Musculoskeletal Hydatid Cyst cases and the success of treatment: A retrospective study

Authors

Abstract

Aim This study aimed to evaluate the clinical features, treatment methods, and outcomes of musculoskeletal hydatid cyst cases treated in our center in the last decade.
Materials and Methods This single-center, retrospective study included patients treated for musculoskeletal hydatid cysts between January 2014 and January 2024. Inclusion criteria were histopathologically confirmed cases treated at our center. Data collected included patient demographics, presenting complaints, cyst location, additional organ involvement, cyst size (measured via MRI or CT), laboratory findings (WBC, eosinophil, CRP, ESR, serology), surgical details, postoperative complications, and recurrence rates. Preoperative imaging (thoracic and abdominal CT) was used to evaluate additional organ involvement. Follow-up included clinical evaluations and an MRI every six months.
Results Thirteen cases (10 females, 3 males; mean age: 45 years) were included. Cysts were located in bones (6 cases: femur, tibia, pelvis) and soft tissues (7 cases: thigh). Additional organ involvement was identified in four cases (liver, ovary, and psoas muscle). Mean cyst diameter was 8 cm (range: 5.4-20 cm). Surgery was performed once in eight cases, while multiple surgeries were required in four cases with bone or pelvic involvement. Postoperative complications included wound problems (2 cases), abscess (1 case), and implant loosening (1 case). Recurrence occurred in four cases. Imaging-guided aspiration and hypertonic saline irrigation were successful in one soft-tissue case. Mean follow-up was 55 months.
Discussion Musculoskeletal hydatid cysts should be considered in endemic regions. Complete excision with wide margins, followed by albendazole therapy, is the most effective approach for musculoskeletal hydatid cysts. Advanced imaging and multidisciplinary management are critical for optimal outcomes.

Keywords

Introduction

Hydatid cyst is a parasitic zoonosis caused by different species of Echinococcus and Echinococcus granulosus (E. granulosus) and is usually seen in Mediterranean countries, the Middle East, India, Australia, and Turkey [1, 2]. Cynical symptoms include a painless, very slowly growing mass [3, 4, 5]. The most common localizations are hepatic (50% to 75%) and pulmonary (20% to 30%), while musculoskeletal involvement is rare (0.5% to 4.7%) [3, 6]. These masses that can be seen in the musculoskeletal system may be located in muscle or bone. Slow-growing hydatid cysts, especially in soft tissues, are usually large at the time of diagnosis as they are late to become symptomatic [7]. The reason for its rare distribution in the muscles may be the presence of lactic acid, which negatively affects the development of parasites [8]. Clinical symptoms are related to the location of the mass. It may cause pain, swelling, pathologic fracture, or neurologic symptoms.
Hydatid cysts located in the musculoskeletal system should be differentiated from other malignant and benign tumors, and their correct treatment is very important [9]. When we look at the literature and daily practical applications, we see that it is not always easy to differentiate hydatid cysts [9]. Radiologic examinations such as Magnetic Resonance Imaging (MRI) and Computed tomography (CT), as well as serologic tests, help the diagnosis. Currently, the most valid treatment is removal of the cyst with appropriate margins without biopsy and subsequent antibiotherapy [10, 11]. Thus, we prevent systemic spread of the cyst and reduce the risk of recurrence.
In our clinic, tumors located in the musculoskeletal system are evaluated by a multidisciplinary council, and surgery is performed in cases diagnosed as hydatid cysts. In the postoperative period, the patient was treated with albendazole for up to 3 months by the Infectious Diseases Clinic; the duration of treatment varied according to the extent of extraskeletal involvement of the disease. This study aimed to investigate cases of hydatid cysts with musculoskeletal involvement and the success of their treatment.

