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ARTICLE
Year : 1966  |  Volume : 14  |  Issue : 3  |  Page : 138-140

Gougerot Sjogren syndrome- treated with parotid duct transplantation


Department of Ophthalmology, Medical College and Hospital, Nagpur, India

Date of Web Publication16-Jan-2008

Correspondence Address:
Ishwarchandra
Department of Ophthalmology, Medical College and Hospital, Nagpur
India
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Source of Support: None, Conflict of Interest: None


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How to cite this article:
Ishwarchandra, Mulak J S. Gougerot Sjogren syndrome- treated with parotid duct transplantation . Indian J Ophthalmol 1966;14:138-40

How to cite this URL:
Ishwarchandra, Mulak J S. Gougerot Sjogren syndrome- treated with parotid duct transplantation . Indian J Ophthalmol [serial online] 1966 [cited 2020 Aug 3];14:138-40. Available from: http://www.ijo.in/text.asp?1966/14/3/138/38645

Duke-Elder (1952) defines the above syndrome as a failure in lacrimal secre­tion associated with an atrophic and cirrhotic condition of the lacrimal gland, occurring as a part of a general systemic disturbance of unknown origin.

The first to associate the salivary deficiency with a deficiency of lacrimal secretion was Fuchs (1910), but the first to establish its widespread nature of affection was Gougerot (1926). In 1933, Sjogren, corroborated and aug­mented the findings of several physi­cians to establish the syndrome with its systemic implications on a sound basis.

Recently it has been noted to be associated with collagen diseases es­pecially lupus-erythematosus, sarcoi­dosis, Mikulicz's disease, Felty's dis­ease, and with lymphadenoid goitre and even with hepatic and gall bladder dis­eases. Hence, it is suggested that there is enough evidence to show that the condition of kerato-conjunctivitis sicca occurs in association with a variety of conditions of probably inter-related aetiology and pathology.


  Clinical Picture Top


Though not uncommon, this syn­drome is not well-known. Kerato-con­junctivitis sicca is more often seen by and is of greatest interest to the oph­thalmologist than the physian, but is often overlooked amongst the many cases of conjunctivitis during a busy out-patient clinic. The condition is usually met with in women after meno­pause, is usually bilateral, of slow and insidious onset, associated with in­volvement of other extraocular struc­tures.

Local signs and symptoms consist of intense photophobia, dryness of the conjunctiva even during weeping. The conjunctiva is congested and dry and there is presence of thick stringy mu­coid discharge in the lower fornix which can be drawn into long fila­ments containing mucus with epithelial cells. Corneal changes are filamentary keratitis, rarely ulcers, leucomas and pannus formation which may be com­plete.


  Case Report Top


G.B., 48 years Hindu female, was transferred to the eye ward on 13-12-1962 from the medical ward. She was treated as a case of rheumatoid arthritis and was complaining of pain in all the joints for the last three months for which she was receiving oral corticosteroids. There was also loss of appetite and insomnia. She was transferred with the complaints of red­ness of both eyes off and on, pain in her eyes, dryness of eyes, diminished vision of two months duration.


  Systemic Examination Top


Systemic examination revealed fis­sures at the angles of the mouth, a fissured tongue, fullness over the paro­tid region [Figure - 1], dryness of the skin, loss of teeth, swelling of the joints especially of the carpometacarpals. [Figure - 1]. The movements were painful and restricted.


  Ocular Findings Top


Lids were normal and , palpebral fis­sure narrowed as the patient had mark­ed photophobia.

Conjunctiva: There was no evidence of any scarring of the palpebral con­junctiva. The bulbar conjunctiva was markedly dry and congested with col­lection of mucoid shreds in the inferior fornix.

Cornea: Cornea showed superficial vascularisation all around upto a dis­tance of 1-2 mm. from the limbus. The cornea on the right side was hazy and showed a central small adherent leucoma. The cornea on the left side was hazy especially in the central part. Cornea in both the eyes showed vesi­cular and filamentary keratitis. The corneal sensation was slightly impaired.

The intra-ocular tension was normal in both the eyes.

Vision: In the right eye it was counting fingers from one meter, whereas with the left eye she could count fingers from 4 meters.

In view of the age, ocular findings and associated rheumatoid arthritis a diagnosis of Sjogren's syndrome was arrived at.

Investigations done:

Blood :
Haemoglobin-65 per cent.

Total leucocyte count-8900/cmm.

Differential count-Poly : 52;

Lympho : 48.

Erythrocyte sedimentation rate-50 mm. Ist hour (Wintrobe).

