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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 56  |  Issue : 1  |  Page : 38-40

Congenital lacrimal gland fistula


1 Department of Orbit and Oculoplasty, Aravind Eye Hospital, Tirunelveli, Tamil Nadu, India
2 Department of Paediatric Ophthalmology, Aravind Eye Hospital, Tirunelveli, Tamil Nadu, India

Date of Web Publication4-Jun-2018

Correspondence Address:
Dr. V Maneksha
Department of Orbit and Oculoplastics, Aravind Eye Hospital and Post Graduate Institute of Ophthalmology, 1, S N High Road, Tirunelveli Junction, Tirunelveli - 627 001, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/tjosr.tjosr_42_18

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  Abstract 


This is a report of a rare case of a lateral type of unilateral congenital lacrimal gland fistula with no systemic anomalies. A 7-year-old girl born of a second-degree consanguineous marriage presented to us with painless, watery discharge from a site close to the left lateral canthus, since early childhood, which increased on crying. There was no underlying swelling or a history of trauma or surgery. On examination, an opening was found 10 mm lateral to the left lateral canthus. Probe test confirmed a tract leading to the lacrimal gland. There were no other systemic or ocular abnormalities. Examination of her parents and the sibling revealed no abnormality. The fistula was excised under general anesthesia. Postoperatively, the child developed signs of inflammation of the left lacrimal gland which subsided with low-dose steroids and antibiotics. After 1 month, the child was asymptomatic, and the fistula had healed well.

Keywords: Congenital lacrimal gland fistula, fistulectomy in lacrimal gland fistula, lateral type of lacrimal gland fistula


How to cite this article:
Maneksha V, Sabyasachi C, Rengappa R. Congenital lacrimal gland fistula. TNOA J Ophthalmic Sci Res 2018;56:38-40

How to cite this URL:
Maneksha V, Sabyasachi C, Rengappa R. Congenital lacrimal gland fistula. TNOA J Ophthalmic Sci Res [serial online] 2018 [cited 2019 Jul 17];56:38-40. Available from: http://www.tnoajosr.com/text.asp?2018/56/1/38/233732




  Introduction Top


A congenital lacrimal gland fistula is a rare occurrence. It was first described by Beer in 1817.[1] Most cases described are unilateral with intermittent tear flow. We reviewed literature and found reports of only 18 cases. Acquired cases following blepharoplasty, ptosis surgeries, and trauma have been described more often. This is a report of a unilateral congenital lacrimal gland fistula diagnosed and treated by us.


  Case Report Top


A 7-year-old girl, born of a second degree consanguineous marriage, presented to us complaining of discharge, from a hole close to the left lateral canthus, since early childhood. The discharge was colorless and odorless. The child and her parents denied any history of pain or swelling at the site of this hole, discharge of bony spicules from the hole or similar lumps or discharging holes elsewhere in the body. There was no other systemic complaint or any history of ocular or periocular surgeries. There was no history of trauma.

On examination, an opening was noted 10 mm lateral to the left lateral canthus, 2 mm in diameter [Figure 1]. No surrounding granulation tissue or tufts of hair was noted. Palpation revealed no mass, induration or irregularity of the orbital margins. There was no discharge on local pressure. Regional lymph nodes were not enlarged. Probe test revealed a 10 mm fistulous tract going upward and medially with a soft stop. Schirmer's test was normal. There was no other systemic or ocular anomaly. Examination of her parents and her sibling revealed no similar anomaly.
Figure 1: The external opening of the lacrimal gland fistula with pouting of tears

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We decided to excise the fistulous tract under general anesthesia. After insertion of the probe into the fistulous tract, an oval incision was given around the orifice. The fistula was dissected out in toto. The wound was closed in layers with 6-0 polyglactin. She was fine on the first postoperative day. On the 4th postoperative day, she came back to us with pain and upper lid edema [Figure 2]. The wound, however, looked healthy. She was put on oral antibiotics (Cephalexin) and steroids (Prednisolone), which was gradually tapered. The swelling subsided by 3 weeks. After 1 month, she was asymptomatic, and the fistula had healed well [Figure 3].
Figure 2: 4th day post-tract excision

