Authorship：Lead author   Language：Japanese  

Language：English   Publisher：鹿児島大学医学会  

ロゼット形成性グリア神経細胞腫瘍は稀な腫瘍で,緩徐に発育し(WHO grade 1),後頭蓋窩に好発する.我々は,亜全摘出後11年以上再発なく経過する,視床を起源とした稀なロゼット形成性グリア神経細胞腫瘍を経験したため報告する.患者は20歳代前半の女性で,6ヵ月前からの記憶障害,頭痛,霧視を自覚し当科を受診した.Magnetic resonance imaging(MRI)では閉塞性水頭症,18mmの右視床から生じた非嚢胞性第三脳室腫瘍を認めた.病変は,T1強調像で等信号,T2強調像では高信号を呈し,ガドリニウムによる造影効果は認めなかった.右側からのtrans-ventricular subchoroidal approachにより,視床後壁にごく少量の遺残を残して,柔らかい腫瘍を亜全摘出した.組織学的には,周囲に好酸球性神経細胞性コアや小血管コアを伴うロゼットを含めたalveolar component,および毛様細胞性星細胞腫に類似した充実性成分で構成されていた.ロゼットを構成する細胞は,Olig-2,MAP,synaptophysinが陽性で,Ki-67 indexは1%であった.術後,症状は消失した.術後11年3ヵ月後のMRIでは再発なく経過していた.本症例は,ロゼット形成性グリア神経細胞腫瘍の発生部位としては稀な視床から発生し,亜全摘出後11年経過しても再発なく経過している点において,極めて稀な症例である.(著者抄録)

Authorship：Corresponding author   Language：English  

DOI： 10.1007/s10143-023-02223-w

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PubMed

Authorship：Lead author   Language：Japanese   Publishing type：Research paper (scientific journal)  

DOI： https://doi.org/10.11477/mf.1436204834

Language：Japanese   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204834

PubMed

Authorship：Lead author   Language：Japanese   Publishing type：Research paper (scientific journal)  

DOI： https://doi.org/10.11477/mf.1436204790

Language：Japanese   Publisher：株式会社医学書院  

DOI： 10.11477/mf.1436204790

PubMed

Language：English  

DOI： 10.1055/s-0043-1763526

PubMed

Language：Japanese   Publisher：Pituitary  

Purpose: Delayed postoperative hyponatremia (DPH) is a unique complication of transsphenoidal surgery (TSS) in pituitary tumors. Growth hormone (GH) enhances renal sodium reabsorption; however, the association between postoperative GH reduction and DPH in acromegaly is unclear. This study was performed to clarify the incidence of and the predictive factors for DPH in patients with acromegaly who underwent TSS. Methods: Ninety-four patients with active acromegaly were examined retrospectively. During the postoperative course, patients with serum sodium levels ≤ 134 mEq/L were classified into the DPH group. We compared basic clinical characteristics, tumor characteristics, and preoperative and postoperative examination findings between the DPH and non-DPH groups. Results: DPH occurred in 39 patients (41.5%), and the lowest serum sodium levels were generally observed during postoperative days (PODs) 7–9. They needed a 3-day longer hospital stay than those without DPH. The DPH group had lower preoperative body weight and body mass index. In addition, a transient increase in body weight during PODs 5–7 occurred with a transient decrease in urinary volume in the DPH group. Preoperative and postoperative GH and insulin-like growth factor-1 levels did not differ between the two groups. Conclusion: The findings suggested that lower preoperative weight and a postoperative transient gain in body weight are associated with an increased risk of DPH in acromegaly patients undergoing transsphenoidal surgery.

