[A CASE OF SUSPECTED ALLERGIC BRONCHOPULMONARY MYCOSIS CAUSED BY SCEDOSPORIUM APIOSPERMUM THAT SHOWED CLINICAL IMPROVEMENT AFTER BRONCHOSCOPIC REMOVAL OF A MUCUS PLUG].

A 69-year-old woman presented with a persistent cough and high fever. Thoracic computed tomography revealed atelectasis and high-attenuation mucus. The blood test results showed eosinophils at 18.2%, an absolute eosinophil count of 980 cells/µL, and a total serum immunoglobulin E of 1980IU/mL. Bronchoscopy revealed a mucous plug, which upon photomicrograph examination, showed eosinophils. A culture study of the mucus yielded Scedosporium apiospermum, leading to the suspicion of allergic bronchopulmonary mycosis (ABPM) caused by the fungus. After the bronchoscopic removal of the mucous plug, her symptoms quickly diminished. She was successfully treated without medication, and ABPM has not recurred for 2 years. To our knowledge, ABPM caused by Scedosporium apiospermum is rare, and close follow-up was effective without the administration of systemic steroids or antifungal drugs.

Fistula formation into other organs secondary to intraductal papillary mucinous neoplasm of the pancreas: A case report and literature review.

INTRODUCTION: Fistula formation from intraductal papillary mucinous neoplasm (IPMN) of the pancreas is uncommon; however, IPMN penetrating various organs has been increasingly reported. To date, there is a lack of literature reviewing recent reports and the clinicopathologic details of IPMN with fistula formation are still poorly understood. METHODS: This study describes the case of a 60-year-old woman presenting with postprandial epigastric pain and diagnosed with main-duct (MD) IPMN penetrating to the duodenum, and presents comprehensive literature review of IPMN with fistulae. A literature review was performed using PubMed for all articles in English using predetermined search terms, including (fistula or fistulization), (pancreas or pancreatic or pancreato or pacreatico), (intraductal papillary mucinous), and (neoplasm or tumor or carcinoma or cancer). RESULTS: A total of 83 cases and 119 organs were identified in 54 articles. Affected organs were as follows: the stomach (34%), duodenum (30%), bile duct (25%), colon (5%), small intestine (3%), spleen (2%), portal vein (1%), and chest wall (1%). Fistula formation into multiple organs was detected in 35% of cases. Approximately one-third of the cases had tumor invasion around the fistula. MD and mixed type IPMN accounted for 82% of cases. IPMN with high-grade dysplasia or invasive carcinoma were over three times more common than IPMN without these components. DISCUSSION AND CONCLUSION: Based on the pathological examination of the surgical specimen, this case was diagnosed of MD-IPMN with invasive carcinoma and mechanical penetration or autodigestion was considered as the mechanism of fistula formation. Given the high risk of malignant transformation and intraductal dissemination of the tumor cells, aggressive surgical strategies, such as total pancreatectomy, should be recommended to achieve complete resection for MD-IPMN with fistula formation.

The efficacy of botulinum toxin type-A for intractable chronic migraine patients with no pain-free time.

AIM: This is a retrospective report of the efficacy of botulinum toxin-A, Botox® (Allergan), in intractable chronic migraine patients non-responsive to previous pharmacological management and with largely no pain-free time, including those with new onset daily persistent headache. METHODS: Thirty-three patients, all with severe Headache Impact Test (HIT)-6 scores at baseline, received 3-monthly injections of Botox® as per Phase III REsearch Evaluating Migraine Prophylaxis Therapy (PRE-EMPT) protocol over a maximum 33-month period. Response criteria were a sustained reduction of HIT-6 scores below 60. RESULTS: Four patients had headache on at least 20 days a month; the remaining patients had daily headache with no pain-free time, including nine patients with new onset persistent migraine. There was a significant reduction in HIT-6 scores following Botox® therapy (x̅ = -5.45, p = 0.000920). Twenty-one percent of the cohort exhibited a sustained reduction in HIT-6 scores below 60. The number of headache days and pain-free time did not change in five of the six responders, but disability improved. There was no difference between patients with episodic migraine evolving to chronic as opposed to those with chronic migraine from onset. CONCLUSION: This report suggests that Botox® treatment is efficacious in intractable chronic migraine without pain-free time. The HIT-6 is a reliable and practical parameter to assess disability in this patient group. Use of such validated parameters should be considered with greater weight in future International Classification of Headache Disorders (ICHD) guidelines for controlled clinical trials.

