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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.
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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
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2
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Prieto Pérez L, Górgolas Hernández-Mora M. An approach to the correct interpretation of intestinal spirochaetosis. ENFERMEDADES INFECCIOSAS Y MICROBIOLOGIA CLINICA (ENGLISH ED.) 2024; 42:229-230. [PMID: 38704192 DOI: 10.1016/j.eimce.2024.03.005] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 03/04/2024] [Accepted: 03/04/2024] [Indexed: 05/06/2024]
Affiliation(s)
- Laura Prieto Pérez
- División de Enfermedades Infecciosas, Hospital Universitario Fundación Jiménez Díaz, Madrid, Spain; Departamento de Medicina, Universidad Autónoma de Madrid, Spain
| | - Miguel Górgolas Hernández-Mora
- División de Enfermedades Infecciosas, Hospital Universitario Fundación Jiménez Díaz, Madrid, Spain; Departamento de Medicina, Universidad Autónoma de Madrid, Spain.
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3
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Rabuszko L, Chessell C, Llewellyn C, Richardson D. Factors and presenting symptoms associated with intestinal spirochaetosis in men who have sex with men: A systematic review. J Eur Acad Dermatol Venereol 2024; 38:680-686. [PMID: 38031524 DOI: 10.1111/jdv.19689] [Citation(s) in RCA: 4] [Impact Index Per Article: 4.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/29/2023] [Accepted: 10/31/2023] [Indexed: 12/01/2023]
Abstract
The transmission dynamics and presenting symptoms of intestinal spirochaetosis (Brachyspira pilosicoli, Brachyspira aalborgi) are poorly understood but is seen more frequently in men who have sex with men (MSM) where sexual transmission may occur. We aimed to systemically review the literature to define the factors and presenting symptoms associated with intestinal spirochaetosis in MSM. We systematically searched three bibliographical databases (MEDLINE, EMBASE and Scopus) for manuscripts written in English up to November 2022. The primary author conducted an initial screen and three authors conducted independent reviews to determine the final eligible manuscripts. We only included manuscripts where there were specific data on factors and presenting symptoms associated with intestinal spirochaetosis in MSM. Fifteen manuscripts published between 1981 and 2022 met the inclusion criteria that included 188 MSM. Sexually transmitted intestinal spirochaetosis in MSM was associated with-living with HIV (including having a low CD4 count <200 mm3), having a concomitant sexually transmitted infection, a history of a sexually transmitted infection, co-infection with intestinal parasites; and sexual behaviour including oro-anal sexual contact, oro-genital contact, fisting, condom-less anal sex, receptive anal sex, multiple non-regular sexual partners and increased number of lifetime sexual partners. The presenting symptoms of MSM with intestinal spirochaetosis were diarrhoea, abdominal pain, constitutional symptoms and proctitis, including rectal pain, rectal bleeding rectal discharge, flatulence and tenesmus. We have highlighted some biological and behavioural factors and presenting symptoms associated with intestinal spirochaetosis in MSM which provide insight for clinicians, guidelines and future public health control strategies.
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Affiliation(s)
- Lucy Rabuszko
- Department of Primary Care and Public Health, Brighton and Sussex Medical School, Brighton, UK
| | - Callum Chessell
- Department of Primary Care and Public Health, Brighton and Sussex Medical School, Brighton, UK
| | - Carrie Llewellyn
- Department of Primary Care and Public Health, Brighton and Sussex Medical School, Brighton, UK
| | - Daniel Richardson
- Department of Primary Care and Public Health, Brighton and Sussex Medical School, Brighton, UK
- Department of Sexual Health and HIV, University Hospitals Sussex NHS Foundation Trust, Brighton, UK
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4
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Ramanathan B, Ramachandran V, Rana A, McDonald C. A Case Study: Incidental Finding of Human Intestinal Spirochetosis in Screening Colonoscopy. Cureus 2024; 16:e55422. [PMID: 38567235 PMCID: PMC10985559 DOI: 10.7759/cureus.55422] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 03/03/2024] [Indexed: 04/04/2024] Open
Abstract
Human intestinal spirochetosis (HIS) is a rare occurrence. We present an interesting case study on an asymptomatic over-60-year-old male who was incidentally discovered to have HIS following a colonoscopy that was conducted for his positive fecal occult blood test (FOBT). Histopathology of the colonic biopsy proved the presence of human intestinal spirochetosis; however, as he was asymptomatic, treatment was not initiated in his case. We discuss here the prevalence, presentation, diagnostic methods, and treatment of colonic HIS.
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Affiliation(s)
| | | | - Abdul Rana
- Surgery, Lyell Mcewin Hospital, Adelaide, AUS
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5
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Chaar AJ, Arbelaez JS, Vargas MJ, Cañadas RA. Human intestinal spirochetosis as a cause of chronic diarrhea in an HIV-positive patient: A case report. REVISTA DE GASTROENTEROLOGIA DE MEXICO (ENGLISH) 2023; 88:443-446. [PMID: 37833137 DOI: 10.1016/j.rgmxen.2023.09.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 05/23/2023] [Accepted: 07/11/2023] [Indexed: 10/15/2023]
Affiliation(s)
- A J Chaar
- Departamento de Gastroenterología y Endoscopia, Hospital Universitario San Ignacio, Pontificia Universidad Javeriana, Bogotá, Colombia.
| | - J S Arbelaez
- Departamento de Patología Clínica, Hospital Universitario San Ignacio, Pontificia Universidad Javeriana, Bogotá, Colombia
| | - M J Vargas
- Departamento de Patología Clínica, Hospital Universitario San Ignacio, Pontificia Universidad Javeriana, Bogotá, Colombia
| | - R A Cañadas
- Departamento de Gastroenterología y Endoscopia, Hospital Universitario San Ignacio, Pontificia Universidad Javeriana, Bogotá, Colombia
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6
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Petruzzellis C, Catino F. Unusual Behavior of Intestinal Spirochetosis: A Case Report. INFECTIOUS DISEASES & IMMUNITY 2023. [PMCID: PMC10368213 DOI: 10.1097/id9.0000000000000095] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 03/20/2025]
Abstract
Abstract
Human intestinal spirochetosis is a rare clinicopathological condition characterized by the presence of spirochetal microorganisms on the surface of the colorectal mucosa and, in some cases, by their invasion into the lamina propria. The most frequent cause of this infection is immunocompromised status. Our case report describes a patient with relevant symptoms and endoscopic alterations, but limited invasion by spirochetes, as shown by histological analysis of ileal and colonic specimens. After histological examination, metronidazole was initiated. This resulted in rapid and complete resolution of the symptoms, which demonstrated an infectious etiology. The peculiarity of our case is that the clinical and endoscopic appearances were typical of the aggressive behavior of the disease, but histological samples showed only superficial colonization of the colonic epithelium without clear invasion of deeper layers. This case report emphasizes that the virulence of spirochetes is not strictly related to intestinal mucosal invasion.
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Affiliation(s)
| | - Federico Catino
- Gastroenterology and Endoscopy Department, Fondazione Poliambulanza Hospital, Brescia, Italy
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7
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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.
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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
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8
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Muacevic A, Adler JR, Fenster M, Correa J, Bansal R. Ulcerative Proctitis: An Unusual Case of Intestinal Spirochetosis. Cureus 2022; 14:e33046. [PMID: 36721604 PMCID: PMC9881603 DOI: 10.7759/cureus.33046] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 12/28/2022] [Indexed: 12/29/2022] Open
Abstract
Human intestinal spirochetosis (HIS) is an uncommon disease characterized by the colonization of spirochetes in the colorectal mucosa and is most often found in individuals who are positive for human immunodeficiency virus (HIV) and in homosexual men. Although HIV is known to cause a variety of infectious colitis, the prevalence has significantly declined with antiretroviral therapy. Intestinal spirochetosis, however, remains meaningful as it can be an infectious cause of colonic ulcerations even with well-controlled HIV. Spirochetosis rarely causes macroscopic changes in the colorectal mucosa and reports of an ulcerated rectum are exceedingly scarce. Here, we report a case of a homosexual man with HIV who is compliant with antiretroviral therapy with high CD4 counts who presented with a six-week history of bloody diarrhea and was found to have multiple ulcerations in the rectosigmoid junction and rectum infected with non-treponemal spirochetes as confirmed on biopsy. To our knowledge, there have not been any reports of multiple rectal ulcerations caused by non-treponemal spirochetes. The patient was treated with metronidazole 500 mg four times daily for 10 days with complete resolution of symptoms. This case is notable as it alerts clinicians to consider intestinal spirochetosis as a differential diagnosis in the workup for bloody stool in the presence of colorectal ulcerations.
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9
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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.
