Chronic pelvic pain due to pelvic lymphangioleiomyomatosis: A case report

AbstractLymphangioleiomyomatosis (LAM), a rare multisystem disease affecting mainly young women, is characterized by an abnormal proliferation of smooth muscle-like cells in the lungs. We report a case of endometriosis with chronic pelvic pain due to pelvic LAM. A 41-year-old gravida 1, para 1 woman had been experiencing intermittent left pelvic pain for several years. She also complained of dyspnea on effort 2 years previously, and was diagnosed with pulmonary LAM. Abdominal magnetic resonance imaging showed a right ovarian endometriotic cyst and a left pelvic mass. She was referred to our hospital for the treatment of pelvic pain; she underwent laparoscopic cystectomy of the right ovarian endometriotic cyst. Her left pelvic cyst was found in the retroperitoneal space, and biopsy confirmed the diagnosis of LAM. Therefore, we suggest that LAM localized in the pelvis should be considered when a patient with pulmonary LAM presents with pelvic pain or abdominal distention

Regulation of 3β-Hydroxysteroid Dehydrogenase Activity in Rat Testis under Hyperprolactinemia and Excessive 17β-Estradiol

The effects of hyperprolactinemia on the activity of 3β-hydroxysteroid dehydrogenase (3β-HSD) in rat testis were studied during excessive 17β-estradiol (E2) administration. Without E2 treatment, 3β-HSD activity did not change significantly in moderate hyperprolactinemic rats (756 ± 179 ng/ml). However, this enzyme activity was significantly decreased in marked hyperprolactinemic rats (3612 ± 1090 ng/ml) compared to that in control rats (45.4 ± 6.2 ng/ml). Under the excessive E2 administration, this enzyme activity was insignificantly decreased in slight hyperprolactinemic rats (218 ± 42 ng/ml1 and inhibited in moderate hyperprolactinemic rats (566 ± 77 ng/ml) as refer to control level. It may be concluded that inhibition of 3β-HSD activity in testis due to the excess administration of estrogen is promoted by the transplantation of MtT/F84 which produces prolactin at the limited level

Long-term Follow-up of Orthodontic Patient with Cervical Lymphangioma

Aim: To report a treatment case of mandibular deviation caused by congenital cervical lymphangioma with traditional orthodontic techniques, following-up by 10-year retention. Background: Lymphangiomas, developmental anomalies, can induce various disturbances of swallowing, mastication, speech, breathing, and skeletal deformities as well as psychological stress and anxiety for the patient and their family. Lymphangiomas are benign with virtually no possibility of turning into a malignant lesion, so clinical management aims to treat the patient functionally. Case description: A girl, aged 6 years and 4 months, complained about facial asymmetry and anterior crossbite caused by congenital cervical lymphangioma. Her facial profile was the straight type with an adequate lip position. Anterior and right-side posterior crossbites were observed. On the frontal cephalogram, the menton shifted 3.0 mm to the right. A functional appliance with an expander was placed to correct her dental midline deviation and posterior crossbite. After 2-year treatment, the anterior and right-side posterior crossbites were improved. Multibracket treatment began after the growth spurt. After 44-month active treatment, a functional occlusion, including a Class I molar relationship with a proper interincisal relationship, was achieved. A functional occlusion was maintained during a 10-year retention period, while a mandibular downward growth was observed through the retention period. Conclusion: Conventional orthodontic techniques enable functional and stable occlusion even in patients with mandibular deviation caused by congenital cervical lymphangioma, although only using early orthodontic management by itself may have some limitations. Clinical significance: The hybrid technique combining functional appliance and intermaxillary elastics proves to be an effective therapy for correcting occlusal cant and mandibular deviation caused by cervical lymphangioma

Causal relations of health indices inferred statistically using the DirectLiNGAM

Causal relations among many statistical variables have been assessed using a Linear non-Gaussian Acyclic Model (LiNGAM). Using access to large amounts of health checkup data from Osaka prefecture obtained during the six fiscal years of years 2012–2017, we applied the DirectLiNGAM algorithm as a trial to extract causal relations among health indices for age groups and genders. Results show that LiNGAM yields interesting and reasonable results, suggesting causal relations and correlation among the statistical indices used for these analyses

Gradient boosting decision tree becomes more reliable than logistic regression in predicting probability for diabetes with big data

