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    Introduction

    Appendicitis is a disease frequently encountered insurgical practice. Complications such as perforation,abscess formation and peritonitis are not uncommon.Most of the time, diagnosis and treatment are straight-forward. On the other hand, a delayed diagnosis can lead

    to rare, but sometimes serious, complications. Wepresent a rare but fatal case of retroperitoneal perforatedappendicitis with subsequent extensive retroperitonealabscess formation and subcutaneous emphysemaresulting in severe sepsis, multiple organ failure anddeath.

    Case report

    A 76-year-old man presented at our emergency depart-ment with progressive abdominal pain and loss ofappetite that had persisted for five days. The patients

    medical history showed non-insulin dependent diabetesmellitus and hypertension. Physical examination showeda slightly ill-looking man with a temperature of 37.5C,a blood pressure of 90/45 mmHg and a pulse rate of117/min. The abdomen was adipose and tender in thelower half and right flank without clinical signs of peri-tonitis. Abnormal laboratory data included leukocytosis(30.3 109/L), anaemia (Hb 6.6 mmol/L) and disturbedrenal function (creatinine of 289 mol/L and urea of23.3 mmol/L). Urinalysis showed 2-5 leucocytes perfield, 2-5 erythrocytes per field and no bacteria. Ultra-sound examination of the abdomen showed a stone in theright pyelum, without signs of obstruction. On suspicionof suffering from urosepsis, the patient was treated with

    intravenous antibiotics, followed by a quick recoverywith disappearance of leucocytosis. After four days hewas discharged with oral antibiotics.

    Five days later the patient returned to our hospitalbecause of persistent abdominal pain, mainly on the rightside. His blood pressure was 85/45 mmHg ; pulse andbody temperature were normal. On physical examination

    a red and tender mass was palpable in the right upperabdominal quadrant. Laboratory tests showed leukocyto-sis (22 109/L), anaemia (6.5 mmol/L), thrombocytosis(631 109/L), progressively disturbed renal function(creatinine of 408 mol/L and urea of 49 mmol/L) andincreased C-reactive protein levels (326 mg/L). Ultra-sonography demonstrated an abscess in the right abdom-inal wall with culture of a subsequent puncture showingcandida glabrata, candida tropicalis, gram-positive floraand anaerobe flora. Subsequently, the patient was treatedwith intravenous fluconazole, ciprofloxacin, amoxicillinand metronidazole. With abdominal computed tomogra-

    phy (CT) abscesses in the retroperitoneum and subcuta-neous emphysema at the right side and in the rightabdominal wall were seen, extending into the leftabdominal wall down to the pelvis (Fig. 1). Emphysemain the left perirenal space with stones in the left kidneyand bladder were also seen. Additional workup with CT-IVP (intravenous pyelogram) and contrast x-ray of thecolon (for colonic perforation) showed no abnormalities.At this moment a urologic origin seemed the most likelycause. The patient was clinically stable and repeated lab-oratory tests did show some improvement (leucocytes of10.1 109/L, C-reactive protein levels of 304 mg/L, cre-atinine of 168 mol/L and urea of 31.5 mmol/L) duringconservative treatment with intravenous antibiotics.

    Acta Chir Belg, 2008, 108, 457-459

    Retroperitoneal Abscess and Extensive Subcutaneous Emphysema in Perforated

    Appendicitis : a Case Report

    N. B. Tomasoa*, J. M. Ultee, B. C. VrouenraetsDepartment of Surgery, Sint Lucas Andreas Hospital, Amsterdam, the Netherlands.*At present : Department of Surgery, Onze Lieve Vrouwe Gasthuis, Amsterdam, the Netherlands.

    Key words. Perforated appendicitis ; retroperitoneal abscess ; subcutaneous emphysema.

    Abstract. Most of the time, the diagnosis and treatment of appendicitis are straightforward. However, a missed diagno-sis can sometimes lead to life-threatening complications. A fatal case of appendicitis in a 76-year-old man who present-ed with progressive abdominal pain, retroperitoneal abscesses and extensive subcutaneous emphysema, is described.

    Eventually, laparotomy showed appendicitis perforated into the retroperitoneum without any signs of peritonitis. Despitemultiple operations the patient died two months after admission due to multiple organ failure.

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    However, on day nine, surgical drainage of retro- andpreperitoneal abscesses took place. At laparotomy,exploration showed an appendicitis perforated into theretroperitoneum without any signs of peritonitis.Appendectomy was performed and several drains wereleft in the abscesses. Post-operatively, the patients con-dition deteriorated and despite multiple operations forpersistent abscess formation and sepsis, he died due tomultiple organ failure two months after admission.

    Discussion

    Worldwide, acute appendicitis is a disease commonlyencountered in everyday practice, with a lifetime risk ofapproximately 7% (1). Proper diagnosis and treatmentare essential to prevent morbidity and mortality (2, 3).Occasionally, serious and sometimes life-threateningcomplications of perforated appendicitis do occur. Fourother cases of retroperitoneal perforated appendicitiswith formation of retroperitoneal abscesses and subcuta-

    neous emphysema have been described (4-7). Amongthese, several similarities are described : the patientspresentation is unusual, a retroperitoneal ruptured appen-dicitis can cause serious complications and CT is thediagnostic tool of choice.

