Hydatid Cyst involving the Biceps Brachii: A Singular Case Report of a Rare Intramuscular Affliction.

doi:10.21203/rs.3.rs-4971680/v1

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Case Report

Hydatid Cyst involving the Biceps Brachii: A Singular Case Report of a Rare Intramuscular Affliction.

https://doi.org/10.21203/rs.3.rs-4971680/v1

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INTRODUCTION: Hydatid disease (HD) is an endemic zoonotic parasitic infection caused by Echinococcus granulosus. The incidence of HD in the musculoskeletal system and soft tissues is extremely rare, ranging from 0.5% to 4.7%.

CASE REPORT: We present a rare case of primary intramuscular hydatid disease in an agricultural worker, diagnosed through clinical evaluation, serological testing, and imaging, and managed with surgical intervention.

DISCUSSION: Hydatid disease is primarily transmitted through ingestion of eggs, leading to infection by the larval form of Echinococcus. While other transmission modes have been suggested, they lack strong evidence. Hydatid cysts most commonly affect the liver and lungs, but can rarely develop in muscles, which generally provide an unfavourable environment due to high lactic acid levels and muscle contractility. Intramuscular hydatid cysts, particularly in the upper limbs, are especially uncommon, likely due to constant muscle activity in this region.

Ultrasound, MRI, and serological tests are crucial diagnostic tools, with MRI being particularly useful for intramuscular cases. Surgical excision is the primary treatment, with pre- and post-operative anthelmintic therapy reducing recurrence rates.

CONCLUSION: Clinicians should maintain a high index of suspicion for hydatidosis in patients from endemic regions, particularly those with an agricultural background, when assessing musculoskeletal swellings. Early detection ensures timely management.

General Surgery

Parasitology

Infectious Diseases

Primary hydatid cyst

Upper extremity hydatid cyst

Musculoskeletal echinococcosis

Intramuscular hydatid cyst management

Endemic echinococcosis

Case Report

Hydatid disease, or echinococcosis, is an endemic zoonotic parasitic infection caused by Echinococcus granulosus, observed in both developing and developed countries. The highest incidence occurs in regions where sheep, cattle, and dogs are raised, particularly in the Middle East, Central Europe, Australia, South America, and the Mediterranean basin.(1)

Although this parasitic disease can affect any part of the body, the liver (55–70%) and lungs (18–35%) are the most frequently affected organs. Rarely, echinococcus can be located in the brain, ovaries, pancreas, thyroid gland, breast, gallbladder, and heart, with a lower incidence of 8–10%. The musculoskeletal system and soft tissue involvement are extremely rare (0.5–4.7%), even in endemic areas.(2) Even rarer are primary hydatid cysts of the muscle and soft tissue, with most cases secondary to hepatic or pulmonary disease or another primary focus.

The clinical presentation of hydatid disease is often subtle and nonspecific, leading to delays in diagnosis. Typically, it manifests as a painless, non-inflammatory swelling that gradually enlarges over many years, while the patient remains in good general health. However, complications such as nerve compression or infections that mimic an acute abscess or malignant tumor may arise, leading to the eventual discovery of the cysts.(1)

Here, we report a very rare case of a hydatid cyst involving the biceps brachii muscle in a 40-year-old female. This case report follows the SCARE 2018 guidelines.(3)

This report describes a rare case of a primary intramuscular hydatid cyst involving the left upper arm in a 40-year-old female without involvement of any other viscera. She hailed from a rural area, had an agrarian background, and presented to the General Surgery OPD with a swelling involving the antero-medial aspect of her right upper arm. The swelling had been gradually increasing in size for about a year since she first noticed it. The swelling itself was painless, but she complained of mild numbness involving the medial aspect of her forearm. There was no history of trauma, and her family history was unremarkable.

On examination, a globular, non-mobile, non-tender mass measuring approximately 13 cm x 8 cm x 7 cm was observed over the antero-medial aspect of the upper arm (Fig. 1), with no overlying skin changes. Laboratory investigations were normal except for a mildly elevated total leukocyte count and a positive anti-echinococcal IgG test.