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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
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10
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Pérez Moux RL, Mude P, Doshi S, Madwani K, Barrett A, Yap JEL. Human Intestinal Spirochetosis: A Rare Case of Intermittent Bloating and Hematochezia. Cureus 2022; 14:e25756. [PMID: 35812607 PMCID: PMC9270075 DOI: 10.7759/cureus.25756] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 06/05/2022] [Indexed: 12/02/2022] Open
Abstract
Human intestinal spirochetosis (HIS) is a condition where spirochetes, a group of spiral-shaped bacteria, attach to the apical membrane of the human colorectal epithelium. Although most findings of HIS are simply incidental discoveries found during screening colonoscopies, the ability to mimic the presentation of inflammatory bowel diseases should prompt consideration of this condition as part of a working differential diagnosis. Herein, we present the case of a 57-year-old bisexual, African American male with a medical history of Human Immunodeficiency Virus (HIV) on antiretroviral therapy (ART) with an undetectable viral load that presented for an elective, outpatient colonoscopy after experiencing four months of intermittent bloating and hematochezia. Histologic examination of colonic biopsies confirmed a diagnosis of HIS. The nonspecific clinical presentation in the setting of well-controlled HIV makes HIS a formidable diagnostic challenge that requires increased awareness.
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11
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Fesen JE, Al-Husseinawi EK, Newman JR. Brushing Up on Brush Borders: Intestinal Spirochetosis Diagnosis and Management. Kans J Med 2021; 14:290-291. [PMID: 34868472 PMCID: PMC8641436 DOI: 10.17161/kjm.vol14.15535] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/27/2021] [Accepted: 07/28/2021] [Indexed: 11/30/2022] Open
Affiliation(s)
- Joy E Fesen
- Department of Internal Medicine, Tulane University School of Medicine, New Orleans, LA
| | | | - Jessica R Newman
- Department of Internal Medicine, Division of Infectious Diseases, University of Kansas Medical Center, Kansas City, KS
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12
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Mucosal eosinophilic infiltration may be a characteristic of human intestinal spirochetosis. BMC Infect Dis 2021; 21:721. [PMID: 34332545 PMCID: PMC8325824 DOI: 10.1186/s12879-021-06418-8] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/19/2021] [Accepted: 07/05/2021] [Indexed: 11/17/2022] Open
Abstract
Background Human intestinal spirochetosis (HIS) is an infectious disease of large intestines caused by Brachyspira species, and most HIS cases are asymptomatic or exhibit mild intestinal symptoms. The host reaction to HIS remains unclear, and we examined HIS-related mucosal inflammatory features histologically. Methods From the archival HIS cases in a single medical center, 24 endoscopically taken specimens from 14 HIS cases (male:female = 10:4; 28–73 yrs) were selected as not containing polypoid or neoplastic lesions. Stromal neutrophils, eosinophils, and mast cells, and intraepithelial neutrophils and eosinophils, (sNeu, sEo, sMast, iNeu, and iEo, respectively) were counted, and the presence or absence of lymphoid follicles/aggregates (LFs) was also examined. Association of the above inflammation parameters and spirochetal infection parameters (such as degrees of characteristic fringe distribution, of spirochetal cryptal invasion, and of spirochetal intraepithelial invasion) were also analysed. Results iNeu was observed in 29.2%, iEo in 58.3%, and LFs in 50.0% of the specimens. Maximal counts of sNeu, sEo, sMast, iNeu, and iEo averaged 8.4, 21.5, 6.0, 0.5 and 1.5, respectively. Strong correlation between the maximum counts of iNeu and iEo (p < 0.001, r = 0.81), and correlations between those of iEo and sNeu (p = 0.0012, r = 0.62) and between those of iEo and sEo (p = 0.026, r = 0.45) were observed. iNeu was influenced by fringe formation (p < 0.05) and spirochetal crypt involvement (p < 0.05). Conclusions HIS was accompanied by inflammatory reactions, and among these, mucosal eosinophilic infiltration may be a central indicator and host reaction of HIS.
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Alnimer L, Zakaria A, Warren B. A Case of Human Intestinal Spirochetosis Diagnosed During Screening Colonoscopy. Cureus 2021; 13:e14829. [PMID: 34094781 PMCID: PMC8173371 DOI: 10.7759/cureus.14829] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/03/2023] Open
Abstract
Human intestinal spirochetosis (HIS) is a rare disease and mostly encountered incidentally during colorectal cancer screening colonoscopy. Risk factors include homosexuality and immunocompromised states. Patients are usually asymptomatic; however, chronic diarrhea and bloody stools have been reported in some cases. Diagnosis is usually confirmed by histopathology. A watch-and-see approach is usually acceptable, but successful treatment with Metronidazole has been reported in symptomatic cases. Its clinical significance remains questionable given that patients are mostly asymptomatic.
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Affiliation(s)
- Lynna Alnimer
- Department of Internal Medicine, Ascension Providence Hospital, Michigan State University/College of Human Medicine, Southfield, USA
| | - Ali Zakaria
- Department of Gastroenterology, Ascension Providence Hospital, Michigan State University/College of Human Medicine, Southfield, USA
| | - Bradley Warren
- Department of Gastroenterology, Ascension Providence Hospital, Michigan State University/College of Human Medicine, Southfield, USA
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14
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Motta JP, Wallace JL, Buret AG, Deraison C, Vergnolle N. Gastrointestinal biofilms in health and disease. Nat Rev Gastroenterol Hepatol 2021; 18:314-334. [PMID: 33510461 DOI: 10.1038/s41575-020-00397-y] [Citation(s) in RCA: 154] [Impact Index Per Article: 38.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 11/25/2020] [Indexed: 01/30/2023]
Abstract
Microorganisms colonize various ecological niches in the human habitat, as they do in nature. Predominant forms of multicellular communities called biofilms colonize human tissue surfaces. The gastrointestinal tract is home to a profusion of microorganisms with intertwined, but not identical, lifestyles: as isolated planktonic cells, as biofilms and in biofilm-dispersed form. It is therefore of major importance in understanding homeostatic and altered host-microorganism interactions to consider not only the planktonic lifestyle, but also biofilms and biofilm-dispersed forms. In this Review, we discuss the natural organization of microorganisms at gastrointestinal surfaces, stratification of microbiota taxonomy, biogeographical localization and trans-kingdom interactions occurring within the biofilm habitat. We also discuss existing models used to study biofilms. We assess the contribution of the host-mucosa biofilm relationship to gut homeostasis and to diseases. In addition, we describe how host factors can shape the organization, structure and composition of mucosal biofilms, and how biofilms themselves are implicated in a variety of homeostatic and pathological processes in the gut. Future studies characterizing biofilm nature, physical properties, composition and intrinsic communication could shed new light on gut physiology and lead to potential novel therapeutic options for gastrointestinal diseases.
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Affiliation(s)
- Jean-Paul Motta
- Institute of Digestive Health Research, IRSD, INSERM U1220, Toulouse, France.
| | - John L Wallace
- Department of Physiology & Pharmacology, Cumming School of Medicine, University of Calgary, Calgary, AB, Canada.,Antibe Therapeutics Inc., Toronto, ON, Canada
| | - André G Buret
- Department of Biological Sciences, University of Calgary, Calgary, AB, Canada
| | - Céline Deraison
- Institute of Digestive Health Research, IRSD, INSERM U1220, Toulouse, France
| | - Nathalie Vergnolle
- Institute of Digestive Health Research, IRSD, INSERM U1220, Toulouse, France. .,Department of Physiology & Pharmacology, Cumming School of Medicine, University of Calgary, Calgary, AB, Canada.
