International Journal of Surgery Case Reports 123 (2024) 110259

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International Journal of Surgery Case Reports

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Case report

Extrasphincteric anal fistula with intrarectal opening extended upto thigh,

successfully treated with a minimally invasive, novel surgical technique- a
rare case report
Swapna Bopparathi a,* , Narasimha Raju K.V b
a
Associate Professor, Department of Shalya Tantra, National Institute of Ayurveda, Deemed to be University, Amer Road, Jaipur, Rajasthan, India
b
Former Head and Professor, Dept. of Kaya Chikitsa, Faculty of Ayurvedic Sciences, Jayoti Vidyapeeth Women’s University, Jaipur, Rajasthan, India

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction: Fistula in ano is a complex disease, and the treatment for it is still a big challenge for surgeons
Anal fistula because of the high recurrence rate (7 %–50 %) and incontinence, and to minimize these complications
Continence preservation numerous surgical interventions are emerging daily in the conventional system of medicine.
Ksharasutra
Presentation of case: A 48- year- old male patient came with complaints of pus discharge from an external opening
Internal opening
in the inner aspect of his right thigh, located about 22 to 25 cm away from the anal verge for the last 15 years and
Recurrence
Retrograde probing was diagnosed as long extrasphincteric fistula with intrarectal opening based on clinical and MRI findings. We
successfully treated this case with a minimally invasive novel surgical technique, RetroGrade Probing and
Application of KharaSutra and Division of the Fistulous Tract (RGPAKS- DFT).
Discussion: Ksharasutra is a well-known method in the treatment of anal fistula for preserving continence and a
low recurrence rate. Performing retrograde probing in every single case of anal fistula can address the involved
anal gland under direct vision, which is essential for preventing recurrence and pairing retrograde probing with
the division of the fistulous tract significantly reduces the treatment duration with minimal tissue loss.
Conclusion: In fistula surgery, successful treatment relies on identifying the internal opening and eradicating the
involved anal glands. Based on the same principle, this rare and complex anal fistula was effectively treated with
this RGPAKS-DFT, resulting in no recurrence and incontinence in two years of follow–up after complete recovery.

1. Introduction Sushruta described anal fistula as Bhagandara under eight grave

disorders i.e., Ashtamahagdas in Ayurveda [6] and he (500 BCE) was the
An abnormal hollow tract that connects an internal opening in the first surgeon who explained the surgical excision and ksharasutra in anal
anal canal to an external opening in the perianal skin is known as an anal fistula management [7]. In ancient Indian texts, Ksharasutra is
fistula. Cryptoglandular infection, which originates in the intersphinc­ mentioned as a treatment modality in anal fistula [8]. It is a “medicated
teric space and spreads in multiple directions, is thought to be the pri­ seton” made from different medicinal plant extracts and latex impreg­
mary cause of anal fistulas in the adult population [1]. nated in multiple layers onto a linen thread. Different Ksharas/ alkali
Treatment of complex anal fistulas is still a challenge to surgeons as will be used for the preparation of different ksharasutras. These ksharas
these fistulas involve a significant involvement of the sphincter complex [9] have anti-inflammatory and chemical debriding properties [10]. It
and eradicating the entire fistulous tract is associated with a high risk of works as a cutting, draining seton and simultaneously promotes healing
incontinence. Additionally, failure to remove or excise the primary and and is a well-accepted treatment procedure for continence preservation
all the secondary tracts may result in the persistence or recurrence of and low recurrence rate. A study on the effectiveness of Ksharasutra
fistulas [2], and this rate of recurrence ranges from 7 % to 50 % in reported a low (3.33 %) recurrence rate over a 4-year follow-up period
various types of anal fistulae [3–5]. To minimize the rate of recurrence [11].
and preserve continence, various surgical procedures are emerging daily To prevent an abnormally high recurrence rate, accurate identifica­
in conventional medicine. tion of the internal opening is an essential step of fistula surgery [12,13].

