Laparoscopic Excision of a Uterine Adenomatoid Tumor and a Coexisting Ovarian Teratoma: A Case Report and Literature Review.

Adenomatoid tumors (ATs) are rare, benign neoplasms occurring mainly in reproductive organs such as the uterus, ovaries, fallopian tubes, and testes. Uterine adenomatoid tumors (UATs) are generally incidentally diagnosed during histopathological examination of excisional biopsies performed for other indications, most commonly uterine leiomyomas. We herein present a 38-year-old woman who underwent laparoscopic excision of a uterine leiomyoma and a right ovarian teratoma. Microscopic examination of the excisional biopsy revealed that the enucleated uterine tumor was composed of proliferating glandular tissue covered with single-layered cells that were surrounded by proliferating smooth muscle cells, corresponding exactly to the features of UATs. The excised ovarian cyst was confirmed to be a typical mature cystic teratoma. According to these histopathological findings, the patient was finally diagnosed with a UAT and coexisting teratoma. No recurrence was detected up to 6 months after excision. To the best of our knowledge, this is the eighth case report on laparoscopically enucleated UATs. Although recurrence risk may be low in UATs, further case reports are necessary to elucidate the safety and validity of laparoscopic excision for UATs.

Microscopic polyangiitis preceded by combined pulmonary fibrosis and emphysema.

A 73-year-old male metalworker was admitted to our hospital with a 3-year history of progressive dry cough. Chest high-resolution computed tomography revealed emphysematous changes and reticular lesions, which is referred to as combined pulmonary fibrosis and emphysema (CPFE). Surgical lung biopsy specimens revealed unclassified interstitial pneumonia, including a nonspecific interstitial pneumonia pattern and usual interstitial pneumonia pattern. Two years after his first admission he developed rapid progressive renal dysfunction with an elevated level of myeloperoxidase-antineutrophil cytoplasmic antibody (428 EU). A renal biopsy specimen revealed interstitial nephritis and glomerulonephritis. Consequently, microscopic polyangiitis preceded by CPFE was diagnosed. Despite transient exacerbation of renal involvement, his general condition remained mostly stable during a 2-year period of corticosteroid treatment. He ultimately died from severe pneumococcal pneumonia associated with acute lung injury.

Complete response of 7 years' duration after chemoradiotherapy followed by gefitinib in a patient with intramedullary spinal cord metastasis from lung adenocarcinoma.

Intramedullary spinal cord metastasis is a rare but serious complication which causes rapid progression of neurological deficits. Here we report a 35-year-old man presenting with increasing leg pain and gait disturbance, 8 months after surgery for lung adenocarcinoma. Spinal magnetic resonance imaging revealed an intramedullary tumor at the Th7/8 level. Radiotherapy at 35 Gy resulted in transient symptomatic improvement, but during chemotherapy with vinorelbine and cisplatin, symptoms worsened again. Gefitinib was then administered; the patient improved after 2 weeks and has now maintained a complete response for 7 years.

Histopathological evaluation of the efficacy of antifungals for experimental Trichosporon bloodstream infection.

The efficacy of polyene macrolides to treat experimental Trichosporon bloodstream infection was evaluated by histopathological examination and viable cell counts in the kidneys of infected mice. Viable cell counts on the 5th day after infection confirmed that liposomal amphotericin B (L-AMB) is a more effective treatment than fluconazole (FLC) for mice infected with an azole-resistant strain of Trichosporon. Histological examination revealed that the administration of L-AMB induced a transformation from acute purulent inflammation caused by both azole-susceptible and -resistant strain infections to a chronic and subsiding form, whereas FLC failed to convert the acute inflammation induced by the azole-resistant strain to a subsiding form. Our results demonstrate that polyene macrolides can be used as an alternative therapy for infection of azole-resistant strains of Trichosporon and that histopathological evaluation is useful for elucidating the pathophysiology of an experimental Trichosporon infection.

Stromal micropapillary component as a novel unfavorable prognostic factor of lung adenocarcinoma.

