962055 case-report2020

EJO0010.1177/1120672120962055European Journal of OphthalmologyHavuz and Güdül Havuz

EJO

Case report

Lyme disease atypically presenting with a singular symptom: Unilateral chorioretinitis Erol Havuz1

European Journal of Ophthalmology

European Journal of Ophthalmology 1­–6 © The Author(s) 2020 Article reuse guidelines: sagepub.com/journals-permissions https://doi.org/10.1177/1120672120962055 DOI: 10.1177/1120672120962055 journals.sagepub.com/home/ejo

and Seda Güdül Havuz2

Abstract Background: Lyme disease, caused by Borrelia burgdorferi, is a spirochetal disease. Lyme disease-related ocular findings may also provide important clues. Ocular involvement is most commonly seen as uveitis, chorioretinitis, conjunctivitis, keratitis, episcleritis, papillitis, panuveitis, ischemic optic neuropathy, papilledema, and retinal vasculitis. Case: A 27-year-old male patient was admitted with a history of fatigue, malaise, and sudden loss of vision in his left eye for 3 days. The best visual acuity was found 20/20 in the right eye and 20/400 in the left eye. Fluorescein fundus angiography showed no pathological findings in the right eye; but hyperfluorescence that was compatible with choroiditis foci was seen in the left eye. Optical coherence tomography (OCT) showed choroidal thickening in the left eye compared to the right eye. Lyme IgM antibody was found to be positive, explaining choroiditis etiology, while IgG values were found to be negative. Western blot verification test was positive. The patient was treated with 2 × 100 mg doxycycline (21 days) with a diagnosis of Lyme disease, prednol 1 mg/kg/day (10 days) for choroiditis. Omeprazole tablets were given 1 × 1 during the period of cortisone intake. On the third day of treatment, visual acuity increased to 20/200 and continued to increase until reaching 20/20 in the second week. Conclusions: Lyme disease is rare, but must be kept in mind when investigating the etiology of chorioretinitis and retinal vasculitis. The patient reported here is, to our knowledge, the second case reported in literature that shows atypical clinic for Lyme disease with unilateral chorioretinitis without Erythema chronicum migrans (ECM). Keywords Lyme borreliosis, posterior uveitis, uveitis, complications of uveitis, pharmacology, preventive medicine/screening Date received: 16 December 2019; accepted: 1 September 2020

Introduction Lyme disease, caused by the spirochetal organism called Borrelia burgdorferi, is an often systemic zoonotic infection transmitted to humans via ticks. This infection is more common in the northern hemisphere. Ticks need 80% humidity, a mild climate, small vegetation, and reservoir rodents to be able to continue their life cycle.1 Lyme disease manifests itself in three clinical phases: early localized infection (Stage 1), early widespread infection (Stage 2), and late disease (Stage 3).2 Ophthalmic findings occur in the second stage of Lyme disease. There are cases with no history of tick bites but diagnosed with uveitis and chorioretinitis in the literature.3 Chorioretinitis is characteristically seen bilaterally in Lyme disease, it is very rare to detect it in only one eye.

However, it is apparent that Lyme disease should be kept in mind when investigating the etiology of chorioretinitis and retinal vasculitis, even when unilateral. This case is the second case in the literature reporting unilateral choroiditis without ECM in Lyme disease.

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 epartment of Ophthalmology, University of Health Sciences, SUAM D Samsun Hospital, Samsun, Turkey 2 Minister of Health, Bafra Hospital, Samsun, Turkey Corresponding author: Erol Havuz, Department of Ophthalmology, University of Health Sciences, SUAM Samsun Hospital, Kadikoy Mahallesi, Barıs Blv. No:199, İlkadim, Samsun, 55090, Turkey. Emails: [email protected]; [email protected]

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European Journal of Ophthalmology 00(0)

Figure 1.  (a) Image of the normal right fundus. (b) Choroiditis foci in left fundus (arrows). (c) Normal fundus fluorescein angiography on right. (d) Hyperfluorescence due to diffuse chorioretinitis foci on lef fundus fluorescein angiogram.

