Open Forum Infectious Diseases ID CASE pericardial cultures grew a curved Gram-negative rod that was Getting to the Heart of the Matter: difficult to visualize on Gram stain but easily seen with carbol A 20-Year-Old Man With Fever, Rash, fuchsin counterstain (Figure 2A) and acridine orange stain and Chest Pain (Figure 2B). The organism grew poorly on Campylobacter or 1,a 1,a 2 Brucella blood agar but grew well on buffered charcoal yeast Michelle C. Sabo, Jim Boonyaratanakornkit, Robert Cybulski, 1 1 1,2 1,3,4 Noam E. Kopmar, Rosario V. Freeman, Ferric C. Fang, and Susan M. Graham extract agar under microaerophilic conditions. Piperacillin- 1 2 3 4 Departments of Medicine, Laboratory Medicine, Global Health, and Epidemiology, tazobactam was empirically started. University of Washington, Seattle e p Th atient’s fevers resolved by the second day of piperacil- lin-tazobactam therapy, and repeat blood cultures on day 5 of Infection with Helicobacter cinaedi can encompass a wide therapy were negative. Repeat echocardiogram showed com- spectrum of clinical manifestations, including fever, rash, plete resolution of the effusion, and the pericardial drain was endocarditis, osteomyelitis, and meningitis. The present case removed on hospital day 5. Mass spectrometry failed to iden- demonstrates the ability of H cinaedi to masquerade as acute tify the organism. However, 458 nucleotides of the 16S riboso- rheumatic fever and represents the first reported case of cardiac mal ribonucleic acid (rRNA) sequence matched perfectly with tamponade caused by H cinaedi. Keywords. helicobacter; pericarditis; tamponade; GenBank accession KJ534298 belonging to Helicobacter cinaedi strain CIP105369. The working diagnosis was thus changed bacteremia. from acute rheumatic fever to H cinaedi bacteremia compli- cated by purulent bacterial pericarditis. Aer t ft he organism was identified, the patient was questioned again about his sexual CASE practices. He reported receptive anal intercourse with a man A 20-year-old student with no past medical history was seen in several weeks before admission. Human immunodeficiency the emergency department (ED) for a painful, nodular rash on virus (HIV) testing by polymerase chain reaction and antibody/ his left thigh (Figure 1A). He denied fever, sore throat, or recent antigen screening was negative. travel. Antistreptolysin O was 220 IU/mL (normal <100), and he Given his clinical improvement and clearance of cultures, he was given intramuscular penicillin G due to concern for acute was discharged on piperacillin-tazobactam to complete a total rheumatic fever. Three weeks later, he returned to the ED with 4-week course. Based on susceptibility testing by E-test, which intermittent pleuritic, midsternal chest pain and electrocardio- was delayed due to slow growth of the organism on suscepti- gram findings consistent with acute pericarditis. Despite ther- bility testing media, he was transitioned to doxycycline mon- apy with ibuprofen and colchicine, his chest pain progressed, otherapy for the final 2 weeks of treatment (Table 1). Three and he returned to the ED 1 week later. Exam was notable for weeks aer co ft mpleting therapy, he remained asymptomatic, a temperature of 38.4°C, tachycardia, distended neck veins, and repeat blood cultures were negative. He was also seen in and 2 erythematous nodules on his left thigh. Echocardiogram follow-up 16 months aer co ft mpleting therapy and was doing demonstrated a large pericardial effusion with evidence of well without any evidence of relapse. tamponade (Figure 1B and Video 1). Peripheral blood cultures were obtained, and he underwent urgent pericardiocentesis of DISCUSSION 600 mL bloody fluid. The differential diagnosis for a curved Gram-negative rod that He was initially treated with penicillin G for presumed can be cultivated microaerobically is fairly limited and includes acute rheumatic fever. Two days aer co ft llection, his blood and species of Helicobacter, Vibrio, and Campylobacter. Helicobacter cinaedi is a curved Gram-negative rod first isolated from rectal Received 26 October 2017; editorial decision 9 December 2017; accepted 19 December 2017. cultures of men who have sex with men (MSM) . It is consid- Correspondence: M. C. Sabo, MD, PhD, Department of Medicine, University of Washington, ered an “enterohepatic Helicobacter” due to its initial isolation 1959 NE Pacific St., Health Sciences Building Box 356423, Seattle, WA 98195 (firstname.lastname@example.org). from the gastrointestinal tract . The most common present- M. C. S. and J. B. contributed equally to this work. ing symptoms of H cinaedi infection are fever and a cellulitic Open Forum Infectious Diseases © The Author(s) 2017. Published by Oxford University Press on behalf of Infectious Diseases or nodular rash [3, 4], although cases of myopericarditis , Society of America. This is an Open Access article distributed under the terms of the Creative endocarditis , osteomyelitis , and meningitis  have been Commons Attribution-NonCommercial-NoDerivs licence (http://creativecommons.org/licenses/ by-nc-nd/4.0/), which permits non-commercial reproduction and distribution of the work, in any reported. Severe disseminated disease has been described in medium, provided the original work is not altered or transformed in any way, and that the work patients with or without immunocompromise. One recent study is properly cited. For commercial re-use, please contact email@example.com DOI: 10.1093/ofid/ofx272 suggested that patients with community-acquired infections are ID CASE • OFID • 1 Downloaded from https://academic.oup.com/ofid/article-abstract/5/1/ofx272/4770303 by Ed 'DeepDyve' Gillespie user on 16 March 2018 Pericardial AB Eusion LV RV RA LA Figure 1. (A) Photo of the nodular rash (indicated by black arrows) on the left thigh. (B) Echocardiogram in the apical 4-chamber view showing a large circumferential pericardial effusion (indicated by a white arrow). LA, left atrium; LV, left ventricle; RA, right atrium; RV, right ventricle. less likely to have an immunocompromising condition such as study of the relationship among H cinaedi, anal sex, the rectal chronic kidney disease, diabetes, or use of immune-modulating microbiome, and HIV transmission risk is warranted . therapy . The present case represents the first report of H Matrix-assisted laser desorption ionization time-of-flight cinaedi pericarditis complicated by cardiac tamponade. mass spectrometry can identify H cinaedi using the Bruker Further work is needed to characterize the epidemiology and Biotyper RUO database, but the organism is not yet included pathogenesis of H cinaedi infection. In the original prospec- in the US Food and Drug Administration-cleared database. tive studies identifying H cinaedi, this bacterium was detected Furthermore, commercial detection kits and biochemical with increased frequency in HIV-negative MSM with proctitis detection methods have proven to be unreliable [12, 13]. Thus, relative to asymptomatic HIV-negative MSM, and it was not 16S rRNA sequencing was used as an alternative method of detected in any asymptomatic heterosexual individuals [1, 9]. identification. Although these studies suggested a predilection for MSM, Successful eradication of invasive H cinaedi infection subsequent case reports and case series have not documented often requires ≥4 weeks of therapy, and recurrences 2–10 patient sexual orientation. e Th related pathogen Campylobacter weeks after completing therapy have been described in jejuni is also thought to have the potential for transmission both immunocompetent and immunocompromised hosts through anal sex and causes proctocolitis . Although sexu- [14–16]. Two studies have shown that H cinaedi is reliably ally transmitted infections and other sources of mucosal inflam- susceptible in vitro to imipenem and minocycline and is var- mation are thought to facilitate HIV transmission, additional iably susceptible to β-lactam antibiotics [13, 17]. In Japan, AB Figure 2. (A) Gram stain of the curved Gram-negative rod using carbol fuchsin as the counterstain and (B) acridine orange stain. 2 • OFID • ID CASE Downloaded from https://academic.oup.com/ofid/article-abstract/5/1/ofx272/4770303 by Ed 'DeepDyve' Gillespie user on 16 March 2018 ×3 Table 1. Antibiotic Susceptibility Testing Potential conifl cts of interest. All authors: No reported conflicts of interest. All authors have submitted the ICMJE Form for Disclosure of Potential Conflicts of Interest. Conflicts that the editors consider relevant to Antibiotic Etest MIC (mcg/mL) the content of the manuscript have been disclosed. Penicillin >256 Ceftriaxone 128 References Ampicillin/sulbactam 64 1. Quinn TC, Goodell SE, Fennell C, et al. Infections with Campylobacter jejuni Azithromycin >256 and Campylobacter-like organisms in homosexual men. Ann Intern Med 1984; Ciprofloxacin >32 101:187–92. 