J Korean Ophthalmol Soc.  2018 Dec;59(12):1181-1184. 10.3341/jkos.2018.59.12.1181.

A Case of Congenital Lacrimal Ductule Fistula on Lateral Skin to the Right Upper Eyelid

• Han GH
• Chung IK
• Lee DH
• Kim JH
• Seo JW

• KMID: 2428691
• DOI: http://doi.org/10.3341/jkos.2018.59.12.1181

Abstract

PURPOSE
To present a rare case of tear drainage since birth from a lacrimal ductule fistula, which is the first report in the Republic of Korea.
CASE SUMMARY
3-month-old female who presented with discharge of clear fluid from a small skin opening lateral to the right upper eyelid since birth visited the outpatient clinic. The patient was healthy and was receiving no medication. She was born on gestation age 35 weeks and 3 days. No other specific history or other ophthalmic abnormality was found. On examination, a small skin orifice approximately 2 mm diameter and no sign of infection or discoloration was observed. To relieve this symptom, the patient underwent a fistulectomy under general anesthesia. Histopathological examination of the surgical specimen was performed. The patient's symptom subsided postoperatively without recurrence.
CONCLUSIONS
This is the first report in the Republic of Korea of tears draining from a lacrimal ductule fistula since birth and the subsequent clinical treatment procedures.

Keyword

Congenital abnormality; Lacrimal ductule fistula; Lacrimal gland

MeSH Terms

Ambulatory Care Facilities
Anesthesia, General
Congenital Abnormalities
Drainage
Eyelids*
Female
Fistula*
Humans
Infant
Lacrimal Apparatus
Parturition
Pregnancy
Recurrence
Republic of Korea
Skin*
Tears

Figure

• 

  Figure 1 These are the patient's clinical photographs and orbit magnetic resonance images. Clinical photograph demonstrates the skin dimpling (black arrow) with clear fluid and hair on superolateral side of the right lateral canthus (A, B). Postoperative photograph at 12 months after procedure showing a good cosmetic appearance and no recurrence (C). The tract from the right lacrimal gland (white arrow) shows high intensity in the T1-weighted fat suppressed images (D).

• 

  Figure 2 Intraoperative findings. Incision of the skin surrounding the orifice (A), dissection of the fistula (B). There was gross specimen including fistula, measuring about 1.1 cm and 0.2 cm (C).

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  Figure 3 Histological examination. (A) Low power (×40) view of proximal ductule (black arrow) lined by columnar epithelial cells including lacrimal gland tissue (red arrow) (H&E stain, ×40). (B) Low power (×40) view of distal ductule (black arrow) showing squamous epithelial lining and the presence of normal hair follicles (red arrow) (H&E stain, ×40). H&E = hematoxylin-eosin.

Reference

1. Cogen MS, Lewis AR, Kelly AG. Anomalous lacrimal ductule: case report and review. J Pediatr Ophthalmol Strabismus. 1994; 31:327–329.

2. Blanksma LJ, vd Pol BA. Congenital fistulae of the lacrimal gland. Br J Ophthalmol. 1980; 64:515–517.

3. Kural G, Serifoglu A, Ertüre E. A case of aberrant lacrimal gland and fistula. Br J Ophthalmol. 1989; 73:376–377.

4. Leelapatranurak K, Kim JH, Woo KI, Kim YD. Lacrimal ductule fistula: a new complication of cosmetic lateral canthoplasty. Aesthetic Plast Surg. 2013; 37:892–895.

5. Mackenzie W. A practical treatise on the disease of the eye. 4th ed. London: Longman;1855. p. 140.

6. Duke-Elder S. Normal and abnormal development. In : Duke-Elder S, editor. System of Ophthalmology. 1st ed. London: Henry Klimpton;1964. v. 3. Part 2:p. 921–923.

7. Carpenter R, Annamunthodo H. Anomalous lacrimal ductule. Case report and review. Br J Ophthalmol. 1966; 50:159–161.

8. Desai HG. Congenital fistula of the lacrimal gland. Br J Ophthalmol. 1967; 51:206–207.