Case report A 27-year-old male patient was admitted with a history of fatigue, malaise, and sudden loss of vision in his left eye for 3 days. The patient was in his military service and was serving in the infirmary as a medical officer. He had no history of travel within the last 3 months. He did not have any field duty and did not have a history of any suspected insect bites or tick bites, swelling of the joints, pain, skin inflammation, or rash. The best visual acuity was 20/20 in the right eye and 20/400 in the left eye. Biomicroscopic anterior segment examination did not reveal any pathological findings or afferent pupil defect in either eye. Fundus examination of the right eye was also considered to be normal (Figure 1(a)). Multipl white-cream colored foci were observed in the posterior pole of the left eye (Figure 1(b)). No vitreous inflammation or vasculitis was detected. Fluorescein fundus angiography showed no pathological findings in the right eye (Figure 1(c)), while hyperfluorescence consistent with chorioretinitis foci was determined in the left eye (Figure 1(d)). Indocyanine green Angiography is an indispensable examination for evaluation of both disease activity and therapeutic response in choriocapillaries. Unfortunately, the evaluation could not be made because we did not have

the Indocyanine Green Angiography device of the in our clinic. The optical coherence tomography angiography (OCT-A) of the patient’s right eye was normal (Figure 2 at the top); but in the left eye, inflammation-related filling defects were observed in the choriocapillary layer (Figure 2 at the bottom) .Furthermore, OCT revealed a choroidal thickness of 305 μm in the right eye and 425 μm in the left eye. Retinal thickness in the left macular temporal region was found to be increased due to inflammation (Figure 3(b)). In addition, complete blood count and biochemistry tests performed to determine the etiology of unilateral choroiditis were found to be normal. ASO was 37.4 IU/ml (normal: 0–200 IU/mL), CRP was 13.0 mg/L (normal: 0–8  mg/L) and sedimentation was 38  mm/h (normal: 0–15 mm/h). Other causes of chorioretinitis were evaluated; the brucella coombs test, syphilis test (VDRL-RPR), Toxoplasma IgM and IgG, and serum angiotensin-converting enzyme (ACE) levels were within normal limits. No abnormalities were reported in the final consultation notes from the departments of Rheumatology for collagen vascular disease, Chest Diseases, and Infectious Diseases for tuberculosis and sarcoidosis. In addition, a thorough and detailed physical examination did not yield any findings such as tick bite, ECM, or arthralgia. The VirClia® Chemiluminescence Monotest (Vircell, S.L.

Havuz and Güdül Havuz

Figure 2.  The optical coherence tomography angiography (OCT-A) of the patient’s right eye was normal (at the top); in the left eye, inflammation-related filling defects were observed in the choriocapillary layer (at the bottom).

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Figure 3.  (a) Normal OCT image on the right. (b) Increased retinal thickness in choroidal and macular nasal region, irregular retinal pigment layer. (c) Normal OCT result obtained after treatment of the left eye.

Granada, Spain) and Borrelia burgdorferi IgM antibody levels that were ordered for the etiological screening of the choroiditis yielded positive results. IgM was positive while IgG was negative with the following index values: IgM = 3.1822 (>1.1 positive), IgG = 0.19 (>1.1 positive). Confirmation with Western blot (Anti-Borrelia

Euroline-WB, Euroimmun, Germany) demonstrated positivity for OspC, 39 and 41 bands. The patient was treated with 2 × 100 mg doxycycline for 21 days with the diagnosis of Lyme disease. He was also prescribed prednol 1 mg/kg/day for choroiditis for 10 days, after which treatment was finalized. Omeprazole tablets were given 1 × 1

Havuz and Güdül Havuz during the period of cortisone intake. On the third day of treatment, visual acuity increased to 20/200 and continued to progress until 20/20 vision was determined in the second week. The OCT taken on the 15th day of treatment is shown in Figure 3(c). A written informed consent was optained from the patient. We obligated to protect patient's privacy and not disclose patient’s personel information. The patient also gave a permission to use the images anonymously.

