Publications by authors named "Tomohiro Saito"

71 Publications

The Prevention Measures for COVID-19 and Changes in Kawasaki Disease Incidence.

J Epidemiol 2021 Sep 4. Epub 2021 Sep 4.

Department of Pediatrics, University of Yamanashi.

BackgroundKawasaki disease is suspected to be triggered by previous infection. The prevention measures for COVID-19 have reportedly reduced transmission of certain infectious diseases. Under these circumstances, the prevention measures for COVID-19 may reduce the incidence of Kawasaki disease.MethodWe conducted a retrospective study using registration datasets of patients with Kawasaki disease who were diagnosed in all 11 in-patient pediatric facilities in Yamanashi prefecture. The eligible cases were 595 cases that were diagnosed before the COVID-19 pandemic (from January 2015 to February 2020) and 38 cases that were diagnosed during the COVID-19 pandemic (from March to November 2020). Incidence of several infectious disease were evaluated using data from the Infectious Disease Weekly Report conducted by the National Institute of Infectious Diseases.ResultsEpidemics of various infectious diseases generally remained at low levels during the first nine months (March to November 2020) of the COVID-19 pandemic. Moreover, the incidence of COVID-19 was 50-80 times lower than the incidence in European countries and the United States. The total number of 38 cases with Kawasaki disease for the nine months during the COVID-19 pandemic was 46.3% (-3.5 standard deviation (SD)) of the average (82.0 (SD, 12.7)) for the corresponding nine months of the previous five years. None of the 38 cases was determined to be triggered by COVID-19 based on their medical histories and negative results of severe acute respiratory syndrome coronavirus 2 testing at admission.ConclusionThese observations provide a new epidemiological evidence for the notion that Kawasaki disease is triggered by major infectious diseases in children.
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http://dx.doi.org/10.2188/jea.JE20210132DOI Listing
September 2021

Mixed alkali-ion transport and storage in atomic-disordered honeycomb layered NaKNiTeO.

Nat Commun 2021 Aug 2;12(1):4660. Epub 2021 Aug 2.

Tsukuba Laboratory, Technical Solution Headquarters, Sumika Chemical Analysis Service (SCAS), Ltd., Tsukuba, Japan.

Honeycomb layered oxides constitute an emerging class of materials that show interesting physicochemical and electrochemical properties. However, the development of these materials is still limited. Here, we report the combined use of alkali atoms (Na and K) to produce a mixed-alkali honeycomb layered oxide material, namely, NaKNiTeO. Via transmission electron microscopy measurements, we reveal the local atomic structural disorders characterised by aperiodic stacking and incoherency in the alternating arrangement of Na and K atoms. We also investigate the possibility of mixed electrochemical transport and storage of Na and K ions in NaKNiTeO. In particular, we report an average discharge cell voltage of about 4 V and a specific capacity of around 80 mAh g at low specific currents (i.e., < 10 mA g) when a NaKNiTeO-based positive electrode is combined with a room-temperature NaK liquid alloy negative electrode using an ionic liquid-based electrolyte solution. These results represent a step towards the use of tailored cathode active materials for "dendrite-free" electrochemical energy storage systems exploiting room-temperature liquid alkali metal alloy materials.
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http://dx.doi.org/10.1038/s41467-021-24694-5DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8329229PMC
August 2021

Legg-Calvé-Perthes disease following Ender nail fixation of a pediatric femoral fracture.

Int J Surg Case Rep 2021 Aug 17;85:106209. Epub 2021 Jul 17.

Department of Orthopaedic Surgery, Jichi Medical University, Tochigi, Japan.

Introduction And Importance: Legg-Calvé-Perthes disease (LCPD) sometimes occur in children, however it is difficult to diagnose it at the early stage especially in the cases there are no complaints of symptoms. Femoral shaft fractures in children cause various complications such as leg-length discrepancy, nonunion and malunion, refracture, and osteonecrosis of the femoral head. We presented a rare case in which a pediatric patient developed LCPD after femoral shaft fracture.

Case Presentation: A healthy 8-year-old boy sustained a left femoral diaphyseal fracture following a pedestrian car accident. Fixation was achieved using retrograde Ender nails; bone union was confirmed at 3 months postoperatively, and the Ender nails were removed at 8 months postoperatively without any problems. Unfortunately, the morphological change of the ipsilateral femoral head and subtle symptoms were missed until the femoral head collapsed. LCPD was successfully treated with intertrochanteric varus osteotomy, which achieved a good clinical result.

Clinical Discussion: Although the reason for the ipsilateral LCPD after the femoral shaft fracture is unclear, this case highlights the need for close postoperative follow-up of pediatric femoral fractures resulting from high-energy trauma to prevent the misdiagnosis of this coincidental complication.

Conclusion: This case report describes a missed ipsilateral LCPD after a femoral diaphyseal fracture caused by high-energy trauma. Close postoperative follow-up with a detailed assessment and vigilant interpretation of postoperative radiography is imperative to avoid delayed/missed diagnosis of conditions for which early management may provide better outcomes.
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http://dx.doi.org/10.1016/j.ijscr.2021.106209DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8319750PMC
August 2021

Screening of frequent variants associated with congenital hypothyroidism: a comparison with next generation sequencing.

Endocr J 2021 Jul 8. Epub 2021 Jul 8.

Department of Pediatrics, Faculty of Medicine, University of Yamanashi, Yamanashi 409-3898, Japan.

