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xxii Contributors
Daniela Pugliese Dipartimento Universitario di Medicina e Chirurgia
Traslazionale, Università Cattolica del Sacro Cuore, Rome, Italy
UOC di Medicina Interna e Gastroenterologia; CEMAD – IBD UNIT; Dipartimento
di Scienze Mediche e Chirurgiche, Fondazione Policlinico Universitario
“A. Gemelli” IRCCS, Rome, Italy
J. M. Ramirez Surgery Department, Hospital Universitario Miguel Servet,
Zaragoza, Spain
Carlo Ratto Proctology Unit, Dept. of Medical and Surgical Sciences, Foundation
University Hospital “A. Gemelli”, IRCCS, Rome, Italy
Catholic University, Rome, Italy
Francisco Sergio P. Regadas Filho School of Medicine, Federal University of
Ceara, Fortaleza, Ceará, Brazil
Anne-Laure Rentien Service de Proctologie Médico-Chirurgicale, Groupe
hospitalier Paris Saint-Joseph, Institut Léopold Bellan, Paris, France
Charlene Sackitey Robin Phillips Fistula Research Unit, St Mark’s Hospital and
Academic Unit, London, United Kingdom
Aynur Safiyeva Central Customs Hospital, Baku, Azerbaijan
Kapil Sahnan Fistula Research Unit, St Mark’s Hospital and Academic Institute,
Harrow, UK
Department of Surgery and Cancer, Imperial College London, St Mary’s Hospital,
London, UK
R. Sanz-Baro Department of Surgery, University Hospital Fundación Jiménez
Díaz, Madrid, Spain
Surgery Department, Universidad Autonoma de Madrid, Madrid, Spain
Franco Scaldaferri Dipartimento di Medicina e chirurgia traslazionale, CEMAD –
UOC di Medicina Interna e Gastroenterologia – Fondazione Policlinico
“A. Gemelli” IRCCS, Rome, Italy
Università Cattolica del Sacro Cuore, Rome, Italy
Tommaso Schepis Dipartimento Universitario di Medicina e Chirurgia
Traslazionale, Università Cattolica del Sacro Cuore, Rome, Italy
M. E. I. Schipper Department of Pathology, St. Antonius Ziekenhuis, Nieuwegein,
The Netherlands
Francis Seow-Choen Seow-Choen Colorectal Surgery, Singapore, Singapore
Isaac Seow-En Department of Colorectal Surgery, Singapore General Hospital,
Singapore, Singapore
Contributors xxiii
Lucas Spindler Service de Proctologie Médico-Chirurgicale, Groupe hospitalier
Paris Saint-Joseph, Institut Léopold Bellan, Paris, France
Antonino Spinelli Division of Colon and Rectal Surgery, Humanitas Clinical and
Research Center IRCCS, Milan, Italy
Department of Biomedical Sciences, Humanitas University, Pieve Emanuele, Milan,
Italy
Scott R. Steele Department of Colorectal Surgery, Clevel and Clinic, Cleveland,
OH, USA
Alessandro Sturiale Proctology and Pelvic Floor Clinical Centre, Cisanello University Hospital, Pisa, Italy
Phil Tozer Robin Phillips Fistula Research Unit, St Mark’s Hospital and Academic
Institute, Harrow, UK
Natalia Uribe Colorectal Surgery Unit, Hospital Arnau de Vilanova, Valencia,
Spain
P. G. Vaughan-Shaw Department of Colorectal Surgery, University of Edinburgh,
Western General Hospital, Edinburgh, UK
Lorenzo Maria Vetrone Dipartimento Universitario di Medicina e Chirurgia
Traslazionale, Università Cattolica del Sacro Cuore, Rome, Italy
Matthew Vincent Department of Metabolism, Digestion, and Reproduction, Imperial College London, Hammersmith Hospital, London, UK
Maria Laura Vuolo Emergency Surgery/Pelvic Floor Center, Careggi University
Hospital, Florence, Italy
Janindra Warusavitarne St. Mark’s Hospital, London, UK
Dakshitha Wickramasinghe Department of Surgery, Faculty of Medicine, Uni-
versity of Colombo, Colombo, Sri Lanka
David D. E. Zimmerman Colorectal Research Group, Department of Surgery,
ETZ (Elisabeth – TweeSteden Ziekenhuis) Hospital, Tilburg, The Netherlands
Part I
Basics for Anal Fistula and Abscess
Management
Epidemiology of Anal Fistula and Abscess
Andrea Bondurri
Contents
1 Introduction . ...... . . . . ....... . . . . ...... . . . . . ...... . . . . ....... . . . . ....... . . . . ....... . . . . ...... . . 4
2 Incidence and Prevalence ........................ ........................................... ... 5
2.1 Europe ........................................................... ......................... 5
2.2 USA and Canada ......................................................................... 6
3 Host Factors: Age, Gender, Lifestyle .......................................................... 6
4 IBD and Other Etiologically Relevant Comorbid Conditions ......... ............. .......... 6
5 Anatomical Presentation and Rectovaginal Fistula ............................. .............. 7
6 Anal Fistula Development: Microbiological Factors .......................................... 8
7 Recurrence and Cancer Risk ... . ......................................... ...................... 8
8 Conclusion ................... ................................ ............................... ... 9
References ..................................................... ..................................... 9
Abstract
1
Perianal abscess and anal fistula are common anorectal benign diseases, known
since ancient Egypt.
