Accepted Manuscript Retroperitoneal, transmuscular appendico-cutaneous fistula, as a hazard of incomplete appendicectomy Raashid Hamid, MBBS,MS,Mch, Senior Resident, Nisar Bhat, Professor PII:
S2213-5766(16)30110-5
DOI:
10.1016/j.epsc.2016.09.001
Reference:
EPSC 630
To appear in:
Journal of Pediatric Surgery Case Reports
Received Date: 2 June 2016 Revised Date:
5 September 2016
Accepted Date: 10 September 2016
Please cite this article as: Hamid R, Bhat N, Retroperitoneal, transmuscular appendico-cutaneous fistula, as a hazard of incomplete appendicectomy, Journal of Pediatric Surgery Case Reports (2016), doi: 10.1016/j.epsc.2016.09.001. This is a PDF file of an unedited manuscript that has been accepted for publication. As a service to our customers we are providing this early version of the manuscript. The manuscript will undergo copyediting, typesetting, and review of the resulting proof before it is published in its final form. Please note that during the production process errors may be discovered which could affect the content, and all legal disclaimers that apply to the journal pertain.
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Manuscript type ;Case Report Title ; Retroperitoneal, transmuscular appendico-cutaneous fistula, as a
Authors; Raashid Hamid , Nisar Bhat Name& Address for corresponding author ;
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hazard of incomplete appendicectomy
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Married doctors hostel room no= S2 A Block SKIMS SRINAGER Jammu and Kashmir
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Pin; 190011 E mail;
[email protected]
1.Raashid Hamid ; MBBS,MS,Mch, Senior Resident, Dept. of Paediatric and
Neonatal Surgery Skims Srinager Jammu and Kashmir INDIA. Phone no,-
9469451875 E-Mail;
[email protected]
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2 .Nisar Bhat; Professor , Dept. of Paediatric and Neonatal Surgery Skims Srinager Jammu and Kashmir INDIA
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E Mail;
[email protected]
ACCEPTED MANUSCRIPT Title; Retroperitoneal, transmuscular appendico-cutaneous fistula, as a hazard of
incomplete appendicectomy
Abstract;
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A male child 9 years of age presented with fever, tender swelling in the right flank, with a right paramedian scar. Child had undergone exploratory laparotomy for peritonitis with appendicectomy one year back. radiological Investigations (Ultrasonography and Computed Tomography) revealed retrocaecal abscess extending to the psoas muscle, which
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was drained via flank incision. Child continued to have an intermittently discharge through an opening in the flank just above the posterior superior iliac spine. Ultrasonograpghy, CT fistulogram revealed a fistulous communication from skin to the caecum / appendix. Upon
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second exploration a fistulous communication from the tip of residual appendix (which was left insitu at the initial exploration) was found with multiple adhesions in the terminal ileum, caecum, and ascending colon. Local resection of the caecum and ascending colon was performed with ileocolic anastomosis. Fistulous tract was curetted out. Exploration confirmed the incomplete appendicectomy as a cause of this hazardous condition.
Introduction;
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Key words; Enterocutaneous fistula, Appendicectomy, Curettage
Only few cases appendico-cutaneous fistulae have been reported in the English [1]
Fistula formation can occur after drainage of the appendix abscess, post
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literature.
