scholarly journals Congenital cystic adenomatoid malformation: a rare serial case report

2021 ◽  
Vol 9 (1) ◽  
pp. 190
Author(s):  
Dedy C. Haryono ◽  
Muhammad Kartika ◽  
Prima K. Hayuningrat ◽  
Darmawan Ismail

Congenital cystic adenomatoid malformation (CCAM) is a rare congenital lung malformation which a part of the lung becomes polycystic. CCAM accounts for 25% of congenital lung malformations and 95% of lung lesions. Case 1 was a 5-month-old female infant who was diagnosed with pneumothorax, with multiple cysts in the right lung, using chest computed tomography (CT). Thoracotomy lung resection was performed. Case 2 was one-day-old newborn infant who had respiratory distress with Downe score 3. Multiple cystic lesions with septations in left lung was observed on chest CT. Lobectomy inferior lobes of left lung was performed. Both patients were diagnosed as CCAM type 1 pathologically. CCAM can be detected in the gestation by ultrasonography or after delivery through the appearance of respiratory distress signs.

2020 ◽  
Vol 1 (1) ◽  
pp. 01-03
Author(s):  
Aamir Mosawi

Background: Congenital cystic adenomatoid lung malformation is a very rare condition characterized by replacement of normal pulmonary tissue with cysts of variable size and distribution. The condition is usually unilateral. Very few cases of bilateral and congenital cystic adenomatoid malformation with good outcome after resections of the lesions have been reported, and most cases were stillborn or died early during life. Patients and methods: A forty-day male infant presented with progressive respiratory distress since the first week of life observed at the Children Teaching Hospital of Baghdad Medical City was studied. The literature was reviewed with aim of describing the early documentation of the condition in the literature. Results: Clinically, the infant had significant respiratory distress interfering with feeding and evidence of shift of the mediastinum to the right. Chest X ray showed increased translucency of left lung field and mediastinal shit to the right. CT-scan showed large air-filled cyst in the left lung and small air-filled cyst in the right lung. The child was treated successfully by left upper lobecomy. The gross examination of two gray pieces of lung tissue showed on cut section microcysts that were apparent grossly. Microscopical examination showed intercommunicating cysts lined by cuboidal epithelium with gland like appearance confirming the diagnosis of congenital cystic adenomatoid malformation of the lung. Conclusion: A very rare case of bilateral congenital cystic adenomatoid malformation of the lung is reported and the early documentation of the condition in the literature is described.


2015 ◽  
Vol 10 (1) ◽  
pp. 41-42
Author(s):  
Asish Banerjee ◽  
Sumita Pal ◽  
Anish Chatterjee ◽  
A Pan

A term neonate developed respiratory distress after 12 hours of birth which was diagnosed as a case of congenital cystic adenomatoid malformation (CCAM) of the right lung by computerized tomography scan. CCAM of the lung is rare congenital cystic lung lesion. DOI: http://dx.doi.org/10.3126/jcmsn.v10i1.12767 Journal of College of Medical Sciences-Nepal, 2014, Vol.10(1); 41-42


2020 ◽  
Vol 4 (Supplement_1) ◽  
Author(s):  
Tobias Skrebsky de Almeida ◽  
Roberta P Borges ◽  
Janeczko Laís ◽  
Giovana Caroline Marx Becker ◽  
Ticiana Costa Rodrigues ◽  
...  

