Masatoshi Kurihara

Juntendo University, Tokyo, Tokyo-to, Japan

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Publications (16)32.58 Total impact

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    ABSTRACT: Thoracic endometriosis-related pneumothorax (TERP) is a secondary condition specific for females, but in a clinical setting, TERP often is difficult to distinguish from primary spontaneous pneumothorax (PSP) based on a relationship between the dates of pneumothorax and menstruation. The purpose of this study was to clarify the clinical features of TERP compared with PSP. We retrospectively reviewed the clinical and histopathological files of female patients with pneumothorax who underwent video-assisted thoracoscopic surgery in the Pneumothorax Research Center during the 6-year period from January 2005 to December 2010. We analyzed the clinical differences between TERP and PSP. The study included a total of 393 female patients with spontaneous pneumothorax, of whom 92 (23.4 %) were diagnosed as having TERP and 33.6 % (132/393) as having PSP. We identified four factors (right-sided pneumothorax, history of pelvic endometriosis, age ≥31 years, and no smoking history) that were statistically significant for predicting TERP and assigned 6, 5, 4, and 3 points, respectively, to establish a scoring system with a calculated score from 0 to 18. The cutoff values of a calculated score ≥12 yielded the highest positive predictive value (86 %; 95 % confidence interval (CI) 81.5-90.5 %) for TERP and negative predictive value (95.2 %; 95 % CI 92.3-98 %) for PSP. TERP has several distinct clinical features from PSP. Our scoring system consists of only four clinical variables that are easily obtainable and enables us to suspect TERP in female patients with pneumothorax.
    Beiträge zur Klinik der Tuberkulose 05/2014; · 2.06 Impact Factor
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    ABSTRACT: To characterize pathologic features of pulmonary cysts and to elucidate the possible mechanism of cyst formation in lungs of patients with Birt-Hogg-Dubé syndrome (BHDS), a tumor suppressor gene syndrome, we used histologic and morphometric analyses. We evaluated 229 lung cysts from 50 patients with BHDS and 117 from 34 patients with primary spontaneous pneumothorax (PSP) for the number, size, location, and absence or presence of inflammation. These BHDS-cysts abutted interlobular septa (88.2%) and had intracystic septa (13.6%) or protruding venules (39.5%) without cell proliferation or inflammation. Frequencies of these histologic characteristics differed significantly from those of patients with PSP (P<0.05). Although the intrapulmonary BHDS-cysts were smaller than the subpleural BHDS-cysts (P<0.001), there was no difference in size between them when there was no inflammation. The number of cysts diminished logarithmically and the proportion of cysts with inflammation increased as their individual sizes enlarged (P<0.05). These results imply that the BHDS-cysts are likely to develop at the periacinar region, an anatomically weak site in a primary lobule, where alveoli attach to connective tissue septa. We hypothesize that the BHDS cysts possibly expand in size as the alveolar walls disappear at the alveolo-septal junction and grow even larger when several cysts fuse. This article is protected by copyright. All rights reserved.
    Histopathology 01/2014; · 2.86 Impact Factor
  • Takahiro Haga, Masatoshi Kurihara, Hideyuki Kataoka
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    ABSTRACT: Re-expansion pulmonary edema is an uncommon condition that occurs when a collapsed lung is expanded. The aim of the present study was to investigate the incidence and risk factors associated with re-expansion pulmonary edema which may occur as a complication when carrying out treatment for spontaneous pneumothorax. A total of 462 patients with spontaneous pneumothorax treated with chest tube drainage in inpatient settings at the Nissan Tamagawa Hospital during the 6-year period between January 2007 and December 2012 were retrospectively evaluated. The data were analyzed to identify any clinical differences between the patients with and without re-expansion pulmonary edema. Re-expansion pulmonary edema occurred on 30 (6.5 %) of the 462 patients. The duration of lung collapse in the patients with re-expansion pulmonary edema was longer than that observed in the patients without re-expansion pulmonary edema. (7.7 ± 9.1 and 2.4 ± 4.6 days). This difference was statistically significant (P < 0.0001). The extent of lung collapse in the patients with re-expansion pulmonary edema was more severe than that observed in the patients without re-expansion pulmonary edema. This difference was also statistically significant (P = 0.004). The results suggest that treating spontaneous pneumothorax using chest tube drainage requires careful consideration in view of the relatively high incidence of re-expansion pulmonary edema, especially in cases associated with long periods of lung collapse or large spontaneous pneumothoraxes.
