Von Recklinghausen`s neurofibromatosis, tuberous sclerosis and encephalotrigeminal angiomatosis[Sturge-Kalischer-Weber syndrome] are frequently classified under the heading of organic neurocutaneous syndromes. Both neurofibromatosis and tuberous sclerosis are believed to represent instances of simple autosomal dominant heredity. Multiple neurofibroma and cafe*-au-lait spots are the hallmarks of the van-Recklinghausen`s disease. The characteristic features of the fully developed syndrome are [1] pigmentation of the skin, including cafe*-au-lait spots, pigmented freckles and males, and occasionally a generalized darkening of the skin; [2] subcutaneous nodules and deep neurofibromatous tumors and diffuse plexiform growths of neural tissue; [3] skeletal anomalies, especially scoliosis; and [4] predilection to malignancy. In recent years cystic lung disease, usually of the so-called honeycomb lung variety, has been reported on several occasions in patients with tuberous sclerosis. This association has been shown to our sporadically as well as in members of a single family. Little attention has been paid to the presence of cystic lung disease in association with neurofibromatosis. Currently, most think of thoracic involvement in neurofibromatosis in terms of posterior mediastinal neuroma, pheochromocytoma, meningocele or, less commonly, parenchymal pulmonary neurofibromatosis. Author have experienced a case of von Recklinghausen`s disease. This case developed a huge neurofibroma in the both side thorax and invaded to the Lt. 7th rib.
Do, Mi Young;Chung, Jae Ho;Kim, Hee Man;Han, Seung Jin;Shim, Jae Min;Hwang, Sang Yon;Park, Moo Suk;Kim, Young Sam;Chang, Joon;Kim, Sung Kyu;Park, Seung Woo;Choe, Kyu Ok;Shin, Dong Hwan;Kim, Se Kyu
Tuberculosis and Respiratory Diseases
/
v.56
no.2
/
pp.216-220
/
2004
Because a cavitary pulmonary metastasis is rare, it may not be readily identified. However, various types of cancers can metastasize to the lung in the form of cavities. We report a case of a multiple cavitary metastases to the lung from a cholangiocarcinoma in a 60-year-old man. He complained of generalized weakness and a poor oral intake for 2 months. The plain chest radiography and the chest computed tomography showed multiple small thick-walled cavities and nodules the both lungs. A bronchoscopic examination revealed a focal irregularly elevated surface of the mucosa at the orifice of the superior segment of the right lower lobe and the biopsy demonstrated an infiltrative metastatic adenocarcinoma. The abdomen-pelvis computed tomography showed an ill-marginated and irregularly low-dense area in the right lobe of the liver and a diffuse dilatation of the peripheral intrahepatic bile ducts. The esophagogastroscopy and colonoscopy showed no abnormal findings. It was concluded that the cholangiocarcinoma of the liver metastasized to the lung in the form of cavities. Thereafter, the patient underwent six cycles of the systemic chemotherapy with gemcitabine and cisplatin, and the follow-up imaging studies showed a partial response.
About one-third of radiologically indeterminate solitary pulmonary nodules (SPN) are eventually turned out to be malignant. It is very important to noninvasively determine whether the SPN is malignant or not for the decision of its way of management. PET imaging is highlighted by its unique ability of imaging the function and metabolism of cells. Glucose metabolism is increased in malignant transformed cells. We peformed FDG-PET studies in patients who had radiologically indeterminate SPN and compared the findings with histologic diagnoses to assess the diagnostic accuracy in the detection of malignancy and to decide which parameter is the most suitable for clinical practice among peak SUV (pSUV), average SUV (aSUV), 50/10 ratio, and time-activity curve (TAC), Thirty patients were included in this study and the most useful parameter was pSUV. The sensitivity and specificity in the detection of malignant SPN using 3.5 as a cut off pSUV were both 87%. Interestingly, all 2 false-negative cases were bronch-ioloalveolar carcinoma on histologic examination. If these cases, which could be strongly suspected by CT findings, were excluded, the sensitivity of pSUV was 100%. In conclusion, PET imaging is very helpful for determining malignancy in indeterminate SPN and pSUV is a conveniently measurable parameter which is valuable for interpretation.
