
==== Front
Ann Med Surg (Lond)
Ann Med Surg (Lond)
MS9
Annals of Medicine and Surgery
2049-0801
Lippincott Williams & Wilkins Hagerstown, MD

AMSU-D-24-00443
10.1097/MS9.0000000000002320
00017
3
Original Research
Minimally invasive spleen-preserving surgery to treat primary splenic hydatidosis: short and long-term outcomes: a cohort study
Aljaiuossi Anas MD aanas.jaiuossi@yu.edu.jo

Ba-shammakh Saleh A. MD d*bashammakh97.2.5@gmail.com

Bani Hani Mohammad MD bMohbanihani@hotmail.com

Al-A’athal Musab S. MD cmsalaathal222@med.just.edu.jo

Elsobuh Yazeed M. MD bYazeedmsuboh@gmail.com

Abu Sarhan Hashem MD eHAbuserhan@hamad.qa

Ennab Raed Mahmoud MD araed.ennab@yu.edu.jo

Al-Zubi Mohammad MD Mzubi@yu.edu.jo
a
Alhwari Mohammad J. MD aM7md.Al7wari@gmail.com

Al Omari Laith G. MD aLaith.omari.d1@gmail.com

Mohammad Feras M. MD aaldweerifiras@gmail.com

AL lami Mohamed S. MD ams9lami@gmail.com

Zeitoon Hammam B. MD aHammam.ziton@gmail.com

Alomari Saad A. MD aAl_omarisaad@yahoo.com

Ababneh Salman M. MD asalman.m.ababneh@gmail.com

a Faculty of Medicine, Yarmouk University, Irbid
b King Abdullah University Hospital, Faculty of Medicine
c Department of Public Health and Community Medicine, Jordan University of Science and Technology, Irbid
d Department of General Surgery, Ministry of Health, Amman, Jordan
e Department of Ophthalmology, Hamad Medical Corporations, Doha, Qatar
* Corresponding author. Address: Department of General Surgery, Ministry of Health, P.O. Box 86, Amman 11118, Jordan. Tel.: + 962 780 762 174. E-mail: bashammakh97.2.5@gmail.com (S. A. Ba-shammakh).
9 2024
1 7 2024
86 9 49995006
4 3 2024
18 6 2024
Copyright © 2024 The Author(s). Published by Wolters Kluwer Health, Inc.
2024
https://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0/

Background:

Primary splenic hydatidosis, a rare manifestation of Echinococcus granulosus infection, presents unique diagnostic and therapeutic challenges. This study compares spleen-preserving surgeries with total splenectomy for treating primary splenic hydatid cysts, focusing on short- and long-term outcomes in the Jordanian context, a region particularly affected by this condition.

Methods:

This retrospective analysis was conducted on 18 patients diagnosed with primary splenic hydatid cysts at two Jordanian hospitals from January 2015 to June 2021. Selection criteria included confirmed diagnosis and complete medical records. Surgical approaches, including laparoscopic partial splenectomy, cystectomy, and cyst deroofing, supplemented by albendazole therapy, were compared based on patient demographics, symptoms, surgical details, complications, and recurrence rates.

Results:

The study group was composed of (n=7, 38.9%) male and (n=11, 61.1%) female patients, with an average age of 33.7 years. Most presented with left upper quadrant pain. Postoperative complications occurred in 22% of patients, with an 11% recurrence rate during follow-up. No significant statistical difference in recurrence rates was observed between spleen-preserving surgeries and total splenectomy. These findings highlight the efficacy of less invasive, spleen-preserving techniques in managing primary splenic hydatidosis, showing comparable outcomes to total splenectomy with minimal impact on recurrence rates.

Conclusion:

Spleen-preserving surgery offers a viable alternative to total splenectomy in treating primary splenic hydatid cysts. This approach maintains immune functionality and reduces septic risks, especially in pediatric patients. The study underscores the importance of individualized treatment approaches and suggests further research with larger cohorts for more comprehensive insights into managing this rare condition. The limitations of this study include its small sample size and retrospective nature.

Keywords:

echinococcus granulosus
hydatid disease
minimally invasive surgery
primary splenic hydatid cyst
recurrence
spleen-preserving surgery
OPEN-ACCESSTRUE
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pmcIntroduction

Highlights

Spleen-preserving surgery effective for hydatid cysts.

Outcomes similar to splenectomy with reduced risks.

Postoperative complications at 22%, 11% recurrence rate.

Efficient for both adult and pediatric patients.

Calls for further research, citing small sample size.

Echinococcosis, also known as cystic echinococcosis (CE) or hydatid disease, is caused by the Echinococcus granulosus larva. Hydatid disease primarily affects the liver and lungs, posing a significant health risk1. This condition is characterized by the formation of cysts, most notably in these organs. Less frequently, it can be attributed to E. oligarthrus, E. multilocularis, and E. vogeli 1,2.

Human infections are mainly due to Echinococcus multilocularis and Echinococcus granulosus, leading to alveolar and cystic echinococcosis, respectively3. The global incidence of cystic echinococcosis varies, with rates ranging from 1 to 200 per 100 000 annually3. Canids, particularly dogs, serve as primary hosts for the adult form of this tapeworm. In contrast, ungulates, like sheep, often become intermediate hosts after consuming the eggs. Humans are at risk, especially in regions where close contact with these animals is common, potentially becoming accidental intermediate hosts3,4.

