|
1
|
Zuercher H, Daneshmand A, Stolow E, Giansiracusa M, Allan R, Sapounas A. Coexistence of Intestinal Spirochetosis and Colorectal Cancer: Could the Coil be Carcinogenic? ACG Case Rep J 2024; 11:e01557. [PMID: 39559787 PMCID: PMC11573331 DOI: 10.14309/crj.0000000000001557] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/20/2024] [Accepted: 10/16/2024] [Indexed: 11/20/2024] Open
Abstract
Intestinal spirochetosis (IS) is an infectious gastrointestinal disease caused by Brachyspira bacteria. We detail an exceedingly rare case of IS with concomitant invasive colorectal adenocarcinoma (CRC) in a 58-year-old man presenting with abdominal discomfort and fever. Colonoscopic evaluation revealed abnormal-appearing, nodular cecal mucosa and a 35 mm rectosigmoid mass. Histopathology confirmed IS infection and CRC. Our case report is the first to detail IS diagnosed concurrently with colorectal cancer. It highlights the necessity of a high index of suspicion for IS in patients presenting with abdominal discomfort and endoscopic evidence of irregular nodular mucosa, particularly in the setting of suspected CRC. It further details potential pathophysiologic links between IS and colorectal malignancy.
Collapse
Affiliation(s)
- Hannah Zuercher
- Divison of Internal Medicine, University of Florida College of Medicine, Gainesville, FL
| | - Arvin Daneshmand
- Divison of Internal Medicine, University of Florida College of Medicine, Gainesville, FL
| | - Eugene Stolow
- Division of Gastroenterology, Hepatology, and Nutrition, University of Florida College of Medicine, North Florida/South Georgia Veterans Health System, Gainesville, FL
| | - Matthew Giansiracusa
- Divison of Internal Medicine, University of Florida College of Medicine, Gainesville, FL
| | - Robert Allan
- Pathology and Laboratory Medicine Service, Department of Pathology, Immunology and Laboratory Medicine, University of Florida College of Medicine, North Florida/South Georgia Veterans Health System, Gainesville, FL
| | - Antonios Sapounas
- Division of Gastroenterology, Hepatology, and Nutrition, University of Florida College of Medicine, North Florida/South Georgia Veterans Health System, Gainesville, FL
| |
Collapse
|
|
2
|
Eslick GD, Fan K, Nair PM, Burns GL, Hoedt EC, Keely S, Talley NJ. Clinical and Pathologic Factors Associated With Colonic Spirochete (Brachyspira pilosicoli and Brachyspira aalborgi) Infection: A Comprehensive Systematic Review and Pooled Analysis. Am J Clin Pathol 2023; 160:335-340. [PMID: 37289435 PMCID: PMC10682506 DOI: 10.1093/ajcp/aqad063] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/21/2023] [Accepted: 05/04/2023] [Indexed: 06/09/2023] Open
Abstract
OBJECTIVES This study aims to determine what pathologic and clinical factors differentiate Brachyspira species that may be useful to clinicians and pathologists. METHODS We identified 21 studies of Brachyspira infection with individual patient information (n = 113) and conducted a pooled analysis comparing each species. RESULTS There were differences in the pathologic and clinical profiles of each Brachyspira species. Patients infected with Brachyspira pilosicoli infection were more likely to have diarrhea, fever, HIV, and immunocompromised conditions. Those patients infected with Brachyspira aalborgi were more likely to have lamina propria inflammation. CONCLUSIONS Our novel data provide potential insights into the pathogenic mechanism(s) and the specific risk factor profile of Brachyspira species. This may be clinically useful when assessing and managing patients.