Discussion Lyme disease has been reported with various clinical findings that primarily depend on the stage of the disease, including ECM, carditis, pericarditis, atrioventricular block, arthritis, meningitis, encephalitis, cranial neuritis, myelitis, and peripheral neuropathy. Some ocular disorders associated with Lyme disease may be undiagnosed because the disease is not well known in terms of ocular clinical features. In an interesting study, it was stated that among the 160 uveitis patients referred to the Helsinki University Eye Clinic – a tertiary healthcare provider in Finland (an endemic region for Lyme disease) – the infectious cause had been identified in only 23 (14.4%) of the cases. Lyme borreliosis was determined in 4.3% of these uveitis cases.4 There are only a few publications that have focused on the characteristics of ocular Lyme borreliosis, and the largest series consists of 20 patients. In this series, only 10 patients had a history of tick bites and seven patients had ECM. Ten of 20 patients had uveitis and one had branch retinal vein occlusion. Posterior uveitis was reported in only two of these 20 patients. Vitritis and posterior uveitis were found in both of these patients, while one of them also had retinal vasculitis and neuroretinitis. Seven out of the 10 patients with uveitis were also found to have vasculitis.5 In a study from Turkey, Anlar et al.3 reported a case series consisting of 12 patients, the largest to date in Turkey. Their results showed that four of these 12 patients had ocular involvement. It was extremely interesting to find that none of the patients with ocular involvement, uveitis, or chorioretinitis had a history of tick bites. In 2016, Müftüoğlu et al.6 reported a case of Lyme disease with uveitis, vasculitis, and diffuse white spots on the posterior pole. In this case, travel to the endemic region and a history of ECM lesion was present. In the case reports by Amer et al.,7 one of the two patients with chorioretinitis had an EM clinic, and both had a history of tick bites, but serological tests were negative for Borreliosis. Although serological results were negative, the history of ECM and tick bites facilitated the diagnosis of the patients. In 2016, two cases with chorioretinitis were published by Al Mousa and Koch.8 Unilateral chorioretinitis associated with Lyme disease has only been reported in the literature by Nuitta et al.,9 in 1993. In our study, the patient presented with a complaint of unilateral sight loss. To our knowledge, this is only the second case in the literature with choroiditis development

5 in only one eye that was in the presence of atypical clinical features (due to absence of symptoms such as ECM and arthralgia). ECM is seen in up to 70% of patients with Lyme. Supportive tests should be performed in patients who are likely to have atypical disease course when this pathognomonic finding cannot be confirmed.3 Borrelia burgdorferi IgM antibody starts to increase in the first few weeks of the disease, peaks in 3 to 6 weeks and begins to reduce within 6 months. IgG antibodies begin to increase after a few weeks.1 The ELISA tests and Western blot test make diagnosis easier. However, serological tests are not always accurate and confirmation with western blot may be required in some cases. In the studies for Lyme disease, seropositivity was detected in 6% to 35.9% of individuals in Turkey.2 This information suggests that Lyme disease is surprisingly common in our country. In regard to ocular manifestations, antibiotic treatment for ocular borreliosis and anti-inflammatory treatments are needed to control choroidal inflammation and to prevent damage to the eyes.7 In this case report, although the patient had very atypical clinical findings, it must be emphasized that serious morbidity can be prevented by rapid diagnosis and early initiation of treatment in patients with Lyme disease. It is also evident that Lyme disease is among the causes of retinal vasculitis and chorioretinitis, and must be considered even in the absence of pathognomonic findings. Declaration of conflicting interests The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding The author(s) received no financial support for the research, authorship, and/or publication of this article.

ORCID iD Erol Havuz

https://orcid.org/0000-0002-1731-8715

References 1. İseri L and Durmaz B. Borrelia ve Lyme hastaliği. Turgut Özal Tıp Merkezi Dergisi 2000; 7. 2. Yemişen M, Bilgul M and Balkan I. Lyme hastalıgı. J ExpClin Med 2012; 29: 169–174. 3. Anlar F, Durlu Y, Aktan G, et al. The clinical characteristics of 12 cases with Lyme disease. Mikrobiyoloji Bulteni 2003; 37: 255–260. 4. Helena Mikkila IS, Marjatta Leirisalo-Repo, Ilkka Immonen, et al. The etiology of uveitis: the role of infections with special reference to Lyme borreliosis. Acta Ophthalmol Scand 1997; 75: 716–719. 5. Mikkilä HO, Seppälä IJ, Viljanen MK, et al. The expanding clinical spectrum of ocular Lyme borreliosis. Ophthalmology 2000; 107: 581–587.

6 6. Müftüoğlu İK, Akova YA and Güngör SG. A case of Lyme disease accompanied by uveitis and white dot syndrome. Turk J Ophthalmol 2016; 46: 241. 7. Amer R, Brannan S and Forrester JV. Inflammatory choroidal neovascular membrane in presumed ocular Lyme borreliosis. Acta Ophthalmol 2009; 87: 346–348.

European Journal of Ophthalmology 00(0) 8. Al Mousa M and Koch F. Acute Borrelia infection inducing an APMPPE-like picture. J Ophthalmic Inflamm Infect 2016; 6: 22. 9. Niutta A, Barcaroli I and Palombi E. Monolateral chorioretinitis with multiple foci in one case of Lyme disease. Ann Ophthalmol 1993; 25: 257–261.

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962055 case-report2020 EJO0010.1177/1120672120962055European Journal of OphthalmologyHavuz and Güdül Havuz EJO Case report Lyme disease atypically...
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