The disease is a considerable zoonotic concern, especially in regions where sheep farming is prevalent5,6. Its high prevalence is notably reported in Middle Eastern countries, such as Jordan, where the close coexistence of sheep, dogs, and humans, particularly among rural and Bedouin communities, increases the risk7. The larval form of E. granulosus, once hatched, can penetrate intestinal walls, enter the bloodstream, and travel to various organs. The liver is the primary site (75%), with the lungs following (15%), and other organs such as the spleen, kidneys, and others are less commonly affected8,9. Primary splenic hydatidosis is rare and often speculated to be due to arterial transmission or transcoelomic spread. It accounts for 0.8–4% of all human hydatid cases and 5.8% of abdominal cases, ranking third in organ involvement after the liver and lungs10,11.

The disease’s nature is insidious, with cysts growing slowly over years before becoming detectable12. Complications can arise at any stage, including cyst rupture, leading to secondary cyst formation13.

The management of primary splenic hydatid cysts remains largely undefined due to its rarity, with no established treatment guidelines. Historically, total splenectomy was the primary approach14. However, laparoscopic techniques have recently been favored, as they demonstrate superior outcomes with notably lower complication rates compared to traditional open surgeries15. Due to the substantial risks associated with total splenectomy, including the increased risk of severe post-splenectomy infections (OPSI) with a mortality rate as high as 50%, there has been a shift towards spleen-preserving procedures16. These less invasive methods, which include techniques like partial cystectomy, cyst unroofing, cystojejunal Roux-en-Y anastomosis and partial splenectomy, aim to retain the spleen’s critical immunological functions while mitigating associated risks17,18. Ideal for cases involving superficial, isolated, simple cysts, particularly in younger patients, these techniques have not shown a significant increase in recurrence rates compared to total splenectomy17,18. However, they may present challenges such as increased technical complexity and potential for greater bleeding due to the preservation of a larger portion of the spleen19.

Historically viewed as non-essential, the spleen is now understood to play key roles in immunity, blood filtration, and iron recycling. The removal of the spleen, particularly in children, can lead to severe complications such as rapid septicemia and increased mortality risk20–22. Hence, preventive strategies such as vaccinations and educating patients on infection risks are crucial post-splenectomy20.

This study is crucial for understanding splenic hydatid cysts in the Jordanian medical context, underscoring the importance of local data and treatment modalities. It aims to compare the outcomes of spleen-preserving surgeries with total splenectomy, a comparison vital for advancing medical practices in Jordan and other endemic areas.

The study’s main objective was to evaluate the short- and long-term outcomes, with a primary focus on recurrence, of spleen-preserving procedures versus total splenectomy procedures in managing primary splenic hydatid cysts. It aimed to contribute significantly to the field of surgical treatment for this condition, offering insights into the most effective treatment methods that could shape future surgical approaches and patient care strategies in managing hydatid disease.

Methods

Participants and settings

This retrospective study involved patients diagnosed with primary splenic hydatid cysts, from January 2015 to June 2021. The study included medical records of 18 patients aged 14 years or older, diagnosed exclusively with primary, non-recurrent splenic hydatid cysts. Exclusion criteria encompassed individuals under 14 years, patients in late pregnancy, those with recurrent cysts, extra-splenic involvement, or less than 2 years of postoperative follow-up.

Study design and procedure

Diagnostic confirmation of primary hydatid cysts was achieved using computed tomography (CT) scans, abdominal ultrasounds, and KUB CTs. Data collected included demographic information, symptoms, medical history, laboratory findings, diagnostic methods, surgical procedures, postoperative complications, durations of operation and hospitalization, and follow-up data. Cysts were classified according to Gharbi’s and WHO classification systems using CT scans and abdominal US21,23 (Tables 1, 2). Surgical options, selected at the time of operation, included laparoscopic partial splenectomy, cystectomy, or cyst deroofing. Patients received three courses of albendazole (15–20 mg/kg, max 800 mg/d): one course before surgery and two after surgery. Follow-up was conducted with an indirect hemagglutination test and ultrasound (US). If the US suggested cyst recurrence, confirmation was obtained through CT scans. Our findings were compared with the existing literature.

Table 1 Classification for hydatid cyst type.

Gharbi’s 1981	WHO classification	
Type I	CE1	Active	
Type II	CE3a	
Type III	CE2	Transition	
CE3b	
Type IV	CE4	Inactive	
Type V	CE5	

Table 2 Type of splenic hydatid cyst according to Gharbi’s classification.

