Spontaneous reno-jejuno-cutaneous fistula with stone: An unusual presentation of genito-urinary tuberculosis

Abstract

We are reporting an unusual case of spontaneous reno-jejuno-cuteneous fistula with staghorn calculus. A 19-year-old female presented with pus discharging sinus in the left lumbar and gluteal region. Fistulography and computed tomography showed nonfunctioning kidney and fistula involving left kidney and jejunal loops. The patient was successfully treated by nephrectomy and excision of fistulous tract followed by antituberculous therapy. Histopathology revealed features of renal tuberculosis. To the best of our knowledge our case is the first case reported having reno-jejunal fistula because of tuberculosis and having cuteneous extension in the lumbar as well as gluteal region.

Keywords: Reno-alimentary fistula, renocutaneous fistula, tubercular fistula

How to cite this article:
Tiwari P, Kumar S, Pandey P, Pratap J, Sharma P. Spontaneous reno-jejuno-cutaneous fistula with stone: An unusual presentation of genito-urinary tuberculosis. Ann Trop Med Public Health 2012;5:253-5

 

How to cite this URL:
Tiwari P, Kumar S, Pandey P, Pratap J, Sharma P. Spontaneous reno-jejuno-cutaneous fistula with stone: An unusual presentation of genito-urinary tuberculosis. Ann Trop Med Public Health [serial online] 2012 [cited 2020 Aug 5];5:253-5. Available from: https://www.atmph.org/text.asp?2012/5/3/253/98631

 

Introduction

Normally, there is no communication between urinary and alimentary systems. However, communication may develop because of various reasons. It may be spontaneous or traumatic and accordingly management differs. Renal tuberculosis is still the principal cause of these spontaneous reno-alimantary fistulas in a developing country. Reno-jejunal fistula is rarest among them. To the best of our knowledge, only three cases have been reported in the world literature. Our case is the first case reported having reno-jejunal fistula because of tuberculosis and having cuteneous extension in the lumbar as well as gluteal region. [1]

Case Report

A 19-year-old female was treated for paraspinal ab­scess on the left side by incision and drainage 8 years back. Since then, she had a persistent intermittent discharging sinus in the left lumbar region. She also developed pus-discharging sinus on the left gluteal region since the last 1 year. She has history of weight loss, anorexia, and evening rise in temperature. Clinically, this seemed to be tuberculous sinus, due to osteomyelitis of the spine considering the high prevalence in this country. Accordingly, radiograph of the dorsolumbar spine and thorax was done [Figure 1]a. They revealed radio-opacity, suggestive of a staghorn cal­culus in the left kidney. There was no bony involvement. Her urine for AFB and culture for mycobacterium was negative. Culture of the draining sinus pus was negative by the BACTAC culture method. AFB of the pus was also negative. Ultrasonography whole abdomen revealed small contracted, and echogenic left kidney with loss of corticomedullary differentiation along with calcification. A sinogram showed bizarre outline of sinus, which communicated with left renal pelvis and jejunal loops [Figure 1]b and c. Intravenous pyelography showed a nonfunction­ing kidney on the left side. A CECT scan with fistulography revealed a small shrunken and scarred left kidney with a staghorn calculus and surrounding fibrosis. On opacifying the tract, the dye was directly entering from the skin to renal pelvis and to the small bowel [Figure 1]d. The fistula was successfully managed by single-stage surgery. The patient was explored through left subcostal incision. Renojejunal fistula disconnected from the bowel loops. Nephrectomy with excision of fistulous tract carried out. Continuity of jejunum restored after resection of diseased segment. Her postoperative recovery was uneventful. Histopathology of specimen was consistent with tuberculosis [Figure 2]. Postoperatively we kept her on antituberculosis therapy with Isoniazid, rifampcin, ethambutal, and pyrizinmaide and streptomycin for initial 2 months followed by isoniazid and rifampcin for another six 4 months to 1 year follow-up did not reveal any evidence of recurrence.

