
==== Front
Int J Surg Case Rep
Int J Surg Case Rep
International Journal of Surgery Case Reports
2210-2612
Elsevier

S2210-2612(24)01047-2
10.1016/j.ijscr.2024.110266
110266
Case Report
Hidden schistosomiasis unveiled by appendicular peritonitis: A case report
Limaiem Faten fatenlimaiem@gmail.com
ab⁎
Zaafouri Montassar ac
Atallah Aziz ac
a University of Tunis El Manar, Faculty of Medicine of Tunis, Tunisia
b Pathology Department, Mongi Slim Hospital La Marsa, Tunisia
c Department of Surgery, Mongi Slim Hospital La Marsa, Tunisia
⁎ Corresponding author at: Department of Pathology, Mongi Slim Hospital, La Marsa, Tunisia. fatenlimaiem@gmail.com
10 9 2024
10 2024
10 9 2024
123 1102665 8 2024
4 9 2024
8 9 2024
© 2024 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Limited.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Introduction and importance

Schistosomal appendicitis is a rare disease, with reported prevalence rates ranging from 1.31 to 3.2 %. The presented case underscores the critical significance of considering appendicular schistosomiasis as a potential etiology in cases of acute appendicitis, emphasizing the necessity of comprehensive histopathological examination for accurate diagnosis and appropriate postoperative management.

Case presentation

A 29-year-old man from Guinea, with no significant medical history, presented with vomiting, persistent abdominal pain, and fatigue over five days. Physical examination revealed signs of peritoneal irritation and imaging showed features indicative of acute appendicitis. An appendectomy was conducted laparoscopically. Histological examination confirmed gangrenous appendicitis with the presence of schistosome eggs, diagnosing acute gangrenous appendicitis with schistosomiasis. The patient recovered well postoperatively and was discharged after treatment with praziquantel.

Clinical discussion

The clinical presentation of schistosomal appendicitis resembles that of other acute appendicitis cases. When suspicion arises due to risk factors, confirming schistosomiasis may involve serology, polymerase chain reaction assays, and identifying eggs in urine or feces. Computed tomography findings cannot distinguish acute appendicitis caused by Schistosoma species from other causes.

Conclusions

Histopathological appendix analysis is crucial for detecting conditions like schistosomiasis, warranting postoperative care. Praziquantel therapy post-surgery is vital for eradicating the disease and preventing complications.

Highlights

• Schistosomal diagnosis depends on histological examination due to no specific indicators.

• In non-endemic areas, healthcare providers should watch for atypical schistosomiasis cases in travelers or immigrants.

• Multidisciplinary collaboration is vital for managing schistosomal infections in low-endemic regions.

• Swift treatment with appendectomy and anti-helminthic therapy is crucial for managing acute appendicitis and preventing schistosomiasis complications.

• In endemic areas, preventing schistosomiasis involves avoiding contaminated water, good hygiene, protective clothing, preventive medication, health education, vector control, sanitation, and regular screening.

Keywords

Appendicitis
Peritonitis
Schistosomiasis
Appendectomy
Schistosoma ova
Histopathology
==== Body
pmc1 Introduction

Schistosomiasis, a prevalent chronic granulomatous disease primarily found in tropical regions, can affect various organs in the body. Schistosomal appendicitis is a rare condition, with reported prevalence rates ranging from 1.31 to 3.2 % [1,2]. The development of acute appendicitis and peritonitis due to schistosomiasis is exceedingly rare, even within regions where the disease is prevalent [3]. While more common in endemic regions, cases have been documented in non-endemic areas due to global travel and migration patterns [3]. Tunisia, though not endemic for schistosomiasis, hosts a diverse population including immigrants from endemic regions. The prevalence and clinicopathological features of appendiceal schistosomiasis in Tunisia, a non-endemic region for the disease, are poorly documented and limited. Confirmation of schistosomal involvement of organs usually depends on histological diagnosis, given the absence of pathognomonic clinical or operative findings. Presented herein is a rare case of acute peritonitis resulting from schistosomal appendicitis in a male immigrant from Guinea.

