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      First case report of thyroid abscess caused by Helicobacter cinaedi presenting with thyroid storm

      case-report

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          Abstract

          Background

          Helicobacter cinaedi is a microaerobic Gram-negative spiral-shaped bacterium that causes enteritis, cellulitis, and bacteremia in both immunocompromised and immunocompetent patients. While there have been increasing numbers of reported H. cinaedi infections recently, there has been no thyroid abscess case caused by H. cinaedi presenting with thyroid storm.

          Case presentation

          A 50-year-old Japanese man presented with a 9-day history of high fever associated with palpitations, dry cough, and chronic diarrhea. The patient had a history of Basedow’s disease that had been treated with thiamazole in the past. During the current episode, the patient was diagnosed with thyroid storm and treated accordingly. The blood culture taken on admission was positive for H. cinaedi. This finding was confirmed by matrix-assisted laser desorption ionization-time of flight mass spectrometry (MALDI-TOFMS). A systemic computed tomography (CT) scan revealed a thyroid abscess as the site of infection and cause of the bacteremia. The 16S rRNA gene sequencing identified the pathogen of thyroid abscess as H. cinaedi. Clinical symptoms and laboratory data normalized on admission day 7 after treatment with both effective antibiotics and antithyroid drugs.

          Conclusions

          The case study described a patient with a history of Basedow’s disease that presented with a thyroid abscess caused by H. cinaedi with symptoms similar to those of thyroid storm. While this bacterium has been implicated in other infections, we believe this is the first time the bacteria has been documented to have caused a thyroid abscess.

          Electronic supplementary material

          The online version of this article (10.1186/s12879-019-3808-7) contains supplementary material, which is available to authorized users.

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          Most cited references27

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          Clinical characteristics of bacteremia caused by Helicobacter cinaedi and time required for blood cultures to become positive.

          The aim of this study was to clarify the clinical characteristics of patients with Helicobacter cinaedi bacteremia and the time required for blood cultures to become positive. The medical records of all patients with H. cinaedi bacteremia at Toranomon Hospital and Toranomon Hospital Kajigaya between March 2009 and March 2013 were retrospectively reviewed. Sixty-three patients, 34 men and 29 women with a median age of 67 years (range, 37 to 88 years), were diagnosed with H. cinaedi bacteremia. A total of 51,272 sets of blood cultures were obtained during the study period, of which 5,769 sets of blood cultures were positive for some organism and 126 sets were H. cinaedi positive. The time required for blood cultures to become positive for H. cinaedi was ≤5 days in 69 sets (55%) and >5 days in 57 sets (45%). Most patients had an underlying disease, including chronic kidney disease (21 cases), solid tumor (19 cases), hematological malignancy (13 cases), diabetes mellitus (8 cases), chronic liver disease (6 cases), and postorthopedic surgery (3 cases). Only 1 patient had no apparent underlying disease. The clinical symptoms included cellulitis in 24 cases, colitis in 7 cases, and fever only in 27 cases, including 7 cases of febrile neutropenia. The 30-day mortality rate of H. cinaedi bacteremia was 6.3%. In conclusion, most cases of H. cinaedi bacteremia occurred in immunocompromised patients. We might have overlooked nearly half of the H. cinaedi bacteremia cases if the duration of monitored blood culture samples had been within 5 days. Therefore, when clinicians suspect H. cinaedi bacteremia, the observation period for blood cultures should be extended.
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            Helicobacter cinaedi-associated bacteremia and cellulitis in immunocompromised patients.

            To define the clinical spectrum of illness associated with Helicobacter cinaedi infection in the United States and to determine associated epidemiologic risk factors and optimal laboratory methods for recovery of H. cinaedi. A retrospective epidemiologic study of 23 patients with H. cinaedi-associated illness. 23 patients with H. cinaedi infection identified between January 1982 and August 1990. Most isolates (22 of 23) were from blood; one was from stool. Ages ranged from 24 to 84 years (mean, 44 years). Eighty-three percent of patients were men; 17% were women. Clinical and laboratory data were obtained from 21 patients. Eighteen patients were febrile (15 required hospitalization); cellulitis was reported in 9 patients. Sixty percent were immunocompromised; 45% were reported to be seropositive for human immunodeficiency virus (HIV). For bacteremic patients, positive blood cultures were detected by a slightly elevated growth index in an automated blood culture system; many hospital laboratories had difficulty isolating the organism. Helicobacter cinaedi appears to cause recurrent cellulitis with fever and bacteremia in immunocompromised hosts. Blood cultures from immunocompromised patients with these symptoms may need special handling to isolate H. cinaedi.
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              Helicobacter cinaedi cellulitis and bacteremia in immunocompetent hosts after orthopedic surgery.

