Intrathoracic multiple recurrence and bilateral endobronchial rupture of cyst hydatid disease; the rare cause of anaphylaxis

Abstract

Hydatid cyst is a disease caused by Echinococcus Granulosus and Alveolaris. Often it is localized in the liver and lung. The disease is endemic in Turkey, Mediterranean countries, South Africa, the Middle East, South America and New Zeland [1]. The majority of patients are asymptomatic, with rarely seen signs of cough, dyspnea. Cysts may be symptomatic depending on its size, location and complications. Significant physical examination findings are not available. In pulmonary cyst hydatid disease, well defined round consolidations suggesting cystic lesions can be seen on chest X-ray and surgical treatment is the first choice in this patients. In endemic areas, pulmonary hydatid cyst is a rare cause of anaphylaxis. We present two cases who came to emergency department with anaphylaxis due to different presentations of hydatid cyst disease.

Full text

Accepted Manuscript
Intrathoracic multiple recurrence and bilateral endobronchial rupture of cyst hydatid
disease; the rare cause of anaphylaxis
Tayfun Kermenli, Kürşad Yalçınöz, M. Emin Polat
PII: S2213-0071(17)30104-1
DOI: 10.1016/j.rmcr.2017.04.002
Reference: RMCR 426
To appear in: Respiratory Medicine Case Reports
Received Date: 27 March 2017
Accepted Date: 3 April 2017
Please cite this article as: Kermenli T, Yalçınöz Küş, Polat ME, Intrathoracic multiple recurrence and
bilateral endobronchial rupture of cyst hydatid disease; the rare cause of anaphylaxis, Respiratory
Medicine Case Reports (2017), doi: 10.1016/j.rmcr.2017.04.002.
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Title:
Intrathoracic Multiple Recurrence and Bilateral Endobronchial Rupture of Cyst Hydatid
Disease; The Rare Cause of Anaphylaxis
Authors:
Tayfun Kermenli
1
, Kürşad Yalçınöz
2
, M. Emin Polat
3
The name of the institutions:
1
Elbistan State Hospital, Thoracic Surgery Clinic, Kahramanmaraş, Turkey
2
Elbistan State Hospital, Radiology Department, Kahramanmaraş, Turkey
3
Elbistan State Hospital, Emergency Department, Kahramanmaraş, Turkey
Conflict interest:
We report no conflict of interest.
Corresponding author:
Tayfun Kermenli
Address: Kızılcaoba mh. Kışla Cd. Yenibahar Site, 95/A D:14 Post code: 46300
Elbistan / KAHRAMANMARAŞ / TURKEY
Telephone number: +90 530 0150226
Fax number: +90 344 4138008
E-mail address: tayfunkermenli@gmail.com
Meeting Presentation:
Oral presentation, TÜSAD 38th National Annual Meeting, 15-19 October 2016,
Izmir, TURKEY

