Recurrent Perianal Fistula Research Articles

Endoscopic Fistulotomy (through the Scope and Freehand) as a Salvage Procedure for Recurrent Perianal Fistula with Abscess Postsurgery in Crohn's Disease

Endoscopic Fistulotomy (through the Scope and Freehand) as a Salvage Procedure for Recurrent Perianal Fistula with Abscess Postsurgery in Crohn's Disease

Technique for treatment of recurrent perianal fistula in Crohn's disease using autologous fat.

Technique for treatment of recurrent perianal fistula in Crohn's disease using autologous fat.

S2151 Unusual Presentation of Cancer of Unknown Primary Origin

Introduction: Colorectal cancer (CRC) is the fourth most diagnosed cancer in the world (1) with an overall lifetime risk of 4.3% for men and 4% for women (2). Adenocarcinomas account for 96% of all CRC (3), while Perianal adenocarcinomas account for 2-3% of all gastrointestinal malignancies (4), usually originating from perianal abscesses and Fistula in Ano. Here we present an unusual case of a 74-year-old man who presented with a right gluteal lesion and was found to have moderately differentiated adenocarcinoma on punch biopsy with immunochemistry (IHC) suggestive of colorectal origin. However, lesion in the colon was not identified. Case Description/Methods: The patient is a 74-year-old man with a past medical history of Crohn's disease with recurrent perianal fistulas and abscesses who presented with a progressively increasing right gluteal lesion for the past 2 years. Physical examination revealed a large fungating friable mass extending over the entire right buttock area, with yellow foul-smelling slough and serosanguineous discharge. Laboratory workup revealed leucocytosis, microcytic anaemia, and elevated carcinoembryonic antigen (CEA) levels. An MRI of the pelvis revealed a 9.8 x 16.1 x 7.7 cm heterogenous mass inseparable from the posterior aspect of the anus and along the course of a previously seen perianal fistula. Histopathology of a punch biopsy of the mass was consistent with moderately differentiated adenocarcinoma, and IHC was suggestive of colorectal origin. Astonishingly, a luminal tumor was not identified on colonoscopy (Figure). Discussion: Cancer of unknown primary (CUP) is a relatively rare clinical entity and their ability to metastasis prior to formation of a clinically identifiable primary is a diagnostic challenge. Although imaging such as CT and PET scans are helpful, light microscopic examination of initial biopsy is the most important clue towards the organ/site of origin. Peculiar findings should prompt IHC as highlighted in this unusual case.Figure 1.: Gross appearance of tumor.

Comparative Study of Conventional versus Marsupilized Fistulotomy of Recurrent Perianal Fistula

Introduction and Aim: There is a paucity of studies comparing marsupialization with the conventional fistulotomy technique in the management of recurrent perianal fistula. The current study aimed to compare between conventional and Marsupilized fistulotomy procedures in the management of recurrent perianal fistula regarding perioperative outcomes (pain, infection, healing time, incontinence, and recurrence).
 Methodology: Twenty four patients with recurrent perianal fistula and underwent conventional surgery (Group A) or fistulotomy with marsupialization, were included. The preoperative assessment achieved by history taking, physical examination and laboratory investigations. The patients were discharged on the first postoperative day. The severity of postoperative pain was assessed by the visual analogue scale. Patients were informed about regular outpatient visits after one week, two weeks, one month, three months, six months and nine months to assess wound healing, recurrence, inflammation and incontinence. Both groups were compared for postoperative pain, infection, rate of healing, time for healing, recurrent and postoperative incontinence.
 Results: Both groups were comparable regarding patient age, sex distribution (most included cases were males), preexisting medical comorbidities. Perianal discharge was present in all cases. Anal pain was reported by 33.33 and 25% of cases in the conventional and Marsupilized groups respectively. There was a significant increase in operative time in the Marsupilized than the conventional group (38.75 vs. 27.08 respectively, p < 0.001). The duration of wound discharge showed a significant reduction in the Marsupilized than conventional (2 weeks versus 3 weeks). Wound size was 2.11 and 3.31 cm3 in the Marsupilized and conventional groups respectively (p = 0.033). Time needed for wound healing showed a significant shortening in the Marsupilized group (p = 0.005).
 Conclusion: Marsupialization during surgical management of perianal fistula is associated with better post-operative outcome regarding wound size, discharge, and time for complete healing.