Congenital hypothyroidism (CH) is considered the most common congenital endocrine disorder of genetic origin. Next generation sequencing (NGS) is the standard method for identifying genetic mutations, but it is an expensive and complex technique. Therefore, we propose to use Sanger sequencing to identify selected variants of the four most common CH-causative genes: DUOX2, TG, TSHR, and PAX8. To analyze the performance of Sanger sequencing, we compared its variant detection ability with that of a CH NGS panel containing 53 genes. We performed Sanger sequencing of selected variants and panel NGS analysis of 25 Japanese patients with CH. Sanger sequencing identified nine variants in seven patients, while NGS identified 24 variants in 14 patients. Of these, eight, five, eight, two, and one were found to be potentially pathogenic in DUOX2, TSHR, TG, UBR1, and TPO genes, respectively. The percentage of detectable variants using Sanger sequencing compared with NGS was 37.5% (9/24 variants), whereas the percentage of detectable cases carrying variants using Sanger sequencing compared with NGS was 50% (7/14 patients). We proposed a system for screening commonly identified CH-related variants by Sanger sequencing. Sanger sequencing could therefore identify about a third of CH-causative variants, so is considered an effective and efficient form of pre-screening before NGS.
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http://dx.doi.org/10.1507/endocrj.EJ21-0353DOI Listing
July 2021

Incidence of childhood type 1 diabetes mellitus in Yamanashi Prefecture, Japan, 1986-2018.

Endocrinol Diabetes Metab 2021 04 14;4(2):e00214. Epub 2020 Dec 14.

Department of Pediatrics Faculty of Medicine Saitama Medical University Saitama Japan.

Introduction: Several studies have examined the incidence of childhood T1DM in Japan from the 1970s onwards, but none have been long-term studies using registration data. We estimate the incidence of childhood type 1 diabetes mellitus (T1DM) from 1986 to 2018 in Yamanashi Prefecture, Japan.

Methods: We began a population-based, long-term study of childhood T1DM in 1986 involving every hospital paediatrics department in Yamanashi Prefecture. In the Prefecture, every child newly diagnosed with T1DM is referred to a hospital, and therefore, almost 100% of new patients aged <15 years are registered. We calculated the incidence of T1DM among children aged <15 years from 1986 to 2018. All cases met the Japan Diabetes Society diagnostic criteria and were tested for T1DM-related autoantibodies whenever possible.

Results: Ninety-nine patients (44 boys and 55 girls) were newly diagnosed with T1DM. The annual incidence among 5- to 9-year-olds increased by 5.35% over the study period (95% confidence interval 2.34%-8.35%, = .0005), and there was a trend towards increasing 3-year incidence (15.52% increase, = .0516). There were also trends towards increasing annual and 3-year incidence among 0- to 14-year-olds. However, there were no changes over time in annual or 3-year incidence in the 0-4 year or 10-14 year age groups.

Conclusions: The incidence of T1DM in Yamanashi Prefecture increased among children aged 0-14 years over the study period, with the most significant increase occurring among 5- to 9-year-olds. These data suggest that the number of children aged <15 years with T1DM is gradually increasing in one of the local prefectures in Japan, Yamanashi Prefecture and that the age of onset is decreasing.
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http://dx.doi.org/10.1002/edm2.214DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8029530PMC
April 2021

Evaluation of Tibial Tunnel Location with the Femoral Tunnel Created Behind the Resident's Ridge in Transtibial Anterior Cruciate Ligament Reconstruction.

J Knee Surg 2021 Jan 22. Epub 2021 Jan 22.

Department of Orthopaedic Surgery, Jichi Medical University, Shimotsuke, Japan.

Few studies have determined whether a femoral bone tunnel could be created behind the resident's ridge by using a transtibial (TT) technique-single bundle (SB)-anterior cruciate ligament (ACL) reconstruction. The aim of this study was to clarify (1) whether it is possible to create a femoral bone tunnel behind the resident's ridge by using the TT technique with SB ACL reconstruction, (2) to define the mean tibial and femoral tunnel angles during anatomic SB ACL reconstruction, and (3) to clarify the tibial tunnel inlet location when the femoral tunnel is created behind resident's ridge. Arthroscopic TT-SB ACL reconstruction was performed on 36 patients with ACL injuries. The point where 2.4-mm guide pin was inserted was confirmed, via anteromedial portal, to consider a location behind the resident's ridge. Then, an 8-mm diameter femoral tunnel with a 4.5-mm socket was created. Tunnel positions were evaluated by using three-dimensional computed tomography (3D-CT) 1 week postoperatively. Quadrant method and the resident's ridge on 3D-CT were evaluated to determine whether femoral tunnel position was anatomical. Radiological evaluations of tunnel positions yielded mean ( ±  standard deviation) X- and Y-axis values for the tunnel centers: femoral tunnel, 25.2% ± 5.1% and 41.6% ± 10.2%; tibial tunnel, 49.2% ± 3.5%, and 31.5% ± 7.7%. The bone tunnels were anatomically positioned in all cases. The femoral tunnel angle relative to femoral axis was 29.4 ± 5.5 degrees in the coronal view and 43.5 ± 8.0 degrees in the sagittal view. The tibial tunnel angle relative to tibial axis was 25.5 ± 5.3 degrees in the coronal view and 52.3 ± 4.6 degrees in the sagittal view. The created tibial bone tunnel inlet had an average distance of 13.4 ± 2.7 mm from the medial tibial joint line and 9.7 ± 1.7 mm medial from the axis of the tibia. Femoral bone tunnel could be created behind the resident's ridge with TT-SB ACL reconstruction. The tibial bone tunnel inlet averaged 13.4 mm from the medial tibial joint line and 9.7 mm medial from the tibia axis.
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http://dx.doi.org/10.1055/s-0040-1722568DOI Listing
January 2021

Tumoral calcinosis due to severe hyperphosphatemia and secondary hyperparathyroidism without vascular calcification in a hemodialysis patient.

