VOLUME 85 NUMBER 1 February 2023

Current Issue

ISSN 2186-3326
(Online ISSN)
ISSN 0027-7622
(Print ISSN--v.72no.3/4)

Nagoya Journal of Medical Science: Celebrating 100 years

From the Editor-in-Chief

Toward the next century of the Nagoya Journal of Medical Science: message from the Editor-in-chief

Shinya Toyokuni
pg(s) 1–4

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From the Dean of Nagoya University Graduate School of Medicine

The 100th anniversary of the Nagoya Journal of Medical Science: a message from the Dean

Hiroshi Kimura
pg(s) 5–6

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Comments to the Highly Cited Articles

The dawn of a new era in surgery for lymphedema

Kazunobu Hashikawa and Yuzuru Kamei
pg(s) 7–9

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Comment to our article; Effects of varicocelectomy on testis volume and semen parameters in adolescents: a randomized prospective study

Hatsuki Hibi
pg(s) 10–12

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Commentary on “Mechanisms of asbestos-induced carcinogenesis” published in 2009

Shinya Toyokuni
pg(s) 13–15

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Improving the maternal mortality rate in Lao PDR: 10 years after the publication of the Yang Ye paper in the Nagoya Journal of Medical Science

Junichi Sakamoto
pg(s) 16–20

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The fruits of CD40 research in basic and clinical medicine will soon be harvested

Tsutomu Kawabe
pg(s) 21–22

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Recent progress in adipocytokine research

Noriyuki Ouchi, Koji Ohashi and Toyoaki Murohara
pg(s) 23–26

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A never-ending FLT3 story

Hitoshi Kiyoi
pg(s) 27–29

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Neuroinflammation in neurodegenerative disease

Koji Yamanaka
pg(s) 30–32

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Immune checkpoint inhibitors for patients with pre-existing autoimmune diseases

Osamu Maeda
pg(s) 33–34

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A systematic review regarding clinical characteristics, complications, and outcomes of surgical and non-surgical patients with fragility fracture of the pelvis

Takaomi Kobayashi, Takayuki Akiyama, Tadatsugu Morimoto, Kensuke Hotta and Masaaki Mawatari
pg(s) 35–49

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We conducted this systematic review to clarify the clinical characteristics, complications, and outcomes of surgical and non-surgical patients with fragility fracture of the pelvis (FFP). We searched PubMed, Google Scholar, Cochrane Library, Web of Science, and MEDLINE for English language articles on FFP. We calculated pooled odds ratios (ORs) or mean differences (MDs) of surgical patients in comparison to non-surgical patients for clinical characteristics (Rommens FFP classification, age, sex, dementia, osteoporosis, diabetes mellitus, pulmonary disease, cardiovascular disease, and malignancy), complications (pneumonia, urinary tract infection, cardiac event, thrombosis, pulmonary embolism, pressure ulcer, multiple organ failure, anemia caused by surgical bleeding, and surgical site infection), and outcomes (hospital mortality and one-year mortality). Five studies involving 1,090 patients with FFP (surgical patients, n=432; non-surgical patients, n=658) were included. FFP type III and IV (OR=8.44; 95% confidence interval [CI] 5.99 to 11.88; p<0.00001), a younger age (MD=–3.29; 95% CI –3.83 to –2.75; p<0.00001), the absence of dementia (OR=0.36; 95% CI 0.23 to 0.57; p<0.0001), and the presence of osteoporosis (OR=1.74; 95% CI 1.29 to 2.35; p=0.0003) were significantly associated with the surgical patients. Urinary tract infection (OR=2.06; 95% CI 1.37 to 3.10; p=0.0005), anemia caused by surgical bleeding (OR=4.55; 95% CI 1.95 to 10.62; p=0.0005), and surgical site infection (OR=16.74; 95% CI 3.05 to 91.87; p=0.001) were significantly associated with the surgical patients. There were no significant differences in the outcomes between the surgical and non-surgical patients. Our findings may help to further understand the treatment strategy for FFP and improve clinical outcomes.


