|Year : 2013 | Volume
| Issue : 2 | Page : 155-157
Primary hydatid cyst of broad ligament
Aruna Bhattacharya1, Rama Saha2, Sudipan Mitra3, Pamela Nayak2
1 Department of Biochemistry, Institute of Post-Graduate Medical Education and Research, Kolkata, West Bengal, India
2 Department of Pathology, Institute of Post-Graduate Medical Education and Research, Kolkata, West Bengal, India
3 Department of Medicine, North Bengal Medical College & Hospital, West Bengal, India
|Date of Submission||08-Apr-2013|
|Date of Web Publication||26-Nov-2013|
35, A K.G., School Road, East Anandapuri, P.O. Nonachandanpukur, Barrackpur, Kolkata 700 122, West Bengal
Source of Support: None, Conflict of Interest: None
| Abstract|| |
Cystic lesions of the female pelvis are common. Clinically, symptomatic lesions are mostly ovarian in origin and neoplastic in nature. Considerable diagnostic dilemma may be encountered if clinical, radiological, and estimation of serum markers failed to classify the origin and nature of such cysts. One such exceptional case is being described where a 35-year-old female presented with a rapidly growing cystic mass in lower abdomen, clinically suspicious of malignancy. Investigations failed to identify the nature. On laparotomy, excision of the mass was done. Suprisingly histopathological examination identified the lesion as hydatid cyst arising from the broad ligament. Female genital tract hydatidosis is uncommon and in most cases the involvement is secondary. Primary hydatid disease of female genital tract is even very rarer and generates considerable diagnostic difficulty. A significant clinical suspicion is necessary in the differential diagnosis of pelvic cystic diseases to identify such a rare entity.
Keywords: Broad ligament, pelvic cyst, primary hydatid cyst
|How to cite this article:|
Bhattacharya A, Saha R, Mitra S, Nayak P. Primary hydatid cyst of broad ligament. Trop Parasitol 2013;3:155-7
One of the most common conditions encountered in gynecological practice is the cystic lesion of the pelvis. Clinically symptomatic cysts originate mostly from the ovaries and are mostly benign.  Cysts may also arise from the Fallopian tube More Details, broad ligaments and other surrounding structures. Considerable diagnostic dilemma may be encountered if clinical, radiological, and estimation of serum markers failed to classify the origin and nature of such cysts.
Hydatid disease, a common public health problem, is caused by the tapeworm of the genus Echinococcus. It involves most commonly liver and lung.  Involvement of female reproductive system is rare and in most of the cases primary site of hydatid disease can be traced to the liver and other abdominal organ.  Thus, the involvement is secondary. Primary broad ligament hydatid cyst is extremely rare. Hydatid cyst in the broad ligament may simulate a pedunculated fibroid  or a parovarian cyst. 
The most important factor in its diagnosis is the awareness of the possibility of hydatid disease especially in the endemic area.
According to study conducted by Bickers et al.  over a 20 year study in an endemic area among 532 cases of hydatid cyst, 12 cases of pelvic hydatid cyst were noted out of which, only two were identified in broad ligament (incidence of 0.37%). One such exceptional case of primary broad ligament hydatid cyst is being described hereby as the condition is rare and clinically difficult to identify.
| The Case|| |
A 35-year-old housewife from East Medinipur District of West Bengal presented with difficulty in micturition over 1 year, heaviness of abdomen, and enlarging abdominal lump over 4 months. She was menstruating regularly. No history of contact with dog or other pet animal could be obtained.
On general examination, the lady was thin built, well-nourished, and not anemic. There was no lymphadenopathy. On systemic examination, respiratory, and cardiovascular systems were normal. On abdominal examination, a cystic mass was palpable in the right lower abdomen with restricted mobility. There was no hepatosplenomegaly or ascitis. Pervaginal examination located the mass high up in right fornix.
Routine blood and urine examination, chest X-ray revealed no abnormality. Serum CA 125, alfa foeto protein (AFP), carcino embryonic antigen (CEA) was within the normal limits. Ultrasonography (USG) of whole abdomen, suggested right sided ovarian cystic mass without any septation, contains non-echogenic mass measuring 10 cm × 8 cm. Computed tomography (CT) Scan abdomen revealed a hypoechoic mass of 10 cm × 10 cm × 6 cm dimension of pelvic origin.