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15
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Akay OI, Schmalzle SA. Bloody Diarrhea in a 17-year-old Male. Clin Infect Dis 2021; 70:2013-2015. [PMID: 32291458 DOI: 10.1093/cid/ciz679] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/12/2022] Open
Affiliation(s)
- Okan I Akay
- Division of Infectious Diseases, University of Maryland School of Medicine, Baltimore
| | - Sarah A Schmalzle
- Division of Infectious Diseases, University of Maryland School of Medicine, Baltimore.,Institute of Human Virology, University of Maryland School of Medicine, Baltimore
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16
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Garcia-Hernandez D, Vall-Mayans M, Coll-Estrada S, Naranjo-Hans L, Armengol P, Iglesias MA, Barberá MJ, Arando M. Human intestinal spirochetosis, a sexually transmissible infection? Review of six cases from two sexually transmitted infection centres in Barcelona. Int J STD AIDS 2020; 32:52-58. [PMID: 33232216 DOI: 10.1177/0956462420958350] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/05/2023]
Abstract
Human intestinal spirochetosis (HIS) is a possible cause of chronic diarrhoea and affects mainly men who have sex with men (MSM) and people living with HIV. Diagnosis is based on colon biopsy, where spirochetes can be observed on the luminal surface, especially with the Warthin-Starry stain or similar silver stains. We conducted a retrospective descriptive study of all HIS cases diagnosed in two sexually transmitted infections (STI) centres in Barcelona from 2009 until 2018. The medical histories were reviewed to gather epidemiological, clinical, and diagnostic variables. Six patients were diagnosed with HIS. All the individuals were MSM, with a median age of 31.5 years (interquartile range [IQR] 29.5;49.25) and half of them were living with HIV. Five patients reported condomless anal intercourse and 4 patients had practised oro-anal sex previously. Concomitantly, two of them had rectal gonorrhoea, one had rectal Chlamydia trachomatis and none of them had syphilis. The predominant clinical symptom was diarrhoea (5 patients). All cases were diagnosed by a Warthin-Starry stain on a colon biopsy specimen, and mild inflammatory changes were found in 5 cases. Five patients were treated with metronidazole and one with benzathine penicillin G. Treatment was successful in all the patients. HIS should be considered in patients with chronic diarrhoea who report risky sexual practices and/or concomitant STI. HIS may also be sexually transmitted according to the context.
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Affiliation(s)
| | - Marti Vall-Mayans
- Infectious Diseases Department, STI Unit Vall d'Hebron Drassanes, Hospital Vall d'Hebron, Barcelona, Spain
| | | | - Lola Naranjo-Hans
- Pathology, Consorci Parc de Salut MAR de Barcelona, Barcelona, Spain
| | - Pere Armengol
- Infectious Diseases Department, STI Unit Vall d'Hebron Drassanes, Hospital Vall d'Hebron, Barcelona, Spain
| | | | - María Jesús Barberá
- Infectious Diseases Department, STI Unit Vall d'Hebron Drassanes, Hospital Vall d'Hebron, Barcelona, Spain
| | - Maider Arando
- Infectious Diseases Department, STI Unit Vall d'Hebron Drassanes, Hospital Vall d'Hebron, Barcelona, Spain.,Medicine Department, Universitat Autonoma de Barcelona, Barcelona, Spain
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17
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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.
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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
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Fuentes Valenzuela EA, Oyarzún Bahamonde E, Núñez Rodríguez H. An uncommon cause of chronic abdominal pain and diarrhea. REVISTA ESPANOLA DE ENFERMEDADES DIGESTIVAS 2020; 112:419-420. [PMID: 32338012 DOI: 10.17235/reed.2020.6657/2019] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 11/20/2022]
Abstract
The intestinal spirochaetosis is defined as the presence of spirochetes on the colonic surface. We present the case of a 57-years-old male, with a history of 4-5 months of diffuse abdominal pain, watery diarrhoea due to this infection. The colonoscopy revealed a serpiginous ulcer in the cecum. The biopsy was positive for intestinal spirochaetosis. It is rare infection, more common among immunocompromised patients and HIV, with a faecal-oral transmission. Most cases are incidental findings in the endoscopic screening. In symptomatic patients the watery diarrhoea and non-specific abdominal pain are the most common symptoms. The macroscopic appearance on the colonoscopy is often normal or non-specific lesions can be identified. The diagnosis is based on the biopsy with haematoxylin and eosin and the confirmation can be made with a Warthin-Starry stain. In symptomatic patients the metronidazole is the preferred treatment option. Although it is rare infection, clinicians should be aware of it in patients with common gastrointestinal symptoms.
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19
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Cheema A, Jejelowo O, Syed M, Ramsakal A, Greene JN. Intestinal Spirochetes in Cancer Patients. INFECTIOUS DISEASES IN CLINICAL PRACTICE 2020. [DOI: 10.1097/ipc.0000000000000846] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/25/2022]
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20
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Ogata S, Shimizu K, Tominaga S, Nakanishi K. Histologic characteristics of human intestinal spirochetosis in operatively resected specimens. Virchows Arch 2020; 477:57-63. [PMID: 32144538 DOI: 10.1007/s00428-020-02785-w] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/02/2019] [Revised: 02/17/2020] [Accepted: 02/24/2020] [Indexed: 11/25/2022]
Abstract
Human intestinal spirochetosis (HIS), one of the zoonoses, is caused by colonization by Brachyspira species bacteria within the large intestine. Histologic diagnosis of HIS is usually established by finding "fringes" on the colonic surface epithelium in biopsy specimens. However, its histologic characteristics, especially beneath the colonic mucosa, have not been elucidated. The present study was designed to examine the histologic characteristics of HIS in operatively resected specimens. We reviewed operatively resected (colectomy or appendectomy) specimens obtained in six consecutive years at a single medical center. HIS was diagnosed histologically by finding "fringes". Immunohistochemical study using anti-Treponema pallidum antibody, which cross-reacts with Brachyspira, was additionally performed. A total of 848 (M:F = 477:371; median age, 59 years; 12-94 years) colectomy and/or appendectomy cases were examined, and the seven cases (0.8%) diagnosed as having HIS were all male (1.5% of male cases). Four HIS cases (0.8% of 508 colectomy cases (1.4% of 285 male-cases)) were colectomy cases with cancers, and the other three (0.9% of 340 appendectomy cases (1.6% of 192 male-cases)) were appendectomy cases for acute appendicitis. Our study revealed (1) a heterogeneous distribution of diagnostically important "fringes" within the large intestine, (2) an ileal presence of Brachyspira, (3) superficial location of HIS-related findings among anatomical wall layers, and (4) the presence of Brachyspira or its derivatives within macrophages in the lamina propria and immune apparatus (lymphoid follicles in superficial wall structures (lamina propria or submucosa) and lymph nodes). Investigation using operatively resected specimens might help elucidate the characteristics of HIS. Brachyspira may have immunogenicity in humans.
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Affiliation(s)
- Sho Ogata
- Department of Pathology and Laboratory Medicine, National Defense Medical College, Tokorozawa, Saitama, 359-8513, Japan. .,Department of Diagnostic Pathology, JCHO Saitama Medical Center, Saitama, Saitama, 330-0074, Japan.
| | - Ken Shimizu
- Department of Diagnostic Pathology, JCHO Saitama Medical Center, Saitama, Saitama, 330-0074, Japan
| | - Susumu Tominaga
- Department of Pathology and Laboratory Medicine, National Defense Medical College, Tokorozawa, Saitama, 359-8513, Japan
| | - Kuniaki Nakanishi
- Department of Pathology and Laboratory Medicine, National Defense Medical College, Tokorozawa, Saitama, 359-8513, Japan.,Department of Laboratory Medicine, National Defense Medical College Hospital, Tokorozawa, Saitama, 359-8513, Japan
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21
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Eguchi T, Inoue K, Horino T, Matsumoto T, Kamioka S, Nishida Y, Morimoto M, Morimoto N, Ichii O, Terada Y. Intestinal Spirochetosis Caused by Brachyspira pilosicoli in a Systemic Lupus Erythematosus Patient. J Clin Rheumatol 2019; 25:e142-e145. [PMID: 31764507 DOI: 10.1097/rhu.0000000000000829] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/26/2022]
Affiliation(s)
- Tomohiro Eguchi
- Department of Endocrinology Metabolism, and Nephrology Kochi Medical School Kochi University Kochi, Japan Department of Endocrinology Metabolism, and Nephrology Kochi Medical School Kochi University Kochi, Japan Department of Endocrinology Metabolism, and Nephrology Kochi Medical School Kochi University Kochi, Japan Department of Clinical Laboratory Kochi Medical School Kochi University Kochi, Japan Laboratory of Anatomy Department of Biomedical Sciences Graduate School of Veterinary Medicine Hokkaido University Sapporo, Japan Department of Endocrinology Metabolism, and Nephrology Kochi Medical School Kochi University Kochi, Japan
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22
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Chatani M, Kishita M, Inatomi O, Takahashi K, Sugimoto M, Sonoda A, Kawahara M, Bamba S, Kito K, Kushima R, Andoh A. Severe Colitis with Portal Venous Gas Caused by Brachyspira pilosicoli Infection. Intern Med 2019; 58:3409-3413. [PMID: 31787630 PMCID: PMC6928490 DOI: 10.2169/internalmedicine.3254-19] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 04/26/2019] [Accepted: 06/12/2019] [Indexed: 01/23/2023] Open
Abstract
We herein report a case of Brachyspira pilosicoli-caused severe colitis presenting with portal venous gas. A 75-year-old man was admitted because of a fever, severe abdominal pain and bloody diarrhea. He was negative for anti-HIV antibodies. He had been in close contact with a dog earlier. Abdominal computed tomography detected severe wall-thickening and fat-stranding of the entire colon accompanied by portal venous gas. A smear examination of his stool showed many Gram-negative spiral rods, suggesting intestinal spirochetosis. A polymerase chain reaction assay using stool samples detected an amplified band specific for B. pilosicoli. He responded well to antimicrobial agents including metronidazole.