appendicectomy or as a complication of perforated appendix.[2,3] Appendicectomy with the
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excision of the fistulous tract is considered optimal management of this condition.[4] We are describing a case of appendico-cutaneous fistula in the flank, following one year of drainage of a post appendicectomy retroperitoneal abscess extending to psoas muscle. Case report;
A 9 year old male student presented as a tertiary care hospital with 3 day history of pain right flank, vomiting and fever, difficulty in walking. Patient was having tachycardia and temperature of 101 0F. Per abdomen examination was soft, except a tender swelling in the right flank, with a right paramedian scar. His previous surgical record revealed appendicectomy with peritoneal mopping ,which was performed at some other tertiary care
ACCEPTED MANUSCRIPT hospital. Investigations were
normal
apart from raised WBC counts of 18000 with
neutrophill count of 80%. Ultrasonography revealed a 8x12 cm echogenic area as right psoas abscess. This abscess was drained through right flank incision. Culture of the pus drained demonstrated
Escherichia coli. Patient received
appropriate antibiotics and antiseptic
dressing. Two month after the abscess drainage patient developed an opening at the lateral
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aspect of the wound, superior to the iliac crest at posterior superior iliac spine. A discharging sinus was located in the right loin. Abdominal examination was normal. WBC count was normal. Tuberculosis, chron’s disease and osteomyelitis were ruled out. Patient was lost to follow-up over next one year. Again patient presented with intermittent drainage of pus
USG and CT fistulogram delineated
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through the same site. A sinogram delineated the tract between skin and caecum. (Figure; 1) a well developed fistulous tract between the skin
,through the psoas muscle to the colon. (Figure; 2)
Laparotomy was done through the
retroperitoneum. The
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previous incision (right paramedian).The colon and the terminal ileum were adherent to the retroperitoneal surface of ascending colon was inflamed due to
recurrent infection in this area . After mobilising the right colon, caecum, appendix was found intact with the lumen
at tip communicating with the fistulous tract, through psoas
muscle to the skin opening. (Figure; 3) (Figure; 4) , (Figure; 5) Whole small and large gut
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was normal except for inflammatory nodes near caecum and terminal ileum. Right colon was resected with ileo-colic anastomosis (single layer interrupted). Fistulous tract was curetted out in toto. (Figure; 6)
Discussion;
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epithelialized tract.
Histopathological examination of the fistulous tract showed a well
Fistulous of appendix with
viscera or skin is a very rare and difficult to manage
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complication of acute appendicitis. [4] Fistula may develop spontaneously as a complication of acute perforating appendicitis.[2,3]
In our case the cause of fistula was inadequate
appendicectomy which was performed one year prior. According to Muthukumarassamy,the main mechanism of the fistula formation is the rupture of appendix in the adjacent bowel or skin and persistence of fistula may be due to carcinoid tumor or tuberculosis of the appendix .[4] In our index case, both
the two conditions were ruled out and the persistence of the
pus discharge was due the epithelialisation of the tract. Fistulas initially manifest as a subcutaneous abscess, which
after rupture may persist as a discharging sinus. [4]
In our
case fistula initially manifested as a psoas abscess fistulating posterioly. All though rare, it
ACCEPTED MANUSCRIPT may be a hazard of incomplete appendicectomy. [6] Our case is second
in literature
were
incomplete appendicectomy was the cause of fistula Our case is unique, as the location of the cutaneous opening was very posterior in the loin. The cutaneous opening have been located in the buttock [1], right iliac fossa,[7] right groin
[8]
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and in the right flank. The effect of the fistula on our patient was least pronounced
except for nutritional
anaemia. As this being a low output fistula, the loss of fluids and electrolytes was minimal. Diagnosis in our case was confirmed by 3-D CT fistulogram. Simple fistulography can also
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be sufficient for the diagnosis .[9] Sometimes the fistulous tract may take a helical course as in our case, in such instances CT fistulography with 3-D reconstruction provides better delineation of the two ends of the fistula and the course of the tract, which latter facilitates be easy to diagnose even with the
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an optimal surgical procedure. At times it may not support of sophisticated diagnostic technique.
Recently interventional radiology has taken a prominent role in the management of these complex patients. As flexible endoscopic technology has improved and new endoscopic devices have been developed, endoscopists are expanding their role in the management of [10]
Endoscopically deployable stents, endoscopic suturing devices,
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gastrointestinal fistulae.
throughthe-scope and over-the-scope clips, sealants, fistula plugs, and vacuum sponges are among the technologies currently being attemped to treat fistulae.[11] Although attempts of edoscopicand laproscopic fistula closure has been
performed in adults, no literature could
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be found about the role of endoscopic attempts of fistula closure in children Laparotomy is the optimal management for the confirmation of this rare complication
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and the same time laparotomy permit to corroborate the exact site of the fistula. Laparotomy occasionally reveals the persistence of any residual stump of the appendix as in our case and permits the safe resection of the fistulous tract which in our case was through the psoas muscle. All though appendicectomy and excision of the caecum
tract or limited resection of
may be therapeutic in many instances, right colectomy with ileo- tranverse
anastomosis may at times be obligatory to cure this condition as in our case.