    Although a frequently encountered disease, the diag-nosis of appendicitis may be missed. Because of its posi-tion, a retroperitoneal perforated appendicitis can causeatypical and confusing physical findings (5). Infectionand air can extend to communicating compartments,resulting in emphysema and abscesses in unexpectedanatomical sites. Other cases have been described with

    extension to the abdominal wall, thigh, and perinephricspace (4-7).

    458 N. B. Tomasoa et al.

    Since the location of infection in our patient wasretroperitoneal, typical peritoneal signs were absent andtreatment for a presumed urosepsis was installed. Promptsurgery and source control, especially during the first

    admission, would most likely have altered the unfortu-nate outcome of our patient. An early appendectomycould have prevented this extensive spreading of appen-dicitis.

    For a definite diagnosis of acute appendicitis, CT scanof the abdomen is considered to be the imaging studywith the highest accuracy and efficiency (8-10). Not onlycan it be of great help in diagnosis, but also in evaluatingthe extension of involvement. Furthermore, an approachfor drainage of abscesses can be made on CT results.Only on CT scanning, emphysema and abscessing of theright flank were seen. As described by ISHIGAMI et al. the

    superior and inferior lumbar triangles, two sites ofanatomical weakness in the flank abdominal wall, allowspreading to the abdominal wall (7). Nonetheless, in ourcase, CT findings were not sufficient for the diagnosisand the cause of abscess and emphysema formation wasunclear until laparotomy.

    In summary, a case of a retroperitoneal perforatedacute appendicitis causing formation of retro- andpreperitoneal abscesses with extensive subcutaneousemphysema was presented : a rare but life-threateningcomplication. Physicians should have in mind that occa-sionally disease extension to unexpected anatomical sitesdoes occur, causing unusual clinical pictures. CT scans

    of the abdomen should be made freely. However, some-times an exploratory laparotomy is necessary to revealthe cause. This case re-emphasizes the importance ofearly management for such a common disease.

    References

    1. GRLEYIK G., GRLEYIK E. Age-related clinical features inolder patients with acute appendicitis.Eur J Emerg Med, 2003,10 : 200-3.

    2. BLOMQVIST P. G., ANDERSSON R. E., GRANATH F., LAMBE M. P.,EKBOM A. R. Mortality after appendectomy in Sweden, 1987-1996.Ann Surg, 2001, 233 : 455-460.

    3. HALE D. A., MOLLOY M., PEARL R. H., SCHUTT D. C., JAQUES D. P.Appendectomy : a contemporary appraisal.Ann Surg, 1997, 225 :252-261.

    4. HSIEH C. H., WANG Y. C., YANG H. R., CHUNG P. K., JENG L. B.,CHEN R. J. Extensive retroperitoneal and right thigh abscess in apatient with ruptured retrocaecal appendicitis ; An extremely ful-minant form of a common disease. World J Gastro-enterol, 2006,12 : 496-499.

    5. KAO C. T.,TSAI J. D., LEE H. C., WANG N. L., SHIH S. L., LIN C. C.,HUANG F. Y. Right perinephric abscess : a rare presentation ofruptured retrocaecal appendicitis. Pediatric Nephrol, 2002, 17 :177-180.

    6. USHIYAMA T., NAKAJIMA R., MAEDA T., KAWASAKI T., MATSUSUE Y.Perforated appendicitis causing thigh emphysema : A case report.J Orthop Surg, 2005, 13 : 93-95.

    7. ISHIGAMI K., KHANNA G., SAMUEL I., DAHMOUSH L., SATO Y. Gas-forming abdominal wall abscess : an unusual manifestation of per-

    forated retroperitoneal appendicitis extending through the superiorlumbar triangle.Emerg Radiol, 2004, 10 : 207-209.

    Fig. 1

    CT abdomen showing extensive subcutaneous emphysema andabscess formation in the abdominal wall and left perirenalspace.

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    Retroperitoneal Perforated Appendicitis 459

    8. EDWARDS J. D , ECKHAUSER FE. Retroperitoneal perforation of theappendix presenting as subcutaneous emphysema of the thigh. DisColon Rectum, 1986, 29, 456-458.

    9. GUTKNECHT D. R. Retroperitoneal abscess presenting as emphyse-ma of the thigh.J Clin Gastro-enterol, 1997, 25 : 685-687.

    10. SHARMA

    S. B., GUPTA

    V., SHARMA

    S. C. Acute appendicitis present-ing as thigh abscess in a child : a case report. Pediatr Surg Int,2005, 21 : 298-300.

    Dr. B. C. Vrouenraets, surgeonDepartment of Surgery, Sint Lucas Andreas HospitalPostbus 92431006 AE AmsterdamTel. : +31 20 5108770

    Fax : +31 20 6838771E-mail : [email protected]