severe eccentric MR due to a flailed posterior mitral valve caused by IE. The presence of atrial septal endocarditis caused by jet streaming was also observed. Blood culture was positive for streptococcus oralis and antibiotic therapy was immediately initiated. Considering the large burden of infective tissue, the patient was planned for an early surgical intervention. A minimally invasive resection of the atrial mass, direct closure of the defect, resection of the diseased portions of mitral leaflets and implantation of a biological mitral valve prosthesis was performed. Intra-operative and histological findings confirmed provisional diagnosis by echocardiography.

Conclusions

Together with comprehensive echocardiographic evaluation, attention should be placed on mural vegetations and excluded among all cases of mitral valve endocarditis, particularly those with severe eccentric regurgitant jets.     

Delayed puberty in an 18-year-old female patient with late diagnosis of celiac disease

A case of a 18-year female patient is presented. The patient suffered from certain symptoms of malabsorption syndrome. She was also retarded sexually. Celiac disease was diagnosed according to ESPGAN criteria. Gluten -free diet produced body weight increase, pain relief, improved well-being and normal menstruation.     

Erdheim Chester disease in a patient with Burkitt lymphoma: a case report and review of literature

Background

Erdheim Chester disease (ECD) is a rare, non-Langerhans cell histiocytosis characterized by widespread tissue infiltration by CD68-positive, CD1a-negative foamy histiocytes. ECD can be difficult to identify, and diagnosis relies on the presence of histiocytes with certain histologic and immunophenotypic features in an appropriate clinical and radiologic setting. Clinical signs and symptoms are variable depending on which organ systems are involved. Most patients have at least skeletal involvement with bone pain as well as fatigue. Other common manifestations include diabetes insipidus, cardiac, periaortic, or retro-orbital infiltration/fibrosis, kidney impairment, xanthelasmas, among others.

Case presentation

Herein, we describe a case of BRAF-mutation positive ECD in a patient with Burkitt lymphoma, and we review recent literature.

Conclusion

Underlying BRAF and other MAPK pathway mutations are identified in approximately 50% of cases of ECD, which aids in diagnosis as well as enables novel targeted treatments. ECD patients have an increased risk of myeloid neoplasms; however, unlike other histiocytoses, an association with lymphoproliferative disorders has not been recognized.

     

Toxic epidermal necrolysis associated with radiotherapy and phenytoin in a patient with non-Hodking's lymphoma: A case report

Toxic epidermal necrolysis (TEN) is a disease which is characterized by fever and desquamation of the skin and mucosal membranes. It is usually related with drugs, especially aromatic anticonvulsants which are recognized as the most common cause of this disorder. Cranial irradiation may act as a precipitating factor along with anticonvulsants for the development of TEN. We report a 28-year-old patient with central nervous system (CNS) relapsed non-Hodgkin lymphoma (NHL) who developed TEN after cranial radiotherapy and concurrent phenytoin treatment.     

鹦鹉热衣原体感染致复视1例

鹦鹉热衣原体是一种胞内寄生的革兰阴性菌,可引发人畜共患病。鹦鹉热衣原体感染多表现为呼吸道症状,在中枢神经系统受累时通常表现为脑膜炎、颅神经麻痹、吉兰巴雷综合征以及癫痫。本文介绍了1例鹦鹉热衣原体致重症肺炎且合并颅神经损害的病例,经过多西环素抗感染治疗后肺部感染及颅神经病变好转。该病例报告不仅帮助临床医师进一步熟悉鹦鹉热衣原体感染累及中枢神经系统的临床表现,更有助于其了解鹦鹉热病原体目前的诊断手段。     

Eosinophilia in a patient with cyclical vomiting: a case report

BACKGROUND: Eosinophilic gastritis is related to eosinophilic gastroenteritis, varying only in regards to the extent of disease and small bowel involvement. Common symptoms reported are similar to our patient's including: abdominal pain, epigastric pain, anorexia, bloating, weight loss, diarrhea, ankle edema, dysphagia, melaena and postprandial nausea and vomiting. Microscopic features of eosinophilic infiltration usually occur in the lamina propria or submucosa with perivascular aggregates. The disease is likely mediated by eosinophils activated by various cytokines and chemokines. Therapy centers around the use of immunosuppressive agents and dietary therapy if food allergy is a factor. CASE PRESENTATION: The patient is a 31 year old Caucasian female with a past medical history significant for ulcerative colitis. She presented with recurrent bouts of vomiting, abdominal pain and chest discomfort of 11 months duration. The bouts of vomiting had been reoccurring every 7-10 days, with each episode lasting for 1-3 days. This was associated with extreme weakness and cachexia. Gastric biopsies revealed intense eosinophilic infiltration. The patient responded to glucocorticoids and azathioprine. The differential diagnosis and molecular pathogenesis of eosinophilic gastritis as well as the molecular effects of glucocorticoids in eosinophilic disorders are discussed. CONCLUSIONS: The patient responded to a combination of glucocorticosteroids and azathioprine with decreased eosinophilia and symptoms. It is likely that eosinophil-active cytokines such as interleukin-3 (IL-3), granulocyte macrophage colony stimulating factor (GM-CSF) and IL-5 play pivotal roles in this disease. Chemokines such as eotaxin may be involved in eosinophil recruitment. These mediators are downregulated or inhibited by the use of immunosuppressive medications.     

