Vol 44 # 4 December 2012 - Kma.org.kw

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329 KUWAIT MEDICAL JOURNAL December 2012 Case Report Double Appendix: Report of a Case Naorem Gopendro Singh 1 , Hisham Kantoush 2 , Mirza Kahvic 1 1 Department of Pathology and 2 Surgery, Al-Jahra Hospital, Kuwait Kuwait Medical Journal 2012; 44 (4): 329 - 331 ABSTRACT A 30-year-old female presented to the surgery outpatient department with complaint of pain in right iliac fossa of one day duration. The pain was associated with nausea and vomiting. On examination, there was localized tenderness and rigidity in the right iliac fossa with positive rebound tenderness. Multiple pigmentation of skin in the abdomen, accessory nipple and scoliosis were also noted. Laboratory investigation revealed neutrophilic leukocytosis. Ultrasound abdomen showed an appendix measuring 4 cm in length and a fibroid uterus. Diagnostic laparoscopy revealed an inflamed appendix with a small blind structure in continuity with the cecum. Operative diagnosis of double appendix was made. Microscopic examination confirmed double appendix with features of appendicitis and periappendicitis in the bigger appendix. We report this case because of it rarity. The surgical resident should be aware of this rare congenital anomaly to avoid missing the other appendix in appendectomy procedures and avoid its medico-legal consequences. KEY WORDS: appendicitis, congenital anomaly, double appendix INTRODUCTION Despite the notorious range of variation in character and position natural to the human vermiform appendix, the extremes of variation (absence or duplication) affect this organ so very rarely that they are worth mentioning and recording. Duplication of vermiform appendix is rare with reported incidence of 0.004% [1] and may be associated with other congenital duplications or other anomalies [2] . Doubled appendix is usually asymptomatic. When symptomatic, they are usually the result of obstruction and inflammation. Majority of the cases are diagnosed at surgery or on postmortem examination. Some of them can be picked up preoperatively on barium enema. The clinical presentation can vary according to the location of the appendices [3] . Here we report a case of double appendix because of its extreme rarity and discuss its etiology and different types of anatomic presentation. CASE REPORT A 30-year-old female patient presented with right lower abdominal pain of one day duration with associated nausea and vomiting. She gave no history of change in bowel habits, urinary symptoms, menstrual disturbance or similar previous attacks. On examination she was afebrile. There was localized tenderness and rigidity with positive rebound tenderness in right iliac fossa. Multiple pigmentation of skin especially in the abdomen, accessory nipple (polythelia) and scoliosis were noted. No mass could be felt. Her pelvic examination did not reveal any positive findings. Laboratory investigations revealed a total white blood count of 24.8 x 10 9 cells per liter with neutrophilia (72%). Other laboratory investigations including blood urea, creatinine and liver function test were unremarkable. Ultrasound abdomen showed an appendix measuring 4 cm in length and a fibroid uterus with minimal localized collection of fluid, in the right iliac fossa. With a presumptive diagnosis of acute appendicitis, diagnostic laparoscopy was performed on the day of admission with insufflation of CO 2 gas and insertion of three trocars, two 10 mm ports and one 5 mm port. There was minimal fluid in the pelvis with an inflamed appendix, which could be easily identified. No evidence of perforation or mass formation was noted. However, another blind structure was seen in continuity with the cecum and was incidentally discovered during dissection of mesoappendix (Fig. 1). An operative diagnosis of double appendix was made. The operation was terminated after retrieval of the appendices with endopouch and exploration of rest Adress correspondence to: Naorem Gopendro Singh, MD, Department of Pathology, Al-Jahra Hospital, P O Box 62276, Jahra 02153, Kuwait. Tel: + 965-97128990, Fax: + 965-24582628, E-mail: gopen71@yahoo.co.in
December 2012 KUWAIT MEDICAL JOURNAL 330 Fig. 1: Laparoscopic photograph showing the base of double appendix (arrows) and attached mesoappendix in-between Fig. 3: Photomicrograph showing the two separate mucosas (arrows) of the double appendix with two separate muscle coats and both muscular walls separated by meso-appendicular fibroadipose tissue ( H & E stain X20) of small and large bowels, which ruled out any other associated congenital anomaly. Her postoperative course was uneventful and she was discharged on postoperative day five. Gross pathological examination of the specimen showed duplex vermiform appendix (Fig. 2). The bigger appendix measured 5.1 cm and the small one measured 1.5 in length. Both the appendices were attached to mesoappendix in close proximity. The specimen displayed focal congestion in the serosal aspect of the bigger appendix. Microscopic examination revealed two lumina of appendices having two separate muscle coats and lymphoid follicles in the mucosa (Fig. 3). The bigger and longer appendix revealed mixed acute and chronic inflammatory infiltrate in the mucosa as well as in the muscle coat showing features of acute appendicitis with periappendicitis. The small one showed regular morphology without any inflammation. Thus the operative diagnosis of Fig. 2: Gross photograph of the double appendix. Arrow pointing the small appendix double appendix was microscopically confirmed with one appendix showing features of acute appendicitis. DISCUSSION Duplication of the vermiform appendix, originally described in 1930, is rare with a reported incidence of 0.004% [1] . The etiology of double appendix is unknown, but the most rational explanation was offered by Kelly and Hurdon who examined 54 human embryos to explain the origin and development of the appendix [4] . They described a minute budding “transient appendix” on the tip of the cecum in a 6-week-old embryo [4] . This small “transient appendix” usually atrophied or disappeared in the 8-week-old embryo [4] . Hence “transient appendix” is a definite potential for the embryological origin for the development of a supernumerary appendix and the most plausible explanation of the appendix duplex. Cave [5] classified three types of appendicular duplication as: Type A: Single cecum with one appendix exhibiting partial duplication Type B: Single cecum with two obviously separate appendices (complete duplicity) Type C: Duplication of cecum with each cecum bearing its proper appendix The simplest illustrations of the first type are those curious specimens of ‘double-barrelled’ appendix wherein the single organ presents two distinct lumina throughout either length or throughout only a part thereof [5] . The second type was first recorded by Paterson and Emrys-Robert (1906) wherein a full-term fetus, the subject of ectopia viscerum, spina bifida and other congenital anomalies showed a small ‘sacculated and curved appendix’ lying on each side of the ileocecal junction [5] . Type C was first reported by Greig (1934) in which the whole bowel duplicates distal to the site of Meckel’s diverticulum; two separate ceca were present, each bearing its proper vermiform appendix [5] .
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Vol 44 # 4 December 2012 - Kma.org.kw

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