Kim, Kyu Sik;Lim, Sung Chul;Ko, Young Chun;Park, Kyung Ha;Ju, Jin Young;Jo, Kae Jung;Kim, Soo Ok;Oh, In Jae;Kim, Yu Il;Kim, Young Chul;Kim, Sung Min;Song, Ho Chun;Bom, Hee Seung;Park, Kyung Ok
Tuberculosis and Respiratory Diseases
/
v.55
no.6
/
pp.589-596
/
2003
Background : The solitary pulmonary nodule(SPN) presents a diagnostic dilemma to the physician and the patients in the our nation with high incidence of tuberculoma. We could not exclude whether the SPN was benign or malignant by the change of the size at chest radiograph and findings of chest CT. Recently, positron emission tomography(PET) have been tried as the differential diagnostic method of SPN. We evaluated the efficacy of PET for differentiating malignant from benign SPN and the relationship between standardized uptake values(SUV) of PET and serum glucose. Method : Between January 2001 and July 2002, sixty-one patients with pulmonary nodule were examined by the chest CT and PET. The SPN has been finally diagnosed by the transthorasic needle aspiration and biopsy, bronchoscopic biopsy, and open lung biopsy. Results : Forty eight patients had a malignant nodule(23 squamous cell lung carcinoma, 16 adenocarcinoma, 9 small cell lung cancer) and thirteen patients had a benign nodule(3 tuberculoma, 9 inflammatory granuloma, 1 cryptococcosis). The mean size of malignant and benign nodule was 40.6 mm and 20.0 mm, respectively. All malignant nodules showed a marked increase in 18 fluorodeoxyglucose (FDG) uptake. Mean SUV of malignant was $9.52{\pm}5.20$ and benign nodule was $1.61{\pm}3.60$. There were false positive cases with an increase in 18-FDG uptake (2 tuberculoma, 1 inflammatory granuloma). The SUV of malignant nodule in diabetes patients has no difference in non diabetes patients($9.10{\pm}4.51$ vs $9.65{\pm}5.46$). The sensitivity and specificity of the PET scan for SPN were 100%, 77%, respectively. The positive and negative predictive values were 94% and 100%. Conclusion : PET scanning showed highly accurate result in differentiating the malignant and benign SPN. There were no significant differences between the SUV and serum glucose in the patients with lung cancer.
Background: Making the histologic diagnosis of small pulmonary nodules and ground glass opacity (GGO) lesions is difficult. CT-guided percutaneous needle biopsies often fail to provide enough specimen for making the diagnosis. Video-assisted thoracoscopic surgery (VATS) can be inefficient for treating non-palpable lesions. Preoperative localization of small intrapulmonary lesions provides a more obvious target to facilitate performing intraoperative. resection. We evaluated the efficacy of CT-guided localization with using a hook wire and this was followed by VATS for making the histologic diagnosis of small intrapulmonary nodules and GGO lesions. Material and Method: Eighteen patients (13 males) were included in this study from August 2005 to March 2008. 18 intrapulmonary lesions underwent preoperative localization by using a CT-guided a hook wire system prior to performing VATS resection for intrapulmonary lesions and GGO lesions. The clinical data such as the accuracy of localization, the rate of conversion-to-thoracotomy, the operation time, the postoperative complications and the histology of the pulmonary lesion were retrospectively collected. Result: Eighteen VATS resections were performed in 18 patients. Preoperative CT-guided localization with a hook-wire was successful in all the patients. Dislodgement of a hook wire was observed in one case. There was no conversion to thoracotomy, The median diameter of lesions was 8 mm (range: $3{\sim}15\;mm$). The median depth of the lesions from the pleural surfaces was 5.5 mm (range: $1{\sim}30\;mm$). The median interval between preoperative CT-guided with a hook-wire and VATS was 34.5 min (range: ($10{\sim}226$ min). The median operative time was 43.5.min (range: $26{\sim}83$ min). In two patients, clinically insignificant pneumothorax developed after CT-guided localization with a hook-wire and there were no other complications. Histological examinations confirmed 8 primary lung cancers, 3 cases of metastases, 3 cases of inflammation, 2 intrapulmonary lymph nodes and 2 other benign lesions. Conclusion: CT-guided localization with a hook-wire followed by VATS for treating small intrapulmonary nodules and GGO lesions provided a low conversion thoracotomy rate, a short operation time and few localization-related or postoperative complications. This procedure was efficient to confirm intrapulmonary lesions and GGO lesions.