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Figure 3: 1-month post surgery

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  Discussion Top


The lacrimal gland is a tubuloacinar racemose gland which is situated in the superotemporal aspect of the orbit. During development, the large inferolateral duct of the lacrimal gland outside the external canthus may play a role in the development of the lacrimal gland fistula.[2] This was the most probable cause in our case. Several other theories have also been put forth for its development. Krause suggested that a congenital lacrimal gland fistula may represent the openings of one or more of the accessory lacrimal glands.[3] Schornstein opined that the fistula may occur due to a rupture of a congenital cyst.[4] Duke–Elder stated that most of these fistulae were due to an invagination of the surface epithelium at a higher level than usual before the lid folds have formed.[5] So according to him, the ductules arise from the cutaneous epithelium.

Irrespective of its embryological source, all congenital lacrimal gland fistulae have similar features. The patient may remain asymptomatic throughout life or may present with features of infection of the tract. Despite the congenital nature of the fistula, the features become apparent usually in late adulthood because of evaporation of discharge. It can be classified into two types:[6]

  1. Lateral type: The orifice is close to the lateral canthus and is not associated with a tuft of hair. Our patient had a lateral type fistula
  2. Tarsal type: The orifice is near the convex border of the upper tarsal plate and is associated with a tuft of hair.


When the opening is situated near the lid margin, occlusion of the orifice may produce a drop in tear secretion.

A congenital lacrimal gland fistula may be isolated or associated with facial dysmorphism or multiple anomalies.[7]

Investigation of lacrimal gland fistulae includes syringing with fluorescein stained normal saline, probing and imaging with contrast fluid injected into the fistula. Analysis of the fluid from the orifice can also be done.

Various ways of managing this anomaly have also been reported in literature. For openings near the lid margin, it is best to transplant the fistulous tract to the upper fornix.[8] Fistulectomy is best reserved for external fistulae.[6],[7] Fistulectomy in lid margin fistulae may produce dry eye, while transplantation of external fistulae to the upper fornix may produce epiphora. As our patient did not have a lid margin fistula, we went ahead with a fistulectomy.[6],[7],[8]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.


  Conclusion Top


A congenital lacrimal gland fistula is a very rare occurrence. A careful clinical examination and investigations will help us to arrive at the diagnosis, and it can be completely cured by appropriate surgical techniques.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Beer SJ. Doctrine of the Eye Diseases. Vol. 2. Vienna: Heubner and Wolfe; 1817. p. 186.  Back to cited text no. 1
    
2.
Desai HG. Congenital fistula of the lacrimal gland. Br J Ophthalmol 1967;51:206-7.  Back to cited text no. 2
[PUBMED]    
3.
Krause W. On the nodes of the conjunctiva. In: Henle J, Pfeufer CV, editors. Zeitschrift fur Rationelle Medizin. 4th ed. Leipzig & Heidelberg: C. F. Winter'sche Verlags Handlung; 1854. p. 337.  Back to cited text no. 3
    
4.
Schornstein T. On the question of congenital lacrimal fistula. Arch Augenheilkd 1936;109:86-102.  Back to cited text no. 4
    
5.
Duke-Elder S, editor. Congenital deformities. In: System of Ophthalmology. Vol. 3. London: Henry Kimpton; 1964. p. 921-3.  Back to cited text no. 5
    
6.
Blanksma LJ, vd Pol BA. Congenital fistulae of the lacrimal gland. Br J Ophthalmol 1980;64:515-7.  Back to cited text no. 6
[PUBMED]    
7.
Ling WP. Anomalous duct of the lacrimal gland associated with other congenital anomalies. Am J Ophthalmol 1926;9:1-2.  Back to cited text no. 7
    
8.
Putterman AM. Eyelid epiphora secondary to lacrimal gland fistula. Ophthalmic Surg 1980;11:682-5.  Back to cited text no. 8
[PUBMED]    


    Figures

  [Figure 1], [Figure 2], [Figure 3]



 

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Abstract
Introduction
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