Very recent studies have better defined their epidemiology with a mean prevalence of 8–23 per 100,000 people. Data confirm that 90% are cryptogenetic but
IBD-associated lesions should always be considered because a mean prevalence of
4 per 100,000 people was recently calculated. The crypto-glandular hypothesis
proposes that infection of an intersphincteric gland leads to fistulation out to the
perianal skin. However, not all anorectal abscesses lead to persistent fistula.
Moreover, other causes such as tuberculosis, trauma, hidradenitis suppurativa,
HIV infection, sexually transmitted disease, radiotherapy, or malignancy should
always be excluded. The mean age of first presentation is reported to be 40 years in
both sexes but men are twice as likely as women to develop perianal abscess or anal
A. Bondurri (*)
General Surgery, Luigi Sacco University Hospital, Milan, Italy
e-mail: andrea.bondurri@asst-fbf-sacco.i t
© Springer Nature Switzerland AG 2022
C. Ratto et al. (eds.), Anal Fistula and Abscess, Coloproctology,
https://doi.org/10.1007/978-3-030-76670-2_1
3
4 A. Bondurri
fistula. Smoking is still an important host risk factor. Research is moving into the
comprehension of the complex interaction of host, local pro-inflammatory signals,
and infections but data about microbiological factors in term of occurrence or
recurrence are controversial.
Keywords
Anal fistula · Epidemiology · Prevalence · Crohn’s disease · Comorbidities · Risk
factors
1 Introduction
Perianal abscess and anal fistula are some of the most commonly presenting
anorectal benign diseases.
Cases of incisions of perianal abscess were sporadic during the Fourth Dynasty of
ancient Egypt (2620–2500 BC) (Győry 2008).
Challenging in management of fistula was already described in The Sushant
Samhita, one of the most important surviving ancient treatises on medicine. Suhruta
(b ~ 800 BC) described both fistulotomy and fistulectomy as well as the use of a
chemical seton (Sankaran 1976).
Hippocrates of Kos (b ~ 460 BC) described the use of horsehair (seta) in the
treatment of anal fistula (Malik and Nelson 2008).
Magister Johannes de Arderne (1307–1390 CE), considered one of the fathers of
proctology, described regimens for treatment of anorectal fistula; a condition
believed secondary to long hours in the saddle by the knightly class and sedentary
habits in association with chronic constipation among the religious and civil population (Bernstein 1970; Beynon and Carr 1988).
Perianal abscess affect primarily young patients of working age (Sneider and
Maykel 2013). This condition is usually idiopathic (approximately 90% of cases)
caused by infection of the so-called anal glands or crypts, whereas about 10% of
cases have an underlying cause such as Crohn’s disease, tuberculosis, trauma,
hidradenitis suppurativa, HIV infection, sexually transmitted disease, radiotherapy,
malignancy, or foreign bodies (Pearce et al. 2016; Vogel et al. 2016). Classically,
perianal fistula has been considered to share a crypto-glandular origin with perianal
abscess and to be part of the same disease (Vogel et al. 2016; Parks et al. 1976).