Collapse
Affiliation(s)
- Guy D Eslick
- NHMRC Centre for Research Excellence in Digestive Health, The University of Newcastle, Hunter Medical Research Institute (HMRI), Callaghan, Australia
| | - Kening Fan
- NHMRC Centre for Research Excellence in Digestive Health, The University of Newcastle, Hunter Medical Research Institute (HMRI), Callaghan, Australia
| | - Prema M Nair
- NHMRC Centre for Research Excellence in Digestive Health, The University of Newcastle, Hunter Medical Research Institute (HMRI), Callaghan, Australia
| | - Grace L Burns
- NHMRC Centre for Research Excellence in Digestive Health, The University of Newcastle, Hunter Medical Research Institute (HMRI), Callaghan, Australia
| | - Emily C Hoedt
- NHMRC Centre for Research Excellence in Digestive Health, The University of Newcastle, Hunter Medical Research Institute (HMRI), Callaghan, Australia
| | - Simon Keely
- NHMRC Centre for Research Excellence in Digestive Health, The University of Newcastle, Hunter Medical Research Institute (HMRI), Callaghan, Australia
| | - Nicholas J Talley
- NHMRC Centre for Research Excellence in Digestive Health, The University of Newcastle, Hunter Medical Research Institute (HMRI), Callaghan, Australia
| |
Collapse
|
|
3
|
Pérez-Tanoira R, Tamarit MDP, Montaña AMV, Carmena D, Köster P, Górgolas M, Fortes Alen JR, Cabello-Úbeda A, Prieto-Pérez L. Increased Prevalence of Symptomatic Human Intestinal Spirochetosis in MSM with High-Risk Sexual Behavior in a Cohort of 165 Individuals. Trop Med Infect Dis 2023; 8:tropicalmed8050250. [PMID: 37235298 DOI: 10.3390/tropicalmed8050250] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/03/2023] [Revised: 04/22/2023] [Accepted: 04/24/2023] [Indexed: 05/28/2023] Open
Abstract
Human intestinal spirochetosis (HIS) can cause gastrointestinal symptoms, although asymptomatic infections have been described. Individuals from low-income countries, people living with HIV, and men who have sex with men (MSM) show increased risk. A retrospective review of all patients diagnosed with HIS (n = 165) between January 2013 and October 2020 at a tertiary hospital in Madrid, Spain, was performed to assess risk factors for symptomatic HIS, symptoms, and response to treatment. Most patients were male (n = 156; 94.5%), 86.7% were MSM, and 23.5% practiced chemsex, of whom most were symptomatic (p = 0.039). Most patients (78.4%) reported unprotected oral-anal intercourse. A total of 124 (81.1%) were symptomatic; diarrhea was the most common complaint (68.3%). Multivariable regression showed increased odds of symptoms associated with age under 41 (odds ratio 5.44, 95% CI 1.87-15.88; p = 0.002). Colonoscopy was normal in 153 (92.7%). Furthermore, 66.7% presented previous or concomitant sexually transmitted diseases (STDs). Among the patients, 102 underwent testing for other gastrointestinal pathogens, with positive results in 20 (19.6%). All symptomatic patients without concomitant gastrointestinal infection presenting improvement on follow-up (42 of 53) had received either metronidazole or doxycycline (p = 0.049). HIS should be considered as a cause of chronic diarrhea in MSM with high-risk sexual behavior after other causes have been ruled out; treatment with metronidazole is recommended. Coinfection with other STDs is common.
Collapse
Affiliation(s)
- Ramón Pérez-Tanoira
- Department of Microbiology, Príncipe de Asturias University Hospital, 28805 Alcalá de Henares, Spain
- Department of Biomedicine and Biotechnology, University of Alcalá, 28040 Madrid, Spain
| | | | | | - David Carmena
- Parasitology Reference and Research Laboratory, Spanish National Centre for Microbiology, Health Institute Carlos III, Majadahonda, 28040 Madrid, Spain
| | - Pamela Köster
- Parasitology Reference and Research Laboratory, Spanish National Centre for Microbiology, Health Institute Carlos III, Majadahonda, 28040 Madrid, Spain
| | - Miguel Górgolas
- Department of Infectious Diseases, IIS-Fundación Jiménez Díaz, 28040 Madrid, Spain
| | - José R Fortes Alen
- Department of Pathology, IIS-Fundación Jiménez Díaz, 28040 Madrid, Spain
| | | | - Laura Prieto-Pérez
- Department of Infectious Diseases, IIS-Fundación Jiménez Díaz, 28040 Madrid, Spain
| |
Collapse
|
|
4
|
Novotny S, Mizrahi J, Yee EU, Clores MJ. Incidental diagnosis of intestinal spirochetosis in a patient with chronic hepatitis B: A case report. World J Clin Infect Dis 2022; 12:69-75. [DOI: 10.5495/wjcid.v12.i2.69] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/22/2022] [Revised: 07/01/2022] [Accepted: 08/22/2022] [Indexed: 02/05/2023] Open
Abstract
BACKGROUND Intestinal spirochetosis (IS) is caused by Brachyspira colonization of the gastrointestinal tract. Some patients are asymptomatic, while others present with gastrointestinal complaints such as abdominal pain, diarrhea, or gastrointestinal bleeding. However, the clinical significance of asymptomatic IS is unclear, and guidelines are lacking regarding decision to treat.