Abstract Introduction: PPGLs are rare neuroendocrine tumors that arise from chromaffin cells of the adrenal medulla or their neural crest progenitors, being able to secrete catecholamines. Its treatment is primarily surgical; however, for metastatic/inoperable tumors, effective treatments are lacking. The use of TMZ, an oral alkylating agent, has been scarcely reported with variable response rates. We report 2 patients with reasonable clinical, biochemical and structural responses. Case Reports: Case 1) A 14-year old girl presented with neck pain, sweating, hypertension and tachycardia. Urinary hormonal profile revealed metanephrines 80 (up to 320 ug/24h) and normetanephrines 2983 (up to 390 ug/24h). Abdominal MRI showed a 10x6x5 cm retroperitoneal lesion in close contact with celiac trunk, superior mesenteric artery, renal arteries, aorta, left renal vein and vertebral bodies of T10, T11 and T12. A chest CT revealed multiple lung metastases. After 11 months, both the primary abdominal lesion and lung metastases increased in size.. Due to disease severity, after excluding surgical possibilities and confirming diagnosis by lesion biopsy, rescue treatment with TMZ was started for 5 days on a 28-day cycle. After 11 cycles, lung and abdominal lesions decreased more than 30% in size, and urinary metanephrines decreased 53.4%. After 21 cycles, there is no evidence of disease progression. Case 2) A 44-year old female was first diagnosed at the age of 31 with a right adrenal mass invading the kidney and the inferior vena cava associated with hypertension, sweating, headaches and palpitations. She underwent right adrenalectomy and nephrectomy. Immunohistochemistry confirmed the diagnosis of pheochromocytoma. Seven years later, follow-up CT`s showed a 3 x 2 cm liver metastasis, which was resected, and two lung lesions, one located at the right inferior lobe (1.6 cm) and the other at the left superior lobe (0.9 cm), which initially were just followed-up. At this time, a 7-month sorafenib trial was performed but the drug was stopped due to intolerable side effects. After 3 years of follow-up, the lung lesions increased in size and the right lesion was resected, but the patient refused surgery for the remainder left lung lesion. After 1 year, left lung lesion increased to 2.4 cm and mediastinal and paratracheal lymphadenomegaly developed. TMZ in the same aforementioned schedule was prescribed and after 7 cycles a new chest CT revealed complete regression of the lung and lymph node metastases.. Urinary metanephrines were 2.1 times the upper limit of normal before TMZ and decreased to normal range. Conclusion: These cases highlight the promising role of a well-tolerated single drug chemotherapy regimen in severe cases of metastatic and inoperable PPGLs. TMZ could be considered an alternative strategy for the treatment of these cases and, if possible, should be tested in adequate clinical trials.


CHEST Journal ◽  
2015 ◽  
Vol 148 (4) ◽  
pp. 624A
Author(s):  
Beatrice Tan ◽  
Jose Paolo Panuda ◽  
Consuelo Tan ◽  
Albert Rafanan

2017 ◽  
Vol 7 (1) ◽  
pp. 1130-1132 ◽  
Author(s):  
S Shukla ◽  
H Kini ◽  
ML Ilias ◽  
K Gautam

Congenital cystic adenomatoid malformation is a hamartomatous lesion. A 30 year old woman, G2P1L1, in the 29th week of gestation presented with pain abdomen, chest pain, cough with expectoration, fever and inability to appreciate fetal movements of 2 days duration. Clinically, she had pneumonia. An ultrasound revealed a single fetus in breech presentation with features of hydrops fetalis, hypoplastic left lung, mediastinal shift to the left and poor diastolic and systolic flow in the umbilical artery.A still born male baby delivered subsequently was found at autopsy to have hydrops fetalis, supradiaphragmatic right sided extralobar sequestration with associated congenital cystic adenomatoid malformation.The right lung also showed congenital cystic adenomatoid malformation. There were no other associated anomalies. We present a rare case of extralobar sequestrationwith congenital cystic adenomatoid malformation.


2015 ◽  
Vol 8 (2) ◽  
pp. 160-163
Author(s):  
Doroteya V. Malinova ◽  
Penka L. Kolova ◽  
Radoslav S. Radev

Summary Congenital cystic adenomatoid malformation (CCAM) is a rare abnormality of lung development. It was classified into 5 types by Stocker in 2002 and is also known under the name of congenital pulmonary airway malformation (CPAM). Cases are typically identified prenatally by routine ultrasonography screening. CCAM may present in the older child and adult as an incidental finding. The case presented is of a 21-year-old male patient who suffered from pulmonary infections with a recurrent productive cough 3-4 times the last 4 years. CT scanning of the thorax showed multiple cystic lesions in the right middle and lower lobes. The areas with the lesions were resected. The macroscopic and histological findings were typical for congenital cystic adenomatoid malformation type 2, which was the final diagnosis. Clinical presentations and prognosis depend on the type of lesion and its sequelae. The diagnosis is confirmed histologically.


2021 ◽  
Author(s):  
Jianneng Li ◽  
Wei Zuo ◽  
Qiang Lei ◽  
Guihua Jiang ◽  
Jianhao yan ◽  
...  