Clin Nephrol 2021 Mar;95(3):166-170

We report a case of a 32-year-old man who was undergoing chronic hemodialysis and had hyperphosphatemia and secondary hyperparathyroidism (SHPT) with multiple tumoral calcinosis (TC) lesions refractory to drug therapy. Total parathyroidectomy and autotransplantation were performed, and he recovered from TC within 3 months. Several soft-tissue calcifications were present, but neither computed tomography (CT) before diagnosis nor CT performed 12 months after surgery detected evidence of vascular calcification (VC), despite persistence of hyperphosphatemia. This patient had a high calcium (Ca) × phosphate (P) product and calciprotein particles, and high serum Ca and P levels are important risk factors for both TC and VC. P plays a crucial role in regulation of VC, but the absence of VC in our case suggests a specific circumstance in which VC does not progress even under a high phosphatemic state, and that P alone may be insufficient for VC progression. TC in our patient was probably due to severe SHPT and continuous high serum P and Ca × P product levels, but the absence of VC suggests that the pathophysiologic process leading to VC requires further investigation, particularly in chronic kidney disease.
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http://dx.doi.org/10.5414/CN110201DOI Listing
March 2021

Clinical results of reverse shoulder arthroplasty for comminuted proximal humerus fractures in elderly patients: a comparison between nonporous stems versus trabecular metal stems.

JSES Int 2020 Dec 6;4(4):952-958. Epub 2020 Oct 6.

Department of Orthopaedics, Jichi Medical University Hospital, Tochigi, Japan.

Background: This study compared the clinical results for nonporous stems vs. trabecular metal (TM) stems used in reverse shoulder arthroplasty (RSA) for comminuted proximal humeral fractures (CPHFs) in elderly patients.

Methods: In this retrospective study, a total of 41 shoulders (39 women) of patients with CPHF aged >70 years who underwent RSA were investigated. The minimum follow-up period was 2 years. A total of 15 shoulders were treated with Grammont-style RSA using nonporous stems (the G-RSA group), and 26 shoulders were treated with RSA combining TM stems (the FR-RSA group). The American Shoulder and Elbow Surgeons (ASES) shoulder score, Constant score, shoulder joint range of motion (ROM), and radiographic findings were compared between the 2 groups.

Results: ASES scores and Constant scores were significantly higher in the FR-RSA group than in the G-RSA group. External rotation at the side in the FR-RSA group was significantly higher than that in the G-RSA group. In the FR-RSA and G-RSA groups, the union rates at the greater tuberosity (GT) were 88.5% and 46.7%, respectively, and scapular notching rates were 20% and 7.7%, respectively. Based on a subanalysis, the age was lower, body mass index was higher, and ASES scores, Constant scores, and external rotation ROM were higher in the GT union group than in the GT nonunion group.

Conclusion: GT bone union rates were high, and external rotation ROM of the shoulder joint were more improved for RSA using TM stems than those for RSA using nonporous stems in elderly patients with CPHF.
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http://dx.doi.org/10.1016/j.jseint.2020.08.010DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7738571PMC
December 2020

Satisfaction With Elbow Function and Return Status After Autologous Osteochondral Transplant for Capitellar Osteochondritis Dissecans in High School Baseball Players.

Am J Sports Med 2020 10 17;48(12):3057-3065. Epub 2020 Sep 17.

Jichi Medical University Hospital, Department of Orthopaedics, Tochigi, Japan.

Background: Osteochondral autograft transplant (OAT), a surgical treatment for capitellar osteochondritis dissecans (OCD), has favorable rates of elbow recovery and return to sports in adolescents. However, few reports have investigated how long patients continue to play baseball after OAT and their satisfaction with their treatment outcome.

Purpose: To evaluate the rate of boys who played baseball and received OAT for OCD in junior high school or earlier (age <15 years) and continued to play baseball in high school and the players' satisfaction with their elbow function during play.

Study Design: Cohort study; Level of evidence, 3.

Methods: A total of 32 elbows of boys who played baseball and received OAT at age ≤15 years (mean, 14.1 years) were examined and divided into pitcher (n = 11) and nonpitcher (n = 21) groups according to their player position before surgery. The clinical Timmerman-Andrews score at the end of their high school baseball, participation percentage of players who continued to play baseball, and satisfaction level during play (on a scale of 0-10 during pitching and batting and in a 4-choice format) were compared between the 2 groups.

Results: The Timmerman-Andrews scores significantly improved after surgery in both groups, with no significant difference between the groups. Of the 32 players, 30 (93.8%) continued to play baseball throughout high school, including all players in the pitcher group and 19 (90.5%) of those in the nonpitcher group. The percentage of players who continued to pitch was 55.6% (6/11) in the pitcher group. Satisfaction with elbow joint function at the time of pitching was significantly lower in the pitcher group. Further, 5 players reported being "a little unsatisfied" because of elbow pain during pitching. All of the players indicated satisfaction with elbow function during batting.

Conclusion: The percentage of players who received OAT for OCD in junior high school and continued to play baseball in high school was favorable. However, satisfaction with elbow function during throwing was lower in pitchers than in nonpitchers.

Clinical Relevance: Before surgery, consent should be obtained from patients who are pitchers after it is explained that satisfaction with elbow joint function during pitching could be decreased after OAT.
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http://dx.doi.org/10.1177/0363546520952782DOI Listing
October 2020

Characteristics of dynamic magnetic resonance imaging of symptomatic chronic calcifying tendinitis: preliminary case reports.

JSES Int 2020 Sep 18;4(3):555-558. Epub 2020 May 18.

Department of Orthopedic Surgery, Jichi Medical University, Tochigi, Japan.

Background: The symptoms of chronic calcifying tendinitis consist of shoulder contracture and impingement sign. However, there have been no reports about the use of imaging studies to differentiate these 2 clinical symptoms. A "burning sign" caused by abnormal blood flow was previously reported in the shoulder joint in patients with frozen shoulder by dynamic magnetic resonance imaging. This burning sign was related to pain. The purpose of this study was to investigate the dynamic magnetic resonance imaging findings in patients with symptomatic chronic calcifying tendinitis and to examine the relationship between the location of the burning sign and the physical findings.

