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Tularemide Nadir Bir Prezentasyon: Inguinal yerlesimli glanduler tularemi/A Rare Presentation in Tularemia: Inguinally Located Glandular Tularemia.

Running Head: Inguinal yerlesimli glanduler tularemi Inguinally Located Gl

Submitted: 10-Jun-2017;

Accept (03-Sep-2017)

Ayse SAGMAK TARTAR, Ayhan AKBULUT

Firat Universitesi Tip Fakultesi Enfeksiyon Hastaliklari ve Klinik Mikrobiyoloji

Anabilim Dali, ELAZIG/ TURKIYE,

Correspondence: Ayse SAGMAK TARTAR, Firat Universitesi Tip Fakultesi

Enfeksiyon Hastaliklari ve Klinik Mikrobiyoloji Anabilim Dali, ELAZIG/ TURKIYE,

dr.ayse01@mail.com

T: 5434088468

Keywords: Tularemia, Glandular tularemia, Inguinal lymphadenopathy

Introduction

Tularemia is a zoonotic disease caused by the gram-negative bacteria Francisella tularensis and is seen in the northern hemisphere (1). F. tularensis is widespread in nature and has been isolated in more than 250 animal species. Natural reservoirs of the bacteria include rodents such as wild rabbits, squirrels, shrews, voles, and mice as well as beaver and deer. Humans and domestic pets are incidental hosts of F. tularensis (2). The disease is transmitted to humans through the tissues of infected animals or by direct contact of their bodily fluids to the skin or mucosal surfaces; arthropod bites; consumption of contaminated water and food; and inhalation of infected aerosols (2,3). The incubation period is about 2-5 days, and the disease develops 1-21 days after infection (4). Human to human infection has not been documented. Depending on the route of infection, the disease may manifest as ulceroglandular, glandular, oculoglandular, oropharyngeal, typhoidal, or pneumonic tularemia (4).

The aim of this study was to review adult cases who presented with inguinal lymphadenopathy (LAP) and were diagnosed with glandular tularemia, which is a rare site of involvement in Turkey.

A 26-year-old male patient presented with fever, fatigue, and inguinal swelling beginning 20 days earlier. The patient resided in Bingol and had previously been treated with amoxicillin/clavulanate by a physician at another center. After 7 days of antibiotic therapy, there was no fever but the inguinal swelling persisted. In his epidemiological history, he reported working with livestock and he had no history of tick bites. His family did not have similar symptoms. He had not consumed any game animals and reported drinking from the municipal water supply. On physical examination, a 4X8 cm palpable LAP with fluctuation was evident in the left inguinal zone and multiple palpable LAPs with millimetric diameter were detected in the bilateral inguinal zone (Figure 1). Other physical examination findings were normal. Results of laboratory tests were WBC: 16,890/[mm.sup.3] (neutrophil 70.4%), hemoglobin: 13.9 gr/dl, hematocrit: 44.6%, platelet count: 235,000/[mm.sup.3], aspartate transaminase: 18 U/L, alanine transaminase: 9 U/L, urea: 20 mg/dl, creatinine: 0.7 mg/dl, erythrocyte sedimentation rate: 29 mm/hour, C-reactive protein: 57 mg/L. Brucella tube agglutination, herpes simplex, toxoplasma, cytomegalovirus, Epstein-Barr virus, and syphilis serological tests were negative. Oral ciprofloxacin 500 mg twice daily and metronidazole 500 mg 4 times daily were initiated as empirical therapy. Interventional radiology was consulted and 15 cc of yellow, purulent fluid was aspirated from the LAP in the left inguinal zone. The fluid was analyzed by Gram, Giemsa, and acid-resistant bacilli (ARB) staining, culture, and cytology examination. No growth was observed in the culture; Gram and ARB staining were unremarkable. Cytological examination and Giemsa staining revealed predominant leukocytes with polymorphic nuclei. Quantiferon test for tuberculosis was negative. A serum sample submitted to the National Tularemia Reference Laboratory of the Public Health Agency of Turkey returned a microagglutination test result of 1/160, and a sample submitted 14 days later returned a result of 1/640. Metronidazole was discontinued after 14 days of antibiotherapy, while ciprofloxacin was administered for 21 days. The patient was discharged with full recovery.