Differences in gaze behaviors between trainees and experts during endovascular therapy for cerebral aneurysms: a preliminary study using a cerebral aneurysm model

Takafumi Otsuka, Takashi Izumi, Masaya Yamamoto, Yusuke Seshita, Takeshi Kohama, Masahiro Nishihori, Tetsuya Tsukada and Ryuta Saito
pg(s) 50–58

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In the neuroendovascular field, the training of operators has become an important issue. Recently, eye-tracking technology has been introduced into various fields of medical education. This study aimed to apply eye-tracking technology to the training of neuroendovascular therapy. Six neurosurgeons, including three neuroendovascular specialists and three trainees, at our institution and related facilities participated in the study. Eye movement was recorded by the eye-tracking device during the microcatheter navigation and coil placement into the silastic aneurysm model under biplane X-ray fluoroscopy. Eye-tracking analysis during neuroendovascular therapy was feasible in all six subjects. In microcatheter navigation, specialists tended to more frequently switch their attention between frontal and lateral images than trainees. In coil embolization, the overall gaze frequency tended to increase, and the average fixation duration tended to decrease as the number of experienced cases increased. Inexperienced operators tend to fix their gaze when they are operators than when they are assistants. More experienced operators tended to look at the microcatheter longer in the coil insertion task. The eye-tracking analysis may be useful for operator training in neuroendovascular therapy. Experts may have moved their eyes more frequently than trainees to gaze at the right place. In the future, it will be necessary to collect gaze data for more operators in various tasks.
Factors associated with acceptance of a digital contact tracing application for COVID-19 in the Japanese working-age population

Tomohiro Ishimaru, Koki Ibayashi, Masako Nagata, Seiichiro Tateishi, Ayako Hino, Mayumi Tsuji, Hajime Ando, Keiji Muramatsu and Yoshihisa Fujino, for the CORoNaWork Project
pg(s) 59–69

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The working-age population is at the epicenter of coronavirus disease 2019 (COVID-19) infections. Therefore, it is important to increase the acceptance of digital contact tracing apps in this population. Contact-Confirming Application (COCOA) is the only digital contact tracing app in Japan. This study aimed to determine factors associated with acceptance of the COCOA for COVID-19 in the Japanese working-age population. A cross-sectional study was performed for 27,036 full-time workers registered with an internet survey company during December 2020 in Japan. Factors associated with COCOA adoption were evaluated by multivariate logistic regression analysis. The rate of downloading the COCOA was 25.1%. The COCOA was more likely to be accepted by people with married status, university graduation or above, higher income, and occupations involving desk work. Fear of COVID-19 transmission, wearing a mask, using hand disinfection, willingness to be vaccinated against COVID-19, and presence of an acquaintance infected with COVID-19 were also associated with a greater likelihood of adopting the app. The rate of downloading the COCOA in Japan was not very high. The present findings have important implications for widespread adoption of digital contact tracing apps in working-age populations in Japan and elsewhere.
Comparison study of the Le Fort I osteotomy using 2- and 4-plate fixation

Masahito Fujio, Akira Sayo, Kota Ogisu, Qi Chang, Makoto Tsuboi and Hideharu Hibi
pg(s) 70–78

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This study was conducted to evaluate the postsurgical stability of Le Fort I osteotomy using zygomatic buttress internal fixation alone with no piriform aperture internal fixation. Patients with maxillary retrognathia and mandibular prognathism underwent the Le Fort I osteotomy with a bilateral sagittal split ramus osteotomy. In group I, fixation was accomplished using titanium plate and screws placed at the piriform aperture and the zygomatic buttress (4 plates). In group II, fixation was accomplished using titanium plate and screws placed at the zygomatic buttress (2 plates). Lateral cephalometric radiographs were taken preoperatively (T1), immediately after surgery (T2), and at 6 months to 1 year (T3) to evaluate skeletal movement. In total, 32 patients were included in this study. None of the patients had wound infection, dehiscence, bone fragment instability, and long-term malocclusion. Regarding point A and the posterior nasal spine (PNS), vertical and horizontal relapse in groups I and II did not differ significantly. In most hospitals, the maxilla was fixed using four plates (piriform aperture and zygomatic buttress); however, within the limitations of the study, the choice of the number of plates for osteosynthesis following Le Fort I osteotomy and repositioning of the maxilla can be left to the discretion of the surgeon without putting the patients at risk for increased relapse by careful intraoperative management.
Validation of the abbreviated version of the World Health Organization Quality of Life in Mongolia: a population-based cross-sectional study among adults in Ulaanbaatar