BACKGROUND: Pulmonary adenocarcinomas with a micropapillary component having small papillary tufts and lacking a central fibrovascular core are thought to result in poor prognosis. However, the component consists of tumor cells often floating within alveolar spaces (aerogenous micropapillary component [AMPC]) rather than invading fibrotic stroma observed in other organs like breast (stromal invasive micropapillary component [SMPC]). We previously observed cases of lung adenocarcinoma with predominant SMPC that was associated with micropapillary growth of tumors in fibrotic stroma observed in other organs. We evaluated the incidence and clinicopathological characteristics of SMPC in lung adenocarcinoma cases. PATIENTS AND METHODS: We investigated the clinicopathological characteristics and prognostic significance of SMPC in lung adenocarcinoma cases by reviewing 559 patients who had undergone surgical resection. We examined the SMPC by performing immunohistochemical analysis with 17 antibodies and by genetic analysis with epidermal growth factor receptor (EGFR) and KRAS mutations. RESULTS: SMPC-positive (SMPC(+)) tumors were observed in 19 cases (3.4%). The presence of SMPC was significantly associated with tumor size, advanced-stage disease, lymph node metastasis, pleural invasion, lymphatic invasion, and vascular invasion. Patients with SMPC(+) tumors had significantly poorer outcomes than those with SMPC-negative tumors. Multivariate analysis revealed that SMPC was a significant independent prognostic factor of lung adenocarcinoma, especially for disease-free survival of pathological stage I patients (p = 0.035). SMPC showed significantly higher expression of E-cadherin and lower expression of CD44 than the corresponding expression levels shown by AMPC and showed lower surfactant apoprotein A and phospho-c-Met expression level than corresponding expression levels shown by tumor cell components without a micropapillary component. Fourteen cases with SMPC(+) tumors (74%) showed EGFR mutations, and none of them showed KRAS mutations. CONCLUSIONS: SMPC(+) tumors are rare, but they may be associated with a poor prognosis and have different phenotypic and genotypic characteristics from those of AMPC(+) tumors.

Prognostic value of preoperative FDG-PET in stage IA lung adenocarcinoma.

BACKGROUND: Maximum standardized uptake value (SUVmax) of 18F-fluorodeoxyglucose positron emission tomography (FDG-PET) has been found to have prognostic value. We previously reported the correlation between SUVmax and pathological invasive area, and determined an SUVmax cut-off value of 2.15 for predicting the recurrence potential of an invasive area of diameter 5mm. Here, we evaluate the validity of FDG-PET for prediction of recurrence in pathological stage IA lung adenocarcinoma. METHODS: From February 2006 to May 2008, 100 patients with pathological stage IA lung adenocarcinoma underwent complete resection at our hospital. Tumors were classified as air-type or solid-type based on thin-section computed tomography (TS-CT) findings and the influence of TS-CT classification, SUVmax, and clinicopathologic features were evaluated in terms of the incidence of recurrence. RESULTS: Unlike air-type adenocarcinomas, recurrent disease was detected in 8 of 62 solid-type adenocarcinomas. SUVmax and diameter of invasive area were significantly correlated with recurrence and a shorter time to recurrence. All 8 recurrent cases had pathological invasive area >5mm. All except one case of recurrence were solid-type adenocarcinomas with SUVmax≥2.15. Three-year disease-free survival rates were 100% in air-type adenocarcinomas, 97.1% in solid-type adenocarcinomas with SUVmax<2.15, and 74.1% in solid-type adenocarcinoma with SUVmax≥2.15. CONCLUSION: Combined evaluation of TS-CT classification and SUVmax had significant value in predicting recurrence in stage IA lung adenocarcinoma, reflecting the aggressiveness of primary lung adenocarcinoma. Prediction of tumor aggressiveness could contribute to decision-making regarding the choice of surgical procedure and treatment after surgery.

Stromal micropapillary pattern predominant lung adenocarcinoma--a report of two cases.

