Duplex Vermiform Appendix Left Behind after Laparoscopic Appendectomy: A Case Report

doi:10.21203/rs.3.rs-3341350/v1

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Duplex Vermiform Appendix Left Behind after Laparoscopic Appendectomy: A Case Report

https://doi.org/10.21203/rs.3.rs-3341350/v1

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Background: Duplex appendix, a rare form of vermiform appendix, is seldom encountered. These anomalies are often found incidentally during surgery for other pathologies or occasionally due to their own involvement.

Case presentation: Here, we present a case of a 19-year-old male who initially presented to the hospital with right lower quadrant abdominal pain accompanied by nausea, vomiting, and anorexia. Physical examination revealed tenderness at the McBurney's point and rebound tenderness along with visible guarding. Blood counts showed leukocytosis. Abdominal ultrasound demonstrated an enlarged and thickened appendix. The patient underwent laparoscopic appendectomy, and then, 4 months later, he returned to the hospital with pain in the right lower quadrant, tenderness, and rebound tenderness. A diagnostic laparoscopic surgery was performed, revealing another appendix in the retrocecal area. Ultimately, the patient underwent a repeat appendectomy and was discharged from the hospital without complications after one day.

Conclusions: Acute appendicitis should not be ruled out definitively in a patient with typical clinical manifestations, especially in those with a history of laparoscopic appendectomy. Complete exploration of the cecum during surgery is the only reliable option to rule out appendix anomalies and prevent future medical complications.

Duplex appendix

Duplicated appendix

Laparoscopic appendectomy

Case report

Abnormalities of the appendix pose a challenging scenario for surgeons (1). Some of these abnormalities include agenesis, atresia, duplication, and the vermix appendix, which are exceedingly rare and are typically found incidentally in adult populations (1, 2). Duplex appendix is a highly uncommon form of vermiform appendix, reported in 0.004–0.009% of appendectomy specimens (3, 4). Duplex appendix was first reported in fetal studies in 1867 by Bartels and in adult studies in 1892 by Picoli (5). The rarity of the duplex vermiform appendix makes it a noteworthy medical occurrence, with only around 100 reported cases since its initial observation in 1892 (6). In 1936, Cave first classified cases of duplex appendix (7). This classification was later modified by Cook and colleagues, breaking it down into 4 categories (8). These classifications are A, B1, B2, and C. However, this classification has been updated to encompass other subgroups such as the horseshoe appendix and triplex appendix (9) (see Table 1). This anomaly is often asymptomatic and is frequently discovered during surgery for other pathologies or occasionally due to its own involvement (e.g., appendicitis, perforation, and tumors), or during post-mortem examinations. Some cases can be diagnosed before surgery using barium enema. Clinical symptoms are typically the result of obstruction and inflammation, and clinical manifestations can vary depending on the location of the appendix (4, 7). Despite the rarity of this report, awareness of the possibility of such anomalies in patients is of paramount importance to prevent medical-legal consequences related to delayed diagnosis and discovery of duplex appendices. Here, we report a case of a remaining duplex appendix after laparoscopic appendectomy in a 19-year-old male.

Table 1

Showing the modified Cave-Wallbridge classification
Classification	Description
Type A	single cecum that has one appendix at its normal location exhibiting incomplete duplication
Type B1	two appendices located on both sides of the ileocecal valve symmetrically
Type B2	one appendix is located at the normal site and the other along the tenia coli
Type C	Double cecum with an appendix each
Type D	One appendix has two opening at the cecum (the horseshoe type)

A 19-year-old male presented to a hospital located in the northern part of the Persian Gulf in Iran with a 2-day history of pain in the right lower quadrant of his abdomen (RLQ), along with nausea, vomiting, and loss of appetite. His bowel habits remained unchanged. On examination, tenderness upon palpation at McBurney's point was evident, along with rebound tenderness and visible guarding. Blood count revealed leukocytosis (WBC, 15,300/mm3). Abdominal ultrasound showed a dilated, non-compressible, and thickened appendix. The patient was diagnosed with acute appendicitis and underwent laparoscopic appendectomy. During the surgery, an appendiceal abscess with surrounding adhesions was observed. Following adhesiolysis, the mesoappendix was ligated and divided using LigaSure, and the base of the appendix was also ligated with 3 LigaClips. The patient was discharged from the hospital without complications after one day. Four months later, the patient returned to the hospital with renewed RLQ pain, tenderness upon palpation, and rebound tenderness. A blood count showed leukocytosis. An ultrasound revealed signs of infection and inflammation in the RLQ. The patient underwent a diagnostic laparoscopic surgery, during which evident purulent material was found in the RLQ. After releasing the surrounding tissue along the lines of Toldt approximately 7 to 8 centimeters farther away from the site of the first removed appendix, a second appendiceal structure, the duplex appendix (B2 type), was discovered. It was surrounded by abundant inflammatory exudates on the right lower side of the abdomen. No other significant surgical findings were present, and the remaining abdominal organs appeared normal. The patient underwent a second appendectomy. Histopathological examination of both appendices revealed inflammatory changes in lymphoid follicles and smooth muscle, confirming our diagnosis. The patient was discharged from the hospital without complications after one day.