There is a clear relationship between anorectal abscesses and fistulas. Most
patients with a fistula will have had a recognizable abscess prior to presentation,
and one-third of patients with an anorectal abscess will have a concomitant fistula at
the time of presentation (Sugrue 2017). However, not all anorectal abscess lead to
persistent fistula. Recent data from an observational population-based study in the
UK demonstrated an overall rate of fistula formation following anorectal abscess of
17.2% (15.5% in idiopathic cases and 47.2% in CD) (Sahnan et al. 2017).
Few translational studies have highlighted the precise natural history of the two
conditions (Parés 2011).
1 Epidemiology of Anal Fistula and Abscess 5
There is no definitive means of preventing or predicting fistula occurrence or
formation after abscess drainage.
2 Incidence and Prevalence
Observational studies on the frequency or incidence of perianal abscess or anal
fistula determinants, as well as their temporal or geographical variation, are lacking.
The prevalence of anal fi stula is estimated to be 1–2 per 10,000 patients, but
population-based studies on anal fistula epidemiology are limited (Hokkanen 2019).
Real world prevalence may be higher, since many patients are treated with
antibiotics in the community and some abscesses spontaneously regress or discharge. Patients usually present with an erythematous swelling near the anus and
may be reluctant to seek treatment. They may present to primary care and emergency
physicians, general and colorectal surgeons, gastroenterologist, and infectious diseases specialists (Sahnan et al. 2017; Wright 2016).
The true incidence is unknown. Most publications on anal fistula reflect the
authors’ experience, some quite large, from a single institution (Nelson and Abcarian
2014).
There are few recent national and even international data on the epidemiology,
classification, and treatment of anal fistulas, so there is a need for research seeking to
show the current status of this condition (Fugita et al. 2020).
Anal abscesses and fistulas account for up to 5% of proctological consultations
(Sugrue 2017).
2.1 Europe
The prevalence of anal fistulas in Europe is 16.9 per 100,000 (García-Olmo et al.
2018) and in Western nations is reported to range from 5.6 to 20.8 people in 100,000,
occurring most frequently in patients in their 30s and 40s (Felt-Bersma and Bartelsman
2009; Schwartz et al. 2002; Rankin et al. 1979). The mean incidence of anal fistula is
estimatedto be 8.6 per 100,000 people, 10.4 per 100,000 people, and 23.2 per 100,000
people in Finland, Spain, and Italy, respectively (Zanotti et al. 2007).
A recent Swedish cohort study estimated the incidence at 16.1 per 100,000
(Adamo et al. 2016).
Sahnan recently studied data from 165,536 patients with an episode of new
perianal abscess between 1997 and 2012: the crude incidence rate was 20.4 per
100,000 (Sahnan et al. 2017).
Hokkanen performed a retrospective population-representative observational
cohort study in a UK primary care database and estimated an overall point prevalence of anal fistula of 18 per 100,000 patients in 2017, while the Europe standardized estimate was 18.3 per 100,000 patients. Both these standardized point
prevalence estimates ranged from 18.9 to 23.6 between 2014 and 2016 (Hokkanen
2019).
6 A. Bondurri
2.2 USA and Canada
Hospital discharges or formal operations in the operation rooms are usually recorded and
are available for statistical evaluation. On those basis, Nelson and Abcarian previously
reported an incidence of anal fistula of 0.69–0.80% (Nelson and Abcarian 2014).
3 Host Factors: Age, Gender, Lifestyle
True perirectal abscesses are very rare in infants and children. These are rarely
visible, and diagnosis is usually based on imaging. Et iology is related to trauma,
Crohn’s disease, immune deficiency, or an infected mass lesion (e.g., rectal duplication or teratoma). Fistulas occur in infants with recurrent perianal abscesses and
rarely signify underlying pathology. However, in older children and teens, these may
be the first recognized manifestation of Crohn’s disease (Jamshidi 2018). The
majority of pediatric patients are male. The fistula tract is typically subcutaneous
and straight. The fi stulas occur evenly around the anal circumference. Multiple
fistula tracts occur in 15–20% of cases (Pakarinen et al. 2020).
The mean age of first presentation is reported to be 40 years in both sexes (Sainio
1984; Abcarian 2011).
Several studies have shown that men are twice as likely as women to develop
perianal abscess and fistula (Fugita et al. 2020; Sainio 1984; Ramanujam et al. 1984;
Read and Abcarian 1979). Reasons for this are unclear. There are no differences in
histology or distribution of anal glands between the sexes (McCol l 1967). There is
evidence that pro-inflammatory cytokines are enhanced by testosterone, but
inhibited by estrogen (El-Tawil 2012). However, there are no differences in sex
hormone concentrations between sufferers of either sex and healthy controls (Phillips and Clark 2014).