Methods: We retrospectively analyzed data for 6 patients with symptomatic chronic calcifying tendinitis (mean age, 55.5 ± 9.3 years; 4 women). The range of shoulder motion, impingement sign, and location of the burning sign were assessed.

Results: Four patients had an impingement sign without shoulder contracture, and the other 2 patients had shoulder contracture. All the patients with an impingement sign also had a burning sign around the calcium deposit and no enhancement in the rotator interval and axillary pouch. Conversely, all the patients with contracture had a burning sign in the rotator interval and axillary pouch and no enhancement around the calcium deposit.

Conclusions: Dynamic magnetic resonance imaging identified 2 types of findings in patients with symptomatic chronic calcifying tendinitis: a burning sign in the rotator interval and axillary pouch or around the calcium deposit. The former pattern may be related to shoulder contracture, whereas the latter may be related to impingement sign.
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http://dx.doi.org/10.1016/j.jseint.2020.02.009DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7478990PMC
September 2020

Anti-MDA-5 antibody-positive clinically amyopathic dermatomyositis with rapidly progressive interstitial lung disease treated with therapeutic plasma exchange: A case series.

J Clin Apher 2021 Feb 21;36(1):196-205. Epub 2020 Aug 21.

Department of Medicine, Division of Nephrology, Showa University School of Medicine, Tokyo, Japan.

We present six cases of antimelanoma differentiation-associated gene 5 antibody (anti-MDA5-Ab)-positive clinically amyopathic dermatomyositis (CADM) with rapidly progressive interstitial lung disease (RP-ILD), which is known to have a poor prognosis. The outcomes of these cases are described after treatment with therapeutic plasma exchange (TPE). Clinical and therapeutic data for patients with CADM with RP-ILD were collected retrospectively from medical records. All six patients received early intensive care including high-dose corticosteroids, intravenous cyclophosphamide, and a calcineurin inhibitor, but lung disease and hypoxia became more severe. TPE was performed over a median of 9.5 sessions (range 3-14) per patient, and the median duration from admission to TPE was 23 days. Three patients received combined direct hemoperfusion using a polymyxin B-immobilized fiber column (PMX-DHP) therapy on successive days to manage acute respiratory failure. Four patients survived and two died due to respiratory failure. In the survival cases, ferritin decreased, and ferritin and KL-6 were lower at diagnosis. The patients who died had a higher alveolar-arterial oxygen difference and more severe lung lesions at the time of initiation of TPE. These findings indicate that a combination of conventional therapy and TPE may be useful for improvement of the prognosis of CADM with RP-ILD at the early stage of onset.
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http://dx.doi.org/10.1002/jca.21833DOI Listing
February 2021

Management of infected non-union of subtrochanteric fracture: Two cases.

Trauma Case Rep 2020 Apr 22;26:100293. Epub 2020 Feb 22.

Department of Orthopedic Surgery, School of Medicine, Jichi Medical University, 3311-1 Yakushiji, Shimotsuke 329-0498, Japan.

Infected non-union of subtrochanteric fractures is challenging to treat. We experienced two cases and had good clinical results. Treatment strategy comprised debridement without hesitation after considering later limb lengthening; insertion of the proximal lateral bone edge spike into the distal bone marrow cavity until achieving medial-side bony contact and holding good alignment to compensate for the medial-side bone loss, according to the modified Dimon method; and internal fixation with an angled plate in the decubitus position. The angle of the angled plate should be directed toward the abundant cancellous bone using preoperative computed tomography. Residual limb shortening after ORIF was improved by limb lengthening.
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http://dx.doi.org/10.1016/j.tcr.2020.100293DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7038002PMC
April 2020

Multidisciplinary management of calciphylaxis: a series of 5 patients at a single facility.

CEN Case Rep 2020 05 17;9(2):122-128. Epub 2019 Dec 17.

Division of Nephrology, Department of Medicine, Showa University School of Medicine, 1-5-8 Hatanodai, Shinagawa-ku, Tokyo, 142-8555, Japan.

Calciphylaxis is a rare and severe disease that manifests with painful skin ulceration and necrosis. Herein, we report five patients of hemodialysis patients with skin biopsy-proven calciphylaxis at a single facility. One patient had undergone parathyroidectomy (PTx) due to severe secondary hyperparathyroidism, four had been treated with vitamin D receptor activators, and two were on warfarin therapy. All patients had hyperphosphatemia, and one had hypercalcemia. The intact parathyroid hormone level at diagnosis was 2 pg/ml in the patient after PTx, while three patients were within the target range. The average period after diagnosis of calciphylaxis was 2 months. Skin lesions were present on the thighs and lower legs in two patients, and on the dorsum of the foot in one patient. In skin biopsy, calcification was found in the arteriolar media in four patients, and calcium (Ca) was deposited in the dermal lesion in one patient. All patients received local cures, surgical debridement, antibiotics to control infectious diseases, and strict control of serum Ca and phosphate. Calcimimetics were used in all patients except one who had undergone PTx one month before, sodium thiosulfate was used in 4 patients, and low Ca dialysate was used in three patients. The average follow-up period was 7.4 months. Four patients were cured, and one died due to infection. We suggest that multidisciplinary management for infectious diseases, surgical debridement, strict control of mineral and bone markers from the early stage, and elimination of risk factors may improve the course of calciphylaxis, which is a life-threatening disease.
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http://dx.doi.org/10.1007/s13730-019-00439-8DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7148403PMC
May 2020

Brachial plexus palsy after clavicle fracture: 3 cases.

