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 31,883 document(s) hit in 31,850,051 MEDLINE articles (0.00 seconds)

It is well known that the administration of high-dose gamma-globulin concentrate is effective in alleviating thrombocytopenia in patients with idiopathic thrombocytopenic purpura (ITP). However, treatment can sometimes induce aseptic meningitis. A 25-year-old Japanese woman with antiphospholipid syndrome and ITP was conditioned with high-dose gamma-globulin concentrate prior to splenectomy. Three days after the initial gamma-globulin course, she experienced severe headache, vomiting, and high-grade fever. Cerebrospinal fluid examination yielded a diagnosis of aseptic meningitis. The patient's clinical symptoms and abnormal cerebrospinal fluid findings disappeared immediately after the discontinuation of gamma-globulin therapy. Cases of aseptic meningitis induced by high-dose gamma-globulin therapy are uncommon in the literature, and most involve children. Moreover, to our knowledge, only 1 Japanese adult ITP case of gamma-globulin-induced aseptic meningitis has been reported to date. Aseptic meningitis may be one of the important adverse effects of the administration of high-dose gamma-globulin concentrate to pediatric as well as adult ITP patients.
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PMID:[Aseptic meningitis induced by high-dose gamma-globulin in an adult woman with antiphospholipid syndrome and chronic idiopathic thrombocytopenic purpura]. 1056 30

To describe the clinical and immunologic characteristics of patients with adrenal involvement and antiphospholipid syndrome (APS), we conducted a computer-assisted (PubMed) search of the literature to identify all cases of primary adrenal insufficiency associated with antiphospholipid antibodies published in English, French, and Spanish from 1983 (when APS was first defined) through March 2002. We reviewed 86 patients (80 from the literature plus 6 from our cohort); 55% were male, and the mean age at presentation was 43 +/- 16 years. Sixty-one (71%) patients had primary APS, and 14 (16%) had systemic lupus erythematosus. In 31 (36%) patients, adrenal insufficiency was the first clinical manifestation of APS. Abdominal pain was present in 55% of patients, followed by hypotension (54%), fever (40%), nausea or vomiting (31%), weakness or fatigue (31%), and lethargy or altered mental status (19%). The main finding in imaging techniques was compatible with adrenal hemorrhage (59%) and in histopathologic study was a hemorrhagic infarction with vessel thrombosis (55%). Lupus anticoagulant was detected in 97% of patients and the anticardiolipin antibodies titer was positive in 93% of patients. Most patients (95%) were positive for the IgG isotype of anticardiolipin antibodies, whereas 40% were positive for the IgM isotype. Baseline cortisol levels were decreased in 98% of patients, ACTH hormone levels were increased in 96% of patients, and the cosyntropin stimulation test was positive in 100% of patients tested. Steroid replacement therapy was the most frequent treatment (84%), followed by anticoagulation (52%) and aspirin (6%). Thirty-two of 35 (91%) patients with prolonged anticoagulant therapy were in good health with a mean follow-up of 25 months, whereas 25 of the 69 (36%) patients with outcome data available had died. The results of the present review stress the clinical importance of systematic screening for lupus anticoagulant and anticardiolipin antibodies in all cases of adrenal hemorrhage or infarction. An initial screening for hypoadrenalism is mandatory in any antiphospholipid antibody-positive patient who complains of abdominal pain and undue weakness or asthenia.
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PMID:Adrenal involvement in the antiphospholipid syndrome: clinical and immunologic characteristics of 86 patients. 1264 Jan 87