Generally, adenocarcinomas with micropapillary pattern, featuring small papillary tufts lacking a central fibrovascular core, are thought to have poor prognosis. This pattern has been described in various organs. However, tumor cells with micropapillary pattern of lung adenocarcinoma are more often seen to float within alveolar spaces (aerogenous micropapillary pattern, AMP) than in fibrotic stroma like other organs (stromal micropapillary pattern, SMP) and SMP predominant lung adenocarcinoma (SMPPLA) has not been well described yet. We presented two cases of SMPPLA which were found in the last four years. Both the cases showed more than 50% of SMP in the tumor area. The majority of the stromal micropapillary clusters expressed MUC1 and epithelial membrane antigen along the outer surface of cell membrane. On the other hand, connective tissues surrounding stromal micropapillary clusters showed no reactivity for epithelial markers (thyroid transcription factor-1 and cytokeratin) or endothelial marker (D2-40 and CD34). It means clusters of SMP do not exist within air space or lymphatic or vessel lumens. The tumors with SMP often presented lymphatic permeation and vessel invasion, and intriguingly, one of the two cases showed metastasis to the mediastinal lymph node. Additionally, both the cases showed EGFR point mutations of exon 21. These results suggest that SMPPLA might be associated with poor prognosis and effective for EGFR tyrosine kinase inhibitors.

Papillary adenoma of the lung with a peculiar raw macroscopic feature.

We report a case of papillary adenoma of the lung with a peculiar raw macroscopic feature at intraoperative consultation. A 52-year-old man was admitted to our hospital for the evaluation of a solitary peripheral nodule in the left lower lobe which was detected with chest CT. When we took staples off from the stump of the partially resected lung in order to make a frozen section diagnosis, granular fragments leaked out from the stump. On the cut surface, the dark reddish and granular tumor grew expansively; however, hemorrhage and necrosis were absent. Histologically, granular fragments were mainly composed of papillary structures, which consisted of a single layer of cuboidal to low columnar cells with round to oval nuclei lining the surface of the fibrovascular cores. Characteristically, papillary structures lacked elastic fibers in the stroma and were packed within an elastic fiber framework derived from pre-existing alveolar structures. We considered that high intratumoral pressure might have made the granular fragments leak out of the stump as soon as we removed staples and that peculiar macroscopic findings before fixation may be a diagnostic clue for papillary adenoma.

Subpleural honeycombing on high resolution computed tomography is risk factor for fatal pneumonitis.

BACKGROUND: Postoperative interstitial pneumonitis is a life-threatening complication after lung cancer surgery. We conducted this study to identify risk factors for postoperative interstitial pneumonitis in patients with no clinical evidence of interstitial lung disease. METHODS: We retrospectively studied patients who underwent lung cancer resection. The characteristics of patients were analyzed by reviewing their clinical and surgical records and preoperative chest high-resolution computed tomographic scans. Postoperative interstitial pneumonitis was defined as acute severe hypoxemia accompanied by radiographic diffuse interstitial infiltrates of the lung with no apparent cause within a few weeks after surgery. RESULTS: From 2002 through 2005, 651 patients were evaluated, operated on, and managed by the same team. Postoperative interstitial pneumonia developed in 7 patients (7 of 651, 1.1%). Five of these patients had local, but not diffuse, dorsal subpleural honeycombing occupying three or more segments in both lower lobes on high-resolution computed tomography (CT honeycombing). During the same period, 46 patients had CT honeycombing. The incidence of postoperative interstitial pneumonia was 10.9% (5 of 46) among patients with CT honeycombing and 0.3% (2 of 605) among those without CT honeycombing. Four of the 7 (57%) patients with postoperative interstitial pneumonia died of respiratory failure. Mortality among the patients who had postoperative interstitial pneumonia as well as CT honeycombing was 80% (4 of 5); in contrast, none of the patients without CT honeycombing died. Multivariate analyses showed that the presence of CT honeycombing and prolonged operation time were significant risk factors. CONCLUSIONS: Subpleural honeycombing on high-resolution computed tomography is a significant predictor of postoperative interstitial pneumonia in asymptomatic patients who undergo resection for lung cancer.

Alveolar space filling ratio as a favorable prognostic factor in small peripheral squamous cell carcinoma of the lung.