* Corresponding author.
E-mail address: [email protected] (S. Bopparathi).

https://doi.org/10.1016/j.ijscr.2024.110259
Received 13 July 2024; Received in revised form 30 August 2024; Accepted 5 September 2024
Available online 7 September 2024
2210-2612/© 2024 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Limited. This is an open access article under the CC BY-NC-ND license
(http://creativecommons.org/licenses/by-nc-nd/4.0/).
S. Bopparathi and N.R. K.V International Journal of Surgery Case Reports 123 (2024) 110259

If an accurate internal opening fails to be identified and addressed, at approximately 8 o’clock position, about 45 mm above the anal verge.
recurrence is certain. This is just because the source of sepsis in these There is noticeable surrounding fibrosis or scarring in this area. There is
cases won’t be eliminated [14]. Hence, identification of the correct a small irregularity with a focal outpouching involving the anorectum
original internal opening and eradication of the involved crypts is the approximately at 9 o’clock position, possibly indicating the internal
ultimate solution to eradicate fistula in ano. This can be achieved with opening. Additionally, there are other ill-defined fibrosed or sinus tracts
the current, minimally invasive, innovative procedure, RetroGrade seen in the right ischioanal fossa. There was no evidence of osteitis in
Probing and Application of KharaSutra and Division of the Fistulous pelvic bones. MRI pelvis, indicated a traceable intrarectal opening at 9
Tract (RGPAKS- DFT). Here we treated this complex, rare anal fistula in o’clock position, but the intra-anal openings were not traceable. The
our hospital by using the above-said procedure. This work has been patient was effectively communicated about the prognosis and the
reported in line with the SCARE criteria [15]. anticipated treatment duration of approximately 8 to 9 months taking
into account the presence of an intrarectal opening.
2. Presentation of case After obtaining written informed consent, the patient was taken for
surgery under spinal anaesthesia on 1st September 2021. Surgery was
A 48-year-old male patient presented to our outpatient unit with a performed by the corresponding author and is experienced with the
complaint of pus discharge from an external opening in the inner aspect technique for the last 11 years. After placing the patient in lithotomy
of his right thigh over the past 15 years. He had no history of chronic position, an intraoperative inspection and digital rectal examination
medical conditions such as diabetes mellitus or hypertension. He is a were conducted. The surgical findings are aligned with the preoperative
chronic smoker since 30 years and has normal bowel movements. He is assessment (Fig. 3A-C). Methylene blue dye was flushed from the
from a middle level of socioeconomic status. The patient reported being external opening to confirm the accuracy of the visible internal openings
asymptomatic 15 years ago and he suddenly developed an abscess on the and dye was expelled from all three internal openings (Fig. 3D). The
upper inner aspect of his thigh, which spontaneously ruptured and shape of the internal (6 and 7 o’clock) openings was noted, with both
healed on its own without treatment. Over two years (2008–2009, directed towards 7 o’clock. Subsequently, a probe and curved fine artery
2009–2010), he had recurrent attacks of abscess at the same site for forceps were cautiously introduced into these internal openings to
which he was operated four times with incision and drainage. The pa­ ascertain the nature of the fistulous tracts, whether separate or inter­
tient was diagnosed as a case of anal fistula and was informed about the connected (Fig. 4A). After confirming that both tracts were separate, a
potential risk of incontinence and recurrence postoperatively. Due to rigid, brass probe was inserted through the 6 o’clock internal opening at
concerns about these complications, the patient opted not to undergo the level of the dentate line. The probe effortlessly navigated towards 7
surgical treatment, resulting in continuous suffering and recurrent ab­ o’clock and travelled approximately 2–3 cm away from the anal verge.
scess episodes over the fifteen years. He sought treatment at our hospital At this point, division of the fistulous tract (DFT) was performed using
on August 24th, 2021. Metzenbaum scissors and created a new external opening, through
On clinical examination, an external opening on the upper poster­ which the probe was brought out and a plain linen thread no. 20 was
omedial aspect of the right thigh, approximately 20 to 25 cm away from applied to this proximal tract (RGPAKS) (Figs. 4B-D, 5A, B). The same
the anal verge, was observed with purulent discharge and previous procedure was repeated through the 7 o’clock internal opening, and a
visible operative scars (Fig. 1A). No perianal external opening was thread was applied by bringing out the probe from the same newly
present. Digital rectal examination revealed three internal openings, created external opening. In this case, since both tracts were separate,
with two at the level of the dentate line at 6 o’ and 7 o’clock positions, separate threads were applied. No surgical intervention was performed
and the third approximately 2 cm above the anorectal ring at 9 o’clock for the intrarectal opening. To ensure proper drainage of the distal tract,
position. Based on the clinical findings (Fig. 1B), the case was diagnosed probing was carried out from the existing external opening. However,
as extrasphincteric fistula secondary to transphincteric fistula; Type 4, due to the tortuosity of the fistulous tract, the probe was non-negotiable
Park’s classification [16]. up to the newly made external opening or DFT wound. Therefore, an
MRI Pelvis revealed (Fig. 2A–D) a long tortuous fistulous tract with opening was created till where the probe was negotiable (Fig. 5C, D) and
an external opening in the posteromedial aspect of the upper right thigh. an infant feeding tube no. 8 was applied (Fig. 6A) for adequate drainage.
The tract was observed to course anteriorly, superiorly, and right Preoperative prophylactic single dose and postoperative Intravenous
laterally up to the right gluteus maximus muscle and then slightly pos­ antibiotics including inj. Ceftriaxone 1 g, intravenous, 12th hourly; inj.
teriorly, medially along the right gluteus maximus muscle with a Gentamycin 80 mg, intravenous, 12th hourly; inj. Diclofenac sodium 50
possible intramuscular component. From there, it courses anteriorly, mg, intramuscular, 8th hourly; and inj. Ranitidine, intravenous, 12th
superiorly and slightly medially, piercing the right puborectalis muscle hourly was administered for 48 h. From 3rd day onwards, suggested to

