• Title/Summary/Keyword: congenital cystic adenomatoid malformation

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A Case of Congenital Cystic Adenomatoid Malformation of the Lung Containing Fungal Ball-like Movable Blood Clots in Healthy Young Woman (진균구로 오인된 낭포내 응혈을 보인 23세 여자에서의 선천성 낭포성 유선종 폐기형 1례)

  • Lee, Gang-Ryong;Lee, Kun-Sun;Joo, In-Kyu;Park, June-Ock;Choi, Seung-Jun;Lee, Won-Seok;Kim, Eui-Sook;Lee, Gyu-Hyeon;Kim, Dae-Ha;Lee, Gwang-Seob;Han, Young-Sook;Jee, Mi-Kyeng;Park, Jung-Woong
    • Tuberculosis and Respiratory Diseases
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    • v.46 no.1
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    • pp.129-135
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    • 1999
  • We describe unusual manifestations of congenital cystic adenomatoid malformation (C.C.A.M.) of the lung, such as movable fungal ball-like intracystic blood clots and hemoptysis, which were detected in previously healthy 23 years-old woman. We identified blood clots only after left upper lobectomy and could not distinguish from fungal ball with radiographic methods. CCAM of the lung, rare and lethal form of congenital pulmonary cystic disease, was initially introduced by Ch, in and Tang in 1949. The histogenesis of this lesion is characterized by polypoid glandular tissue proliferation and overgrowth of mesenchymal elements due to cessation of bronchiolar maturation which occurred in after 16weeks intrauterine period. In 80-95% of reported cases, the lesion was confined to a single lobe and there was no lobe and right and left lung predilection The clinical presentation may be widely variable, ranging from intrauterine fetal death to late discovery in childhood with recurrent pulmonary infection. But there,s no reports which were misdiagnosed with intracystic fungal ball. The treatment choice is lobectomy of affected lobe. There,s a few case reports with rhabdomyosarcoma, bronchiolar cell carcinoma and myxosarcoma arising in CCAM patients. Therefore, early resection is recommended even if asymtomatic cases. We experienced a rare case of CCAM of the lung in 23 years old female, and there were intracystic fungal ball-like movable blood clots in lower portion of left lung. After left upper lobectomy was performed, now she is discharged and followed up without any complications.

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Prenatally Diagnosed Extrapulmonary Sequestration - 2 cases - (산전 진단된 폐외분리증 2예)

  • Kim, Hyun-Young;Son, Dong-Woo;Kim, Seok-Yong;Kim, Jee-Eun;Ha, Seung-Yeon
    • Advances in pediatric surgery
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    • v.15 no.2
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    • pp.173-179
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    • 2009
  • Congenital thoracic malformations such as intra- and extra-pulmonary sequestration, cystic adenomatoid malformation, congenital pulmonary airway malformation, malinosculation, bronchogenic cyst, reduplication cyst, and foregut cyst are frequently detected on routine prenatal ultrasound. There are some controversies about treatment for postnatally persistent pulmonary sequestration. Some authors recommend expectant long term follow up but most authors advocate elective surgical excision because of complication such as respiratory distress, infection, intrathoracic bleeding, haemoptysis, cardiac failure, and potential risk of malignancy. We experienced 2 cases of prenatally diagnosed extrapulmonary sequestration which were located in the subdiaphragmatic retroperitoneum. Resections were performed at 2 months and 4 months of age using intraabdominal approach. There were no complications. In conclusion, if the prenatally diagnosed extrapulmonary sequestration remained postnatally, early operation might reduce morbidity related to extrapulmonary sequestration and parental anxiety without any postoperative complication.

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Congenital Cystic Adenomatoid Malformation of Lung in Adults: Clinical, Pathologic and Radiologic Evaluation of Six Patients (성인에서 진단된 선천성 낭포성 유선종 폐기형 6예)

  • Park, Young Jin;Jung, Hoon;Park, I-Nae;Choi, Sang Bong;Hur, Jin-Won;Lee, Hyuk Pyo;Yum, Ho-Kee;Choi, Soo Jeon;Koo, Ho-Seok;Lee, Yang-Haeng;Choi, Suk-Jin;Jung, Soo-Jin;Lee, Hyun-Kyung;Kim, Ae Ran
    • Tuberculosis and Respiratory Diseases
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    • v.65 no.2
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    • pp.110-115
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    • 2008
  • Background: Congenital cystic adenomatoid malformation of the lung (CCAM) is a rare congenital developmental anomaly of the lower respiratory tract. Most cases are diagnosed within the first 2 years of life, so adult presentation of CCAM is rare. We describe here six adult cases of CCAM and the patients underwent surgical resection, and all these patients were seen during a five and a half year period. The purpose of this study was to analyze the clinical, radiological and histological characteristics of adult patients with CCAM. Methods: Through medical records analysis, we retrospectively reviewed the clinical characteristics, the chest pictures (X-ray and CT) and the histological characteristics. Results: Four patients were women and the mean age at diagnosis was 23.5 years (range: 18~39 years). The major clinical presentations were lower respiratory tract infection, hemoptysis and pneumothorax. According to the chest CT scan, 5 patients had multiseptated cystic lesions with air fluid levels and one patient had multiple cavitary lesions with air fluid levels, and these lesions were surrounded by poorly defined opacities at the right upper lobe. All the patients were treated with surgical resection. 5 patients underwent open lobectomy and one patient underwent VATS lobectomy. On the pathological examination, 3 were found to be CCAM type I and 3 patients were CCAM type II, according to Stocker's classification. There was no associated malignancy on the histological studies of the surgical specimens. Conclusion: As CCAM can cause various respiratory complications and malignant changes, and the risks associated with surgery are extremely low, those patients who are suspected of having or who are diagnosed with CCAM should go through surgical treatment for making the correct diagnosis and administering appropriate treatment.