Enkhjin Bat-Erdene, Enkhnaran Tumurbaatar, Gantsetseg Tumur-Ochir, Otgonbold Jamiyandorj, Tsolmon Jadamba, Eiko Yamamoto, Nobuyuki Hamajima, Takakazu Oka and Battuvshin Lkhagvasuren
pg(s) 79–92

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There is currently no validated tool to measure the quality of life (QOL) in the Mongolian language. This study aimed to validate the Mongolian version of the World Health Organization Quality of Life – Brief (WHOQOL-BREF) questionnaire for the general population of Mongolia. The subjects were 301 adults aged 18–65 years selected randomly by a computer from 30 centers in 8 districts of Ulaanbaatar, Mongolia, in 2020. Reliability was measured using Cronbach’s α and intraclass correlation coefficients. Convergent, discriminant, and construct validities were examined using exploratory and confirmatory factor analyses for a four-domain factor structure. Among the participants, 56.1% were women, 32.9% held a bachelor’s degree or higher, 48.8% were employed, and 61.8% were married. The overall Cronbach’s α coefficient of the WHOQOL-BREF questionnaire was 0.804. Correlations between the component scores of the WHOQOL-BREF ranged from 0.581–0.822. All items showed higher item-total correlations with their corresponding domains than with other domains, except the mobility item from the physical domain. Discriminative validity was evident in physical and psychological domains. Exploratory and confirmatory factor analyses revealed a four-factorial structure consisting of 24 items that provided an acceptable fit to the data (RMSEA=0.084; CFI=0.860). In conclusion, the Mongolian version of the WHOQOL-BREF demonstrated evidence of good reliability and validity for assessing QOL in the general population of Mongolia. These findings indicate that it allows the comparison of QOL of adults in Mongolia with those in other countries.
Negative impact of behavior restriction amidst a clustered COVID-19 infection on immobility syndrome in older patients negative for COVID-19: report from a chronic care hospital in Japan

Yoshinori Maki, Akio Goda, Masatoshi Koumo, Tetsuya Yamamoto, Kouta Yokoyama, Masashi Ueno, Asuka Kishimoto, Masami Kusumoto, Mayumi Takagawa, Tamako Takahashi, Chiharu Takahashi, Keisuke Nakajima, Mika Hashimoto, Tsumugi Hosokawa, Misuzu Kobayashi, Junichi Katsura and Ken Yanagibashi
pg(s) 93–102

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Previous studies have reported on associations between immobility syndrome and the COVID-19 pandemic. However, little is known about the aggravation of this syndrome in older patients negative for COVID-19 infection amidst behavior restriction due to a clustered COVID-19 infection. Patients hospitalized one month before a clustered COVID-19 infection occurred in our hospital were recruited. Rehabilitation therapy was suspended for 25 days during behavior restriction. The ability of daily living of the patients was evaluated with the functional independence measure and Barthel index. Chronological changes in the functional independence measure and Barthel index scores were evaluated monthly, beginning one month before the clustered COVID-19 infection to one month after re-initiation of rehabilitation therapy. Patients with minimum scores in the functional independence measure (18) and Barthel index (0) prior to the clustered COVID-19 infection were excluded. Functional independence measure scores of 73 older patients and the Barthel index scores of 48 patients were analyzed. The mean total functional independence measure score amidst the behavior restriction significantly changed from 36.3 to 35.1 (p = 0.019), while statistical significance was not detected in the mean motor subtotal (from 21.6 to 20.9 with p = 0.247) or cognitive subtotal functional independence measure scores (from 14.6 to 14.2 with p = 0.478). During the behavior restriction, the mean Barthel index scores declined from 25.8 to 23.2 without statistical significance (p = 0.059). Behavior restriction due to a clustered COVID-19 infection may aggravate immobility syndrome in older patients who are negative for COVID-19.
Neck pain after cervical laminoplasty is associated with postoperative atrophy of the trapezius muscle