    Surgery Today 09/2013; · 0.96 Impact Factor
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    ABSTRACT: Lymphangioleiomyomatosis (LAM) is a rare disease caused by dysregulated activation of the mammalian target of rapamycin (mTOR). Sirolimus, an inhibitor of mTOR, has been reported to decrease the size of angiomyolipomas and stabilize pulmonary function in patients with LAM. However, the optimal dose for the treatment of LAM remains unclear. We conducted a retrospective, observational study of 15 patients with LAM who underwent sirolimus therapy for more than 6 months. The efficacy was evaluated by reviewing the patients' clinical courses, pulmonary function and chest radiologic findings before and after the initiation of sirolimus treatment. All patients had blood trough levels of sirolimus lower than 5ng/mL. Sirolimus treatment improved the annual rates of change in FVC and FEV1 in the 9 patients who were free from chylous effusion (FVC, -101.0 vs. +190.0mL/y, p=0.046 and FEV1, -115.4 vs. +127.8mL/y, p=0.015). The remaining 7 patients had chylous effusion at the start of sirolimus treatment; the chylothorax resolved completely within 1-5 months of treatment in 6 of these cases. These results resembled those of previous studies in which blood trough levels of sirolimus ranged from 5 to 15ng/mL. Low-dose sirolimus (trough level, 5ng/mL or less) performed as well as the higher doses used previously for improving pulmonary function and decreasing chylous effusion in patients with LAM.
    Respiratory investigation. 09/2013; 51(3):175-83.
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    ABSTRACT: Thoracic endometriosis is a rare disease responsible for catamenial pneumothorax. The immunohistochemical features of thoracic endometriosis are not well understood. An immunohistochemical examination of 84 diaphragmatic specimens of catamenial pneumothorax using antibodies against estrogen receptor (ER), progesterone receptor (PgR), CD10 and smooth muscle actin (SMA) was conducted. The endometrial tissue was small, and focally located around the chasm of the tendon on the side of the thoracic cavity. Endometrial stroma were detected in 84/84 (100%) of the specimens, endometrial glands were detected in 21/84 (25%) and smooth muscle was detected in 1/84 (1.2%). The endometrial stroma exhibited positive staining for ER in 74/84 (88.1%) of the specimens, PgR in 84/84 (100%), CD10 in 74/84 (88.1%) and SMA in 46/84 (54.8%). Because thoracic endometriosis is small in size, and only 25% of the resected tissue specimens were accompanied with the endometrial gland, an immunohistochemical analysis can be useful for their detection. The fact that over half of the thoracic endometrial stroma showed positive staining for SMA, and the existence of thoracic endometriosis accompanied by smooth muscle, indicated that some part of the thoracic endometriosis may have the ability to differentiate into smooth muscle, although further studies are needed to confirm this hypothesis.
    Pathology International 09/2013; 63(9):429-34. · 1.72 Impact Factor
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    ABSTRACT: Purpose: Catamenial pneumothorax (CP) is classified as thoracic endometriosis syndrome. There are few reports of CP, and the clinical manifestations of this disease are unclear. The aim of the present study is to clarify the features of CP.Methods: The clinical and pathological files of the 92 female patients pathologically diagnosed with thoracic endometriosis are included in this study. The clinical data and pathological findings of the recurrent and non-recurrent groups are compared.Results: Thirty-six patients (39.1%) experienced recurrence, 37 (40.2%) patients did not, and 19 (20.4) patients could not be evaluated. The ratio of the endometrial gland in the diaphragm is significantly higher in the recurrent cases in comparison to non-recurrent cases (66.7% vs. 37.8%, P = 0.01).Conclusions: The recurrence rate of CP is high. Further study of the optimal management strategies is needed, especially for CP cases with the endometrial gland in the diaphragm.