              At various times after orthopedic operations (more than a few weeks, with an average of 29.9 days), 11 patients had a sudden onset of high temperature (average 38.9 degrees C) and local cellulitis at different sites on the operated sides. The wounds had completely healed, without complicated infections, when the cellulitis occurred. The clinical picture of cellulitis in all patients was atypical: diffuse salmon-pink skin color, local heat, swelling, spontaneous pain, and tenderness but no eruptions. No patient had any underlying immunocompromising conditions or had been given immunosuppressive agents. Gram-negative spiral bacteria were isolated from blood cultures and were identified as Helicobacter cinaedi on the basis of 16S rRNA gene sequencing and DNA-DNA hybridization using standard strains. By means of phylogenetic analysis, we divided these clinical isolates into two clones. The H. cinaedi strain isolated via fecal cultures from two patients without intestinal symptoms was the same clone as the blood isolate. All isolates were quite susceptible to various antibiotics, and clinical and inflammatory symptoms of bacteremia and cellulitis improved after treatment with penicillins and cephalosporins. A relatively high incidence of recurrence of the same disease was observed, however. Almost all patients responded immunologically to the infection, as evidenced by the production of serum antibody against H. cinaedi. We thus suggest that H. cinaedi should not be regarded as simply an opportunistic pathogen but that it may be a pathogen in immunocompetent hosts and may cause infections together with bacteremia and cellulitis.
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                Author and article information

                Contributors
                tomohirotakehara@keio.jp
                +81-42-523-3131 , tetsuo19840324@yahoo.co.jp
                k-yajima@rondo.plala.or.jp
                m.mizuguchi@tachikawa-hosp.gr.jp
                otsuka.yoshihito@kameda.jp
                hdfmkoh@yahoo.com
                Journal
                BMC Infect Dis
                BMC Infect. Dis
                BMC Infectious Diseases
                BioMed Central (London )
                1471-2334
                15 February 2019
                15 February 2019
                2019
                : 19
                : 166
                Affiliations
                [1 ]ISNI 0000 0004 1936 9959, GRID grid.26091.3c, Division of Pulmonary Medicine, Department of Medicine, , Keio University School of Medicine, ; Tokyo, Japan
                [2 ]GRID grid.416823.a, Division of Pulmonary Medicine, Department of Internal Medicine, Federation of National Public Service Personnel Mutual Aid Associations, , Tachikawa Hospital, ; 4-2-22 Nishikicho, Tachikawa, Tokyo, 190-8531 Japan
                [3 ]GRID grid.416823.a, Division of Diabetes, Endocrinology and Metabolism, Department of Internal Medicine, Federation of National Public Service Personnel Mutual Aid Associations, , Tachikawa Hospital, ; Tokyo, Japan
                [4 ]GRID grid.416823.a, Department of Microbiological Laboratory, Federation of National Public Service Personnel Mutual Aid Associations, , Tachikawa Hospital, ; Tokyo, Japan
                [5 ]ISNI 0000 0004 0378 2140, GRID grid.414927.d, Department of Clinical Laboratory, , Kameda Medical Center, ; Chiba, Japan
                Author information
                http://orcid.org/0000-0001-5719-2697
                Article
                3808
                10.1186/s12879-019-3808-7
                6377776
                30770725
                bf2a588e-c0e1-45ca-b910-bc98a8f33be5
                © The Author(s). 2019

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                : 5 October 2018
                : 12 February 2019
                Categories
                Case Report
                Custom metadata
                © The Author(s) 2019

                Infectious disease & Microbiology
                helicobacter cinaedi,thyroid abscess,basedow’s disease,thyroid storm

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