Congenital Cystic Adenomatoid Malformation (CCAM) Type II: A Rare Case of Sudden Infant Death
Abstract
1. Introduction
2. Case Presentation
Histopathological Investigation
3. Discussion
4. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Acknowledgments
Conflicts of Interest
References
- Sirithangkul, S.; Chuengchitraks, S.; Staworn, D.; Laohapand, C.; Silarat, T. Late Manifestation of Congenital Cystic Adenomatoid Malformation with Lung Abscess: A Case Report. J. Med. Assoc. Thail. 2010, 93 (Suppl. 6), 223–227. [Google Scholar]
- Strumillo, B.; Jóźwiak, A.; Palka, A.; Szaflik, K.; Piaseczna-Piotrowska, A. Congenital Cystic Adenomatoid Malformation-Diagnostic and Therapeutic Procedure: 8-Year Experience of One Medical Centre. Kardiochirurgia i Torakochirurgia Polska 2018, 15, 10–17. [Google Scholar] [CrossRef] [PubMed]
- Viana, R.; Carvalho, L.; Santos, C. Congenital Pulmonary Airway Malformation: A Rare Diagnosis in Adulthood. Respirol. Case Rep. 2022, 10, e0895. [Google Scholar] [CrossRef] [PubMed]
- Koga, H.; Ochi, T.; Hirayama, S.; Watanabe, Y.; Ueno, H.; Imashimizu, K.; Suzuki, K.; Kuwatsuru, R.; Nishimura, K.; Lane, G.J.; et al. Congenital Pulmonary Airway Malformation in Children: Advantages of an Additional Trocar in the Lower Thorax for Pulmonary Lobectomy. Front. Pediatr. 2021, 9, 722428. [Google Scholar] [CrossRef] [PubMed]
- Stocker, J.T.; Madewell, J.E.; Drake, R.M. Congenital Cystic Adenomatoid Malformation of the Lung. Classification and Morphologic Spectrum. Hum. Pathol. 1977, 8, 155–171. [Google Scholar] [CrossRef]
- Widiyanto, M.K.; Ismail, D.; Hayuningrat, P.K.; Muhammad, F. Congenital Cystic Adenomatoid Malformation Type i in a Newborn with Sepsis: A Case Report. Malays. J. Med. Health Sci. 2021, 17, 179–181. [Google Scholar]
- Kotecha, S.; Barbato, A.; Bush, A.; Claus, F.; Davenport, M.; Delacourt, C.; Deprest, J.; Eber, E.; Frenckner, B.; Greenough, A.; et al. Antenatal and Postnatal Management of Congenital Cystic Adenomatoid Malformation. Paediatr. Respir. Rev. 2012, 13, 162–171. [Google Scholar] [CrossRef]
- Stocker, J.T. Cystic Lung Disease in Infants and Children. Fetal. Pediatr. Pathol. 2009, 28, 155–184. [Google Scholar] [CrossRef]
- Argeitis, J.; Botsis, D.; Kairi-Vassilatou, E.; Hasiakos, D.; Papakonstantinou, K.; Kondi-Pafiti, A. Congenital Cystic Adenomatoid Lung Malformation: Report of Two Cases and Literature Review. Clin. Exp. Obstet. Gynecol. 2008, 35, 76–80. [Google Scholar]
- Yamashita, A.; Hidaka, N.; Yamamoto, R.; Nakayama, S.; Sasahara, J.; Ishii, K.; Mitsuda, N. In Utero Resolution of Microcystic Congenital Cystic Adenomatoid Malformation after Prenatal Betamethasone Therapy: A Report of Three Cases and a Literature Review. J. Clin. Ultrasound 2015, 43, 451–457. [Google Scholar] [CrossRef]