Gharbi’s	WHO	Number, n (%)	
Type I	CE1		2 (11.1)	
Type II	CE3a		3 (16.7)	
Type III	CE2	3 (16.7)	7 (38.9)	
CE3b	4 (22.2)	
Type IV	CE4		4 (22.2)	
Type V	CE5		2 (11.1)	

Surgical technique

Patients eligible for spleen-preserving surgery underwent a thorough preoperative assessment, which included imaging studies to characterize the splenic cysts. Cysts considered for spleen-preserving resection were those located at the periphery of the spleen, had a well-defined wall, and lacked extensive vascular involvement or deep parenchymal connections that might necessitate total splenectomy. All patients received preoperative necessary vaccines 2 weeks prior to surgery, in case total splenectomy was deemed necessary intraoperatively. The position of patients was right decubitus with a 45° tilt, with a sandbag under the lumbar region. A suprarumbilical 11 mm port was inserted using the open Hasson technique; CO2 was insufflated to achieve a pneumoperitoneum at 14 mm Hg. Three more working ports were inserted under direct vision: a 10 mm in the left midclavicular line, a 5 mm in the left anterior axillary line at the level of the umbilicus, and a 5 mm in the midline with appropriate triangulation distances. The location of the splenic cyst(s) was determined. Dissection started with Ligasure freeing the splenic attachments, taking down the splenic flexure to varying extents depending on the cyst location. In the case of an upper pole cyst location, the lesser sac was opened and the gastrosplenic ligament was dissected to gain access to the cyst and allow mobilization. After mobilization, gauze soaked with 0.5% cetrimide was packed around the cyst to reduce the chance of dissemination in case of spillage. After that, the cyst was aspirated to collapse and then 0.5% cetrimide was injected inside the cyst—avoiding overdistension of the cyst—and left for 10 min, then aspirated completely. This maneuver was repeated twice. The puncture in the cyst was then widened to allow careful extraction of the germinal membrane and daughter cysts into a preplaced endobag positioned near the cyst to diminish spillage. The cyst wall was then excised and put in the endobag, and the inner epithelial lining was cauterized using monopolar diathermy. The specimen was then sent for histopathology. Suction was further done, and hemostasis was ensured in all cases before a 16 Fr redivac drain was placed near the cyst.

Outcomes measured

Short-term outcomes included:Fluid collection

Blood transfusion

Atelectasis

Surgical site infection

Hospitalization time

Long-term outcomes focused on recurrence of cysts, assessed through postoperative follow-ups at 3 months, 1 year, and 2 years.

Statistical analysis

SPSS version 25 (IBM Corp) was utilized for statistical analysis. To explore relationships between variables, descriptive statistics, such as means and standard deviations, were calculated for continuous variables, while frequencies and percentages summarized categorical variables. Inferential statistics, including t-tests and correlation tests, were employed to assess the significance of relationships among variables. The analysis was conducted with a significance level set at 5%.

Ethical considerations

The Institutional Review Board (IRB) of our university approved the study. The study adhered to ethical guidelines, ensuring confidentiality and compliance with healthcare standards. The reporting of this work conforms to the STROCSS criteria24.

Results

Our study involving 18 patients, with a gender distribution of 38.9% male (n=7) and 61.1% female (n=11), the age range was 15–75 years, with an average age of 33.67 years. The majority (72.22%) of patients presented with left upper quadrant pain (n=13), followed by incidental findings (n=3), left back pain (n=1), and left flank pain (n=1). Medical histories indicated one patient with hypertension and another with both hypertension and diabetes mellitus (Table 3).

Table 3 Patients demographic and clinical characteristics.

No	Sex	Age	Symptom	Cyst diameter	No. cysts	Operative procedure	Postoperative complications	Hospitalization time (day)	
1	F	22	Left upper quadrant pain	9.6	Multiple	LPS	—	5	
2	F	22	Left upper quadrant pain	17.1	1	LSC, LSD	—	4	
3	F	15	Incidental finding	5.6	1	LPS	—	4	
4	F	24	Left upper quadrant pain	13.8	1	LSC, LSD	—	3	
5	M	44	Left upper quadrant pain	4.6	1	LSD	—	4	
6	F	20	Left upper quadrant pain	13.2	1	LPS, LSC	—	6	
7	M	23	Left upper quadrant pain	2.2	1	LSC	—	4	
8	M	35	Left upper quadrant pain	4.9	1	LSD	—	4	
9	F	26	Left upper quadrant pain	5.9	1	LSD	—	5	
10	F	43	Incidental finding	8.2	1	LSD	Fluid collection	9	
11	M	38	Left upper quadrant pain	7.7	1	LSD	Blood transfusion	4	
12	M	31	Left upper quadrant pain	7.2	1	LSD	—	2	
13	F	27	Left upper quadrant pain	7.9	2	LSD	Atelectasis	5	
14	F	75	Left back pain	10.2	1	LSD	Surgical site infection and atelectasis	6	
15	M	44	Left upper quadrant pain	6.6	1	LSD	—	3	
16	F	29	Incidental finding	5.8	2	LSD	—	3	
17	M	33	Left flank pain	8.7	1	LSD	—	2	
18	F	55	Left upper quadrant pain	9.7	1	LSD	—	4	
F, female; LPS, laparoscopic partial splenectomy; LSC, laparoscopic splenic cystectomy; LSD, laparoscopic splenic deroofing; M, male

Preoperative complete blood counts (CBCs) identified eosinophilia in 16.67% of patients (n=3). Indirect hemagglutination (IHA) tests were positive in 72.22% of the cohort (n=13). Imaging assessments varied, with 10 patients undergoing CT scans, 7 receiving both CT scans and abdominal US, and 1 undergoing CT scans and KUB CT. Gharbi’s Classification revealed the distribution of cyst types as: type I (n=2), type II (n=3), type III (n=7), type IV (n=4), and type V (n=2) (Table 3).

