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幼儿先天性囊性腺瘤样畸形:伴有胸腔积液的不寻常表现。

Congenital cystic adenomatoid malformation in a toddler: Unusual presentation with pleural effusion.

作者信息

Laanait Radia, El Fakiri Karima, Rada Noureddine, Draiss Ghizlane, Bouskraoui Mohammed

机构信息

Pediatric A Department, Mohammed VI University Hospital, Marrakesh, Morocco, Faculty of Medicine and Pharmacy, University Cadi Ayyad, Morocco.

出版信息

Radiol Case Rep. 2024 Aug 18;19(11):5013-5017. doi: 10.1016/j.radcr.2024.07.096. eCollection 2024 Nov.

DOI:10.1016/j.radcr.2024.07.096
PMID:39247462
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11378084/
Abstract

Congenital cystic adenomatoid malformation encompasses a series of cystic malformative lesions characterized by aberrant bronchiolar formations of varying size and dispersion. Most cases of this illness are detected in the first few years of life, usually affecting infants. We report a case of CCAM presenting as pleural effusion in a 15-month-old boy who presented with acute respiratory distress. Chest CT revealed a cystic image in the right lower lobe of the lung. The patient had surgical excision, and a pathological examination validated the diagnosis of CCAM type 1 with no malignant material. Following surgery, the patient's general condition improved, and no new respiratory symptoms were observed during an 8-month follow-up period. Increased awareness of this rare condition among pediatricians and radiologists is crucial for facilitating early diagnosis and appropriate treatment.

摘要

先天性囊性腺瘤样畸形包括一系列以大小和分布各异的异常细支气管形成为特征的囊性畸形病变。这种疾病的大多数病例在生命的最初几年被发现,通常影响婴儿。我们报告一例15个月大患有急性呼吸窘迫的男孩,其先天性囊性腺瘤样畸形表现为胸腔积液。胸部CT显示右肺下叶有囊性影像。患者接受了手术切除,病理检查证实为1型先天性囊性腺瘤样畸形,无恶性物质。手术后,患者的一般状况有所改善,在8个月的随访期内未观察到新的呼吸道症状。儿科医生和放射科医生对这种罕见疾病的认识提高对于促进早期诊断和适当治疗至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d85/11378084/34da3f977faa/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d85/11378084/5cda44381574/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d85/11378084/d4532476d142/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d85/11378084/34da3f977faa/gr3.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d85/11378084/5cda44381574/gr1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d85/11378084/d4532476d142/gr2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/6d85/11378084/34da3f977faa/gr3.jpg

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本文引用的文献

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Congenital pulmonary airway malformation (CPAM): A case report and review of the literature.先天性肺气道畸形(CPAM):一例病例报告及文献综述
Radiol Case Rep. 2023 Jul 25;18(10):3483-3486. doi: 10.1016/j.radcr.2023.07.018. eCollection 2023 Oct.
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A rare case of congenital cystic adenomatoid malformation: Mimics pneumonia manifestations.先天性囊性腺瘤样畸形罕见病例:酷似肺炎表现。
Ann Med Surg (Lond). 2021 May 27;66:102433. doi: 10.1016/j.amsu.2021.102433. eCollection 2021 Jun.
3
The natural history of prenatally diagnosed congenital cystic lung lesions: long-term follow-up of 119 cases.
产前诊断的先天性肺囊性病变的自然病史:119例长期随访
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Congenital cystic adenomatoid malformation in an adolescent: an unusual presentation with pleural effusion and pneumatocele.青少年先天性囊性腺瘤样畸形:一种伴有胸腔积液和气囊肿的罕见表现。
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Malignancy of congenital cystic adenomatoid malformation of lung in aged.老年先天性肺囊性腺瘤样畸形恶变
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From cystic pulmonary airway malformation, to bronchioloalveolar carcinoma and adenocarcinoma of the lung.从肺囊性气道畸形到细支气管肺泡癌和肺腺癌。
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