Allogeneic hematopoietic stem cell transplantation for Chediak-Higashi syndrome.

The clinical outcome of allogeneic HSCT was retrospectively analyzed in eight patients with CHS. In total, six of these eight patients are alive. Four of five patients transplanted with MAC achieved prompt engraftment, and three of the four patients, including two patients with AP before transplant, are alive without disease. In contrast, three patients transplanted with RIC without active AP disease achieved prompt engraftment and survive long term. RIC-HSCT might be an alternative treatment for CHS similar to other types of HLH, at least for patients without active AP disease.

Generalized infantile myofibromatosis with a monophasic primitive pattern.

Infantile myofibromatosis (IM) is a rare disorder present at birth or in early infancy with a biphasic histological pattern. We present a neonatal-onset case of generalized IM with visceral (central nervous system, heart, lungs, liver, spleen, small intestine, kidneys and bones) and placental involvement, showing a monophasic histological pattern through the lesions during the course of disease. Histologically, the tumor was composed of a solid proliferation of cytologically uniform, 'primitive' mesenchymal cells associated with a hemangiopericytoma-like vascular pattern. Immunohistochemical analysis and ultrastructural study revealed that the tumor cells exhibited primitive features without mature myofibroblastic differentiation. Neither ETV6-NTRK3 nor ACTB-GLI fusion gene was identified. The patient died of cerebral hemorrhage and respiratory failure at four months of age despite intensive therapy. Generalized IM characterized by monophasic primitive pattern could be related to poor clinical outcome.

Dentilisin involvement in coaggregation between Treponema denticola and Tannerella forsythia.

Periodontitis arises from a biofilm consisting of gram-negative anaerobic rods and spirochetes. Porphyromonas gingivalis, Treponema denticola, and Tannerella forsythia, termed the Red complex, have been co-isolated with high frequency from chronic periodontitis lesions, and these microorganisms are thought to be major pathogens of the disease. Coaggregation is an important strategy in the colonization of dental plaque biofilm by these bacteria. In the present study, we investigated the coaggregation of T. denticola strains with T. forsythia ATCC 43037 by use of visual grading or spectrophotometry. T. denticola ATCC 35405 coaggregated with T. forsythia, reaching a plateau at approximately 60 min. This coaggregation was inhibited by heat treatment of T. denticola ATCC 35405, but not of T. forsythia. Disaccharides such as sucrose, maltose, and lactose inhibited coaggregation by approximately 50%. The coaggregation reaction varied among T. denticola strains. There was somewhat less coaggregation between T. denticola ATCC 33520 and T. forsythia than between T. denticola ATCC 35405 and T. forsythia, although this difference was not statistically significant; T. denticola ATCC 33521 showed a trace level of coaggregation with T. forsythia. The magnitude of coaggregation among the three T. denticola strains was proportional to their dentilisin activities. Inactivation of dentilisin abolished coaggregation activity, but inactivation of the major outer sheath protein did not. In addition, phenylmethylsulfonyl fluoride did not affect coaggregation. These results indicate that dentilisin is involved indirectly in the coaggregation between T. denticola and T. forsythia, because its proteolytic activity is not required, possibly via ligand maturation.

Hemagglutinin/Adhesin domains of Porphyromonas gingivalis play key roles in coaggregation with Treponema denticola.