The average cyst diameter was 8.3 cm, ranging from 2.2 to 17.1 cm. Most patients had a single cyst (n=15), while two had two cysts, and one had multiple cysts (Figs. 1, 2). Cyst locations varied across the spleen. Surgical approaches included laparoscopic partial splenectomy (n=2), splenic cystectomy (n=1), and cyst deroofing (n=12), with some undergoing combined procedures. Postoperative complications occurred in four patients, including fluid collection, blood transfusion, atelectasis, and surgical site infections. The average hospital stay was 4.28 days (range: 2–9 days). There were no intraoperative complications or postoperative mortalities (Table 3).

Figure 1 Computed tomography scans of a single splenic hydatid cyst. (A) Axial view showing a unilocular cyst (red arrow). (B) Coronal view with the cyst’s position (red arrow). (C) Sagittal view detailing the cyst’s anteroposterior dimension (red arrow).

Figure 2 Computed tomography scans of multiple hydatid cysts in the spleen. (A) Axial view of multiple multilocular hydatid cysts (red arrow). (B) Coronal view showing the cysts’ distribution (red arrow). (C): Sagittal view of the cysts’ vertical spread (red arrow).

A correlation analysis showed a moderate positive association between operative time and estimated blood loss (EBL) (r=0.498, P=0.036). t-tests indicated significant gender differences in cyst diameter and hospitalization time, with longer operation times and hospitalization in patients with postoperative complications and those positive for eosinophilia (P values: 0.028, 0.036, 0.044, 0.012, respectively).

Postoperative follow-ups at three months, one year, and two years revealed two recurrences (11%), consisting of one intrasplenic cystic lesion and another multiloculated cyst. Both patients presented with left upper quadrant (LUQ) pain at 1 year and 2 years, respectively. Statistical analysis using χ2 tests showed no significant association between the types of spleen-preserving surgeries and recurrence, with a P value of 0.186 (Table 4). However, the limited number of recurrences observed warrants further investigation in larger cohorts to confirm these findings.

Table 4 Spleen-preserving surgery outcomes.

	n (%)	
Spleen-preserving surgery	Patient	Short-term complications	Long-term complications	
LPS	2 (11.1)	0	1 (50)	
LSC	1 (5.6)	0	0	
LCD	12 (66.7)	4 (100)	0	
LSC, LCD	2 (11.1)	0	1 (50)	
LPS, LSC	1 (5.6)	0	0	
Total	18 (100)	4 (22.2)	2 (11.1)	
LCD, laparoscopic cyst deroofing; LPS, laparoscopic partial splenectomy; LSC, laparoscopic splenic cystectomy.

Discussion

Jordan, located in the Middle East north of the Gulf region, is distinguished for its vast sheep-rearing territories. A prevalent health concern in Jordan is hydatid disease (HD), a condition endemic to this area25. The exploration and comprehension of HD in Jordan began to intensify in the late 19th century26, with the earliest recorded cases dating to 196627. Further studies have confirmed HD’s endemic status in Jordan, with significant research highlighting an incidence rate ranging from 0.5 to 8.2 cases per 100 000 individuals, averaging 2.9, and identifying the liver and lungs as the most commonly affected organs28,29.

Notably, spleen involvement in HD is infrequent, even in endemic regions, accounting for merely 2–3.5% of all cases30–32. This rarity is attributable to the distinct dissemination mechanisms of Echinococcus Granulosus eggs, which occasionally bypass hepatic and pulmonary defenses to reach the spleen33. Additional, less frequent spread pathways include the portal, lymphatic, and colonic trans-parietal routes12,34,35.

Splenic hydatidosis constitutes a mere 0.5–4% of abdominal hydatid disease cases12,34,35. Its slow, covert growth—typically 0.3–1 cm per year—often results in prolonged undetected periods, sometimes extending to two decades, thereby contributing to significant clinical latency36–38. In our study, 17% of patients were asymptomatic, discovered incidentally during unrelated evaluations. The average age at diagnosis was 33.7 years, with primary symptoms such as left upper quadrant abdominal pain and a palpable mass occurring in 72% of cases.

HD can evolve into complex forms. Complications such as cyst infection, pressure effects, fistula formation, or rupture into adjacent organs can manifest in varied symptoms, including fever, dyspepsia, and anaphylactic shock33,39–42.

Diagnostic approaches typically involve routine blood tests, which often yield normal results. Serological tests, such as hydatid immunophoresis and ELISA, demonstrate sensitivity rates between 85 and 90%33,35,43,44. However, their efficacy in diagnosing extrahepatic localizations is limited to 65%19,45. Our study identified a 28% false-negative rate in indirect hemagglutination tests, suggesting their limitations as sole diagnostic tools.