DOI： 10.1007/s11102-022-01288-y

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PubMed

Language：Japanese   Publisher：Neuro-Oncology Advances  

Background: Glioblastoma (GBM) is a malignant brain tumor, with radiological and genetic heterogeneity. We examined the association between radiological characteristics and driver gene alterations. Methods: We analyzed the driver genes of 124 patients with IDH wild-type GBM with contrast enhancement using magnetic resonance imaging. We used a next-generation sequencing panel to identify mutations in driver genes and matched them with radiological information. Contrast-enhancing lesion localization of GBMs was classified into 4 groups based on their relationship with the subventricular zone (SVZ) and cortex (Ctx). Results: The cohort included 69 men (55.6%) and 55 women (44.4%) with a mean age of 66.4 ± 13.3 years. EGFR and PDGFRA alterations were detected in 28.2% and 22.6% of the patients, respectively. Contrast-enhancing lesion touching both the SVZ and Ctx was excluded because it was difficult to determine whether it originated from the SVZ or Ctx. Contrast-enhancing lesions touching the SVZ but not the Ctx had significantly worse overall survival than non-SVZ lesions (441 days vs. 897 days, P = .002). GBM touching only the Ctx had a better prognosis (901 days vs. 473 days, P < .001) than non-Ctx lesions and was associated with EGFR alteration (39.4% vs. 13.2%, P = .015). Multiple contrast lesions were predominant in PDGFRA alteration and RB1-wild type (P = .036 and P = .031, respectively). Conclusions: EGFR alteration was associated with cortical lesions. And PDGFRA alteration correlated with multiple lesions. Our results suggest that clarifying the association between driver genes and tumor localization may be useful in clinical practice, including prognosis prediction.

DOI： 10.1093/noajnl/vdad110

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PubMed

Language：Japanese   Publisher：Cancer Medicine  

Background: We aimed to evaluate the mutation profile, transcriptional variants, and prognostic impact of the epidermal growth factor receptor (EGFR) gene in isocitrate dehydrogenase (IDH)-wildtype glioblastomas (GBMs). Methods: We sequenced EGFR, evaluated the EGFR splicing profile using a next-generation sequencing oncopanel, and analyzed the outcomes in 138 grade IV IDH-wildtype GBM cases. Results: EGFR mutations were observed in 10% of GBMs. A total of 23.9% of the GBMs showed EGFR amplification. Moreover, 25% of the EGFR mutations occurred in the kinase domain. Notably, EGFR alterations were a predictor of good prognosis (p = 0.035). GBM with EGFR alterations was associated with higher Karnofsky Performance Scale scores (p = 0.014) and lower Ki-67 scores (p = 0.005) than GBM without EGFR alterations. EGFRvIII positivity was detected in 21% of EGFR-amplified GBMs. We identified two other EGFR variants in GBM cases with deletions of exons 6–7 (Δe 6–7) and exons 2–14 (Δe 2–14). In one case, the initial EGFRvIII mutation transformed into an EGFR Δe 2–14 mutation during recurrence. Conclusions: We found that the EGFR gene profiles of GBM differ among cohorts and that EGFR alterations are good prognostic markers of overall survival in patients with IDH-wildtype GBM. Additionally, we identified rare EGFR variants with longitudinal and temporal transformations of EGFRvIII.

DOI： 10.1002/cam4.4939

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PubMed

Language：English  

Language：English   Publishing type：Research paper (scientific journal)  

Language：English   Publishing type：Research paper (scientific journal)  

Language：English   Publishing type：Research paper (scientific journal)  

Language：Japanese   Publisher：Surgical Neurology International  

Background: Idiopathic normal pressure hydrocephalus (iNPH) is a neurological disorder presenting a triad including dementia and ventricular enlargement. The mechanism causing excessive cerebrospinal fluid (CSF) accumulation in the ventricles in iNPH is poorly understood. We hypothesized that the age-related degradation of the spinal shock-absorbing system composed of a spinal dural sac (SDS) and surrounding soft tissue, preventing ventricular enlargement caused by wide CSF pulsation driven by heartbeats, may be involved in the ventricular enlargement observed in iNPH. Methods: Sixty-four patients with iNPH in their seventies who underwent a lumboperitoneal shunt and a control group of 79 people in the same age group who underwent brain check-ups were included in the study. We compared the sizes of the cervical and upper parts of the thoracic SDS using magnetic resonance imaging between the two groups. Results: The anterior-posterior distances of the dural sac at C5 were shorter in patients with iNPH of both sexes than those in the control group (P = 0.0008 in men and P = 0.0047 in women). The number of disc levels with disappeared CSF space surrounding the cervical cord was more in iNPH (P = 0.0176 and P = 0.0003). The midsagittal area of the upper part of the spinal sac, C2-Th4, was smaller in iNPH (P = 0.0057 and P = 0.0290). Conclusion: Narrowing of the cervical dural sac and midsagittal area in the upper part of the SDS in patients with iNPH may reflect the degradation of the shock-absorbing mechanism for CSF pressure pulsations, which may cause iNPH or at least aggravate iNPH by other unknown causes.

