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   Table of Contents      
CASE REPORT
Year : 2018  |  Volume : 66  |  Issue : 8  |  Page : 1220-1223

Live encysted Thelazia callipaeda presenting as a lump adjacent to the right lacrimal sac in a 42-year-old female: A rare case report


1 Department of Ophthalmic Plastic and Reconstructive Surgery, Sri Sankaradeva Nethralaya, Guwahati, Assam, India
2 Department of Ocular Pathology, Uveitis and Neuro-Ophthalmology Services, Guwahati, Assam, India
3 Department of Ophthalmology, Sri Sankaradeva Nethralaya, Guwahati, Assam, India

Date of Submission25-Mar-2018
Date of Acceptance23-Apr-2018
Date of Web Publication23-Jul-2018

Correspondence Address:
Dr. Jayanta Kumar Das
Department of Ophthalmic Plastic and Reconstructive Surgery, Sri Sankaradeva Nethralaya, 96, Basistha Road, Beltola, Guwahati, Assam
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijo.IJO_317_18

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  Abstract 


Thelazia callipaeda or the oriental eye worm infects a wide variety of hosts including dogs, cats, foxes, rabbits, and humans through the Drosophila flies. We report here a case of a 42-year-old female who presented with pain, redness, and cystic swelling in the right lacrimal sac area for 8 months. Ocular examination was within normal limits. Excision biopsy of the cyst revealed a live worm. In humans, the worm is usually found in the conjunctival sac, lacrimal canaliculi, or the lacrimal sac leading to a disease which can be subclinical or symptomatic. Thus, we report a rare presentation of thelaziasis, that is, periocular involvement. This case presentation is first of its kind in the Indian literature.

Keywords: Human thelaziasis, oriental eyeworm, periocular thelaziasis, Thelazia callipaeda, zoonoses


How to cite this article:
Das JK, Das D, Deshmukh S, Gupta K, Tomar SS, Borah E. Live encysted Thelazia callipaeda presenting as a lump adjacent to the right lacrimal sac in a 42-year-old female: A rare case report. Indian J Ophthalmol 2018;66:1220-3

How to cite this URL:
Das JK, Das D, Deshmukh S, Gupta K, Tomar SS, Borah E. Live encysted Thelazia callipaeda presenting as a lump adjacent to the right lacrimal sac in a 42-year-old female: A rare case report. Indian J Ophthalmol [serial online] 2018 [cited 2019 Oct 20];66:1220-3. Available from: http://www.ijo.in/text.asp?2018/66/8/1220/237332



In 1910, Railliet and Henry first described the arthropod-borne zoonosis by a nematode, Thelazia callipaeda.[1] In 1917, the first human case was reported by Stucky in China.[2]T. callipaeda is also known as the “Oriental eye worm” because it is mainly confined to South Asian countries.[3] The two important species causing infection in humans are T. callipaeda and Thelazia californiensis. T. callipaeda is prevalent in India, Thailand, China, Korea, Japan, Russia, Northern Europe, and Southern Italy. However, T. californiensis is exclusively seen in the United States.[4] The transmission is through the tear-seeking flies, Phortica okadai and Phortica variegate. It has been found that the Musca domestica (Common fly) is not a vector of T. callipaeda.[5]


  Case Report Top


A 42-year-old female, farmer and cattle breeder by occupation, presented with chief complaint of swelling, medial to the medial canthus of the right eye associated with mild pain, and redness for the past 8 months [Figure 1]a. The patient complained of minimal watering from the right eye which was occasional with no history of discharge. On clinical examination, her best-corrected visual acuity in both eyes was 6/6 and N6. Slit-lamp biomicroscopy showed normal anterior segment with normal intraocular pressure. Fundus examination was within normal limits. Syringing showed a patent lacrimal system. There was no associated history of any systemic illness.
Figure 1: (a) Clinical photograph of the patient showing swelling medial to the right side medial canthus. (b) Computed tomography scan showing a mass of dimension 1.11 cm × 0.78 cm over the lacrimal sac area