Aggressive Burkitt-like lymphoma of colon in a patient with prior celiac disease

Background: Celiac disease (CD) and immunosuppression are the two risk factors for gastrointestinal, as well as non-gastrointestinal, non-Hodgkin’s lymphomas (NHL). Recent large retrospective studies confirm that celiac disease significantly increases risk of developing small bowel lymphomas by 30 to 40 percent and other gut malignancies by 83-fold. Case Report: A 75-year-old man with a history of CD of two-year duration presented with pallor, fatigue, and 20-pound weight loss of three weeks duration. There was a vague non-tender mass in the right hypochondrium, and his stools tested positive for occult blood. The lab values were within normal range, except for hemoglobin of 11mg/dL, MCV 75, mildly elevated SGOT of 61 IU/L, and LDH of about 5000 IU/L. Work-up including computerized tomography (CT) scan, positron emission tomography (PET) scan, and colonoscopy were performed. Results: A CT scan of the abdomen showed extensive carcinomatosis, scattered lymphadenopathy, and small pleural effusions. PET scan results coincided with CT findings. Colonoscopy revealed a friable nodular mass in the hepatic flexure, histopathology of which confirmed a high-grade B-cell lymphoma. Flow cytometry following immunostaining was positive for CD10, CD19, CD20, CD45, CD79a, and Ki-67. FISH assay demonstrated t (14:18) translocation and bcl-2 rearrangement. The bone marrow biopsy showed evidence of disease. The patient was treated with rituximab, plus cyclophosphamide, Adriamycin, vincristine, and prednisone (CHOP-R), with intrathecal methotrexate prophylaxis. Currently, the patient remains in remission. Conclusion: This is the first case of aggressive Burkitt-like lymphoma (BLL) occurring in a patient with celiac disease in his eighth decade of life. It is possible that chronic inflammation, profound immunosuppression, and nutritional deficit could lead to development of high-grade B-cell lymphoproliferative disorders. Further molecular studies are warranted to the investigate the link between certain polymorphisms of human leukocyte antigens (HLA) in B-cell populations in the gut, and this might be useful to identify high-risk individuals in the population of patients with CD.     

Suppression of frequent ventricular ectopy in a patient with hypertrophic heart disease with ranolazine: a case report

Background

Pro-arrhythmic concerns with most anti-arrhythmic agents in patients with significant left ventricular hypertrophy (LVH) limits options when anti-arrhythmic therapy is indicated. Ranolazine, an anti-anginal agent which inhibits late Na+ currents, indirectly causes a decrease in diastolic cardiomyocyte Ca++ levels producing an energy sparing effect. Ranolazine also inhibits triggered activity in animal studies and has anti-arrhythmic properties in patients with ischemic heart disease. Here we report the dramatic anti-arrhythmic effects of ranolazine in a patient with frequent ventricular and supraventricular ectopy in the setting of hypertrophic heart disease without significant coronary artery disease.

Methods

A 72 year old hypertensive patient with palpitations and significant exercise intolerance due to dyspnea was evaluated with echocardiography, thallium stress testing and cardiac catheterization. Holter monitor data prior to, and after institution of ranolazine 1000 mg twice daily was compared. Patient tolerance and sense of well being after ranolazine was assessed.

Results

Significant LVH was noted and obstructive coronary artery disease was ruled out by cardiac catheterization. Within two hours of the initial dose of ranolazine a marked decrease in ventricular ectopy was observed. Ventricular ectopy on Holter monitor decreased approximately 12 fold (23.8% of beats to1.9%) while supraventricular ectopy decreased approximately 7 fold (5.3% of beats to 0.8%). The decrease in ectopy was associated with an improved sense of well being.

Conclusion

Ranolazine had rapid onset, potent anti-arrhythmic properties in the absence of obstructive coronary artery disease in a patient with LVH and may be an ideal agent in patients where few anti-arrhythmic options exist.     

Severe metabolic alkalosis: a case report.

A 45-year-old man who was admitted with nausea, vomiting, and abdominal pain was found to have severe metabolic alkalosis, with a PaCO2 of 11.4kPa (85.5 mm Hg), PaO2 of 5.8 kPa (43.5 mm Hg), pH of 7.61, and plasma bicarbonate concentration of 82.0 mmol/l. He was treated with oxygen, intravenous physiological saline, and phenytoin and improved within 48 hours. Radiographs showed gastric outlet obstruction secondary to peptic ulcer, which was treated by surgery. Though sever, the rise in carbon dioxide concentration in this patient was probably lifesaving. The PaCO2 was therefore allowed to fall gradually as the alkalosis was treated. The return of both PaCO2 and plasma bicarbonate values to normal in parallel suggests that hypoventilation compensated for the metabolic alkalosis and emphasises the importance of conservative treatment in cases of metabolic alkalosis.     

VACTERL-H associated with central hypothyroidism: a case report

The VACTERL-H syndrome is a rare combination of vertebral anomalies, anal atresia, congenital heart defects, tracheo-esophageal fistula, abnormalities of kidneys and limb anomalies together with hydrocephalus. This condition is recognized as a hereditary entity with poor prognosis. We present a newborn weighing 3400 g, born by cesarean section to a 27 years old mother who had had an irregular antenatal follow-up. The patient had severe hydrocephalus, proximal esophageal atresia and distal tracheoesophageal fistula, gastric outlet obstruction, imperforated anus and recto-urethral fistula, patent ductus arterious, a bifid scrotum, a vertebral defect, sacral dimple and central hypothyroidism. The patient had no limb defects. The association of central hypothyroidism and VACTERL-H has previously not been reported.