For imaging, ultrasound and CT scans are pivotal, with ultrasound recommended as the primary examination due to its high sensitivity19,46. CT scans, offering even higher sensitivity, are essential for detailed assessment of the cyst’s characteristics19,46. Our study indicates that a combination of ultrasonography and CT scans yields a diagnostic accuracy of 100%47.

Treatment strategies for splenic hydatid cysts vary between conservative management and surgical intervention. Small, asymptomatic cysts may be managed with anthelmintic drugs, while surgical options, including aspiration and total splenectomy, are primary for more significant cases39,43,49. However, there is no consensus on the optimal surgical approach. Surgeons often opt for surgery in symptomatic or larger cysts, mainly due to the risk of complications such as spontaneous rupture43. Total splenectomy is typically favored for larger, multiple, or symptomatic cysts, particularly those in central or hilar locations or with concurrent involvement of other organs33,43. Despite its efficacy, total splenectomy carries a notable risk of sepsis-related mortality, 4% in children and 1.9% in adults, leading to an increasing preference for spleen-saving surgical procedures17.

Spleen-saving techniques, suitable for small, solitary cysts in peripheral locations, include partial splenectomy, enucleation, deroofing with omentoplasty, and internal or external drainage17,38. These techniques are complemented by Albendazole-based perioperative therapy, aimed at reducing cyst volume and sterilizing disseminated protoscoleces17,47.

Total splenectomy, though technically challenging due to risks like adhesions from chronic pericystic inflammation and potential injury to adjacent organs, stands out as a definitive intervention for preventing recurrences and complications associated with residual cavities in splenic pathologies33,39,41,48. Despite its effectiveness, this procedure harbors inherent risks, including overwhelming post-splenectomy infections (OPSI) and thromboembolic events, with mortality rates of 1.9% in adults and 4% in pediatric patients19,39,49. Total splenectomy is recommended in specific circumstances such as multiple cysts, spleens with pathological changes, ambiguous protruding domes, centroparenchymal cysts, cysts proximal to the splenic hilum, and cases with significant surrounding organ adhesions19,33,40,45,50. It is particularly advocated for managing giant splenic cysts that compromise over 75% of the spleen parenchyma, where substantial parenchymal damage and reduction are observed due to pericystic fibrosis19,41,45.

Spleen-preserving surgery emerges as a preferable choice for polar cysts with accessible domes or in scenarios where adhesions elevate the risk of total splenectomy45. This approach, while having a lower hemorrhage risk compared to partial splenectomy, is not free from risks, such as postoperative suppuration stemming from residual cavities51. Nonetheless, spleen-preserving surgeries, including partial or hemi-splenectomy, are infrequently employed in cases of splenic hydatidosis, with only 24% of 333 surgically managed cases opting for these techniques42,52–55. Partial cystectomy remains a preferred method, although laparoscopic partial splenectomy is limited by risks such as intra-abdominal contamination and cyst rupture. Interestingly, laparoscopic partial splenectomy is contemplated for pre-cancerous and cancerous lesions, indicating a potential for broader application in parasitic and oncological contexts52,55,56.

Comparing the outcomes of partial splenectomy for hydatidosis with other indications such as hereditary spherocytosis reveals distinct challenges, especially in pediatric surgeries. In children, longer operative times are common, and although morbidity rates are low, significant complications can arise in adults, including hemorrhage and infarction necessitating conversion to total splenectomy52–54,57,60. Spleen-preserving surgeries offer the advantage of maintaining immune function and reducing septic postoperative risks. Notably, partial splenectomy, suitable for polar cysts, demands the preservation of at least 25% of splenic tissue but is associated with considerable hemorrhagic risk, particularly in adult patients37,43,61. Furthermore, longer hospital stays are often observed when conservative surgical methods encounter complications37.

The laparoscopic approach is increasingly favored due to its minimally invasive nature and effective outcomes. However, it bears risks such as accidental cyst rupture and anaphylactic shock, potentially leading to spillage of cystic fluid and secondary peritoneal echinococcosis. This technique is particularly suitable for small splenic hydatid cysts but is generally not recommended for multiple or infected cysts19,45,51. Its limitations also encompass the high costs of training and equipment, as well as the necessity for advanced surgical skills.

Puncture Aspiration Injection and Reaspiration (PAIR) has emerged as a promising therapeutic strategy, increasingly favored for its minimally invasive approach. Esteemed for its safety profile, PAIR facilitates reduced hospitalization and mitigates morbidity, markedly outperforming spleen-preserving surgeries and total splenectomy in these aspects50. It serves as a formidable option for patients contraindicated for general anesthesia19,45. Nevertheless, its applicability remains confined to uncomplicated type I or II cysts, preferably smaller than 5 cm in diameter31,41,45. A significant gap in the literature persists, as no comprehensive studies have yet juxtaposed its long-term complications, notably recurrence rates, against other surgical interventions.