Most appendix abnormalities are observed in adults and are found incidentally during surgeries that do not primarily involve the appendix (10). The appendix first appears as an outgrowth of the cecum in the eighth week of gestation. However, the embryology of duplex appendix and its variations are less understood. Picoli reported the first case of appendiceal duplication in 1892 in a female patient who had associated anomalies, including duplication of the entire large intestine, double uterus with two vaginas, ectopia vesicae, and exomphalos (5). Diagnosis can be an incidental finding on imaging or through complex clinical presentations. In the neonatal stage, it appears along with other congenital anomalies, but in older age groups, it may manifest as acute appendicitis, colonic perforation, obstruction, enuresis, bleeding, pain, growth retardation, or abdominal mass. It can remain asymptomatic in many patients throughout life (1, 4, 11–14). Our patient had a duplex vermiform appendix, classified as B2 according to the Cave Wallbridge classification. Because the thorough exploration of the cecum and retrocecal space was not adequately performed during the first appendectomy, the second appendix had been overlooked. It has been demonstrated that misdiagnosis of duplex appendix has detrimental clinical and medical consequences (15–17). In a systematic review conducted by Negzvar and colleagues (2018), out of 141 reported cases of duplex appendix, 67.5% were of the B1 type, and 32.5% were of the B2 type, making them among the most common types (18). B1 and C types are usually associated with structural abnormalities of the urinary and large intestinal systems (18, 19). In our case, the duplex appendix was of the B2 type and was not associated with any other anomalies.The reported age range for duplex appendix varies between fetuses and 69 years old, with an average age of 20 years, and a male-to-female ratio of 4:1 (19). In our report, this type of appendix was diagnosed in a 19-year-old male patient. Therefore, we are presenting this case to highlight that this anomaly went undetected during the initial laparoscopic appendectomy for this patient. Consequently, awareness of this rare anomaly in patients with a history of previous appendectomy is crucial in case clinical symptoms and signs of appendicitis arise. Similarly, considering this type of anomaly in individuals undergoing appendectomy, especially in the age range between fetuses and 69 years old, and in males, as per the existing literature, holds even greater importance.

While the use of the McBurney incision is highly practical for the majority of cases of acute appendicitis, it provides minimal access to the intra-abdominal organs. The laparoscopic approach undoubtedly can offer better visualization of the area around the cecum and minimize surgical complications. Laparoscopic appendectomy has been successfully reported in cases of duplex appendices in the pediatric population (20). However, even though the laparoscopic approach is the standard method for appendectomy in our department, we did not diagnose the patient's duplex appendix during the initial appendectomy. Retrocecal duplex appendices of type B are not easily identifiable (16). Failure to remove the sister appendix and failure to diagnose this condition can have serious clinical, medical, and legal consequences (21). Therefore, it's essential for the surgeon to be aware of such anomalies. In cases of duplex appendix, when only one appendix is inflamed, both should be removed to prevent potential diagnostic challenges that may arise later. However, non-inflamed duplex appendices found incidentally during exploration for other conditions do not require appendectomy (10). Hence, awareness of this rare anomaly in patients with a history of previous appendectomy is of utmost importance if they exhibit clinical symptoms and signs of appendicitis (9). In conclusion, our case illustrates that even though a patient may have recently undergone laparoscopic appendectomy, a duplex appendix in the patient may not be diagnosed during the initial procedure, and the patient may return to the hospital with symptoms of acute appendicitis several months after the first appendectomy.

This case report has been reported following the SCARE 2020 criteria (22).

In the differential diagnosis of lower abdominal pain, even if a patient has previously reported an appendectomy, a duplex appendix should be considered. Surgeons should be aware of potential anatomical variations of the vermiform appendix, and a thorough examination of the cecum should be performed during laparoscopy. A misdiagnosis can lead to serious life-threatening complications for

Acknowledgements

We would like to express our sincere appreciation to the patient and their esteemed family for providing the necessary information and cooperating throughout all stages of this study. Additionally, we extend our gratitude to the Clinical Research Development Unit of Bushehr University of Medical Sciences for their dedicated support in conducting this research.

Funding

There was no funding for this case report.

Availability of data and materials

Data are available from the corresponding author on reasonable request.