Porphyromonas gingivalis and Treponema denticola are major pathogens of periodontal disease. Coaggregation between microorganisms plays a key role in the colonization of the gingival crevice and the organization of periodontopathic biofilms. We investigated the involvement of surface ligands of P. gingivalis in coaggregation. Two triple mutants of P. gingivalis lacking Arg-gingipain A (RgpA), Lys-gingipain (Kgp) and Hemagglutinin A (HagA) or RgpA, Arg-gingipain B (RgpB) and Kgp showed significantly decreased coaggregation with T. denticola, whereas coaggregation with a major fimbriae (FimA)-deficient mutant was the same as that with the P. gingivalis wild-type parent strain. rgpA, kgp and hagA code for proteins that contain 44 kDa Hgp44 adhesin domains. The coaggregation activity of an rgpA kgp mutant was significantly higher than that of the rgpA kgp hagA mutant. Furthermore, anti-Hgp44 immunoglobulin G reduced coaggregation between P. gingivalis wild type and T. denticola. Treponema denticola sonicates adhered to recombinant Rgp domains. Coaggregation following co-culture of the rgpA kgp hagA mutant expressing the RgpB protease with the rgpA rgpB kgp mutant expressing the unprocessed HagA protein was enhanced compared with that of each triple mutant with T. denticola. These results indicate that the processed P. gingivalis surface Hgp44 domains are key adhesion factors for coaggregation with T. denticola.

Results of 1-year follow-up examinations after intravitreal bevacizumab administration for idiopathic choroidal neovascularization.

PURPOSE: The purpose of this study was to report the results of 1-year follow-up examinations after intravitreal bevacizumab injection for the treatment of idiopathic choroidal neovascularization. METHODS: Seven eyes in 7 patients with idiopathic choroidal neovascularization were intravitreally injected with 1.25 mg/0.05 mL of bevacizumab. The need for retreatment was evaluated if spectral-domain optical coherence tomography showed intraretinal edema or subretinal fluid at the time of a 1-month follow-up examination. Fluorescein angiography was performed 1 year after the first injection. The primary outcome measures were best-corrected visual acuity and central foveal thickness using spectral-domain optical coherence tomography. RESULTS: All 7 eyes were assessed at a 1-year follow-up examination. The mean number of injections per eye was 2.7. The mean logarithm of the minimum angle of resolution best-corrected visual acuity improved significantly from 0.31 +/- 0.29 to 0.15 +/- 0.38 (P < 0.05). The mean central foveal thickness decreased from 332 +/- 83 microm to 261 +/- 66 microm (P < 0.01). Fluorescein angiography showed no leakage at 1 year in all eyes. All patients whose best-corrected visual acuity improved by > or =0.2 logarithm of the minimum angle of resolution had a visual acuity of > or =20/40 when first injected at baseline. CONCLUSION: The intravitreal injection of bevacizumab is effective for stabilizing or improving vision in patients with idiopathic choroidal neovascularization, as evaluated at a 1-year follow-up examination. In particular, this treatment may be well tolerated in patients with a visual acuity of > or =20/40 at baseline. Additional investigations are needed to assess the long-term safety and the optimal protocol for intravitreal bevacizumab administration.

Combined assessment of left ventricular dyssynchrony and contractility by speckled tracking strain imaging: a novel index for predicting responders to cardiac resynchronization therapy.

BACKGROUND: Mechanical dyssynchrony is an important factor in the response to cardiac resynchronization therapy (CRT). However, no echocardiographic measure can improve prediction of case selection for CRT. OBJECTIVE: The purpose of this study was to assess the efficacy of a newly combined echocardiographic index for ventricular dyssynchrony and contractility using speckled tracking strain analysis to predict responders to CRT. METHODS: Forty-seven patients with severe heart failure in New York Heart Association functional class III/IV, left ventricular ejection fraction /=130 ms were included in the study. Echocardiography was performed, and a novel index (i-Index), the product of radial dyssynchrony and radial strain, was calculated. Responder to CRT was defined as a patient with a >/=15% decrease in left ventricular end-systolic volume at 6-month follow-up. RESULTS: Thirty-two patients (68%) were classified as responders. The i-Index was significantly higher in responders than in nonresponders (3,450 +/- 1180 vs 1,481 +/- 841, P <.001). The area under receiver operator characteristic curve was 0.92 for the i-Index, which was better than the index of radial dyssynchrony only (0.74). A cutoff value of i-Index >2,000 predicted responders with 94% sensitivity and 80% specificity. The index using only radial dyssynchrony had 81% sensitivity and 53% specificity. Furthermore, i-Index decreased in responders (1,985 +/- 1261, P <.001) but not in nonresponders (1,684 +/- 866, P = .48). CONCLUSION: Our findings suggest that a novel combined index by radial strain echocardiography might be a predictor of response to CRT. The value of this novel echocardiographic index requires further assessment in larger studies.

