Rare presentation of isolated hydatid disease of the breast

  1. Ronal Kori 1,
  2. Sudhir Kumar Jain 2 and
  3. Rehan Nabi Khan 3
  1. 1 General Surgery, Conquest Hospital, Saint Leonards on Sea, UK
  2. 2 Urology, Rotherham General Hospital, Rotherham, UK
  3. 3 General Surgery, Christie NHS Foundation Trust, Manchester, UK
  1. Correspondence to Dr Ronal Kori; ronal_kori@yahoo.co.in

Publication history

Accepted:24 Jun 2021
First published:21 Jul 2021
Online issue publication:21 Jul 2021

Case reports

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Abstract

Isolated hydatid disease of the breast is a rare condition, possessing a diagnostic dilemma for the clinicians. Hydatid disease is common in endemic areas affecting most commonly the liver and lungs. Other organs rarely involved are the kidney, bone and brain. It is caused by the parasite Echinococcus granulosus, widely spread by cattle and sheep. Humans are the accidental host for this organism. We present a similar case of isolated hydatid cyst of the breast, which was diagnosed preoperatively and managed successfully.

Background

Hydatid disease is most prevalent in Andhra Pradesh and Tamil Nadu in India.1 The disease is caused by Echinococcus granulosus species, most commonly found in sheep and cattle. Humans are the accidental intermediate host. The most common site in humans is liver (75%) and lungs (15%).2 Uncommon organs include the spleen (5%), brain and breast. Hydatid disease has been reported in the past, poses a diagnostic dilemma for the clinician. It mimics fibroadenoma, cystic mastopathies, phyllodes tumour or chronic breast abscess. It is essential to identify this condition because the cyst’s antigen can cause a significant anaphylactic reaction if appropriate measures are not taken during surgery.

Ultrasound is beneficial to identify cyst morphology. An ultrasound-guided aspiration can also be performed for cytology to rule out other causes.3 A CT scan can be helpful in undetermined cases. An X-ray of the chest, CT and ultrasound of the abdomen are also essential to rule out systemic disease. Specific antibodies against hydatid antigen (immunodiagnostic test) are also helpful in establishing the diagnosis.

Most patients are benefited from a course of albendazole before the operation to control the systemic disease. The breast cyst is removed intact without rupturing the cyst cavity.

Case presentation

A 32-year-old, non-lactating woman presented to the outpatient department reporting of a progressively enlarging, painless lump in the upper outer quadrant of the left breast for the past 2 years. There was no associated nipple discharge or history of trauma to the breast. No record of fever could be elicited. There is no other swelling noted elsewhere in the body. No family history of breast cancer was present.

On examination, an 8 cm×8 cm, rounded, non-tender, smooth-surfaced swelling was present in the breast’s upper, outer quadrant with a positive transillumination test. There was no discharge from the nipple expressed on pressing. The rest of the left breast, right breast and bilateral axillae were normal.

The blood examination was unremarkable. The ultrasonography of the breast revealed an 8 cm×6 cm cystic swelling with clear fluid and smooth margins (figure 1). A mammogram revealed a simple cyst with posterior acoustic enhancement (Breast Imaging-Reporting And Data Systems II, BIRADS II). Aspiration of cyst showed the serous aspirate, which was found to be acellular on microscopy. The microscopy does show scolices and brood capsule. The aspirate tested negative for Mycobacterium spp. ELISA test performed on aspirated fluid was positive for hydatid antigen. Hydatid serology was positive for the antigen. An ultrasound of the abdomen and plain radiograph of the chest done subsequently taken to rule out concomitant lesions, which was reported normal.

Figure 1

An ultrasound image of breast cyst.

Wide local excision of the cyst was performed (figure 2). Intraoperatively, a 7 cm×6 cm subcutaneous cyst was found, revealing clear, serous fluid and a smooth laminated membrane on subsequent sectioning (figure 3). The histopathology examination showed a laminated membrane with daughter cysts suggestive of hydatid disease (figure 4). Postoperatively, the patient was started on albendazole 200 mg two times per day for 6 weeks. The patient completed the course of albendazole successfully and did not show any recurrence sign at the end of 6 months.

Figure 2

Wide local excision of the cyst.

Figure 3

Opened cyst cavity showing lamellated membrane.

Figure 4

Histopathology of hydatid wall.

Investigations

  1. Ultrasound of the breast: Left breast showed 8 cm×6 cm cystic lesion with clear fluid and smooth margins. The right breast and both the axilla are normal.

