Figure 1
Teaching Case
SectionChest imaging
Case TypeClinical Cases
Authors
Marcela Martin Solon de Pontes;
Guilherme Girardi May;
Antonio Carlos Maciel
Connected authors
1 years, female
The patient arrived at our emergency department with cough, dyspnea, fever and rhinorrhea, showing signs of respiratory distress and decreased vesicular murmur in the left hemithorax. She had a history of repeated respiratory infections since 3 months old. We started antibiotics and requested chest X-Ray and CT.
Anatomopatologic results confirmed both masses were bronchogenic cysts.
Enteric cyst
Abscess
Neoplasms
Infected bullas
Vascular malformations
Hydatidosis
Fungal disease
The X-Ray showed an opacity predominantly atelectatic on the lower third of the lung, as well as a rounded opacity by the left hilum. The trachea is pushed to the right, and there is also a possible compression of the left main bronchus.
The CT demonstrated two hypo-dense nodular images without impregnation, one measuring 2,2x2,2x1,5 cm near the left main bronchus causing impression and reduction of it's caliber, and another one measuring 2,0x1,6x1,5 cm localized near the left hilum. It was suspected of bronchogenic cyst and scheduled a surgery for remotion.
The anatomopathological result confirmed bronchogenic cyst from the material of the mediastinal lesion, and infected bronchogenic cyst with chronic suppurative inflammation and granular tissue from the pulmonary lesion. The patient did not present any complication from the surgery and was released from the hospital without symptoms.
The CT demonstrated two hypo-dense nodular images without impregnation, one measuring 2,2x2,2x1,5 cm near the left main bronchus causing impression and reduction of it's caliber, and another one measuring 2,0x1,6x1,5 cm localized near the left hilum. It was suspected of bronchogenic cyst and scheduled a surgery for remotion.
The anatomopathological result confirmed bronchogenic cyst from the material of the mediastinal lesion, and infected bronchogenic cyst with chronic suppurative inflammation and granular tissue from the pulmonary lesion. The patient did not present any complication from the surgery and was released from the hospital without symptoms.
Bronchogenic cysts represent 18% of all mediastinal masses. They are a result of abnormal bronchial budding of the foregut during embryological formation of the lungs, between the 20th and the 40th day of gestation [1]. The moment this phenomenon happens determines the location of the cysts, whether in the mediastinum (most common) if it happens sooner, or in the lungs if it happens later [5,10]. They are usually diagnosed during adulthood, being rarely seen in elder or infants [2,10]. Although mostly asymptomatic, patients may present complications from extrinsic compression or from infection [1].
We found just six cases of double bronchogenic cysts in the literature so far [2,3,4,9,11,13], and, in only four, both cysts were in separate locations, and none had age less than 33 years old [2,3,9,13]. Our case reveal the diagnosis in an infant (1 year and 8 months) of double cysts, one infected in the left lung parenchyma and another one in the mediastinum causing compressive symptoms.
The main reason to request radiologic images and diagnose correctly is based on the importance of solving or preventing symptoms, given that most patients will develop some type of complication over time, such as infection, superior vena cava obstruction, pneumothorax, atrial fibrillation, hemoptysis, or even malignancy [1,2,5,6,12].
Surgical correction must be done as soon as possible, even if the patient remains asymptomatic, and it should be completely resected in order to avoid recurrence [1,7,8]. In this case, it was decided to perform muscle sparing lateral thoracotomy, removing the infected pulmonary mass with teleobstructive pneumonia from the anterior segment of the left upper lobe, and the cystic lesion in the pulmonary aortic window with reactional aortopulmonary lymph nodes. The patient had no complications and was discharged asymptomatic.
This case is remarkable given it had two concomitant bronchogenic cyst with different characteristics. Whilst one was confined in the mediastinum causing compression of the left bronchi, the second was in the lung parenchyma exhibiting signs of infection. Since there was an obstruction of the air way, the infant manifested recurrent pneumonias in the left lower lobe, which led her to consult a doctor numerous times. Therefore, it is essential to highlight the importance of remembering this potential congenital threat when analysing a radiographic double cystic lesion at a age far from standart.
Written informed patient consent for publication has been obtained.
We found just six cases of double bronchogenic cysts in the literature so far [2,3,4,9,11,13], and, in only four, both cysts were in separate locations, and none had age less than 33 years old [2,3,9,13]. Our case reveal the diagnosis in an infant (1 year and 8 months) of double cysts, one infected in the left lung parenchyma and another one in the mediastinum causing compressive symptoms.