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Affiliation(s)
- Motoharu Chatani
- Department of Medicine, Shiga University of Medical Science, Japan
| | - Megumi Kishita
- Division of Clinical Laboratory Medicine, Shiga University of Medical Science, Japan
| | - Osamu Inatomi
- Department of Medicine, Shiga University of Medical Science, Japan
| | | | | | - Ayano Sonoda
- Department of Medicine, Shiga University of Medical Science, Japan
| | | | - Shigeki Bamba
- Division of Nutrition, Shiga University of Medical Science, Japan
| | - Katsuyuki Kito
- Department of Medicine, Shiga University of Medical Science, Japan
| | - Ryoji Kushima
- Division of Clinical Laboratory Medicine, Shiga University of Medical Science, Japan
| | - Akira Andoh
- Department of Medicine, Shiga University of Medical Science, Japan
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23
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Severe Human Intestinal Spirochetosis: An Unusual Cause of Diffuse Colonic Ulcerations in a Patient Living with HIV. Case Rep Gastrointest Med 2019; 2019:1504079. [PMID: 31737379 PMCID: PMC6815633 DOI: 10.1155/2019/1504079] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/02/2019] [Revised: 07/14/2019] [Accepted: 07/22/2019] [Indexed: 11/18/2022] Open
Abstract
We describe a case of a homosexual male with human immunodeficiency virus (HIV) and CD4 count of 246 presenting with acute severe bloody diarrhea. Infectious work up was negative, and colonoscopy revealed severe diffuse colonic ulcerations. Histopathologic analysis and Treponemal pallidum immunostaining confirmed the diagnosis of intestinal spirochetosis. There was no evidence of co-infection with other pathogens. His symptoms completely resolved after a 14-day course of metronidazole. This case is notable as colonic ulceration of any severity in patients living with HIV is rarely identified with intestinal spirochetosis. Hence, it should be considered in the differential diagnosis of colonic ulcerations.
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24
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Tornero C, Frutos JM, Orta N, Lopez C, Castello I. Two Birds, One Stone: Two Unusual Causes of Diarrhea in a Nonimmunocompromised Human Immunodeficiency Virus-Infected Patient. Clin Infect Dis 2019; 67:1775-1776. [PMID: 30423036 DOI: 10.1093/cid/ciy282] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/13/2022] Open
Affiliation(s)
- Carlos Tornero
- Department of Internal Medicine, Hospital Francesc De Borja Gandia, Valencia, Spain
| | - Jose Maria Frutos
- Department of Internal Medicine, Hospital Francesc De Borja Gandia, Valencia, Spain
| | - Nieves Orta
- Department of Microbiology, Hospital Francesc De Borja Gandia, Valencia, Spain
| | - Cecilia Lopez
- Department of Pathology, Hospital Francesc De Borja Gandia, Valencia, Spain
| | - Inmaculada Castello
- Department of Digestive Diseases, Hospital Francesc De Borja Gandia, Valencia, Spain
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25
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Lemmens R, Devreker T, Hauser B, Degreef E, Goossens A, Vandenplas Y. Intestinal Spirochetosis: A Case Series and Review of the Literature. Pediatr Gastroenterol Hepatol Nutr 2019; 22:193-200. [PMID: 30899696 PMCID: PMC6416382 DOI: 10.5223/pghn.2019.22.2.193] [Citation(s) in RCA: 13] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/08/2018] [Revised: 04/17/2018] [Accepted: 05/16/2018] [Indexed: 11/21/2022] Open
Abstract
A clinical suspicion of intestinal spirochetosis is required when patients have long lasting complaints of abdominal pain, diarrhea, rectal bleeding, weight loss, and nausea. An endoscopy with biopsies needs to be performed to confirm the diagnosis of intestinal spirochetosis. The diagnosis of intestinal spirochetosis is based on histological appearance. Intestinal spirochetosis can also be associated with other intestinal infections and juvenile polyps (JPs). JPs seem to be more frequent in patients with intestinal spirochetosis than in patients without intestinal spirochetosis. Intestinal spirochetosis in children should be treated with antibiotics. Metronidazole is the preferred option. In this article, we describe 4 cases of intestinal spirochetosis in a pediatric population and provide a review of the literature over the last 20 years. Intestinal spirochetosis is a rare infection that can cause a variety of severe symptom. It is diagnosed based on histological appearance.
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Affiliation(s)
- Roel Lemmens
- Kidz Health Castle, UZ Brussel, Vrije Universiteit Brussel, Brussels, Belgium.,Department of Pathology, UZ Brussel, Vrije Universiteit Brussel, Brussels, Belgium
| | - Thierry Devreker
- Kidz Health Castle, UZ Brussel, Vrije Universiteit Brussel, Brussels, Belgium
| | - Bruno Hauser
- Kidz Health Castle, UZ Brussel, Vrije Universiteit Brussel, Brussels, Belgium
| | - Elisabeth Degreef
- Kidz Health Castle, UZ Brussel, Vrije Universiteit Brussel, Brussels, Belgium
| | - Annieta Goossens
- Department of Pathology, UZ Brussel, Vrije Universiteit Brussel, Brussels, Belgium
| | - Yvan Vandenplas
- Kidz Health Castle, UZ Brussel, Vrije Universiteit Brussel, Brussels, Belgium
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26
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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.
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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
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27
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Abstract
The alimentary tract serves as host to a large number of diseases. In the non-neoplastic group of disorders, conventional histochemistry continues to play an important diagnostic role. It is particularly important in recognizing specific infectious diseases, such as Helicobacter gastritis, Whipple disease, intestinal tuberculosis and other forms of mycobacteriosis, malakoplakia, intestinal spirochetosis, fungal enteritides, amebiasis, cryptosporidiosis, isosporiasis, and microsporidiosis. Those conditions and their histochemical properties are discussed in this review, along with the use of histochemistry in the characterization of structural gastrointestinal disorders. The latter include mucosal metaplasias, amyloidosis, glycogenic acanthosis of the esophagus, lymphocytic-collagenous colitis, gastric neuroendocrine hyperplasia, and pill gastritis.
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Affiliation(s)
- Patrick L Fitzgibbons
- Department of Pathology, St. Jude Medical Center, Fullerton, CA 92835, United States.
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28
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Green KR, Harris C, Shuja A, Malespin M, De Melo SW. Intestinal Spirochetosis: An Obscure Cause of Lower Gastrointestinal Bleeding. Cureus 2018; 10:e2970. [PMID: 30221098 PMCID: PMC6136888 DOI: 10.7759/cureus.2970] [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] [Indexed: 01/03/2023] Open
Abstract
Adherence of spirochetes to the apical membrane of the colonic epithelium has been well-described in the literature, but the exact pathogenesis leading to symptomatic clinical manifestations is poorly understood. Most cases are found incidentally on the pathological evaluation of colonic biopsies taken during diagnostic or therapeutic colonoscopies. However, whether the colonization of the intestinal mucosa can be attributed to clinical symptoms is a matter of debate. Here, we present a case of intermittent hematochezia attributed to the overwhelming invasion of the colonic mucosa by intestinal spirochetes.
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Affiliation(s)
- Kevin R Green
- Internal Medicine, University of Florida College of Medicine-Jacksonville, Jacksonville, USA
| | - Ciel Harris
- Internal Medicine, University of Florida College of Medicine-Jacksonville, Jacksonville, USA
| | - Asim Shuja
- Division of Gastroenterology, University of Florida College of Medicine-Jacksonville, Jacksonville, USA
| | - Miguel Malespin
- Division of Gastroenterology, University of Florida College of Medicine-Jacksonville, Jacksonville, USA
| | - Silvio W De Melo
- Division of Gastroenterology, Oregon Health and Science University, Portland, USA
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29
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Graham RP, Naini BV, Shah SS, Arnold CA, Kannangai R, Torbenson MS, Lam-Himlin DM. Treponema pallidum Immunohistochemistry is positive in human intestinal Spirochetosis. Diagn Pathol 2018; 13:7. [PMID: 29378606 PMCID: PMC6389163 DOI: 10.1186/s13000-017-0676-6] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/29/2017] [Accepted: 12/08/2017] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND Human intestinal spirochetosis (IS) has been recognized for decades, but whether it represents commensalism or a pathogenic process remains controversial. IS is diagnosed on routine stains with confirmation by silver stains but these stains are labor intensive and slow to read. We evaluated the Treponema pallidum immunostain as a diagnostic adjunct for IS. METHODS We retrieved biopsies from 33 patients with IS for this study. Each case was tested by Warthin-Starry (WS) and T. pallidum immunohistochemistry (IHC). Species specific genotyping was performed in 3 cases. RESULTS Patients with IS ranged from 22 to 82 years without gender predilection. IS involved normal (n = 15), and inflamed (n = 5) mucosa and colonic polyps (n = 13). Warthin-Starry and T. pallidum IHC were positive in all cases including both species of Brachyspira. Six (18%) symptomatic patients were treated for IS, and experienced resolution. In patients diagnosed with incidental IS on cancer screening (n = 5), follow up biopsies, without therapy, were negative for IS. T. pallidum IHC required 75 min less hands-on time than WS for performance and was faster to interpret. CONCLUSIONS T. pallidum IHC can be used to confirm the diagnosis of IS and is easier to perform and faster to interpret than WS.