- Miller, J.A.; Corteville, J.E.; Langer, J.C. Congenital Cystic Adenomatoid Malformation in the Fetus: Natural History and Predictors of Outcome. J. Pediatr. Surg. 1996, 31, 805–808. [Google Scholar] [CrossRef] [PubMed]
- Kersten, C.M.; Hermelijn, S.M.; Mullassery, D.; Muthialu, N.; Cobanoglu, N.; Gartner, S.; Bagolan, P.; Mesas Burgos, C.; Sgrò, A.; Heyman, S.; et al. The Management of Asymptomatic Congenital Pulmonary Airway Malformation: Results of a European Delphi Survey. Children 2022, 9, 1153. [Google Scholar] [CrossRef] [PubMed]
- Mardy, C.; Blumenfeld, Y.J.; Arunamata, A.A.; Girsen, A.I.; Sylvester, K.G.; Halabi, S.; Rubesova, E.; Hintz, S.R.; Tacy, T.A.; Maskatia, S.A. In Fetuses with Congenital Lung Masses, Decreased Ventricular and Atrioventricular Valve Dimensions Are Associated with Lesion Size and Clinical Outcome. Prenat. Diagn. 2020, 40, 206–215. [Google Scholar] [CrossRef]
- Pomara, C.; Fineschi, V. Forensic and Clinical Forensic Autopsy. An Atlas and Handbook—2nd Edition; Pomara, C., Fineschi, V., Eds.; CRC Press: Boca Raton, FL, USA, 2020; ISBN 9780367330712. [Google Scholar]
- Mackenzie, T.C.; Guttenberg, M.E.; Nisenbaum, H.L.; Johnson, M.P.; Adzick, N.S. A Fetal Lung Lesion Consisting of Bronchogenic Cyst, Bronchopulmonary Sequestration, and Congenital Cystic Adenomatoid Malformation: The Missing Link? Fetal Diagn. Ther. 2001, 16, 193–195. [Google Scholar] [CrossRef] [PubMed]
- Seear, M.; Townsend, J.; Hoepker, A.; Jamieson, D.; McFadden, D.; Daigneault, P.; Glomb, W. A Review of Congenital Lung Malformations with a Simplified Classification System for Clinical and Research Use. Pediatr. Surg. Int. 2017, 33, 657–664. [Google Scholar] [CrossRef]
- Müller, A.M. Pathology of Pediatric Lung Diseases and Malformations|Pathologie von Pädiatrischen Lungenerkrankungen Und Fehlbildungen. Pneumologe 2015, 12, 52–58. [Google Scholar] [CrossRef]
- Annam, V.; Korishetty, S.I.; Yelikar, B.R.; Hippargi, S.B.; Shivalingappa, D.B. Bilateral Congenital Cystic Adenomatoid Malformation, Stocker Type III with Associated Findings and Review of Literature. Indian J. Pathol. Microbiol. 2010, 53, 331–333. [Google Scholar] [CrossRef]
- Kurland, G.; Deterding, R.R.; Hagood, J.S.; Young, L.R.; Brody, A.S.; Castile, R.G.; Dell, S.; Fan, L.L.; Hamvas, A.; Hilman, B.C.; et al. An Official American Thoracic Society Clinical Practice Guideline: Classification, Evaluation, and Management of Childhood Interstitial Lung Disease in Infancy. Am. J. Respir. Crit. Care Med. 2013, 188, 376–394. [Google Scholar] [CrossRef]
- Deutsch, G.H.; Young, L.R.; Deterding, R.R.; Fan, L.L.; Dell, S.D.; Bean, J.A.; Brody, A.S.; Nogee, L.M.; Trapnell, B.C.; Langston, C.; et al. Diffuse Lung Disease in Young Children: Application of a Novel Classification Scheme. Am. J. Respir. Crit. Care Med. 2007, 176, 1120–1128. [Google Scholar] [CrossRef]