The clinical picture of venous or arterial thrombosis in the presence of circulating antiphospholipid antibodies is referred to as the antiphospholipid syndrome. A 5-month-old baby girl who was quite healthy so far was referred to our clinic with irritability, vomiting, and abdominal distension for 30 hours. Surgical exploration exposed a gangrenous ileal segment about 15 cm long. The postoperative period was unremarkable. Investigation to identify the risk factors for mesenteric thrombosis found anticardiolipin antibodies (isotype Ig G) and decreased protein C level. Protein S and antithrombin III were within normal levels. Hb electrophoresis results showed no HbS, and neither Factor V Leiden nor prothrombin 20210 mutations were detected. Eight months postoperatively, anticardiolipin antibodies were found within normal levels. Lupus anticoagulant, ds DNA, and ss DNA were negative. Direct coombs test and protein C, C3, and C4 were also within normal levels. She had no thrombotic episode in the 24 months postoperatively, although no anticoagulant medication was administered. To the authors' knowledge this case is the first report of segmental intestinal infarction in transient antiphospholipid syndrome in the pediatric population.
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PMID:Transient antiphospholipid syndrome in an infant with segmental small bowel infarction. 1469 90

The authors report a case of a 56-year-old Thai woman with a history of recurrent venous thrombosis, spontaneous abortion and Graves' disease who presented with bilateral flank pain, nausea, vomiting and low-grade fever followed by hypotension. Adrenal crisis from bilateral adrenal hemorrhage was diagnosed by a low serum cortisol level during hypotension and bilateral hyperdense oval masses in each of the adrenal glands in a computerized tomographic study. Her hemostatic and serologic profile was compatible with primary antiphospholipid syndrome. Rapid improvement was observed after the administration of intravenous hydrocortisone. She was discharged on long-term glucocorticoid replacement for her primary adrenal insufficiency as well as an anticoagulant for prevention of thrombosis. The antiphospholipid syndrome should be suspected in a patient presenting with adrenal crisis without a distinct etiology.
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PMID:Adrenal crisis due to bilateral adrenal hemorrhage in primary antiphospholipid syndrome. 1614 61

We report an unusual case with macro-amylasemia with coexistent selective IgA deficiency and antiphospholipid antibodies. A 16-year-old girl was referred to us with a history of episodic abdominal pain accompanied by vomiting and diarrhea. Macroamylasemia was demonstrated by precipitation of 99% amylase activity with polyethylene glycol 6000. She had high levels of anticardiolipin IgG and beta2 glycoprotein 1 IgG antibodies in the blood, but no evidence of clinical criteria of antiphospholipid syndrome. In the literature, although macro-amylasemia has been found to occur in a variety of diseases including autoimmune disorders, to our knowledge, this is the first well-documented case of macro-amylasemia associated with selective IgA deficiency and the presence of antiphospholipid antibodies. It is important that clinicians be aware of their existence in order to avoid unnecessary procedures and that the patient is informed of the macro-amylasemia; moreover, it should be stated in the patient's health record.
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PMID:Macro-amylasemia in a patient with selective IgA deficiency and antiphospholipid antibodies. 1683 Mar 1

Anti Phospholipid Syndrome (APS) is a relatively new conception of syndrome complex first noticed in 1983. It may be primary or secondary to other diseases like SLE, RA, Systemic sclerosis, behchet's syndrome, temporal arteritis, sjogren's syndrome psoriatic arthropathy etc. Clinical manifestations are consequences of vascular thrombosis and embolism like DVT, pulmonary embolism, stroke, TIA, complication of pregnancy with pregnancy loss. We report a 34 years married female housewife who presented with sudden onset of nausea, vomiting, vertigo, dysphagia, dysarthria and ataxia. She had a chronic leg ulcer. Neurological findings were consistent with lateral medullary syndrome due to stroke though she was normotensive, nondiabetic with normal lipid profile. She had history of two abortions in last three years. Investigations were done accordingly and she fulfilled the diagnostic criteria of APS. No secondary cause was detected after thorough clinical examination and laboratory investigations. She was treated symptomatically along with oral anticoagulation. She improved slowly but steadily.
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PMID:Anti phospholipid syndrome. 1918 54

