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Interactive Cardiovascular and Thoracic Surgery 2:395-397(2003)
© 2003 European Association of Cardio-Thoracic Surgery


Case report - Thoracic general

Multiple primary chest wall hydatid cysts associated with spinal canal involvement

Dalokay Kiliça,*, Bulent Erdoganb, Mehmet Ali Habesogluc and Ahmet Hatipoglud

a Department of Thoracic Surgery, Baskent University, School of Medicine, Baskent University Hospital, 01250 Adana, Turkey
b Department of Neurosurgery, Baskent University, School of Medicine, Baskent University Hospital, 01250 Adana, Turkey
c Department of Chest Disease, Baskent University, School of Medicine, Baskent University Hospital, 01250 Adana, Turkey
d Department of Thoracic Surgery, Baskent University, School of Medicine, 06490 Ankara, Turkey

* Corresponding author. Tel.: +90-322-3272727; fax: +90-322-3271274
dalokay7{at}hotmail.com

Received April 6, 2003; received in revised form April 30, 2003; accepted May 16, 2003


    Abstract
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
Primary multiple chest wall hydatid cysts associated with spinal canal involvement through an intervertebral foramen is an uncommon clinical entity. We present a 54-year-old man who underwent cystotomy and total resection of ribs five through seven via a left posterolateral thoracotomy followed by Th5–Th6 anterolateral partial pediculotomies for removal of cysts in the spinal canal. Although spinal reconstruction was not required, the chest wall defect was repaired with mersilene mesh–methyl methacrylate sandwich graft. Hydatid disease should be considered in the differential diagnosis of mass lesions located in the chest wall. In cases of spinal canal involvement, detailed visualization of spinal canal utilizing MRI and/or CT is essential for planning surgical approach.

Key Words: Hydatid cyst; Chest wall; Spinal canal; Chest wall reconstruction


    1. Introduction
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
Hydatid disease is a parasitosis caused by Echinoccosus granulosus and has been known since Hippocrates [1]. Living in a rural area is an important risk factor for this disease. It is still an endemic disease in some regions of the world, particularly in many Mediterranean countries [2–4]. In adults the lung is the second most common organ for hydatid disease after the liver [1]. A hydatid cyst can be encountered in almost every part of the body, however, a chest wall hydatidosis, especially of the rib is very rare. Multiple, some of them complicated chest wall hydatid disease, and its association with spinal canal extension in a patient constitutes an extremely rare presentation.

We report a case of primary chest wall hydatid disease with secondary spinal canal involvement successfully treated by resection of the involved ribs followed by removal of cysts via Th5–Th6 partial pediculotomies, and chest wall reconstruction with mersilene mesh–methyl methacrylate (MM-MM).


    2. Case report
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
A 54-year-old-man was admitted to our hospital with chest pain and swelling of 3 months duration on the left chest wall. His medical and familial histories were unremarkable except for living in a rural area. On physical examination, he had an irregular, palpable mass over his left lateral chest wall 10x7 cm in diameter, and his neurological examination was completely normal. Routine laboratory studies yielded normal values. A computed tomography (CT) scan revealed hypodense, complicated cystic lesions with lobulations and septations located in the chest wall causing destruction of ribs five through seven, and underlying pectoral muscle (Fig. 1). Magnetic resonance imaging (MRI) revealed numerous daughter vesicles extending to spinal canal through intervertebral foramen (Fig. 2). The liver, spleen, brain, and other parts of spinal canal were entirely normal at a detailed work-up by ultrasound and/or CT.



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Fig. 1 CT scans of thorax (a,b) showing hypodense, complicated cystic lesions with lobulations and septations located in the chest wall causing destruction of ribs five through seven, and underlying pectoral muscle.

 


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Fig. 2 Axial T2W (a) and coronal enhanced T1W (b) Magnetic resonance images showing spinal canal involvement by numerous daughter vesicles through intervertebral foramen at level of Th5–Th6. No bony structure destruction is noted.

 
Total resection of the fifth, sixth, and seventh ribs was performed via a left posterolateral thoracotomy. Some of the cysts in the chest wall were thick-walled and lobulalated. After performing rib resection and cystotomy, the cysts, which extended into spinal canal through intervertebral foramen were removed unruptured via Th5–Th6 pediculotomies. No vertebral bone destruction was noted. Although, chest wall defect was repaired with MM-MM, pediculotomy defect did not require stabilization. Histological examinations confirmed a diagnosis of hydatid cyst. Postoperative course was uneventful, and the patient was discharged on day 10 post-surgery. Albendazole (10 mg/kg daily; Andozid, Biofarma Istanbul, Turkey) was administered postoperatively.