ACCEPTED MANUSCRIPT We recommend that the complicated appendicitis should be managed very carefully and meticulously in an attempt to avoid such hazardous consequence, which may result due to the persistence of the part or whole of the appendix in situ while managing a complicated appendicitis.
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Figures; Figure 1; Sinogram showing fistulous communication (Thin arrow- Fistula Tract and thick arrow- Dye in left colon) Figure 2; CT fistulogram showing the fistulous tract (thick arrows) and the communication with skin (thin arrows)
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Figure 5; Fistulous tract being curetted out posterioly
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Figure 3; Feeding tube through the fistula patient in lateral position
Figure 4; Fistulating tract site in the retroperitoneum (Black arrow) Figure 6; Colonic specimen with residual appendix
References;
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1. Hender J, Jansson R, Lindberg B. Appendico-cutaneous fistula. A case report. Acta Chir Scand 1978; 144:123-4.
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2. Deorah S, Seenu V, Pradeep KK, Sharma S. Spontaneous appendico-cutaneous fistula e a rare complication of acute appendicitis. Trop Gastroenterol 2005 ;26:48-50
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3. Brunagel G, Decker P, Hirner A. Delayed appendico-cutaneous fistulae a rare complication of simple abdominal drainage. Zentralbl Chir 1996; 121:67-9. 4. Muthukumarassamy R, Shankar R, Sistla S, Jagdish S. Appendico- cutaneous fistula. Indian J Surg 2006; 68:318-9. 5. Kjellman T Appendiceal fistulae and calculi. Review of literature and report of three cases Acta Chir Scand 1957; 113: 123 -9. 6. Hyett A. Appendico-cutaneous fistula: a hazard of incomplete appendicectomy. Aust N Z J Surg 1995 ;65:144-5
ACCEPTED MANUSCRIPT 7. Koak Y, Jeddy, T A and Giddings A E. Appendico-cutaneous fistula. J R Soc Med 1999; 92: 639 - 40.
8. Jacob, V and Mohan CVR. Spontaneous appendico-cutaneous fistula: A case report. Christian Med Coll Alumni J 2002; 36: 15 - 8.
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9. Genier F, Plattner V, Letessier E, Armstrong O, Heloury Y, LeNeel JC. Postappendicectomy fistula of cecum. Apropos of 22 cases. J Chir (Paris) 1995; 132:3938. 10. Kwon SH, Oh JH, Kim HJ, Park SJ, Park HC. Interventional management of gastrointestinal
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fistulas. Korean J Radiol. 2008;9(6):541-549.
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11. Kumar N , Larsen MC , Thompson CC Endoscopic Management of Gastrointestinal Fistulae. Gastroenterology & Hepatology 2014 ; 10: 495-502
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1. Only few cases appendico-cutaneous fistulae have been reported in the English literature. Fistula formation can occur after drainage of the appendix abscess, post appendicectomy or as a complication of perforated appendix. 2. In this case ,Ultrasonograpghy, CT fistulogram revealed a fistulous communication from skin to the caecum / appendix 3. A sinogram delineated the tract between skin and caecum. (Figure; 1) USG and CT fistulogram delineated a well developed fistulous tract between the skin ,through the psoas muscle to the colon. 4. Laparotomy was done through the previous incision (right paramedian).The colon and the terminal ileum were adherent to the retroperitoneum.
5. Whole small and large gut was normal except for inflammatory nodes near caecum
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and terminal ileum. Right colon was resected with ileo-colic anastomosis (single layer interrupted). Fistulous tract was curetted out in toto