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There was a 4 cm × 2 cm swelling over the right lacrimal sac area. On palpation, it was nontender and cystic in consistency with restricted mobility both sideways and vertically. The overlying skin had normal texture and was freely mobile. Thus, a differential diagnosis of dermoid, mucocele, or soft-tissue tumor was made. A computed tomography scan of orbit was advised to define the limits of swelling and to image the bony architecture and paranasal sinuses [Figure 1]b. It showed a mass lesion of dimension 1.11 cm × 0.78 cm over the lacrimal sac area which was isodense with the surrounding soft tissue with smooth margins. Complete blood count including red blood cell count, white blood cell count, and differential leukocyte count were within normal limits.

Under local anesthesia, soft-tissue dissection was done and it was found that the cystic lesion was not adherent to the lacrimal sac. Excision biopsy of the cystic lesion was done and sent for histopathological examination. On exploration of the cyst, a creamy white thread-like live worm was found [Figure 2]a and [Figure 2]b. The worm showed active motility when transferred to a container containing normal saline and was video documented.
Figure 2: (a and b) Dissection of the cyst showing creamy white thread-like worm

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Histopathological examination of the cyst revealed granulomatous inflammation. Morphologically, the worm was creamy white in color and 36 cm long and 1.75 mm wide in dimension. Microscopically, the worm had a buccal opening [Figure 3]a with upper alimentary canal and cuticular striation all over the surface [Figure 3]b. The middle body segment was filled with eggs [Figure 3]c. The tail was blunted. Based on the morphological features, the worm was identified as adult female T. callipaeda.
Figure 3: (a) Microscopic view of the worm showing buccal opening (white arrow), (b) cuticular striation (white arrow), and (c) middle body segment filled with eggs (white arrow)

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The patient is under constant follow-up. In the recent visit, the incision site had completely healed with minimal scarring and no recurrence. The syringing was patent with normal tear meniscus height [Figure 4].
Figure 4: Clinical photograph of the patient at 1-year follow-up showing healed incision with minimal scarring and no recurrence

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  Discussion Top


Review of the Indian literature shows that 10 cases of ocular thelaziasis have been reported all over the country. All these cases are of ocular thelaziasis, but ours is the first case report describing the periocular thelaziasis.[1] [Table 1] shows epidemiological and geographical distribution of human thelaziasis in India. Considering the global scenario, more than 250 cases of T. callipaeda have been reported in the literature. Maximum number of cases have been reported from China, Japan, India, Russia, Thailand, and Korea.[6]
Table 1: Epidemiological and geographical distribution of human thelaziasis in India

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The patient was a farmer and a cattle breeder by occupation, which is a predisposing factor for thelaziasis. Occupational history is important because the source of infection for thelaziasis is an infected animal (cows, horses, and dogs – definitive host) or human (accidental host) who harbors the first-stage larvae in the lacrimal secretions. The flies which are the intermediate host, feed on the infected lacrimal secretions, and ingest these larvae. These larvae then develop into infective third-stage larvae in the intermediate host. This stage is infective to humans. These infective larvae are transmitted to another susceptible host by the flies when it feeds on their lacrimal secretions which then develop into adult worms. Humans are considered accidental hosts.[7] Human thelaziasis usually presents with mild signs and symptoms such as foreign body sensation, epiphora, follicular hypertrophy, and less often with severe signs and symptoms such as keratitis, photophobia corneal opacities, ectropion, and secondary bacterial infections.[8]

The first case of thelaziasis was reported in India from Salem in 1948.[9] Subsequently, nine cases were reported from various parts of the country including Himachal Pradesh, Manipur, Assam, Kerala, and Karnataka. Maximum number of cases have been reported from the Northeastern part of the country, that is, seven cases, four from Assam and three from Manipur.[1],[10],[11],[12],[13] This could be attributed to the tropical climate with hilly terrain and longer monsoon period, which is favorable for the growth of arthropods, and subsequently, the vector-borne diseases.[14]

Preventive measures include use of bed nets, maintenance of personal hygiene, and keeping surroundings clean to control the vector population responsible for the transmission of infection. Since the main reservoir of Thelazia are the domestic animals, maintaining the hygiene and cleanliness of them would go a long way in controlling the spread within population of domestic animals which eventually become a source of infection to humans.

