*Agnieszka Białas
Multiple anal fistulas are the first symptom of Crohn’s disease – a difficult diagnostic path. Case report
Mnogie przetoki odbytnicze pierwszym objawem choroby Leśniowskiego-Crohna – trudna droga diagnostyczna. Opis przypadku
Department of General Surgery, BCM Hospital in Bielsko-Biała
Streszczenie
Pacjenci z anorektalną postacią choroby Leśniowskiego-Crohna (CD) stanowią ok. 20% wszystkich pacjentów cierpiących na to schorzenie. Częstość jej występowania i rozpowszechnienie rośnie na całym świecie wraz ze wzrostem zapadalności na poziomie 4-15% rocznie w ciągu ostatnich trzech dekad. Choroba Leśniowskiego-Crohna jest chorobą na całe życie, jej optymalne leczenie jest multidyscyplinarne i staje się coraz bardziej złożone. W pracy przedstawiono przypadek 28-letniego pacjenta z mnogimi przetokami odbytu, z powikłanym przebiegiem choroby, które okazały się pierwszym objawem choroby Leśniowskiego-Crohna. Rozpoznanie zapalenia jelit nie zostało postawione od razu, gdyż objawy ogólne (bóle brzucha, biegunki) pojawiły się później niż objawy przetoki. Pacjent był kilkukrotnie operowany, jednak dopiero wprowadzone leczenie biologiczne doprowadziło do ustąpienia objawów zapalnych i wygojenia przetok. Autorka w podsumowaniu podkreśla, że wnikliwa diagnoza, szczególnie w przypadkach nietypowych, m.in. wystąpienia przetok mnogich, i wdrożenie odpowiedniego leczenia interdyscyplinarnego mogą doprowadzić do sukcesu terapeutycznego.
Summary
Patients with anorectal Crohn’s disease account for approximately 20% of all patients suffering from this condition. Its incidence and prevalence are increasing worldwide, with an annual increase of 4-15% over the last three decades. Crohn’s disease is a lifelong condition, and its optimal treatment is multidisciplinary and becoming increasingly complex. This paper presents the case of a 28-year-old patient with multiple anal fistulas and a complicated course of the disease, which turned out to be the first symptom of Crohn’s disease. The diagnosis of inflammatory bowel disease was not made immediately, as the general symptoms (abdominal pain, diarrhoea) appeared later than the fistula symptoms. The patient underwent several surgeries, but it was only after the introduction of biological treatment that the inflammatory symptoms subsided and the fistulas healed. In her conclusion, the author emphasises that a thorough diagnosis, especially in atypical cases, such as multiple fistulas, and the implementation of appropriate interdisciplinary treatment can lead to therapeutic success.
Introduction
Patients with the anorectal form of Crohn’s disease (CD) constitute approximately 20% of all patients with this condition. These patients also often experience a more aggressive course of the disease. In approximately 30% of patients, perianal symptoms are the first symptom – they may also be the only symptom of CD. Fistulas affect one-third of patients with CD, with the anorectal location being the most common. The fistulas formed in the course of CD are so specific that the American Gastroenterological Association (AGA) has developed a special classification for patients with Crohn’s disease, dividing fistulas into simple fistulas, in which the canal runs below the dentate line with a single internal opening, without purulent collections, and complex fistulas, in which the canal runs above the dentate line: high intersphincteric, high transsphincteric, suprasphincteric and extrasphincteric with multiple external openings, the presence of abscesses, rectal strictures and penetrating into the pelvic organs, e.g. the vagina or urinary bladder (1).
Case description
The paper presents a case of a 28-year-old patient with multiple anal fistulas and a complicated course of the disease, which turned out to be the first symptom of CD.
The patient reported to the Proctology Clinic urgently in May 2022 due to multiple anal fistulas. A year earlier (2021), he had undergone sclerotherapy for hemorrhoidal nodules twice due to bleeding after defecation (no documentation was provided). The patient tolerated the procedure well, and the bleeding stopped. Approximately six months after sclerotherapy, he experienced discomfort and pain in the left buttock area. He was consulted at the center performing the procedure and treated with oral antibiotics (Augmentin and Metronidazole). A magnetic resonance imaging (MRI) of the pelvis was recommended.
An MRI performed in October 2021 showed the presence of 3 fistulas, without signs of abscess:
• at 6 o’clock, simple transsphincteric fistula,
• at 1 o’clock complex transsphincteric fistula,
• at 11:30, suspicion of simple intersphincteric fistula.