J Shoulder Elbow Surg 2020 Feb 26;29(2):e60-e65. Epub 2019 Nov 26.

Department of Orthopaedic Surgery, Jichi Medical University, Tochigi, Japan.

Background: Brachial plexus palsy after clavicle fracture is extremely rare. We experienced 3 cases of brachial plexus palsy after clavicle fracture and investigated the findings that such patients have in common and the clinical results of these cases.

Methods: We retrospectively analyzed the data of 3 patients with clavicle fracture who had no neurovascular symptoms at the time of the initial injury but gradually developed brachial plexus palsy within 1 month after the injury. The patients were aged 70, 62, and 68 years; 2 patients were male and 1 was female. The patients' backgrounds and clinical results were assessed.

Results: All patients had a displaced middle-third clavicle fracture and underwent conservative therapy with a figure-8 bandage. The intervals between fracture and symptoms of brachial plexus palsy were 8, 30, and 14 days. The times from symptoms of brachial plexus palsy to surgery were 27, 75, and 28 days. In all patients, surgery revealed a ruptured subclavius muscle and abnormal development of granulation tissue around the fracture site, compressing the brachial plexus. Open reduction and plate fixation was performed in 2 patients, and clavicle resection was performed in 1 patient. The intervals between surgery and full recovery of muscle strength were 11, 6, and 6 months.

Conclusion: The findings our 3 patients with brachial plexus palsy after clavicle fracture had in common are old age, middle-third displaced clavicle fracture, and abnormal development of granulation tissue around the fracture site. Surgical intervention yielded good clinical outcomes.
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http://dx.doi.org/10.1016/j.jse.2019.09.027DOI Listing
February 2020

Norwood Operation With Anterior Translocation of Pulmonary Artery.

Ann Thorac Surg 2019 12 31;108(6):e387-e388. Epub 2019 May 31.

Department of Cardiovascular Surgery, Chiba Children's Hospital, Chiba, Japan.

Even in the current era of improved clinical outcome of Norwood operation, postoperative pulmonary artery stenosis, recoarctation of the aorta, ventricular dysfunction, and atrioventricular valve regurgitation remain serious clinical concerns. We present 2 cases of successful total cavopulmonary connection after Norwood operation using the right ventricle-to-pulmonary artery shunt with translocation of the pulmonary bifurcation anterior to the reconstructed neoaortic arch. This modification provided a better surgical access to an obstructive pulmonary artery at staged single ventricle strategy and a space for the growth of the pulmonary arteries.
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http://dx.doi.org/10.1016/j.athoracsur.2019.04.038DOI Listing
December 2019

The course and clinical impact of articular magnetic resonance imaging findings 6 months after shoulder manipulation under ultrasound-guided cervical nerve root block for frozen shoulder.

JSES Open Access 2019 Mar 18;3(1):21-24. Epub 2019 Mar 18.

Department of Radiology, Jichi Medical University, Tochigi, Japan.

Background: In our previous study, iatrogenic capsular tears, bone bruises of the humeral head, and labral tears were detected on magnetic resonance imaging (MRI) performed 1 week after manipulation following ultrasound-guided cervical nerve root block in patients with frozen shoulder 6 months after manipulation.

Methods: We studied 25 patients with frozen shoulder. MRI was performed before, 1 week after, and 6 months after manipulation. On the basis of the course of MRI findings over a period of 6 months, the patients were divided into 2 groups: those with MRI findings of bone bruises, capsular tears, and/or labral tears (19 patients) and those with no MRI findings (6 patients). The clinical outcomes of the 2 groups at 6 months after manipulation were compared using the Wilcoxon matched-pairs test, the Mann-Whitney test, and the Fisher exact probability test for statistical analysis.

Results: At 1 week after manipulation, 96% of patients had capsular tears, 40% had bone bruises, and 20% had labral tears; these percentages had decreased at 6 months after manipulation to 4%, 20%, and 8%, respectively. No significant differences in clinical outcomes were noted between patients with residual MRI findings 6 months after manipulation and those without any MRI findings.

Conclusion: Most of the iatrogenic capsular tears, bone bruises, and labral tears detected 1 week after manipulation had disappeared 6 months later. Residual MRI findings 6 months after manipulation had no significant correlation with clinical symptoms.
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http://dx.doi.org/10.1016/j.jses.2018.11.001DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6443643PMC
March 2019

Clinical outcome of conversion from external fixation to definitive internal fixation for open fracture of the lower limb.

J Orthop Sci 2019 Sep 13;24(5):888-893. Epub 2019 Feb 13.

Department of Orthopaedic Surgery, School of Medicine, Jichi Medical University, Shimotsuke, Japan.

Introduction: Few reports have examined the outcomes and complications of temporary bridging external fixation (EF) in open fracture of the lower limb followed by conversion to open reduction internal fixation (ORIF). The purpose of this study was to evaluate healing rates and complications in patients treated with conversion from external fixation to definitive internal fixation for open fracture of the lower limb.

Method: Patients who underwent temporary bridging EF and subsequent conversion to internal fixation (IF) for open fracture of the lower limb, with follow-up period ≥12 months were included in this study. Demographic data, Gustilo-Anderson classification, fracture type, duration to definitive surgery, surgical procedure, perioperative complications, and additional procedures for cases with complications were obtained.

Results: In total, 58 patients (43 males, 15 females), 63 fractures were included in this study. Four fractures (6.3%) were Gustilo grade I, 11 fractures (17.5%) were grade II, 34 fractures (54.0%) were grade IIIa, 12 fractures (19.0%) were grade IIIb, and two fractures (3.2%) were grade IIIc. Mean duration of the application of EF was 12.4 days (range, 3-45 days) until conversion to definitive IF. Rates of deep infection and nonunion were both 9.5%, with two cases showing concomitant infection and nonunion. Rates of infection were 8.8% (3/34) in grade IIIa and 25% (3/12) in grade IIIb. Rates of nonunion were 9.1% (1/11) in grade II, 2.9% (1/34) in grade IIIa and 33% (4/12) in grade IIIb.

