Cases reported "Chronic Disease"

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21/192. An unusual case of chronic neuropathic pain responds to an optimum frequency of intravenous ketamine infusions.

    The effective treatment of patients suffering from a variety of difficult pain syndromes, including phantom pain and other neuropathic pains, remains a clinical challenge. Neuropathic pain has been shown to respond to drugs that block the N-methyl-D-aspartate (NMDA) receptor, such as ketamine and amantidine. A 44-year-old woman with a previous right-sided forequarter amputation presented to the Palliative medicine Team complaining of neuropathic pain in her left arm, which was neurologically intact. The pain was treated with repeated infusions of intravenous ketamine. Twenty-one infusions were given over a period of four months. The pain intensity experienced by the patient lessened as the frequency of the ketamine infusions increased. This finding has not been described previously and supports the theory that there may be an optimum frequency of ketamine infusions to achieve adequate pain control.
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22/192. Atypical left atrial flutter after intraoperative radiofrequency ablation of chronic atrial fibrillation: successful ablation using three-dimensional electroanatomic mapping.

    Curative treatment of chronic atrial fibrillation (AF) remains a challenging task for electrophysiologists. Eliminating the initiating triggers by focal radiofrequency ablation in a subset of patients with paroxysmal AF and modifying the maintaining substrate by performing linear lesions within the left atrium in patients with prolonged episodes of AF are among the alternative approaches for management of these patients. Recently, a new intraoperative treatment procedure aimed at eliminating left atrial anatomic "anchor" reentrant circuits by induction of contiguous lesions using radiofrequency energy under direct vision was introduced. However, atypical left atrial flutter may occur during follow-up after intraoperative ablation of AF. These arrhythmias most likely are due to discontinuities in linear lesions; therefore, they can be successfully mapped and ablated in a subsequent percutaneous catheter ablation procedure. We report and discuss the case of a patient who underwent successful intraoperative ablation of chronic AF, but who developed atypical left atrial flutter postoperatively. Three-dimensional nonfluoroscopic electroanatomic mapping revealed a gap in the linear lesion line connecting the left upper and right upper pulmonary vein orifices. Ablation at the exit site of the breakthrough was successful.
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23/192. Fatal paradoxic embolism occurring during IVC filter insertion in a patient with chronic pulmonary thromboembolic disease.

    A patient diagnosed with chronic thromboembolic disease experienced a fatal intraprocedural paradoxic embolism during inferior vena cava (IVC) filter insertion. The frequency of patent intra-atrial shunts in patients with chronic thromboembolic disease is surprisingly high, occurring in approximately 29.5% of patients with chronic thromboembolic disease. Prevention of embolic events during venous interventions in such cases requires that the operator recognize that central shunts may be present and strictly adhere to meticulous technique during IVC filter insertion.
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24/192. A case of familial clustering of interstitial cystitis and chronic pelvic pain syndrome.

    A case of familial clustering of interstitial cystitis (IC) and chronic pelvic pain syndrome (CPPS), a symptom complex similar to IC that occurs in men, is reported. The proband was a 28-year-old woman with a 6-month history of severe frequency, urgency, and genital pain. After cystoscopy with hydrodistention and biopsy, a diagnosis of IC was made. IC was also diagnosed in the patient's mother and in two of her brothers, previously considered to have CPPS (category IIIB CPPS). A third brother was asymptomatic. This case highlights the importance of genetic factors in the onset of symptoms and natural history of IC and CPPS.
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25/192. IgM monoclonal antibody against terminal moiety of GM2, GalNAc-GD1a and GalNAc-GM1b from a pure motor chronic demyelinating polyneuropathy patient: effects on neurotransmitter release.

    We describe a patient with a pure motor chronic demyelinating polyneuropathy with an IgM monoclonal component showing anti-GM2, GalNAc-GD1a and GalNAc-GM1b reactivity whose common epitope appears to be -[GalNAcbeta1-4Gal(3-2alphaNeuAc)beta1]. We used intracellular recording to study how IgM from this patient affected neurotransmitter release in the mouse diaphragm in vitro.Adding serum (and specifically, the purified monoclonal IgM component) blocked the nerve-evoked response in both quantal content and evoked endplate potential (EPP) amplitude in a complement-independent and reversible manner. The IgM increased the frequency of spontaneous miniature endplate potentials (MEPPs) in a complement-dependent and reversible manner but had no effect on MEPP amplitude.
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26/192. Differential cellular targets of Epstein-Barr virus (EBV) infection between acute EBV-associated hemophagocytic lymphohistiocytosis and chronic active EBV infection.

    Unusual Epstein-Barr virus (EBV) infection into T or natural killer cells plays a pivotal role in the pathogenesis of acute EBV-associated hemophagocytic lymphohistiocytosis (EBV-HLH) and chronic active EBV infection (CAEBV). The precise frequency and localization of EBV genome in lymphocyte subpopulations especially within T-cell subpopulations are unclear in these EBV-related disorders. This study analyzed the frequency of EBV-infected cells in circulating lymphocyte subpopulations from 4 patients with acute EBV-HLH and 4 with CAEBV. EBV- encoded small rna-1 in situ hybridization examination of peripheral blood lymphocytes showed a significantly higher frequency of EBV-infected cells of 1.0% to 13.4% in EBV-HLH and 1.6% to 25.6% in CAEBV, respectively. The patterns of EBV infection in lymphocyte subpopulations were quite different between acute EBV-HLH and CAEBV. EBV infection was predominant in CD8( ) T cells in all EBV-HLH patients, whereas the dominant EBV-infected cell populations were non-CD8( ) lymphocyte subpopulations in CAEBV patients. Phenotypical analysis revealed that EBV-infected cell populations from both EBV-HLH and CAEBV were activated. There was no predominance of any EBV substrain of latent membrane protein-1, EBV-associated nuclear antigen (EBNA)-1, and EBNA-2 genes between the 2 abnormal EBV-associated disorders, and self-limited acute infectious mononucleosis. These results showing differential virus-cell interactions between acute EBV-HLH and CAEBV indicated different pathogenic mechanisms against EBV infection between the 2 EBV-associated diseases, which accounts for the difference in clinical manifestations between the 2 diseases.
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27/192. Chronic T-cell lymphoproliferative disease expressing natural killer cell receptors: clinicopathological and molecular features.