Authors’ contributions

A.E. and S.T. prepared the case and conducted the literature review,A.E. prepared the manuscript draft. All authors analyzed, interpreted the case and literature, and critically reviewed and revised the manuscript for important intellectual content. All authors read and approved the final manuscript.

Competing interests

The authors declare that they have no competing interests

Consent for publication

The patient signed an informed consent for this case report and the document is attached with the cover letter.

Ethics approval and consent to participate

Not applicable (That is because all procedures described were carried out as part of standard care).

Sagoe R, Seidu AS, Assim AO, Amoah M, Davor A, Galley F, et al. Ruptured Meckel's diverticulum coexisting with a duplex appendix. J Pediatr Surg Case Rep. 2021;73:101990.
Ademaj I, Kurshumliu F, Syla A. Congenital stenosis of appendix vermiformis: A case report. J Pediatr Surg Case Rep. 2023;92:102619.
Mathur V, Garg D, bahadur Singh U, Grover J. Duplication of Appendix in four cases of Congenital pouch colon: Case series. J Dent Med Sci (IOSR-JDMS). 2023;22(3):62–5.
Bakhteyar MAK, Prasad R, Chaubey D, Yadav R, Kumar P. Appendicular Duplication–Variations in Anatomy and Associations. Iran J Pediatr Surg. 2023;9(1):43–55.
Khanna AK. Appendix vermiformis duplex. Postgrad Med J. 1983;59(687):69–70.
Tinkham M, Ziesat M, Straumanis J, Heisler S. Double Appendix: Implications for the Emergency Department. J Emerg Med. 2021;61(2):180–3.
Mushtaque M, Mehraj A, Khanday SA, Dar RA. Double appendicitis. Int J Clin Med. 2012;3(1):60–1.
Coker DD, Hartong JM, Conklin HB. Duplication of the vermiform appendix: case report. Mil Med. 1975;140(5):331–2.
Dhannoon A, Kunna M, Baynes D, Hynes SO, Nugent E. Case report of the duplex vermiform appendix: A rare presentation in an elderly patient. Int J Surg Case Rep. 2022;92(1):106829.
Dubhashi SP, Dubhashi UP, Kumar H, Patil C. Double Appendix. Indian J Surg. 2015;77(3):1389–90.
Bali R, Wani M, Mir S, Thakur S, Bhat R. Appendiceal duplication with simultaneous acute appendicitis and appendicular perforation causing small bowel obstruction. J Surg case Rep. 2011;2011(2):3–.
Dhar PP, Mohanty U. A rare case report of infant ileal atresia with double appendix. Int J Surg Case Rep. 2020;74(1):226–9.
Damera VV, Rathlawath SK. A rare case of duplicated appendix presenting as acute appendicitis. Indian J Colo-Rectal Surg. 2021;4(2):47–9.
Macpherson RI. Gastrointestinal tract duplications: clinical, pathologic, etiologic, and radiologic considerations. Radiographics. 1993;13(5):1063–80.
Samee A, Alibhai M, Ameer N, Sparks M, Morgan A, Halliday M. Appendicitis in a duplex appendix mimicking intussusception. Case Rep. 2010;2010(1):bcr1220092516.
Christodoulidis G, Symeonidis D, Spyridakis M, Koukoulis G, Manolakis A, Triantafylidis G, et al. Acute appendicitis in a duplicated appendix. Int J Surg case Rep. 2012;3(11):559–62.
Alves JR, Maranhão IG, de Oliveira PV. Appendicitis in double cecal appendix: Case report. World J Clin cases. 2014;2(8):391–4.
Nageswaran H, Khan U, Hill F, Maw A. Appendiceal Duplication: A Comprehensive Review of Published Cases and Clinical Recommendations. World J Surg. 2018;42(2):574–81.
Panda SK, Prasad C, Tirkey R, Rajesh V, Mishra J, Dora RK. Unusual association of Meckel's diverticulum with double appendix - A rare finding. Int J Surg Case Rep. 2014;5(11):879–81.
Christodoulidis G, Symeonidis D, Spyridakis M, Koukoulis G, Manolakis A, Triantafylidis G, et al. Acute appendicitis in a duplicated appendix. Int J Surg Case Rep. 2012;3(11):559–62.
Mitchell I, Nicholls J. Duplication of the vermiform appendix: report of a case: review of the classification and medicolegal aspects. Med Sci Law. 1990;30(2):124–6.
Agha RA, Franchi T, Sohrabi C, Mathew G, Kerwan A, Thoma A, et al. The SCARE 2020 Guideline: Updating Consensus Surgical CAse REport (SCARE) Guidelines. Int J Surg. 2020;84(1):226–30.

No competing interests reported.

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Duplex Vermiform Appendix Left Behind after Laparoscopic Appendectomy: A Case Report

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2. Case presentation

3. Discussion

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