CASE SUMMARY A 73-year-old male with peptic ulcer disease and gastroesophageal reflux was evaluated for elevated liver enzymes. He was diagnosed with chronic hepatitis B virus and prescribed entecavir. Additionally, he was leukopenic and had stage 4 liver fibrosis on transient elastography. After 5 mo, the patient returned for esophagogastroduodenoscopy and screening colonoscopy. He denied any gastrointestinal symptoms at that time. Findings included grade I distal esophageal varices, mild portal hypertensive gastropathy, and patchy nodular gastric antral mucosa. On colonoscopy, several polyps were removed. Hematoxylin and eosin stain of mucosa adjacent to the polyps revealed a “false brush border,” and Steiner stain identified spirochetes adherent to the mucosa. These pathology findings confirmed the diagnosis of IS. He was managed conservatively with careful observation and without antibiotic therapy via a multidisciplinary approach between gastroenterology and infectious disease. He remained asymptomatic at the 7-wk follow-up.
CONCLUSION This case reports the finding of incidental, asymptomatic IS in a leukopenic patient with hepatitis B virus. Conservative management was appropriate.
Collapse
Affiliation(s)
- Samantha Novotny
- Renaissance School of Medicine, Stony Brook University, Stony Brook, NY 11794, United States
| | - Joseph Mizrahi
- Division of Gastroenterology and Hepatology, Stony Brook Medicine, Stony Brook, NY 11794, United States
| | - Eric U Yee
- Department of Pathology, Stony Brook Medicine, Stony Brook, NY 11794, United States
| | - Michael J Clores
- Division of Gastroenterology and Hepatology, Stony Brook Medicine, Stony Brook, NY 11794, United States
| |
Collapse
|
|
5
|
Fan K, Eslick GD, Nair PM, Burns GL, Walker MM, Hoedt EC, Keely S, Talley NJ. Human intestinal spirochetosis, irritable bowel syndrome, and colonic polyps: A systematic review and meta-analysis. J Gastroenterol Hepatol 2022; 37:1222-1234. [PMID: 35385602 PMCID: PMC9545717 DOI: 10.1111/jgh.15851] [Citation(s) in RCA: 20] [Impact Index Per Article: 6.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/15/2022] [Accepted: 03/24/2022] [Indexed: 12/09/2022]
Abstract
Human colonic spirochetosis (CS) is usually due toBrachyspira pilosicolior Brachyspira aalborgiinfection. While traditionally considered to be commensal bacteria, there are scattered case reports and case series of gastrointestinal (GI) symptoms in CS and reports of colonic polyps with adherent spirochetes. We performed a systematic review and meta-analysis investigating the association between CS and GI symptoms and conditions including the irritable bowel syndrome (IBS) and colonic polyps. Following PRISMA 2020 guidelines, a systematic search of Medline, CINAHL, EMBASE, and Web of Science was performed using specific keywords for CS and GI disease. Pooled odds ratios (ORs) and 95% confidence intervals (CIs) were calculated using a random-effects model. Of 75 studies identified in the search, 8 case-control studies met the inclusion criteria for meta-analysis and 67 case series studies met the inclusion criteria for pooled prevalence analysis. CS was significantly associated with diarrhea (n = 141/127, cases/controls, OR: 4.19, 95% CI: 1.72-10.21, P = 0.002) and abdominal pain (n = 64/65, OR: 3.66, 95% CI: 1.43-9.35, P = 0.007). CS cases were significantly more likely to have Rome III-diagnosed IBS (n = 79/48, OR: 3.84, 95% CI: 1.44-10.20, P = 0.007), but not colonic polyps (n = 127/843, OR: 8.78, 95% CI: 0.75-103.36, P = 0.084). In conclusion, we found evidence of associations between CS and both diarrhea and IBS, but not colonic polyps. CS is likely underestimated due to suboptimal diagnostic methods and may be an overlooked risk factor for a subset of IBS patients with diarrhea.
