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Iran J Parasitol: Vol. 15, No. 1, Jan-Mar 2020, pp.147-152 Iran J Parasitol Tehran University of Medical Open access Journal at Iranian Society of Parasitology Sciences Public a tion http:// ijpa.tums.ac.ir http://isp.tums.ac.ir http://tums.ac.ir Case Report Ocular Dirofilariasis after Clinically Manifested Subcutaneous Migration of the Parasite: A Case Report František ONDRISKA 1,2, Vojtech BOLDIŠ 1, Marta STANISLAVOVÁ 3, *Daniela ANTOLOVÁ 4, Martina MITERPÁKOVÁ 4, Anton HANÁČEK 5, Soňa VEŠPEROVÁ 6, Ivan JANČOVIČ 7 1. Department of Laboratory Testing Methods in Health Care, Faculty of Health Care and Social Work, Trnava University, Trnava, Slovakia 2. Department of Parasitology, Medirex (Ltd), Bratislava, Slovakia 3. Department of Ophthalmology, University Hospital of Bratislava, Bratislava, Slovakia 4. Department of Zoonotic Diseases, Institute of Parasitology SAS, Košice, Slovakia 5. Department of Ophthalmology, Faculty Hospital of Trnava, Trnava, Slovakia 6. Department of Infectology, Faculty Hospital of Trnava, Trnava, Slovakia 7. ENT Outpatient Department, Bánovce nad Bebravou, Slovakia Received 19 Jul 2019 Abstract Accepted 11 Sep 2019 Dirofilaria repens is the causative agent of human subcutaneous or, less often, ocular dirofilariasis. The work presents a rare case of ocular dirofilariasis manifested by previous subcutaneous migration accompanied by severe headache symptoms. In Keywords: February 2017, a 58-yr-old man from Trnava region, western Slovakia, noticed red Dirofilariasis; and itchy stripes on his left leg. Inflamed but painless stripes disappeared and Dirofilaria repens; showed up again every 5–7 days, migrating gradually towards the head. Approxi- Subcutaneous migra- mately one month after the first skin´s alterations, strong pain in the left temple, tion; with the swelling of the left face and the enlargement of mandibular lymph nodes Ocular infection; appeared. Several days later, the patient felt excruciating pain of the right eyeball accompanied by strong nausea and subsequent vomiting. Ocular examination re- Slovakia vealed the presence of a live worm in the subconjunctival space and morphological *Correspondence and molecular analyses of extracted helminth confirmed D. repens as etiological Email: agent of the infection. According to clinical manifestation of the infection, it could [email protected] be supposed that ocular form of the disease was the result of the migration of a parasite through the subcutaneous tissues. Moreover, a rare phenomenon of lym- phadenitis of underlying lymph nodes and the swelling of left face accompanied the migration. 147 Available at: http://ijpa.tums.ac.ir Ondriska et al.: Ocular Dirofilariasis after Clinically Manifested … Introduction number of different helminthic cm below the crotch. Within next five days, species can affect human eye. Some of the stripes disappeared and showed up again, A them, like Wuchereria bancrofti, Brugia first on the right side of the back and later un- malayi, Loa loa, and zoonotic Onchocerca der the scapula. The lesions were itchy, elon- volvulus and Angiostrongylus cantonensis, represent gated, ascended about 0.5 mm above skin´s public health problem in developing countries; surface and reached about 2 cm in length. The the spread of other, e.g. Thelazia callipaeda or nodules under the scapula were not pruritic Dirofilaria repens, are connected with climate and reminded of purulent ulcer. Approximate- and environmental changes (1). ly one month after the first skin alterations Dirofilariasis is considered fast spreading had been observed; strong pain in the left disease with the causative agent being trans- temple, swelling of the left half of the face and mitted by mosquitoes of various species. The the enlargement of the mandibular lymph most frequently detected Dirofilaria-species nodes appeared. Several days later, the patient parasites in humans is D. repens, which causes had felt excruciating, one-hour lasting pain of subcutaneous and ocular form of the infection the right eyeball, which ceased and reappeared (2). Since 2007, when the first autochthonous next day. The pain was accompanied by strong case of human dirofilariasis in Slovakia was nausea and subsequent vomiting. recorded, a total of 16 19 human cases caused Clinical examination at the Clinic of Oph- by D. repens have been reported in Slovakia; thalmology revealed the presence of a live four of them of ocular form (3,4; Antolová, worm in the subconjunctival space (Fig. 1A). Miterpáková and Ondriska – personal com- The patient agreed with all examinations and munication). Concerning ocular dirofilariasis, signed the informed content. No identifying adult parasites are often present subconjuncti- data are presented in the paper. The study and vally, intravitreally or directly in orbit (5). subsequent publication of results was ap- Presence of the parasite in eye structures is proved by the Ethics Committee of Institute clinically manifested mainly by