Predictors of reoperation for perianal fistula in Crohn's disease

Treating perianal fistula in cases of Crohn's disease (CD) remains challenging and the postoperative recurrence rate of perianal fistula is 22%-28%. This study aimed to identify the predictive risk factors for reoperation in Korean CD patients with perianal fistula. Medical records of the patients with clinically and pathologically confirmed CD who underwent surgical treatment for perianal fistulas at four referral centers in Korea between March 2010 and February 2020 were retrospectively reviewed. The rate of reoperation due to perianal fistula recurrence, which was defined as any subsequent surgery for perianal fistula or abscess, and the potential risk factors for reoperation were analyzed. Fifty-one patients at a mean age of 22 years were included in the study. During a median follow-up period of 26 months (range 2-89 mo), 21 (41.2%) patients underwent reoperation because of recurrent perianal fistula or abscess. The median interval from the first surgery to reoperation was 13 months. A multivariate Cox regression analysis revealed that drug escalation (from 5-aminosalicylic acid [5-ASA] to thiopurine or from 5-ASA or thiopurine to anti-tumor necrosis factor agents) after the first surgery was associated with a reduced likelihood of reoperation (hazard ratio 0.316, 95% confidence interval 0.117-0.858, P=0.024). The postoperative recurrence rate was relatively high (41.2%) after the first surgery for perianal fistula in Korean patients with CD. Drug escalation therapy after the first surgery may help reduce the need for reoperation for perianal fistula.

Is routine pathological analysis of perianal fistula specimen necessary in diagnosis of crohn's disease?

Background: Crohn's disease causes recurrent and complex perianal fistulas. Although the prevalence of it is up to 30%, the common cause of perianal fistula is thought to be the crypto-glandular theory. Surgeons send perianal fistula specimens for histopathological analysis; however, it is unclear whether such practices should be performed routinely. Objective: Evaluating the utility of routine histopathology on perianal fistula specimens during surgery to exclude the diagnosis of Crohn's disease. Design: Multicenter retrospective study was conducted from January 2012 to October 2018 on patients who underwent surgery for perianal fistula and specimen sent for histology. Setting: Metropolitan tertiary referral center in Melbourne, Australia. Patients and Methods: 105 patients who underwent 124 anal fistula procedures and their perianal fistula specimens sent for histopathological analysis were selected from the medical database. Medical and pathology reports were analyzed and data were reviewed by a second author for consistency. Sample Size: 105 patients, 124 procedures. Main Outcome Measures: Histopathological results suspicious for Crohn's disease and endoscopic examination results to confirm Crohn's disease. Results: 41 together, 124 perianal fistula specimens were collected from 105 patients. The male to female ratio was 2.9:1 and the average age was 43.6 years. Nonspecific inflammation was seen in 121 (97.5%) specimens. Three specimens had granulomatous inflammation of which, only two (1.6%) had Crohn's disease confirmed on endoscopic biopsy of the terminal ileum. In 15 patients with Crohn's disease, none of the 19 specimens sent for histology demonstrated histopathological features of Crohn's disease. Conclusion: Routine histopathological analyses of perianal fistula specimens provide limited clinical value. Clinicians should selectively send specimens for histopathological analysis to limit the use of resources. Limitations: Retrospective study. Not all perianal fistula specimens were routinely sent for the analysis. Data prior to 2012 was not collected as we are limited by an electronic database which was commenced in 2012. Conflict of Interest: None.

IL-10 and IL-10 Receptor Mutations in Very Early Onset Inflammatory Bowel Disease.

