Poor outcome of bilateral lower extremity morel-lavallee lesions: a case report.
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ABSTRACT:
SUBMITTER: Stanley SS
PROVIDER: S-EPMC3933474 | biostudies-other | 2014 Mar
REPOSITORIES: biostudies-other
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Project description:INTRODUCTION: Polymelia, or congenital duplication of a limb, is an extremely rare entity in humans, with few cases reported in the literature. CASE PRESENTATION: We present the case of a six-month-old Hispanic boy born with a lower limb bud on the left posterior thigh. CONCLUSION: The infant had a favorable outcome and evolution after surgical treatment of his supernumerary limb, with no after-effects or impairment whatsoever.
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Project description:BACKGROUND:Bilateral thalamic lesions are rare. Here, we describe a case of probable acute disseminating encephalomyelitis (ADEM) with symmetrical bilateral thalamic lesions. CASE PRESENTATION:An 85-year-old man presented with weakness of the lower limbs and urinary retention for 1 day, soon followed by coma. He had an H1N1 influenza vaccination 3 months ago. A lumbar puncture showed positive oligoclonal bands and negative results for anti-AQP4 antibodies. A head MRI demonstrated focal symmetrical bilateral thalamic lesions. An MRI of the thoracic spinal cord showed longitudinally extensive lesions in the spinal cord. He was diagnosed with probable ADEM. Despite being treated with IVIG, the patient remained unconscious and died a month later from pneumonia. CONCLUSIONS:In cases with bilateral thalamic lesions, the possibility of ADEM should be considered. The characteristics of the thalamic lesions and imaging findings in other parts of the brain or spinal cord should be taken into account in association with the clinical and laboratory information in making a correct diagnosis.
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Project description:BackgroundVenous thrombo-embolic events have been described in hospitalized patients with coronavirus disease 2019 (COVID-19), suggesting the presence of coagulopathy induced by the viral infection. To date, only rare cases of arterial thrombosis related to COVID-19 have been reported.Case summaryA 54-year-old patient with an influenza-like illness 15 days earlier, which resolved, and no known cardiovascular risk factor presented with acute right lower limb ischaemia. A computed tomography angiogram of the abdominal aorta and lower extremities showed, in the absence of vascular disease, a subocclusive thrombosis of the right common iliac artery and an occlusion of the right internal iliac, profunda femoral, and popliteal arteries. On the left side, the computed tomography angiogram demonstrated a non-occlusive thrombosis of the common femoral artery. The patient underwent emergency surgical thrombectomy as well as endovascular revascularization on the right side followed by therapeutic anticoagulation, with normalization of the limb perfusion. A nasopharyngeal swab for severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) by real-time reverse transcription-PCR (rRT-PCR) was negative three times. Haemostasis analysis showed a mild hyperfibrinogenaemia and a shortening of the activated partial thromboplastin time. An extensive screening for cardio-embolism was negative. As the thrombotic event was unexplained, antibody testing for SARS-CoV-2 was performed and the result was positive.DiscussionVenous thrombosis and pulmonary embolisms have been observed in COVID-19. As in our case, the first reports on COVID-19-associated arterial thrombotic events have emerged. A better understanding of the coagulopathy in COVID-19 is essential to guide prevention and treatment of venous as well as arterial thrombo-embolic events.
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Project description:Although open surgical bypass remains the standard revascularization strategy for patients with critical limb ischemia (CLI), many centers now perform peripheral endovascular intervention (PVI) as the first-line treatment for these patients. We sought to determine the effect of a prior ipsilateral PVI (iPVI) on the outcome of subsequent lower extremity bypass (LEB) in patients with CLI.A retrospective cohort analysis of all patients undergoing infrainguinal LEB between 2003 and 2009 within hospitals comprising the Vascular Study Group of New England (VSGNE) was performed. Primary study endpoints were major amputation and graft occlusion at 1 year postoperatively. Secondary outcomes included in-hospital major adverse events (MAE), 1-year mortality, and composite 1-year major adverse limb events (MALE). Event rates were determined using life table analyses and comparisons were performed using the log-rank test. Multivariate predictors were determined using a Cox proportional hazards model with multilevel hierarchical adjustment.Of 1880 LEBs performed, 32% (n = 603) had a prior infrainguinal revascularization procedure (iPVI, 7%; ipsilateral bypass, 15%; contralateral PVI, 3%; contralateral bypass, 17%). Patients with prior iPVI, compared with those without a prior iPVI, were more likely to be women (32 vs 41%; P = .04), less likely to have tissue loss (52% vs 63%; P = .02), more likely to require arm vein conduit (16% vs 5%; P = .001), and more likely to be on statin (71% vs 54%; P = .01) and beta blocker therapy (92% vs 81%; P = .01) at the time of their bypass procedure. Other demographic factors were similar between these groups. Prior PVI or bypass did not alter 30-day MAE and 1-year mortality after the index bypass. In contrast, 1-year major amputation and 1-year graft occlusion rates were significantly higher in patients who had prior iPVI than those without (31% vs 20%; P = .046 and 28% vs 18%; P = .009), similar to patients who had a prior ipsilateral bypass (1 year major amputation, 29% vs 20%; P = .022; 1 year graft occlusion, 33% vs 18%; P = .001). Independent multivariate predictors of higher 1-year amputation and graft occlusion rates were prior iPVI, prior ipsilateral bypass, dialysis dependence, prosthetic conduit and distal (tibial and pedal) bypass target.Prior iPVI is highly predictive for poor outcome in patients undergoing LEB for CLI with higher 1-year amputation and graft occlusion rates than those without prior revascularization, similar to prior ipsilateral bypass These findings provide information, which may help with the complex decisions surrounding revascularization options in patients with CLI.