Ultrasound (US) revealed a 12 cm × 7 cm cystic lesion on the medial aspect of the right arm, characterized by multiple well-defined internal cysts (Fig. 2). Magnetic Resonance Imaging (MRI) further demonstrated a multi-cystic lesion in the same region, with part of the cyst extending intramuscularly (Fig. 3). Chest X-ray, abdominal ultrasound, and contrast-enhanced CT of the abdomen excluded other primary foci of hydatid disease.

She was diagnosed with primary intramuscular hydatid disease and was scheduled for definitive surgery. Preoperatively, she was started on albendazole 400 mg twice daily for a month, which was continued for three months postoperatively. Pericystectomy was performed with assistance from a General Surgeon under general anaesthesia, and the cyst was dissected out from the intramuscular plane while conserving the overlying neurovascular structures (Fig. 4) (Fig. 5). The wound was closed after thorough saline irrigation, and a 14F closed suction drain was placed in the subcutaneous plane. Histopathological examination of the cyst confirmed the diagnosis of hydatid disease. She was free of any local or systemic recurrence at the three-month follow-up.

Hydatid disease is an endemic parasitic disease caused by the larval form of Echinococcus. E. granulosus and E. multilocularis cause cystic echinococcosis and alveolar echinococcosis in humans.(4)

The most common mode of transmission is through the alimentary tract. The adult tapeworm of Echinococcus granulosus resides in the small intestine of canids (definitive hosts), and human infestation usually results from ingestion of gravid proglottids (eggs) shed in feces. After ingestion, larvae are released in the small intestine, penetrate the intestinal wall with their hooks, and reach the circulatory system, typically lodging in the liver and lungs.(5)

Although airborne transmission and penetration of bronchial venules to reach the heart and systemic circulation have been suggested, they have not been well demonstrated. Direct subcutaneous contamination through injured skin has also been considered, but this theory is unlikely since the hands (most prone to contact via injury) are much less commonly affected than other musculoskeletal parts of the body. The hypothesis that contamination by means other than egg ingestion is not possible is supported by the fact that eggs transform into larvae in the gastrointestinal environment. Therefore, it is generally accepted that parasites might reach organs and tissues other than the liver and lungs through a lymphatic or venous shunt that bypasses the portal filter.(5)

Although primary hydatid cysts of the thigh, gluteal muscles, or other uncommon anatomical sites have been documented, hydatid cysts typically affect the liver and lungs, and the exact pathophysiology of musculoskeletal hydatid disease remains unclear.

Several factors may contribute to the rarity of muscular involvement in hydatid disease. First, muscles are considered an unfavourable environment for Echinococcus due to high lactic acid levels, which inhibit cyst growth. Second, the contractility of muscle tissue restricts the development of intramuscular cysts. Third, the hepatic barrier plays a significant role, as the formation of cysts in muscles would require bypassing the liver and lung filters.(1)

Parasitic cysts tend to develop around muscles of the neck, trunk, and limb roots—areas characterized by reduced muscular activity and increased vascularization. These cysts grow slowly over time and eventually act as space-occupying lesions, leading to pressure-related effects on the surrounding tissues.(6) We hypothesize that the rarity of primary intramuscular hydatid cysts in the upper limb, even compared to other intramuscular sites, is likely due to the constant muscle activity in the upper limb.

While serological tests are available for diagnosing hydatid cysts, a negative test result does not entirely rule out the disease.(6, 7) Ultrasound is highly accurate in detecting cystic membranes, floating membranes, daughter cysts, septa, and hydatid sand.(2, 6) MRI is the preferred examination for suspected intramuscular hydatid disease. The detection of multiloculated polycystic mass lesions on MRI, along with the presence of a two-layered wall of collagen tissue and a vascularized pericyst, aids in the differential diagnosis of cystic lesions.(6)

It is crucial to identify or suspect a hydatid cyst before surgery, as improper handling during resection could lead to the rupture of the cyst and the release of its contents into the systemic circulation, potentially triggering an anaphylactic reaction.(6)