In the realm of short-term complications, our study found no significant differences in postoperative hospitalization duration or the emergence of other complications when comparing spleen-preserving procedures with total splenectomies17,36,43. It is noteworthy that 22% of the patients (n=4) encountered postoperative complications, which varied from fluid collection and atelectasis to surgical site infections. Furthermore, there was a case where a patient necessitated a postoperative blood transfusion owing to intraoperative bleeding. These complications were effectively managed and resolved. In contrast, several studies highlight the advantages of spleen-preserving surgeries, especially in laparoscopic techniques. These studies underline the less invasive nature of spleen-preserving surgeries, faster recovery periods, and lower rates of postoperative complications compared to total splenectomy62–64.

In terms of long-term outcomes, research suggests no significant disparity in recurrence rates between spleen-preserving surgeries and total splenectomy17,36,43. Our study noted a recurrence in a mere 2 cases (11%, n=2), a statistically insignificant figure. However, other studies indicate lower rates of complications and recurrence62–64, a finding particularly crucial in pediatric cases due to the spleen’s vital immunological functions.

To conclude, spleen-preserving surgery emerges as a beneficial treatment alternative for splenic hydatid cysts. It upholds immune functionality and minimizes septic risks, especially in pediatric patients. Customizing the surgery based on individual patient needs and cyst characteristics is imperative for optimal outcomes.

Our study, albeit enlightening, is not without inherent limitations that merit acknowledgement. Firstly, the limited sample size, consisting of only 18 patients, cautions against the broad extrapolation of our findings to a wider population. Secondly, being conducted exclusively at two centers, the generalizability of our findings to diverse geographical or clinical settings may be constrained. Furthermore, the absence of a randomized control group within our study framework restricts our ability to definitively attribute outcomes solely to the treatment methods, as other variables could potentially influence the results. Given the rare occurrence of primary splenic hydatid cysts, our study, though offering valuable insights, underscores the pressing need for larger cohorts to corroborate our findings. Lastly, the limited number of recurrences observed underscores the necessity for further exploration through more extensive studies. To enhance the transparency and integrity of our research, we emphasize the importance of presenting our findings honestly and advocating for more comprehensive future studies to fortify these initial observations.

In conclusion, this study comprehensively compares spleen-preserving surgeries with total splenectomy for treating primary splenic hydatid cysts. We demonstrate that techniques like laparoscopic partial splenectomy, cystectomy, and cyst deroofing are effective, with similar recurrence rates to total splenectomy.

These findings are vital for guiding clinical decisions in managing primary splenic hydatidosis. However, given the study’s limited sample size, further research is essential, especially to understand the long-term impacts of these surgical options. Future studies should focus on larger cohorts to validate our results and explore broader implications for patient health.

Ethical approval

Ethical approval for this retrospective study was duly obtained in accordance with standard research ethics guidelines. The study was reviewed and approved by the Institutional Review Board (IRB) of Al-Yarmouk University. The ethical approval encompasses the review and analysis of patient records under the specified conditions of confidentiality and data protection.

Consent

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

Source of funding

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

Author contribution

A.A. and S.A.B.: both authors were significantly involved in the study design, equally contributing to the conceptualization, planning of the research, and manuscript drafting. M.B.H. and M.S.A.-A.: participated in data analysis and interpretation, providing critical insights for the study's findings and manuscript drafting. Y.M.E., H.A.S., R.M.E., M.A.-Z., M.J.A., L.G.A.O., F.M.M., M.S.A., H.B.Z., S.A.A., S.M.A.: engaged in data collection and contributed to the manuscript drafting process. All authors have reviewed and approved the final draft of the manuscript, affirming their agreement with its content and conclusions.

Conflicts of interest disclosure

The authors have no conflicts of interest to disclose relevant to this article.

Research registration unique identifying number (UIN)

Our research was rigorously registered, aligning with the ethical guidelines outlined in the World Medical Association's Declaration of Helsinki (2013), which governs the conduct of research involving human subjects. The registration details are as follows:Registry: ClinicalTrials.gov

Registration ID: NCT06289816

Registration URL: https://clinicaltrials.gov/study/NCT06289816

Guarantor

Saleh A. Ba-shammakh.

Data availability statement

Data are available from the corresponding author upon reasonable request and with permission of the respective hospitals.

Provenance and peer review

Not commissioned, externally peer-reviewed.

A.A. and S.A.B. contributed equally to this study.

Sponsorships or competing interests that may be relevant to content are disclosed at the end of this article.
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References