itching and in- of Parasitology of SAS (No. EK 04/2015). flammatory reaction, which may result in im- From the time of the first dermal symptoms paired vision of the patient. to the appearance of the worm in the eye it Dirofilarial infections are relatively often ac- took about six weeks. Common laboratory companied with the parasite subcutaneous findings as well as the numbers of erythro- migration, and even cutaneous larva migrans cytes and leucocytes were within the normal syndrome was recorded in Slovak patient re- range, while the absolute count of eosinophils cently (6). Herein we present a rare and re- was slightly increased to 0.69 x 10^9/L (refer- markable case of ocular dirofilariasis manifest- ence value 0.0 – 0.50 x 10^9/L). The extracted ed by foregoing subcutaneous migration ac- helminth was whitish, thin and measured companied by severe headache symptoms. about 8.3 cm in length (Fig. 1B). Based on microscopic examination, the presence of Case presentation multi-layered cuticle with external longitudinal ridges was observed (Fig. 1C). Preliminary di- In February 2017, a 58-yr-old man from agnosis of dirofilariasis caused by D. repens Trnava region, western Slovakia, noticed red species was pronounced. and itchy stripes appeared on his left leg, 10 Available at: http://ijpa.tums.ac.ir 148 Iran J Parasitol: Vol. 15, No. 1, Jan-Mar 2020, pp.147-152 Fig. 1: Dirofilaria repens worm found in ocular structures of the patient. A) The patient´s eye with a live worm in the subconjunctival space; B) The worm extracted from the right eye; C) Multi-layered cuticle with external longitudinal ridges typical for D. repens observed by the microscopic examination Subsequently, the nematode was sent for long band on 1.5 % agarose gel confirmed molecular analysis. Genomic DNA was isolat- that our nematode belongs to D. repens species. ed by using DNeasy Blood and Tissue Kit The patient was treated with antibiotic eye (Quiagen, Hilden Germany) according to the drops (ofloxacin), ophthalmic corticosteroids manufacturer´s instructions. PCR reactions (dexamethasone), and ophthalmic gel contain- were performed using 5x FIREPol® Master ing dexpanthenol. No new symptoms have Mix Ready to Load (SOLIS Biodyne, Estonia). been observed after the worm extraction. Mitochondrial gene for cytochrome oxidase From epidemiological point of view, the au- subunit 1 (cox1) was amplified using specific tochthonous origin of the infection cannot be primers for both, D. immitis (DI COI -F1: unambiguously confirmed, because in August AGT GTA GAG GGT CAG CCT GAG 2016 patient had spent his holiday in Calabria, TTA and DI COI-R1: ACA GGC ACT GAC southern Italy. AAT ACC AAT) and D. repens (DR COI-F1: AGT GTT GAT GGT CAA CCT GAA TTA Discussion and DR COI-R1: GCC AAA ACA GGA ACA GAT AAA ACT) (7). As positive con- Until recently, human dirofilariasis was con- trol, DNA isolated from D. immitis microfilari- sidered rare infection in the territory of Cen- ae from dog´s blood and DNA from D. repens tral Europe. Nevertheless, the incidence of the worm isolated from human subcutaneous tis- disease is increasing, especially in countries sue was used. Amplification consisted of de- regarded as non-endemic (2, 8). Until in the naturing step (94 °C/2 min), 35 cycles at the dogs D. repens is mostly located free in subcu- temperatures 94°C/30 sec, 57°C/30 sec and taneous tissues, the parasite in human body 72°C/30 sec and final extension (72 °C/7 usually forms a nodule, with the exception of min). The presence of approximately 200 bp ocular form of the infection. 149 Available at: http://ijpa.tums.ac.ir Ondriska et al.: Ocular Dirofilariasis after Clinically Manifested … In case of ocular dirofilariasis, the nematode by several authors. For instance, Ermakova et is considered migratory and not trapped by al. (17) in her study describes parasite´s migra- the host´s defense mechanisms (2). In the eye, tion in more than 43 % of 266 patients with Dirofilaria worms can be present in eyelid, con- subcutaneous form of the infection. In more junctiva, inside the bulb, but most often sub- than 17% of the cases, the worms moved for conjunctivally, with the presence of helminths quite long distances with the most common usually being accompanied by redness, burn- final localization in upper half of the body ing, eye pain, itching and sensation of the for- (head, neck and periorbital area). An interest- eign item in the eye (9,10). The helminths oc- ing case was described also in Slovakia when cur in the form of a cystic lesion or are direct- clinical case of D. repens infection was con- ly visible (3,11), with one, exceptionally two nected with cutaneous larva migrans syn- individuals detected (12). Despite of ocular drome accompanied by severe pain, burning dirofilariasis represents still less common form and erythema of the skin (6). In our patient, of the infection in comparison with subcuta- a parasite´s migration was visible in a form of neous form, during the last two decades pro- ascended subcutaneous strips.