INTRODUCTION: Squamous cell carcinomas (SqCCs) of the lung can be divided into two types according to the location of primary site; one is central type and another is peripheral type. Many reports on the central type revealed the clinicopathological characteristics relating carcinogenesis, therapeutics and prognosis. On the other hand, those on the peripheral type are very a few and prognostic indicators of peripheral type have not been enough elucidated. The aim of this study was to clarify clinicopathological prognostic factors of small peripheral SqCCs of the lung 30 mm or less. MATERIALS AND METHODS: We evaluated various 15 clinicopathological parameters in 81 patients with peripheral type SqCCs, which are defined as tumors located in or more peripheral from the third branching bronchus, measuring 30 mm or less in diameter. RESULTS: Univariate analyses were performed using the log lank test and multivariate analyses using logistic regression model. As a result, two factors had a statistically significant influence on outcome of the patients in the univariate analysis; no relapse was observed in the patients with the ratio of alveolar space filling (ASF) area to tumor area of 70% or more and the maximum diameter of invasive area measuring 10 mm or less in size (P=0.0214, P=0.0373, respectively). Meanwhile, multivariate analysis showed that the ASF ratio of 70% or more significantly affected the outcome of the patients (P=0.0337), however the maximum diameter of invasive area did not (P=0.2136). We could not show the unfavorable prognostic factor contributory to tumor relapse. CONCLUSIONS: We have shown that the ASF ratio is a significantly favorable prognostic factor for small peripheral type. Especially the focally invasive tumors with ASF ratio of 70% or more might be classified as "a microinvasive carcinoma" of the peripheral SqCCs of the lung and tumors with ASF ratio 100% as noninvasive carcinoma.

Recurrent EML4-ALK-associated lung adenocarcinoma with a slow clinical course.

The fusion gene EML4-ALK (echinoderm microtubule-associated protein-like 4 gene and the anaplastic lymphoma kinase gene) was recently identified as a novel genetic alteration in non-small-cell lung cancer. The clinicopathological features of EML4-ALK-positive adenocarcinoma are reported to include its high incidence in young, non-smoking patients, tumors that show distinct solid or acinar growth patterns with or without signet-ring cell histology, and its mutually exclusive occurrence with mutations in EGFR and KRAS. However, the clinical findings have not been well described. Here, we report a case of EML4-ALK-positive lung adenocarcinoma that showed multiple metachronous lesions on the pleura and pulmonary field, suspected to be a recurrence of lung adenocarcinoma after a 20-year disease-free interval. The slow clinical course may be characteristic of EML4-ALK-positive lung adenocarcinoma. Therefore, long-term observation of patients with EML4-ALK-positive lung adenocarcinomas is required after surgery.

Duodenal gangliocytic paraganglioma showing lymph node metastasis: a rare case report.

We describe a case of duodenal gangliocytic paraganglioma showing lymph node metastasis. A 61-year-old Japanese man underwent pylorus preserving pancreaticoduodenectomy to remove a tumor at the papilla of Vater. The section of the tumor extending from the mucosa to submucosa of the duodenum was sharply demarcated, solid, and white-yellowish. Neither necrosis nor hemorrhage was present. Histological examination confirmed the immunohistochemical identification of three components comprising epithelioid cells, spindle-shaped cells, and ganglion-like cells. Epithelioid cells showed positive reactivity for synaptophysin, somatostatin, and CD56. In contrast, spindle-shaped cells showed positive reactivity for S-100 protein, but not for synaptophysin, somatostatin or CD56. Furthermore, we found lymph node metastasis despite lack of bcl-2 and p53 expression. In addition to the rarity of the tumor, we are describing here the present case suggests the malignant potency of the tumor despite lack of acceptable prognostic indicators for neuroendocrine tumor.

Surgical outcomes and reevaluation of treatment strategies for thymomas.