In recent weeks, the patient has developed abdominal pain, without weight loss, no bowel movements disorders, regular stools 1-2 times a day, no constipation or diarrhea.
Apart from inhalant allergies, the patient was not undergoing any chronic treatment or taking any medications on a regular basis. He denied anal sexual intercourse. His family history of cancer and inflammatory bowel disease was negative.
Rectal examination at 6 and 1 o’clock revealed visible skin openings of fistulas, no purulent discharge, tender to palpation; at 11 o’clock there was no visible opening of fistula, no palpable openings in the canal, increased sphincter tone (very painful examination for the patient – therefore anoscopy was omitted) (fig. 1).
Fig. 1. Examination at the first visit, two cutaneous fistula openings visible
The patient was prescribed Metronidazole suppositories, a repeat MRI or transrectal ultrasound, a fecal calprotectin test, and a colonoscopy. He was informed of the possibility of a multi-stage treatment.
In June 2022, the patient came for a check-up with the following MRI results:
• at 1-2 o’clock – stable image,
• at 6 o’clock worsening – two fistula channels? fissure?,
• at 9 o’clock – extrasphincteric fistula,
• at 11-12 o’clock – intersphincteric fistula as before.
The patient reported a reduction in pain after treatment with suppositories.
Physical examination: during rectal examination and anoscopy at 6 and 1 o’clock two cutaneous fistula openings were visible, without purulent discharge, tender to palpation – a cannula was inserted – it penetrates both channels; at 9 and 11 o’clock no fistula opening, increased sphincter tone, but less than previously, an anoscope was introduced – no obvious internal openings were visualized.
The patient was qualified for examination under anesthesia with attempted fistula drainage (no internal fistula openings were visible, hence the uncertainty as to whether drainage would be possible).
However, approximately four days before hospitalization, the patient’s condition worsened: a painful swelling appeared in the area of the gluteal cleft on the spine. The patient was afebrile. A rectal examination at 6 o’clock revealed a palpable rectal abscess measuring approximately 2 × 3 cm, tender to palpation, with a distinct fluctuating sensation and severe pain. Augmentin was administered orally, and the patient was referred to the emergency surgical ward with a diagnosis of an abscess.
The surgery was performed on July 19, 2022 – an incision of the perianal abscess and thread drainage of the fistulas were performed (fig. 2a-d). The postoperative course and early wound healing were normal (fig. 3).
Fig. 2a-d. a) Intraoperative: two fistula openings and an abscess visible at 7 o’clock; b) intraoperative: abscess at 7 o’clock; c) intraoperative: incised abscess at 7 o’clock and drained fistulas at 5 and 7 o’clock; d) intraoperative: drained fistulas at 5 and 7 o’clock, with a common internal opening
Fig. 3. Follow-up: healing of the abscess and reduction of inflammation, posterior fistulas drained
The patient had his calprotectin level measured – 865 mcg/g (norm 150), and was then referred for an urgent colonoscopy, the description of which is attached below.
Rectal examination: Visible perianal fistulas, sutured, sphincters tight, and within fingertip reach, no abnormalities. Examination of the large intestine up to the cecum revealed a normal Bauhin’s valve and appendix opening. An instrument was then inserted through the Bauhin’s valve into the terminal ileum. Bowel preparation was performed using a Boston scale of 6 (P2/S2/L2). Further insertion of the instrument was impossible. The mucosa was friable, with contact bleeding, covered with shallow ulcerations, irregular, and with several pseudopolyps – numerous biopsies were taken for histopathological examination. The mucosa of the large intestine was smooth, shiny, with preserved vascular patterns, and without morphological changes.
The result of the histopathological examination showed: inflammatory infiltrates focally penetrating the muscularis mucosa, mixed inflammatory infiltrates, inflammatory granulation tissue, formation of micro-cracks – the image may indicate CD.
The patient was referred for a gastroenterology and proctology consultation at a reference center, where gastroenterology treatment for enteritis, fistula drainage, and consideration of possible biological treatment were recommended.
From October 2022, the gastroenterologist recommended Entocort and Imuran and a check-up after 3 months (then a possible qualification for biological treatment would take place).
A month later, the patient developed a small inflammatory infiltrate around the cutaneous opening of the fistula at 1 o’clock. The man received local Metronidazole suppositories and was referred for fistula drainage.
Surgery performed in December 2022. A 1 o’clock drainage of the rectal fistula was performed, followed by excision of the peripheral fragment of the fistula. The postoperative course was complicated by prolonged wound healing.