Abstract BackgroundCoronavirus disease 2019 (COVID-19) is a global catastrophic disease that has severely affected more than 185 countries. The key steps in fighting against COVID-19 involve early detection and tracking of the treatment effects. A large number of studies highlighted computed tomography (CT) as a reliable method for early diagnosis and follow-up monitoring of the disease. However, there are limited data on quantitative analysis of the follow-up images. In this study, we used a deep learning model using a neural network with high accuracy in automatic segmentation and quantification to analyze the infected lesions on chest CT images.MethodsWe used a deep learning model using a neural network with high accuracy in automatic segmentation and quantification to analyze the infected lesions on chest CT images. A total of 14 patients (mean age, 53±14 years; age range, 23–74 years; 42.9% men and 57.1% women) with confirmed mild-type COVID-19 from January 1 to May 7, 2020, were retrospectively reviewed. Initial and follow-up original CT images were collected, and CT quantitative parameters, including percentage of infection (POI) and density variation of pneumonia, were determined.ResultsThe median initial POI was 3.4% (interquartile range, IQR 0.5%–8.4%) for the whole lung, 0.8% (IQR 0.2%–6.7%) for the left lung, and 5.8% (IQR 0.5%–9.7%) for the right lung. The infection was more serious in the right than in the left lung. The infected region mainly involved bilateral lower lobes, more pronounced on the right side. Quantitative CT showed that POI significantly decreased throughout the follow-up period in all 14 patients (p < 0.001). Among them, 50% of the patients had a more significant decrease in POI (51.3%) after a negative nucleic acid test. Moreover, there was a significant decrease in the CT number range of ground-glass opacities (GGO) and consolidation (p < 0.001).ConclusionsThis study demonstrated the quantitative analysis of follow-up CT scans plays an important role in the monitoring of COVID-19 treatment, which could help in treatment planning and standardizing the assessment for discharge.


Author(s):  
M. Manjunath ◽  
M. Vishnu Sharma ◽  
Kollanur Janso ◽  
Praveen Kumar John ◽  
N. Anupama ◽  
...  

Abstract Introduction Refinements in the modern computed tomography (CT) imaging techniques have led to anatomical variations in the fissures of lung being diagnosed more frequently. So far, majority of the studies conducted are cadaveric. There is paucity of studies in this aspect based on chest CT images. Hence, we undertook this study to find the anatomical variations in the fissures. Prior detection of anatomical abnormalities is important to reduce postoperative complications in lung resection surgeries. Materials and Methods This was a cross-sectional study conducted over a period of 2 years. Data were collected from the patients who underwent CT scan thorax. Patients in whom normal anatomy of lung was distorted and cases where both lungs were not visualized completely were excluded from the study. All the CT images were reviewed by a single radiologist. The presence or absence of the normal and accessory pulmonary fissures, as well as the continuity of each fissure, was recorded by the radiologist. Data were compiled and analyzed. Results The study population consisted of 394 (70.4%) males and 166 (29.6%) females, totaling 560 cases. Fissural variations were detected in 22.9% (n = 128). Also, 17.5% (n = 98) fissural variations were seen in males and 5.4% (n = 30) fissural variations were seen in females. Further, 54.7% (n = 70) of variations were detected in the right lung and 45.3% (n = 58) in the left lung. The most common fissural variation noted was right incomplete oblique fissure with a frequency of 8.4% cases (n = 47). The most common accessory fissure detected was inferior accessory fissure. Total 22 cases were detected in both the lungs, 17 cases in male and 5 in female. Conclusion Anatomical variations in fissures were found to be more in the right lung than the left lung. Accessory fissures were detected in higher incidence on the right side.


2021 ◽  
Vol 14 (7) ◽  
pp. e243760
Author(s):  
Mohsin F Butt ◽  
Maggie Symonds ◽  
Ruhaid Khurram

Unilateral pleural effusions are uncommonly reported in patients with SARS-CoV-2 pneumonitis. Herein, we report a case of a 42-year-old woman who presented to hospital with worsening dyspnoea on a background of a 2-week history of typical SARS-CoV-2 symptoms. On admission to the emergency department, the patient was severely hypoxic and hypotensive. A chest radiograph demonstrated a large left-sided pleural effusion with associated contralateral mediastinal shift (tension hydrothorax) and typical SARS-CoV-2 changes within the right lung. She was treated with thoracocentesis in which 2 L of serosanguinous, lymphocyte-rich fluid was drained from the left lung pleura. Following incubation, the pleural aspirate sample tested positive for Mycobacterium tuberculosis. This case demonstrates the need to exclude non-SARS-CoV-2-related causes of pleural effusions, particularly when patients present in an atypical manner, that is, with tension hydrothorax. Given the non-specific symptomatology of SARS-CoV-2 pneumonitis, this case illustrates the importance of excluding other causes of respiratory distress.


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