Liver function tests: Within normal limits.

Basal metabolic rate : + 10.

Conjunctival smear:
Epithelial cells were not to be seen. Mucoid shreds stained homogeniously with eosin.

Corneal staining with fluorescin: Ne­gative.

Schirmer's Test : Less than 0.5 cm. soaked even after the onion test.

The patient had great discomfort due to her ocular and associated ge­neral complaints. Rheumatoid arthri­tis would get aggravated on stappage of oral cortisone therapy.

In the left eye occlusion of the punc­tum with tying of the canaliculi was done.

For the right eye she was referred to the Department of Plastic Surgery with a view to do a parotid duct im­plantation in the right eye, which was getting worse day by day. A preli­minary test with a chewing gum, one three times a day for one week was carried out to determine if there was any increase in parotid secretion. Since there was definite increase, she was advised an operation and an implan­tation of the parotid duct was success­fully carried out on 16-1-1963 by Prof. Balkrishnan. The post-operative period was uneventful and the patient was discharged on 11-2-1963 with a moist right eye.

The follow up on 1-4-64 revealed that although the patient complained of slight photophobia, there was a marked subjective improvement. The pannus was still present, but the blood vessels were not so much engorged. The cornea was smoother and brighter. There was no oversecretion of the parotid, which was just enough to keep the conjunctiva moist. Vision: Right Eye-counting fingers 3 meters, Left Eye--6/18.


  Discussion Top


The association of hypothyroidism, lymphadenoid goitre, and raised serum cholesterol with Sjogren's syndrome (Gifford et al, 1943 and Gruber, 1956) is interesting. However, our case showed normal basal metabolic rate. John Me Lanachen (1956) noting the occurrence of disease almost exclusive­ly in women (89.4 per cent) strongly suggests an endocrinal origin with liver dysfunction. An inability of the liver to utilise vitamin A may explain the changes in the epithelial structures. In our case the values of liver function tests were within normal limits.

J. H. Ramage and W. F. Kinnear (1956) described six cases of kerato­conjunctivitis sicca with a related paro­tid involvement; two cases had asso­ciated rheumatoid arthritis. The pre­set case had associated rheumatoid arthritis.


  Summary Top


A case report of Sjogren's syndrome with typical ocular findings and asso­ciated systemic involvement (rheuma­toid arthritis) is reported, in a lady in her menopause.

Oral cortisone therapy did not re­lieve her ocular complaints although she was temporarily relieved of the painful movements of joints.

Parotid duct transplantation mark­edly improved the clinical picture in the right eye.

A simple occlusion of the puncta and tying of canaliculi have also been instrumental in improving the clinical picture in the left eye. Moreover, be­cause of the diminished parotid secre­tion there is no epiphora in the right eye. Hence it is suggested that this procedure be given a trial in a larger number of cases.


  Summary Top


A case of Sjogren's syndrome with associated rheumatoid arthritis is re­ported.

Occlusion of puncta and tying of canaliculi in one eye and a parotid duct transplantation into the conjunctiva in the other eye, considerably ameliorated the condition with improvement in vision.


  Acknowledgement Top


Our thanks are due to Prof. Bal­krishna, for performing the parotid duct transplant.[8]

 
  References Top

1.
Barrie, R. J. and Stevenson. C. J. (1957): Brit. Jour. Ophthal. 41: 153.  Back to cited text no. 1
    
2.
Duke-Elder (1952): Textbook of Oph­thalmology. Lond., Henry Kimpton. Vol. 5. pp. 5244.  Back to cited text no. 2
    
3.
Edies and Thompson (1955): Brit. Jour. Ophthal. 39: 90.  Back to cited text no. 3
    
4.
Heaton, J. M. (1959): Brit. Med. .Jour. 1: 466.  Back to cited text no. 4
    
5.
Henderson. J. W. (1950): Amer. Jour. Ophthal. 33: 197.  Back to cited text no. 5
    
6.
Joachim Weber (1957): Year Book of Ophthalmology. Chicago. Year Book Publishers. pp. 86.  Back to cited text no. 6
    
7.
John Mclenachan (1956): Tr. Ophthal. Soc. U.K. 76: 413.  Back to cited text no. 7
    
8.
Ramage, J. H. and Kinnetar. W. F. (1956): Brit. Jour. Ophthal. 40: 416.  Back to cited text no. 8
    


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  In this article
Clinical Picture
Case Report
Systemic Examination
Ocular Findings
Discussion
Summary
Summary
Acknowledgement
References
Article Figures

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