Very early onset inflammatory bowel disease (VEO-IBD) is a unique disease entity with a complex genetic susceptibility in affected patients. Next-generation gene sequencing techniques have revealed various monogenetic mutations contributing to the pathogenesis of VEO-IBD, including interleukin 10 (IL-10) and IL-10 receptor (IL-10R) mutations. In this article, we reviewed the features of and effective therapeutic options for VEO-IBD with IL-10 and/or IL-10R mutations. The IL-10 signal pathway inhibits the release of several key cytokines and thereby has a significant anti-inflammatory effect in the gastrointestinal tract. Mutations of the genes encoding IL-10 and/or IL-10R have been detected in VEO-IBD patients among myriad populations throughout the world. VEO-IBD patients with IL-10 or IL-10R mutations often present with repeated bouts of bloody diarrhea, marked weight loss, growth retardation, and recurrent perianal problems, including abscesses, fistulas, and significant fissures. Moreover, some patients may have folliculitis and present with pulmonary infections. While the therapeutic efficacy of immunosuppressants is typically poor in these patients, allogeneic hematopoietic stem cell transplantation (HSCT) has been reported to improve symptoms significantly. However, the long-term prognosis of VEO-IBD patients with IL-10 or IL-10R gene mutations treated with HSCT requires further exploration to verify the efficacy and safety of this treatment. We concluded that clinicians should recognize the clinical phenotype of VEO-IBD, as mutational analysis of the IL-10 pathway can support the diagnosis and prompt early treatment of this complicated disease.

P-197 Identification of a Homozygous Mutation in the ZBTB24 Gene in a Patient with Very Early Onset Inflammatory Bowel Disease

Very early onset inflammatory bowel disease (VEO-IBD) occurs in children less than 5 years old and is frequently characterized by a more severe disease course than older onset IBD. The etiology likely includes a significant genetic contribution, particularly genes associated with immunodeficiency. The increased utilization of whole exome sequencing (WES) has identified several rare and novel variants in immune pathways that are associated with this phenotype. A 17-month-old boy with a family history of consanguinity and Crohn disease in his maternal grandmother initially presented with poor growth, recurrent fever, constipation, and intermittent blood in his stool. He was discovered to have a recurrent perianal fistula. Colonoscopy revealed multiple ulcerations in the distal 10cm of his colon and rectum. He was treated with azathioprine, corticosteroids, mesalamine enemas, and metronidazole and had resolution of his symptoms and fistula within 6 months. He developed serious infections as well, including multiple episodes of febrile pneumonia requiring hospitalization and IV antibiotics. Additionally he had 2 episodes of acute otitis media but no sinusitis or cellulitis. His growth remained poor. His physical exam was notable for hypertelorism, epicanthal folds, low set posteriorly rotated ears, and developmental delay. Immune work up revealed combined immunodeficiency including total hypogammaglobulinemia, decreased production and maturation of B cells, decreased NK cells, and poor titer response to vaccines. WES detected a potential causative homozygous frameshift mutation, c.1492-1493delCA: p.Q498VfsX15, in ZBTB24, which is involved in B cell differentiation. The subject's mother and father are heterozygous for the same mutation. Homozygous and compound heterozygous mutations of ZBTB24 have been described in patients with immunodeficiency, centromeric instability, and facial anomalies syndrome type 2 (ICF2, OMIM 614069). Karyotype to confirm this diagnosis was performed. Underlying immunodeficiencies can present with phenotypic characteristics of VEO-IBD. Here whole exome sequencing together with immunophenotyping detected a novel variant in a known immunodeficiency gene presenting as recurrent pneumonia and inflammatory bowel disease.

EFFICACY OF ARGENTUM-QUARTZ SOLUTION IN THE TREATMENT OF PERIANAL FISTULAS: A PRELIMINARY STUDY

Objective: Nowadays, an optimal and effective medical surgery remains the gold standard for perianal fistulas. Hereby we reported preliminary results in favor of using Argentum-quartz solution for both primary and recurrent perianal fistulas. Methods: Three patients with intersphincteric and extrasphincteric fistulas were enrolled. Argentum-quartz solution was administrated twice a week for a period of 4 weeks, followed by a pause of 8 days and then another 4 weeks of treatment, totally 16 administrations. After treatment, all patients were monitored for a 4-month follow-up. Results: Complete closures of 2 extrasphincteric fistulas and a partial closure with absence of inflammation and superative phenomena in the intrasphincteric fistula were both manifested. Conclusion: Selective treatment of perianal fistulas with an argentum-quartz solution is safe and effective, and may represent a reliable alternative.

