Management of rare, low anal anterior fistula exception to Goodsall's rule with Kṣārasūtra.

Anal fistula (bhagandara) is a chronic inflammatory condition, a tubular structure opening in the ano-rectal canal at one end and surface of perineum/peri-anal skin on the other end. Typically, fistula has two openings, one internal and other external associated with chronic on/off pus discharge on/off pain, pruritis and sometimes passing of stool from external opening. This affects predominantly male patients due to various etiologies viz., repeated peri-anal infections, Crohn's disease, HIV infection, etc., Complex and atypical variety is encountered in very few patients, which require special treatment for cure. The condition poses difficulty for a surgeon in treating due to issues like patient hesitation, trouble in preparing kṣārasūtra, natural and routine infection with urine, stool etc., and dearth of surgical experts and technique. We would like to report a complex and atypical, single case of anterior, low anal fistula with tract reaching to median raphe of scrotum, which was managed successfully by limited application of kṣārasūtra.

INTRODUCTION

Suśruta saṁhitā elaborately describes bhagandara (anal fistula). The condition is termed bhagandara as it does dāraṇā (tearing) of bhaga (perineum), guda (rectum) and basti pradeśa (pelvis). Bhagadara is enlisted among aṣṭamahāgada (eight intricate diseases)[1] which by nature are difficult to cure considering the morbidity, reoccurrence and social burden. The causative factors are attributed to the doṣa viz., vāta, pitta, śleṣma, sannipāta and the external factors such as trauma, infection etc., (āgantuja). The aggravation of the doṣa is through its causative factors. Repeated exposure to the causative factors causes the doṣa to get aggravated, confounded and immobilized vitiating māṁsa and rakta in the area measuring about one/two fingers around anus producing a reddish boil. If not managed, the boil suppurates and develops into a wound with single or multiple holes through which clear, frothy copious or slimy discharge passes constantly according to the doṣa involved.[2]

Contemporary medicine describes the causes of anal fistula as, abscess in one or more potential spaces, source of infection from anal fissure, an ulcer at the root of the pile mass, inflamed anal crypt, infection from hair follicle, infected sebaceous gland, infected sweat gland, inflamed/thrombosed pile mass, retained sutures after hemorrhoidectomy, foreign body penetrating from outside, radiation burns from X-rays and radio therapy. Systemic diseases like tuberculosis of intestine, ulcerative colitis, regional ileitis, recurrent appendicitis, urinary tract infections, prostatic infection, pilo-nidal sinuses, pott's spine and osteo-myelitis of pelvic bones may also cause fistula-in-ano.

In general, the external openings of the tract are found around the peri-anal area. In the present case the external openings were at septum of the scrotum, which is an unusual presentation and poses great difficulty to operate for a surgeon due to high chances of urethra and testis getting injured in the course of surgery.

Ayurvedic surgeons usually adopt kṣārasūtra in multiple sittings in order to cut through whole of the tract and simultaneously heal; which is quite a lengthy procedure and even painful. In the present case, we adopted a new technique where the kṣārasūtra was not replaced until the fistulous tract was completely cut, instead the replacement of kṣārasūtra was limited to two times and tested for its efficacy. Kṣārasūtra is a medicated thread prepared by utilizing barbour thread of size 20 number anointed with total 21 coatings of snuhi ksheera (latex of Euphorbia neriifolia), haridra (Curcuma longa Linn) and apāmārga (Achyranthes aspera) kṣāra.[3]

CASE REPORT

A 50-year-old male patient presented with complaints of persistent foul smelling discharge from his scrotal wound with pain while sitting and riding vehicle, since 6 months. Patient had undergone surgery thrice under spinal anesthesia at a private hospital, and yet the complaints were persisting. All the previous investigations were normal and the histopathology report showed a fistula tract with chronic inflammation. Finally, he approached our institute for further treatment. There was no associated history of fever, bleeding per rectum or constipation. He did not have a history of major illnesses apart from present illness. He hadn’t undergone any major surgery in the past and was on higher antibiotics and antiinflammatory drugs. Personal history of patient revealed that he was Hindu, vegetarian with good appetite, married, teacher by occupation with no habits of tobacco and alcohol use.

On examination, the patient's vital parameters were stable. On local examination, there were two visible external openings on the scrotum anteriorly on median raffe [Figure 1]. The internal opening was not palpable on per rectal examination. Both the external openings were four centimeters apart on palpation. There was hard indurated swelling with tenderness possibly due to chronic infection and heavy usage of antibiotics (antibioma), both external sinuses were nonmovable suggesting that they were adhered to hard indurated swelling. Probing was done to ensure direction and position of internal opening, but probing was not possible due to induration.

Figure 1

External opening of fistula at scrotum during inspection

The patient was asked to stop antibiotics, antiinflammatory drugs and advised to take avagāha sveda (sitz bath) with pañcavalkala kaṣāya[4] along with internal administration of tablet Nimbādi Guggulu,[5] two BD for first 10 days. This was prescribed given the pitta kaphaja prakṛiti of the patient. On next visit, it was observed that the size of the indurated swelling was reduced significantly with increased discharge from external opening and patient was able to sit and ride vehicle with mild pain. Subsequently patient was advised for fistulography so that the dye can easily enter the tract and give complete picture of the internal opening so as to help plan further management. The antero-posterior [Figure 2] and lateral view of fistulogram[Figure 3] showed that the internal opening was at 12’O clock position with involvement of few fibers of external sphincters.