During follow-up visits in January and February 2023, the inflammatory infiltrate was less severe, but purulent material continued to drain when the surrounding tissue was compressed. Therefore, in March 2023, a resection of the peripheral portion of the fistula, along with the inflammatory infiltrate around the external opening, was performed. During follow-up visits, the wound healed normally, with intermittent reports of mild pain and occasional bleeding when the thread was moved.
Control calprotectin level determination in March 2023 – 731 mcg/g (previous test 865 mcg/g) – intensification of gastroenterological treatment was recommended.
The patient was finally consulted with a gastroenterologist in June 2023, then hospitalized in the Gastroenterology Department in July 2023, where a repeat colonoscopy was performed – inflammatory changes in the ileum and Bauhin’s valve were found.
The man was qualified for biological treatment. He scored 320 points on the CDAI scale. On August 30, 2023, the first dose of infliximab was administered. Since then, the patient has been on continuous biological treatment.
Powyżej zamieściliśmy fragment artykułu, do którego możesz uzyskać pełny dostęp.
Mam kod dostępu
- Aby uzyskać płatny dostęp do pełnej treści powyższego artykułu albo wszystkich artykułów (w zależności od wybranej opcji), należy wprowadzić kod.
- Wprowadzając kod, akceptują Państwo treść Regulaminu oraz potwierdzają zapoznanie się z nim.
- Aby kupić kod proszę skorzystać z jednej z poniższych opcji.
Opcja #1
29 zł
Wybieram
- dostęp do tego artykułu
- dostęp na 7 dni
uzyskany kod musi być wprowadzony na stronie artykułu, do którego został wykupiony
Opcja #2
69 zł
Wybieram
- dostęp do tego i pozostałych ponad 7000 artykułów
- dostęp na 30 dni
- najpopularniejsza opcja
Opcja #3
129 zł
Wybieram
- dostęp do tego i pozostałych ponad 7000 artykułów
- dostęp na 90 dni
- oszczędzasz 78 zł
Piśmiennictwo
1. American Gastroenterological Association Clinical Practice Committee. American Gastroenterological Association medical position statement: perianal Crohn’s disease. Gastroenterology 2003; 125(5): 1503-1507.
2. Adamina M, Minozzi S, Warusavitarne J et al.: ECCO Guidelines on Therapeutics in Crohn’s Disease: Surgical Treatment. J Crohns Colitis 2024; 18(10): 1556-1582.
3. Tsai L, McCurdy JD, Ma C et al.: Epidemiology and Natural History of Perianal Crohn’s Disease: A Systematic Review and Meta-Analysis of Population-Based Cohorts. Inflamm Bowel Dis 2022; 28(10): 1477-1484.
4. Singh A, Midha V, Kochhar GS et al.: Management of Perianal Fistulizing Crohn’s Disease. Inflamm Bowel Dis 2024; 30(9): 1579-1603.
5. Swaminathan A, Sparrow MP: Perianal Crohn’s disease: Still more questions than answers. World J Gastroenterol 2024; 30(39): 4260-4266.
6. Munster LJ, Meriba GR, Schuitema J et al.: Early diagnosis of Crohn’s disease in patients presenting with a perianal fistula: systematic review and development of a perianal red flags index. Tech Coloproctol 2025; 29(1): 89.
7. Munster LJ, Mönnink GLE, van Dieren S et al.: Fistulizing Perianal Disease as a First Manifestation of Crohn’s Disease: A Systematic Review and Meta-Analysis. J Clin Med 2024; 13(16): 4734.
8. Munster LJ, Pronk AJM, Mundt MW et al.: Prolonged Time to Diagnosis of Crohn’s Disease in Patients With Perianal Fistulas Negatively Affects Long-Term Outcomes. J Crohns Colitis 2025; 19(3): jjae146.
9. Eglinton TW, Barclay ML, Gearry RB, Frizelle FA: The spectrum of perianal Crohn’s disease in a population-based cohort. Dis Colon Rectum 2012; 55: 773-777.
10. Munster LJ, Hanna LN, Hart AL et al.; TOpCLASS perianal Crohn’s disease Consortium, the Dutch Initiative on Crohn and Colitis (ICC) and the Initiative on Crohn and Colitis – Surgery (ICC-S): Diagnosing Crohn’s disease in presumed cryptoglandular perianal fistulas: an expert Delphi consensus on early identification of patients at risk of Crohn’s disease in perianal fistulas (PREFAB). J Crohns Colitis 2025; 19(1): jjaf002.