Sadayuki Ito, Yoshihito Sakai, Kei Ando, Hiroaki Nakashima, Masaaki Machino, Naoki Segi, Hiroyuki Tomita, Hiroyuki Koshimizu, Tetsuro Hida, Kenyu Ito, Atsushi Harada and Shiro Imagama
pg(s) 103–112

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Cervical laminoplasty is a safe and effective treatment for cervical myelopathy. However, it has a higher frequency of postoperative axial pain than other methods. A variety of causes of postoperative axial pain have been reported, but these have not been fully elucidated. This study aimed to investigate the association between postoperative axial pain and changes in the posterior neck muscles before and after surgery. The study included 93 patients with cervical myelopathy who underwent surgery at our institute between June 2010 and March 2013. The patients with greater preoperative and 1-year postoperative neck pain visual analog scale scores comprised the neck pain group. The cross-sectional area of the cervical posterior extensor muscles and the trapezius muscle were measured by magnetic resonance imaging before and 1 year after surgery at the C3/4, C4/5, and C5/6 levels to compare with neck pain. The total cross-sectional area atrophy rate (C3/C4, C4/C5, and C5/C6) of the trapezius muscle was significantly larger in patients with neck pain (12.8 ± 13.2) than in those without (6.2 ± 14.4; p<0.05). The cross-sectional area atrophy rate of the trapezius muscle at only the C5/6 level was significantly higher in patients with neck pain (16.7 ± 11.7) compared to those without (3.3 ± 14.4; p<0.001). No significant differences were found in the cross-sectional areas of the cervical posterior extensor muscles. Trapezius atrophy, especially at the lower cervical vertebrae, was associated with neck pain after cervical laminoplasty.


Reporting system on mortality statistics in Lao People’s Democratic Republic

Souphalak Inthaphatha, Viengsakhone Louangpradith, Bounfeng Phoummalaysith, Bounbouly Thanavanh, Tetsuyoshi Kariya, Eiko Yamamoto and Nobuyuki Hamajima
pg(s) 113–122

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The system to collect information on mortality statistics in Lao PDR is not well established, accurate and timely death information is therefore not available. This article reports the system and process to make the mortality statistical data of Lao PDR. The country has a paper-based resident registration system, using a death notification document, a death certificate, and a family census book. The death notification document is important as it provides the cause of death, which is issued from a health facility and the village office. In the event of a death occurring at home, the family representative needs to report to the village office verbally to obtain a death notification document. On the other hand, if the death occurred in a medical facility, a death notification document from a health facility is provided. The family representative should bring the death notification document to the district Home Affairs office to register the death and obtain a death certificate. After that, the family representative needs to bring the death certificate to the district Public Security office for an amendment in the family census book. ICD-10 is under development regarding death notification from health facilities under the Ministry of Health. However, it is unclear how death notification from village offices can adopt ICD-10 as the majority of deaths occur outside health facilities. A comprehensive and integrated mortality reporting system is necessary in order to create a holistic health policy and welfare for the country.


Prominent dermal accumulation of Russell bodies underlying pseudocarcinomatous hyperplasia with fungal infection

Tatsuhiro Noda, Norika Akashi, Maiko Shimomura, Haruka Koizumi, Miyuki Mizuta, Kosei Nakajima, Takuya Takeichi, Teruyuki Mitsuma and Masashi Akiyama
pg(s) 123–126

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Blockade of the secretion of immunoglobulins leads to their accumulation in plasma cells, resulting in condensed immunoglobulins in the rough endoplasmic reticulum of plasma cells, termed Russell bodies. They are sometimes found in lymphoplasmacellular inflammation of the intestinal mucosa and in lymphoid cell malignancies, but only very rarely in skin diseases. Here, we report an 86-year-old female who presented with a lesion with the prominent accumulation of Russell bodies underlying pseudocarcinomatous hyperplasia with fungal infection in the face. Immunohistochemical staining showed the cells containing Russell bodies to be positive for CD138 and the Russell bodies to be positive for immunoglobulin κ and λ light chains. The present case suggests that when inflammatory cell infiltration with abundant round intracellular eosinophilic materials is observed in the dermis, the dermal accumulation of Russell bodies should be considered in cases with reactive pseudocarcinomatous hyperplasia with fungal infection.
The usefulness of three-dimensional fusion imaging of spinal arteriovenous malformation by a workstation connected to angiography systems