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Affiliation(s)
- Rondell P. Graham
- Department of Laboratory Medicine and Pathology, Division of Anatomic Pathology, Mayo Clinic, 200 First Street SW, Rochester, MN 55905 USA
| | - Bita V. Naini
- Department of Pathology and Laboratory Medicine, David Geffen School of Medicine, University of California Los Angeles, 10833 Le Conte Ave. Suite 27-061C7 CHS, Los Angeles, CA 90095 USA
| | - Sejal S. Shah
- Department of Laboratory Medicine and Pathology, Division of Anatomic Pathology, Mayo Clinic, 200 First Street SW, Rochester, MN 55905 USA
| | - Christina A. Arnold
- Department of Pathology, The Ohio State University Wexner Medical Center, 410 West 10th Avenue, Columbus, OH 43210 USA
| | - Rajesh Kannangai
- Department of Clinical Virology, Christian Medical College, Vellore, 632004 India
| | - Michael S. Torbenson
- Department of Laboratory Medicine and Pathology, Division of Anatomic Pathology, Mayo Clinic, 200 First Street SW, Rochester, MN 55905 USA
| | - Dora M. Lam-Himlin
- Department of Laboratory Medicine and Pathology, Division of Anatomic Pathology, Mayo Clinic, Scottsdale, AZ 85259 USA
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Nishii S, Higashiyama M, Ogata S, Komoto S, Ito S, Mizoguchi A, Terada H, Furuhashi H, Takajo T, Shirakabe K, Watanabe C, Tomita K, Nagao S, Miura S, Hokari R. Human intestinal spirochetosis mimicking ulcerative colitis. Clin J Gastroenterol 2017; 11:145-149. [PMID: 29204849 DOI: 10.1007/s12328-017-0807-3] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/11/2017] [Accepted: 11/25/2017] [Indexed: 10/18/2022]
Abstract
Human intestinal spirochetosis (HIS) is a colorectal infection caused by the Brachyspira species of intestinal spirochetes, whose pathogenicity in humans remains unclear owing to the lack of or mild symptoms. We monitored the 5-year clinical course of a woman diagnosed with HIS in whom ulcerative colitis (UC) had been suspected. Following a positive fecal occult blood test, she underwent a colonoscopic examination at a local clinic where she was diagnosed with "right-sided" UC concomitant with incidentally detected HIS, and was referred to our hospital. Colonoscopic, histopathological, and cytological examination revealed localized erosive colitis in the ascending and the right transverse colon concomitant with HIS resembling skip lesions of UC. Initially, we chose the wait-and-watch approach; however, she gradually developed bloody diarrhea. Metronidazole improved her abdominal symptoms, as well as her colonoscopic and histopathological findings, suggesting that HIS was responsible for her colorectal inflammation. This case reveals (1) a possible pro-inflammatory role of HIS, (2) difficulties in diagnosing HIS in chronic proctocolitis, and (3) a possible inclusion of some HIS cases in "UC". HIS could mimic UC and might be included in differential diagnoses for UC. Antibiotic administration is necessary following the detection of HIS, particularly in patients demonstrating an atypical presentation of UC.
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Affiliation(s)
- Shin Nishii
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Masaaki Higashiyama
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan.
| | - Sho Ogata
- Department of Laboratory Medicine, National Defense Medical College Hospital, Tokorozawa, Japan
| | - Shunsuke Komoto
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Suguru Ito
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Akinori Mizoguchi
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Hisato Terada
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Hirotaka Furuhashi
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Takeshi Takajo
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Kazuhiko Shirakabe
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Chikako Watanabe
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Kengo Tomita
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Shigeaki Nagao
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Soichiro Miura
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
| | - Ryota Hokari
- Department of Internal Medicine, National Defense Medical College, 3-2 Namiki, Tokorozawa, Saitama, 359-8513, Japan
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31
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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.
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Gan J, Bryant C, Arul D, Parmar C. Intestinal spirochaetosis mimicking acute appendicitis with review of the literature. BMJ Case Rep 2017; 2017:bcr-2017-221574. [PMID: 29103010 DOI: 10.1136/bcr-2017-221574] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/03/2022] Open
Abstract
Human intestinal spirochaetosis is a well-established micro-organism existing in the colon. It is less commonly seen in the appendix, and rarely presents as acute appendicitis. We present a case of a man presenting with symptoms consistent with acute appendicitis. The literature on spirochaetosis presenting as acute appendicitis is also reviewed.
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Affiliation(s)
- Jason Gan
- Department of Surgery, Whittington Hospital, London, UK
| | | | - Dhili Arul
- Department of Surgery, Whittington Hospital, London, UK
| | - Chetan Parmar
- Department of Surgery, Whittington Hospital, London, UK
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Abstract
Bacteremia and sepsis are conditions associated with high mortality and are of great impact to health care operations. Among the top causes of mortality in the United States, these conditions cause over 600 fatalities each day. Empiric, broad-spectrum treatment is a common but often a costly approach that may fail to effectively target the correct microbe, may inadvertently harm patients via antimicrobial toxicity or downstream antimicrobial resistance. To meet the diagnostic challenges of bacteremia and sepsis, laboratories must understand the complexity of diagnosing and treating septic patients, in order to focus on creating algorithms that can help direct a more targeted approach to antimicrobial therapy and synergize with existing clinical practices defined in new Surviving Sepsis Guidelines. Significant advances have been made in improving blood culture media; as yet no molecular or antigen-based method has proven superior for the detection of bacteremia in terms of limit of detection. Several methods for rapid molecular identification of pathogens from blood cultures bottles are available and many more are on the diagnostic horizon. Ultimately, early intervention by molecular detection of bacteria and fungi directly from whole blood could provide the most patient benefit and contribute to tailored antibiotic coverage of the patient early on in the course of the disease. Although blood cultures remain as the best means of diagnosing bacteremia and candidemia, complementary testing with antigen tests, microbiologic investigations from other body sites, and histopathology can often aid in the diagnosis of disseminated disease, and application of emerging nucleic acid test methods and other new technology may greatly impact our ability to bacteremic and septic patients, particularly those who are immunocompromised.
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Rojas P, Petrich A, Schulze J, Wiessner A, Loddenkemper C, Epple HJ, Sterlacci W, Vieth M, Kikhney J, Moter A. Distribution and phylogeny of Brachyspira spp. in human intestinal spirochetosis revealed by FISH and 16S rRNA-gene analysis. Anaerobe 2017; 47:25-32. [PMID: 28300642 DOI: 10.1016/j.anaerobe.2017.03.012] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 12/13/2016] [Revised: 03/09/2017] [Accepted: 03/10/2017] [Indexed: 01/01/2023]
Abstract
During six years as German National Consultant Laboratory for Spirochetes we investigated 149 intestinal biopsies from 91 patients, which were histopathologically diagnosed with human intestinal spirochetosis (HIS), using fluorescence in situ hybridization (FISH) combined with 16S rRNA gene PCR and sequencing. Aim of this study was to complement histopathological findings with FISH and PCR for definite diagnosis and species identification of the causative pathogens. HIS is characterized by colonization of the colonic mucosa of the human distal intestinal tract by Brachyspira spp. Microbiological diagnosis of HIS is not performed, because of the fastidious nature and slow growth of Brachyspira spp. in culture. In clinical practice, diagnosis of HIS relies solely on histopathology without differentiation of the spirochetes. We used a previously described FISH probe to detect and identify Brachyspira spp. in histological gut biopsies. FISH allowed rapid visualization and identification of Brachyspira spp. in 77 patients. In most cases, the bright FISH signal already allowed rapid localization of Brachyspira spp. at 400× magnification. By sequencing, 53 cases could be assigned to the B. aalborgi lineage including "B. ibaraki" and "B. hominis", and 23 cases to B. pilosicoli. One case showed mixed colonization. The cases reported here reaffirm all major HIS Brachyspira spp. clusters already described. However, the phylogenetic diversity seems to be even greater than previously reported. In 14 cases, we could not confirm HIS by either FISH or PCR, but found colonization of the epithelium by rods and cocci, indicating misdiagnosis by histopathology. FISH in combination with molecular identification by 16S rRNA gene sequencing has proved to be a valuable addition to histopathology. It provides definite diagnosis of HIS and allows insights into phylogeny and distribution of Brachyspira spp. HIS should be considered as a differential diagnosis in diarrhea of unknown origin, particularly in patients from risk groups (e.g. patients with colonic adenomas, inflammatory polyps, inflammatory bowel disease or HIV infection and in men who have sex with men).