Rectovaginal Fistula and Uterine Prolapse: A Transvaginal Uterine-Sparing Single-Stage Surgical Approach

Abstract Background: In cases of women with inflammatory bowel disease and pelvic floor disorders, the common practice has been to manage their complaints in separate surgical stages. Case: We present a patient with Crohn's disease complicated by prior colostomy and recurrent rectovaginal and perianal fistulas with symptomatic stage 3 uterovaginal prolapse and rectovaginal fistula. She successfully underwent a transvaginal fistula repair and uterine-sparing prolapse correction without intestinal diversion. Conclusions: Patients with inflammatory bowel disease and multiple pelvic floor disorders may be successfully managed with a single-stage surgical approach within a multidisciplinary setting. (J GYNECOL SURG 31:289)

Direct MRI fistulography with hydrogen peroxide in patients with recurrent perianal fistulas: a new proposal of extended diagnostics.

BackgroundPerianal fistulas are malformations of the anorectal area. Accurate preoperative assessment of perianal fistula tract is a main assumption in diagnosis of the disease, affecting the operation efficiency. The aim of the study was to present our experience in application of a new diagnostic protocol based on the magnetic resonance imaging (MRI) examination using a mixture of hydrogen peroxide (HP) and gadolinium as a direct contrast medium in evaluation of recurrent fistulas tract. The method is referred to as HPMRI.Material/MethodsThe study group consisted of 12 subjects operated on from 2011. Direct HPMRI fistulography was performed in all subjects before the operation. All types of fistulas were precisely evaluated by HPMRI examination.ResultsIntraoperative state confirmed complete course of fistulas in 11 cases. In 1 case, an internal opening was not found.ConclusionsWe suggest that this new method of direct HPMRI fistulography may improve visualization of the tracts of recurrent fistulas and improve efficacy of surgical procedures.

Fecal Microbiota Transplantation (FMT) in the Treatment of Inflammatory Bowel Disease (IBD): A Case Report: ACG/AstraZeneca Clinical Vignette Award: Presidential Poster

Case: A 26-year-old man with recurrent perianal fistula presented in August 2012 with a onemonth history of bloody diarrhea, abdominal pain and 10-lb weight loss. He had been previously healthy, and did not take any medication. Further investigation at that time revealed Hb 11.4, MCV 91, WBC 14.8, PLT 309, CRP 24.8, normal electrolytes, creatinine and LFT, negative stool C&S, O&P, and Clostridium difficile. Colonoscopy demonstrated moderately severe and continuous mucosal inflammation from rectum to cecum, with normal terminal ileum. Colonic biopsies demonstrated chronic active inflammation with architectural distortion, consistent with IBD. He was started on Asacol, prednisone, and imuran. He responded well, but started to flare (Harvey Bradshaw Index=5) when prednisone was tapered to 20 mg per day. He quickly responded with an increased prednisone dose, but bloody diarrhea and abdominal pain recurred (HBI=12) when prednisone dose was tapered over the following 4 weeks. Stool was again negative for C&S and C. difficile. He needed to go on a biological agent, but his insurer refused to cover the cost. The discussion of fecal microbiota transplantation (FMT) as an experimental therapy was brought up with the patient as an option, and he agreed to proceed. He underwent FMT (universal donor) by colonoscopy, which demonstrated diffuse moderate mucosal inflammation, more severe in the proximal than the distal colon, and normal terminal ileum (Photo 1). Colonic biopsy subsequently ruled out CMV. Approximately 400 cc of fresh fecal suspension was infused in the cecum. Two days after FMT, he started to have 2-3 soft BMs per day, and his abdominal pain started to improve. Two weeks after FMT, his HBI was 0. The patient decided to pursue further FMT therapy as his maintenance therapy, and had a second FMT by colonoscopy 4 weeks after initial FMT. It showed complete mucosal healing (Photo 2). He continued to do well 6 weeks after the second FMT, and had the third FMT 8 weeks after the second FMT. Conclusion: To our knowledge, this is the first report of complete mucosal healing following a single FMT infusion in an IBD patient. These results suggest that FMT may be a promising therapy in a subgroup of IBD patients. Further research is needed to further characterize the IBD population most likely to benefit from FMT.Figure 1: Cecum at day 0.Figure 2: Cecum at day 28 following FMT.