Yoshitaka Nagashima, Takashi Izumi, Yusuke Nishimura, Masahiro Nishihori, Takahiro Oyama, Mamoru Matsuo, Hiroshi Ito, Tomoya Nishii and Ryuta Saito
pg(s) 127–133

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Digital subtraction angiography (DSA) is the most useful technique for diagnosing spinal arteriovenous malformations (AVM). In recent years, with the improvement of imaging capabilities, the usefulness of three-dimensional (3D) imaging by fusing various modalities has been recognized. The use of 3D fusion imaging with a workstation connected to an angiography system has been reported in many cases of intracranial disease, but less frequently for spinal AVM. In this article, we describe two illustrative cases of spinal AVM in which 3D fusion imaging was useful for treatment. Although 3D fusion images using the system have the disadvantage that only a maximum of two images can be fused, it provides spinal surgeons with useful information for preoperative evaluation in a small amount of time.
Successful prevention of stenosis after circumferential endoscopic resection of esophageal cancer

Hitoshi Tanaka, Naomi Kakushima, Koichi Muroi, Takashi Hirose, Tomohiko Suzuki, Takahiro Suzuki, Emiko Hida, Keiko Hirai, Hiroyuki Shibata, Nobuhito Ito, Satoshi Furune, Kazuhiro Furukawa and Mitsuhiro Fujishiro
pg(s) 127–133

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Circumferential resection of a >5-cm longitudinal mucosal defect following esophageal endoscopic submucosal dissection (ESD) is a risk factor for refractory stenosis. Circumferential ESD was performed in 3 patients with 64, 69, and 70 mm longitudinal mucosal defects. A local steroid injection was used to treat the postoperative ulcer, followed by an oral steroid. In all three cases, the ulcer healed without the need for endoscopic dilation. A combination of local injection and oral steroids effectively prevented esophageal stenosis in patients with high-risk stenosis after ESD.
Surgical revascularization for quasi-moyamoya disease associated with polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes (POEMS) syndrome: a case report and literature review

Yuki Kubota, Yoshiki Hanaoka, Jun-ichi Koyama, Yusuke Takahashi, Nagaaki Katoh, Mai Iwaya, Yu Fujii, Toshihiro Ogiwara and Tetsuyoshi Horiuchi
pg(s) 141–149

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POEMS (polyneuropathy, organomegaly, endocrinopathy, monoclonal gammopathy, and skin changes) syndrome is a rare multisystem disease characterized by plasma cell dyscrasia and overproduction of vascular endothelial growth factor, which is related to disease activity. Recent treatment strategies have improved survival of patients suffering from this disorder; however, ischemic stroke remains a poor prognostic factor. POEMS patients with ischemic stroke frequently develop cerebral large artery stenosis/ occlusion, followed by progressive stroke. Post literature review, we present an ischemic stroke case of quasi-moyamoya disease linked with this syndrome that was successfully treated with surgical revascularization. A 41-year-old woman diagnosed with POEMS syndrome developed progressive ischemic stroke due to quasi-moyamoya disease, despite decreased vascular endothelial growth factor level with lenalidomide and dexamethasone treatment. She underwent superficial temporal artery to middle cerebral artery bypass with encephalo-duro-myo-synangiosis bilaterally. The postoperative course was uneventful. Two years and five months after the stroke, neuroimaging demonstrated bypass patency, neovascularization after encephalo-duro-myo-synangiosis, and no recurrence of stroke. Our case is the first to report successful surgical revascularization for a POEMS patient. Surgical revascularization may be a useful treatment option for patients with quasi-moyamoya disease associated with POEMS syndrome, especially for those who develop refractory ischemic stroke despite reduced vascular endothelial growth factor level.
A case of Erdheim–Chester disease with the BRAF V600E mutation diagnosed via endoscopic sinus surgery

Takaharu Matsuhisa, Masaaki Teranishi, Yuta Tsuyuki, Michihiko Sone and Juichi Sato
pg(s) 150–156