- Nogee, L.M. Interstitial Lung Disease in Newborns. Semin. Fetal Neonatal Med. 2017, 22, 227–233. [Google Scholar] [CrossRef]
- Khan, H.; Kurup, M.; Saikia, S.; Desai, A.; Mathew, M.; Sheikh, A.; Goonasekera, C.D.A. Morbidity after Thoracoscopic Resection of Congenital Pulmonary Airway Malformations (CPAM): Single Center Experience over a Decade. Pediatr. Surg. Int. 2021, 37, 549–554. [Google Scholar] [CrossRef] [PubMed]
- Hermelijn, S.M.; Mackenbach, M.J.; van Horik, C.; Ciet, P.; Wolf, J.L.; von der Thüsen, J.H.; Wijnen, R.M.H.; Tiddens, H.A.W.M.; Schnater, J.M. Quantitative CT Imaging Analysis to Predict Pathology Features in Patients with a Congenital Pulmonary Airway Malformation. J. Pediatr. Surg. 2022, 57, 1567–1572. [Google Scholar] [CrossRef] [PubMed]
- Ibrahim, H.; Hafiz, A.; Rabiu, B.; Abdullahi, U.; Ghidazuka, Y.; Aliyu, I. Spontaneous Resolution of a Congenital Multicystic Lung Lesion in a Newborn. Med. J. Dr. D.Y. Patil Vidyapeeth 2019, 12, 352–355. [Google Scholar] [CrossRef]
- Garg, S.; Singh, R.S.; Singh, H. Congenital Cystic Adenomatoid Malformation of the Lung in Adults: Report of Two Cases and Review of the Literature. Indian J. Thorac. Cardiovasc. Surg. 2018, 34, 488–490. [Google Scholar] [CrossRef] [PubMed]
- Sharma, A.; Pannu, M.S.; Lata, N.; Dhillon, S.P.S.; Singh, N.; Sareen, A. Rare Presentation of Congenital Cystic Adenomatoid Malformation of the Lung. J. Nepal Paediatr. Soc. 2016, 36, 91–93. [Google Scholar] [CrossRef]
- Badour, M.; Hussain, B.; Hammed, A.; Sawssan, A.; Saeed, F. A Rare Case of Congenital Cystic Adenomatoid Malformation: Mimics Pneumonia Manifestations. Ann. Med. Surg. 2021, 66, 102433. [Google Scholar] [CrossRef]
- Hermelijn, S.M.; Elders, B.B.L.J.; Ciet, P.; Wijnen, R.M.H.; Tiddens, H.A.W.M.; Schnater, J.M. A Clinical Guideline for Structured Assessment of CT-Imaging in Congenital Lung Abnormalities. Paediatr. Respir. Rev. 2021, 37, 80–88. [Google Scholar] [CrossRef]
- Zhang, Z.-J.; Huang, M.-X. Children with Congenital Cystic Adenomatoid Malformation of the Lung CT Diagnosis. Int. J. Clin. Exp. Med. 2015, 8, 4415–4419. [Google Scholar]
- Sessa, F.; Esposito, M.; Messina, G.; di Mizio, G.; di Nunno, N.; Salerno, M. Sudden Death in Adults: A Practical Flow Chart for Pathologist Guidance. Healthcare 2021, 9, 870. [Google Scholar] [CrossRef]
- di Nunno, N.; Patanè, F.G.; Amico, F.; Asmundo, A.; Pomara, C. The Role of a Good Quality Autopsy in Pediatric Malpractice Claim: A Case Report of an Unexpected Death in an Undiagnosed Thymoma. Front. Pediatr. 2020, 8, 8. [Google Scholar] [CrossRef]
- Schiavone, S.; Mhillaj, E.; Neri, M.; Morgese, M.G.; Tucci, P.; Bove, M.; Valentino, M.; di Giovanni, G.; Pomara, C.; Turillazzi, E.; et al. Early Loss of Blood-Brain Barrier Integrity Precedes NOX2 Elevation in the Prefrontal Cortex of an Animal Model of Psychosis. Mol. Neurobiol. 2017, 54, 2031–2044. [Google Scholar] [CrossRef] [PubMed]