Materials and Methods

This was planned as a single-center, retrospective study. The study was conducted in accordance with the ethical principles outlined in the Declaration of Helsinki. Cases diagnosed with hydatid cysts treated in our clinic between January 2014 and January 2024 were included in the study.
Inclusion criteria: cases whose treatment was performed by our clinic and histopathologically diagnosed; exclusion criteria: patients whose treatment and follow-up were not performed by our clinic and histopathologically diagnosed. Thirteen of the 36 cases evaluated by our clinic with the diagnosis of hydatid cyst met the study criteria.
The age, gender, complaints at initial presentation, cyst location, additional organ involvement, radiologic cyst size, laboratory tests (white blood cell [WBC], eosinophil, C-reactive protein [CRP], erythrocyte sedimentation rate [ESR], serologic test (enzyme-linked immunosorbent assay [ELISA]), surgery, and medical treatment were determined. Radiologically, cyst size was determined as the largest distance measured on MRI or CT sections. All patients underwent abdominal and thoracic CT scans before treatment. They were evaluated for additional organ involvement. All cases were evaluated in a multidisciplinary council, and treatment was initiated. Hydatid cysts with musculoskeletal involvement were removed with appropriate surgical margins, or curettage was performed (figure 1). Curettage was performed using mechanical curettage, high- speed burr, and cauterization until only healthy tissue remained around the cyst (figures 2,3). Postoperative antibiotherapy was administered by the Infectious Diseases department. Postoperative follow-up with MRI and clinical examination was performed every 6 months. Cases with complications or recurrence were treated by us. Any postoperative complications and follow-up periods were evaluated.
Ethical Approval
This study was approved by the Ethics Committee of Bursa Uludağ University (Date: 2024-09-11, No: 2024-14/7).

Results

The study included 13 cases, 10 females and 3 males, with a mean age of 45 years. The most common reason for presentation was pain and swelling, while three patients had difficulty in walking, especially due to lower extremity and pelvic involvement. One patient presented with pain and skin ulceration. Anatomical localizations of the cysts were 6 bone localizations, including 2 femur, 1 tibia, 3 pelvis, and 7 soft tissue localizations, including 7 thighs.
When additional organ involvement was investigated, the liver was found in 3 cases, and the liver, ovary, and psoas muscle in one case. The mean diameter of the cysts was 8 cm (minimum: 5.4 cm, maximum: 20 cm) according to radiologic measurements. In the laboratory tests of the cases, white blood cell (x109cells per L) was 8 (lowest 5,4, highest 11,), eosinophile (x109 cells per L) was 2 (lowest 1,5, highest 3,8), C-reactive protein (mg/L) was 6 (lowest 0,5, highest 15), sedimentation (mm/h) was 35 (lowest 4, highest 59).
Serology tests were positive in all cases. Surgery was performed once in 8 cases, two of which had bone involvement. Two patients with pelvis and femur involvement underwent surgery 3 times, one patient with pelvis involvement underwent surgery 2 times, and one patient with soft tissue involvement in the thigh underwent surgery 4 times. A 35-year-old female patient with a hydatid cyst in the thigh underwent imaging- guided aspiration and irrigation with hypertonic saline solution (figure 4). In the study group with a mean follow-up period of 55 months, hydatid cyst recurrence was observed in 4 cases, skin wound in 2 cases, abscess in the operation area in 1 case, and loosening of the implant in 1 case. Revision surgery was performed on the patient with a loose implant. Debridement and primary suturing were performed in cases with skin wounds. Abscess drainage and antibiotherapy were performed in the patient with an abscess. Albendazole (15 mg/kg for three months) was administered after surgery. We have shown our case examples in Figures 1,2,3.

