Infection DOI 10.1007/s15010-014-0613-5

CASE REPORT

An atypical case of tularemia presented with pseudoptosis T. Celik • M. Kosker • K. Kirboga

Received: 3 December 2013 / Accepted: 5 March 2014 Ó Springer-Verlag Berlin Heidelberg 2014

Abstract We report an unusual case of tularemia involving pseudoptosis associated with deterioration of the lymphatic drainage system extending from the upper eyelid to the preauricular lymph nodes. A 16-week pregnant patient was admitted with an absess on the neck, preauricular lymphadenopathy, and pseudoptosis on the left side. The micro-agglutination test was positive for Francisella tularensis antibody at a titer of 1/200. The absess was surgically drained and oral cefuroxime was given for 6 weeks. Two weeks after drainage, the pseudoptosis improved due to the recovery of lymphatic drainage system of the eyelid and remission of the absess on the neck. Our case report contributes to the medical literature on tularemia during pregnancy and informs healthcare professionals on the management of the infection in such cases. Keywords Oculoglandular  Tularemia  Pregnancy  Pseudoptosis

Introduction Tularemia is an uncommon but potentially severe zoonotic infection caused by the Gram-negative coccobacillus Francisella tularensis. The microorganism can be transmitted to humans through arthropod bites, the handling of infected animal tissues, ingestion of contaminated water or food, and inhalation of contaminated aerosols [1, 2]. The disease has varied clinical manifestations, and the symptoms may vary depending on which part of the body is affected. The infection generally presents in a ulceroglandular form (70–85 %), which is more severe than other forms (i.e., glandular, oculoglandular, oropharyngeal, pneumonic, and typhoidal forms [2]). We describe here the case of a 16-week pregnant woman with an unusual form of tularemia who presented with pseudoptosis.

Case report

T. Celik (&) Department of Ophthalmology, Bolu Gerede State Hospital, Seviller St., Gerede-Bolu 14900, Turkey e-mail: [email protected] M. Kosker Department of Ophthalmology, Ulus State Hospital, Ankara, Turkey K. Kirboga Department of Ophthalmology, Yozgat State Hospital, Yozgat, Turkey

An 18-year-old female patient from central Anatolia was referred to our hospital with complaints of fever, generalized pain, localized swelling and redness on the left side of the neck, and puffy eyelids. The patient was 16 weeks pregnant. At presentation, her body temperature was 38 °C, heart rate was 90/min, and arterial blood pressure was 130/70 mmHg. Physical examination revealed the presence of an absess on the left side of the neck; preauricular lymphadenopathy and pseudoptosis were also seen on the same side (Fig. 1). Further examination revealed abnormalities in the oral cavity, and an ophthalmic examination also did not detect abnormalities. Blood values were as follows: hemoglobin, 12.8 g/dl; haematocrit, 44 %;

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T. Celik et al. Fig. 1 Presentation of the patient with preauricular lymph node, absess on the neck, and pseudoptosis (before the drainage). (The patient provided written informed consent for publication of these photographs)

Fig. 2 Recovery of absess on the neck and pseudoptosis after the drainage. (The patient provided written informed consent for publication of these photographs)

platelets, 175,000/mm3; C-reactive protein, 6.07 mg/l, white blood cell count, 7,400/mm3, with 80 % polymorphonuclear cells and 20 % lymphocytes. No atypical cells were observed. The abscess was drained surgically and the drainage material cultured on human blood agar. The cultures were negative for pyogenic bacteria. Pathological examination of the aspiration material indicated suppurative inflammation. Serological results were negative for human immunodeficiency virus, hepatitis B and C virus, cytomegalovirus, Herpes simplex virus, Epstein–Barr virus, rubella virus, Treponema pallidum, Borrelia, and Bartonella. The micro-agglutination test was positive for Francisella tularensis with a titer at 1/200. Treatment modalities for pregnant patients and the potential side effects of these regimens on the fetus were explained to the patient in detail. She refused the aminoglycoside or quinolone treatment because of the side

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effects on the fetus, thus we applied the cefuroxime protocol (oral formulation, 500 mg, twice per day for 6 weeks). We observed a gradual improvement in the pseudoptosis 2 weeks after the drainage (Fig. 2). The patient recovered, with no complications of the treatment on the fetus. The patient was followed up weekly during the first month, then monthly during the subsequent 6 months. No relapses were seen in control examinations during the pregnancy. The delivery was without any complications, and we did not observe any problems in the mother and her baby after the birth.