    Annals of thoracic and cardiovascular surgery : official journal of the Association of Thoracic and Cardiovascular Surgeons of Asia. 05/2013;
  • Takahiro Haga, Masatoshi Kurihara, Hideyuki Kataoka
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    ABSTRACT: Objective Prolonged air leaks in patients with spontaneous pneumothorax are not infrequent. The aim of this study was to assess the duration of air leaks and define the clinical variables associated with the therapeutic success of chest tube drainage for spontaneous pneumothorax. Methods A total of 441 patients with spontaneous pneumothorax treated with chest tube drainage between 2008 and 2012 were retrospectively evaluated. The clinical differences between the patients successfully treated with drainage and those who required more invasive procedures were analyzed. Results Invasive procedures, such as video-assisted thoracic surgery (n=121), fibrin glue administration through a chest tube (n=8) and pleurodesis with OK-432 (n=21), were performed in 34% (150/441) of the patients. The treatment rate of chest drainage alone was higher in the patients with initial pneumothorax (72%; 124/170) than in those with recurrent pneumothorax (62%; 167/271) (p=0.015). In addition, this rate was higher in the patients with moderate lung collapse (70%; 167/237) than in those with severe lung collapse (61%; 124/204) (p=0.032). Conclusion Patients with recurrent pneumothorax or severe lung collapse associated with prolonged air leakage are more likely to receive invasive procedures.
    Internal Medicine 01/2013; 52(19):2189-2192. · 0.97 Impact Factor
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    ABSTRACT: BACKGROUNDS: Lymphangioleiomyomatosis (LAM) is a destructive lung disease that share clinical, physiologic, and radiologic features with chronic obstructive pulmonary disease (COPD). This study aims to identify those features that are unique to LAM by using quantitative CT analysis. METHODS: We measured total cross-sectional areas of small pulmonary vessels (CSA) less than 5mm(2) and 5-10mm(2) and calculated percentages of those lung areas (%CSA), respectively, in 50 LAM and 42 COPD patients. The extent of cystic destruction (LAA%) and mean parenchymal CT value were also calculated and correlated with pulmonary function. RESULTS: The diffusing capacity for carbon monoxide/alveolar volume (DL(CO)/VA %predicted) was similar for both groups (LAM, 44.4±19.8% vs. COPD, 45.7±16.0%, p=0.763), but less tissue damage occurred in LAM than COPD (LAA% 21.7±16.3% vs. 29.3±17.0; p<0.05). Pulmonary function correlated negatively with LAA% (p<0.001) in both groups, yet the correlation with %CSA was significant only in COPD (p<0.001). When the same analysis was conducted in two groups with equal levels of LAA% and DL(CO)/VA %predicted, %CSA and mean parenchymal CT value were still greater for LAM than COPD (p<0.05). CONCLUSIONS: Quantitative CT analysis revealing a correlation between cystic destruction and CSA in COPD but not LAM indicates that this approach successfully reflects different mechanisms governing the two pathologic courses. Such determinations of small pulmonary vessel density may serve to differentiate LAM from COPD even in patients with severe lung destruction.
