The idiopathic hypereosinophilic syndrome

The purpose of this study was to review the positive angiographic findings in patients with polyarteritis nodosa (PAN). The authors reviewed the angiograms of 56 consecutive patients (25 women and 31 men; age range, 18-81 years; mean age, 55 years) with PAN and arterial abnormalities consistent with necrotizing vasculitis. Aneurysms were present in 27 patients and segments of ectasia were present in seven patients, for a total of 34 (61%) of 56 patients with aneurysmal lesions. The remaining 22 (39%) patients had arterial lesions that were occlusive: luminal irregularity, stenosis, or occlusion. All but one of the patients with an aneurysm also had occlusive lesions. Therefore, 55 (98%) of the 56 patients were found to have occlusive lesions. Skeletal muscle arteries were affected in 18 patients, nine in the extremities. The most frequent finding in patients with PAN was occlusive arterial lesions. Although the presence of aneurysms increases specificity for the diagnosis of PAN, many patients have only occlusive lesions. Involvement of skeletal muscle arteries was common.

Polyarteritis nodosa: spectrum of angiographic findings.

The use of intra-articular injections of high molecular weight elastoviscous solutions of hyaluronan or hylans (cross-linked derivatives of hyaluronan) to treat arthritis is termed viscosupplementation. The function of viscosupplementation is to restore the rheologic properties of synovial fluid. Although anecdotal data exist, no long-term studies regarding the use of viscosupplementation in the ankle have been published to date. The goal of this clinical trial was to compare pain reduction following ankle arthroscopy versus that following ankle arthroscopy combined with weekly intra-articular instillation of hylan G-F 20 during the first 3 postoperative weeks. In the series of patients described in this report, we found that both treatment groups experienced statistically significantly decreased pain following the intervention (P = .002 and P = .0009 for the arthroscopy alone and arthroscopy plus hylan groups, respectively), and that those who received 3 intra-articular injections of hylan G-F 20 following ankle arthroscopy improved statistically significantly (P = .0014) more than did those who underwent arthroscopy as a sole therapy. These preliminary results suggest that viscosupplementation combined with arthroscopy may be more beneficial than arthroscopy alone, and provide further insight into the role of viscosupplementation in the treatment of ankle osteoarthritis. Level of Clinical Evidence: 4

The Role of Viscosupplementation in the Ankle Using Hylan G-F 20

Summary

Four cases of extraperitoneal haemorrhage occurring in the setting of anticoagulation or coagulopathy are presented. Treatment of this condition has traditionally consisted of reversal of anticoagulation and supportive therapy. Diagnosis was made on contrast-enhanced computed tomography. The finding of active contrast extravasation was found to be a factor predictive of failure of conservative therapy and, therefore, an indication for angiography and embolization. In all four cases presented in the present paper, the bleeding vessel(s) were identified and significant active bleeding arrested by transcatheter embolization.

Spontaneous extraperitoneal haemorrhage: Computed tomography diagnosis and treatment by selective arterial embolization

Sir, The Ehlers±Danlos syndrome (EDS) is a heterogeneous group of heritable connective tissue disorders characterized by articular hypermobility, skin hyperextensibility, and tissue fragility. Type IV (EDSIV), the ecchymotic or vascular type, is a rare EDS subtype which is characterized by spontaneous vascular rupture and bowel perforation. The disease is generally inherited in an autosomal dominant fashion, but frequently there may be single affected individuals in families; it has been proposed that autosomal recessive forms also exist. The gene for EDSIV has been identified as the COL3A1 gene, located at 2q31±q32, encoding the chains of type III procollagen. A 31-year-old white female was referred to our department with suspected EDSIV. She was the second child of unrelated parents. After a normal pregnancy, she was born prematurely at 24 weeks' gestation weighing 1750 g. She failed to thrive and remained in an incubator for 2 months. At the time of delivery, her father was aged 40 years and her mother 39 years. No other members of the family were reported to be affected by skin problems. The patient's skin changes and articular hypermobility were noted immediately after birth. A tendency toward easy bruising and bleeding was noted when she was 6 years old. At the age of 14 years, marked shortening of the right achilles tendon causing talipes equinovarus was noted. In addition, she had a history of recurrent bilateral pneumothoraces treated by pleural stripping when she was 19 years old. At the age of 29 years there was a subarachnoidal haemorrhage due to rupture of an intracranial aneurysm. On physical examination, she had a thin, pinched, curved nose, thin lips, tight skin, hollow cheeks, and large prominent eyes. Tight, firm, lobeless ears were also present. There was prominent capillary telangiectasia and prominent venules over her upper anterior chest (Fig. 1). The skin on the dorsum of her hands was thin and had a prematurely aged appearance. Acrocyanosis of both hands was also noted. Mild hyperextensibility of the digits, hands, and elbows was present. Cardiological examination revealed low-grade pulmonary valve stenosis and mitral valve prolapse with thickening and prolapse of the anterior mitral valve leaflet. A skin biopsy from the left hand showed both collagen depletion and relative elastic fibre accumulation with shortening and fragmentation. Biochemical analysis by sodium dodecyl sulphatase± polyacrylamide gel electrophoresis showed decreased secretion of collagen type III by skin fibroblasts from the patient (Fig. 2). Separation of the collagen molecules retained in the cell layer showed an accumulation of collagen type III chains that migrated with a slight smear, which could indicate the presence of slower migrating, mutant collagen type III molecules. In addition to these findings there was an additional band right above the band representing the a2(V) collagen chains. In situ cyanogen bromide digestion and electrophoresis in the second dimension confirmed that

Digital necrosis in a patient with hypereosinophilic syndrome in the absence of cutaneous eosinophilic vasculitis.

We describe a patient with polyarteritis nodosa who developed a large retroperitoneal hemorrhage from a ruptured suprarenal capsule artery aneurysm. Platinum coils were embolized selectively during arteriography and successfully controlled the aneurysmal bleeding. The vasculitis was controlled ten weeks later but our patient developed anemia and computerized tomography scan revealed a large retrogastric hemorrhage. A further arteriograstric confirmed the presence of a ruptured gastric coronary artery aneurysm. A second embolization was performed with complete success

Embolization of two bleeding aneurysms with platinum coils in a patient with polyarteritis nodosa

Digital necrosis has been exceptionally described in association with the hypereosinophilic syndrome. We report a case of a smoking patient with idiopathic hypereosinophilic syndrome who developed digital arteritis with necrosis. A radial artery biopsy confirmed the vasculitis with inflammatory infiltrates which were predominantly eosinophilic granulocytes. The eosinophils may have an active role in the development of the vasculitis.

Artérite digitale, thrombose et syndrome hyperéosinophilique : une complication exceptionnelle

We present a case of retroperitoneal fibrosis associated with scleroderma (CREST syndrome). The pathogenic implications of such association with systemic vasculitis and connective tissue disease is discussed.

Fibrose rétropéritonéale et sclérodermie. Association fortuite ou mécanisme commun

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Embolization of two bleeding aneurysms with platinum coils in a patient with polyarteritis nodosa

Artérite digitale, thrombose et syndrome hyperéosinophilique : une complication exceptionnelle

Fibrose rétropéritonéale et sclérodermie. Association fortuite ou mécanisme commun