We sought to verify the reliability of machine learning (ML) in developing diabetes prediction models by utilizing big data. To this end, we compared the reliability of gradient boosting decision tree (GBDT) and logistic regression (LR) models using data obtained from the Kokuho-database of the Osaka prefecture, Japan. To develop the models, we focused on 16 predictors from health checkup data from April 2013 to December 2014. A total of 277,651 eligible participants were studied. The prediction models were developed using a light gradient boosting machine (LightGBM), which is an effective GBDT implementation algorithm, and LR. Their reliabilities were measured based on expected calibration error (ECE), negative log-likelihood (Logloss), and reliability diagrams. Similarly, their classification accuracies were measured in the area under the curve (AUC). We further analyzed their reliabilities while changing the sample size for training. Among the 277,651 participants, 15,900 (7978 males and 7922 females) were newly diagnosed with diabetes within 3 years. LightGBM (LR) achieved an ECE of 0.0018 ± 0.00033 (0.0048 ± 0.00058), a Logloss of 0.167 ± 0.00062 (0.172 ± 0.00090), and an AUC of 0.844 ± 0.0025 (0.826 ± 0.0035). From sample size analysis, the reliability of LightGBM became higher than LR when the sample size increased more than 104. Thus, we confirmed that GBDT provides a more reliable model than that of LR in the development of diabetes prediction models using big data. ML could potentially produce a highly reliable diabetes prediction model, a helpful tool for improving lifestyle and preventing diabetes

Fetal and Neonatal Goiter in Cynomolgus Monkeys Following Administration of the Antithyroid Drug Thiamazole at High Doses to Dams During Pregnancy

To evaluate morphologic alterations in the thyroid gland in the second generation in cynomolgus monkeys, pregnant dams were exposed to high doses of thiamazole. In Experiment A, dams received thiamazole intragastrically via a nasogastric catheter from gestation day (GD) 50 to GD 150 or on the day before delivery. Initially, the dose level was 20 mg/kg/day (10 mg/kg twice daily); however, the dose level was subsequently decreased to 5 mg/kg/day (2.5 mg/kg twice daily), since deteriorated general conditions were observed in two dams. Six out of seven neonates died on the day of birth. The cause of neonatal death was tracheal compression and suffocation from goiter. The transplacental exposure to thiamazole affected the fetal thyroid glands and induced goiter in all neonates. The surviving neonate was necropsied 767 days after discontinuation of thiamazole exposure and showed reversibility of the induced changes. In Experiment B, dams were intragastrically administered thiamazole at 5 mg/kg/day (2.5 mg/kg twice daily) for treatment periods from GDs 51 to 70, 71 to 90, 91 to 110, 111 to 130 and 131 to 150. All fetuses showed enlarged thyroid glands but were viable. Histopathologically, hypertrophy and/or hyperplastic appearance of the follicular epithelium of the thyroid gland was observed at the end of each treatment period. The most active appearance of the follicular epithelium, consisting of crowded pedunculated structure, was demonstrated at end of the treatment period from GD 131 to 150. This is the first report on the morphology of fetal and neonatal goiter in the cynomolgus monkey

Primary tumor-secreted lymphangiogenic factors induce pre-metastatic lymphvascular niche formation at sentinel lymph nodes in oral squamous cell carcinoma

Objectives: The objectives of this study were to evaluate the formation of lymphvascular niches in lymph nodes of patients with oral squamous cell carcinoma (OSCC), and investigate the roles of lymphangiogenic and angiogenic factors, such as vascular endothelial growth factor (VEGF)-A, VEGF-C, and VEGF-D, expressed in the primary tumors. Materials and Methods: Forty-four patients with previously untreated clinically late T2 or T3 OSCC of cN0 were evaluated for primary tumors and 166 sentinel lymph nodes (SLNs). Primary tumors were immunohistochemically analyzed for expressions of VEGFs. Densities of lymphatic vessels (LVDpodoplanin) and high endothelial venules (HEVD) in the SLNs were also calculated using antibodies for each marker, podoplanin and MECA-79, respectively. Results: In 25 patients, all lymph nodes were metastasis-negative, whereas, in 19 patients, metastasis was positive for at least one lymph node (either at SLN, non-SLN, or nodal recurrence). From the analyses of 140 SLNs without metastasis, LVDpodoplanin in 50 SLNs of metastasis-positive cases was significantly higher than that in 90 SLNs of metastasis-negative cases (p = 0.0025). HEVD was not associated with lymph node metastasis. The patients with VEGF-A-High or VEGF-D-High tumors had significantly higher LVDpodoplanin than patients with their Low counterparts (p = 0.0233 and p = 0.0209, respectively). In cases with lymph node metastasis, the VEGF-D-expression score was significantly higher than in those without lymph node metastasis (p = 0.0006). Conclusions: These results suggest that lymph node lymphangiogenesis occurs before metastasis in OSCC. VEGF-A and VEGF-D play critical roles in this process. VEGF-D is a potential predictive marker of positive lymph node metastasis in cN0 patients. © 2015 Wakisaka et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited

Dysconnectivity of the Agency Network in Schizophrenia: A Functional Magnetic Resonance Imaging Study