Tularemia is a zoonotic disease. Except for the typhoidal and pneumonic forms, the mortality rate is less than 1%. Globally, the most common mode of transmission for the disease is via contact with infected animals and ticks; however, in our country it is most often transmitted through consumption of contaminated water and food. Therefore, oropharyngeal tularemia is the most prevalent clinical form in Turkey (5). If diagnosis of oropharyngeal tularemia is delayed, glandular forms characterized by cervical lymphadenitis may develop. An analysis of the recent cases revealed that the glandular form is the second most common manifestation of tularemia (5). The most common form worldwide is the ulceroglandular form, which develops after contacting infected animals and ticks (3). Glandular tularemia accounts for 15-44% of all cases of tularemia worldwide (6). These patients exhibit painful LAP primarily involving the submandibular, cervical, axillary and inguinal lymph nodes. If fever is absent, diagnosis may be delayed. Later, suppurative lymph nodes may develop fluctuation. Glandular tularemia is more common in children than adults (6). Nevertheless, our adult patient was diagnosed with glandular tularemia. On presentation, he exhibited a lymph node with fluctuation due to delayed diagnosis. The differential diagnosis of ulceroglandular and glandular tularemia includes diseases such as cat scratch disease, malignancy, mycobacterial infections, syphilis, lymphogranuloma venereum, chancroid, streptococcal or staphylococcal lymphadenitis, fungal infection, toxoplasmosis, mouse bite fever, herpes simplex infection, and anthrax (7). According to the guidelines on tularemia from the World Health Organization, tularemia can be confirmed when at least one microagglutination titration is [greater than or equal to]1:128 or when titration quadruples between serum samples obtained at an interval of 14 days (2). In our case, microagglutination test results were 1/160 in the initial sample and 1/640 in the sample obtained 14 days later. The patient showed full response the treatment and was discharged with complete recovery.

There have been cases of glandular tularemia with inguinal LAP reported in Turkey, but they were in pediatric patients (8). Glandular tularemia cases in adults are generally in the form of cervical and axillary LAP. There is a report of an adult patient from Ankara with bilateral inguinal LAP and intraabdominal abscess (9). Although tularemia-related inguinal LAP is rarely seen in Turkey, it accounts for 30% of the tularemia cases in North America (6).

To date, there have been 3 cases of tickborne tularemia reported in our country, and all were ulceroglandular tularemia (10,11). No data are available in Turkey with regard to the presence of F. tularensis in ticks.

Early treatment of tularemia helps reduce complications, relapse, and mortality. Delayed diagnosis leads to more complications, suppuration, and the need for surgical intervention. However, drainage may be required in spite of appropriate and early antibiotic therapy. Our case presented late, so the abscess was drained. Tularemia is diagnosed sporadically in our region, and typically present nationwide as oropharyngeal and glandular tularemia.

In conclusion, although oropharyngeal tularemia is the most common clinical form in Turkey, tularemia is endemic in our region and may exhibit atypical involvement, as in our case.

References

(1.) Penn RL. Francisella tularensis (Tularemia). In: Mandell GL, Bennett JE, Dolin R, eds. Principles and Practice of Infectious Diseases. 7th ed. Philadelphia: Churchill Livingstone; 2010. p.2927-37.

(2.) WHO Guidelines on Tularemia. http://www.cdc.gov/tularemia/resources/whotularemiamanual. Pdf

(3.) Ellis J, Oyston PC, Green M, Titball RW. Tularemia. Clin Microbiol Rev. 2002; 15(4): 631-46.

(4.) Harik NS. Tularemia: epidemiology, diagnosis and treatment. Pediatr Ann. 2013; 42: 288-92.

(5.) Kilic S. Tularemi: Etken ve Epidemiyoloji. Turkiye Klinikleri J Inf Dis-Special Topics 2014; 7(2): 52-61

(6.) Snowden J, Stovall S. Tularemia: retrospective review of 10 years' experience in Arkansas. Clin Pediatr. 2011; 50: 64-8.

(7.) Asano S. Granulomatous lymphadenitis. J Clin Exp Hematop 2012; 52(1): 1-16. (PMID: 22706525).

(8.) Bayhan-Tas GI, Tanir G, Celebi B. Two Cases of Glandular Tularemia From Turkey. Turk J Pediatr 2012; 54(2): 203-6.

(9.) Grassie SS, Kilic S, Kilickalp G, Ocak F, Akduran S, Kayaaslan B. Bilateral inguinal LAP ve intraabdominal Apse ile Seyreden Bir Tularemi Vakasi, 5. EKMUD Bilimsel Platformu, 1-4 Nisan 2015, Izmir. Kongre Kitabi, s: 221, PB-052.

(10.) Yesilyurt M, Kilic S, Cagasar O, Celebi B, Gul S. Two cases of tick-borne tularemia in Yozgat province, Turkey. Mikrobiyol Bul 2010; 45(4): 746-54.

(11.) Aydinli S, Atasoylu G, Ince A, Karadenizli A. The first ulceroglandular tularemia case occurred after a tick bite in Turkey where oropharyngeal tularemia is prevalent. (Poster presentation no:072) The 4th Eurasia Congress of Infectious Diseases, 01-05 June 2011, Sarajevo, Bosnia-Herzegovina, p.183
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Title Annotation:Mjima-0009
Publication:Mediterranean Journal of Infection, Microbes and Antimicrobials
Article Type:Letter to the editor
Date:Jan 1, 2017
Words:1360
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