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SN Comprehensive Clinical Medicine

, Volume 1, Issue 2, pp 96–98 | Cite as

Concomitant Intrathoracic Extrapulmonal and Cervical Hydatid Cyst—a 10-Year Follow-up

  • Laura Chiara GuglielmettiEmail author
  • Claudio Caviezel
  • Sven Hillinger
  • Isabelle Opitz
  • Walter Weder
  • Ilhan Inci
Surgery
  • 116 Downloads
Part of the following topical collections:
  1. Topical Collection on Surgery

Abstract

Hydatosis is a clinical entity known since the time of Hippocrates with Echinococcus granulosus as most common cause of hydatidosis. Humans as well as herbivores are infected as intermediate hosts and the zoonosis is most commonly observed in the liver and lungs. Intrathoracic extrapulmonary cysts are uncommon. We present the rare presentation of concomitant intrathoracic extrapulmonary and cervical hydatid cysts after a follow-up of 10 years. The patient presented with a soft palpable supraclavivular mass and increasing pain. Thoracoscopic exploration (VATS, video-assisted thoracic surgery) revealed a cystic lesion at the apex of the left hemithorax without contact to the lung. We performed a cystotomy and removed the laminated membrane. Later, the cervical hydatid cyst was removed. Five years later, a recurrence of the disease was again resected. Currently, 10 years after initial referral, the patient remains without any signs of active disease. To our knowledge, this is the first report of an intrathoracic extrapulmonary hydatidosis removed by VATS. Our report presents VATS as a surgical option in the treatment of intrathoracic extrapulmonary hydatid disease of the thoracic apex and confirms the feasibility of minimal invasive surgery for intrathoracic hydatid disease.

Keywords

Echinococcosis Hydatid disease VATS 

Introduction

Hydatosis is a clinical entity known since the time of Hippocrates, and it is endemic in some parts of the world. The Mediterranean, South America, and China are reported to have the highest prevalence, with Echinococcus granulosus as most common cause of hydatidosis [1]. Humans as well as herbivores are infected as intermediate hosts and the zoonosis is most commonly observed in the liver and lungs. Intrathoracic extrapulmonary cysts are uncommon with a reported incidence of 3.4–7.5% [2, 3, 4]. We present the rare presentation of concomitant intrathoracic extrapulmonary and cervical hydatid cysts after a follow-up period of 10 years.

Case Presentation

A 30-year-old patient presented with a soft palpable mass in the left supraclavicular region. He was referred after two unsuccessful attempts of fine needle aspiration and reported progressive growth of the lesion and increasing pain over the course of the last 5 months. Computer tomography (CT) revealed 5 × 9-cm infraclavicular and 7.4 × 4.6-cm supraclavicular lesions with erosion of the first rib but no evidence of intrapulmonary lesions (Fig. 1a, b).
Fig. 1

a, b Computer tomography images at initial presentation

Thoracoscopic exploration (VATS, video-assisted thoracic surgery) revealed a cystic lesion at the apex of the left hemithorax without contact to the lung (Fig. 2). We performed a cystotomy and removed the laminated membrane, which confirmed the macroscopic diagnosis of hydatid disease. The left hemithorax was irrigated twice with 2 L of 10% povidone-iodine solution as scolicidal agent. Two days later, the cervical hydatid cyst was removed via left supraclavicular incision. After application of gauzes soaked with 10% povidone-iodine solution around the cyst, the same solution was injected into the cyst cavity to achieve scolicidal effect. Cystotomy was performed and the laminated membrane and daughter cysts were removed.
Fig. 2

VATS view of the intrathoracic cyst with its laminated membrane

Histopathologic examination revealed Echinococcus granulosus. The patient was discharged with a prescription of 10 mg/kg/day albendazole for 3 months.

Five years after initial presentation, the patient showed recurrence of the disease at the apex of the left hemithorax. Due to the increased destruction of the first rib, intraosseous cysts were suspected which are known to be hardly reachable by medical treatment. New CT images demonstrated liquid levels within the cyst as sign of active disease while serology results remained unchanged. Due to the proximity to vital structures and adherence to the brachial plexus, only a macroscopic resection was feasible without considerable morbidity for the patient. The recurrent lesion was again resected (supraclavicular incision and thoracotomy, Fig. 3a, b) and the patient discharged with albendazole.
Fig. 3

Recurrence of the disease with a cervical cyst (a) and underlying destruction of the first rib (b)

Currently, 10 years after initial referral, the patient remains without any signs of active disease.

Discussion and Conclusion

Intrathoracic extrapulmonary hydatid cysts are rare [4] and involvement of the chest wall is even less frequent with reports of 0.09% in a series of 6500 patients [5]. Bone involvement as confirmed after the recurrence and resection of the destroyed rib in our case is reported in 3% of all cases [6].

To our knowledge, this is the first report of an intrathoracic extrapulmonary hydatidosis at the apex of the thoracic cavity removed by VATS. Treatment of pulmonary hydatidosis with VATS has become more popular in the past years with promising results [7] although VATS echinococcectomy was considered a contraindication at the beginning of the 1990s [8]. Findikcioglu et al. published a case series including three mediastinal cases removed by VATS [3].

The same protection from spillage as in open techniques has been performed with careful aspiration of the cyst fluid and removal of the laminated membrane. To prevent possible spillage, the chest cavity was irrigated with a large volume of scolicidal agent.

VATS results in lower morbidity and shorter hospitalization with better esthetic results [8]. In the literature, a follow-up of up to 29 months revealed no recurrence in patients who underwent VATS for mediastinal hydatidosis [3]; nevertheless, our patient suffered from recurrence of the intrathoracic cyst. The recurrence of the disease in our patient is most likely due to bone involvement, which includes cysts that are hardly reached by systemic medical treatment. Recurrent disease after 5 years is in concordance with the reported latency in the literature from 2 to 9 years [2]. Our report presents VATS as a surgical option in the treatment of intrathoracic extrapulmonary hydatid disease of the thoracic apex and confirms the feasibility of minimal invasive surgery for intrathoracic hydatid disease.

Notes

Compliance with Ethical Standards

Conflict of Interest

The authors declare that they have no conflict of interest.

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Copyright information

© Springer Nature Switzerland AG 2018

Authors and Affiliations

  1. 1.Department of Thoracic SurgeryUniversity Hospital ZurichZurichSwitzerland

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