1 Morris PJ Malt RA . Hydatid disease. Oxford Textbook of Surgery. Oxford University Press; 2000:41–48.
2 Derbel F Mabrouk MB Hamida MB . Hydatid cysts of the liver-diagnosis, complications and treatment. Abdom Surg 2012;5 :105–138.
3 Nunnari G Pinzone MR Gruttadauria S . Hepatic echinococcosis: clinical and therapeutic aspects. World J Gastroenterol 2012;18 :1448–1458.22509076
4 Kayaalp C . Hydatid cyst of the liver. In: Blumgart LH, Belghiti J, Jarnagin WR, et al, eds. Surgery of the Liver, Biliary Tract, and Pancreas, 4th ed.. Saunders; 2006:952–970.
5 Amr SS Amr ZS Jitawi S . Hydatidosis in Jordan: an epidemiological study of 306 cases. Ann Trop Med Parasitol 1994;88 :623–627.7893176
6 Larrieu E Gavidia CM Lightowlers MW . Control of cystic echinococcosis: background and prospects. Zoonoses Public Health 2019;66 :889–899; Epub 2019 Sep 17. PMID: 31529690.31529690
7 Alghamdi FA . Hydatid disease in the Western Region of Saudi Arabia, a university medical center experience. SJPM 2019;04 :747–754.
8 Talib H . Some surgical aspects of hydatid diseases in Iraq. Br J Surg 1968;55 :576–585.5690993
9 Wen H Vuitton L Tuxun T . Echinococcosis: advances in the 21st century. Clin Microbiol Rev 2019;32 :e00075–18.30760475
10 Uriarte C Pomares N Martin M . Splenic hydatidosis. Am J Trop Med Hyg 1991;44 :420–423.2042707
11 Aydin U Yazici P Onen Z . The optimal treatment of hydatid cyst of the liver: radical surgery with a significant reduced risk of recurrence. Turk J Gastroenterol 2008;19 :33–39.18386238
12 Safioleas M Misiakos E Manti C . Surgical treatment for splenic Hydatidosis. World J Surg 1997;21 :374–377.9143567
13 Duthie GS . Hamilton Bailey’s emergency surgery. 13th ed. Edited by Ellis BW, Paterson-Brown S. Br J Surg 2002;89 :631.
14 Tarcoveanu E Bradea C Vasilescu A . Splenic Hydatid Cyst: Open Or Laparoscopic Approach? Surgery 2018;14 :39–44.
15 Polat FR . Hydatid cyst: open or laparoscopic approach? A retrospective analysis. Surg Laparosc Endosc Percutan Tech 2012;22 :264–266.22678325
16 Ellison EC Fabri PJ . Complications of splenectomy: etiology, prevention, and management. Surg Clin North Am 1983;63 :1313–1330.6417808
17 Atmatzidis K Papaziogas B Mirelis C . Splenectomy versus spleen-preserving surgery for splenic echinococcosis. Dig Surg 2003;20 :527–531.14534375
18 Ran B Shao Y Yimiti Y . Spleen-preserving surgery is effective for the treatment of spleen cystic echinococcosis. Int J Infect Dis 2014;29 :181–183.25448337
19 Rasheed K Zargar SA Telwani AA . Hydatid cyst of spleen: a diagnostic challenge. North Am J Med Sci 2013;5 :10–20.
20 Williams NS O’Connell PR McCaskie A . Bailey & Love’s Short Practice of Surgery, 27th Edition. CRC Press; 2018:1176–1187.
21 Gharbi HA Hassine W Brauner MW . Ultrasound examination of the hydatic liver. Radiology 1981;139 :459–463.7220891
22 Tahir F Ahmed J Malik F . Post-splenectomy Sepsis: a review of the literature. Cureus 2020;12 :e6898.32195065
23 WHO Informal Working Group. International classification of ultrasound images in cystic echinococcosis for application in clinical and field epidemiological settings. Acta Trop 2003;85 :253–261.12606104
24 Mathew G Agha R for the STROCSS Group. STROCSS 2021: Strengthening the Reporting of cohort, cross-sectional and case-control studies in Surgery. Int J Surg 2021;96 :106165.34774726
25 Singal R Sandhu KS Mittal A . A giant splenic hydatid cyst. Baylor Univ Med Center Proc 2016;29 :55–57.
26 Vilgrain V . Cystic lesions of the liver. Clin Biol Gastroenterol 2001;25 :B167–B177.
27 Budke CM White C Jr Garcia HH . Zoonotic larval cestode infections: neglected, neglected tropical diseases? PLoS Negl Trop Dis 2009;3 :e319.19238190
28 Kodama Y . Alveolar Echinococcosis: MR findings in the liver 1. Radiology 2003;228 :172–177.12750459
29 Kalovidouris A . CT characterization of multivesicular hydatid cysts. J Comput Assist Tomogr 1985;10 :428–431.
30 Soufi M Kharrasse G Wafae K . Giant isolated hydatid cyst of spleen. JSLR 2015;1 :7.
31 Hoteit A Alimoradi M Aljoubaei M . Primary splenic hydatid cyst treated with laparoscopic spleen preserving surgery: a case report. Int J Surg Case Rep 2020;74 :95–98.32829017
32 Jahic D Kapur E Begic E . Splenic localization of primary hydatid cyst in a 27-year-old sportsman treated by PAIR technique: imaging anatomy assessment. Acta Inform Med 2017;25 :277–279.29284921
33 Culafić DM Kerkez MD Mijac DD . Spleen cystic echinococcosis: clinical manifestations and treatment. Scand J Gastroenterol 2010;45 :186–190.20095883