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Affiliation(s)
- Pablo Rojas
- Institute for Microbiology and Hygiene, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Annett Petrich
- Institute for Microbiology and Hygiene, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | - Julia Schulze
- Biofilmcenter, Deutsches Herzzentrum Berlin, Berlin, Germany
| | | | | | - Hans-Jörg Epple
- Medical Clinic I, Gastroenterology, Rheumatology, Infectiology, Charité - Universitätsmedizin Berlin, Berlin, Germany
| | | | - Michael Vieth
- Institute of Pathology, Klinikum Bayreuth GmbH, Bayreuth, Germany
| | - Judith Kikhney
- Institute for Microbiology and Hygiene, Charité - Universitätsmedizin Berlin, Berlin, Germany; Biofilmcenter, Deutsches Herzzentrum Berlin, Berlin, Germany
| | - Annette Moter
- Biofilmcenter, Deutsches Herzzentrum Berlin, Berlin, Germany; Former German Consultant Laboratory for Treponema Identification, Charité - Universitätsmedizin Berlin, Berlin, Germany.
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Ogata S, Shimizu K, Tominaga S, Nakanishi K. Immunohistochemical study of mucins in human intestinal spirochetosis. Hum Pathol 2017; 62:126-133. [PMID: 28188751 DOI: 10.1016/j.humpath.2017.01.013] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 12/02/2016] [Revised: 01/11/2017] [Accepted: 01/26/2017] [Indexed: 01/27/2023]
Abstract
Most patients with human intestinal spirochetosis (HIS; a colorectal bacterial infection caused by Brachyspira species) seem asymptomatic, and its pathogenicity remains unclear. Recently, alterations in mucin expression were reported in animal Brachyspira infection. The present question was "Is mucin expression altered in HIS?" Using antibodies for MUCs 1, 2, 4, 5AC, and 6, we immunohistochemically compared 215 specimens from 83 histology-confirmed HIS cases with 106 specimens from 26 non-HIS cases. Positive staining (which included even focal positive staining) was rated "high (+)" or "low (+)." Results were analyzed for 4 categories of lesions, and associations between MUC expression and spirochetal presence were also analyzed. In the "specimens without polyps or adenocarcinoma" category, high (+) MUC2 positivity was more frequent in HIS than in control. In the hyperplasia/serrated polyp category, in HIS (versus control), the MUC5AC positivity rate was lower, whereas high (+) MUC4 positivity was more frequent. In the conventional adenoma category, in HIS (versus control), the MUC1 positivity rate was lower, whereas both high (+) MUC2 positivity and high (+) MUC5AC positivity were less frequent. In the adenocarcinoma category, high (+) MUC2 positivity was more frequent in HIS than in control. Among the above mucins, only MUC1 positivity was significantly associated with an absence of the so-called fringe formation, an absence of spiral organisms within mucus, and an absence of strong immunopositive materials within the epithelial layer and within the subepithelial layer. The results suggest that Brachyspira infection or a related change in the microbiome may alter the large intestine mucin expression profile in humans.
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Affiliation(s)
- Sho Ogata
- Department of Laboratory Medicine, National Defense Medical College Hospital, Tokorozawa, Saitama 359-8513, Japan; Department of Diagnostic Pathology, JCHO Saitama Medical Center, Saitama, Saitama 330-0074, Japan.
| | - Ken Shimizu
- Department of Diagnostic Pathology, JCHO Saitama Medical Center, Saitama, Saitama 330-0074, Japan
| | - Susumu Tominaga
- Department of Pathology and Laboratory Medicine, National Defense Medical College, Tokorozawa, Saitama 359-8513, Japan
| | - Kuniaki Nakanishi
- Department of Laboratory Medicine, National Defense Medical College Hospital, Tokorozawa, Saitama 359-8513, Japan; Department of Pathology and Laboratory Medicine, National Defense Medical College, Tokorozawa, Saitama 359-8513, Japan
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Ruiz SJ, Procop GW. Cross-reactivity of Anti-Treponema Immunohistochemistry With Non-Treponema Spirochetes: A Simple Call for Caution. Arch Pathol Lab Med 2016; 140:1021-2. [DOI: 10.5858/arpa.2016-0004-le] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.9] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/06/2022]
Affiliation(s)
- Sory J. Ruiz
- Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, Cleveland, Ohio
| | - Gary W. Procop
- Robert J. Tomsich Pathology and Laboratory Medicine Institute, Cleveland Clinic, Cleveland, Ohio
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Ogata S, Shimizu K, Oda T, Tominaga S, Nakanishi K. Immunohistochemical detection of human intestinal spirochetosis. Hum Pathol 2016; 58:128-133. [PMID: 27581381 DOI: 10.1016/j.humpath.2016.07.032] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/21/2016] [Revised: 07/28/2016] [Accepted: 07/29/2016] [Indexed: 11/17/2022]
Abstract
Human intestinal spirochetosis (HIS) is a colorectal infection by Brachyspira species of spiral bacteria. Immunohistochemical cross-reaction to an antibody for Treponema pallidum aids its histologic diagnosis. This study's aim was to analyze the immunohistochemical characteristics of HIS. In this analysis, on 223 specimens from 83 HIS cases, we focused on so-called fringe formation (a histologic hallmark of HIS), spiral organisms within mucus or within crypts, and strong immunopositive materials in the mucosa, together with their location and the types of lesions. Fringe formation was found in 81.6% of all specimens and spiral organisms within mucus or within crypts in 97.3% and 57.0%, respectively. Strong immunopositive materials were observed in the surface epithelial layer in 87.9%, in the subepithelial layer in 94.6%, and in deeper mucosa in 2.2% of all specimens. The positive rates in conventional adenomas (24.0%, n = 146) and hyperplastic nodules (100%, n = 17) were each different from that found in inflammation (70.8%, n = 24), and spiral organisms were seen more frequently in the right-side large intestine than in the left (within mucus, 100%, n = 104 versus 95.0%, n = 119; within crypts, 65.4%, n = 104 versus 49.6%, n = 119). Thus, immunohistochemistry was effective not only in supporting the diagnosis of HIS but also in highlighting spiral organisms within mucus or crypts that were invisible in routine histology. Possibly, these spiral organisms may spread throughout the entire large intestine, although there is a potential problem with antibody specificity.
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Affiliation(s)
- Sho Ogata
- Department of Laboratory Medicine, National Defense Medical College Hospital, Tokorozawa, Saitama, 359-8513, Japan; Department of Diagnostic Pathology, JCHO Saitama Medical Center, Saitama, 330-0074, Japan.
| | - Ken Shimizu
- Department of Diagnostic Pathology, JCHO Saitama Medical Center, Saitama, 330-0074, Japan
| | - Tomohiro Oda
- Department of Laboratory Medicine, National Defense Medical College Hospital, Tokorozawa, Saitama, 359-8513, Japan
| | - Susumu Tominaga
- Department of Pathology and Laboratory Medicine, National Defense Medical College, Tokorozawa, Saitama, 359-8513, Japan
| | - Kuniaki Nakanishi
- Department of Laboratory Medicine, National Defense Medical College Hospital, Tokorozawa, Saitama, 359-8513, Japan; Department of Pathology and Laboratory Medicine, National Defense Medical College, Tokorozawa, Saitama, 359-8513, Japan
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Ngwa T, Peng JL, Choi E, Tayarachakul S, Liangpunsakul S. Colonic Spirochetosis in a 60-Year-Old Immunocompetent Patient: Case Report and Review. J Investig Med High Impact Case Rep 2016; 4:2324709616662671. [PMID: 27570780 PMCID: PMC4984321 DOI: 10.1177/2324709616662671] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/13/2016] [Accepted: 07/02/2016] [Indexed: 01/03/2023] Open
Abstract
Spirochetes, a genetically and morphologically distinct group of bacteria, are thin, spiral-shaped, and highly motile. They are known causes of several human diseases such as syphilis, Lyme disease, relapsing fever, and leptospirosis. We report a case of colonic spirochetosis in a healthy patient presenting for surveillance colonoscopy. The diagnosis of intestinal spirochetosis was made accidentally during the histological examination of colonic polyps, which were removed during colonoscopy. We also performed an extensive review on intestinal spirochetosis with a focus on clinical presentation and outcomes of reported cases from the past two decades.