This case report adheres to the SCARE Criteria [4].

2 Case presentation

A 29-year-old man from Guinea, with an unremarkable medical history, sought medical attention at the emergency department with complaints of vomiting, persistent abdominal pain, and fatigue over the past five days. Upon admission, the patient presented with a fever of 38 °C, tachypnea, a blood pressure of 110/70 mmHg, and a heart rate of 100 beats per minute. A physical examination unveiled abdominal pain in the right lower quadrant accompanied by signs of peritoneal irritation, such as rebound tenderness and generalized guarding. The blood laboratory results indicated a white blood cell count of 10,640, C-reactive protein (CRP) level of 468, and a hemoglobin level of 14.3 g/dL. The patient's liver function tests, including total bilirubin at 0.7 mg/dL (normal range: 0.1–1.2 mg/dL), AST at 13 U/L (normal range: 10–40 U/L), ALT at 12 U/L (normal range: 7–56 U/L), ALP at 50 U/L (normal range: 44–147 U/L), and GGT at 15 U/L (normal range: 9–48 U/L), were within normal ranges. Urea: 12 mg/dL (normal range: 7–20 mg/dL); Creatinine: 103 μmol/L (normal range: 53–106 μmol/L); Na+: 131 mmol/L (normal range: 135–145 mmol/L); K+: 4.5 mmol/L (normal range: 3.5–5.1 mmol/L).

The abdominal CT scan (Fig. 1A) revealed a complex retrocecal hydroaeric multilocular collection, characterized by a thickened enhanced wall, stretching along the entire right paracolic gutter where it abuts the liver. This was accompanied by a diffuse distension of the intestinal loops filled with hydroaeric content. Additionally, there was a moderate amount of intra-peritoneal fluid present in the perihepatic and perisplenic regions, as well as along the left paracolic gutter between the loops and in the pelvic area. These findings were further accentuated by a diffuse thickening of the peritoneal layers and a generalized densification of the mesenteric fat. The radiological findings suggested a generalized peritoneal reaction likely originating from the appendix, associated with probable acute functional intestinal obstruction. During the exploratory laparoscopy, the retrocecal appendix was identified as gangrenous accompanied by generalized peritonitis with pseudomembranes. An appendectomy was conducted laparoscopically, followed by a comprehensive saline irrigation quadrant by quadrant. The macroscopic analysis of the appendix specimen revealed its surface to be coated with fibropurulent materials, indicating signs of gangrene and perforation (Fig. 1B, C). Histological examination of the surgical specimen revealed features consistent with gangrenous appendicitis (Fig. 1D). The appendix exhibited widespread necrosis of its wall, a prominent infiltration of inflammatory cells, marked vascular congestion, and fibrin thrombi within the blood vessels. Notably, multiple calcified eggs of diverse shapes (round, ovoid, elongate, or lemon-shaped, some with small subterminal spines or none) were observed, surrounded by a varied inflammatory infiltrate, indicative of schistosomiasis (Fig. 2A, B, C, D). The final diagnosis was acute gangrenous appendicitis associated with the presence of schistosome eggs. The patient's postoperative recovery was uncomplicated and he was discharged on postoperative day four. Stool and urine samples were collected, and parasitological analysis confirmed the presence of Schistosoma japonicum eggs in the stool sample. Following treatment with a single dose of praziquantel (40 mg/kg), the patient was instructed to return for a follow-up appointment in two weeks. Throughout the follow-up period, the patient remained generally well, albeit experiencing mild persistent abdominal pain.Fig. 1 A: Axial CT scan of the abdomen depicting dilated bowel loops.

B: Macroscopic examination revealing gangrenous appendix post-resection.

C: Macroscopic examination of gangrenous appendix upon sectioning.

D: Histopathological section revealing gangrenous appendicitis. (Hematoxylin and eosin, magnification ×100).

Fig. 1

Fig. 2 A: Histological section of the appendix displaying numerous calcified schistosomal eggs in a background of acute gangrenous appendicitis. (Hematoxylin and eosin, magnification ×40).