Conclusion: Temporary EF for open fracture of the lower limb followed by conversion to IF, as early as soft tissue and general condition permit, may be a safe and effective procedure for patients with lower-limb open fracture of Gustilo grade IIIa or less.

Level Of Evidence: Level IV, Case series.
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http://dx.doi.org/10.1016/j.jos.2019.01.009DOI Listing
September 2019

Non-occlusive intestinal ischemia in the ascending colon and rectum: a pediatric case occurring during encephalitis treatment.

Surg Case Rep 2019 Feb 15;5(1):23. Epub 2019 Feb 15.

Department of Pediatrics, Yamanashi Prefectural Central Hospital, Kofu, Japan.

Background: Non-occlusive mesenteric ischemia (NOMI) is a rare and severe pathological condition that can cause intestinal necrosis without mechanical obstruction of the mesenteric artery. NOMI often develops during the treatment of severe disease in elderly patients and mostly occurs in the intestine supplied by the superior mesenteric artery (SMA). We experienced a 12-year-old patient with NOMI that was segmentally localized in the ascending colon and rectum during encephalitis treatment.

Case Presentation: A 12-year-old boy was hospitalized with limbic encephalitis. On day 41 after admission, he abruptly developed hypotension following diarrhea and fever, and presented abdominal distension. A computed tomography scan revealed pneumatosis intestinalis localized in the ascending colon and rectum coexisting with portal venous gas. The presence of peritoneal signs required an emergency laparotomy. Intraoperatively, skip ischemic lesions were found in the ascending colon and the rectum without bowel perforation. SMA and superior rectal arterial pulsation were present, and the patient was diagnosed with NOMI. The remaining colon, from the transverse to the sigmoid colon, appeared intact. We performed a distal ileostomy without bowel resection. Postoperative colonoscopies were carried out and revealed rectal and ascending colon stenosis with ulceration but demonstrated the patency of the two lesions. We confirmed the improvement of the transient bowel strictures; therefore, the ileal stoma was closed 14 months after the previous laparotomy.

Conclusion: NOMI can be present in childhood during encephalitis treatment and can be segmentally localized in the ascending colon and the rectum. Although NOMI is most often seen in elderly patients, we should also consider the possibility of NOMI when pediatric patients with severe illness manifest abdominal symptoms.
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http://dx.doi.org/10.1186/s40792-019-0592-yDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6377685PMC
February 2019

Clinical significance of serum and mesangial galactose-deficient IgA1 in patients with IgA nephropathy.

PLoS One 2018 2;13(11):e0206865. Epub 2018 Nov 2.

Division of Nephrology, Department of Medicine, Showa University School of Medicine, Tokyo, Japan.

Introduction: Galactose-deficient IgA1 (Gd-IgA1) is a critical pathogenic factor for IgA nephropathy (IgAN), but its value as a disease-specific biomarker remains controversial. We aimed to clarify the clinical significance of Gd-IgA1 in patients with IgAN.

Methods: We retrospectively reviewed 111 patients who were diagnosed with IgAN based on the findings of renal biopsies (RB) at Showa University Hospital since 2007. Serum Gd-IgA1 (s-Gd-IgA1) at the time of RB was compared among 111 IgAN patients, 18 Henoch-Schönlein purpura nephritis (HSPN) patients, 29 lupus nephritis (LN) patients, 28 ANCA-associated vasculitis (AAV) patients, and 13 minimal change disease (MCD) patients using ELISA with an anti-human Gd-IgA1-specific monoclonal antibody (KM55). We also immunohistochemically stained paraffin-embedded sections for mesangial Gd-IgA1 (m-Gd-IgA1) deposition using KM55.

Results: Although levels of s-Gd-IgA1 were comparable among IgAN and HSPN, s-Gd-IgA1 levels were significantly elevated in patients with IgAN compared with LN, AAV and MCD (IgAN vs. HSPN, LN, AAV, and MCD: 16.2 ± 9.1 vs. 14.2 ± 10.8, p = 0.263; 12.7 ± 9.4, p = 0.008; 13.1 ± 7.3, p = 0.059; and 8.2 ± 4.8 μg/mL, p<0.001, respectively). Mesangial-Gd-IgA1 deposition was specifically detected in IgAN or HSPN. The increase in s-Gd-IgA1 significantly correlated with m-Gd-IgA1 positivity in patients with IgAN, and s-Gd-IgA1 elevation and m-Gd-IgA1 deposition were evident in patients with histopathologically advanced IgAN. Moreover, s-Gd-IgA1 levels were significantly higher in IgAN patients with glomerular sclerosis and tubulo-interstitial lesions. Mesangial-Gd-IgA1 intensity negatively correlated with eGFR in IgAN. Multivariate analysis selected s-Gd-IgA1 elevation as a significant risk factor for a 30%-reduction in eGFR in IgAN (HR, 1.37; 95% CI, 1.02-1.89; p = 0.038).

Conclusions: Although IgAN and HSPN remain difficult to differentiate, s-Gd-IgA1 elevation and m-Gd-IgA1 deposition are reliable diagnostic factors that reflect IgAN severity. Serum-Gd-IgA1 could serve as a predictor of renal outcomes in IgAN. Thus, Gd-IgA1 could be significant biomarker for patients with IgAN.
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http://journals.plos.org/plosone/article?id=10.1371/journal.pone.0206865PLOS
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6214568PMC
April 2019

Blood flow evaluation by dynamic magnetic resonance imaging of symptomatic rotator cuff tears and frozen shoulders.

