Recurrent Bacteremia and Multifocal Lower Limb Cellulitis Due to<i>Helicobacter</i>‐Like Organisms in a Patient with X‐Linked Hypogammaglobulinemia

Gerrard, John; Alfredson, David; Smith, Ina

doi:10.1086/323405

Cited by 42 publications

(46 citation statements)

References 11 publications

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“…Although the majority of these species have been associated with infection of the gastrointestinal tract, several species, e.g., H. bilis, H. canis, H. cinaedi, H. canadensis, H. fennelliae, and H. pullorum, have been found to cause bacteremia and systemic diseases (5,8,9,12,13,19). The course of our patient's illness was similar to those of other X-linked agammaglobulinemia patients (1,6,7,10,12,16,22). Skin lesions raised the suspicion of bacterial infection; however, the etiological agent initially remained unidentified since routine cultures were negative, leading to empirical antibiotic treatment.…”

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confidence: 63%

Bacteremia Caused by a Novel Helicobacter Species in a 28-Year-Old Man with X-Linked Agammaglobulinemia

et al. 2010

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We report on a case of bacteremia caused by a previously unknown urease-negative Helicobacter strain, IMMIB HP-28/08, isolated from blood cultures of a 28-year-old man with X-linked agammaglobulinemia. The identification of the isolate was based on 16S rRNA gene sequencing. In the phylogenetic tree, the isolate fell into a cluster which included Helicobacter canadensis, Helicobacter equorum, and Helicobacter pullorum. This is the first report of bacteremia caused by this fastidious organism. Further investigations are necessary to determine the potential role of this species as a pathogen of bloodstream infections. CASE REPORTThe patient was a 28-year-old man with X-linked agammaglobulinemia, which was diagnosed at age 8. He was started on monthly intravenous (i.v.) immunoglobulin therapy and remained relatively free of symptoms until 2004, when he noticed a demarcated, hyperpigmented, painful macule on the extensor surface of his right lower leg. He appeared healthy and received several empirical antibiotic therapies on suspicion of bacterial infection. He was admitted to our hospital in May 2006 with several hyperpigmented macules on the flexor surfaces of both forearms, his left ankle, and the extensor surface of his right lower leg and systemic symptoms of fatigue, night sweats, and pain in his right lower leg (Fig. 1). Laboratory analysis showed leucocytosis with a count of 23,700 leukocytes/liter with 89% neutrophils in the differential blood count, and the C-reactive protein (CRP) (115 mg/liter) and erythrocyte sedimentation rate (44 mm/h) were elevated. Several blood cultures and microbiological, virological, and autoimmune serological parameters remained negative. Next, the patient was treated empirically with multiple courses of antibiotics, including ampicillin-sulbactam, ciprofloxacin, clarithromycin, ceftazidime, and clindamycin. However, antibiotics were associated with only a brief improvement of symptoms. Skin biopsy gave the suspicion of fibrosing lymphocytic panniculitis, and thus, the patient was put on immunosuppressive therapy with tacrolimus in December 2006. Four weeks after starting immunosuppressive therapy, the patient had severe pains in his lower right leg and became septic. Blood cultures taken at this time signaled bacterial growth after 3 days of incubation. Gram staining revealed the presence of Gram-negative thin rods with a fusiform appearance. Later, this was identified by means of the 16S rRNA gene sequence analysis as a Helicobacter canadensis-like organism.Subsequently, the patient was treated with intravenous amoxicillin and gentamicin. Laboratory results (CRP, erythrocyte sedimentation rate, leukocytes, and differential blood count) normalized rapidly; however, the hyperpigmented macula cleared only slowly. It took several courses of intravenous therapy, with amoxicillin and gentamicin switched to imipenem and fosfomycin due to ototoxicity associated with gentamicin, to prevent a recurrence of symptoms. After a total of 40 weeks of i.v. therapy, the patient was stable on a...

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confidence: 63%

Bacteremia Caused by a Novel Helicobacter Species in a 28-Year-Old Man with X-Linked Agammaglobulinemia

et al. 2010

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“…Non-pylori Helicobacter species (including H. canis) that colonize the mucosal surfaces of the intestinal tract and/or liver are mostly zoonotic pathogens of mammals or birds, infrequently reported to cause invasive infection in humans (1). Especially susceptible are humans with immunodeficiencies such as AIDS (2) and X-agammaglobulinemia (3)(4)(5). These organisms may be associated with diarrhea, bacteremia, and/or visceral disease, including cholecystitis, hepatitis, and possibly hepatic cancer (6), and have been detected in livers of patients with ulcerative colitis and concomitant liver disease, as well as in children with other liver diseases (7,8).…”