34 Rodríguez-Leal GA Morán-Villota S Milke-García Mdel P . Splenic hydatidosis: a rare differential diagnosis in a cystic lesion of the spleen. Rev Gastroenterol Mex 2007;72 :122–125.
35 Malik AA ul Bari S Younis M . Primary splenic hydatidosis. Indian J Gastroenterol 2011;30 :175–177.21744126
36 Kaya B Uçtum Y Kutaniş R . Splenic hydatid cyst attacking retroperitoneum. Turkiye Parazitol Derg 2010;34 :193–195.21391192
37 Cebollero MP Cordoba E Escartin J . Hydatic cyst of spleen. J Clin Gastroenterol 2001;33 :89–90.11418806
38 Fernández-Ruiz M Guerra-Vales JM Enguita-Valls AB . Splenic hydatid cyst, a rare location of extrahepatic echinococcosis: report of six cases. Eur J Intern Med 2008;19 :51–53.18206602
39 Belli S Akbulut S Erbay G . Spontaneous giant splenic hydatid cyst rupture causing fatal anaphylactic shock: a case report and brief literature review. Turk J Gastroenterol 2014;25 :88–91.24918138
40 Garg RK Katyal A Bishnoi SR . Isolated hydatid cyst of spleen with cystocolic fistula: an unusual case. Trop Parasitol 2018;8 :114–117.30693220
41 Ben Ameur H Affes N Abdelhedi C . Hydatid cyst of the spleen: Tunisian series of 21 cases. Indian J Surg 2015;77 :515–519.26730056
42 Akbulut S Sogutcu N Eris C . Hydatid disease of the spleen: single-center experience and a brief literature review. J Gastrointest Surg 2013;17 :1784–1795.23949423
43 Balik AA Celebi F Başglu M . Intra-abdominal extrahepatic echinococcosis. Surg Today 2001;31 :881–884.11759882
44 Kalinova K Stefanova P Bosheva M . Surgery in children with hydatid disease of the spleen. J Pediatr Surg 2006;41 :1264–1266.16818060
45 Eris C Akbulut S Yildiz MK . Surgical approach to splenic hydatid cyst: single center experience. Int Surg 2013;98 :346–353.24229022
46 Zhuoli Z Yu Z Liya X . Case report: laparoscopic excision of a primary giant splenic hydatid cyst: literature review. Am J Trop Med Hyg 2019;101 :821–827.31407654
47 Ozogul B Kisaoglu A Atamanalp SS . Splenic hydatid cysts: 17 cases. Indian J Surg 2015;77 :257–260.26730005
48 Meimarakis G Grigolia G Loehe F . Surgical management of splenic echinococcal disease. Eur J Med Res 2009;14 :165–170.
49 Örmeci N Kalkan Ç Karakaya F . Percutaneous treatment with the Örmeci technique for hydatid disease located in the spleen: Single center experience for twenty six years. Turk J Gastroenterol 2018;29 :566–573.30260779
50 Pukar MM Pukar SM . Giant solitary hydatid cyst of spleen—a case report. Int J Surg Case Rep 2013;4 :435–437.23500751
51 Milosavljevic V Veselinovic M Tadic B . Laparoscopic management of initially unrecognized splenic hydatid cysts: a case report and review of the literature. Medicina 2019;55 :771.31817008
52 Vasilescu C Tudor S Popa M . Robotic partial splenectomy for hydatid cyst of the spleen. Langenbecks Arch Surg 2010;395 :1169–1174.20393743
53 Dar MA Shah OJ Wani NA . Surgical management of splenic hydatidosis. Surg Today 2002;32 :224–229.11991507
54 Karadayi K Turan M Sen M . A new technique for partial splenectomy with radiofrequency technology. Surg Laparosc Endosc Percutan Tech 2011;21 :358–361.22002274
55 Akkoyun I Akbiyik F Altunkeser A . Laparoscopic cystectomy for splenic hydatid cyst: a case report. J Pediatr Surg 2011;46 :E1–E3.
56 Uranues S Grossman D Ludwig L . Laparoscopic partial splenectomy. Surg Endosc 2007;21 :57–60.17031738
57 Buesing KL Tracy ET Kiernan C . Partial splenectomy for hereditary spherocytosis: a multi-institutional review. J Pediatr Surg 2011;46 :178–183.21238662
58 Slater BJ Chan FP Davis K . Institutional experience with laparoscopic partial splenectomy for hereditary spherocytosis. J Pediatr Surg 2010;45 :1682–1686.20713220
59 Louis D Pham Duc ML Reix P . Partial splenectomy for portal hypertension in cystic fibrosis related liver disease. Pediatr Pulmonol 2007;42 :1173–1180.17968998
60 Rice HE Crary SE Langer JC . Comparative effectiveness of different types of splenectomy for children with congenital hemolytic anemias. J Pediatr 2012;160 :684–689.22050869
61 Berrada S Ridai M Mokhtari M . Hydatid cysts of the spleen: Splenectomy or conservative surgery? Ann Chir 1991;45 :434–436.1859117
62 Misiakos EP Bagias G Liakakos T . Laparoscopic splenectomy: Current concepts. World J Gastrointest Endosc 2017;9 :428.28979707
63 Costi R Castro Ruiz C Zarzavadjian le Bian A . Spleen hydatidosis treated by hemi-splenectomy: a low-morbidity, cost-effective management by a recently improved surgical technique. Int J Surg 2015;20 :41–45.26074292
64 Arce MA Garcia HH Valcarcel M . Primary giant splenic echinococcal cyst treated by laparoscopy. Am J Trop Med Hyg 2016;94 :161–165.26556833