The antiphospholipid antibody syndrome is the most common acquired thrombophilia; it is a systemic autoimmune disease characterized by recurrent arterial and venous thrombosis and/or pregnancy loss, in association with circulating antiphospholipid antibodies. The pathogenic mechanisms in antiphospholipid antibody syndrome that lead to in vivo injury are incompletely understood. Like other autoimmune diseases, a combination of genetic and environmental factors is involved. We report the case of a 50-year-old woman suffering from an antero-lateral non-ST-elevation myocardial infarction. After few days, coronary angiography showed a severe occlusive arterial disease, involving anterior descending, circumflex e right coronary arteries. Percutaneous coronary intervention was performed with the implantation of a drug-eluting stent in the proximal segment of the anterior descending coronary artery. One day after discharge (10 days after the first hospitalization) the patient experienced dizziness, nausea, vomiting, swelling in absence of any electrocardiographic abnormalities or myocardial enzyme elevation; then she was hospitalized in the neurology department. Because of a similar episode, urgent cerebral computed tomography scan was performed 5 days later; it revealed two different acute ischemic areas, parietal in the right hemisphere and cerebellar in the left hemisphere. The diagnosis of antiphospholipid antibody syndrome was confirmed by high anticardiolipin antibody titers, also present in medium titer at 5 and 17 weeks apart. She was discharged without any sequelae, on warfarin and double antiplatelet therapy (aspirin and clopidogrel for 6 months), then warfarin and aspirin.
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PMID:[Myocardial and cerebral infarction as initial presentation of antiphospholipid syndrome]. 1947 82

Antiphospholipid antibodies may signal the formation of vascular thrombi in the Antiphospholipid syndrome (APS). A rare complication of APS is adrenal insufficiency resulting from venous thrombus followed by hemorrhagic infarction. We describe the case of a 42-year-old male with APS presenting with vomiting and abdominal pain. Through laboratory and imaging diagnostic exams, we confirmed the diagnosis of bilateral adrenal hemorrhage and subsequent adrenal failure. We also conducted a search of literature associating bilateral adrenal thrombosis to APS, and describe the two pathological mechanisms most often cited to explain this phenomenon. To our knowledge, this is the first Portuguese case of adrenal insufficiency due to APS-associated bilateral adrenal hemorrhage.
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PMID:Definitive bilateral adrenal failure in antiphospholipid syndrome. 2278 16

An 11-year-old girl presented to the hospital with vomiting, left upper-quadrant pain and blood in the stool. An ultrasound scan showed no blood flow in the coeliac axis or the splenic artery. A contrast-enhanced CT scan then demonstrated no flow beyond the coeliac axis origin, a large clot in the distal superior mesenteric artery (SMA), a large splenic infarct and bilateral renal infarcts. Investigations revealed a positive lupus anticoagulant and she was given a presumptive diagnosis of catastrophic antiphospholipid syndrome (CAPS). She was anticoagulated and started on combination immunosuppressive therapy. Her surgical management was by SMA embolectomy, small bowel resection and proximal stoma formation. Stoma closure was performed at 3 months, and she was eventually established on full enteral feeds. Childhood CAPS is a rare condition with a very high mortality rate, and the successful outcome in this case reflects close multidisciplinary teamwork between medical and surgical specialties.
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PMID:CAPS: a rare acute abdomen. 2390 21

A 27-year-old man with a history of migraines, epilepsy and pulmonary stenosis presented to the emergency department with symptoms of vomiting, headache, visual disturbance and problems with balance. The team considered the possibility of intracranial pathology and an urgent CT head with contrast showed what appeared to be a large posterior fossa mass with an appearance suggestive of a primary haemangioblastoma, which was causing considerable mass effect. The patient had neurosurgery to relieve the obstruction and a biopsy of the area showed the mass to be an ischaemic infarct rather than a tumour. Further investigations following the stroke confirmed that the cause was due to having antiphospholipid syndrome and a patent foramen ovale. The patient made a good recovery following the operation and remains well.
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PMID:'A stroke of luck': a case report of a right-sided cerebellar cerebrovascular accident in a young man. 2489 86


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