Congenital pulmonary airway malformation in adult: report of two rare cases and literature review

Massimo Galia, Domenico Albano, Massimo Midiri, Cesare Gagliardo, Tommaso Vincenzo Bartolotta, Alberto Bruno, Antonio Sidoti Pinto, Claudio Leto, Dario Giambelluca, Giuseppe Cutaia, Giuseppe Salvaggio, Leonardo Salvaggio

Research output: Contribution to journalArticle

Abstract

Congenital pulmonary airway malformations (CPAMs) are a heterogeneous group of congenital, non-hereditary lesions of the lung, cystic and non-cystic type with clear developmental anomalies and evident malformative changes, which derive from the excessive proliferation of the tubular bronchial structures. We described two rare cases of type IV CPAM detected in adult patients with different clinical presentation: one of these cases was symptomatic, complaining of cough and hemoptysis. Conversely, the other case was reported as incidental findings in patient with abdominal pain without respiratory symptoms. Chest computed tomography (CT) scan was performed showing voluminous cystic formations with fibrotic septa within the lesions in both patients. The patient with hemoptysis was subjected to video assisted thoracoscopic lobectomy, while the other one was hospitalized to perform biopsy, which confirmed the diagnosis of CPAM. All patients were advised to follow-up. The incidence of type IV CPAM in adult patients is probably underestimated; the knowledge of the CT features of CPAM is crucial for a correct diagnosis and an appropriate management of asymptomatic patients, which may be conservative.
Original languageEnglish
Pages (from-to)45-51
Number of pages7
JournalDefault journal
Volume58
Publication statusPublished - 2019

All Science Journal Classification (ASJC) codes

  • Pulmonary and Respiratory Medicine

Cite this

Congenital pulmonary airway malformation in adult: report of two rare cases and literature review. / Galia, Massimo; Albano, Domenico; Midiri, Massimo; Gagliardo, Cesare; Bartolotta, Tommaso Vincenzo; Bruno, Alberto; Sidoti Pinto, Antonio; Leto, Claudio; Giambelluca, Dario; Cutaia, Giuseppe; Salvaggio, Giuseppe; Salvaggio, Leonardo.

In: Default journal, Vol. 58, 2019, p. 45-51.

Research output: Contribution to journalArticle

Galia, M, Albano, D, Midiri, M, Gagliardo, C, Bartolotta, TV, Bruno, A, Sidoti Pinto, A, Leto, C, Giambelluca, D, Cutaia, G, Salvaggio, G & Salvaggio, L 2019, 'Congenital pulmonary airway malformation in adult: report of two rare cases and literature review', Default journal, vol. 58, pp. 45-51.
Galia, Massimo ; Albano, Domenico ; Midiri, Massimo ; Gagliardo, Cesare ; Bartolotta, Tommaso Vincenzo ; Bruno, Alberto ; Sidoti Pinto, Antonio ; Leto, Claudio ; Giambelluca, Dario ; Cutaia, Giuseppe ; Salvaggio, Giuseppe ; Salvaggio, Leonardo. / Congenital pulmonary airway malformation in adult: report of two rare cases and literature review. In: Default journal. 2019 ; Vol. 58. pp. 45-51.
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abstract = "Congenital pulmonary airway malformations (CPAMs) are a heterogeneous group of congenital, non-hereditary lesions of the lung, cystic and non-cystic type with clear developmental anomalies and evident malformative changes, which derive from the excessive proliferation of the tubular bronchial structures. We described two rare cases of type IV CPAM detected in adult patients with different clinical presentation: one of these cases was symptomatic, complaining of cough and hemoptysis. Conversely, the other case was reported as incidental findings in patient with abdominal pain without respiratory symptoms. Chest computed tomography (CT) scan was performed showing voluminous cystic formations with fibrotic septa within the lesions in both patients. The patient with hemoptysis was subjected to video assisted thoracoscopic lobectomy, while the other one was hospitalized to perform biopsy, which confirmed the diagnosis of CPAM. All patients were advised to follow-up. The incidence of type IV CPAM in adult patients is probably underestimated; the knowledge of the CT features of CPAM is crucial for a correct diagnosis and an appropriate management of asymptomatic patients, which may be conservative.",
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AU - Galia, Massimo

AU - Albano, Domenico

AU - Midiri, Massimo

AU - Gagliardo, Cesare

AU - Bartolotta, Tommaso Vincenzo

AU - Bruno, Alberto

AU - Sidoti Pinto, Antonio

AU - Leto, Claudio

AU - Giambelluca, Dario

AU - Cutaia, Giuseppe

AU - Salvaggio, Giuseppe

AU - Salvaggio, Leonardo

PY - 2019

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N2 - Congenital pulmonary airway malformations (CPAMs) are a heterogeneous group of congenital, non-hereditary lesions of the lung, cystic and non-cystic type with clear developmental anomalies and evident malformative changes, which derive from the excessive proliferation of the tubular bronchial structures. We described two rare cases of type IV CPAM detected in adult patients with different clinical presentation: one of these cases was symptomatic, complaining of cough and hemoptysis. Conversely, the other case was reported as incidental findings in patient with abdominal pain without respiratory symptoms. Chest computed tomography (CT) scan was performed showing voluminous cystic formations with fibrotic septa within the lesions in both patients. The patient with hemoptysis was subjected to video assisted thoracoscopic lobectomy, while the other one was hospitalized to perform biopsy, which confirmed the diagnosis of CPAM. All patients were advised to follow-up. The incidence of type IV CPAM in adult patients is probably underestimated; the knowledge of the CT features of CPAM is crucial for a correct diagnosis and an appropriate management of asymptomatic patients, which may be conservative.

AB - Congenital pulmonary airway malformations (CPAMs) are a heterogeneous group of congenital, non-hereditary lesions of the lung, cystic and non-cystic type with clear developmental anomalies and evident malformative changes, which derive from the excessive proliferation of the tubular bronchial structures. We described two rare cases of type IV CPAM detected in adult patients with different clinical presentation: one of these cases was symptomatic, complaining of cough and hemoptysis. Conversely, the other case was reported as incidental findings in patient with abdominal pain without respiratory symptoms. Chest computed tomography (CT) scan was performed showing voluminous cystic formations with fibrotic septa within the lesions in both patients. The patient with hemoptysis was subjected to video assisted thoracoscopic lobectomy, while the other one was hospitalized to perform biopsy, which confirmed the diagnosis of CPAM. All patients were advised to follow-up. The incidence of type IV CPAM in adult patients is probably underestimated; the knowledge of the CT features of CPAM is crucial for a correct diagnosis and an appropriate management of asymptomatic patients, which may be conservative.

UR - http://hdl.handle.net/10447/356449

UR - https://www.minervamedica.it/en/journals/minerva-pneumologica/article.php?cod=R16Y2019N02A0070

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