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Erdheim–Chester disease is characterized by the infiltration of foamy histiocytes in tissues. Lesional tissue biopsy is recommended to confirm diagnosis and establish the BRAF mutational status. A 52-year-old man presented to our hospital with hydronephrosis. Computed tomography showed enhancement of soft shadows around the left renal pelvis transition area and the aorta. He was treated with prednisolone 0.2 mg/kg for 1 year; however, no improvement was observed. 18Fluorodeoxyglucose–positron emission tomography/computed tomography revealed increased fluorodeoxyglucose uptake in various body parts, including the maxillary sinuses, indicative of Erdheim–Chester disease. He refused further examination, and the maxillary sinus lesions were treated with antibiotics and intranasal steroids, but no improvement was observed. Two years later, he underwent biopsy with endoscopic sinus surgery of the maxillary sinus, which showed the highest increase in fluorodeoxyglucose uptake on repeat 18fluorodeoxyglucose–positron emission tomography/computed tomography. Endoscopic findings showed only nonspecific inflammatory findings, but pathological findings revealed the proliferation of cells with abundant foamy cytoplasms. Sufficient tumor volume was available to perform PCR for BRAF V600E mutation analysis, which was positive and resulted in a diagnosis of Erdheim–Chester disease with the BRAF V600E mutation. This is the first case of a patient with Erdheim–Chester disease with the BRAF V600E mutation identified in a sinus lesion. Endoscopic sinus surgery biopsy of the paranasal sinuses was considered to contribute to the histological and genetic diagnosis of Erdheim–Chester disease, particularly following the notable increase in fluorodeoxyglucose uptake.
Single-lane clipping technique for a ruptured aneurysm of A1 fenestration of the anterior cerebral artery: a case report and literature review

Yuki Kubota, Yoshiki Hanaoka, Tatsuro Aoyama, Yu Fujii, Toshihiro Ogiwara, Tatsuya Seguchiand Tetsuyoshi Horiuchi
pg(s) 157–166

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Fenestration of the A1 segment of the anterior cerebral artery is a rare vascular anomaly with a high risk of saccular aneurysm at the proximal end of the A1 fenestration. These aneurysms have a high risk of rupture. However, conventional surgical clipping can be technically challenging due to the anatomical characteristics. We report a case of A1 fenestration with a ruptured aneurysm wherein we successfully achieved complete obliteration of the aneurysm with a new “single-lane” clipping technique. A 64-year-old woman presented with a ruptured saccular A1 aneurysm at the proximal end of an A1 fenestration, resulting in subarachnoid hemorrhage. Microsurgical clipping was attempted; however, adequate exposure of the aneurysm could not be achieved. The recurrent artery of Heubner originated near the distal end of the lateral limb of the A1 fenestration. The lateral limb of the A1 fenestration had no perforating arteries, according to surgical examination. Thus, the aneurysm neck and lateral limb were concurrently obliterated using a nonfenestrated clip, preserving the medial limb of the A1 fenestration. The antegrade flow of the recurrent artery of Heubner was detected using the retrograde flow of the distal part of the lateral limb of the A1 fenestration during intraoperative indocyanine green video angiography. The postoperative course was uneventful without any evidence of ischemic stroke. For A1 aneurysms arising from the proximal end of the A1 fenestration, this technique may be a useful treatment option. Before using this technique, careful surgical exploration should be performed to assess the A1 perforating arteries.
Successful perioperative management of simultaneous transcatheter aortic valve implantation and hip fracture surgery: a case report

Kochi Yamane, Tadashi Aoyama, Yohei Aoki, Hironori Momohara and Mikio Nonogaki
pg(s) 167–170