    European journal of radiology 09/2012; · 2.65 Impact Factor
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    ABSTRACT: Purpose: Primary spontaneous pneumothorax is believed to result from blebs, or from other abnormalities of the pleural surface. There is no consensus as to whether a change in weather conditions can precipitate spontaneous pneumothorax. The aim of the present study was to investigate the influence of weather conditions on the onset of primary spontaneous pneumothorax.Methods: The case histories of 1051 inpatients with primary spontaneous pneumothorax treated at Nissan Tamagawa Hospital between January 2006 and December 2011 were analyzed retrospectively. Data on weather conditions were collected daily throughout the 6-year period. The data were analyzed to determine differences in weather conditions between days on which primary spontaneous pneumothorax occurred and those on which it did not. Results: Primary spontaneous pneumothorax occurred on 819 (37.3%) of 2191 study days. On days before and the day of primary spontaneous pneumothorax onset, the difference in mean atmospheric pressure was 0.6 hPa lower than on days in which no primary spontaneous pneumothorax occurred. This difference was statistically significant (P = 0.015). There was no statistical difference in mean, maximum, and minimum temperature, hours of sunshine, amount of precipitation, and mean and minimum humidity between days with and those without primary spontaneous pneumothorax.Conclusion: This largest study of the literature showed decreased atmospheric pressure might play an important role in the occurrence of primary spontaneous pneumothorax.
    Annals of thoracic and cardiovascular surgery : official journal of the Association of Thoracic and Cardiovascular Surgeons of Asia. 08/2012;
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    ABSTRACT: Since Birt-Hogg-Dubé syndrome (BHDS) and lymphangioleiomyomatosis (LAM) share some clinical manifestations (multiple pulmonary cysts with pneumothorax, renal tumors, and sometimes skin lesions), the differential diagnosis of the two diseases becomes problem especially in female patients. This study aims to quantify pulmonary cysts in computed tomography (CT) of females with BHDS and those with LAM and also to identify the independent parameters for differentiating the two diseases. Fourteen patients with BHDS and 52 with LAM were studied. In CT scans, lung fields were defined as areas with fewer than -200 Hounsfield units (HU) and pulmonary cysts as areas consisting of 10 or more consecutive pixels with fewer than -960 HU. The extent, number, size and circularity of cysts were calculated by using hand-made software and compared between the two diseases. Moreover, the lung fields were divided into six zones and analyzed for the distribution of cysts. Finally, a stepwise discriminant analysis employing quantitative measurements of cysts and clinical features was performed. The two diseases were significantly different in all quantitative measurements of cysts. Stepwise discriminant analysis accepted the following four variables: the family history of pneumothorax within the second degree relatives, lower-medial zone predominance of cysts, diffusing capacity and mean size of cysts in this order. The quantitative characteristics of pulmonary cysts are significantly different between BHDS and LAM. The independent parameters for differentiating the two diseases are the family history of pneumothorax, zonal predominance of cysts, diffusing capacity and size of cysts.
    European journal of radiology 05/2011; 81(6):1340-6. · 2.65 Impact Factor
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    ABSTRACT: Birt-Hogg-Dubé syndrome (BHDS) is an inherited autosomal genodermatosis characterised by fibrofolliculomas of the skin, renal tumours and multiple lung cysts. Genetic studies have disclosed that the clinical picture as well as responsible germline FLCN mutations are diverse. BHDS may be caused by a germline deletion which cannot be detected by a conventional genetic approach. Real-time quantitative polymerase chain reaction (qPCR) may be able to identify such a mutation and thus provide us with a more accurate clinical picture of BHDS. This study analysed 36 patients with multiple lung cysts of undetermined causes. Denaturing high performance liquid chromatography (DHPLC) was applied for mutation screening. If no abnormality was detected by DHPLC, the amount of each FLCN exon in genome was quantified by qPCR. An FLCN germline mutation was found in 23 (63.9%) of the 36 patients by DHPLC and direct sequencing (13 unique small nucleotide alterations which included 11 novel mutations). A large genomic deletion was identified in two of the remaining 13 patients by qPCR (one patient with exon 14 deletion and one patient with a deletion encompassing exons 9 to 14). Mutations including genomic deletions were most frequently identified in the 3'-end of the FLCN gene including exons 12 and 13 (13/25=52.0%). The BHDS patients whose multiple cysts prompted the diagnosis in this study showed a very low incidence of skin and renal involvement. BHDS is due to large deletions as well as small nucleotide alterations. Racial differences may occur between Japanese and patients of European decent in terms of FLCN mutations and clinical manifestations.