As no definite proof of origin and nature of the mass could be derived by clinical, radiological, and laboratory investigations, a laparotomy was performed as lesion was suspicious of a malignant ovarian cyst. Total abdominal hysterectomy with bilateral oophorectomy was performed. Perioperatively, a cyst showing dense adhesion with omentum, uterus, and colon was noted. Whole resected specimen was sent for the histopathological examination.
On gross examination of specimen, an elongated cyst measuring 13 cm × 6 cm × 4 cm on right side of the uterus arising from the broad ligament. On opening, albuminous egg white membrane like structure came out [Figure 1].
Microscopic examination revealed a cyst wall composed of lamellated ectocyst with chronic inflammatory cell infiltrate in pericyst, consistent with the diagnosis of hydatid cyst of broad ligament [Figure 2].
|Figure 2: Photomicrograph shows wall of hydatid cyst with mild inflammation (inset shows lamellated membrane of hydatid cyst wall)|
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Post-operatively, the recovery was uneventful. The patient underwent further clinical and the radiological evaluation for detection of hydatid disease of other organs. As no evidence of such lesion was identified, a final diagnosis of Primary broad ligament hydatid cyst was rendered.
| Conclusions|| |
Hydatid cyst involving female pelvis is rare and isolated primary pelvic hydatid disease is still uncommon. According to clinicoepidemiological study conducted by Akhtar et al.,  among 117 cases of hydatid disease in central India there is only 1 case of pelvic hydatidotosis. In Libiya, where the hydatid disease is endemic, 14 cases of pelvic hydatidotosis were encountered in one hospital department between 1971 and 1979. 
A solitary pelvic hydatid cyst is considered primary when no other such lesion is found in liver, lung, kidney, spleen, and peritoneal cavity.  In our case also, we did not find such lesion anywhere else other than broad ligament. Primary broad ligament hydatid cyst is very rare. Bellil et al.,  conducted a study among 265 cases of extrapulmonary hydatid disease over 18 year period from 1990 to 2007, where only one case of broad ligament cyst was documented. Arora et al.,  Roychowdhury et al.,  Kriplani and Kriplani  have reported few isolated case of primary broad ligament hydatid cyst from different parts of India.
In female, genital organs are reported to be the most affected areas in pelvis, which can be related to their relatively high vascularity and true invasions from connective tissue of peritoneum of Douglas and suspensory ligaments. , Pelvic hydatid disease can be presented with vague abdominal pain due to irritation, swelling, menstrual irregularities, infertility, and pressure symptoms involving the adjacent organs (bladder, ureters, rectum, and vascular structures). , In our case, patient presented with abdominal lump with heaviness of the abdomen and difficulty in micturition.
Pre-operative diagnosis of primary pelvic hydatidosis may be difficult as the lesion mimics malignancy.  A preliminary diagnosis by cytology or fine needle aspiration cytology (FNAC) may not always be helpful as thick mucin aspirated with scanty cellularity may simulate laminated membrane of hydatid cyst and easily be misdiagnosed as ectocyst of hydatid disease.  In our case, FNAC and other serological tests were not carried out as there was no suspicion of hydatid cyst in the pelvic region.
Diagnosis of hydatid cyst of female pelvic organ is usually possible only after operation.  In our case, also it was diagnosed only after exploratory laparotomy, which was carried out with provisional diagnosis of right sided ovarian tumor.
However, pre-operative diagnosis of pelvic hydatid diseases can be possible with imaging study such as USG, CT Scan by noting features such as multilocular appearance, cyst wall calcification, a fluid level from hydatid sand, and the ultrasonic water lily sign.  However, in the present case, USG and CT Scan could not detect such abnormality. According to Gharbi's classification Type 1 Cystic echinococcosis (CE) and World Health Organization Type 1 CE, present as unilocular cystic lesion with an anechoic content, which is similar to ours. Non-complicated Isolated type I pelvic CE mimicking ovarian tumor Type 1 CE is a diagnostic dilemma as diagnostic criteria of advanced disease are not seen in Type 1.  The condition was identified post-operatively with gross and histopathological examination of the cyst. Therefore, a high degree of clinical suspicion is essential in the differential diagnosis of pelvic cystic diseases to identify such a rare entity especially in endemic areas and hence that the patient can be managed accordingly.
Apart from surgery, three other treatment modalities are established. 
- Chemotherapy with albendazole/mebendazole
- Percutaneous drainage sterilization (PAIR)
- Observation of inactive Echinococcal stages 'watch and wait' approach.