Background: Self-disturbances in schizophrenia have recently been explained by an abnormality in the sense of agency (SoA). The cerebral structures of SoA in healthy people are considered to mainly include the insula and inferior parietal lobule. In contrast, the functional lesion of aberrant SoA in schizophrenia is not yet fully understood. Considering the recent explanation of establishing SoA from the standpoint of associative learning, the “agency network” may include not only the insula and inferior parietal lobule but also the striatum. We hypothesized that aberrant SoA in schizophrenia is based on a deficit in the “agency network.”Methods: Functional magnetic resonance imaging data were acquired while patients with schizophrenia (n = 15) and matched controls (n = 15) performed our adaptation method of agency attribution task on a trial-by-trial basis to assess participants' explicit experience of the temporal causal relationship between an action and an external event with temporal biases. Analysis of functional connectivity was done using the right supramarginal gyrus and the right middle frontal gyrus as seed regions.Results: In healthy controls, analyses revealed increased activation of the right inferior parietal lobule (mainly the supramarginal gyrus), right insula, and right middle frontal gyrus as an activation of the agency condition. We defined activated Brodmann areas shown in the agency condition of healthy controls as the seed region for connectivity analysis. The connectivity analysis revealed lower connectivity between the head of the left caudate nucleus and right supramarginal gyrus in the patients compared to healthy controls.Conclusions: This dysconnectivity of the agency network in schizophrenia may lead to self-disturbance through deficits in associative learning of SoA. These findings may explain why pathological function of the striatum in schizophrenia leads to self-disturbance

Genetic cholesteryl ester transfer protein deficiency caused by two prevalent mutations as a major determinant of increased levels of high density lipoprotein cholesterol

金沢大学大学院医学系研究科病態検査学Genetic determinants of HDL cholesterol (HDL-C) levels in the general population are poorly understood. We previously described plasma cholesteryl ester transfer protein (CETP) deficiency due to an intron 14 G(+1)-to-A mutation(Int14 A) in several families with very high HDL-C levels in Japan. Subjects with HDL-C ≥ 100 mg/dl (n = 130) were screened by PCR single strand conformational polymorphism analysis of the CETP gene. Two other mutations were identified by DNA sequencing or primer-mediated restriction map modification of PCR products: a novel intron 14 splice donor site mutation caused by a T insertion at position +3 from the exon14/intron14 boundary (Int14 T) and a missense mutation (Asp442 to Gly) within exon 15 (D442G). The Int14 T mutation was only found in one family. However, the D442G and Int14 A mutations were highly prevalent in subjects with HDL-C ≥ 60 mg/dl, with combined allele frequencies of 9%, 12%, 21%, and 43% for HDL-C 60-79, 80-99, 100-119, and ≥ 120 mg/dl, respectively. Furthermore, prevalences of the D442G and Int14 A mutations were extremely high in a general sample of Japanese men (n = 236), with heterozygote frequencies of 7% and 2%, respectively. These two mutations accounted for about 10% of the total variance of HDL-C in this population. The phenotype in a genetic compound heterozygote (Int14 T and Int14 A) was similar to that of Int14 A homozygotes (no detectable CETP and markedly increased HDL-C), indicating that the Int14 T produces a null allele. In four D442G homozygotes, mean HDL-C levels (86±26 mg/dl) were lower than in Int14 A homozygotes (158±35 mg/dl), reflecting residual CETP activity in plasma. In 47 D442G heterozygotes, mean HDL-C levels were 91±23 mg/dl, similar to the level in D442G homozygotes, and significantly greater than mean HDL-C levels in Int14 A heterozygotes (69±15 mg/dl). Thus, the D442G mutation acts differently to the null mutations with weaker effects on HDL in the homozygous state and stronger effects in the heterozygotes, suggesting dominant expression of a partially defective allele. CETP deficiency, reflecting two prevalent mutations (D442G and Int14 A), is the first example of a genetic deficiency state which is sufficiently common to explain a significant fraction of the variation in HDL-C in the general population

Effects of the development changes in dentition on the retention of mouthguards

Summary The use of mouthguards is one of the main strategies that are considered effective in preventing sports–related trauma.Although mouthguards are recommended for use in children during sports activities,little is known as to how growth and developmental changes in the oral cavity, jaw, and dentition affect the retention of mouthguards. In the present study, we designed mouthguards for primary,mixed(4 types),and permanent detention stages and performed tensile testing to determine the effects of dental growth and development on their retention.We also designed mouthguards for the left maxillary second milk molar,left maxillary second premolar, and left maxillary first molar to determine how the shape of the teeth affects their retention.Data was analyzed statistically,and the following results were obtained:1.Mouthguards for permanent detention stage had a significantly higher level of retention than those for primary and mixed detention stages.2.Mouthguards for primary detention stage had a significantly lower level of retention than those for mixed and permanent detention stages. 3.The retention of mixed detention stage mouthguards became poorer with the number of teeth requiring relief.4.Differences in the shape of the teeth(left maxillary second milk molar, left maxillary second premolar, left maxillary first molar)had no significant effect on the retention of mouthguards.These findings indicate that the level of mouthguard retention is lower in children at primary and mixed detention stages than in adults and children with complete eruption of central incisor to second molar.This difference may be attributed to the differences in the coverage areas of mouthguard