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Affiliation(s)
- Taiwo Ngwa
- Division of Gastroenterology and Hepatology, Department of Medicine, Indiana University School of Medicine, Indianapolis, IN, USA
| | - Jennifer L Peng
- Division of Gastroenterology and Hepatology, Department of Medicine, Indiana University School of Medicine, Indianapolis, IN, USA
| | - Euna Choi
- Department of Pathology and Laboratory Medicine, Indiana University School of Medicine, Indianapolis, IN, USA
| | - Sucharat Tayarachakul
- Division of Gastroenterology and Hepatology, Department of Medicine, Indiana University School of Medicine, Indianapolis, IN, USA
| | - Suthat Liangpunsakul
- Division of Gastroenterology and Hepatology, Department of Medicine, Indiana University School of Medicine, Indianapolis, IN, USA; Roudebush Veterans Administration Medical Center, Indianapolis, IN, USA
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Chebli JMF, de Abreu NC, Chebli LA, Reboredo MM, Pinheiro HS. Intestinal spirochetosis: an unusual cause of IBD flare-up during anti-TNF therapy. Int J Colorectal Dis 2016; 31:745-746. [PMID: 25982466 DOI: 10.1007/s00384-015-2240-8] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Accepted: 05/08/2015] [Indexed: 02/04/2023]
Affiliation(s)
- Julio Maria Fonseca Chebli
- Inflammatory Bowel Diseases Center, Division of Gastroenterology, University Hospital of the Federal University of Juiz de Fora, Av. Eugênio do Nascimento - s/n°, Dom Bosco, Juiz de Fora, Minas Gerais, 36038-330, Brazil.
- Department of Medicine, School of Medicine, Federal University of Juiz de Fora, Av. Eugênio do Nascimento - s/n°, Dom Bosco, Juiz de Fora, Minas Gerais, 36038-330, Brazil.
- , Rua Maria José Leal, 296, Juiz de Fora, MG, 36036-247, Brazil.
| | - Nathália Chebli de Abreu
- Inflammatory Bowel Diseases Center, Division of Gastroenterology, University Hospital of the Federal University of Juiz de Fora, Av. Eugênio do Nascimento - s/n°, Dom Bosco, Juiz de Fora, Minas Gerais, 36038-330, Brazil
| | - Liliana Andrade Chebli
- Inflammatory Bowel Diseases Center, Division of Gastroenterology, University Hospital of the Federal University of Juiz de Fora, Av. Eugênio do Nascimento - s/n°, Dom Bosco, Juiz de Fora, Minas Gerais, 36038-330, Brazil
- Department of Medicine, School of Medicine, Federal University of Juiz de Fora, Av. Eugênio do Nascimento - s/n°, Dom Bosco, Juiz de Fora, Minas Gerais, 36038-330, Brazil
| | - Maycon Moura Reboredo
- Department of Medicine, School of Medicine, Federal University of Juiz de Fora, Av. Eugênio do Nascimento - s/n°, Dom Bosco, Juiz de Fora, Minas Gerais, 36038-330, Brazil
| | - Helady Sanders Pinheiro
- Department of Medicine, School of Medicine, Federal University of Juiz de Fora, Av. Eugênio do Nascimento - s/n°, Dom Bosco, Juiz de Fora, Minas Gerais, 36038-330, Brazil
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40
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Answer to December 2015 Photo Quiz. J Clin Microbiol 2015. [DOI: 10.1128/jcm.00015-14] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/20/2022] Open
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41
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[A colonic mucosae with brushing border]. Ann Pathol 2015; 35:536-8. [PMID: 26586089 DOI: 10.1016/j.annpat.2015.05.020] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/04/2014] [Revised: 05/18/2015] [Accepted: 05/31/2015] [Indexed: 11/23/2022]
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Warren S, Freedman A, Lomax N, Christian A. Intestinal spirochaetosis causing chronic diarrhoea in a patient with HIV infection. Int J STD AIDS 2015; 28:616-618. [PMID: 26494705 DOI: 10.1177/0956462415614724] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022]
Abstract
A 36-year-old Caucasian homosexual man was found to have HIV infection on routine screening. He had an eight-year history of chronic diarrhoea, which pre-dated the HIV diagnosis and did not improve after the introduction of combination antiretroviral therapy. After referral to the Gastroenterology department, he underwent fibreoptic colonoscopy. Colonic biopsies revealed the presence of intestinal spirochaetosis. He received a two-week course of metronidazole, which led to complete resolution of his diarrhoea. Intestinal spirochaetosis should be considered in the differential diagnosis of patients with HIV infection and chronic diarrhoea without other apparent cause.
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Affiliation(s)
- Sian Warren
- 1 Department of Integrated Sexual Health, Cardiff Royal Infirmary, Cardiff, UK
| | - Andrew Freedman
- 2 Infectious Diseases Unit, University Hospital of Wales, Cardiff, UK
| | - Nicola Lomax
- 1 Department of Integrated Sexual Health, Cardiff Royal Infirmary, Cardiff, UK
| | - Adam Christian
- 3 Department of Histopathology, University Hospital of Wales, Cardiff, UK
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Human intestinal spirochetosis: right-side preference in the large intestine. Ann Diagn Pathol 2015; 19:414-7. [PMID: 26597024 DOI: 10.1016/j.anndiagpath.2015.10.004] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 10/08/2015] [Accepted: 10/12/2015] [Indexed: 11/24/2022]
Abstract
Human intestinal spirochetosis (HIS) is a colorectal bacterial infection, and its clinicopathologic features remain unclear. The aim of this study was to examine its characteristics. We histologically reviewed paraffin-embedded section slides made in 2001, 2006, and 2011 at a single institution in Japan. Cases histologically exhibiting a distinct fringe formation were considered to have HIS. Information was obtained from pathology request forms. We identified 85 HIS cases among 4930 patients (7 cases [0.5%) in 2001, 29 [1.7%] in 2006, and 49 [2.8%] in 2011]. Gastrointestinal symptoms were observed in 7.1% of HIS cases. Human intestinal spirochetosis was more frequent in the right-side large intestine than in the left side. Among 224 samples from HIS cases, conventional (tubular, tubulovillous, and villous) adenomas were found in 148 samples. These adenomas were more frequent in the right side than in the left side, although neither their size nor morphology differed between the sides. Histopathologic evaluation suggested a year-upon-year increasing prevalence of HIS in Japan. A small number exhibited gastrointestinal symptoms. Both histologic sign of HIS and conventional adenomas were more frequent in the right-side large intestine. Therefore, a right-side preference may be a characteristic of HIS.
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Walker MM, Talley NJ, Inganäs L, Engstrand L, Jones MP, Nyhlin H, Agréus L, Kjellstrom L, Öst Å, Andreasson A. Colonic spirochetosis is associated with colonic eosinophilia and irritable bowel syndrome in a general population in Sweden. Hum Pathol 2015; 46:277-83. [DOI: 10.1016/j.humpath.2014.10.026] [Citation(s) in RCA: 37] [Impact Index Per Article: 3.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/24/2014] [Revised: 10/26/2014] [Accepted: 10/27/2014] [Indexed: 12/21/2022]
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Tateishi Y, Takahashi M, Horiguchi SI, Funata N, Koizumi K, Okudela K, Hishima T, Ohashi K. Clinicopathologic study of intestinal spirochetosis in Japan with special reference to human immunodeficiency virus infection status and species types: analysis of 5265 consecutive colorectal biopsies. BMC Infect Dis 2015; 15:13. [PMID: 25582884 PMCID: PMC4300994 DOI: 10.1186/s12879-014-0736-4] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/12/2014] [Accepted: 12/22/2014] [Indexed: 01/03/2023] Open
Abstract
Background Previous studies reported that the incidence of intestinal spirochetosis was high in homosexual men, especially those with Human Immunodeficiency Virus infection. The aim of the present study was to clarify the clinicopathological features of intestinal spirochetosis in Japan with special reference to Human Immunodeficiency Virus infection status and species types. Methods A pathology database search for intestinal spirochetosis was performed at Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital between January 2008 and October 2011, and included 5265 consecutive colorectal biopsies from 4254 patients. After patient identification, a retrospective review of endoscopic records and clinical information was performed. All pathology slides were reviewed by two pathologists. The length of the spirochetes was measured using a digital microscope. Causative species were identified by polymerase chain reaction. Results Intestinal spirochetosis was diagnosed in 3 out of 55 Human Immunodeficiency Virus-positive patients (5.5%). The mean length of intestinal spirochetes was 8.5 μm (range 7–11). Brachyspira pilosicoli was detected by polymerase chain reaction in all 3 patients. Intestinal spirochetosis was also diagnosed in 73 out of 4199 Human Immunodeficiency Virus-negative patients (1.7%). The mean length of intestinal spirochetes was 3.5 μm (range 2–8). The species of intestinal spirochetosis was identified by polymerase chain reaction in 31 Human Immunodeficiency Virus-negative patients. Brachyspira aalborgi was detected in 24 cases (78%) and Brachyspira pilosicoli in 6 cases (19%). Both Brachyspira aalborgi and Brachyspira pilosicoli were detected in only one Human Immunodeficiency Virus-negative patient (3%). The mean length of Brachyspira aalborgi was 3.8 μm, while that of Brachyspira pilosicoli was 5.5 μm. The length of Brachyspira pilosicoli was significantly longer than that of Brachyspira aalborgi (p < 0.01). The lengths of intestinal spirochetes were significantly longer in Human Immunodeficiency Virus-positive patients than in Human Immunodeficiency Virus-negative patients (p < 0.05). Conclusions The incidence of intestinal spirochetosis was slightly higher in Human Immunodeficiency Virus-positive patients than in Human Immunodeficiency Virus-negative patients. However, no relationship was found between the Human Immunodeficiency Virus status and intestinal spirochetosis in Japan. Brachyspira pilosicoli infection may be more common in Human Immunodeficiency Virus-positive patients with intestinal spirochetosis than in Human Immunodeficiency Virus-negative patients with intestinal spirochetosis.