In our case, as the diagnosis was incidental, pre-operative medication with albendazole was not possible; hence, surgery followed by chemotherapy with albendazole was the treatment of option.
| References|| |
|1.||Pudasaini S, Lakhey M, Hirachand S, Akhter J, Thapa B. A study of ovarian cyst in a tertiary hospital of Kathmandu valley. Nepal Med Coll J 2011;13:39-41. |
|2.||Bickers WM. Hydatid disease of the female pelvis. Am J Obstet Gynecol 1970;107:477-83. |
|3.||Georgakopoulos PA, Gogas CG, Sariyannis HG. Hydatid disease of the female genitalia. Obstet Gynecol 1980;55:555-9. |
|4.||Chakrabarti D, Sarkar SK, Ghosal KK. Hydatid cyst of the broad ligament. J Indian Med Assoc 1974;63:193-4. |
|5.||Dadhwal V, Kochhar S, Vimala N, Singh MK, Mittal S. An unusual cyst in the broad ligament. Trop Doct 2002;32:114-5. |
|6.||Akhtar MJ, Khanam N, Rao S. Clinico epidemiological profile of hydatid diseases in Central India, a retrospective and prospective study. Int J Biol Med Res 2011;2:603-6. |
|7.||Rahman MS, Rahman J, Lysikiewicz A. Obstetric and gynaecological presentations of hydatid disease. Br J Obstet Gynaecol 1982;89:665-70. |
|8.||Selvaggi FP, Fabiano G, Santacroce S, Traficante A. A retrovesical echinococcal cyst: Unusual cause of acute urinary retention. Eur Urol 1978;4:60-2. |
|9.||Bellil S, Limaiem F, Bellil K, Chelly I, Mekni A, Haouet S, et al. Descriptive epidemiology of extrapulmonary hydatid cysts: A report of 265 Tunisian cases. Tunis Med 2009;87:123-6. |
|10.||Arora M, Gupta CR, Jindal S, Kapoor N. An unusual case of hydatid cyst of broad ligament.JIACM 2005;6:86-7. |
|11.||Roychowdhury A, Bandopadhyay A, Bhattacharya P, Mitra RB. An unusual case of primary intrapelvic hydatid cyst. Indian J Pathol Microbiol 2010;53:588-9. |
|12.||Kriplani A, Kriplani AK. Primary echinococcal cyst of the broad ligament (a case report). J Postgrad Med 1989;35:57-8. |
|13.||Terek MC, Ayan C, Ulukuþ M, Zekioðlu O, Ozkinay E, Erhan Y. Primary pelvic hydatid cyst. Arch Gynecol Obstet 2000;264:93-6. |
|14.||Gupta A, Kakkar A, Chadha M, Sathaye CB. A primary intrapelvic hydatid cyst presenting with foot drop and a gluteal swelling: A case report. J Bone Joint Surg Br 1998;80:1037-9. |
|15.||Martín-Serradilla JI, Guerrero-Peral AL, Marcos-Alvarez R, Mohamed-Buskri A, Hernández-Carrero MT, Zatarain-Vázquez MT. Lumbar plexopathy secondary to pelvic hydatid cyst. Rev Neurol 2002;34:944-9. |
|16.||Varedi P, Saadat Mostafavi SR, Salouti R, Saedi D, Nabavizadeh SA, Samimi K, et al. Hydatidosis of the pelvic cavity: A big masquerade. Infect Dis Obstet Gynecol 2008;2008:782621. |
|17.||Godara R, Dhingra A, Ahuja V, Garg P, Sen J. Primary peritoneal hydatidosis: Clinically mimicking carcinoma of ovary. Internet J Gynecol Obstet 2007;7:2. |
|18.||Pedrosa I, Saíz A, Arrazola J, Ferreirós J, Pedrosa CS. Hydatid disease: Radiologic and pathologic features and complications. Radiographics 2000;20:795-817. |
|19.||Vural M, Yalcin S, Yildiz S, Camuzcuoglu H. Isolated type I pelvic cystic echinococcosis mimicking ovarian tumor. N Am J Med Sci 2011;3:289-91. |
|20.||Hosch W, Junghanss T, Werner J, Düx M. Imaging methods in the diagnosis and therapy of cystic echinococcosis. Rofo 2004;176:679-87. |
[Figure 1], [Figure 2]