    The frequency and clinicopathological significance of the expression of natural killer cell receptors (NKRs) in T-cell malignancies remain undefined. A 71-year-old man presented with leukocytosis, generalized lymphoadenopathy, and hepatosplenomegaly. bone marrow and lymph node biopsies showed a T-cell lymphoproliferative disease expressing NKRs (CD2( ), CD3( ), CD4( ), CD5( ), CD7( ), CD8(-), CD56(-), CD94( ), CD158a( ), CD158b( ), CD161(-), p70(-), TCRalphabeta(1), TCRgammadelta(2), TIA-1(-)). An abnormal clone, 46,Y,add(X)(p14),der(1)t(1;6)(p33;p21),t(7;12)(p10;q10), was found on conventional karyotyping. comparative genomic hybridization confirmed these findings, and showed a deletion of 12p that was not apparent on karyotyping. Clinically, the disease remained indolent and responded transiently to purine analogs but not to intensive chemotherapy. Peripheral T-cell lymphoproliferative disease of CD4( )alphabeta(1)NKR( ) phenotype is hitherto undescribed. The issues of whether this case was derived from transformation of a rare T-cell subtype or represented aberrant T-cell expression of NK-cell antigens, and the clinicopathologic significance of these T-cell neoplasms warrant further studies.
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28/192. Circulatory arrest to protect transplant kidney in a patient with chronic type III dissection.

    With broader indications for renal transplantation and improved allograft survival, it is anticipated that the problem of aortic disease in the post-transplant patient will be encountered with increasing frequency. We report a technique of protecting the transplant kidney from ischemic damage during distal aortic surgery. A 30-year-old renal transplant patient who had undergone an operation for ruptured chronic type III dissection 3 years previously underwent abdominal aortic aneurysm repair under hypothermic circulatory arrest. The patient recovered uneventfully and is presently doing well 1 year after the operation. Hypothermic circulatory arrest could be used in selected cases as a useful alternative for transplant kidney protection.
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29/192. Chronic high-frequency globus pallidus internus stimulation in different types of dystonia: a clinical, video, and MRI report of six patients presenting with segmental, cervical, and generalized dystonia.

    The results of deep brain stimulation (DBS) of the globus pallidus internus (Gpi) in six patients with generalized, focal, and segmental dystonia are presented. Pre- and postoperative assessments are given for one patient with generalized inherited dystonia and for five patients with idiopathic segmental or cervical dystonia. Clinical symptoms were evaluated before and 3-12 months after surgery using the Burke-Fahn-Marsden (BFM) dystonia rating scale for primary torsion dystonia and the Tsui scale for cervical dystonia. The Short-Form Health Survey (SF-36) was completed by each patient to document preoperative and postoperative health status. Also, neurological status was documented by video before and during chronic stimulation. magnetic resonance imaging studies were performed to show the anatomical localization of the electrode leads. Five patients showed a progressive improvement within 7 days. One patient with cervical dystonia and Meige's syndrome showed no improvement for 3 months, but beneficial effects were observed after 12 months. On average, the BFM movement scale scores decreased by 72.5% and Tsui scale scores by 63%. SF-36 showed an improvement in health status by an average of 36% according to eight different health categories. We conclude that chronic high-frequency Gpi stimulation in different types of dystonia is a very effective and safe treatment.
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30/192. Management of life-threatening autonomic hyper-reflexia using magnesium sulphate in a patient with a high spinal cord injury in the intensive care unit.

    We report the successful use of i.v. magnesium sulphate to control life-threatening autonomic hyper-reflexia associated with chronic spinal cord injury in the intensive care environment. A 37-yr-old, male was admitted to the intensive care unit with a diagnosis of septic shock and acute renal failure secondary to pyelonephritis. He had been found unresponsive at home following a 2-day history of pyrexia and purulent discharge from his suprapubic catheter. He had sustained a T5 spinal cord transection 20 yr previously. Initial management included assisted ventilation, fluid resuscitation, vasopressor support, and continuous veno-venous haemofiltration. The sepsis was treated with antibiotic therapy and percutaneous nephrostomy drainage of the pyonephrosis. On the fifth day, the patient developed profuse diarrhoea. This was associated with paroxysms of systemic hypertension and diaphoresis, his arterial pressure rising on occasion to 240/140 mm Hg. A diagnosis of autonomic hyper-reflexia was made and a bolus dose of magnesium sulphate 5 g was administered over 15 min followed by an infusion of 1-2 g h(-1). There was an almost immediate decrease in the severity and frequency of the hypertensive episodes. There were no adverse cardiac effects associated with the administration of magnesium, only a slight decrease in minute ventilation as the plasma level approached the upper end of the therapeutic range (2-4 mmol litre(-1)). In view of the beneficial effects observed in this case we advocate further research into the use of magnesium sulphate in the treatment or prevention of autonomic hyper-reflexia secondary to chronic spinal cord injury in the intensive care unit.
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