    Journal of Medical Genetics 04/2010; 47(4):281-7. · 5.70 Impact Factor
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    ABSTRACT: BACKGROUND: Lymphangioleiomyomatosis (LAM) is a rare neoplastic disease in which abnormal smooth muscle-like cells (LAM cells) proliferate in the lungs and along the axial lymphatic systems, including the lymph nodes and thoracic ducts. LAM cells are transformed due to loss-of-function type mutations of either the TSC1 or TSC2 tumor suppressor genes. The pathological features include the proliferation of benign-looking LAM cells and the existence of abundant lymphatic vessels that are associated with clinical conditions such as chyle leakage. LAM cells produce potent lymphangiogenic growth factors (VEGF-C and VEGF-D) and the lymphatic vessel density within LAM lesions correlates with the histologic severity of LAM. The serum VEGF-D level increases in LAM, especially in patients with lymphatic involvement. LAM cell clusters (LCCs), which are postulated pathologically to be generated by lymphangiogenesis-mediated fragmentation and subsequent shedding into the lymphatic circulation, are observed in both chylous effusion and LAM-associated lymphatics within LAM tissue specimens. The identification of LCCs in chylous effusion together with the characteristic clinical manifestations can therefore be an alternative for a lung biopsy if LAM patients are complicated with chylous effusion. CONCLUSION: LAM appears to be a disease involving a dysfunction of the lymphatic system and a fascinating model of tumor dissemination that is exclusively lymphangitic. LAM-associated lymphangiogenesis that mediates the shedding of LCCs seems to play a central role in the dissemination of LAM cells and progression in LAM and it may also be a potential therapeutic target as well as the dysregulated mTOR signaling pathway.
    Lymphatic Research and Biology 03/2010; 8(1):21-31. · 2.33 Impact Factor
  • Masatoshi Kurihara, Hideyuki Kataoka, Aki Ishikawa, Reina Endo
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    ABSTRACT: Currently, the treatment of pneumothorax varies among hospitals. Three factors account for the differences. First, pneumothorax expresses the state of disease instead of the disease itself. Some pneumothoraces heal on their own but patients with pneumothorax due to chronic diseases may need further intervention. The decision for the choice of treatment is up to the physician. Second, it is insufficient to treat pneumothorax with current treatment guidelines. Third, prognosis and follow-up after pneumothorax treatment is not well documented. Therefore, verification/assessment of treatment and its effect is insufficient. To prevent postoperative bulla neogenesis, the "covering technique" is most effective. Chemical pleurodesis has shown a great number of recurrences and has led to inconveniences, such as various results among individuals. Therefore, it should not be the recommended therapy. Intractable pneumothorax in elderly patients has been increasing year after year. Interventional treatments such as endobronchial Watanabe spigot embolization and the thoracographic fibrin glue sealing method are expected to become more common in the future, keeping some patients from undergoing surgery. Causes are still unknown for certain pneumothoraces in women, and there are still numerous cases of postoperative recurrence among young males. Further studies in this field are expected in future.
    General Thoracic and Cardiovascular Surgery 03/2010; 58(3):113-9.
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    ABSTRACT: To describe in detail the characteristic chest computed tomography (CT) findings of Birt-Hogg-Dubé (BHD) syndrome. Thin-section chest CT scans of consecutive 12 patients with genetically diagnosed BHD syndrome were retrospectively evaluated by two observers, especially about the characteristics (distribution, number, size, shape and relation to pleura) of pulmonary cysts. Interobserver agreement in the identification of abnormalities on the CT images was achieved using the κ statistic, and the degree of interobserver correlation for the characterization of pulmonary cysts was assessed using the Spearman rank correlation coefficient. Multiple pulmonary cysts were seen in all patients. The number of cysts in each patient was various (range, 29-407), and cysts of various sizes (from a few mm to 2 cm or more) were seen in all patient. 76.6% (mean) of cysts were irregular-shaped, and 40.5% (mean) of cysts were located along the pleura. The mean extent score of cysts was 13% of the whole lung, and the distribution of cysts was predominantly in the lower medial zone. Finally, cysts abutting or including the proximal portions of lower pulmonary arteries or veins were also seen in all patients. Multiple, irregular-shaped cysts of various sizes with lower medial lung zone predominance are characteristic CT findings of BHD syndrome. Cysts abutting or including the proximal portions of lower pulmonary arteries or veins may also exist in this syndrome in a high probability.