Improved histological typing systems for thymic tumors and advances in induction and adjuvant therapy have created the need to reevaluate strategies for the management of thymoma. We retrospectively studied 73 patients with completely resected thymomas unassociated with myasthenia gravis. The World Health Organization (WHO) histologic classification, clinicopathological features and surgical outcomes were analyzed. Overall survival was 66.2% at 10 years, and the median survival time was 169 months. According to the Masaoka staging system, overall survival rates at 10 years were 94.7% in stage I, 76.1% in stage II, 30% in stage III and 0% in stage IV. In the WHO classification, overall survival rates at 10 years were 91.9% in types A and AB, 50.9% in type B2 and not achieved in type B3. The disease-free interval was slightly shorter in patients with B2 and B3 disease than in those with type A, AB and B1 disease. Advanced thymomas were significantly associated with type B2 and B3 (p<0.01). In stage III and IV disease, adjuvant or neoadjuvant therapy was associated with better survival as compared to no adjuvant therapy (p=0.07). On multivariate analysis, Masaoka stage III and IV disease and extended thymectomy indicated significant, negative and independent risk factors for survival (p<0.01). Masaoka stage I and II thymomas or WHO type A and AB thymomas have favorable prognoses and do not require postoperative adjuvant therapy. Patients with stage III and IV thymomas require additional therapy after surgery.

Churg-Strauss syndrome presenting with diffuse alveolar hemorrhage and rapidly progressive glomerulonephritis.

A 46-year-old man with a 4-month history of bronchial asthma was admitted to our hospital complaining of progressive dyspnea, weakness of the lower extremities, multiple truncal erythematous purpura and hemoptysis. Neurological examination identified the presence of mononeuritis multiplex. Laboratory data indicated marked anemia, eosinophilia, severe renal failure with nephrotic condition and elevated serum myeloperoxidase anti-neutrophil cytoplasmic antibody (MPO-ANCA) levels (1,050 EU). Chest computed tomography showed diffuse ground glass opacity in both lungs. Bronchoalveolar lavage fluid revealed bloody fluid with eosinophilia (81%). Microscopic findings of a transbronchial lung biopsy were consistent with alveolar hemorrhage. A skin biopsy revealed eosinophilic vasculitis consistent with Churg-Strauss syndrome (CSS). A renal biopsy specimen revealed pauci-immune crescentic necrotizing glomerulonephritis. Consequently, he was diagnosed as having CSS presenting with diffuse alveolar hemorrhage (DAH) and rapidly progressive glomerulonephritis (RPGN) with MPO-ANCA-associated systemic vasculitis. His clinical condition markedly improved with the administration of intravenous corticosteroid (CS) and cyclophosphamide (CY). Thus, we report a case of CSS presenting with the rare complication of DAH and RPGN.

[Pathophysiology of invasive fungal infection in diabetic patients].

It has been known that diabetes mellitus impairs functioning of neutrophil, macrophage, cellular immunity, humoral immunity, and iron metabolism. In addition to them, diabetes-related angiopathy leads a patient to being at high-risk individual for several kinds of infectious diseases. Therefore, diabetes has been accepted as one of the important risk factors for invasive fungal infection. From the viewpoint of pathology, the present review describes both pathophysiology of immunosuppression induced by diabetes and histopathological characteristics of typical forms in invasive fungal infection when it occurred as an opportunistic infection; those are candidiasis, aspergillosis, and cryptococcosis. We wish to draw that pathophysiological explanation still remains obscuring of relationship between diabetes and invasive fungal infection.

Pathophysiological study of chronic necrotizing pulmonary aspergillosis.

The aim of the present study is to define the characteristics of the clinical and histopathological features of chronic necrotizing pulmonary aspergillosis (CNPA) cases with severe hemoptysis. We conducted a histological study of three patients clinically diagnosed as having CNPA who had hemoptysis for 5 years. A tuberculosis sequelae was found as the underlying disorder in all three cases. All patients had fever, general fatigue, and hemoptysis, and their chest computed tomographic images revealed fungus balls, cavity wall thickening, consolidation surrounding the cavity, and satellite foci. All had been treated with anti-fungal drugs and corticosteroids. However, all patients died from respiratory failure due to massive hemoptysis. Histopathological examination revealed that the cavity wall consisted of three layers comprised of necrotic, granulation, and fibrous tissue layers. Aspergilli were found in both the fungus ball and necrotic tissue comprising the inner layer of the cavity. In addition, most of the vessels were incompletely occluded with thrombosis and were necrotic, as well as showing local invasion of Aspergilli. Surgical intervention should be considered as a prior procedure for CNPA patients, because vessels at the cavity wall, whether occluded completely or incompletely, are usually necrotic and/or show local invasion of Aspergilli.