Fig. 1. Prepoperative findings.

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S. Bopparathi and N.R. K.V International Journal of Surgery Case Reports 123 (2024) 110259

Fig. 2. MRI pelvis findings (25/08/2021).

Fig. 3. Intraoperative findings (01-09-2021).

perform daily regular chores and initiated ayurvedic conservative Ksharasutra was changed and tightened once a week for next five weeks
treatment such as oral administration of anti-inflammatory drug, tablet and the tract was eventually laid opened with Ksharasutra. To ensure
triphala guggulu, 1 g, 12th hourly, after meals; isabgol husk 2tsf with proper drainage of the distal tract, an infant feeding tube was kept in
lukewarm water at bedtime; for a month. Patient was instructed, to have place without being changed over for these five months. On 22nd of
a warm water sitz bath by adding 3–4 pinches of purified borax (Tankan) February, after inserting sims speculum into the anal canal, the floor of
powder, once daily after defecation; wound dressing with Jatyadi tail the wound was examined for remnant tracts and a ramification was
smeared wick or gauze daily; and weekly once visit. identified at 6 o’clock position, below the level of the dentate line. To
The patient was kept on plain linen thread no. 20 (Fig. 6B) for four address this issue patient was taken for surgery under local anaesthesia
months to aid the healing of an intrarectal opening and other ramifi­ on 24th of February 2022 and RGPAKS was performed in the ramifica­
cations. After confirming the healing of the intrarectal opening by digital tion and the infant feeding tube in the distal tract was removed and
rectal examination (four months after surgery) treatment with apamarg replaced with plain linen thread no. 20 for drainage. Subsequently, the
ksharasutra was started for cutting and healing of the fistulous tract. The ksharasutra was changed once a week for three weeks and the tract was

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S. Bopparathi and N.R. K.V International Journal of Surgery Case Reports 123 (2024) 110259

Fig. 4. Steps of operative procedure (01-09-2021).

Fig. 5. Steps of operative precedure (01-09-2021).

successfully laid open with ksharasutra. On 19th of April 2022 the floor patient with high satisfaction score. There are no signs of recurrence
of the wound was re-examined (Fig. 6C) and after confirming the floor to after two years of follow-up after recovery. The patient expressed
be healthy, plain linen thread from the distal tract was removed. overwhelming gratitude for the treatment, stating, “I am excited and
Notably, no curettage or coring was performed for the distal tract, and it overjoyed with my complete recovery and I feel like I got a new life after
got healed completely (Fig. 6D) within the subsequent ten days. suffering for fifteen long years.”
The patient has not experienced any postoperative complications
such as incontinence to flatus, liquids, and stool (as per Wexner’s
continence score), and the operative procedure is well tolerated by the

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S. Bopparathi and N.R. K.V International Journal of Surgery Case Reports 123 (2024) 110259

Fig. 6. Images captured during treatment and follow - up.