Even though 10 cases of thelaziasis have been reported in India, only one study is available regarding its epidemiology.[1],[3],[9],[10],[11],[12],[13],[14],[15] Thus, an epidemiological study should be undertaken to study the true picture of ocular thelaziasis in India.


  Conclusion Top


Ocular thelaziasis can manifest as a lacrimal sac mass.

Acknowledgment

The authors would like to thank Sri Kanchi Sankara Health and Educational Foundation, Guwahati, India.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Singh RM, Singh HL, Gurumayum P, Gambhir Singh RK. Human ocular thelaziasis: A case report from Manipur, India. Ann Trop Med Public Health 2015;8:13-5.  Back to cited text no. 1
  [Full text]  
2.
Leiper RT. Thelaziasis in man: A summary of recent reports on “circumocular filariasis” in Chinese literature, with a note on the zoological position of the parasite. Br J Ophthalmol 1917;1:546-9.  Back to cited text no. 2
    
3.
Krishnachary PS, Shankarappa VG, Rajarathnam R, Shanthappa M. Human ocular thelaziasis in Karnataka. Indian J Ophthalmol 2014;62:822-4.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Anderson RC. Nematode Parasites of Vertebrates: Their Development and Transmission. 2nd ed. Guilford, UK: CABI Publishing; 2000. p. 404-5.  Back to cited text no. 4
    
5.
Otranto D, Lia RP, Cantacessi C, Testini G, Troccoli A, Shen JL, et al. Nematode biology and larval development of Thelazia callipaeda (Spirurida, Thelaziidae) in the drosophilid intermediate host in Europe and China. Parasitology 2005;131:847-55.  Back to cited text no. 5
    
6.
Koyama Y, Ohira A, Kono T, Yoneyama T, Shiwaku K. Five cases of thelaziasis. Br J Ophthalmol 2000;84:441.  Back to cited text no. 6
    
7.
Hong ST, Park YK, Lee SK, Yoo JH, Kim AS, Chung YH, et al. Two human cases of Thelazia callipaeda infection in Korea. Korean J Parasitol 1995;33:139-44.  Back to cited text no. 7
    
8.
Shen J, Gasser RB, Chu D, Wang Z, Yuan X, Cantacessi C, et al. Human thelaziosis – A neglected parasitic disease of the eye. J Parasitology 2006;92:872-6.  Back to cited text no. 8
    
9.
Friedmann M. Thelazia callipaeda, the oriental eye worm. Antiseptic 1948;45:620-6.  Back to cited text no. 9
    
10.
Singh TS, Singh KN. Thelaziasis: Report of two cases. Br J Ophthalmol 1993;77:528-9.  Back to cited text no. 10
    
11.
Mahanta J, Alger J, Bordoloi P. Eye infestation with Thelazia species. Indian J Ophthalmol 1996;44:99-101.  Back to cited text no. 11
[PUBMED]  [Full text]  
12.
Nath R, Narain K, Saikia L, Pujari BS, Thakuria B, Mahanta J, et al. Ocular thelaziasis in Assam: A report of two cases. Indian J Pathol Microbiol 2008;51:146-8.  Back to cited text no. 12
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13.
Das D, Islam S, Bhattacharjee H, Deka A, Yambem D, Tahiliani PS, et al. Parasitic diseases of zoonotic importance in humans of Northeast India, with special reference to ocular involvement. Eye Brain 2014;6:1-8.  Back to cited text no. 13
    
14.
Handique AK, Tamuli A, Khan AM. Ocular thelaziasis in a 7-month-old infant. Indian J Med Microbiol 2014;32:84-6.  Back to cited text no. 14
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15.
Sharma A, Pandey M, Sharma V, Kanga A, Gupta ML. A case of human thelaziasis from Himachal Pradesh. Indian J Med Microbiol 2006;24:67-9.  Back to cited text no. 15
[PUBMED]  [Full text]  


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