- Neri, M.; Othman, S.M.; Cantatore, S.; de Carlo, D.; Pomara, C.; Riezzo, I.; Turillazzi, E.; Fineschi, V. Sudden Infant Death in an 8-Month-Old Baby with Dengue Virus Infection: Searching for Virus in Postmortem Tissues by Immunohistochemistry and Western Blotting. Pediatr. Infect. Dis. J. 2012, 31, 878–880. [Google Scholar] [CrossRef] [PubMed]
- Bertozzi, G.; Maglietta, F.; Baldari, B.; Besi, L.; Torsello, A.; di Gioia, C.R.T.; Sessa, F.; Aromatario, M.; Cipolloni, L. Mistrial or Misdiagnosis: The Importance of Autopsy and Histopathological Examination in Cases of Sudden Infant Bronchiolitis-Related Death. Front. Pediatr. 2020, 8. [Google Scholar] [CrossRef] [PubMed]
- Santacroce, R.; Santoro, R.; Sessa, F.; Iannaccaro, P.; Sarno, M.; Longo, V.; Gallone, A.; Vecchione, G.; Muleo, G.; Margaglione, M. Screening of Mutations of Hemophilia A in 40 Italian Patients: A Novel G-to-A Mutation in Intron 10 of the F8 Gene as a Putative Cause of Mild Hemophilia a in Southern Italy. Blood Coagul. Fibrinolysis 2008, 19, 197–202. [Google Scholar] [CrossRef] [PubMed]
- Sintim-Damoa, A.; Cohen, H.L. Fetal Imaging of Congenital Lung Lesions with Postnatal Correlation. Pediatr. Radiol. 2022, 52, 1921–1934. [Google Scholar] [CrossRef] [PubMed]
- Zhu, P.; Cheng, K.; He, M.; Wang, Y.; Shen, P.; He, K.; Xu, C.; Zhang, B.; Liu, Z. Diagnostic Value of Congenital Pulmonary Airway Malformation Volume Ratio for Fetal Hydrops Due to Congenital Lung Malformations: A Systematic Review and Meta-Analysis. Orphanet J. Rare Dis. 2022, 17, 1–10. [Google Scholar] [CrossRef] [PubMed]
- Huang, Y.-Y.; Chang, Y.-J.; Chen, L.-J.; Lee, C.-H.; Chen, H.-N.; Chen, J.-Y.; Chen, M.; Hsiao, C.-C. Survival of Hydrops Fetalis with and without Fetal Intervention. Children 2022, 9, 530. [Google Scholar] [CrossRef]
- Jeong, B.-D.; An, S.-A.; Lee, M.-Y.; Won, H.-S.; Han, M.; Yoon, H.; Lee, J.-H.; Cho, Y.-J. Comparison of the Prognostic Factors of Fetuses with Congenital Pulmonary Airway Malformations According to Type. J. Ultrasound Med. 2020, 39, 2243–2252. [Google Scholar] [CrossRef]
- Mon, R.A.; Johnson, K.N.; Ladino-Torres, M.; Heider, A.; Mychaliska, G.B.; Treadwell, M.C.; Kunisaki, S.M. Diagnostic Accuracy of Imaging Studies in Congenital Lung Malformations. Arch. Dis. Child. Fetal Neonatal Ed. 2018, 104, F372-2018. [Google Scholar] [CrossRef]
- Chaouachi, S.; ben Hamida, E.; ben Fraj, N.; Blibèche, S.; Marrakchi, Z. Congenital Cystic Adenomatoid Malformation of the Lung: Two Cases Report. Tunis. Med. 2011, 89, 55–58. [Google Scholar]
- Shupe, M.P.; Kwon, H.P.; Morris, M.J. Spontaneous Pneumothorax in a Teenager with Prior Congenital Pulmonary Airway Malformation. Respir. Med. Case Rep. 2014, 11, 18–21. [Google Scholar] [CrossRef] [PubMed][Green Version]
