Literature DB >> 2777100

Retinal and choroidal ischemic syndrome, digestive tract and renal small vessel hyalinosis, intracerebral calcifications and phenotypic abnormalities: a new family syndrome.

G van Effenterre1, J Haut, A Brezin, Y Le Mer, J C Rambaud, A Galian, G Touchard, E Rothschild.   

Abstract

A new family syndrome is described that affected three of seven siblings and another patient who had been abandoned at birth but came from the same area of France. All four patients were young women with a very peculiar phenotype, poikiloderma and greying of the hair, and idiopathic non-arteriosclerotic cerebral calcifications. Pathological studies demonstrated small-vessel hyalinosis due to basal membrane thickening, mainly in the digestive tract, kidneys and calcified areas of the brain. The clinical and biological expressions of these vascular changes varied. Peripheral retinal ischemic syndrome and chorioretinal scars were found in the ocular fundi of three patients. Malabsorption and protein-losing enteropathy was the main problem in all four, and was the cause of one patient's death. A subarachnoid hemorrhage due to a right sylvian aneurysm also occurred in two of the three sisters and was lethal for one. Nephropathy with renal failure and systemic hypertension is the major problem of the two surviving patients.

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Year:  1989        PMID: 2777100     DOI: 10.1007/BF02169404

Source DB:  PubMed          Journal:  Graefes Arch Clin Exp Ophthalmol        ISSN: 0721-832X            Impact factor:   3.117


  16 in total

1.  Cerebroretinal vasculopathy. A new hereditary syndrome.

Authors:  M G Grand; J Kaine; K Fulling; J Atkinson; S B Dowton; M Farber; J Craver; K Rice
Journal:  Ophthalmology       Date:  1988-05       Impact factor: 12.079

Review 2.  Basal lamina layering in diabetes mellitus. Evidence for accelerated rate of cell death and cell regeneration.

Authors:  R Vracko
Journal:  Diabetes       Date:  1974-02       Impact factor: 9.461

3.  Electron microscopical study of Coat's disease.

Authors:  R Tripathi; N Ashton
Journal:  Br J Ophthalmol       Date:  1971-05       Impact factor: 4.638

4.  Light and electron microscopic studies on the sclerotic changes in human central retinal arteries. I. The "crossing phenomenon".

Authors:  T Kimura
Journal:  Nippon Ganka Gakkai Zasshi       Date:  1970-09

5.  [A concept of "capillarosclerosis retinae". I. Pathological changes of the capillaries in the retina and the optic nerve head in senescence and in benign hypertension].

Authors:  H Ikui; Y Tominaga; H Inomata; S Uga; T Okada
Journal:  Nippon Ganka Gakkai Zasshi       Date:  1968-05-10

6.  Retinal vascular pattern in familial exudative vitreoretinopathy.

Authors:  H Miyakubo; K Hashimoto; S Miyakubo
Journal:  Ophthalmology       Date:  1984-12       Impact factor: 12.079

7.  The clinical picture of Eales' disease.

Authors:  M Spitznas; G Meyer-Schwickerath; B Stephan
Journal:  Albrecht Von Graefes Arch Klin Exp Ophthalmol       Date:  1975

8.  Pathophysiology of hypertensive retinopathy.

Authors:  M O Tso; L M Jampol
Journal:  Ophthalmology       Date:  1982-10       Impact factor: 12.079

9.  Digestive tract and renal small vessel hyalinosis, idiopathic nonarteriosclerotic intracerebral calcifications, retinal ischemic syndrome, and phenotypic abnormalities. A new familial syndrome.

Authors:  J C Rambaud; A Galian; G Touchard; L Morel-Maroger; J Mikol; G Van Effenterre; J P Leclerc; Y Le Charpentier; J Haut; C Matuchansky
Journal:  Gastroenterology       Date:  1986-04       Impact factor: 22.682

10.  Parafoveal retinal telangiectasis. Light and electron microscopy studies.

Authors:  W R Green; H A Quigley; Z De la Cruz; B Cohen
Journal:  Trans Ophthalmol Soc U K       Date:  1980-04
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  3 in total

1.  Protein losing enteropathy in critically ill adult patients with burns: a preliminary report.

Authors:  Balasubramanian Venkatesh; Jenny Gough; David R Ralston; Michael Muller; Stuart Pegg
Journal:  Intensive Care Med       Date:  2003-11-21       Impact factor: 17.440

2.  Familial vascular retinopathy. A preliminary report.

Authors:  C W Storimans; J A Oosterhuis; M J van Schooneveld; P J Bos; P D Maaswinkel-Mooy
Journal:  Doc Ophthalmol       Date:  1990-10       Impact factor: 2.379

3.  Fatal gastrointestinal bleeding in a case report of Coat's plus syndrome.

Authors:  Mohammed Jeraq; Valerie Armstrong; Grigoriy Klimovich; Krishnamurti Amrit Rao; Patricia Byers
Journal:  Int J Surg Case Rep       Date:  2019-12-16
  3 in total

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