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A 90-year-old woman with severe aortic stenosis experienced hospital readmission for chronic heart failure exacerbations many times and was admitted to our hospital for undergoing transcatheter aortic valve implantation. Thereafter, she fell in the ward and fractured her femoral trochanter, requiring early hip fracture surgery. We proposed that we should perform simultaneous transcatheter aortic valve implantation and hip fracture surgery to cardiologist and orthopedist from anesthetic and perioperative management perspective. We considered that it was difficult to maintain cardiovascular function without cardiac intervention during hip fracture surgery and starting rehabilitation as early as possible was important. General anesthesia was induced without any complications, and the tracheal tube was removed after the successive surgeries. On postoperative day 1, bedside rehabilitation was started, and on postoperative day 3, she was transferred from the intensive care unit to the general ward. On postoperative day 32, she was transferred to another hospital. Anesthesiologist should play an important role for decision making in not only intraoperative but perioperative management for critical case, we should communicate with other departments. The successful perioperative management of simultaneous transcatheter aortic valve implantation and hip fracture surgery enabled to start rehabilitation early and prevented further patient hospitalization.
A case of eosinophilic polyangiitis with granulomatosis that evolved to cardiac arrest due to advanced atrioventricular block

Yuichiro Sakurai, Chiyako Oshikata, Takaharu Katayama, Shunsuke Takagi, Yasushi Kaneko, Kikuo Yo, Takeshi Kaneko, Hiroyuki Kubota, Takashi Matsubara and Naomi Tsurikisawa
pg(s) 171–178

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Cardiac manifestations are the major cause of mortality in patients with eosinophilic granulomatosis with polyangiitis (EGPA). Among these manifestations in EGPA patients, in the literature, there are fewer reports describing bradycardia in EGPA patients than those describing tachycardia. A 50-year-old woman with a history of childhood-onset asthma. At age 28, she was diagnosed with eosinophilic gastroenteritis without the diagnosis of EGPA and was started on a systemic steroid and had maintenance daily dose of 2.5 mg after gradually tapered. She had experiencing dizziness and palpitations 2 weeks after discontinuation of the steroid treatment. At emergency visit, electrocardiography revealed an advanced atrioventricular block of 3:1 or less. Forty-eight minutes after the start of electrocardiography, only a P wave was observed and cardiac arrest occurred for 9 s and temporary emergency pacing was performed immediately. She was diagnosed as EGPA presenting leukocyte count, 16,500/µL, 42.8% of which were eosinophils and sinusitis in computed-tomography. She could be survival by treatment of steroid, following the patient to withdraw from an external pacemaker. She received prednisolone of 60 mg, intravenous cyclophosphamide and intravenous immunoglobulin. She had relapsed presenting peripheral eosinophilia, abdominal and numbness in the toes of the left leg pain, but not arrythmia after tapered of prednisolone. Following additional steroid pulse, she had an increase of prednisolone and continued by intravenous cyclophosphamide, intravenous immunoglobulin and started mepolizumab. We presented a severe case of EGPA presenting an advanced atrioventricular block into cardiac arrest.
Transorbital penetrating head injury by a wooden chopstick in the cavernous sinus: a case report and literature review

Daisuke Yamazaki, Naoki Ogihara, Takehiro Yako, Yu Fujii, Yoshiki Hanaoka, Toru Kurokawa and Tetsuyoshi Horiuchi
pg(s) 179–184

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Penetrating head injury is a relatively rare condition associated with high morbidity and mortality. Although the immediate treatment of penetrating head injury is needed, surgical strategies are varied based on the trajectory of the penetrating objects in the cranium. We present a case of 24-year-old man who sustained a transorbital penetrating injury caused by a wooden chopstick. Neuroimages revealed a linear lesion extending from the left intraorbital segment to the cavernous sinus passing through the superior orbital fissure. The foreign body was successfully removed via the transcranial approach without complications. A careful management based on the perioperative images and correct diagnosis is necessary to avoid unfavorable complications. Four cases of transorbital penetrating injuries have been previously reported, in which the foreign body penetrated through the superior orbital fissure and lodged in the cavernous sinus. The frontotemporal craniotomy with extradural approach can be a useful option to remove foreign bodies around the cavernous sinus regions.
Failure of internal fixation for ankle joint Charcot neuroarthropathy with beta(2)-microglobulin amyloidosis: a case report

