Literature DB >> 8516621

Widespread vasculopathy with hemolytic uremic syndrome, perimyocarditis and cystic pancreatitis in a young woman with mixed connective tissue disease. Case report and review of the literature.

J Braun1, J Sieper, A Schwarz, F Hiepe, T Lenz, F Keller, H Herbst, A Distler.   

Abstract

A 15-year-old girl had severe Raynaud's phenomenon and arthralgias. A high ANA-IF titer was found and undifferentiated connective tissue disease was diagnosed. After 7 years of moderately flaring disease the patient deteriorated and presented with congestive heart failure, pleuropericardial effusion, hemolytic uremic syndrome, proteinuria and moderate hypertension. Autoantibodies against DNA, Sm-protein, and very high titers against U1RNP were detected. Therapy with high steroid doses, a cyclophosphamide pulse and 4 weeks of plasmaphresis with plasma exchange improved the heart, but not the renal condition. Symptomatic pancreatitis became the dominant problem of a progressively consuming process that resulted in the death of the patient. Post-mortem examination revealed widespread vasculopathy with intima proliferation and only minimal fibrosis involving the kidneys, heart and other main organs, including the pancreas. Taken together, the clinical picture was of an overlap between scleroderma and systemic lupus erythemathosus; the serologic and histopathologic findings suggest a diagnosis of a severe form of mixed connective tissue disease (MCTD).

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Year:  1993        PMID: 8516621     DOI: 10.1007/BF00290331

Source DB:  PubMed          Journal:  Rheumatol Int        ISSN: 0172-8172            Impact factor:   2.631


  35 in total

1.  Heterogeneity of RNP and Sm autoantigens in relation to the cell sources and the activated state of the cells.

Authors:  F Hiepe; K Yamamoto; A Takeuchi; E Apostoloff; T Miyamoto
Journal:  Rheumatol Int       Date:  1990       Impact factor: 2.631

2.  SLE or MCTD and the snurp in the splicing.

Authors:  S Whittingham
Journal:  J Rheumatol       Date:  1990-10       Impact factor: 4.666

3.  Development of a solid phase enzyme immunoassay for the detection of anti-Ro autoantibodies.

Authors:  T Dörner; F Hiepe; F Mielke; S T Kiessig; A Lukowsky; E Apostoloff
Journal:  Scand J Rheumatol       Date:  1991       Impact factor: 3.641

Review 4.  HUS and TTP: variable expression of a single entity.

Authors:  G Remuzzi
Journal:  Kidney Int       Date:  1987-08       Impact factor: 10.612

5.  Mixed connective tissue disease: a subsequent evaluation of the original 25 patients.

Authors:  S H Nimelstein; S Brody; D McShane; H R Holman
Journal:  Medicine (Baltimore)       Date:  1980-07       Impact factor: 1.889

6.  Diagnostic potential of in vivo capillary microscopy in scleroderma and related disorders.

Authors:  H R Maricq; E C LeRoy; W A D'Angelo; T A Medsger; G P Rodnan; G C Sharp; J F Wolfe
Journal:  Arthritis Rheum       Date:  1980-02

7.  Comparison between 3 diagnostic criteria for mixed connective tissue disease. Study of 593 patients.

Authors:  D Alarcón-Segovia; M H Cardiel
Journal:  J Rheumatol       Date:  1989-03       Impact factor: 4.666

8.  The relationship of hypertension and renal failure in scleroderma (progressive systemic sclerosis) to structural and functional abnormalities of the renal cortical circulation.

Authors:  P J Cannon; M Hassar; D B Case; W J Casarella; S C Sommers; E C LeRoy
Journal:  Medicine (Baltimore)       Date:  1974-01       Impact factor: 1.889

9.  Myocarditis in mixed connective tissue disease. Association of myocarditis with antibody to nuclear ribonucleoprotein.

Authors:  P L Whitlow; J N Gilliam; A Chubick; M Ziff
Journal:  Arthritis Rheum       Date:  1980-07

10.  Thrombotic thrombocytopenic purpura and systemic lupus erythematosus.

Authors:  D A Fox; J D Faix; J Coblyn; P Fraser; B Smith; M E Weinblatt
Journal:  Ann Rheum Dis       Date:  1986-04       Impact factor: 19.103

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