There was no evidence that abscesses are related to personal hygiene or sedentary
lifestyles, but Wang recently compared 1342 patients with anal fistulas to controls
with other anorectal complaints and found several independent risk factors for anal
fistula on multivariate analysis including: body mass index greater than 25 kg/m
high daily salt inta ke, diabetes, hyperlipidemia, dermatosis, anorectal surgery, history of smoking and alcohol intake, sedentary lifestyle, excessive intake of spicy/
greasy food, infrequent participation in sports, and prolonged sitting on the toilet for
defecation (Wang et al. 2014).
Smoking is a well-known risk factor for anal abscess or fistula development, but
the risk returns to baseline 5–10 years after smoking cessation (Devaraj et al. 2011).
2
,
4 IBD and Other Etiologically Relevant Comorbid Conditions
Perianal abscess and anal fistula are less frequently associated with other causes:
inflammatory bowel diseases; abdominal or pelvic infections (such as from diverticulitis); trauma (3.3%) or direct penetration of the anal wall (by chicken or fish
1 Epidemiology of Anal Fistula and Abscess 7
bones or by anal digitation); surgery (1%); perfor ation from low rectal or anal
cancers; tuberculosis (0.2%); actinomycosis; hidradenitis suppurativa; sexually
transmitted diseases and penetrating ulcers; irradiation. Approximately 7% of
patients with lymphogranuloma venereum and 80–91% of patients with anorectal
tuberculosis present with anal fistula. Patients with chronic diseases like diabetes
mellitus are also discussed to be at increased risk of anal fistula, due to their
susceptibility to skin lesions and systemic infections (Sainio 1984; Goligher et al.
1967; Lunniss and Phillips 1994; Parks 1961; Williams et al. 2007; Lillius 1968;
Abeysuriya et al. 2010; Marks et al. 1981; Culp 1983; Shukla et al. 1988; Scieux
et al. 1989; Gupta 2005).
The first fistula in Crohn’s disease was described in 1921 by Gabriel, 9 years
before Crohn identified CD as a clinical entity.
It was already known from the 1980s that near one-third of patients with CD have
at least one anal fistula during the course of the disease. A population-based study
from Olmsted County documented that the cumulative risk of a patient with CD to
develop perianal fistulas was 22% at 10 years and 26% at 20 years after diagnosis
(Schwartz et al. 2002). In approximately 10% of patients, the presence of perianal
fistulas can be the initial manifestation of CD (Molendijk et al. 2014).
Hokkanen recently conducted a retrospective population-representative observa-
tional cohort study and found that the overall crude point prevalence of anal fistula
with CD per 100,000 patients was 4.5 in 2017 and ranged from 4.9 to 5.2 in
2014–2016. The standardized point prevalence estimate of anal fistula with CD
was 4.4 for both United Kingdom and Europe (Hokkanen 2019).
5 Anatomical Presentation and Rectovaginal Fistula
In patients presenting for treatment of anorectal abscesses and fistulas, perianal
abscess occurred in 40–42.7%, ischiorectal in 20–22.7%, intersphincteric in
21.4%, and supra-levator in 7.0–7.33% of cases (Read and Abcarian 1979).
Rectovaginal fistula (RVF) is distressing and has a significant negative impact on
physical and psychological well-being. Crohn’s disease is the second most common
cause of RVF after obstetrical trauma. Other causes include abdominal or anal
surgery, inflammatory or malignant processes (e.g., diverticulitis, rectal cancer),
radiation, and trauma (Pinto et al. 2010; Homsi et al. 1994; Venkatesh et al. 1989;
Bahadursingh and Longo 2003; Saclarides 2002).
The exact incidence of RVF is unknown, but it is seen in 9–10% of patients with
Crohn’s disease (Radcliffe et al. 1988; Andreani et al. 2007) and in approximately
10% of patients following low anterior resection (Bahadursingh and Longo 2003;
Saclarides 2002). It is a complication in 0.1% of vaginal births: obstetric trauma
leading to RVF formation results from perineal laceration or from prolonged ischemia and necrosis of the tissue following obstr ucted labor (Saclarides 2002). Risk
factors associated with severe perineal lacerations at the time of vaginal delivery
include episiotomy, older maternal age, higher birth weight, primiparity, and assisted
vaginal delivery (Angioli et al. 2000). A recent study highlighted the lack of a
8 A. Bondurri
scientific basis for this incidence and prevalence of fistula (Stanton et al. 2007).