    European journal of radiology 09/2009; 77(3):403-9. · 2.65 Impact Factor
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    ABSTRACT: Birt-Hogg-Dubé (BHD) syndrome, a rare inherited autosomal genodermatosis first recognised in 1977, is characterised by fibrofolliculomas of the skin, an increased risk of renal tumours and multiple lung cysts with spontaneous pneumothorax. The BHD gene, a tumour suppressor gene located at chromosome 17p11.2, has recently been shown to be defective. Recent genetic studies revealed that clinical pictures of the disease may be variable and may not always present the full expression of the phenotypes. We hypothesised that mutations of the BHD gene are responsible for patients who have multiple lung cysts of which the underlying causes have not yet been elucidated. We studied eight patients with lung cysts, without skin and renal disease; seven of these patients have a history of spontaneous pneumothorax and five have a family history of pneumothorax. The BHD gene was examined using PCR, denaturing high-performance liquid chromatography and direct sequencing. We found that five of the eight patients had a BHD germline mutation. All mutations were unique and four of them were novel, including three different deletions or insertions detected in exons 6, 12 and 13, respectively and one splice acceptor site mutation in intron 5 resulting in an in-frame deletion of exon 6. We found that germline mutations of the BHD gene are involved in some patients with multiple lung cysts and pneumothorax. Pulmonologists should be aware that BHD syndrome can occur as an isolated phenotype with pulmonary involvement.
    Journal of Medical Genetics 10/2007; 44(9):588-93. · 5.70 Impact Factor
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    ABSTRACT: Lymphangioleiomyomatosis (LAM) is a rare destructive lung disease characterized by an abnormal proliferation of smooth muscle-like cells (LAM cells) in the lung and along the axial lymphatics. LAM demonstrates a heterogeneous clinical course, but there is no serum surrogate marker available for assessing the disease severity or predicting the disease progression. Since the authors have recently demonstrated the extensive LAM-associated lymphangiogenesis and its potential role in progression and metastasis of LAM cells, they hypothesized that serum levels of lymphangiogenic growth factors might be increased in LAM and become a surrogate marker for disease severity. VEGF-A, VEGF-C, and VEGF-D in serum of 44 patients with LAM were measured by enzyme-linked immunosorbant assay. Only VEGF-D was significantly increased in LAM patients as compared with age- and gender-matched healthy volunteers (n=24) (LAM vs. control, geometric mean 95% CI; 1069.3 pg/mL (809.4 approximately 1412.6) vs. 295.9 pg/mL (262.6 approximately 333.5), p<0.0001). Serum VEGF-D levels negatively correlated with variables of pulmonary function tests, FEV1/FVC (forced expiratory volume in one second/forced vital capacity) (r=-0.365, p<0.05) and %DLco/VA (the percentage of diffusing capacity for carbon monoxide/alveolar volume to the predicted value) (r=-0.560, p<0.001). As expected, the group who received hormone therapy showed more deteriorated pulmonary function with higher serum VEGF-D levels than the group who was just observed without hormone therapy. Immunohistochemical examination of lung specimens demonstrated the positive immunoreactivity of LAM cells for VEGF-D. Serum VEGF-D levels may be a valuable surrogate marker for evaluating the disease severity in LAM.
    Lymphatic Research and Biology 02/2006; 4(3):143-52. · 2.33 Impact Factor