Excision of a hydatid cyst in the muscle can be challenging, particularly in the absence of natural cleavage lines, and is further complicated when the cyst is infected. The cyst’s tight adherence to blood vessels and nerves adds to the difficulty of the procedure. Despite these challenges, pericystectomy remains the preferred treatment for musculoskeletal hydatid cysts. Percutaneous aspiration, infusion of scolicidal agents such as chlorhexidine gluconate, and re-aspiration under ultrasound or CT guidance (PAIR) can be used as an alternative to surgery in inoperable cases.(1)

Thorough intraoperative irrigation of the soft tissues surrounding the cyst with hypertonic saline has been found to reduce the risk of recurrence. Additionally, administering anthelmintic drug therapy both before and after surgery has proven effective in reducing the recurrence rate.(6) Newer alternatives to surgical treatment of intramuscular hydatid cysts include the percutaneous Örmeci technique, in which a solution of 2% alcohol and 1% polidocanol is injected after aspiration from the active cyst.(8)

Intramuscular hydatid disease is an extremely rare entity, particularly in the upper extremities. A high degree of suspicion, especially in endemic areas, is necessary for diagnosis, as blindly proceeding with excision or aspiration cytology may result in local or systemic spillage, fistula formation, or, in the worst cases, anaphylaxis.

INFORMED CONSENT:

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

CONFLICT OF INTEREST:

The authors have no conflict of interest to declare.

SOURCES OF FUNDING:

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

ETHICAL APPROVAL:

No ethical clearance was needed for this case report at our Institution.

ACKNOWLEDGEMENTS:

I would like to express my sincere gratitude to Dr. Arup Kr Mohanta for his invaluable assistance during the surgical procedure, which was crucial to the successful outcome of this case. I am also deeply thankful to Dr. Gautam Das for his expert guidance in writing this manuscript and for his unwavering encouragement to pursue its publication. Finally, I would like to extend my heartfelt thanks to Dr. Satakshi Shome Purkayastha for her immense help with language skills and for meticulously proofreading the article, ensuring its clarity and precision.

Zazo A, Zazo R, Shashaa MN, Alkarrash MS, Brimo Alsaman MZ, Niazi A (2020) Uncommon musculoskeletal femoral hydatid cyst disturbs a female for a year: Case report. Annals Med Surg 55:30–32
Samsami M, Qaderi S, Zebarjadi Bagherpour J, Lucero-Prisno DE (2021) A case report of primary isolated extrahepatic hydatid cyst of the soft tissues of the breast and thigh. Int J Surg Case Rep 79:475–478
Agha RA, Borrelli MR, Farwana R, Koshy K, Fowler AJ, Orgill DP et al (2018) The SCARE 2018 statement: Updating consensus Surgical CAse REport (SCARE) guidelines. Int J Surg 60:132–136
Arslan RS, Çiçek M, Diker S, Küçük Ş (2023) A Case Report of Primary Isolated Extrahepatic Hydatid Cyst of the Thigh. Hamidiye Med J 4(2):152–155
VECCHIO R, VECCHIO V (2020) Transmission ways of Echinococcus granulosus in rare muscular locations of hydatid disease. Annals Med Surg 55:332–333
Lakhanpal V, Badgurjar M, Saxena P (2021) An arcane case report of primary intramuscular hydatid cyst of thigh. Int J Surg Case Rep [Internet]. Mar 1 [cited 2024 Aug 24];80. https://pubmed.ncbi.nlm.nih.gov/33592423/
Mihmanli M, Oguz Idiz U, Kaya C, Demir U, Bostanci O, Omeroglu S et al Current status of diagnosis and treatment of hepatic echinococcosis. 1169 [cited 2024 Aug 24]; http://dx.doi.org/10.4254/wjh.v8.i28.1169
ÖRMECİ N, ASİLLER ÖÖ, YAĞCI C, ÖRMECİ T, ELLİK Z, KALKAN Ç et al (2022) Muscle hydatid diseases: percutaneous treatment with Örmeci technique. J Health Sci Med 5(3):732–739

The authors declare no competing interests.

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Hydatid Cyst involving the Biceps Brachii: A Singular Case Report of a Rare Intramuscular Affliction.

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