Co-occurrence of Cardiac and Cerebral Hydatid Cysts: A Case Report 

K Itumur¹, Y Tamam² , A Karabulut¹, A Guzel³ and N Kilic⁴

Dicle University Faculty Of Medicine^{, 1}Department Of Cardiology, ²Neurology, ³Neurosurgery and ⁴Pathology,  Diyarbakir, Turkey 

Correspondence Address: 

Dr. Yusuf Tamam, Department of Neurology, Faculty of Medicine, Dicle University, 21280 Diyarbakir, Turkey 

E-Mail : yusuta@yahoo.com

SMJ 2006 51(3): 50

Abstract

Cardiac and cerebral hydatid cysts are rarely encountered. In this case, we report a male patient admitted to our hospital with hemiparesis, headache and dysphasia which occurred as a result of complications of both 3,1x3,5 cm single hydatid cyst in the left cardiac ventricle and  multiple cerebral hydatid cysts (approximately 18 particles, the largest being 3× 2,2cm). He had undergone surgery 17 month earlier due to a multiple brain hydatid cysts. Although he had been  treated with albendazole, multiple cerebral hydatid cysts re-appeared 17 months after operation. This was a rare case in which left ventricular intracavitary hydatid cyst occurred together with brain multiple cysts at the same time. Physicians should be alert about the probability of cardiac involvement when a cerebral hydatid cyst is diagnosed and   appropriate investigations should always be conducted.

Key Words: Cardiac hydatid cyst,  cerebral hydatid cysts, hemiparesis, stroke.

Introduction

Hydatid Cyst (HC) is a parasitic disease caused by the larval stages of echinococcus granulosus, and the most common sites of involvement are liver and lungs (1-3). However, cardiac and brain echinococcosis are rare parasitic diseases, accounting for 0,5 to 2 % of all cases (1-7). Hydatid cyst rupture into left-sided chambers may cause systemic emboli, and rupture into right-sided chambers may cause pulmonary emboli (1, 3). Brain involvement generally occur secondary to dissemination from a cardiac source (5, 6).

We would like to report a case with concurrent cerebral and cardiac cysts which presented to our hospital with symptoms of hemiparesia, headache, and dysphasia.

Case Report

A 24-years-old male patient was admitted to our hospital with right hemiparesis associated with headache and dysphasia. Seventeen month before the admission, the patient underwent a brain surgery due to left brain hydatid cyst. Multiple cysts (6 particles) had been removed during the operation without any consequent complications including cyst rupture. The patient received albendazol therapy for 6 months as postoperative chemotherapy to which he was fully compliant. We could not be able to find any echocardiography result performed during that period. However at that time before the surgery, his cardiac examination and electrocardiography findings were within normal limits.  In postoperative period, he had been healthy until one month earlier before his current presentation and did not show any sign or symptom of any other heart or lung diseases. The chest and abdominal examinations were normal. The cardiovascular examination, however, revealed a 1-2 / 6 regurgitant systolic murmur in the left parasternal area. In his neurological examination he had right hemiparesis with all other findings being normal. The electrocardiogram and chest X-ray were within normal limits.

Two-dimensional echocardiographic examination performed by SONOS 4500 in his current referral revealed a cystic lesion with a dimension 3,1×3,5 cm in the left ventricle. The cyst was originating from the left ventricular posterolateral wall, protruding into the left ventricle (LV). There was also mild mitral insufficiency (Figure 1). Transesophageal echocardiography supported the same findings.

----Insert Figure 1 here----

The brain magnetic resonance (MR) imaging revealed multiple cysts (approximately 18 particles, the largest being  3× 2,2cm ) inside the left cerebral hemisphere in different localisations.  The MR demonstrated  a spherical and well-defined, smooth, thin walled, homogeneous cystic lesion with fluid  density similar to the cerebrospinal fluid, without septations or calcification (Figure 2).

-----Figure 2------

Hydatid Cysts both in the brain and heart of the patient were surgically removed without causing any cyst rupture. Hydatid cyst in left ventricle was removed with complete excision of the cyst. Albendazol therapy had been initated in pre-operative period (1 week before operation) which was then continued for 6 months in post-operative period. Histopathological findings confirmed the radiological and  echocardiographic diagnosis of hydatid cyst (Figure 3). Analysis of the aspirated fluid  in cardiac cross section revealed small daughter cysts. The histology of the membrane showed a homogeneously eosinophilic, laminated acellular wall (Figure 3B). Similarly cross sections of brain tissue revealed normal brain tissue and material with homogeneously eosinophilic, laminated acellular walls as well (Figure 3A). 

-----Figure 3------

The clinical examination in the postoperational period showed that the neurological symptoms of patient have significantly improved (i.e. dysphasia and  hemiparesis).  During our six months follow-up, we did not detect any other visceral localisation nor any recurrence of previous hydatid cyst  lesions.

