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 Table of Contents  
CASE REPORT
Year : 2018  |  Volume : 3  |  Issue : 3  |  Page : 54-56

Multiple pelvic osteomyelitis combined with adenocarcinoma: An extremely rare complication following intestinal fistula


Department of Orthopedics and Trauma, General Hospital of Xinjiang Military Region, Xinjiang, China

Date of Submission31-May-2018
Date of Acceptance23-Jul-2018
Date of Web Publication28-Sep-2018

Correspondence Address:
Dr. Shuzhang Guo
Department of Orthopedics and Trauma, General Hospital of Xinjiang Military Region, Xinjiang 830000
China
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ts.ts_7_18

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  Abstract 


Aim: Intestinal fistula is a common condition with numerous infective complications. Adenocarcinoma in the anal fistula is a rare tumor, and most of the known cases have been documented as case reports. Osteomyelitis of the pelvis is uncommon. Whereas, adenocarcinoma associated with osteomyelitis of the pelvis and anal fistula is a rare case in clinical practice. We report an unusual case of multiple pelvic osteomyelitides, combined with intestinal tumors as a late complication following intestinal fistula. Methods: A case report and review of the pertinent English language literature. Results: To the best of our knowledge, this is the fist report in the English literature to describe multiple pelvic osteomyelitides combined with intestinal tumors as a serious complication following intestinal fistula. The diagnosis was made with the aid of computed tomography imaging, sinus imaging, X-ray film, needle aspiration biopsy, tumor marker tests, surgery, and pathological section. The complication was treated successfully with surgery. Conclusion: Multiple pelvic osteomyelitides combined with adenocarcinoma and anal fistula was indeed a rare case in clinical practice. Heightened awareness and better understanding of this complication would necessitate the early diagnosis and treatment.

Keywords: Adenocarcinoma, fistula, pelvic osteomyelitis


How to cite this article:
Zhang Z, Ji M, Guo S. Multiple pelvic osteomyelitis combined with adenocarcinoma: An extremely rare complication following intestinal fistula. Transl Surg 2018;3:54-6

How to cite this URL:
Zhang Z, Ji M, Guo S. Multiple pelvic osteomyelitis combined with adenocarcinoma: An extremely rare complication following intestinal fistula. Transl Surg [serial online] 2018 [cited 2018 Dec 19];3:54-6. Available from: http://www.translsurg.com/text.asp?2018/3/3/54/242492




  Introduction Top


Pelvic osteomyelitis is a bacterial infection of the pelvic skeleton. Osteomyelitis of the pelvis is uncommon. It was first described 100 years ago. Its estimated incidence varies from 2% to 11% of all bone infections.[1] Apart from rare hematogen diffusion, mild trauma, and urinary tract infection as the possible causes, Crohn's disease has been associated with the occurrence of pelvic osteomyelitis.[2],[3] Due to the anatomic complexity and the complicated clinical picture of the pelvis, this infection can be neglected or underdiagnosed. Chronic perianal fistulas are a common clinical condition. However, their evolution into adenocarcinoma is rare.[4] Multiple pelvic osteomyelitides combined with adenocarcinoma and anal fistula are indeed a rare case in clinical practice. Here, we report such a case of adenocarcinoma and pelvic osteomyelitis that evolved from chronic perianal fistulas.


  Case Report Top


A 70-year-old male with a 50-year history of perianal fistula and right hip sinus who had been suffering from mucinous, pyoid secretion and pain for about 20 years was referred to our hospital for treatment. About 40 years back, he had been treated at a different hospital for the right hip mass with abscess formation, for which abscesses incision and drainage had been performed. About 1 year later, he noticed the recurrence of the right hip mass but did not seek treatment until July 2010, which is when he visited our hospital due to an increase in pain. His pain was described as a steady aching discomfort in the right pelvis and hip region exacerbated by standing, sitting, or walking. The pain increased gradually to a point where he could not ambulate. During the past 50 years, the patient complained of purulent secretion from the sinus tract. He also reported a weight loss of about 10 kg over the past 3-month period. The patient was admitted to our hospital with severe, disabling pain in the right hip, only for pain relief. He complained of hip pain, perineal induration, and the secretion of pus from sinus tract at the perineal area and the buttocks. No alteration in the bowel habit and no previous colorectal diseases were reported. The procedures were in accordance with the ethical standards of the responsible committee on human experimentation, and the patient consent form has been obtained.

Physically, the patient was very lean, and on palpation, marked tenderness of the right iliac crest, the anterior superior iliac spine, and the right hip was seen. Active movement of the right hip was stiff and painful. His routine blood test results were within the normal range except the C-reactive protein, which was 96 mg/L (normal value <10 mg/L). Tumor marker tests showed that the carcinoembryonic antigen was 19.02 KU/L (normal value <5 KU/L), CA19-9 was 196 KU/L (normal value <35 KU/L), and CA242 was 153.86 KU/L (normal value <20 KU/L). A plain X-ray film of the pelvis showed the extensive destruction of the pubis, ischium, ilium, and hip [Figure 1]. The opacification of sinuses showed that they communicated with the rectum [Figure 2]. The computed tomography scan of the pelvis [Figure 3] showed signs of inflammation and bone destruction of the right pelvis and soft-tissue induration at the site of the iliac fossa. A diagnosis of pelvic osteomyelitis was made. No intestinal lesion was seen on endoscopic examination. The sinus culture results obtained a few days later were positive for  Escherichia More Details coli, which were sensitive to amikacin and gentamycin. Preoperative histological examination of a biopsy specimen showed enterocyte. It was surprising that the skin of perisinus was not cancerous. After consultation with both the general surgery and urology departments, the general surgery team created a colostomy. One week after colostomy, we performed a local bony resection. Histological examination of the excised specimen confirmed poorly differentiated adenocarcinoma. Metastases to the mesenteric lymph nodes were not detected. Postoperatively, the patient developed a surgical site infection with E. coli. The patient was subsequently treated with surgery; incisional drainage was carried out. After an uneventful postoperative course, the patient was discharged from hospital on the 14th postoperative day. The orificium fistulae were not removed at discharge.
Figure 1: Plain anterior-posterior pelvis radiograph showing right hip bony ankylosis, bony changes in the ischium, ilium, and pubis