Posterior Thoracotomy under the Prone Position in Children - Report of 3 Cases - (소아에서 복와위하의 개흉술 3례 보고)

  • 김기봉
    • Journal of Chest Surgery
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    • v.24 no.10
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    • pp.1000-1004
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    • 1991
  • Children`s small airway precludes the use of standard methods of bronchial separation. So, we performed the posterior thoracotomy under the prone position in 3 cases to avoid endobronchial gravity spillage of secretion and infected debris from the diseased lung to the contralateral sound lung. The advantages of the posterior thoracotomy under the prone position was discussed. In two cases, empyema with total collapse of left lung and congenital cystic adenomatoid malformation [CCAM] of right lung, copious secretion was spilled through the endotracheal tube but could be removed successfully by the endotracheal suction. In the third case of bilateral peripleural abscess, bilateral posterior thoractomy was done without position change. All procedures were performed without any technical difficulty and complication.

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Surgical Treatment of Congenital Cystic Lung Disease (선천성 낭성 폐질환의 수술적 치료)

  • Wi, Jin-Hong;Lee, Yang-Haeng;Han, Il-Yong;Yoon, Young-Chul;Hwang, Youn-Ho;Cho, Kwang-Hyun
    • Journal of Chest Surgery
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    • v.41 no.3
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    • pp.335-342
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    • 2008
  • Background: Congenital cystic diseases of the lung are uncommon, and they share similar embryogenic and clinical characteristics. But they are sometimes vary widely in their presentation and severity. Therefore they are often difficult to make different diagnosis each other, and all require surgical treatment. Material and Method: From 1993 to 2006, 38 patients underwent surgical procedures under these diagnostic categories in the Depart. of Thoracic and. Cardiovascular Surgery, Busan-Paik Hospital, College of Medicine, Inje University. And we retrospectively reviewed these patients' charts for clinical presentations, surgical procedures, pathologic findings and postoperative morbidity and mortality. Result: There were 22 males and 16 females, ages ranged from 1 month after birth to 51 years and mean age was 20.8 years. The main symptoms were 19 fever, cough, sputum production due to recurrent infection, 7 dyspnea, 8 chest discomfort, 4 hemoptysis, but eight patients were asymptomatic. Computed tomography was chosen as diagnostic modalities and available for operation plan for all of patients. For all the cases, surgical resection were performed. Lobectomy was performed in 28 patients, simple excision (resection) in 8 patients, segmentectomy or wedge resection in 2 patients. There were 10 pulmonary sequestrations, 15 congenital cystic adenomatoid malformations (CCAM), 11 bronchogenic cysts, and 2 congenital lobar emphysemas. They all were confirmed by pathologic exams. The complications were 6 wound disruption or infection, 2 chylothorax, 1 ulnar neuropathy, but all of them were resolved uneventful. There was no persistent air leakage, respiratory failure, operative mortality and recurrence. Conclusion: We performed immediate surgical removal of congenital cystic lung lesions after diagnosis and obtained good results, so reported them with literature review.

Vertical Axillary Muscle Sparing Thoracotomy in Thoracic Surgery (흉부 수술에 있어 수직액와 근육보존 개흉술의 적용)

  • Won, Tae-Hui;Seong, Suk-Hwan
    • Journal of Chest Surgery
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    • v.28 no.1
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    • pp.42-46
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    • 1995
  • Vertical axillary muscle sparing thoracotomy is newly appeared and excellent alternative method of standard posterolateral thoracotomy.It has many advantages compared to standard posterolateral thoracotomy , less postoperative pain, well preserved thoracic muscle strength, full range of motion of the shoulder girdle and attractive cosmetic results. We performed vertical axillary muscle sparing thoracotomy in 36 patients from November 1993 to July 1994. The ages of the patients ranged from 6 months to 71 years[mean 45.1 years , and the patients consisted of 20 males and 16 females.The preoperative diagnosis were as follows : lung cancer in 17 patients, tbc destroyed lung in 7, bronchiectasis in 3, bullous emphysema in 3 and the others are mediastinal tumor, bronchogenic cyst, lung abscess, empyema, esophageal diverticulum, and CCAM [congenital cystic adenomatoid malformation . The operative procedures were as follows : lobectomy and bilobectomy in 16 patients, segmentectomy in 4, wedge resection in 3, penumonectomy in 7, and the others were open biopsy, lobectomy with diaphragm excision, sleeve right upper lobectomy, decortication, mediastinal mass excision, and esophageal diverticulectomy. We had 6 complications : postoperative bleeding in 2 cases, operative wound infection, arrrhythmia[atrial fibrillation , Horner`s syndrome, hoarseness. The subcutaneous seroma occurred in 4 cases but did not require drainage and relieved within 4 weeks spontaneously. We concluded that vertical axillary muscle sparing thoracotomy could be done in most of all thoracic surgery with safety. Comparing to standard posterolateral thoracotomy vertical axillary muscle sparing thoracotomy has many advantages such as less postoperative pain, well preserved muscle strengths and good cosmetic results.

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