J Shoulder Elbow Surg 2018 Dec 13;27(12):e372-e379. Epub 2018 Aug 13.

Department of Orthopaedic Surgery, Jichi Medical University, Tochigi, Japan.

Background: This study compared imaging findings of blood flow changes between symptomatic rotator cuff tear (RCT) and frozen shoulder (FS) by using 3-dimensional dynamic magnetic resonance imaging (MRI) to determine the clinical characteristics of symptomatic RCT.

Methods: The 2 study groups comprised 31 symptomatic RCT patients who underwent arthroscopic rotator cuff repair and 30 patients with FS. We denoted abnormal blood flow detected around the glenohumeral joint as the burning sign (BS). We evaluated the characteristics of dynamic MRI and compared them between BS-positive and BS-negative patients in the RCT group.

Results: All members of the FS group showed the BS. Conversely, the incidence of the BS in RCT patients was 53% (16 of 31). The BS in RCT and FS patients was observed in the rotator interval in 16 shoulders, in the axillary pouch in 3 shoulders (P < .01), and in the intertubercular groove in 10 RCT and 12 FS patients. In the RCT group, 16 patients with BS had a statistically significantly higher Numeric Rating Score at rest (P = .0005) and in motion (P = .04) than the 15 patients without BS and exhibited a higher rate of small and medium tears and a higher rate of shoulder contracture.

Conclusion: Dynamic MRI of symptomatic RCT (53.3%) highlighted abnormal vascularization around the glenohumeral joint, which may be associated with pain and contracture in RCT as in FS.
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http://dx.doi.org/10.1016/j.jse.2018.05.042DOI Listing
December 2018

Giant umbilical cord and hypoglycemia in an infant with Proteus syndrome.

Am J Med Genet A 2018 05;176(5):1222-1224

Department of Pediatrics, University of Yamanashi, Yamanashi, Japan.

Proteus syndrome (PS) is characterized by the progressive, segmental, or patchy overgrowth of the skin, and other tissues. This is the first case report of recurrent severe insulin-independent hypoglycemia in an infant with PS. Somatic p.E17K of AKT1 mutation was confirmed. The patient also had a giant umbilical cord, which has not yet been reported in PS.
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http://dx.doi.org/10.1002/ajmg.a.38674DOI Listing
May 2018

Erlotinib attenuates the progression of chronic kidney disease in rats with remnant kidney.

Nephrol Dial Transplant 2018 04;33(4):598-606

Division of Nephrology, Department of Medicine, Showa University School of Medicine, Tokyo, Japan.

Background: Increasing evidence indicates that epidermal growth factor receptor (EGFR) has a pathogenic role in renal fibrosis. Currently no effective treatment can completely halt the progression of chronic kidney disease (CKD). This study was undertaken to investigate the renoprotective effects of erlotinib, a tyrosine kinase inhibitor that can block EGFR activity in the progression of CKD and the mechanisms involved.

Methods: Sprague Dawley rats with 5/6 nephrectomy were administered either erlotinib or vehicle from 2 weeks after surgery and for a period of 8 weeks. Blood pressure, proteinuria and serum creatinine were measured periodically. Renal morphological investigations were performed at sacrifice. In vitro, we used normal human mesangial cells (NHMCs) and human proximal tubular cells to investigate the inhibitory effects of erlotinib on renal fibrosis-associated signaling pathways by western blotting.

Results: Erlotinib treatment significantly blunted the progression of CKD as evidenced by reduced levels of serum creatinine, proteinuria and renal cortical profibrogenic genes and scores of glomerulosclerosis and tubulointerstitial damage. Tubulointerstitial macrophage infiltration and multiple pro-inflammatory cytokine gene expression levels were also attenuated by erlotinib treatment. In vitro, heparin-binding epidermal growth factor-like growth factor-induced Akt and extracellular-regulated kinase (ERK) 1/2 activation in normal human mesangial cells and human proximal tubular cells was inhibited by pretreatment with erlotinib.

Conclusions: EGFR blocking by erlotinib protected against renal fibrosis in 5/6 nephrectomized rats via inhibition of Akt and ERK 1/2 signaling pathways, which are associated with renal fibrosis. Erlotinib also has anti-inflammatory properties, which may contribute to its renoprotective effects. Erlotinib represents a potential novel therapeutic strategy for the treatment of CKD.
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http://dx.doi.org/10.1093/ndt/gfx264DOI Listing
April 2018

Shprintzen-Goldberg syndrome associated with first cervical vertebra defects.

Pediatr Int 2017 Oct 31;59(10):1098-1100. Epub 2017 Aug 31.

Department of Pediatrics, University of Yamanashi, Chuo, Yamanashi, Japan.

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http://dx.doi.org/10.1111/ped.13354DOI Listing
October 2017

Fetal goitrous hypothyroidism treated by intra-amniotic levothyroxine administration: case report and review of the literature.

J Pediatr Endocrinol Metab 2017 Aug;30(9):1001-1005

.

Background: Fetal goitrous hypothyroidism is mainly caused by maternal treatment of Graves' disease. Fetal goiter sometimes compresses the trachea and esophagus and may cause polyhydramnios, preterm labor, complications of labor and delivery, and neonatal respiratory disorder.

Case Presentation: We report a case of fetal goitrous hypothyroidism in which the mother had Graves' disease, which was treated with propylthiouracil. Intra-amniotic levothyroxine (L-T4) administration was performed, and the fetal goiter decreased in size. A female infant was delivered without goiter and complications. Thyroid function was within the normal range.

Conclusions: Previous reports on fetal goitrous hypothyroidism that was treated with intra-amniotic L-T4 showed that patients who had intra-amniotic L-T4 administration were likely to have a good outcome compared with patients who did not have L-T4. Thyroid function of the mother and fetus should be carefully monitored and treated appropriately.
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http://dx.doi.org/10.1515/jpem-2017-0094DOI Listing
August 2017

Repair of Atrioventricular Septal Defect and Transposition in Left Isomerism.