Collapse
Affiliation(s)
- Kening Fan
- School of Biomedical Sciences and Pharmacy, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
| | - Guy D Eslick
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
- School of Medicine and Public Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
| | - Prema M Nair
- School of Biomedical Sciences and Pharmacy, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
| | - Grace L Burns
- School of Biomedical Sciences and Pharmacy, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
| | - Marjorie M Walker
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
- School of Medicine and Public Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
| | - Emily C Hoedt
- School of Biomedical Sciences and Pharmacy, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
| | - Simon Keely
- School of Biomedical Sciences and Pharmacy, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
| | - Nicholas J Talley
- Hunter Medical Research InstituteNew Lambton HeightsNew South WalesAustralia
- NHMRC Centre for Research Excellence in Digestive Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
- Australian Gastrointestinal Research Alliance (AGIRA)NewcastleNew South WalesAustralia
- School of Medicine and Public Health, College of Health, Medicine and WellbeingUniversity of NewcastleNewcastleNew South WalesAustralia
| |
Collapse
|
|
6
|
Miwa W, Hiratsuka T, Sato K, Kato Y. Pneumatosis cystoides intestinalis accompanied by intestinal spirochetosis. Clin J Gastroenterol 2020; 13:545-551. [PMID: 31898208 DOI: 10.1007/s12328-019-01087-2] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/06/2019] [Accepted: 12/18/2019] [Indexed: 11/28/2022]
Abstract
Pneumatosis cystoides intestinalis (PCI) is a rare but well-recognized entity characterized by multiple gas-filled cysts in the intestinal wall. Although the pathogenesis of PCI remains unclear, several theories, including a bacterial theory, have been postulated. Intestinal spirochetosis (IS) is an uncommon condition defined by the presence of spirochetes attached to the surface of the colonic epithelium. The nature of IS as a commensal or pathogenic process remains debatable. However, recent evidence supports the idea that IS can be invasive and highly pathogenic in both immunocompromised and immunocompetent individuals. We present the case of a 35-year-old asymptomatic and immunocompetent man who underwent colonoscopy because of a positive fecal blood test. Multiple submucosal cystic lesions were detected accompanied by erythematous areas along the ascending colon. Computed tomography-colonography and biopsy specimens from the erythematous areas confirmed coexisting PCI and IS. Both PCI and IS recovered completely 3 months after administration of metronidazole. To the best of our knowledge, this case represents only the second report of the extremely rare concurrence of PCI with IS. Taking into account the published literature, we also discuss the possibility that the development of PCI may be related to IS.
Collapse
Affiliation(s)
- Wataru Miwa
- Division of Internal Medicine, Hiratsuka Gastroenterological Hospital, 3-2-16 Nishiikebukuro, Toshima-ku, Tokyo, 171-0021, Japan.