Influence of activity level on the outcome of treatment of lateral ankle ligament rupture.

BACKGROUND: Few studies of treatment of lateral ankle ligament rupture have given careful consideration to the activity levels of patients. In the present study, we investigated whether a patient's activity level influences the outcome of the treatment of lateral ankle ligament rupture and whether treatment should be determined according to activity level. METHODS: A total of 115 consecutive patients with lateral ankle ligament rupture were treated with short-term cast immobilization followed by functional bracing. We divided preinjury activity levels and activity levels at follow-up into four classes according to the International Knee Documentation Committee standards. Follow-up examination 1 year after injury included clinical assessment (anterior drawer test, range of motion, maximum circumference of the calf, tenderness, swelling), reinjury assessment, and functional evaluation by means of the Karlsson scoring system. RESULTS: We found significant differences in ankle stability and reinjuries between the four groups classified by their preinjury activity level. The number of patients with ankle instability and reinjury was significantly higher in the high-activity group than in the low-activity groups. We also found significant difference between groups classified by activity level at follow-up with regard to the Karlsson score. CONCLUSIONS: The outcome of treatment of lateral ankle ligament rupture is significantly influenced by the patient activity level, and treatment may need to be determined according to this activity level.

[A case of sarcomatoid malignant peritoneal mesothelioma responding to combination chemotherapy of cyclophosphamide, vincristine, adriamycin and dacarbazine(CYVADIC)].

A 66-year-old woman was seen at our hospital because of abdominal fullness. A computed tomography(CT)revealed massive tumors in abdominal cavity. The patient underwent surgery consisting of tumorectomy, segmental gastrectomy, partial resection of small intestin, transverse colectomy, left oophorectomy and gastrostomy. By using immunohistochemical staining, the patient was diagnosed as sarcomatoid malignant peritoneal mesothelioma. Rapidly abdominal fullness occurred as of 22 days after the operation, and an abdominal CT revealed the massive recurrent tumors. We started a combination chemotherapy of cyclophosphamide, vincristine, adriamycin and dacarbazine (CYVADIC). The recurrent tumors showed remarkable reduction after the two courses of CYVADIC chemotherapy. Although we next started carboplatin and paclitaxel combination chemotherapy, she died due to rapidly progression of the disease with disseminated intravascular coagulation after 132 days of the operation. Malignant mesothelioma, especially sarcomatoid mesothelioma, is known to have a poor prognosis. However, our case suggests that we could improve the prognosis of sarcomatoid malignant mesothelioma by aggressive chemotherapy.

Minute ampullary carcinoid tumor with lymph node metastases: a case report and review of literature.

BACKGROUND: Carcinoid tumors are usually considered to have a low degree of malignancy and show slow progression. One of the factors indicating the malignancy of these tumors is their size, and small ampullary carcinoid tumors have been sometimes treated by endoscopic resection. CASE PRESENTATION: We report a case of a 63-year-old woman with a minute ampullary carcinoid tumor that was 7 mm in diameter, but was associated with 2 peripancreatic lymph node metastases. Mild elevation of liver enzymes was found at her regular medical check-up. Computed tomography (CT) revealed a markedly dilated common bile duct (CBD) and two enlarged peripancreatic lymph nodes. Endoscopy showed that the ampulla was slightly enlarged by a submucosal tumor. The biopsy specimen revealed tumor cells that showed monotonous proliferation suggestive of a carcinoid tumor. She underwent a pylorus-preserving whipple resection with lymph node dissection. The resected lesion was a small submucosal tumor (7 mm in diameter) at the ampulla, with metastasis to 2 peripancreatic lymph nodes, and it was diagnosed as a malignant carcinoid tumor. CONCLUSION: Recently there have been some reports of endoscopic ampullectomy for small carcinoid tumors. However, this case suggests that attention should be paid to the possibility of lymph node metastases as well as that of regional infiltration of the tumor even for minute ampullary carcinoid tumors to provide the best chance for cure.