These authors reported an estimated prevalence of 188 per 100,000 women aged
15–49 years in South Saharan Africa and emphasized the need for population-based
studies.
In modern obstetrics, improved care and timely intervention in labor as a result of
the unrestricted use of caesarean section have reduced the occurrence of genital
fistula (Tebeu et al. 2012).
6 Anal Fistula Development: Microbiological Factors
Gut-derived organisms contribute to the development of anal fistulas or they are
simply a marker of a fistulous process in a patient with an anorectal abscess?
Literature presents controversial data. Sugrue and colleagues recently presented
several studies with similar results: they all found that abscesses with gut-derived
microorganisms were significantly more likely to be associated with an anal fistula
rather than skin-derived organisms (Sugrue 2017).
However, a case series of 164 patients found no statistically significant associa-
tion between the presence of gut-derived organisms and the development of a fistula
or recurrence of perianal abscess (Xu and Tan 2016).
7 Recurrence and Cancer Risk
The development and persistence of anal fistulas remains incompletely understood.
In addition to patient and surgeon factors, current data suggest that histological,
microbiological, and molecular factors probably play a role (Sugrue 2017).
Factors associated with a higher likelihood of developing a fistula, recurrence,
and the need for early repeat drainage incl ude patients younger than 40 years without
diabetes, incomplete initial drainage and failure to break up loculations within the
abscess, missed abscess, and undiagnosed fistula (Sozener et al. 2011; Hamadani
et al. 2009; Onaca et al. 2001).
Based upon a retrospective case study of 500 consecutive patients undergoing
drainage of a perirectal abscess, horseshoe-type abscesses had been associated with
especially high rates of persistence and recurrence ranging between 18% and 50%
(Onaca et al. 2001).
Bacteria are not often found in chronic fistulas. Therefore, their contribution to
poor wound healing following an attempt at repair is less clear. Seow-Choen and
colleagues (1992) cultured granulation tissue from curettage samples obtained from
18 patients with anal fistulas, excluding those with inflammatory bowel disease and
acute suppuration. They found a low number of bacteria that grew only from
enrichments with the most common types being gut-derived organisms: E. coli
(22%), B. fragilis (20%), and enterococcus (16%). Based on these results, they
concluded that chronic inflammation in anal fistulas does not seem to be maintained
by either excessive numbers of organisms or organisms of an unusual type.
1 Epidemiology of Anal Fistula and Abscess 9
Tozer et al. (2015) obtained biopsy specimens from patients with both idiopathic
and Crohn’s fistulas aiming to characterize the microbiota within the fistula tracts.
Samples were carefully obtained near the internal opening, taking precautions to
avoid contamination from surrounding skin, and analyzed using fluorescent in situ
hybridization, Gram staining, and scanning electron microscopy. Surprisingly, only
1 of the 32 fistula specimens was found to contain bacteria associated with the wall
of the tract. The authors concluded that anal fistula tracts do not harbor high levels of
mucosa-associated bacteria, and alternative explanations are needed to explain the
persistence of anal fistulas.
Abcarian pointed that long-standing chronic draining wounds, such as anal
fistula, predispose to development of cancer; as it has been well documented in
diseases such as ulcerative colitis. Nelson and colleagues reported six cancers in
chronic fistulas which on the average were present for 13.8 years (Nelson and
Abcarian 2014). Several case-control and cohort studies have also shown an association between intr actable fistulas and development of cancer (Nelson and Abcarian
1996).
8 Conclusion
Very recent studies have better defined the epidemiology of perianal abscess and anal
fistula with a mean prevalence of 8–23 per 100,000 people. Data confirm that 90%
are cryptogenetic but IBD-associated fistula should always be considered because a
mean prevalence of 4 per 100,000 people was recently calculated. Other epidemiologically relevant comorbid condition should always be considered. Perianal abscess
and anal fistula are most common benign anorectal diseases but the risk of malignancy should be excluded. Research is moving into the comprehension of the
complex interaction of host, local pro-inflammatory signals, and infections.
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