| | - Takashi Hiratsuka
- Division of Internal Medicine, Hiratsuka Gastroenterological Hospital, 3-2-16 Nishiikebukuro, Toshima-ku, Tokyo, 171-0021, Japan
| | - Ken Sato
- Division of Surgery, Hiratsuka Gastroenterological Hospital, Tokyo, Japan
| | - Yo Kato
- Department of Pathology, Nikko Medical Center, Dokkyo Medical University, Tochigi, Japan
| |
Collapse
|
|
7
|
Iwamoto J, Adachi Y, Honda A, Monma T, Matsuzaki Y. The comparison of the intensity of human intestinal spirochetes between Brachyspira pilosicoli and Brachyspira aalborgi infections. J Clin Biochem Nutr 2018; 64:86-90. [PMID: 30705517 PMCID: PMC6348408 DOI: 10.3164/jcbn.18-68] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/10/2018] [Accepted: 07/31/2018] [Indexed: 11/22/2022] Open
Abstract
The agglutination titers of Brachyspira pilosicoli (B. pilosicoli) and Brachyspira aalborgi (B. aalborgi) were examined in colitis patients with human intestinal spirochetes. Among three cases of colitis patients, the titer of B. pilosicoli was extremely high in two cases while the titer of B. aalborgi was extremely high in one case. These three cases had symptoms of colitis, such as watery diarrhea, and we diagnosed the case as Brachyspira- related colitis. These findings suggest that the agglutination titers of Brachyspira may be useful in cases of Brachyspira- related colitis. Severe symptoms, such as abdominal pain and diarrhea, were observed in cases with high antibody titer of B. aalborgi, as well as B. pilosicoli, indicating that B. aalborgi could also cause symptomatic colitis.
Collapse
Affiliation(s)
- Junichi Iwamoto
- Department of Gastroenterology, Tokyo Medical University Ibaraki Medical Center, 3-20-1 Ami-machi Chuo, Inashiki-gun, Ibaraki 300-0395, Japan
| | - Yoshikazu Adachi
- Animal Health Laboratory, School of Agriculture, Ibaraki University, 3-21-1 Ami, Ibaraki 300-0393, Japan
| | - Akira Honda
- Joint Research Center, Tokyo Medical University Ibaraki Medical Center, 3-20-1 Ami-machi Chuo, Inashiki-gun, Ibaraki 300-0395, Japan
| | - Tadakuni Monma
- Department of Gastroenterology, Tokyo Medical University Ibaraki Medical Center, 3-20-1 Ami-machi Chuo, Inashiki-gun, Ibaraki 300-0395, Japan
| | - Yasushi Matsuzaki
- Department of Gastroenterology, Tokyo Medical University Ibaraki Medical Center, 3-20-1 Ami-machi Chuo, Inashiki-gun, Ibaraki 300-0395, Japan
| |
Collapse
|
|
8
|
The Spirochete Brachyspira pilosicoli, Enteric Pathogen of Animals and Humans. Clin Microbiol Rev 2017; 31:31/1/e00087-17. [PMID: 29187397 DOI: 10.1128/cmr.00087-17] [Citation(s) in RCA: 39] [Impact Index Per Article: 4.9] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/12/2023] Open
Abstract
Brachyspira pilosicoli is a slow-growing anaerobic spirochete that colonizes the large intestine. Colonization occurs commonly in pigs and adult chickens, causing colitis/typhlitis, diarrhea, poor growth rates, and reduced production. Colonization of humans also is common in some populations (individuals living in village and peri-urban settings in developing countries, recent immigrants from developing countries, homosexual males, and HIV-positive patients), but the spirochete rarely is investigated as a potential human enteric pathogen. In part this is due to its slow growth and specialized growth requirements, meaning that it is not detectable in human fecal samples using routine diagnostic methods. Nevertheless, it has been identified histologically attached to the colon and rectum in patients with conditions such as chronic diarrhea, rectal bleeding, and/or nonspecific abdominal discomfort, and one survey of Australian Aboriginal children showed that colonization was significantly associated with failure to thrive. B. pilosicoli has been detected in the bloodstream of elderly patients or individuals with chronic conditions such as alcoholism and malignancies. This review describes the spirochete and associated diseases. It aims to encourage clinicians and clinical microbiologists to consider B. pilosicoli in their differential diagnoses and to develop and use appropriate diagnostic protocols to identify the spirochete in clinical specimens.
Collapse
|