High volume acupuncture clinic (HVAC) for chronic knee pain--audit of a possible model for delivery of acupuncture in the National Health Service.

Recent research has established the efficacy, effectiveness and cost effectiveness of acupuncture for some forms of chronic musculoskeletal pain. However, there are practical problems with delivery which currently prevent its large scale implementation in the National Health Service. We have developed a delivery model at our hospital, a 'high volume' acupuncture clinic (HVAC) in which patients are treated in a group setting for single conditions using standardised or semi-standardised electroacupuncture protocols by practitioners with basic training. We discuss our experiences using this model for chronic knee pain and present an outcome audit for the first 77 patients, demonstrating satisfactory initial (eight week) clinical results. Longer term (one year) data are currently being collected and the model should next be tested in primary care to confirm its feasibility.

[Renal parencymal malacoplakia successfully treated with conservative therapy; a case report].

Malacoplakia is a relatively rare form of chronic granulomatous inflammation. Which occurs most frequently in the genito-urinary system, but renal parenchymal involvement is rare. We present a case of malacoplakia of the renal parenchyma. A 69-year-old woman with left renal mass was referred to our department. Abdominal enhanced CT scan revealed heterogeneous mass in the left kidney (50 x 45 mm). CT-guided percutaneous left renal needle biopsy confirmed the diagnosis of renal malakoplakia. We successfully treated her with oral levofloxacin. The lesion resolved after the 4-months-therapy, and there has been no recurrence for 22 months since the treatment.

Sex differences in the relationships between electrocardiographic abnormalities and the extent of left ventricular hypertrophy by echocardiography.

BACKGROUND: Several criteria have been proposed for the electrocardiographic diagnosis of left ventricular hypertrophy (LVH). However, their diagnostic accuracy is questionable. Furthermore, the diagnostic accuracy of abnormalities in ST-T patterns for LVH is known to be uncertain, especially in women. We examined the relationship between electrocardiographic abnormalities and the extent of LVH. METHODS: We studied 76 men and 48 women who satisfied electrocardiographic voltage criteria for LVH (RV(5) or RV(6)> or = 2.6 mV, SV(1) + RV(5) or SV(1) + RV(6)> or = 3.5 mV). They were classified into three groups based on ST-T pattern: normal, early strain, and strain. We defined echocardiographic evidence of LVH as an LV wall thickness > or = 12 mm. RESULTS: LVH was identified by echocardiography in 55.3% of men and in 47.9% of women. In strain and early strain groups, the prevalence of echocardiographic LVH was significantly higher in men than in women (strain group: 100 vs 75%, P < 0.05, early strain group: 81.8 vs 42.1%, P < 0.05), it did not differ significantly between men and women in normal group. In men, QRS voltage values were significantly correlated with echocardiographic indices. In group strain of men, significant good correlations were observed between QRS voltage values and echocardiographic indices. However, in women, there were no significant correlation between QRS voltage values and echocardiographic indices even in strain group. CONCLUSIONS: The combined criteria of both QRS voltage and ST-T classification could provide a greater accuracy in diagnosing LVH compared to the criteria using QRS voltage alone in men rather than in women.

Correlation between detection rates of periodontopathic bacterial DNA in coronary stenotic artery plaque [corrected] and in dental plaque samples.

Utilizing PCR, the 16S rRNA detection rates for Porphyromonas gingivalis, Actinobacillus actinomycetemcomitans, Bacteroides forsythus, Treponema denticola, and Campylobacter rectus in samples of stenotic coronary artery plaques were determined to be 21.6, 23.3, 5.9, 23.5, and 15.7%, respectively. The detection rates for P. gingivalis and C. rectus correlated with their presence in subgingival plaque.