B: Microscopic analysis of the appendix reveals calcified schistosomal eggs of varying shapes distributed across the appendiceal wall. (Hematoxylin and eosin, magnification ×100).

C: Histological section of the appendix illustrating schistosoma eggs alongside ulceration of the appendiceal mucosa. (Hematoxylin and eosin, magnification ×100).

D: Schistosome eggs within the appendiceal wall showing the presence of terminal spines (black arrows). (Hematoxylin and eosin, magnification ×400).

Fig. 2

3 Discussion

Schistosomiasis represents a significant global health burden, posing a major threat to public health worldwide. Nearly 800 million individuals are susceptible to this disease, making it the second most prevalent infectious disease after malaria [3]. It is estimated that schistosomiasis afflicts over 250 million individuals across 78 countries annually and leads to approximately 280,000 to 500,000 deaths each year [3]. The primary schistosome species affecting humans include Schistosoma mansoni, S. japonicum, and S. haematobium. [[5], [6], [7]]. The occurrence of appendicitis due to schistosomiasis is rare, even in areas where the disease is common. A systematic review of international literature indicated an overall prevalence of 1.3 %. When stratified by continents, Africa showed a prevalence of 2.8 %, while the Middle East had a prevalence of 0.5 % [2]. In non-endemic countries, studies reported a lower prevalence rate ranging from 0.1 % to 0.2 % [8,9]. The prevalence and clinicopathological characteristics of appendiceal schistosomiasis in Tunisia, a non-endemic region for the disease, are limited. The reported cases of schistosomiasis in Tunisia are often linked to global travel and migration patterns. Globally, there are few documented instances of peritonitis stemming from schistosomal appendicitis. Gali et al. detailed a single case of generalized peritonitis among 27 cases of schistosomal appendicitis [8]. An occurrence of schistosomal peritonitis, where eggs were discovered in the greater omentum and peritoneal cavity, was documented in an Egyptian migrant in Greece [10]. Schistosomal appendicitis is categorized into two forms: ‘granulomatous acute appendicitis,’ triggered by an immune reaction to fresh eggs causing tissue damage quickly, and ‘obstructive acute appendicitis,’ resulting from long-term inflammation and fibrosis around dead eggs, obstructing the appendiceal lumen and raising infection risks over time. [6].

The clinical presentation of schistosomal appendicitis closely resembles that of other causes of acute appendicitis. Standard hematological and biochemical tests offer limited additional insights to indicate a schistosomal origin [1,7]. When suspicion arises based on risk factors, serology, polymerase chain reaction assays, and the detection of eggs in urine or feces may be employed to confirm schistosomiasis. Computed tomography results are unable to differentiate acute appendicitis caused by Schistosoma species from other etiologies. The diagnosis of schistosomal appendicitis requires histopathological confirmation, demonstrating the presence of schistosomal ova. Histologically, schistosomiasis is characterized by the presence of foreign body granulomas surrounding eggs or an infiltrate of eosinophils and neutrophils. While hematoxylin and eosin staining can reveal schistosomal eggs, special stains may be necessary for clearer identification. The egg shells of S. mansoni and Schistosoma intercalatum are acid-fast, unlike those of S. haematobium.

The management of schistosomal appendicitis involves appendectomy and anthelmintic therapy with praziquantel at 40 mg/kg, while for intestinal and urogenital schistosomiasis, praziquantel at the same dosage is the main treatment, with consideration for individualized second doses and treatment duration for effective parasite eradication and complication prevention [1,7,10,11].

The strength of our case report lies in the uncommon occurrence of appendiceal schistosomiasis with an atypical presentation of peritonitis, facilitating the diagnosis of this condition. This rare instance, observed in an immigrant patient from Guinea, an endemic region for this parasitic disease, underscores the importance of considering schistosomiasis in cases of appendiceal peritonitis among immigrants and travelers to endemic regions, compelling clinicians and surgeons in non-endemic countries to maintain a high suspicion for such infections. The weakness of our case report lies in the absence of pertinent statistical data on schistosomiasis, particularly regarding appendiceal schistosomiasis in Tunisia, as well as the limited follow-up period for this recently diagnosed case of appendiceal schistosomiasis.