Discussion             

Hydatidosis is a parasitic disease caused by echinococci granulosus, which usually settles in liver and lungs (1,2), and is a serious health issue in undeveloped  and developing countries. The incidence of hydatid disease is 1:2000 in Turkey (2). However, cardiac hydatidosis is a rare parasitic disease, and it has been reported that cardiac involvement is about 0,5-2% of all human hydatidosis (1-3). Similarly cerebral hydatidosis is reported to have a rare incidence (4-9).

Cardiac hydatidosis is often primitive and unique (3,9). Similarly, intracranial hydatid cysts are usually single, and multiple intracranial cysts are rare (4-9). Unlike this patient, hydatid cysts of the left ventricle are usually localised subepicardially, because of the pressure in the cardiac chambers. In the intracavitary cyst,  the risk of intracavitary rupture is higher(3). Echinococcus granulosus reaches the left side of the heart from the coronary circulation, the lymphatics or through the pulmonary veins (2). Visceral metastatic hydatidosis, such as brain, spleen and kidneys , occurs when cyst rupture takes place in the left cavities (1,3). Brain involvement of hydatid disease caused by the echinococcus granulosus parasite is a rare condition generally resulting from the rupture of a cardiac hydatid cyst (4-9).

There are different clinical presentations of cardiac hydatidosis. Main symptoms of an uncomplicated hydatid cysts are chest pain, palpitations and dyspnea (1,2). However, some patients with cardiac echinococcosis as in our patient may remain asymptomatic for many years or have minor nonspecific complaints, but it is associated with an increased risk of lethal complications if left undiagnosed and untreated (1,7,10-12). The prominent symptoms of our patient were hemiparesis, headache and dysphasia .The clinical presentation depends upon the number, site and size of the brain hydatid cysts (9). The presence of multiple brain HC should especially raise suspicion of an embolism arising from a hydatid cyst located in the heart.

In the present case, despite optimal treatment, recurrence of cerebral hydatid cysts could be due to undetected minimal cysts which could not be removed during first brain operation and did not respond adequately to albendazole therapy in postoperative period. It might also be a result of dissemination of a cardiac hydatid cyst which might have been present in left ventricle at the time of initial operation and went unnoticed in previous cardiac physical assessments and laboratory examinations (1-3,9).

In conclusion, this was a rare case with concurrent left ventricular intracavitary hydatid cyst and multiple cerebral  cysts. Though, intracavitary cardiac hydatid cysts are rare and may be asymptomatic, the diagnosis of cerebral hydatid cyst should alert physicians about the probability of cardiac involvement of hydatid cyst, and appropriate investigations should be conducted.

References

 1-Thameur H, Abdelmoula S, Chenik S, et al. Cardiopericardial hydatid cysts. World J Surg  2001;25:58-67

2-Akar R, Eryılmaz S,Yazıcıoğlu L, et al.Surgery for cardiac hydatid disease: an Anatolian experience. Anadolu Kardiyol Derg 2003;3:238-44

3- Kaplan M, Demirtas M, Cimen S, et al. Cardiac hydatid cyst with intracavitary expansion. Ann Thorac Surg 2001;71: 1587–90.  

4-Altınörs N, Bavkek M,  Caner HH, Erdoğan B. Central nervous system hydatidosis in

Turkey: a cooperative study and literature survey analysis of 458 cases . J Neurosurg

2000;93:1–8

 5- Palankar T, Chotai N, Prasad S, el al. Intracerebral hydatid cyst in a child with atrial septal defect. Indian Pediatr 1999;36:406-8.

6-  Turgut M, Benli K, Eryilmaz M. Secondary multiple intracranial hydatid cysts caused by intracerebral embolism of cardiac echinococcosis: an exceptional case of hydatidosis. Case report. J Neurosurg 1997;86: 714–718

 7-Singh NP, Arora SK, Gupta A, et al. Stroke: A rare presentation of card.iac hydatidosis. Neurol India 2003;51:120-121

8- Erashin Y, Mutluer S, Guzelbag E, et al. Intracranial hydatid cysts in children. Neurosurgery 1993;33:219-24.

9- Bukte Y, Kemanoglu S, Nazaroglu H, Ozkan U, Ceviz A, Simsek M. Cerebral hydatid

disease: CT and MR imaging findings. Swiss Med Wkly  2004;134:459–467.

10-Kurtoglu N, Ermeydan C, Akdemir R, Basaran Y, Dindar I. Interventricular septal hydatid cyst. Echocardiography 2000 ;17:693-95.

 11- Sinha PR, Jaipuria N, Avasthey P, Intracardiac hydatid cyst and sudden death in a child. Int J Cardiol  1995;51: 293–95

12- Ulgen MS, Alan S, Karadede A, Aydinalp O, Toprak N. Cardiac hydatid cysts located in both the left ventricular apex and the intraventricular septum: case report. Heart Vessels  2000;15:243-44.

Back to August Contents