Yang Zheng, Jiu-Dan Zhang, Jie-Feng Huang and Ya-Hong Zhu
pg(s) 185–194

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Charcot neuroarthropathy (CN) is a serious diabetic complication with a poor prognosis and a high rate of misdiagnosis. Furthermore, beta(2)-microglobulin amyloidosis (Abeta2M) makes the diagnosis and therapy more difficult and complex. This case report highlights the pathophysiology, clinical evaluation, treatment, and prevention of the major diabetic complications associated with CN and Abeta2M that cause poor quality of life, limit the patient’s ability to walk independently, and are directly or indirectly linked with a high risk for lower limb amputation. Ankle CN was discovered in a 36-year-old single female with a history of type 1 diabetes mellitus and diabetic nephropathy. We performed early internal fixation. However, because she lived alone and needed hemodialysis three times a week, wearing a brace and non-weight-bearing were extremely inconvenient. Furthermore, she did not experience any pain and only some edema; thus, she proceeded to bear weight ahead of schedule without authorization. Due to the premature weight-bearing and poor compliance, the patient suffered severe bone resorption and infection and eventually had to undergo amputation. Abeta2M was suggested by bone pathological sections. We present a case of failed internal fixation of ankle CN with Abeta2M, emphasizing the importance of social factors and postoperative management.
Myelodysplastic syndrome with trisomy 8 presenting periodic fever and multiple MEFV gene variants outside exon 10: a case report

Noriyuki Takahashi, Ryo Hanajiri, Masashi Suzuki, Chise Anan, Atsushi Inagaki, Dai Kishida, Shohei Ozawa, Sho Kohri, Nobuhide Kamiya, Motoki Sato and Juichi Sato
pg(s) 195–203

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Myelodysplastic syndrome is associated with the development of autoinflammatory conditions, such as recurrent fever, polymyalgia, arthralgia, and erythema. Trisomy 8 is a common chromosomal abnormality in patients with myelodysplastic syndrome. Myelodysplastic syndrome with trisomy 8 involves autoinflammatory conditions, especially Behçet’s disease-like symptoms with intestinal mucosal damage. MEFV variants, particularly those in exon 10, are pathogenic in familial Mediterranean fever, the most common autoinflammatory disease, presenting typical symptoms such as periodic fever and pleuritis/pericarditis/ peritonitis. MEFV variants outside exon 10 are common in Japanese patients with familial Mediterranean fever and are associated with atypical symptoms, including myalgia and erythema. MEFV variants in myelodysplastic syndrome with trisomy 8 have rarely been investigated, although myelodysplastic syndrome with trisomy 8 might develop autoinflammatory conditions similar to those in familial Mediterranean fever. We encountered a 67-year-old man who had myelodysplastic syndrome with trisomy 8 and multiple MEFV variants outside exon 10. He presented with periodic fever, as well as chest/abdominal pain, myalgia, and erythema, although the symptoms did not fulfill the diagnostic criteria of familial Mediterranean fever. We discussed the possibility that these symptoms are modified by MEFV variants outside exon 10 in myelodysplastic syndrome with trisomy 8.
Multiple tendon transfer for a case of radial nerve palsy in hereditary neuropathy with liability to pressure palsy

Federico Palumbo, Michiro Yamamoto and Hitoshi Hirata
pg(s) 204–210

<Abstract> - < PDF >

Hereditary neuropathy with liability to pressure palsy (HNPP) is a rare autosomal dominant disease characterized by focal, recurrent, demyelinating peripheral neuropathies. It is caused by deletions of the gene encoding for peripheral myelin protein 22 (PMP22) on chromosome 17. While it may range widely, the most common clinical presentation is an acute, focal mononeuropathy with numbness or muscle weakness after trauma or compression. Diagnostic tools include electrophysiological studies, genetic tests and nerve biopsies. There is no standard surgical or pharmacological treatment. The course of the disease is usually benign, with spontaneous improvement after most episodes of peripheral nerve palsy. HNPP is best managed by early detection, preventative measures, and subsequent treatment of symptoms. According to the medical literature, operative treatment was undertaken in few cases and limited to decompression of the nerve at the classic entrapment sites of the carpal or cubital tunnels. We present a case of multiple tendon transfer (pronator teres to extensor carpi radialis brevis and flexor carpi radialis to extensor digitorum communis) with a two-year follow-up in a 24-year-old woman with HNPP who was affected by irreversible radial nerve palsy, and conclude with a review of the medical literature related to the disease.