Case report: misdiagnosis of tailgut cyst presenting as recurrent perianal fistula with pelvic abscess

Tailgut cysts are uncommon lesions that usually occur within the presacral space. The relative rarity and nonspecific complaints associated with these lesions often lead to misdiagnosis or unnecessary procedures before the correct diagnosis is made. We describe a case of a 16-year-old female who presented with pelvic pain. She had previously undergone several procedures at an outside institution for recurrent perianal fistula and perirectal abscess. Subsequent evaluation under anesthesia revealed a presacral cystic mass with a well-developed tract within the anorectal ring in the posterior midline. This mass was surgically removed using a combined transanal and posterior sagittal excision technique and was found to be a tailgut cyst upon pathologic evaluation. Tailgut cysts and other presacral masses should be included in the differential for patients with recurrent abscess in the presacral space or fistula within the anal canal. A variety of surgical approaches are available depending on the anatomy of the lesion.

A Case of Pulmonary Carcinoid Tumor in a Patient on Adalimumab for Crohnʼs Disease

Purpose: We report a unique case of pulmonary carcinoid tumor associated with adalimumab therapy for Crohn's disease. Methods: Adalimumab is a fully human monoclonal antibody against tumor necrosis factor alpha (TNFα). TNFα is a protein synthesized by activated macrophages and T cells. It is approved for the treatment of Crohn's disease and its' safety is established by clinical trials. However serious side effects can occur. Patients on TNFα inhibitors are at risk for injection reactions, serious infections and can develop malignancy. There is an FDA warning regarding risk of B-cell lymphomas and other tumors. Results: 63 year old female with fisutlizing, structuring Crohn's colitis and ileitis diagnosed at age 18, required multiple bowel resections and ileocolonic anastomosis. Presented with recurrent peri-anal fistulas and was started on adalimumab 10-11 months ago. Recently had developed a peri-rectal fistula and abscess requiring drainage. Previous smoker. No family history of Inflammatory Bowel Disease (IBD) or colorectal cancer. Physical exam notable for a non-tender, non-distended abdomen and rectal exam revealing non-tender peri-anal excoriations and a draining sinus fistula tract. Abdominal CT done for follow up of perirectal abcess had incidental finding of pulmonary nodules. This was a new finding; a chest CT then showed a 2cm RUL lung mass and multiple bibasilar sub-centimeter pulmonary nodules. Prior chest x-rays upon close inspection did not reveal the mass. Bronchoscopy with bronchiolar lavage and transbronchial biopsy was inconclusive. Whole body PET scan was remarkable for the right upper lung bronchial nodular density with low level FDG activity. Surgery performed a thoracotomy and wedge resection for diagnosis and cure. Pathology review confirmed a typical carcinoid tumor with pathologic stage of pT1a pN0. Adalimumab was stopped and she was placed on antibiotics for her fistulas along with prednisone taper. Patient later underwent an uncomplicated right lung wedge resection with curative intent and has since been doing well. She denies rectal pain or discharge and has 1-2 formed bowel movements daily. Post operatively she was placed on methotrexate for Crohn's maintenance therapy. Conclusion: Studies have demonstrated the clinical association of biologic therapy and Non-hodgekin's lymphomas. The action of TNFα antagonists has been shown in laboratory models to possibly potentiate tumor growth. Pulmonary carcinoid is a slow growing neuroendocrine tumor, but in this case appears to have developed in conjunction with adalimumab therapy. To our knowledge this is the first case of primary pulmonary carcinoid to be associated with adalimumab.