Discussion

While rare, musculoskeletal hydatid cysts can and should be treated with antibiotics after surgical excision with appropriate margins. The successful treatment results in this study prove that this is an effective approach. However, we must acknowledge that complications and recurrence are possible. Furthermore, the successful treatment of a case with hydatid cyst involvement in the thigh with imaging-guided aspiration and irrigation with hypertonic saline solution demonstrates that there is still a viable treatment option for hydatid cyst cases with soft tissue involvement.
The most reliable quantitative laboratory tests are indirect hemagglutination, ELISA (Enzyme-Linked ImmunoSorbent Assay), and latex agglutination reaction. The most accurate qualitative Western Blot test is also essential. [12, 13, 14]. A negative serologic test does not rule out the diagnosis. The serologic response is dependent on the location, number, and evolution of the cysts. Immunologic reactions also allow for postoperative follow-up and early detection of hydatid recurrence. It is possible to detect parasitic structures after aspiration of cyst fluid, in surgical specimens, or in sputum fluid. All cases in this study group had a positive serology result, which was confirmed by histopathology. Our study group did not include any cases where a non-specific eosinophil increase in blood tests may be present in cases with hydatid cystic involvement of the musculoskeletal system. White blood cell count was above normal limits in only two cases. CRP was above the upper limit in only five cases. Eleven patients had elevated ESR. This study series definitively shows that the increase in ESR value is more prominent than CRP and WBC in cases with musculoskeletal system involvement diagnosed with hydatid cysts. Elevated ESR was also found in the study by Safalı and Aydın [15].
X- ray findings are crucial in diagnosing bone-localized hydatid cysts. However, ultrasound, tomography, and MRI are the gold standard for identifying soft-tissue-localized hydatid cysts. An ultrasound will show intramuscular, hypoechogenic, and various masses in soft tissue. Multivesicular hydatid cysts can and should be seen on MRI. This MRI clearly shows that the morphologic features and signal intensity characteristics vary according to specific stages of cyst development [16]. A simple, viable hydatid cyst stage is indicated by a cyst wall that appears isointense to the fluid within the cyst on T1-weighted images. T2-weighted images show a low signal intensity edge surrounding high signal intensity contents. Abdominal ultrasound (US) or thoraco-abdomino-pelvic computed tomography, usually combined with chest radiography, is used to screen for hydatid cysts, including the primary hepatic and pulmonary filters of the parasite, and to exclude disseminated hydatid cysts [17, 18]. In our study group, a CT scan of the thorax and abdomen was performed in all cases, and the presence of additional organ involvement was examined. Liver involvement was present in four cases, and opinions were obtained from the departments of general surgery, thoracic surgery, and infection before treatment. MRI and CT images were utilized for surgical planning in the preoperative period. Cyst excisions were attempted with appropriate surgical margins, which is the most important step in preventing recurrence or anaphylaxis. Indeed, the importance of an oncologic approach to these masses has been previously emphasized [15].
Hydatid cysts in the musculoskeletal system are rare. However, in endemic regions, they should be considered in tumors with soft tissue involvement. These masses may present late, so it is important to measure them. In this study, the largest diameter of the masses was measured and found to be 8 cm on average (minimum: 5.4 cm, maximum: 20 cm).
Hydatid disease involving bone presents X-ray and CT features similar to those of tuberculosis, metastases, giant cell tumors, and bone cysts. The treatment of hydatid cysts in bone is surgical. The objective is to remove the cyst and surrounding bone, replace bone defects with bone grafts or prostheses, prevent secondary infection, and prevent recurrence. Unfortunately, these goals are rarely fully achieved in this relentless disease. It is a simple fact that most “scolesidal” agents do not kill all microscopic daughter cysts. This means that recurrence is highly likely, and surgery is usually only palliative. While long- term survival is possible, eradicating the disease is challenging, and a cure remains elusive. Effective chemotherapy would be a significant advancement. Our study demonstrates that single surgery is often unsuccessful in hydatid cyst patients with bone involvement. Patients typically present at advanced stages, leading to recurrence and the risk of conversion to osteomyelitis. It must be acknowledged that this study has some limitations. Firstly, it is a single-center retrospective study, which introduces a potential for bias. Secondly, the sample size of this study is relatively small, which may reduce the convincing power of our results. Therefore, multicenter prospective studies are essential to gain a full understanding of the treatment outcomes, complications, and recurrence rates of patients with musculoskeletal hydatid cysts.

Limitations

The limitations of this study are that the number of patients included in the study is not large enough to provide clear statistical results. In addition, the differences between the cases in terms of anatomical location make it difficult to present clear common results.

Conclusion

In societies where hydatid cysts are endemic, soft tissue or bone malignancies must be included in the differential diagnosis. Complete excision of the mass with wide margins after investigation of additional organ involvement and subsequent Albendazole treatment is the most successful treatment method for hydatid cysts with musculoskeletal manifestations. MRI and CT are essential in surgical planning and the investigation of additional organ involvement.

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