Parkinsonism-hyperpyrexia syndrome (PHS) is a neurologic potentially fatal emergency that mimics neuroleptic malignant syndrome. It commonly presents as systemic inflammatory response syndrome, acute onset worsening of muscular rigidity, autonomic instability, hyperpyrexia, confusion, diaphoresis and high creatine phosphokinase. The most common trigger for PHS is reduction or withdrawal of anti-Parkinson’s medications, especially levodopa. It was also reported in a few cases following deep brain stimulation of the subthalamic nucleus surgery shortly after anti-Parkinson’s medications were discontinued. Rare causes of PHS include deep brain stimulator (DBS) malfunction due to battery depletion. To the best of our knowledge, PHS following DBS battery depletion was reported only in three occasions. Here, we report a case of PHS due to DBS battery depletion presented as sepsis and was successfully treated with the administration of dopamine agonists, intravenous fluids and changing the DBS battery.
Pulmonary arterial hypertension (PAH) describes a rare, progressive disease of the pulmonary vasculature, involving a group of clinical conditions that result in precapillary pulmonary hypertension (PH). PAH is characterized by proliferative vasculopathy, and subsequent right heart failure. For the past several decades research has focused on identification of underlying molecular causes of this disease. Recently, there have been a number of reported cases of patients with scurvy developing pulmonary arterial hypertension. Thus, it has been hypothesized that vitamin C deficiency results in non-hypoxic activation of hypoxia inducible transcription factors (HIF) and low nitric oxide (NO) level in the pulmonary vasculature, leading to subsequent pulmonary vasculopathy and an exaggerated pulmonary vasoconstrictive response. Immediate supplementation of vitamin C is considered the definitive treatment, preventing an otherwise fatal outcome. In this paper, we describe a patient with a fatal case of scurvy related PAH, who was admitted with ecchymosis, oral ulceration, and dyspnea.
Objectives HIV elite controllers (ECs) are a unique subgroup of HIV-positive patients who are long-term virologically suppressed in the absence of antiretroviral treatment (ART). The prevalence of this subgroup is estimated to be < 1%. Various cohorts of ECs have been described in developed countries, most of which have been demographically heterogeneous. The aim of this study was to identify ECs in two large African cohorts and to estimate their prevalence in a relatively genetically homogenous population. Methods We screened two cohorts of HIV-positive Ethiopian patients. The first cohort resided in Mekelle, Ethiopia. The second was comprised of HIV-positive Ethiopian immigrants in Israel. In the Mekelle cohort, ART-naïve subjects with stable CD4 counts were prospectively screened using two measurements of viral load 6 months apart. Subjects were defined as ECs when both measurements were undetectable. In the Israeli cohort, subjects with consistently undetectable viral loads (mean of 17 viral load measurements/patient) and stable CD4 count > 500 cells/µL were defined as ECs. Results In the Mekelle cohort, 16 of 9515 patients (0.16%) fitted the definition of EC, whereas seven of 1160 (0.6%) in the Israeli cohort were identified as ECs (P = 0.011). Conclusions This is the first large-scale screening for HIV-positive ECs to be performed in entirely African cohorts. The overall prevalence of ECs is within the range of that previously described in developing countries. The significant difference in prevalence between the two cohorts of similar genetic background is probably a consequence of selection bias but warrants further investigation into possible environmental factors which may underlie the EC state.
Pulmonary arterial hypertension (PAH) is a progressive disease of the pulmonary vasculature that results in precapillary pulmonary hypertension. PAH is caused by a group of clinical conditions involving multiple organ systems. Several cases have been reported in the literature demonstrating an association between vitamin C deficiency and PAH. Low endothelial nitric oxide levels in the pulmonary vasculature, combined with the inappropriate activation of hypoxia-inducible transcription factors, seen in patients with ascorbic acid deficiency, are believed to be the main contributors to the pathogenesis of pulmonary vasculopathy and the exaggerated pulmonary vasoconstrictive response seen in patients with scurvy-induced PAH. Vitamin C supplementation is considered the definitive treatment.
The presence of chyle in the pleural cavity is referred to as chylothorax. Exudative chylothorax is usually related to damage or obstruction of the lymphatic vasculature with subsequent leakage into the pleural space. In contrast, transudative chylothorax is related to increased hydrostatic pressure caused by elevated intra-abdominal pressure, which leads to the translocation of chylous fluid into the pleural space. Cirrhosis is the most common cause of transudative chylothorax, commonly presenting with ascites and portal hypertension. To the best of our knowledge, isolated transudative chylothorax as a consequence of cirrhosis is exceptionally rare and has been scarcely reported in the literature. We herein report a female patient in her fifties who presented to our hospital with isolated unilateral transudative hepatic chylothorax, with no clinical evidence of cirrhosis or any stigmata of portal hypertension at the time of presentation.
We present a case of rituximab-induced organizing pneumonia (OP) along with bronchiectasis and pulmonary fibrosis, in a patient with a history of granulomatosis with polyangiitis (GPA), on long-term maintenance therapy with rituximab. T-cell dysregulation and B-cell depletion associated with the chronic use of rituximab often lead to a profound immunosuppressed state with hypogammaglobulinemia and unbalanced T-cell response. This acquired immunodeficient state with severe immune dysregulation predisposed this patient to recurrent pulmonary infection and ultimately led to bronchiectasis and pulmonary fibrosis.
Parkinsonism-hyperpyrexia syndrome (PHS) is a rare, potentially fatal neurological emergency, that is seen in Parkinson's Disease (PD) patients and mimics neuroleptic malignant syndrome. The most common trigger for PHS is sudden withdrawal of anti-parkinsonian medications, specifically levodopa. However, it can also be due to Deep Brain Stimulation (DBS) device malfunction. In this work, we describe three cases of PHS; the first of which is related to DBS battery depletion, and the remaining two to dopaminergic withdrawal. Additionally, we will include the results of a literature review on PHS, its etiologies, presentation, and management.
Stress (Takotsubo) cardiomyopathy (CM) is defined as a transient regional systolic dysfunction of the left ventricle that mimics acute coronary syndrome (ACS) in the absence of significant coronary artery disease or plaque rupture. This phenomenon commonly occurs in postmenopausal females in the presence of emotional, physical and psychological stressors with excess catecholamine stimulation, resulting in diffuse microvascular spasm and subsequently myocardial stunning. Here we report a 58-year-old postmenopausal female patient with end stage renal disease (ESRD) who was presented to our hospital with a clinical picture suggesting ACS. Shortly after admission she progressed to cardiogenic shock and pulseless electrical activity (PEA) and was diagnosed retrospectively with Takotsubo cardiomyopathy. She was successfully treated with beta-blockers, IV fluids and inotropic agents. Intra-aortic balloon pump (IABP) was considered, however, the patient gradually improved to full recovery with resolution of left ventricular function back to normal.
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