Figure 2

Fistulogram antero-posterior view

Figure 3

Fistulogram lateral view

The patient was posted for kṣārasūtra[6] ligation under spinal anesthesia confirming all the routine electrocardiogram, hematological, biochemical and urine investigations. The entire tract was meticulously probed to see internal opening. Both the external openings were connected to a single internal tract. The internal opening was dissected and widened to drain completely (cryptoglandular), and kṣārasūtra was tied [Figure 4]. Patient was kept on oral antibiotics for 3 days as prophylaxis. Subsequently the kṣārasūtra was changed twice at intervals of 7 days. Pañcavalkala avagāha was continued for 2 weeks after kṣārasūtra ligation. During this time, it was observed that the tract was patent, healthy with low discharge Kṣārasūtra in situ itself shows that the tract was patent and through history it was evaluated that the patient was able to sit and perform normal activities without pain, discharge (undergarment staining was very less) and induration around both external openings were reduced.

Figure 4

During ksharasutra ligation

The thread was removed to encourage the healing of the tract without expecting the tract to be cut through in its entirety as it would have taken more time and would have been painful. Next visit it was observed that the whole tracts had healed completely with minimal scar [Figure 5]. Later, the patient was followed-up for 1-year to observe for recurrence. No recurrence was observed post kṣārasūtra ligation.

Figure 5

After treatment

DISCUSSION

Ano-rectal sepsis can be complicated by anal fistula in about 25% of patients during the acute phase of sepsis or within 6 months thereafter. It is now a widely accepted fact that infection of the anal gland is the cause of the cryptoglandular infection that results in an abscess in the acute stage and in a fistula in its chronic stage. Pus from this abscess contains intestinal microorganisms. From the intersphincteric space, the infection can spread in three directions - downwards to the perineal space; laterally piercing the external sphincter to the ischiorectal fossa and upwards in the intersphincteric plane to the supralevator space. From the above-mentioned spaces, infection can spread anteriorly to scrotum/vulva and groin; posteriorly to pre- and post-sacral spaces and then, rarely, to the gluteal region and thigh, and superiorly to pre- and retroperitoneal spaces.

The patient presented with hard indurated swelling with two external openings which was due to injudicious use of antibiotics. To soften the induration and to drain the swelling, avagāha sveda was employed along with tablet Nimbādi Guggulu internally in the dose of 2 tablet BD, which possesses lekhana, śodhana properties. With this preliminary management the tract became clear and probing was possible to some extent.

Conventional surgeries like fistulectomy, fistulotomy were not done as the tract was lengthy and there were chances of injuring urethra, testis and anal sphincters hence the kṣārasūtra ligation were adopted. The subsequent changing of the kṣārasūtra was limited for 3 weeks considering the pain involved in changing the thread, and also lengthy time involved in cutting of the entire tract. Moreover, in anterior low anal fistulae there is no potential space (fossa) to form abscess cavity, hence such anal fistulae can be well managed by limited application of kṣārasūtra without requiring cutting of the entire tract. Simultaneously, it was ensured that the tract would be properly scraped and completely drained. Internally, tablet Nimbādi Guggulu was continued for a week with a dose of 2 tablets twice daily.

According to Goodsall's rule[7] the external opening situated behind the transverse anal line will open into the anal canal in the midline posteriorly. An anterior opening is usually associated with a radial tract. In this case the two external openings were on median raphe of the scrotum beyond 3 cm from the anus, which may have opened into the posterior midline of the anal canal. On the contrary, the tracts were direct in this case, which is a rare finding. The author has encountered such exceptions in his clinical practice where in anterior low anal fistula beyond 3 cm follow a simple direct course whereas high anal fistula beyond 3 cm tend to follow curved course and open posteriorly.

CONCLUSION

Bhagandara is one of the aṣṭamāhāgada considering its morbidity, recurrence and social burden by its nature. Hence meticulous planning of management is utmost essential. Nonjudicious usage of antibiotics may, in fact, worsen the condition. Depending on the stage of the disease, simple measures like avagāha sveda will help alleviate the pathology. The kṣārasūtra need not always be used to cut the tract completely but it may also be used to facilitate faster recovery. However, in other varieties of fistula this technique fails as there will be potential space for the development of abscess, which requires multiple replacement of kṣārasūtra to drain and heal the tract completely. Hence, this technique should be only adopted in anterior low anal fistula.

The classical intention of kṣārasūtra is to cut the entire tract and simultaneously heal. However, in the present case, the tract was more than 10 cm which requires multiple sittings of kṣārasūtra and may require more than 4-5 months for complete cutting of the tract and healing. Hence in this case, cutting of entire tract was not necessary due to above mentioned reasons. Once the unhealthy tract and tissue is cleared with the help of kṣāra, there is growth of healthy granular tissue, and kṣārasūtra can be removed and the tract can be left for secondary healing. Before leaving the tract for secondary healing, the surgeon should ensure that the pus collected is drained entirely and unhealthy tissue is removed.