Background : The number of immunocompromised hosts has been increasing steadily and a new pulmonary infiltrate in these patients is a potentially lethal condition which needs rapid diagnosis and treatment. In this study we sought to examine the clinical manifestations, radiologic findings, and therapeutic outcomes of pulmonary mycoses presenting as a new pulmonary infiltrate in immunocompromised hosts. Method : All cases presenting as a new pulmonary infiltrate in immunocompromised hosts and confirmed to be pulmonary mycoses by pathologic examination or by positive culture from a sterile site between October of 1996 and April of 1998 were included in the study and their chart and radiologic findings were retrospectively reviewed. Results : In all, 14 cases of pulmonary mycoses from 13 patients(male : female ratio = 8 : 5, median age 47 yr) were found. Twelve cases were diagnosed as aspergillosis while two were diagnosed as mucormycosis. Major risk factors for fungal infections were chemotherapy for hematologic malignancy(10 cases) and organ transplant recipients(4 cases). Three cases were receiving empirical amphotericin B at the time of appearance of new lung infiltrates. Cases in the hematologic malignancy group had more prominent symptoms : fever(9/10), cough(6/10), sputum(5/10), dyspnea(4/10), chest pain(5/10). Patients in the organ transplant group had minimal symptoms(p<0.05). On simple chest films, all of the cases presented as single or multiple nodules(6/14) or consolidations(8/14). High resolution computed tomograph showed peri-lesional ground glass opacities(14/14), pleural effusions(5/14), and cavitary changes(7/14). Definitive diagnostic methods were as follows : 10 cases underwent minithoracotomy, 2 underwent video-assisted thoracoscopic surgery, 1 underwent percutaneous needle aspiration and 1 case was diagnosed by culture of abscess fluid. All cases received treatment with amphotericin B with 1 case each being treated with liposomal amphotericin B and itraconazole due to renal toxicity. Lung lesion improved in 12 of 14 patient but 4 patients died before completing therapy. Conclusion : When a new lung infiltrate develops presenting either as a nodule or consolidation in a neutropenic patient with hematologic malignancy or in a transplant recipient, you should always consider pulmonary mycoses as one of the differential diagnosis. By performing aggressive work up and early treatment, we may improve prognosis of these patients.
Song, Suk Ho;Hahn, Hye Sook;Kyung, Sun Young;Hwang, Jun Kyu;An, Chang Hyeok;Lim, Young Hee;Park, Gye Young;Park, Jeong Woong;Jeong, Seong Hwan
Tuberculosis and Respiratory Diseases
/
v.52
no.4
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pp.330-337
/
2002
Background : A pulmonary tuberculoma is one of the most common causes of a solitary pulmonary lesion. Treating a tuberculoma is still controversial and there are few reports on antituberculosis chemotherapy. In this study, the clinical findings and changes in the size of tuberculomas on a radiograph after completing antituberculosis chemotherapy was investigated. Methods : The medical records, an chest radiographs of 18 pulmonary tuberculoma patients who were admitted to the Gachon medical school, Ghil medical center between April 1998 and August 2001, were reviewed. The symptomatic changes were recorded and the size of the tuberculomas following treatment were compared. To compare the size, the long distance of each tuberculoma on the chest radiographs were measured and the additional radiological findings of calcification, satellite nodules and cavities were investigated. Results : Fifteen patients were men and 3 were women. The median age was 46 (24-74). Among these 18 patients, 14 patients had clinical symptoms. The other 4 patients were diagnosed incidentally as during a routine chest radiograph. The mean size of the tuberculomas on the initial plain chest film was $4.3{\pm}2.3cm$(range : 1.7-10 cm) and after 6 months treatment, it had decreased to $1.68{\pm}2.00cm$(range : 1.5-6.5 cm) (P<0.05). At least 6 months of antituberculosis chemotherapy resulted in the findings of a tuberculoma with a disappearance in 9, a decreased size in 4, and no change in 5 on the chest radiograph. Calcifications were found in 3 patients on the initial chest film and the chest CT and all calcified tuberculomas had disappeared after treatment. Conclusion : Although a pulmonary tuberculoma can remain as an inactive lesion for a long time, if it is confirmed by pathological or bacteriological methods, antituberculosis chemotherapy will be beneficial despite the presence of calcification.