Figure 1: (a) Sinogram – radiograph of the dorsolumbar spine and thorax-radio-opacity, suggestive of a staghorn calculus in the left kidney. (b) Sinogram (left lumbar sinus) – sinus communicating with left renal pelvis and bowel loops. (c) Sinogram (left gluteal region) – across midline to reach opposite side along with multiple branching. (d) CECT with fistulogram-dye was seen to be directly entering from the skin to renal pelvis to the small bowel

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Figure 2: (a) Histopath – gross specimen with cut surface: ureteric catheter in fistuous tract, (b) Histology of nephrectomy specimen – high magnification shows collection of epitheliod cells and Langhans type of giant cells. H and E, ×40, (c) Histology – low-power view showing areas of necrosis with fibrous tissue and well-formed epithelioid granuloma. H and E, ×10

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Discussion

Spontaneous renal fistulization to neighboring viscera is not uncommon but spontaneous fistulization to jejunum is rare and its cuteneous extension is extremely rare. Arthur and Morris [2] provide a good review of the literature on the types and frequency of these spontaneously occurring fistulas. Reno-colic is the most common variety followed by Reno-duodenal, while reno-gastric and reno-jejunal fistulas are rarer. The jejunum is away from the left renal hilum and the interposition of the peritoneum may explain the rarity of this type of lesion. Cuteneous extension of these fistulas is reported in 10% of cases. [3] The majority of such fistulas present as spontaneous drainage through the lumbar region. Other unusual sites of spontaneous reno-cutaneous fistula are groins, gluteal, and even knee. [4] In almost all cases, the disease leading to reno-enteric fistula formation is originated from kidney. In a developing country TB is the principal cause of these kinds of fistula. The clinical features related to renal tuberculosis are variable and range from simple fatigue, evening rise in temperature, and anorexia and weight loss to attacks of loin pain and hematuria. Other features depend upon the stage of disease and extent of involvement. The presence of reno-cutaneous fistula overshadows other symptoms and draws most of patient/doctor’s attention. Tuberculous spondylitis is an important differential diagnosis and should be excluded by MRI.

Recurrent acute attacks of infection due to calculus obstruction of the kidney lead to adhesion formation between the kidney and surrounding viscera. The pyonephrosis leads to perinephric abscess, which ultimately ruptures and the pus may discharge along the path of least resistance forming a reno-cutaneous fistula, reno-enteric fistula, etc. Once fistulas have developed, little renal function is preserved, which indicates that nephrectomy should be done [5] in our case, this resulting in reno-jejunal and reno-cuteneous fistula in the left lumbar and left gluteal region.

The presence of communication between the kidney and the bowel is important to document before any operative intervention. Many imaging methods are available, but reno-jejuno-cutaneous fistula CT in conjunction with fistulography is the best method to establish diagnosis. [5],[6] There are only three previous such case reports of spontaneously occurring renojejunal fistula. [1] In all these cases there is no cuteneous extension as in our case.

We concede that in the new era of prompt multidrug tuber­cular chemotherapy has significantly retarded the inci­dence of spontaneous reno-enteric fistula. However, renal tuberculosis being endemic in our coun­try, we cannot dismiss altogether an insidious develop­ment of such fistulas in nonfunctioning retained kidneys.

The standard treatment of spontaneous reno-enteric-cutaneous fistula is nephroureterectomy with complete debridement of affected tissue and excision of fistulous tract. The surgery is followed by anti-TB regimen and long-term follow-up.

References

 

1. Evans PF. Spntaneous pyelo-jejunal fistula. Postgrad Med J 1990;66:965-7.
2. Arthur GW, Morris DG. Reno-alimentary fistulae. Br J Surg 1966;53:396-402.
3. Terries MK, Collins MA, Shah SR. Renoalimentary fistula. e-Medicine Jan, 2012.
4. Hanchanale VS, Rao AR, Motiwala HG. Renogluteal fistula: An unusal complication of genito-urinary tuberculosis. Indian J Urol 2006;22:270-1.
5. Wani NA, Zargar HU, Akhtar MA, Khan M. Cutaneonephrocolic fistula. Int Surg 1977;62:545-6.
6. Cohen EL, Greenstein AJ, Katz SE. Nephrocolo cutaneous fistula: Use of CT scan to aid diagnosis. Comput Radiol 1983;7:291-4.

Source of Support: None, Conflict of Interest: None

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DOI: 10.4103/1755-6783.98631

Figures

[Figure 1], [Figure 2]

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