  2. Mammogram: Left breast shows a simple cyst with posterior acoustic shadow (BIRADS II). The right breast is normal.

  3. Ultrasound-guided aspiration: Serous aspirate, no cells found.

  4. Hydatid serology: Positive for the E. granulosus antigen.

  5. Ultrasound of the abdomen: No abnormalities were found.

  6. X-ray of the chest: No abnormalities were found.

  7. Histopathology of the specimen:

    • Gross: 7 cm×6 cm cystic lesion with clear fluid. A laminated membrane can be seen lining the cyst wall inside.

    • Microscopy: Laminated membrane with daughter cyst can be seen.

Differential diagnosis

We kept cystic mastopathies as our differential diagnosis because of the patient’s age. The patient does not have pain or signs of inflammation which excludes cystic mastopathies and chronic breast abscess. A suspicion of hydatid cyst was made because the swelling was brilliantly transilluminated. A diagnosis of fibroadenoma was excluded as the swelling is cystic with positive fluctuation. A diagnosis of phyllodes tumour was excluded as the patient had no pain or any risk factors.

Treatment

The patient was taken for the wide local excision of the cystic mass. The patient was started on albendazole 200 mg two times per day for 6 weeks postoperatively.

Outcome and follow-up

She showed no signs of remnant disease or recurrence. The patient was fit and healthy at 6 months of follow-up.

Discussion

Echinococcosis is an infection caused in humans by the larval stage of the E. granulosus complex, E. multilocularis or E. vogeli. The E. granulosus complex parasites producing unilocular cystic lesions are prevalent in those areas where livestock is raised in association with dogs. The definitive hosts are canines that pass eggs in their faeces. Cysts develop after ingesting these eggs in the intermediate hosts such as sheep, cattle, humans, goats, camels and horses for the E. granulosus complex.

The liver is involved in about two-thirds of E. granulosus infections and nearly all E. multilocularis infections. Breast is very infrequently affected, accounting for only 0.27% of all cases.4 According to the work of Barrett and Thomas, the distribution of the disease in the various organs are 60% in the liver, 30% in lungs, 2.5% in kidneys, 2.5% in heart and pericardium, 2% in bone, 1.5% in the spleen, 1% in muscle and 0.5% in the brain.5 6 Clinically, a hydatid cyst in the breast might mimic cysts, fibroadenomas, phyllodes tumours, circumscribed carcinomas, chronic abscesses and cystic mastopathies.4 7 8

No single biochemical test can definitively establish the diagnosis. The primary tests for antibody detection are (1) ELISA, (2) indirect hemagglutination antibody test, (3) latex agglutination test, (4) immunofluorescence antibody test and (5) immunoelectrophoresis. The secondary tests in specialised laboratories are (1) detection of precipitation line (arc 5), (2) identification of IgG subclasses, (3) immunoblotting and (4) PCR. Determining specific antigens and immune complexes with ELISA gives a positive result in >90% of patients. Specific IgE antibodies are demonstrated with ELISA and radioallergosorbent test if the active disease is present. Positivity for the arc 5 antibody test (which involves precipitation during immunoelectrophoresis of the patient’s blood with the antigen) is 91%.

Fine needle aspiration cytology from mammary hydatid cyst can show hooklets or the laminated membrane or, as was the case here, and acellular smear.9 No urticarial or anaphylactic reactions have been reported as a complication of this procedure.10 11

Treatment is based on considerations of the size, location and manifestations of cysts and the patient’s overall health. Surgery has traditionally been the principal definitive method of treatment. Currently, ultrasound staging is recommended for E. granulosus infections.

Patient’s perspective

I was having a swollen left breast for a long time. I was advised to take antibiotics which I took for 2 months but the swelling was not resolved. I came to this hospital following my family member’s recommendation. After taking antibiotics again, the swelling did not resolve. Doctors performed an ultrasound and aspirated some liquid from the breast. I was advised to have surgery to which I agreed. The doctors explained me about the disease, and I was surprised and I agreed to share my case as knowledge to others. I became pain-free after 2 weeks and I thank doctors who have helped me.

Learning points

  • If a patient presents with a brilliantly transilluminating cystic lesion of the breast, a suspicion of hydatid cyst of the breast should be kept in mind.

  • It is essential to investigate these patients for the presence of the disease elsewhere in the body.

  • The author recommends a full dosage of albendazole for a complete elimination of the disease.

  • The author also recommend a trial of aspiration of breast cyst after 3–4 weeks course of albendazole for diagnosis.

Ethics statements

Footnotes

  • Contributors RK: manuscript preparation and editing, submission, gaining consent from the patient. SKJ: chief surgeon, manuscript preparation and management advise. RNK: manuscript editing and advise.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests None declared.

  • Provenance and peer review Not commissioned; externally peer reviewed.

References

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