Background: The lung is the most common site of metastasis and usually it manifests as a single or multiple nodules in chest X-ray. But less commonly the cancer spreads through the lymphatics and X-ray shows diffuse reticulonodular densities. Sometimes, patient is presented with respiratory symptoms only with interstitial lung infiltration before the signs of primary tumor and in that cases, the differential diagnosis with other interstitial lung disease is required. We have experienced 5 such cases, who were diagnosed as lymphangitic carcinomatosis by transbronchial lung biopsy. Methods: Clinical manifestation, pulmonary function test, modified thin section CT, bronchoalveolar lavage and transbronchial lung biopsy were done. Results: The primary tumor was gastric cancer in 3, lung cancer in 2. Pulmonary function test showed restrictive pattern with low DLco in 2 patients and obstructive pattern in one. Bronchoalveolar lavage showed lymphocytosis in 4 patients and malignant cells were found in one patient. Transbronchial lung biopsy revealed malignant cells localized to the lymphatics (peribronchial, perivascular and perialveolar). Cell type was adenocarcinoma in 4 and squamous cell carcinoma in one. Conclusion: Rarely lymphangitic carcinomatosis can be presented as diffuse interstitial lung disease and easily diagnosed by transbronchial lung biopsy.
Hong, Kyung Soo;Ahn, June Hong;Choi, Eun Young;Jin, Hyun Jung;Shin, Kyeong-Cheol;Chung, Jin Hong;Lee, Kwan Ho
Journal of Yeungnam Medical Science
/
v.32
no.2
/
pp.71-79
/
2015
Background: Recent studies have shown that the nontuberculosis mycobacterium (NTM) recovery rate in clinical cultures has increased within Korea. However, another study conducted by a secondary hospital within Daegu reported different results. Therefore, the purpose of this study is to understand and evaluate the microbiological distribution and clinical features of NTM in Daegu. Methods: A retrospective study was conducted on 11,672 respiratory specimens undergoing acid fast bacilli (AFB) culture from 6,685 subjects who visited Yeungnam University Respiratory Center from January 2012 to December 2013. Results: Of the 11,672 specimens undergoing AFB culture, 1,310 specimens (11.2%) showed positive results. Of these specimens, NTM was recovered from 587 specimens, showing a recovery rate of 44.8%. Identification test for NTM was performed on 191 subjects; the results were as follows: M. avium-intracellulare complex (MAC) 123 (64.4%), M. abscessus 20 (10.5%), M. kansasii 12 (6.3%), and 33 other NTM germ strains. Of the 382 subjects with NTM, 167 were diagnosed with pulmonary NTM disease (43.7%), however virulence differed depending on NTM strain. Multivariate analysis showed that nodular bronchiectasis, the nodules, and finding consistent with cavity under imaging study were statistically significant for triggering pulmonary NTM disease. AFB culture showing MAC and M. abscessus was statistically significant as well. Positive predictive value for NTM polymerase chain reaction (NTM-PCR) was 88.6%. Conclusion: Results for NTM recovery rate within the Daegu area were similar to those for the Seoul metropolitan area. We can assume that NTM infection is increasing in our community, therefore AFB-positive subjects (1) should undergo NTM-PCR, (2) should have their culture results checked for differentiation of mycobacterium tuberculosis complex (MTB) from NTM, and (3) undergo NTM identification test to confirm its type. Administration of treatment with the above results should be helpful in improving the patients' prognosis.
Malignant melanoma is a highly malignant form of cutaneous cancer derived from melanocytes. The lesion frequently metastasizes to the lymph nodes, lung, liver and bone. However, an endobronchial metastasis and a primary malignant. melanoma of the lung are quite rare. We report a case of an unknown primary malignant melanoma with a pulmonary and endobronchial metastasis in a 34 years old male. He complained of coughing and black-colored sputum. Abnormal skin and mucosal lesions were not found during a physical examination. A chest X-ray revealed multiple nodular masses in both lung fields. A flexible bronchoscopy showed two yellowish small nodules at the entry of left lower bronchus. Vimentin, the S-100 protein, and HMB-45 stain positive melanoma cells were detected at the bronchoscopic biopsy specimen.
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