    3. Discussion
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 
Intrathoracic extrapulmonary hydatid disease constitutes 7.4% of all hydatid diseases [2]. Among intrathoracic extrapulmonary hydatid cysts, 55% of the cysts are located in the fissure, 18% within the parietal pleura, 14% in the chest wall, 4.5% in the mediastinum, and 4.5% in the diaphragm [2]. When an intrathoracic extrapulmonary hydatid cyst lies in the vicinity of bone structures, it may result in bone and muscle destruction. In case of extrapulmonary involvement by hydatid cysts, rib involvement account for only 2.3% [3]. The disease can also be seen in the musculoskeletal system in 1–4% [5]. There is only one chest wall involvement in the report of 842 hydatid cysts in the series of Qian [6].

Reconstruction of chest wall defect with rigid material must be performed in cases where more than two ribs are resected in order to prevent paradoxical respiration caused by chest wall instability [7]. In this case, we used MM-MM for repairing chest wall defect.

When we reviewed literature, we found out that primary chest wall hydatid disease associated with spinal canal involvement had not been reported previously. Although they are nonneoplastic, in the spinal canal, hydatid cysts are generally space-occupying lesions that produce signs and symptoms due to their mass effect. Although there were numerous thin-walled cysts located extradurally at the thoracic levels 5–6, our patient had no neurological deficits. Surgical removal is the first and most effective option for treating spinal canal hydatid cysts [8]. The aim of the surgery is removal of the entire cyst(s) without rupture. Despite therapy, the disease frequently relapses with progressive destruction of the vertebral column and neurological deterioration. Altinors et al. [8] in their cooperative study, reported the rate of recurrence in patients with spinal cysts to be 24.32%, and the incidence of paraplegia due to recurrent disease to be 25–45%. However, the possibility of cure is high when there is no bony involvement [9]. In the presented case, there was no vertebral bony destruction, all of the thin-walled daughter vesicles were removed without rupturing them, and after performing pediculotomies of Th5 and Th6, spinal stabilization was not required. Albendazole treatment was started in postoperative stage for preventing late recurrences.

Detailed preoperative evaluation with MRI and CT scanning is essential in localizing the lesions and planning the surgical approach [8]. CT and MRI not only have tremendously increased diagnostic specificity, but have also allowed us to visualize the multiplicity, viability, and outermost margin of the cysts, thus helping the spinal surgeon to optimize surgical approach to the lesion. CT is helpful especially in visualization of details of bone destruction and spinal canal involvement.

In conclusion, hydatid disease should be considered in the differential diagnosis of mass lesions located in the chest wall. After performing multiple rib resections, the chest wall defect should be reconstructed despite the challenge of repair. MRI and CT should be considered in suspected cases of spinal canal involvement. Total removal of the cysts without rupture appears to be effective in the prevention of late recurrences.

doi:10.1016/S1569-9293(03)00108-7


    References
 Top
 Abstract
 1. Introduction
 2. Case report
 3. Discussion
 References
 

  1. Aletros H, Symbos PN. Hydatid disease of the lung. Shields TW, Lo Cicero J III, Poon RB. General thoracic surgery. Philadelphia, PA: Lippincott; 2000. p. 1113–1122
  2. Oguzkaya F, Akcali Y, Kahraman C, Emirogullari N, Bilgin M, Sahin A. Unusually located hydatid cysts: intrathoracic but extrapulmonary. Ann Thorac Surg. 1997;64:334–337[Abstract/Free Full Text]
  3. Burgos L, Baquerizo A, Munoz W, de Aretxabala X, Solar C, Fonseca L. Experience in the surgical treatment of 331 patients with pulmonary hydatidosis. J Thorac Cardiovasc Surg. 1991;102:427–430[Abstract]
  4. Ozdemir N, Akal M, Kutlay H, Yavuzer S. Chest wall echinococcosis. Chest. 1994;105:1277–1279[Abstract]
  5. Merkle AM, Schulte M, Vogel J, Tornczak R, Reiber A, Kern P, Goerich J, Brambs HJ, Sokiranski R. Musculoskeletal involvement in cystic echinococcosis: report of eight cases and review of the literature. AJR Am J Roentgenol. 1997;168:1531–1534[Abstract/Free Full Text]
  6. Qian ZX. Thoracic hydatid cysts: a report of 842 cases treated over a thirty-year period. Ann Thorac Surg. 1988;46:342–346[Abstract]
  7. McCormack PM. Use of prosthetic materials in chest wall reconstruction. Surg Clin North Am. 1989;69:965–975[Medline]
  8. Altinors N, Bavbek M, Caner HH, Erdogan B. Central nervous system hydatidosis in Turkey: a cooperative study and literature survey analysis of 458 cases. J Neurosurg. 2000;93:1–8[Medline]
  9. Hamdan TA, Al-Kaisy MA. Dumbbell hydatid cyst of the spine: case report and review of the literature. Spine. 2000;25:1296–1299[CrossRef][Medline]




This Article
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Right arrow Articles by Kiliç, D.
Right arrow Articles by Hatipoglu, A.
Related Collections
Right arrow Lung - other
Right arrow Chest wall


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