3. Discussion opening. Once the probe enters the original fistulous tract it navigates
effortlessly into the fistulous tract as per its course. In small fistulous
The current surgical procedures available for the extrasphincteric tracts, this procedure alone can be performed whereas in long fistulous
anal fistulas with an intrarectal opening often involves a temporary tracts (more than 4 cm), to minimize the tissue loss and to reduce the
diversion colostomy or the placement of a seton from an intrarectal overall duration of the treatment, RGPAKS should be paired with DFT.
opening, which has a high risk of incontinence or resection of the fis­ In DFT, division of the fistulous tract (DFT) will be done approxi­
tulous tract followed by surgical closure of the intrarectal opening with mately 2 to 4 cm away from the anal verge (according to the course of
unsatisfactory results. the tract) considering this point as a new external opening/ DFT wound.
The use of ksharasutra in the treatment of anal fistula is an estab­ The fistulous tract towards the anal verge is the proximal tract and treat
lished and safe technique for preserving continence. Ksharasutra is a it with Ksharasutra, the tracts away from the DFT wound are the distal
medicated thread made from linen no. 20 (Barbour) and is coated with tracts. If the DFT wound is kept well patent for adequate drainage, no
various herbal medicines and herbal extracted alkali (Kshara). These intervention such as applying thread/ feeding tube in the distal tracts is
ksharas have anti-inflammatory and anti-debriding properties with necessary. Once the source of sepsis is removed, the distal tracts will heal
which draining, cutting and healing of the tracts will be achieved in the next 10 to 15 days, without performing coring or curettage.
simultaneously. After the application of ksharasutra in the fistulous The following are some of the crucial points that contributed to the
tract, it remains in direct contact with the tract and curettes out the inner complete recovery and the prevention of recurrence in this case:
epithelial lining chemically and physically, allowing the fistulous tract
to cut and heal simultaneously [17,18]. Ksharasutra treatment involves 1. Addressing the correct internal openings according to their shape
the tightening and changing of the medicated thread every seven days. and nature
This process facilitates the cutting of the muscle complex, while the 2. Providing adequate drainage healed the intrarectal opening, all the
medicinal properties of the thread support the healing of the wound at ramifications, and secondary tracts.
the same time [19]. Moreover, Ksharasutra aids in draining and pro­ 3. Identifying the nature of multiple internal openings as separate and
moting the healing of the ramifications too. In this particular case, treating them with ksharasutra
apamarga ksharasutra was used for the treatment, where 21 coatings of 4. Examining the floor for remnant tracts
different herbal medicines were done on thread, such as 11 coatings of 5. Addressing the ramification with ksharasutra
latex of Euphorbia nerifolia Linn. (snuhi ksheer), 7 coatings of Achyr­
anthes aspera Linn. plant extracted alkali and 3 coatings of turmeric The advantages of this procedure are, that it is safe and cost-
powder [20]. effective, causes only minimal tissue loss, allows the patient to resume
Retrograde probing is a technique where probing will be performed daily activities from 2nd or 3rd postoperative day, preserves continence,
from inside the anal canal through the internal opening and surgeons leaves a minimal scar and maintains the integrity, anatomy and contour
usually choose this procedure when probing is failed from the external of the perineum with a single disadvantage, i.e., burning pain, which
opening or where an external opening is absent. But by performing lasts for ten minutes to two days after changing ksharasutra every week,
retrograde probing in every single case of anal fistula, surgeons can till the anoderm is laid open. During this period patients will be sug­
carefully examine the shape, direction, site, number, and nature of in­ gested to take ice compression and oral analgesics (if the pain is
ternal openings under direct vision, which is essential for preventing intolerable).
recurrence. In every fistula surgery, identifying the original internal opening is
In the RGPAKS procedure, the initial step involves thorough identi­ critical in minimising the risk of a high recurrence rate. To effectively
fication of the internal opening’s shape, direction, site, nature, and address this issue, it is important to perform retrograde probing through
number. Subsequently, a rigid probe is carefully passed through the the internal opening based on its shape, number, and nature to eliminate

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S. Bopparathi and N.R. K.V International Journal of Surgery Case Reports 123 (2024) 110259

the involved anal crypt and gland. This rare, complex extrasphincteric Conflict of interest statement
and long anal fistula with the intrarectal opening was successfully
treated with this minimally invasive, innovative surgical procedure, None.
without causing any postoperative complications such as incontinence
to liquids, gases and stool and there was no sign of recurrence in two Acknowledgements
years of follow-up.
We would like to thank Dr. Utkarsh Kabra, MD, EDIR, D-ICRI,
4. Conclusion Radiologist for his comments on MRI Pelvis.

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