Ann Thorac Surg 2017 Apr;103(4):e353-e355

Department of Cardiovascular Surgery, Chiba Children's Hospital, Chiba, Japan.

We report the successful primary anatomic correction of an atrioventricular septal defect with transposition of the great arteries and pulmonary stenosis in a 4-year-old girl with left isomerism by common atrioventricular valve division, Mustard-type atrial septation, and a Rastelli operation with ventricular septal defect enlargement. To the best of our knowledge, this is the first report of total correction for this combination of cardiac anomalies.
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http://dx.doi.org/10.1016/j.athoracsur.2016.09.080DOI Listing
April 2017

Evaluation of the association between locomotive syndrome and depressive states: a cross-sectional study.

Nagoya J Med Sci 2017 02;79(1):43-46

Department of Orthopaedic Surgery, Jichi Medical University, Shimotsuke, Japan.

The Japanese Orthopaedic Association has proposed the term "locomotive syndrome" to designate a condition that places a person at high risk for long-term care. However, in daily clinical practice, even when a diagnosis of locomotive syndrome is made, exercise therapy often cannot be successfully performed in some patients owing to their lack of motivation. We speculated that locomotive syndrome and depressive states co-exist in elderly people. The purpose of this study was to determine the presence or absence of depressive states in older patients aged ≥ 65 years who were diagnosed with locomotive syndrome. A questionnaire survey, the 25-Question Geriatric Locomotive Function Scale and Self-Rating Questionnaire for Depression was conducted. The items of the interview survey were sex, age, and history of treatment for hypertension or diabetes mellitus. For somatometry, height and body weight were measured. Patients diagnosed with locomotive syndrome (LS group) were compared with those without locomotive syndrome (non-LS group). The LS group included 99 patients, mean age was 79.4 years old, while the non-LS group included 101 patients, mean age was 76.3 years old. The number of patients with depressive states and number of females were significantly higher in the LS group. In addition, the LS group was significantly older and shorter. Multivariate analysis revealed depressive states and age to be independent factors. Therapy for patients with LS should include evaluation and, if necessary, treatment for concomitant depression.
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http://dx.doi.org/10.18999/nagjms.79.1.43DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5346619PMC
February 2017

Short-term clinical results of frozen shoulder treated with shoulder manipulation under ultrasound-guided cervical nerve root block at outpatient setting: A case series.

J Orthop Sci 2017 Mar 25;22(2):275-280. Epub 2016 Nov 25.

Department of Orthopaedic Surgery, Jichi Medical University, Tochigi, Japan.

Background: We evaluated the short-term clinical outcomes of frozen shoulder treatment via shoulder manipulation under ultrasound-guided cervical nerve root block (MUC).

Methods: This study included frozen shoulder patients who were unresponsive to conservative therapy for at least 6 months and were then treated with MUC. Patients with a rotator cuff tear, calcifying tendinitis, osteoarthritis, or any other shoulder disorder were excluded following X-ray, ultrasound, and magnetic resonance imaging evaluation. Although 25 patients were initially included, three patients were not followed-up for at least 1 year. We investigated a final total of 22 patients; the average age was 58 years and 59% were female. We measured shoulder pain, shoulder range of motion, and American Shoulder and Elbow Surgeons shoulder scores immediately prior to MUC, 1 week after MUC, and 1 year after MUC. A Short-Form 36-Item Health Survey was administered before MUC and 1 year after MUC. We used the Friedman and Wilcoxon signed-rank tests to identify statistical differences. Significance was defined as p < 0.05.

Results: MUC significantly improved shoulder pain during motion, range of motion, and American Shoulder and Elbow Surgeons scores 1 week after MUC. This improvement persisted at the 1-year follow-up. Seven of the eight Short-Form 36-Item Health Survey measures were significantly improved 1 year after MUC. One patient (4.5%) developed Horner's syndrome, although symptoms resolved within several hours without treatment.

Conclusion: MUC for frozen shoulder was safe and resulted in a significant improvement in shoulder pain and range of motion 1 week after the procedure. This improvement persisted at the 1-year follow-up.
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http://dx.doi.org/10.1016/j.jos.2016.11.007DOI Listing
March 2017

Locomotive syndrome is associated with large blood pressure variability in elderly hypertensives: the Japan Ambulatory Blood Pressure Prospective (JAMP) substudy.

J Clin Hypertens (Greenwich) 2017 Apr 15;19(4):388-394. Epub 2016 Nov 15.

Division of Cardiovascular Medicine, Department of Medicine, Jichi Medical University, Shimotsuke, Tochigi, Japan.

Chronic pain, represented by locomotive syndrome (LS), and psychosocial factors are possible factors of blood pressure (BP) variability (BPV). The authors tested the hypothesis that there are links among LS, depression, and BPV. In 85 Japanese elderly hypertensive patients with normal daily activities, the authors performed ambulatory BP monitoring, determined the LS scale (LSS), and administered the Self-Rating Questionnaire for Depression (SRQD). The LSS score but not the SRQD score was associated with the standard deviation (SD) and coefficient of variation (CV) of daytime systolic BP (SBP) and SD of nighttime SBP (all P<.05). Higher LSS score (in quartiles) was associated with a higher SD of daytime SBP (P=.041), even after adjusting for covariates. Regarding the components of the LSS score, movement-related difficulty and usual care difficulty were associated with the SD and CV of daytime SBP. In elderly hypertensive patients, the LSS score was associated with exaggerated systolic BPV. The LS state could be an important determinant of systolic BPV.
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http://dx.doi.org/10.1111/jch.12946DOI Listing
April 2017
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