Recurrent Bacteremia Caused by a “ Flexispira ”-Like Organism in a Patient with X-Linked (Bruton’s) Agammaglobulinemia

Abstract

Helicobacter spp., except for Helicobacter cinaedi, have only rarely been reported in cases of septicemia. A patient with X-linked (Bruton’s) agammaglobulinemia was found to have persistent sepsis with a Helicobacter-like organism despite multiple courses of antibiotics. His periods of sepsis were associated with leg swelling thought to be consistent with cellulitis. The organism was fastidious and required a microaerophilic environment containing H2 for growth. Optimal growth was observed at 35 to 37°C on sheep blood, CDC anaerobe, and Bordet-Gengou agars. Serial subcultures every 4 to 5 days were required to maintain viability. The organism was strongly urease positive and showed highest relatedness toHelicobacter-like organisms with the vernacular name “Flexispira rappini” by 16S rRNA gene sequence analysis. Genomic DNA hybridization studies, however, found 24 to 37% relatedness to “F. rappini” and even less to otherHelicobacter spp. Although the organism phenotypically resembles “Flexispira” and Helicobacter, it is thought to represent a new taxon. The patient’s infection was eventually cleared with a prolonged (5-month) course of intravenous imipenem and gentamicin.

Experimental Helicobacter marmotae infection in A/J mice causes enterohepatic disease

“…The liver of a puppy with multifocal hepatitis was colonized with Helicobacter canis, and the organism was cultured from diarrhoeic faeces of a child and bacteraemic humans (Burnens et al, 1993;Fox et al, 1996a). 'Helicobacter rappini' (now classified in the Helicobacter bilis taxon) was recovered from ovine fetuses with hepatic necrosis, and has been described in association with gastroenteritis and bacteraemia in humans (Archer et al, 1988;Kirkbride et al, 1985;Weir et al, 1999). A novel helicobacter belonging to the H. bilis taxon has been identified in the livers and colons of hamsters with hepatobiliary and intestinal disease (Fox et al, 2009).…”

Pyoderma gangrenosum-like ulcer caused by Helicobacter cinaedi in a patient with x-linked agammaglobulinaemia

“…Over the past decade, Helicobacter and Campylobacter species have been recognized as significant pathogens in patients with XLA (Table 1). 6 They have been reported to cause a range of clinical symptoms including fever, chronic leg ulcers resembling PG, 3–5 transient joint swellings 7,8 cellulitis, synovitis, osteomyelitis, bacteraemia and diarrhoea.…”

Identification of a Unique Helicobacter Species by 16S rRNA Gene Analysis in an Abdominal Abscess from a Patient with X-Linked Hypogammaglobulinemia

“…Helicobacters have been identified in the gastrointestinal tract, liver, gallbladder, bile, peripheral blood, and joint effusions. Flexispira rappini, which is also considered a helicobacter, has been isolated from stool specimens from a diarrhea patient (3), and bacteremia in a child with pneumonia (10), in an adult patient undergoing hemodialysis (12), and in a patient with X-linked (Bruton) agammaglobulinemia (14) has been reported.…”

“…Helicobacters are very fastidious organisms. Isolation of some helicobacters, e.g., F. rappini, requires the use of microaerophilic conditions with H 2 , which is not a common feature of the microaerophilic systems used by clinical laboratories (10,14). Even if primary cultures are obtained, subculture attempts may not turn out to be successful (11).…”

“…Several species of Helicobacter, including H. cinaedi, H. fennelliae, H. westmeadii, H. sp. strain Mainz, and F. rappini, have been isolated from immunocompromised patients (1,3,6,7,10,(13)(14)(15). Recently, a case of septic shock due to H. fennelliae was reported (6).…”