4 Conclusion

In regions like Tunisia where schistosomiasis is non-endemic, surgeons must be vigilant for appendiceal schistosomiasis, particularly in patients with travel history to endemic areas or within immigrant populations. Given the absence of definitive clinical or operative signs, histological confirmation is vital for identifying schistosomal involvement in organs. Effective coordination between surgeons and pathologists is essential for accurate diagnosis. It is imperative that all appendectomy specimens undergo histopathological evaluation, irrespective of their appearance. Swift treatment, involving appendectomy and anti-helminthic therapy, is essential for managing acute appendicitis and averting schistosomiasis-related complications.

Consent statement

Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Ethical approval

Ethical approval for this study was provided by the Ethical Committee of Mongi Slim University Hospital, Marsa, Tunisia.

Funding

This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Author contribution

Dr. Faten LIMAIEM: Prepared, organized, wrote, and edited all aspects of the manuscript. Performed the gross and microscopic pathologic evaluation of the pathology specimen.

Dr. Montassar ZAAFOURI and Dr. Aziz ATALLAH: Read, edited, and approved the final version of the manuscript. Contributed to data acquisition, analysis, and interpretation. Provided final approval of the manuscript before its submission.

Guarantor

Dr. Faten Limaiem.

Conflict of interest statement

None declared.
==== Refs
References

1 Khalifa M. Elhassan E. Abdel Rahim S. Schistosomal appendicitis: a case report J. Med. Case Rep. 18 1 2024 283 38890741
2 Zacarias M. Pizzol D. de Miranda H. Colangelo A.C. Veronese N. Smith L. Schistosomal appendicitis: case series and systematic literature review PLoS Negl. Trop. Dis. 15 2021 e0009478
3 LoVerde P.T. Schistosomiasis Adv. Exp. Med. Biol. 1454 2024 75 105 39008264
4 Sohrabi C. Mathew G. Maria N. Kerwan A. Franchi T. Agha R.A. The SCARE 2023 guideline: updating consensus Surgical CAse REport (SCARE) guidelines Int. J. Surg. 109 5 May 2023 1136 37013953
5 Gundlapalli V. Shah M. Baskara A. Atypical appendicitis: schistosomal infection causing perforated appendicitis Int. J. Surg. 28 2012 1 6
6 Hasan A. Elhussiny M.E.A. Nagaty M.E. Eid M. Elias A.A.K. Abdulmohaymen A. Clinico-pathological profile of schistosomal appendicitis detected in surgically resected appendices: a retrospective study Int. J. Surg. Open. 54 2023 100606
7 Weber G. Borer A. Zirkin H.J. Riesenberg K. Alkan M. Schistosomiasis presenting as acute appendicitis in a traveler J. Travel Med. 5 1998 147 9772333
8 Karatepe O. Adas G. Tukenmez M. Battal M. Altiok M. Karahan S. Parasitic infestation as a cause of acute appendicitis G. Chir. 30 2009 426 428 19954582
9 Gali B.M. Nggada H.A. Eni E.U. Schistosomiasis of the appendix in Maiduguri Trop Doctor 36 2006 162 163
10 Konstantinidou E. Alexiou C. Deminakou M. Sakellaridis T. Fotopoulos A. Antsaklis G. Schistosomal peritonitis: a rare cause of acute abdomen Trans. R. Soc. Trop. Med. Hyg. 103 2009 1068 1070 19135220
11 Nwabuoku S.E. Mukoro G.D. Daniyan M. Dauda M.M. Khalid L. Aliyu H.O. Shittu S.M. Gana S.G. Suraj A. Idokoko E. Okeke C. Iji L. Audu J.A. Appendiceal schistosomiasis presenting as an appendiceal mass J. West Afr. Coll. Surg. 12 1 2022 100 103 36203918