Discussion Francisella tularensis is a highly virulent bacteria that can be spread from animals to humans by vectors such as ticks, deer flies, and mosquitos [2, 3]. Ticks are the most common

An atypical case of tularemia

insect vectors of tularemia in the USA, and the most common tick species that transmit tularemia to humans are Amblyomma, Dermacentor, Haemaphysalis and Ixodes. Rabbits, rodents, and hares serve as reservoir hosts [3]. Although tularemia is known to occur throughout the Northern Hemisphere, F. tularensis subspecies vary with respect to their geographic distribution [4]. There are four subspecies of F. tularensis, with F. tularensis subsp. tularensis (Type A) and F. tularensis subsp. holarctica (Type B) being the main causes of the disease. Type A is more virulent and mainly found in North America; Type B is found primarily in Eastern Europe and Asia and causes a milder infection than Type A [1, 2, 4]. In the USA, 90–154 cases of tularemia were reported yearly to the Centers for Disease Control and Prevention between 2001 and 2010 [5]. The disease typically manifests as fever and regional lymphadenopathy [1, 2]. After infecting the human body, the bacteria replicate inside the macrophage and spread to regional lymph nodes. An inflammatory response is induced in all affected tissues [2]. The incubation period is generally 1–3 days but may extend to 14–15 days. At presentation to our hospital, our patient had fever, generalized pain, preuricular lymphadenopathy, neck absess, and pseudoptosis. She denied exposure to wild animals but she does live in central Anatolia, where tularemia is endemic. Clinical findings combined with serological tests are the preferred methods to diagnose tularemia infection. Positive PCR test results in a clinical series and at least a 1/160 antibody titer in the serum sample indicate tularemia [6]. In our case, although we did not isolate the bacteria on culture medium, the micro-agglutination test was positive with a titer of 1/200. The oculoglandular form of tularemia, which is one of the rarest forms, occurring in only 4 % of all cases, generally affects the conjunctiva and more rarely the adnexes, eyelids, and lacrimal system [7–9]. The preauricular lymph node basin is the primary site of eyelid lymphatic drainage. The classic lymphatic drainage pattern of most of the upper eyelid and the lateral half of the lower eyelid is to the preauricular lymph nodes; some of the medial part of the upper lid and the medial half of the lower lid is to the submandibular nodes [10, 11]. Nijhavan et al. [10] used lymphoscintigraphy to describe the drainage patterns of the human eyelid. However, their results conflict with the previously described traditional drainage patterns of eyelid lymphatics in that they reported the primary sentinel node to be the preauricular node in 72 % (18/25) of the cases studied. Another lymphoscintigraphy study involving 15 patients with eyelid carcinoma corroborated the data reported in the literature by defining the primary lymph node of the eyelids as preauricular node [11].

Here we report a patient who had an unusual form of tularemia that presented with pseudoptosis associated with deterioration of the lymphatic drainage pattern of the upper eyelid to the preauricular lymph nodes. To the best of our knowledge, this is the first report that identifies tularemia as causing pseudoptosis in association with the preauricular lymph node. This should be taken into account by clinicians for patients who are referred to the hospital with puffy eyelid and lymphadenopathy. Azithromycin represents a first-line treatment option for tularemia during pregnancy in regions where the infectious strains of F. tularensis are not resistance to macrolides [12]. However, F. tularensis subsp. holarctica biovar, which is mainly found in Eastern Europe and Asia, is generally resistant to macrolides [13]. A semi-synthetic erythromycin derivative, telithromycin, is a ketolide which is highly active against F. tularensis in vitro, but its use in pregnant women is currently discouraged [14]. The firstchoice antibiotics recommended for tularemia, such as aminoglycosides, fluoroquinolones, or tetracyclines, may be toxic to the fetus; therefore, no treatment is often recommended for tularemia during pregnancy in mild cases [1, 12, 14]. Our patient did not agree to treatment with firstline antibiotics because of the potential side effects on fetus. She was treated with oral cefuroxime and recovered with no complication. This option is currently controversial since tularemia may be associate with severe complications, such as osteomyelitis, pneumonia, and meningitis. Consequently, clinicians should be aware of these complications and observe patients closely in these situations. Conflict of interest No author has a financial or proprietary interest in any material or method mentioned.

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12. Ahmad S, Hunter L, Qin A, Mann B, Van Hoek M. Azithromycin effectiveness against intracellular infections of Francisella. BMC Microbiol. 2010;10:123. 13. Gestin B, Valade E, Thibault F, Schneider D, Maurin M. Phenotypic and genetic characterization of macrolide resistance in Francisella tularensis subsp. holarctica biovar I. J Antimicrob Chemother. 2010;65:2359–67. 14. Dentan C, Pavese P, Pelloux I, Boisset S, Brion JP, Stahl JP, Maurin M. Treatment of tularemia in pregnant woman, France. Emerg Infect Dis. 2013;19:996–8.

An atypical case of tularemia presented with pseudoptosis.

We report an unusual case of tularemia involving pseudoptosis associated with deterioration of the lymphatic drainage system extending from the upper ...
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