Acta chir belg, 2004, 104, 736-738
The Case of Acute Appendicitis and Appendiceal Duplication H. Yanar*, C. Ertekin*, E. S. Unal*, K. Taviloglu*, R. Guloglu*, O. Mete** Department of General Surgery*, Department of Pathology**, Emergency Surgery Unit, Istanbul Medical Faculty, Istanbul University.
Key words. Appendix vermiform ; duplication ; acute abdomen. Abstract. Background : Appendiceal anomalies are extremely rare malformations that are usually found in the adult population as an incidental finding during laparotomy performed for other reasons. Abnormal development of the appendix usually takes the form of a double appendix. Accompanying intestinal, genito-urinary or vertebral malformations may be present when appendiceal duplications are detected in childhood. Case Report : Presented herein is a case of perforated double appendix, which causes acute abdomen in a child, without any co-existing pathology. Conclusion : Appendiceal anomalies are of great practical importance and a surgeon must bear them in mind during an operation. If he overlooks them, the patient undergoing surgery may experience grave consequences. They also may be a forensic issue in cases when a second explorative laparotomy reveals ‘previously removed’ vermiform appendix.
Introduction Duplication of the vermiform appendix is rare, with a reported incidence of 0.004%. Less than 100 appendiceal anomalies have been reported in the literature (1, 2, 3, 4, 5). Most anomalies of the appendix have been observed in adults and most were noticed incidentally during surgery not primarily involving the appendix (1). Duplication of the vermiform appendix causing small bowel obstruction (2), mimicking adenocarcinoma of the colon (6), hypotrophic and duplicated appendix (9) and unusual duplication of appendix and cecum (7) have also been reported. Appendiceal duplication have with colonic duplication and genito-urinary abnormalities (3), or with gastroschisis (5) can exhibit life-threatening conditions. Case Report A 15-year-old boy was admitted on an emergency basis with a 3-day colicky abdominal pain, loss of appetite, nausea, and bile-stained vomiting. His bowel habits were not disturbed and he continued to pass flatus normally. There was localized tenderness at McBurney’s point, moderate rebound tenderness, guarding, positive psoas signs and increased pain with coughing. The rectal examination showed normal soft stool without tenderness. The blood count showed leukocytosis (WBC : 14200/mm3) with a shift to the left. Plain abdominal radiographs were normal. Ultrasonographic examination was conclusive for acute appendicitis (a dilated, noncompressible, thickened-walled appendix vermiformis).
Fig. 1 Intraoperative appearance of the duplication
Laparotomy was performed via McBurney’s incision. The operative findings were a mass surrounded by omentum and purulent intra-abdominal fluid. After the omentum was dissected, a 10 5 cm perforated appendix was observed and an intact second appendix 4 cm in length (Fig. 1). Both appendices opened into the caecum with a single base. There were no other noteworthy surgical findings and the remaining abdominal viscera were normal. A formal appendectomy was performed.
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Fig. 2 Histological analysis confirms the typical appearance of appendiceal wall with lymphoid follicles and smooth muscle (HE-staining, 10).