- Barikbin, P.; Roehr, C.C.; Wilitzki, S.; Kalache, K.; Degenhardt, P.; Bührer, C.; Schmalisch, G. Postnatal Lung Function in Congenital Cystic Adenomatoid Malformation of the Lung. Ann. Thorac. Surg. 2015, 99, 1164–1169. [Google Scholar] [CrossRef] [PubMed]
- Isabellea, V.; Wildhaber Barbara, E.; Ueli, M.; Nicolas, R.; Daniel, T.; Dietmar, C.; Juerg, B.; Constance, B.-A.; Isabelle, R.-M. A Swiss Database and Biobank to Better Understand and Manage Congenital Lung Anomalies. Swiss Med. Wkly. 2019, 149, w20081. [Google Scholar] [CrossRef]
- Davenport, M.; Warne, S.A.; Cacciaguerra, S.; Patel, S.; Greenough, A.; Nicolaides, K. Current Outcome of Antenally Diagnosed Cystic Lung Disease. J. Pediatr. Surg. 2004, 39, 549–556. [Google Scholar] [CrossRef]
- Stanton, M.; Davenport, M. Management of Congenital Lung Lesions. Early Hum. Dev. 2006, 82, 289–295. [Google Scholar] [CrossRef]
- Tsai, A.Y.; Liechty, K.W.; Hedrick, H.L.; Bebbington, M.; Wilson, R.D.; Johnson, M.P.; Howell, L.J.; Flake, A.W.; Adzick, N.S. Outcomes after Postnatal Resection of Prenatally Diagnosed Asymptomatic Cystic Lung Lesions. J. Pediatr. Surg. 2008, 43, 513–517. [Google Scholar] [CrossRef]
- Jaffé, A.; Chitty, L.S. Congenital Cystic Adenomatoid Malformations May Not Require Surgical Intervention. Arch. Dis. Child. Fetal Neonatal Ed. 2006, 91, F464. [Google Scholar] [CrossRef][Green Version]
- Chetcuti, P.A.J.; Crabbe, D.C.G. CAM Lungs: The Conservative Approach. Arch. Dis. Child Fetal Neonatal Ed. 2006, 91, F463. [Google Scholar]
Developmental diseases | Genetic mutations |
---|---|
|
|
Growth disorders | Undefined etiology |
|
|
Publisher’s Note: MDPI stays neutral with regard to jurisdictional claims in published maps and institutional affiliations. |
© 2022 by the authors. Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
Share and Cite
Salerno, M.; Sessa, F.; Cocimano, G.; Roccuzzo, S.; Esposito, M.; Pomara, C. Congenital Cystic Adenomatoid Malformation (CCAM) Type II: A Rare Case of Sudden Infant Death. Children 2022, 9, 1830. https://doi.org/10.3390/children9121830
Salerno M, Sessa F, Cocimano G, Roccuzzo S, Esposito M, Pomara C. Congenital Cystic Adenomatoid Malformation (CCAM) Type II: A Rare Case of Sudden Infant Death. Children. 2022; 9(12):1830. https://doi.org/10.3390/children9121830
Chicago/Turabian StyleSalerno, Monica, Francesco Sessa, Giuseppe Cocimano, Salvatore Roccuzzo, Massimiliano Esposito, and Cristoforo Pomara. 2022. "Congenital Cystic Adenomatoid Malformation (CCAM) Type II: A Rare Case of Sudden Infant Death" Children 9, no. 12: 1830. https://doi.org/10.3390/children9121830
APA StyleSalerno, M., Sessa, F., Cocimano, G., Roccuzzo, S., Esposito, M., & Pomara, C. (2022). Congenital Cystic Adenomatoid Malformation (CCAM) Type II: A Rare Case of Sudden Infant Death. Children, 9(12), 1830. https://doi.org/10.3390/children9121830