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Affiliation(s)
- Yoko Tateishi
- Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan. .,Department of Pathology, Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital, Tokyo, Japan.
| | - Masae Takahashi
- Department of Pathology, Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital, Tokyo, Japan.
| | - Shin-ichiro Horiguchi
- Department of Pathology, Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital, Tokyo, Japan.
| | - Nobuaki Funata
- Department of Pathology, Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital, Tokyo, Japan.
| | - Koichi Koizumi
- Department of Internal Medicine, Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital, Tokyo, Japan.
| | - Koji Okudela
- Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan.
| | - Tsunekazu Hishima
- Department of Pathology, Tokyo Metropolitan Cancer and Infectious Disease Center Komagome Hospital, Tokyo, Japan.
| | - Kenichi Ohashi
- Department of Pathology, Yokohama City University Graduate School of Medicine, Yokohama, Japan.
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Iwamoto J, Ogata S, Honda A, Saito Y, Murakami M, Ikegami T, Adachi Y, Matsuzaki Y. Human intestinal spirochaetosis in two ulcerative colitis patients. Intern Med 2014; 53:2067-71. [PMID: 25224189 DOI: 10.2169/internalmedicine.53.2386] [Citation(s) in RCA: 12] [Impact Index Per Article: 1.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/06/2022] Open
Abstract
A histological examination of colonic biopsies of the longitudinal and irregularly-shaped ulcerative lesions of a 37-year-old man and 61-year-old man with ulcerative colitis showed so-called "fringe formation," a typical finding of Brachyspira infection. The antibody titer to Brachyspira aalborgi showed marked elevation in both cases, and the patients were each treated with 1,000 mg of metronidazole for 14 days. Colonoscopy performed after treatment showed an improvement in the ulcerative lesions in both patients. These results indicate the possibility that intestinal spirochaetosis infection should be considered as an infectious complication in patients with ulcerative colitis receiving long-term steroid therapy.
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Affiliation(s)
- Junichi Iwamoto
- Department of Gastroenterology, Tokyo Medical University Ibaraki Medical Center, Japan
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47
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Burks ML, Kundrotas L. Unusual colon biopsy. Gastroenterology 2013; 145:e10-1. [PMID: 23810345 DOI: 10.1053/j.gastro.2013.04.054] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/22/2013] [Revised: 04/22/2013] [Accepted: 04/29/2013] [Indexed: 12/02/2022]
Affiliation(s)
- Margaret L Burks
- University of Texas Health Science Center at San Antonio, San Antonio, Texas, USA
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48
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Abstract
Brachyspira species have been implicated as a potential cause of gastroenteritis in humans; this is, however, controversial. In 733 gastroenteritis cases and 464 controls, we found 29 samples positive for Brachyspira species (2.3% of cases and 2.6% of controls; P = 0.77). Brachyspira species were not associated with gastroenteritis in humans.
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49
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Chopra S, Wu MLC. Specimens from biopsies of colorectal polyps often harbor additional diagnoses. PATHOLOGY RESEARCH INTERNATIONAL 2013; 2013:570526. [PMID: 24455417 PMCID: PMC3886612 DOI: 10.1155/2013/570526] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 07/26/2013] [Accepted: 11/13/2013] [Indexed: 12/11/2022]
Abstract
Objectives. The utility of examining specimens from colorectal biopsies of polyps for nonneoplastic diseases is currently unknown. Our objectives were to characterize such additional diagnoses that could be rendered. Methods. We retrospectively and prospectively reviewed specimens from endoscopic biopsies of colorectal polyps obtained during routine screening or surveillance. Results. 17 of 168 specimens (10.1%) contained additional diagnoses, including schistosomiasis, eosinophilic colitis, intestinal spirochetosis, melanosis coli, and other entities. These findings were easily overlooked because they often affected mucosa that was spared by the polyps or were often evident only at high magnification. Schistosomiasis, eosinophilic colitis, and intestinal spirochetosis were clinically occult. Conclusions. Specimens from biopsies of colorectal polyps often harbor other diagnoses, in addition to polyps, and can be simultaneously screened for polyps and examined for nonneoplastic diseases. Detection of other diagnoses in addition to polyps requires awareness, examination at high magnification, and examination of areas spared by the polyps.
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Affiliation(s)
- Shefali Chopra
- Department of Pathology and Laboratory Medicine, University of California Irvine School of Medicine, Irvine, CA 92868, USA
| | - Mark Li-cheng Wu
- Department of Pathology and Laboratory Medicine, University of California Irvine School of Medicine, Irvine, CA 92868, USA
- *Mark Li-cheng Wu:
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Anthony NE, Blackwell J, Ahrens W, Lovell R, Scobey MW. Intestinal spirochetosis: an enigmatic disease. Dig Dis Sci 2013; 58:202-8. [PMID: 22851039 DOI: 10.1007/s10620-012-2305-2] [Citation(s) in RCA: 15] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/04/2012] [Accepted: 06/25/2012] [Indexed: 01/03/2023]
Abstract
INTRODUCTION Intestinal spirochetosis (IS) is a condition in which colonic and appendiceal epithelial cells are colonized by one of two anaerobic spirochetes, either the Brachyspira aalborgi or Brachyspira pilosicoli. There is much debate in the literature as to whether IS is a pathogen or a commensal inhabitant. A recent case of IS at our institution prompted a retrospective database search and review of the literature. METHODS A pathology database search for IS was performed at Carolinas Medical Center from 2003 through 2007. After patient identification, a retrospective review of the endoscopic record and the pathology report was performed. Pathology slides were reviewed for accuracy and special silver stains and/or immunostains were performed if needed. The following data were collected for each patient when available: age, gender, nationality, HIV status, and other co-morbid conditions when noted. We attempted to determine whether patients were treated for spirochetosis and if so, the treatment regimen used as well as the results. RESULTS The database search detected 29 patients with biopsies showing IS. Three patients were subsequently removed due to incorrect identification. A total of 26 patients with an average age of 45 years were reviewed. The most common symptoms were abdominal pain, diarrhea, and rectal bleeding. Most patients did not exhibit inflammatory changes despite the presence of spirochetosis. Pathologic examination revealed a relative increase in intra-epithelial lymphocytes in a subset of cases, a non-specific finding. Acute colitis or architectural distortion was not seen in any of the study cases. We were only able to obtain follow-up of two patients after treatment with metronidazole and both responded to therapy. CONCLUSIONS To date, our study is the largest case series that includes both endoscopic and pathologic descriptions and confirms the "bland" nature of the condition. In <20 % of our patients inflammation was present microscopically and it did not correlate well with endoscopic appearance. Symptoms reported by our patients were similar to those reported in previous studies, although our lack of endoscopic changes was different from one previous paper. There is no established standard of care for the treatment of IS and our study, reflects the enigmatic nature of IS as a disease process. In the absence of rigorous literature, physicians will need to use a logical and pragmatic approach to the evaluation and treatment of IS.
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Affiliation(s)
- Nicholas E Anthony
- Department of Internal Medicine, Carolinas Medical Center, 1000 Blythe Blvd., Charlotte, NC 28203, USA.
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