The main reason to request radiologic images and diagnose correctly is based on the importance of solving or preventing symptoms, given that most patients will develop some type of complication over time, such as infection, superior vena cava obstruction, pneumothorax, atrial fibrillation, hemoptysis, or even malignancy [1,2,5,6,12].
Surgical correction must be done as soon as possible, even if the patient remains asymptomatic, and it should be completely resected in order to avoid recurrence [1,7,8]. In this case, it was decided to perform muscle sparing lateral thoracotomy, removing the infected pulmonary mass with teleobstructive pneumonia from the anterior segment of the left upper lobe, and the cystic lesion in the pulmonary aortic window with reactional aortopulmonary lymph nodes. The patient had no complications and was discharged asymptomatic.
This case is remarkable given it had two concomitant bronchogenic cyst with different characteristics. Whilst one was confined in the mediastinum causing compression of the left bronchi, the second was in the lung parenchyma exhibiting signs of infection. Since there was an obstruction of the air way, the infant manifested recurrent pneumonias in the left lower lobe, which led her to consult a doctor numerous times. Therefore, it is essential to highlight the importance of remembering this potential congenital threat when analysing a radiographic double cystic lesion at a age far from standart.
Written informed patient consent for publication has been obtained.
References
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[3] Gillion J, Lagneau M, Balaton A, Convard J, Laroudie M, Hovasse D (2007) Kyste bronchogénique de l’estomac juxtacardial simulant une tumeur stromale, associé à un kyste bronchogénique de l’œsophage sus-diaphragmatique. Gastroenterol Clin Biol 31:1036-8 (PMID: 18166903)
[4] Hashimoto M, Hanaoka J, Kitano H, Oshio Y, Igarashi T, Kataoka Y, et al (2014) Resection of double bronchogenic cysts within the anterior mediastinum; report of a case. Kyobu Geka 67(13):1198-200 (PMID: 27222776)
[5] Kosar A, Tezel C, Orki A, Kiral H, Arman B (2009) Bronchogenic Cysts of the Lung: Report of 29 Cases. Heart, Lung and Circulation 18:214–8 (PMID: 19119078)
[6] Lee T, Tsai IC, Tsai WL, Jan YJ, Lee CH (2005) Bronchogenic Cyst in the Left Atrium Combined with Persistent Left Superior Vena Cava: The First Case in the Literature. AJR 185:116-9 (PMID: 15972410)
[7] Lin F, Zhang C, Cheng K, Dang D, Zhao Y (2015) Thoracoscopic excision of two bronchogenic cysts located in highest upper mediastinum: Report of two cases. Pak J Med Sci 31(3):721-3 (PMID: 26150875)
[8] Liu HS, Li SQ, Cao ZL, Zhang ZY, Ren H (2009) ) Clinical features and treatment of bronchogenic cyst in adults. Chin Med Sci J 24(1):60-3 (PMID: 22263169)
[9] Matsuoka T, Sugi K, Matsuda E, Okabe K, Hirazawa K, Azuma T (2008) Intrapulmonary and mediastinal, double bronchogenic cysts; report of a case. Kyobu Geka 61(5)419-22 (PMID: 27222776)
[10] Michel E. Ribet, MD, Marie-Christine Copin, MD, and Bernard H. Gosselin, MD (1996) Bronchogenic Cysts of the Lung. Ann Thorac Surg 61:1636-40 (PMID: 8651761)
[11] Ohbayashi T, Sudo K, Unno T, Hamada Y, Morita H, Hayashi N, et al ( (1990) A case of double bronchogenic cysts in the mediastinum. Kyobu Geka 43(6):487-90 (PMID: 2385026)
[12] Vilela TT, Daher RT, Nóbrega MDA, Filho CAX, Montandon C, Júnior MEM (2009) 12. Vilela TT, Daher RT, Nóbrega MDA, Filho CAX, Montandon C, Júnior MEM. Radiol Bras 42(1):57–62
[13] Yernault JC, Kuhn G, Dumortier P, Rocmans P, Ketelbant P, Vuyst PD (1986) “Solid” Mediastinal Bronchogenic Cyst: Mineralogic Analysis. AJR 146:73-4 (PMID: 3484413)
Case information
| URL: | https://www.eurorad.org/case/16313 |
| DOI: | 10.1594/EURORAD/CASE.16313 |
| ISSN: | 1563-4086 |
License
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Figure 1
Department of Radiology, ISCMPA, Porto Alegre, Brazil.
Figure 2
Department of Radiology, ISCMPA, Porto Alegre, Brazil.
Figure 3
Department of Radiology, ISCMPA, Porto Alegre, Brazil.
Figure 4
Department of Radiology, ISCMPA, Porto Alegre, Brazil.