Histopathological examination revealed lymphoid follicles and smooth muscle, which resembled the wall of the vermiform appendix (Fig. 2). Thus, our macroscopic diagnosis of appendiceal duplication was confirmed by histological examination. The patient was discharged after an uneventful postoperative period. Discussion Appendiceal duplications were classified by WALLBRIDGE (11) and WAUGH (12). Wallbridge’s 1962 classification divides these duplications into three groups (11) (Fig. 3). Type A consists of various degrees of partial duplication on a normally localized appendix with a single caecum. Type B includes a single caecum with two completely separated appendices. This type has two subgroups. In the B1 group, there are two appendices localized symetrically on either side of the ileo-cecal valve ; this resembles the normal phylogenet-
ical arrangement in birds, so this group was called the ‘bird-like or avian’ type. B2 (taenia-coli type) has a normally localized appendix arising from the caecum at the usual site and a second, separate, rudimentary appendix localized along the taenia line. In type C ; there is a double cecum, each having its own appendix. In Wallbridge’s original paper, this classification was based on the reported cases of which there were less than 50 at that time. Because of the difficulty of categorizing some cases into a suitable type, the authors started to add additional types. Type D is a horse-shoe appendix with two openings at the common cecum (10). The case presented in this paper could be classified as type A. COLLINS reported only two cases of duplication in 50,000 autopsies (13). Accompanying duplications that affect the large intestine and genito-urinary tract may be explained by the close anatomic association of the distal hindgut and the urogenital septum in the embryologic origin, but the precise mechanism is still unknown (3). These anomalies are mostly associated with types B1 and C duplications. There is not yet enough knowledge about the reason for this relationship (1). In the literature, the cases reported as type-A were never accompanied by associated anomalies. Duplication of the appendix must be distinguished from solitary diverticulum of the cecum, and from appendiceal diverticulosis. This distinction can be best made histopathologically (1, 11). Besides duplication and diverticulosis, the horse-shoe and triple appendix anomalies should be considered in the differential diagnosis (1, 10, 11, 14). All these anomalies are of great practical importance and a surgeon must bear them in mind during an operation. If he overlooks them, the operated patient may experience serious consequences, which may be of legal importance in cases where repeated exploratory laparotomy reveals a ‘previously removed’ vermiform appendix (8, 10). We also believe that junior surgical staff must be aware of these conditions due to the medicolegal aspects.
Fig. 3 Wallbridge-Waugh Classification of Appendiceal Duplication
738 References 1. EROGLU E., ERDOGAN E., GUNDOGDU G., DERVISOGLU S., YEKER D. Duplication of appendix vermiformis : a case in a child. Tech Coloproctol, 2002, 6 : 55-7. 2. CHEW D. K. W., BORROMEO J. R., GABRIEL Y. A., HOLGERSEN L. O. Duplication of the vermiform appendix. J Pediatr Surg, 2000, 35 : 617-18. 3. MCNEILL S. A., RANCE C. H., STEWART R. J. Fecolith impaction in a duplex vermiform appendix : An unusual presentation of colonic duplication. J Pediatr Surg, 1996, 31 : 1435-37. 4. BIERMANN R., BORSKY D., GOGORA M. Die Appendicitis duplexeine pathologische Raritat. Chirurg, 1993, 64 : 1059-61. 5. GILCHRIST F. B., SCRIVEN R., NGUYEN M., NGUYEN V., KLOTZ D., RAMENOFSKY M. L. Duplication of the vermiform appendix in gastroschisis. J Am Coll Surg, 1999, 189 : 426. 6. BLUETT M. K., HALTER S. A., SALHANY K. E., O’LEARLY J. P. Duplication of the appendix mimicking adenocarcinoma of the colon. Arch Surg, 1987, 122 : 817-20. 7. KIM E. P., MCCLENATHAN J. H. Unusual duplication of appendix and cecum : extension of the Cave-Wallbridge classification. J Pediatr Surg, 2001, 36 : E18. 8. MITCHELL I. C., NICHOLLS J. C. Duplication of the vermiform appendix. Report of a case : review of the classification and medicolegal aspects. Med Sci Law, 1990, 30 : 124-6.
H. Yanar et al. 9. DE LAGAUSIE P., BILLING A., EYMERI J. C., TAVAKOLI D. Hypotrophic and duplicated appendix. A case in a child. Chir Pediatr, 1989, 30 : 216-7. 10. DRINO E., RADNIC D., KOTJELNIKOV B., AKSAMIJA G. Rare anomalies in the development of the appendix. Acta Chir Iugosl, 1991, 38 : 103-11. 11. WALLBRIDGE P. H. Double appendix. Br J Surg, 1963, 50 : 346. 12. WAUGH T. R. Appendix vermiformis duplex. Arch Surg, 1941, 42 : 311-20. 13. COLLINS D. C. A study of 50000 specimens of the human vermiform appendix. Surg Gynecol Obstet, 1955, 101 : 437-46. 14. MESKO T. W., LUGO R., BREITHOITZ T. Horseshoe anomaly of the appendix : a previously undescribed entity. Surgery, 1989, 106 : 563-66.
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