Haemorrhoids, rectal prolapse, anal fissure, peri-anal fistulae and sexually transmitted diseases

Anorectal disorders like haemorrhoids, rectal prolapse, anal fissures, peri-anal fistulae and sexually transmitted diseases are bothersome benign conditions that warrant special attention. They, however, can all be diagnosed by inspection or proctoscopy (sexually transmitted proctitis). Constipation can play an underlying role in haemorrhoids, rectal prolapse and anal fissures, and it is important to treat these conditions in order to avoid recurrences. Haemorrhoids and anal fissures are generally treated conservatively and surgery is seldom required. Rectal prolapse and cryptoglandular peri-anal fistulae are treated surgically. In a recurrent peri-anal fistula, the fistular tract needs to be visualised with anal ultrasound or magnetic resonance imaging (MRI). There are different techniques available for this evaluation, and care must be taken not to damage the anal sphincter. Peri-anal fistulae in Crohn's disease are treated conservatively and surgery is only required in cases with abscesses. Sexually transmitted proctitis needs to be adequately recognised and treated according to the infectious agent.

Perianal eccrine adenocarcinoma

BackgroundEccrine carcinoma is a quite rare malignant tumor that typically arises from a normal sweat gland and that features a rather high recurrence rate subsequent to simple excision. Given its rather poor response to adjuvant therapy, wide excision of the lesion with tumor-free margins may offer a reasonable chance for long-term control of this neoplasm.Case presentationHerein, we report on an unusual case of perianal eccrine carcinoma, initially presenting as a perianal abscess.ConclusionEven though eccrine carcinomas would appear to be rare, when dealing with recurrent skin tumors or recurrent perianal fistulas, the possibility of eccrine carcinoma should be considered by consulting clinicians.

Mucinous adenocarcinoma arising from chronic perianal fistula

Perianal mucinous adenocarcinoma is a rare tumour which may be associated with long‐standing chronic perianal sepsis. Early diagnosis is challenging and is based on a high index of clinical suspicion and specific histological features. Definitive treatment is surgical, in the form of an abdomino‐perineal resection. We hereby describe a case of a perianal mucinous adenocarcinoma arising from long‐standing recurrent perianal fistula and complement this with a brief review of the literature pertaining in particular to the management of this condition.

Endosonographic Imaging of Anorectal Diseases

The normal sonographic anatomy of the anorectum, sonographic findings of anorectal diseases, and indications and limitations of endosonography compared with magnetic resonance imaging are reviewed. Methods. Endosonographic imaging was performed with a Siemens (Erlangen, Germany) FI 400 ultrasound scanner with an end-fire 7.5-MHz biplane endorectal probe and a B-K Medical (Sandhoften, Denmark) scanner with an 1850 axial-type side-fire 5.0- to 10.0-MHz rotating endoscopic probe. Rectal carcinoma appears on endorectal sonography as a low-echogenicity lesion that abruptly interrupts the normal sequence of layers. The internal anal sphincter is seen very clearly on endoanal sonography, and it is easy to appreciate atrophy and small tears of this sphincter. Endoanal sonography cannot accurately show thinning of the external anal sphincter. Peroxide-enhanced endoanal sonography is especially useful for patients with recurrent perianal fistulas in whom scarring should be distinguished from recurrent fistulas and detection of the internal opening. However, sonography does not provide an adequate deep and global display of all adjacent pelvic and perineal spaces. Endosonography can accurately stage primary rectal tumors and assess the internal anal sphincter. Peroxide-enhanced 3-dimensional imaging can increase the utility of endoanal sonography in detection and characterization of perianal fistulas and planning of optimal therapy. However, magnetic resonance imaging can be used a complementary modality to endosonography, especially for evaluation of external anal sphincter atrophy and deep pelvic inflammation.

Carcinoma of anal-ductal origin

A 57-year-old man who had a carcinoma proven to be of anal ductal origin is presented. Recurrent perianal fistulas were treated locally for two years until biopsy proved the malignancy. Gross and histologic examination of the specimen removed by abdominoperineal resection demonstrated the anal ductal origin of this tumor. Recurrence was seen in less than a year.