2. Solnick JV, Schauer DB. Emergence of diverse Helicobacter species in the Ertapenem 0.25 pathogenesis of gastric and enterohepatic diseases. Clin Microbiol Rev 2001; Meropenem 0.032 14:59–97. Gentamicin 1 3. Shimizu S, Shimizu H. Cutaneous manifestations of Helicobacter cinaedi: a review. Tetracycline 1 Br J Dermatol 2016; 175:62–8. 4. Uwamino Y, Muranaka K, Hase R, et al. clinical features of community-acquired Doxycycline 0.5 Helicobacter cinaedi bacteremia. Helicobacter 2016; 21:24–8. Abbreviations: MIC, minimum inhibitory concentration. 5. Lewis GD, Holmes CB, Holmvang G, Butterton JR. Case records of the Massachusetts General Hospital. Case 8-2007. A 48-year-old man with chest pain followed by cardiac arrest. N Engl J Med 2007; 356:1153–62. 6. Bartels H, Goldenberger D, Reuthebuch O, et al. First case of infective endocardi- intrinsic mutations in the gyrA gene and 23S rRNA genes tis caused by Helicobacter cinaedi. BMC Infect Dis 2014; 14:586. 7. Murata S, Suzuki H, Sakamoto S, et al. Helicobacter cinaedi-associated vertebral have resulted in increased H cinaedi resistance to fluoro- osteomyelitis in an immunocompetent patient. Intern Med 2015; 54:3221–4. quinolones and macrolides, respectively. To our knowledge, 8. Okubo H, Goto M, Sato M, et al. Helicobacter cinaedi meningitis: a case report and no reports of multidrug-resistant H cinaedi have been pub- review of previous cases. J Neurol Sci 2014; 347:396–7. 9. Totten PA, Fennell CL, Tenover FC, et al. Campylobacter cinaedi (sp. nov.) and lished; however, genomic studies have identified several Campylobacter fennelliae (sp. nov.): two new Campylobacter species associated putative efflux pumps . Our patient improved on pip- with enteric disease in homosexual men. J Infect Dis 1985; 151:131–9. 10. Rompalo AM. Diagnosis and treatment of sexually acquired proctitis and procto- eracillin/tazobactam, which was reported to be effective in colitis: an update. Clin Infect Dis 1999; 28(Suppl 1):S84–90. an asplenic patient with H cinaedi bacteremia . Further 11. Fleming DT, Wasserheit JN. From epidemiological synergy to public health pol- icy and practice: the contribution of other sexually transmitted diseases to sexual characterization of H cinaedi susceptibility patterns may transmission of HIV infection. Sex Transm Infect 1999; 75:3–17. be warranted to help guide empiric therapy in critically ill 12. Bateman AC, Butler-Wu SM. The brief case: bacteremia caused by Helicobacter cinaedi. J Clin Microbiol 2017; 55:5–9. patients. 13. Kawamura Y, Tomida J, Morita Y, et al. Clinical and bacteriological characteristics of Helicobacter cinaedi infection. J Infect Chemother 2014; 20:517–26. 14. Shimizu Y, Gomi H, Ishioka H, Isono M. Refractory to treat Helicobacter cinaedi CONCLUSIONS bacteremia with bilateral lower extremities cellulitis in an immunocompetent patient. IDCases 2016; 5:9–11. In summary, H cinaedi should be considered in patients pre- 15. Sugimoto M, Takeichi T, Muramatsu H, et al. Recurrent cellulitis caused by Helicobacter cinaedi in a patient with X-linked agammaglobulinaemia. Acta senting with fever and nodular rash, particularly in MSM, Derm Venereol 2017; 97:277–8. and disseminated disease may rarely present as cardiac 16. Adachi Y, Moriya C, Fujisawa T, et al. Recurrent superficial cellulitis-like erythema associated with Helicobacter cinaedi bacteremia. J Dermatol 2016; 43:844–6. tamponade. 17. Rimbara E, Mori S, Matsui M, et al. Molecular epidemiologic analysis and anti- microbial resistance of Helicobacter cinaedi isolated from seven hospitals in Japan. Acknowledgments J Clin Microbiol 2012; 50:2553–60. We gratefully acknowledge the staff at the University of Washington 18. Kim SK, Cho EJ, Sung H, et al. A case of Helicobacter cinaedi bacteremia in an Medical Center Clinical Microbiology Laboratory. asplenic patient. Ann Lab Med 2012; 32:433–7. ID CASE • OFID • 3 Downloaded from https://academic.oup.com/ofid/article-abstract/5/1/ofx272/4770303 by Ed 'DeepDyve' Gillespie user on 16 March 2018
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