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Intrathoracic Multiple Recurrence and Bilateral Endobronchial Rupture
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of Cyst Hydatid Disease; The Rare Cause of Anaphylaxis
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Introduction:
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Hydatid cyst is a disease caused by Echinococcus Granulosus and Alveolaris. Often
5
it is localized in the liver and lung. The disease is endemic in Turkey, Mediterranean
6
countries, South Africa, the Middle East, South America and New Zeland (1). The majority of
7
patients are asymptomatic, with rarely seen signs of cough, dyspnea. Cysts may be
8
symptomatic depending on its size, location and complications. Significant physical
9
examination findings are not available. In pulmonary cyst hydatid disease, well defined round
10
consolidations suggesting cystic lesions can be seen on chest X-ray and surgical treatment is
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the first choice in this patients. In endemic areas, pulmonary hydatid cyst is a rare cause of
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anaphylaxis. We present two cases who came to emergency department with anaphylaxis due
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to different presentations of hydatid cyst disease.
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Case 1:
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A 28-year-old woman was brought to emergency department with anaphylaxis. She
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had hypotension (arterial blood pressure 70/45 mmHg), dyspnea, tachypnea (45/min.) and
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tachycardia (135/min.). On physical examination, lung sounds were decreased at the left
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lower zone and there was a left posterolateral thoracotomy scar. Erythrocyte sedimentation
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rate was measured as 57 mm/h in biochemical tests and there were no abnormality in
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hemogram and liver function tests.
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Quickly we were started anaphylaxis treatment to the patient. Inhaled β2 mimetic,
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inhaled corticosteroid, methylprednisolone intravenous (120 mg) and intravenous 0.9% NaCl
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fluid support were given. Then patient was taken to the intensive care unit after improving the
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overall condition.
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Chest X-ray showed left diaphragm is elevated, there were many well defined round
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consolidations suggesting cystic lesions at the left lower zone and adjacent to the mediastinum
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(Figure 1a). Thorax CT scan showed multiple cystic lesions in the left lower lobe (3x3 cm),
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adjacent to the left pericardium (5x5 and 2x2 cm) and adjacent to the upper mediastinum (2x4
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cm) (Figure 1b). Indirect hemagglutination (IHA) test was positive. The abdomen
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ultrasonography showed no cyst in the liver.
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The patient was operated after completion of preoperative preparations. Cysts
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adjacent to the upper mediastinum and left pericardium were removed by cystectomy (Figure
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2). Cystotomy and capitonnage were performed in the left lower lobe cyst. Pathological
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diagnosis was Echinococcus alveolaris. After expanded of lung in postoperative 4th day,
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thorax drain was removed. The patient was discharged on the 10th day of hospitalization.
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Case 2:
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A 42-year-old female patient was admitted to our hospital emergency department
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with a saliva-like vomiting after a severe cough. There was no previous illness story in her
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anamnesis. On physical examination, body temperature was 38.7 ᵒ C, arterial blood pressure
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was measured 65/40 mm/Hg, there were occasional rashes in the body. She had tachycardia
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(132/min), wheezing respiration and dyspnea (oxygen saturation 87%). Crepitations were
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heard at the lower zone of right lung and upper zone of left lung. Hemogram, sedimentation
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and liver function tests were normal. On chest X-ray cystic lesions were detected in the left
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upper zone and right lower zone. Then anaphylactic treatment was started to the patient,
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adrenalin 0.5 mg intramuscular, methylprednisolone 120 mg intravenous, inhaler salbutamol,
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oxygen (5 L/min.) and bolus 0.9% NaCl fluid intravenous support were given. The patient
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was taken to the thoracic surgery clinic after improving the overall condition.
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In the thoracic CT, perforated cystic areas were found in the left upper lobe posterior
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segment and right lower lobe superior segment of the lung (Figure 3). Albendazole
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prophylaxis treatment was started to the patient (2x400 mg oral). Abdomen ultrasonography
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showed a 8 mm diameter cyst in the liver.
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After one week of medical treatment, the patient was operated with bilateral
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Videothoracoscopy (VATS). Cystotomy and capitonnage were performed and parenchymal
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protective technique was applied to the bilateral pulmonary cysts. Pathological diagnosis was
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Echinococcus granulosus.
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Thoracic drains removed on the postoperative 3rd day when bilateral expansion seen
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on chest X-ray. Albendazole therapy was started to the patient 2x400 mg and liver function
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tests were checked every 2 weeks. The patient's 6th months policlinic control was normal.
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Discussion:
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Hydatid lung cysts are one of the most common helminth zoonotic diseases in
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humans, resulting in the infestation of the larval form of Echinococcus granulosus. The adult
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worm live in the small intestine of the dogs, and leaves a lot of eggs to the environment with
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the dogs feces. People who come into contact with contaminated food or water become sick.
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It is seen as endemic in regions such as Middle Eastern countries where animal breeding is
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common like eastern part of our country. There are also reports in the literature suggesting
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that family screening should be performed, especially when a newly diagnosed patient is
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identified in endemic areas (2).
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Echinococci IHA, ELISA IgG, immunoelectrophoresis, indirect fluorescent
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antibody tests can be used in the diagnosis and follow-up of cyst hydatid (3). Chest X-ray and
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thorax CT are the first step in the diagnosis of pulmonary cyst hydatid and MR can be used to
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evaluate the cyst adjacent to the diaphragm (4). The majority of patients are asymptomatic,
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but they can be caused by compression findings according to the location of the cyst, and may
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also cause anaphylaxis and sudden death. There are many reports in the literature that it is the
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cause of anaphylaxis (5-7).
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In all age groups, the first treatment of lung hydatid disease should be surgery.
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Recent developments have also been reported with minimally invasive techniques such as
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VATS (8). Treatment of hydatid cyst with albendazole after surgery is recommended to
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prevent recurrences. In our first case, albendazole treatment was not given after the first
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operation, which posed a risk for recurrence.
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Conclusion:
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Liver and lung are the most common sites of hydatid cysts and abdomen
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ultrasonography should be performed if lung cyst hydatid is detected. Surgery should be the
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first option in the treatment of lung cyst hydatid.
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Treatment of hydatid cyst with albendazole after surgery is recommended to prevent
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recurrences. In our first case, albendazole treatment was not given after the first operation,
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which posed a risk for recurrence. When cyst perforation occurs, albendazole treatment
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should be given without time and anaphylactic precautions should be taken.
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References:
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1. Özdemir A, Bozdemir ŞE, Akbiyik D, Daar G, Korkut S, Korkmaz L, Baştuğ O.
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Anaphylaxis due to ruptured pulmonary hydatid cyst in a 13-year-old boy. Asia Pac
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Allergy. 2015 Apr;5(2):128-31
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2. Karadağlı E, Gürses D, Akpınar F, Herek Ö, Birsen O, Aydın Ç, Four Hydatid Cysts
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in One Family: Is Family Screening Necessary? Turkiye Parazitol Derg. 2015
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3. Koca T, Dereci S, Gençer A, Duman L, Aktaş AR, Akçam M, Akçam FZ, Cystic
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Pulmonary Hydatid Cyst, Turkish J Thorac Cardiovasc Surg 2005;13:62-64
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Figure 1: Chest X-ray shows left diaphragm is elevated (black arrow), cystic lesions at the left
lower zone and adjacent to the mediastinum (a). Non-contrast thorax computed tomography
axial images shows cystic lesions in the left lower lobe, adjacent to the left pericardium and
adjacent to the upper mediastinum (b).
Figure 2: Operation photo shows cysts adjacent to the upper mediastinum and left
pericardium.
Figure 3: Non-contrast thorax CT axial images shows perforated cystic areas in the left upper
lobe posterior segment and right lower lobe superior segment of lungs.

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