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Figure 2: The opacification of sinuses showing the sinuses communicating with the rectum

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Figure 3: Computed tomography scan of the pelvis showed signs of inflammation and bone destruction at the right pelvis and the soft-tissue induration at the site of iliac fossa

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  Discussion Top


Osteomyelitis is a progressive infection that results in inflaammatory destruction, necrosis, and bone neoformation, which can progress to a chronic and persistent stage.[5] There are two major osteomyelitis classification systems, one based on the duration of illness (acute versus chronic) and the other based on the mechanism of infection (hematogenous versus exogenous spread).[6] The rarity of the condition and the way in which its clinical signs imitate other diseases make it an intriguing diagnostic challenge.[7] The pelvic bones are deeply placed, and their movements are minimal, missing the usual signs of bone infection such as tenderness to palpation and limited motion. Further problems are fistulas and abscesses. With the involvement of symphysis or sacroiliac joint, it is possible to develop septic pelvic instability [Figure 4].
Figure 4: In this case, the relationship between pelvic osteomyelitis and adenocarcinoma

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Primary adenocarcinomas in the anal canal are rare tumors, representing only 3%–11% of all perianal carcinomas.[8] Majority of them grow extraluminally and in some cases, are not exposed to the mucosal surface of the anal canal or rectum. There have been many discussions on the origin of these tumors. Although etiology is controversial, mucinous adenocarcinoma may originate from chronic perianal fistulas, abscesses, anal glands, or malformations such as intestinal duplications.[9],[10],[11]

We speculated the cause of osteomyelitis and adenocarcinoma in our patient, which is depicted below:

To the best of our knowledge, our case report is the first in the English literature reporting a rare complication associated with the intestinal fistula, osteomyelitis of pelvis, adenocarcinoma, and sinuses as a late infective complication of intestinal fistula. We emphasize that although osteomyelitis of pelvis, adenocarcinoma, and sinuses are a rare complication of intestinal fistula, they require prompt diagnosis and treatment.[12] Despite all the advanced imaging techniques available, diagnosis is still established through meticulous physical examination of the patient and his medical history. To conclude, this case report aims to generate awareness and understanding of this rare complication. At the same time, we want to call doctors attention that in case of long-standing osteomyelitis or fistula, cancerization should be suspected.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Beaupré A, Carroll N. The three syndromes of iliac osteomyelitis in children. J Bone Joint Surg Am 1979;61 (7):1087-92.  Back to cited text no. 1
    
2.
Armstrong N, Schurr M, Helgerson R, Harms B. Fungal sacral osteomyelitis as the initial presentation of Crohn's disease of the small bowel, report of a case. Dis Colon Rectum 1998;41 (12):1581-4.  Back to cited text no. 2
    
3.
Chandler JT, Riddle CD Jr. Osteomyelitis associated with Crohn's disease. A case report and literature review. Orthopedics 1989;12 (2):285-8.  Back to cited text no. 3
    
4.
Leal RF, Ayrizono ML, Coy CS, Fagundes JJ, Góes JR. Mucinous adenocarcinoma derived from chronic perianal fistulas: Report of a case and review of the literature. Tech Coloproctol 2007;11 (2):155-7.  Back to cited text no. 4
    
5.
Smith IM, Austin OM, Batchelor AG. The treatment of chronic osteomyelitis: A 10 year audit. J Plast Reconstr Aesthet Surg 2006;59 (1):11-5.  Back to cited text no. 5
    
6.
Lew DP, Waldvogel FA. Osteomyelitis. N Engl J Med 1997;336 (14):999-1007.  Back to cited text no. 6
    
7.
Hughes CD, Axt MW. Life-threatening pelvic osteomyelitis: Pelvic destruction in an 8-year-old boy. Med J Aust 2009;190 (12):709-10.  Back to cited text no. 7
    
8.
Patrinou V, Petrochilos J, Batistatou A, Oneniadum A, Venetsanou-Petrochilou C. Mucinous adenocarcinoma arising in chronic perianal fistulas. J Clin Gastroenterol 2001;33 (2):175-6.  Back to cited text no. 8
    
9.
Shinohara T, Hara H, Kato Y, Asano M, Nakazawa Y, Kato T, Nogaki T, Yamashita Y. Implantation of rectal cancer cells in a fistula in Ano: Report of a case. Surg Today 2001;31 (12):1094-6.  Back to cited text no. 9
    
10.
Venclauskas L, Saladzinskas Z, Tamelis A, Pranys D, Pavalkis D. Mucinous adenocarcinoma arising in an anorectal fistula. Medicina (Kaunas) 2009;45 (4):287-90.  Back to cited text no. 10
    
11.
Winkler R, Wittmer A, Heusermann U. Cancer and Crohn's disease. Z Gastroentero l 2002;40 (8):569-76.  Back to cited text no. 11
    
12.
Wong NA, Shirazi T, Hamer-Hodges DW, Corfield AP, Lessells AM. Adenocarcinoma arising within a Crohn's-related anorectal fistula: A form of anal gland carcinoma? Histopathology 2002;40 (3):302-4.  Back to cited text no. 12
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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