Section: Case Reportmentioning

confidence: 99%

Helicobacter canis Bacteremia in a Patient with Fever of Unknown Origin

et al. 2013

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A 57-year-old woman with common variable immune deficiency and liver failure of unknown etiology presented with recurrent fevers over a 5-month period. She was found to have Helicobacter canis bacteremia. Immunocompromised hosts with exposure to cats or dogs may be at risk for infection with this organism, which may be challenging to diagnose. CASE REPORTA 57-year-old woman from Montana was referred to our institution for Infectious Diseases consultation for recurrent fever and chills for 3 months. She had common variable immunodeficiency (for which she received intermittent intravenous immune globulin infusions), and presumed pulmonary sarcoidosis (treated intermittently with prednisone), and had undergone prior splenectomy. She had a 2-year history of elevated alkaline phosphatase. She had developed intermittent fever 3 months prior to consultation and had been previously hospitalized locally for fever and clinical sepsis with negative blood cultures. Laparoscopic liver biopsy had shown mildly active steatohepatitis with periportal fibrosis, and had been procedurally complicated by intra-abdominal hemorrhage. She had been hospitalized for fever on several subsequent occasions and had received multiple courses of empirical antibacterial therapy despite negative blood cultures and no clear definition of an infectious process.Upon presentation for initial Infectious Diseases consultation, fevers were described as low grade and intermittent and were accompanied by chills and sweats. She had taken prednisone (20 mg daily) for the previous 3 months for abdominal pain ascribed to possible gastrointestinal sarcoidosis. She reported a 1-year history of diarrhea and a 2-year 160-pound weight loss. A colonoscopic biopsy 5 months prior had shown findings consistent with common variable immunodeficiency. Stool studies showed no evidence of enteric infection. She had two dogs and three cats. The patient's blood cultures, serologic testing for HIV, blood PCR for cytomegalovirus, and chest X-ray were negative. Serum IgG was 408 mg/dl (767 to 1,590 mg/dl), and IgA was 2 mg/dl (61 to 356 mg/dl). Total bilirubin was 0.4 mg/dl (0.1 to 1 mg/dl), alkaline phosphatase 566 units/liter (46 to 118 units/liter), and alanine transaminase 155 units/liter (7 to 45 units/liter). Computed tomography of the chest, abdomen, and pelvis was notable only for a fluid collection around the liver. Aspiration of this collection was attempted, but no fluid was obtained, and the finding was ascribed to a resolving hematoma. A positron emission tomography scan was unremarkable. She was empirically treated with ciprofloxacin and metronidazole for 5 days. Fever resolved and she was discharged home.She was admitted to a local hospital 2 months later for recurring fevers and worsening cholestasis. Total bilirubin was 21.6 mg/dl (0.1 to 1 mg/dl), alkaline phosphatase 359 units/liter (46 to 118 units/liter), and alanine transaminase 384 units/liter (7 to 45 units/liter). She was subsequently transferred to our hospital, at which time she was afebrile in the a...

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“…Considerable diversity in the 16S rRNA genes of 35 F. rappini strains has been observed so that the International Subcommittee on the Taxonomy of Campylobacter and Helicobacter decided that F. rappini is not a well-defined species but encompasses multiple Helicobacter species (3,27). Therefore, the several interesting reports of bacteremia due to Helicobacter (Flexispira) rappini in HIVseropositive patients and in a patient with X-linked hypogammaglobulinemia should be interpreted with care (8,20,21,30).…”

Section: Discussionmentioning

confidence: 99%

Genotypic Identification of Erythromycin-Resistant Campylobacter Isolates as Helicobacter Species and Analysis of Resistance Mechanism

et al. 2003

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The correct identification of Campylobacter species remains cumbersome, especially when conventional biochemical tests and antimicrobial susceptibility patterns are used for a phenotypical identification. Correct identification is important for epidemiological purposes and for studying changes in antimicrobial resistance patterns. Six erythromycin-resistant campylobacter strains were investigated by 16S ribosomal DNA (rDNA) sequencing, 23S rDNA sequencing, and restriction fragment length polymorphism analysis of a putative heme-copper oxidase domain described as being specific for thermophilic Campylobacter species. Three erythromycin-resistant isolates from feces of human immunodeficiency virus (HIV)-seropositive patients with diarrhea and one blood isolate of from HIV-seropositive patient with cellulitis were identified by 16S rDNA analysis as Helicobacter cinaedi, whereas 23S rDNA sequencing suggested Wolinella succinogenes. The 16S rDNA sequence data of fecal isolates of two patients with travelers diarrhea revealed Helicobacter pullorum and were also in contrast with 23S rDNA sequencing. Of 4 H. cinaedi isolates, 1 contained the putative heme-copper oxidase gene thought to be specific for thermophilic species. The six erythromycin-resistant Helicobacter species had a similar point mutation A2143G in 23S rDNA resembling the macrolides resistance in Helicobacter pylori. We conclude that 16S rDNA sequencing should be preferred to 23S rDNA analysis and that macrolide-resistant campylobacter strains should be investigated by this approach for a correct identification.

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Recurrent Bacteremia and Multifocal Lower Limb Cellulitis Due toHelicobacter‐Like Organisms in a Patient with X‐Linked Hypogammaglobulinemia

Cited by 42 publications

References 11 publications

Bacteremia Caused by a Novel Helicobacter Species in a 28-Year-Old Man with X-Linked Agammaglobulinemia

Bacteremia Caused by a Novel Helicobacter Species in a 28-Year-Old Man with X-Linked Agammaglobulinemia

Helicobacter canis Bacteremia in a Patient with Fever of Unknown Origin

Genotypic Identification of Erythromycin-Resistant Campylobacter Isolates as Helicobacter Species and Analysis of Resistance Mechanism

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