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Project description:OBJECTIVE:To determine the clinical correlates bilateral independent periodic discharges (BIPDs) and their association with electrographic seizures and outcome. METHODS:Retrospective case-control study of patients with BIPDs compared to patients without periodic discharges ("No PDs") and patients with lateralized periodic discharges ("LPDs"), matched for age, etiology and level of alertness. RESULTS:We included 85 cases and 85 controls in each group. The most frequent etiologies of BIPDs were stroke, CNS infections, and anoxic brain injury. Acute bilateral cerebral injury was more common in the BIPDs group than in the No PDs and LPDs groups (70% vs. 37% vs. 35%). Electrographic seizures were more common with BIPDs than in the absence of PDs (45% vs. 8%), but not than with LPDs (52%). Mortality was higher in the BIPDs group (36%) than in the No PDs group (18%), with fewer patients with BIPDs achieving good outcome (moderate disability or better; 18% vs. 36%), but not than in the LPDs group (24% mortality, 26% good outcome). In multivariate analyses, BIPDs remained associated with mortality (OR: 3.0 [1.4-6.4]) and poor outcome (OR: 2.9 [1.4-6.2]). CONCLUSION:BIPDs are caused by bilateral acute brain injury and are associated with a high risk of electrographic seizures and of poor outcome. SIGNIFICANCE:BIPDs are uncommon but their identification in critically ill patients has potential important implications, both in terms of clinical management and prognostication.
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Project description:Ochronosis is a syndrome characterized by bluish black discoloration due to the deposition of polymerized products of homogentisic acid (HGA) in the connective tissues. The endogenous variety (alkaptonuria), is a rare autosomal recessive metabolic disorder. The disorder is manifested by deficiency of the enzyme homogentisate 1,2-dioxygenase. The characteristic of the condition is a triad of pigmentation of skin, cartilage, and sclera; ochronotic arthropathies and homogentisic aciduria (resulting in darkening of urine). More rarely, it may affect the breast. This rare and interesting case of a woman with ochronosis of both breasts and chest wall, prompted us to write this case report.
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Project description:Hidradenocarcinoma (HC) is a rare malignant sweat gland tumor with metastatic potential primarily located in the head, neck, and trunk. We present an unusual case of a large lower extremity Clear Cell HC managed with surgical resection and adjuvant locoregional radiation after excluding lymph node involvement.
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Project description:BackgroundOcular neuromyotonia (ONM) is characterized by episodic diplopia, which is usually triggered by prolonged eccentric gaze of the affected extraocular muscles. The spell is characterized by involuntary, occasionally painful, sustained contraction of one or more extraocular muscles innervated by the oculomotor, trochlear, or abducens nerve. ONM usually occurs as a late consequence of radiotherapy around the parasellar area, although idiopathic cases have been reported. Most cases are unilateral; however, bilateral ONM has occasionally been described.Case presentationA 60-year-old woman presented with a 4-month history of episodic, painful, horizontal binocular diplopia. She underwent external beam radiotherapy to the skull base for treatment of nasopharyngeal carcinoma. The tumor was well controlled. General neurological examination findings were unremarkable. Neuro-ophthalmic examination revealed normal visual acuity, visual fields, pupils, and fundi. Ocular alignment showed orthotropia with normal ocular motility. Myasthenic eyelid signs were absent. However, she developed episodes of involuntary sustained contraction of the medial rectus muscle following prolonged eccentric gaze toward the affected medial rectus muscle, which resulted in esotropia upon returning to the primary position. The esotropic episodes spontaneously resolved after approximately 2 min. These spells affected both medial rectus muscles. Both pupils remained normal throughout the examination. Magnetic resonance imaging revealed neither brain parenchyma/brain stem lesions nor tumor recurrence. Her symptoms were successfully treated with carbamazepine.ConclusionsEpisodic esotropia in the adducting eye following prolonged horizontal eccentric gaze is a significant characteristic of ONM affecting the bilateral medial rectus muscles (i.e., bilateral oculomotor ONM). In spite of its extreme rarity, ONM should be considered as a differential diagnosis of episodic diplopia, especially in patients with a history of radiotherapy around the parasellar area. Careful examination with prolonged eccentric gaze should be performed to achieve a correct diagnosis and avoid an extensive unnecessary workup.
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Project description:IntroductionThe clinical impact of vascular calcification is well established in the context of cardiovascular morbidity and mortality, but other clinical syndromes, such as calciphylaxis, although less frequent, have a significant impact on chronic kidney disease.MethodsCase report of a 27-year-old woman, who had complained of bilateral pain in her toes for 3 days, with the presence of small necrotic areas in the referred sites. She had a history of type 1 diabetes (25 years ago), with chronic kidney disease, on peritoneal dialysis, in addition to rheumatoid arthritis. She was admitted to the hospital, which preceded the current condition, due to exacerbation of rheumatoid arthritis, evolving with intracardiac thrombus due to venous catheter complications, when she started using warfarin. Ischemia progressed to her feet, causing the need for bilateral amputations. Her chirodactyls were also affected. Thrombophilia, vasculitis, endocarditis or other embolic sources were investigated and discarded. Her pathology report evidenced skin necrosis and superficial soft parts with recent arterial thrombosis, and Monckeberg's medial calcification. We started treatment with bisphosphonate and sodium thiosulfate, conversion to hemodialysis and replacement of warfarin with unfractionated heparin. Despite all the therapy, the patient died after four months of evolution.DiscussionCalciphylaxis is a rare microvasculature calcification syndrome that results in severe ischemic injuries. It has pathogenesis related to the mineral and bone disorder of chronic kidney disease combined with the imbalance between promoters and inhibitors of vascular calcification, with particular importance to vitamin K antagonism.ConclusionThe preventive strategy is fundamental, since the therapy is complex with poorly validated effectiveness.
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