A case of concomitant occurrence of struma ovarii and malignant transformation of cystic teratoma.

A 77-year-old woman received a total abdominal hysterectomy and bilateral salpingo-oophorectomy because of a tumor in the left ovary. The surgical specimen measured 8.5x4.5x4.0 cm, and the solid lesion measured 4.0x3.5x3.5 cm. The solid lesion was diagnosed as struma ovarii. The cyst wall partially comprised squamous epithelium-like and ciliated columnar epithelium-like cells. The tumorous lesion of the cyst wall revealed a poorly differentiated adenocarcinoma. Immunohistochemically, the tumor cells were positive for cytokeratin7, and were negative for cytokeratin20 and thyroid transcription factor-1. The authors diagnosed that struma ovarii and other parats coexisted as a poorly differentiated adenocarcinoma that had arisen from a mature ovarian cystic teratoma. As for the identification of the origin of adenocarcinomas arising from mature ovarian cystic teratomas, more cases need to be identified and investigated.

[Clinicopathological study of invasive pulmonary aspergillosis complicated by leukemia].

OBJECTIVE: We assessed the clinicopathological features of invasive pulmonary aspergillosis. PATIENTS AND METHODS: We conducted chest radiograph-pathologic correlations in 8 cases with invasive pulmonary aspergillosis diagnosed at autopsy. RESULTS: Subjects were 4 men and 4 women with a mean age of 57.4 years and all having leukemia as an underlying hematological disorder. All had been treated with anticancerous drugs and corticosteroids. Histopathologically, coagulation necrosis with marginal hemorrhage and agranulocytosis were observed in 6 whose initial abnormal shadows in chest X-ray appeared during the neutropenic phase less than 500/microL. In contrast, numerous infiltration of neutrophils and colliquation necrosis at the center of lesions were observed in 2 whose initial abnormal shadows in chest X-ray appeared having more than 500/microL of neutrophils in the peripheral blood.

[A case of pleural sarcoidosis in which vats lung biopsy, obtained pleural and pulmonary lesions].

Here we report a case that was diagnosed as sarcoidosis but required differential diagnosis from pneumoconiosis. A 51-year-old asymptomatic man, who showed signs of bilateral hilar lymphadenopathy (BHL) on a chest X-ray taken during a medical check, was given a diagnosis of sarcoidosis, based on the results of mediastinoscopic mediastinal lymph node biopsy. Because of the presence of large and small nodular lesions adjacent to the pleura extending from the bilateral upper lobes into the lung field, and continuous bead-like, small nodular lesions in the right interlobar pleura, pleural sarcoidosis was suspected and thoracoscopy was performed. Macroscopically, multiple grayish-white nodules with distinct margins, up to 1cm in diameter surrounded by a proliferation of capillaries were found in the pleura, particularly in the upper lobes. Lesions were also scattered over the interlobar pleura and diaphragmatic surface. Histopathologically, several non-caseous epithelioid cell granulomas and silicotic nodule-like lesions of hyaline degeneration were found; therefore, pneumoconiosis, or more specifically chronic berylliosis, was suspected. Despite these symptoms, the patient did not have a history of exposure, and the results of the lymphocyte stimulation test using beryllium were negative in blood and bronchoalveolar lavage fluid. The patient was given a diagnosis of pleural sarcoidosis and has been observed without treatment.

[Nursing experience in palliative care at the outpatient clinic].

Seven of the 17 patients reported that they were satisfied with the palliative care at our outpatient clinic. A patient's will to stay home, a devoted key person to the patient, procedures such as good pain control and an oral intake were considered to be important factors in palliative care at the outpatient clinic. We should understand that the patient's family attending the outpatient clinic is a part of their important daily life, so we try to make them feel comfortable whenever they come to see us. We should also strive for building more experience in palliative care for patients and their families. Based on our experience, we anticipate that outpatient care will be increased in the future. In the meantime, advanced nursing skills and techniques are needed.