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   Table of Contents      
CASE REPORT
Year : 1992  |  Volume : 40  |  Issue : 4  |  Page : 115-116

Ipsilateral Coat's reaction in the eye of a child withen coup de sabre morphoea-A case report


1 Department of Ophthalmology, Mahatma Gandhi Institute of Medical Sciences, Sevagram, Wardha, MS-442 102, India
2 Department of Medicine, (Dermatology Unit), Mahatma Gandhi Institute of Medical Sciences, Sevagramn, Wardha- Maharashtra-442 102, India

Correspondence Address:
A S Neki
Lecturer in Ophthalmology, M.G. Institute of Medical Sciences, Sevagram, Wardha, Maharashtra-442 102
India
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Source of Support: None, Conflict of Interest: None


PMID: 1300302

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  Abstract 

A female child with post-traumatic en coup de sabre type of morphoea (fronto-parietal circumscribed scleroderma) involving the left side of the forehead and face, who developed a Coat's disease-like fundus picture over the following two years, is being reported.

Keywords: en coup de sabre morphoea, Coat′s disease


How to cite this article:
Neki A S, Sharma A. Ipsilateral Coat's reaction in the eye of a child withen coup de sabre morphoea-A case report. Indian J Ophthalmol 1992;40:115-6

How to cite this URL:
Neki A S, Sharma A. Ipsilateral Coat's reaction in the eye of a child withen coup de sabre morphoea-A case report. Indian J Ophthalmol [serial online] 1992 [cited 2020 May 28];40:115-6. Available from: http://www.ijo.in/text.asp?1992/40/4/115/24383


  Introduction Top


Morphoea is a disorder of unknown cause in which there is localised sclerosis of the skin. Morphoea differs from systemic scleroderma in being localised and reversible [1]. It may be of the circumscribed, linear or, frontoparietal (en coup de sabre types [1]sub . Etiologies speculated include: trauma , post radiotherapy [1], pregnancy [1], phenylketonurea [3], autoim­munity [1] etc. Morphoea has been associated with several systemic and a few ocular conditions. The latter include: increased corneal thickness [4]sub , cataract [5]sub , iritis and iris atrophy [6]. To the best of our knowledge, this is the first report of the association of Coat's reaction with this type of morphoea [Figure - 1].


  Case report Top


A six year old female child presented with a diminution of vision in the left eye for two years, three months following a fall from a swing. The child had fallen on the leftside of her face, and had spontaneous loss of hair from the left frontoparietal scalp.

There was a mild hematoma, but no laceration. Over the next three months, the development of a scar-like depression of the left frontal and supraorbital areas and an apparently painless diminution of vision to the left eye were noticed. There was no history of headaches or seeing floaters, nor any family history of significant ocular problems. The wide skin lesion running from beyond the left orbit till the bregma with mild left facial hemiatrophy, was diagnosed as post-traumatic en coup de sabre type of morphoea [Figure - 1].

The IQ was normal. The visual acuity was 6/9 (OD) and 6/36 (OS). The anterior segments of both eyes and posterior segments of the right eye were normal. The left eye fundus however showed large posterior pole consolidated exudates (4 disc diameters in size) with chorioretinal degeneration and pigment clumping at the margins. Veins were markedly tortuous and dilated, with several veins showing beading and fusiform aneurysms in the inferotemporal quadrant. The superior and inferior peripheral fundus showed clusters of multiple small and glistening fleck exudates. The optic disc was normal. Considering the typical fundus picture, unilaterality and age of presentation, the left ocular condition was diagnosed as Coat's reaction [7].


  Discussion Top


Frontoparietal lesions of morphoea (en coup de sabre) have been reported in all age groups [1], especially children [8]. Trauma may be the provocative factor . Vascular anomalies have been reported associated with this condition [1],[9],as also with Coat's disease, in which exudative retinopathy is known to be sub­sequent to retinal vascular anomalies and consequent leakage from impaired blood vessels [7].

It is tempting to hypothesize that post-traumatic morphoea in the case at hand in some way depended in its manifestation upon vascular anomalies; the same kind of vascular anomalies was possibly respon­sible for Coat's disease in the eye. Alternatively it could be just a coincidental association. Never­the-less, the appearance of the two conditions in a single case, ipsilaterally, deserves clinical attention.

 
  References Top

1.
Rook A, Wilkinson DS, et al., Textbook of Dermatology Vol.2. 4th edn., Oxford University Press, Bombay 1987 p.1334.  Back to cited text no. 1
    
2.
Christiansen HB, et al., Localised scleroderma: a clinical study of two hundred thirty five cases, Arch. Dermatol 1956, 74, 629-639.   Back to cited text no. 2
    
3.
Kornreich H., et al., Phenylketonurea and Scleroderma, J. Pediatr, 1968, 73, 571-575.  Back to cited text no. 3
    
4.
Serup J. et al., Increased central corneal thickness in localised scleroderma (morphoea). Metab pediatr Syst Ophthalmol, 1985, 8 (2-3): 11-14.  Back to cited text no. 4
    
5.
Wallis C, et al., Cataracts, alopecia and sclerodactyly: a previously apparently undescribed ectodermal dysplasia syndrome on the island of Rodrigues, Am J Med Genet 1989 Apr; 32 (4): 500-503.  Back to cited text no. 5
    
6.
Segal P. et al., Ocular changes in linear scleroderma, Am J Ophthalmol 1961, 51, 807-813.  Back to cited text no. 6
    
7.
Duke-Elder S: System of Ophthalmology, Vol. X. Henry Kimpton, London 1967. p. 164-179.  Back to cited text no. 7
    
8.
Domenia R. et al., (Localised Scleroderma in childhood) Pediatr Med Chir, 1988 May-June; 10(3): 297-302 (Ita), (Eng abs).  Back to cited text no. 8
    
9.
Kovalev VM, et al., ( A rare combination of linear-scleroderma with cavernous hemangioma of the glans penis) 1988; (6): 60-61 (Rus) (Eng abs).  Back to cited text no. 9
    


    Figures

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