DOI： 10.25259/SNI_474_2023

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PubMed

Authorship：Lead author   Language：Japanese  

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Rapid technological advances in molecular biology, including next-generation sequencing, have identified key genetic alterations in central nervous system (CNS) tumors. Accordingly, the fifth edition of the World Health Organization (WHO) CNS tumor classification was published in 2021. We analyzed 303 patients with diffuse glioma using an amplicon-based glioma-tailored gene panel for detecting 1p/19q codeletion and driver gene mutations such as <i>IDH1/2</i>, <i>TERTp</i>, <i>EGFR</i>, and <i>CDKN2A/B</i> on a single platform. Within glioblastomas (GBMs), the most commonly mutated genes were <i>TERTp</i>, <i>TP53</i>, <i>PTEN</i>, <i>NF1</i>, and <i>PDGFRA</i>, which was the most frequently mutated tyrosine kinase receptor in GBM, followed by <i>EGFR</i>. The genes that most commonly showed evidence of loss were <i>PTEN</i>, <i>CDKN2A/B</i>, and <i>RB1</i>, whereas the genes that most commonly showed evidence of gain/amplification were <i>EGFR</i>, <i>PDGFRA</i>, and <i>CDK4</i>. In 22 grade III oligodendroglial tumors, 3 (14%) patients had <i>CDKN2A/B</i> homozygous deletion, and 4 (18%) patients had <i>ARID1A</i> mutation. In grade III oligodendroglial tumors, an <i>ARID1A</i> mutation was associated with worse progression-free survival. Reclassification based on the WHO 2021 classification resulted in 62.5% of grade II/III <i>isocitrate dehydrogenase</i> (<i>IDH</i>) -wildtype astrocytomas being classified as <i>IDH</i>-wildtype GBM and 37.5% as not elsewhere classified. In summary, our glioma-tailored gene panel was applicable for molecular diagnosis in the WHO 2021 classification. In addition, we successfully reclassified the 303 diffuse glioma cases based on the WHO 2021 classification and clarified the genetic profile of diffuse gliomas in the Japanese population.</p>

DOI： 10.2176/jns-nmc.2022-0103

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PubMed

Language：English  

Language：English   Publisher：(一社)日本脳神経外科学会  

日本人患者のびまん性神経膠腫にみられる、分子遺伝学的プロファイルについて調査した。著者等の大学で収集した、びまん性神経膠腫患者303例の腫瘍組織試料を用いて、神経膠腫に特化した遺伝子パネルから、分子遺伝学的プロファイルを解析した。その結果、膠芽腫(GBM)患者185例に最も変異が多く認められた遺伝子は、TERTp遺伝子、TP53遺伝子、PTEN遺伝子、NF1遺伝子とPDGFRA遺伝子であった。また、欠失が最も多く認められた遺伝子は、PTEN遺伝子およびCDKN2A/B及びRB1遺伝子で、増加/増幅が最も多く認められた遺伝子は、EGFR遺伝子とPDGFRA遺伝子およびCDK4遺伝子であった。さらに、グレードIII乏突起膠腫22例のうち3例には、CDKN2A/B遺伝子のホモ接合性欠失が、4例にはARID1A遺伝子変異が検出され、ARID1A遺伝子変異が無増悪生存転帰不良と関連することが示唆された。なお、WHO脳腫瘍分類2021により、グレードII/IIIでIDH野生型星状細胞腫の62.5%が、IDH野生型のGBMに再分類された。本報により、神経膠腫に特化した遺伝子パネルが、WHO脳腫瘍分類2021に準じた分子診断に適用可能であることが確認された。

Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.98.s.hpt_63

Language：Japanese   Publisher：(一社)日本内分泌学会  

50歳代男性。頭痛の精査目的に施行された頭部MRIで鞍上部腫瘍を認められ、当科に紹介された。MRI所見は鞍上部に最大径17mmの腫瘍性病変を認め、腫瘍は視路に接していた。内分泌学的には明らかな異常は認めず、視野検査でも明らかな障害は認めなかった。無症候性ではあるものの、腫瘍が視路に接しており将来的には症候性となることが予想されたことなどから早期の手術介入の方針とし、開頭経鼻同時手術を施行した。腫瘍は赤色調で周囲脳や硬膜との癒着は目立たなかったが、右の視神経との癒着が強固であった。術中所見から右海綿静脈洞内への腫瘍進展が疑われたため、同部位と視神経周囲は愛護的に操作して腫瘍を摘出し、病理組織所見から傍神経節腫と診断した。術後に全身検索を行い、中枢神経単発の非機能性傍神経節腫と最終診断した。右海綿静脈洞周囲の残存腫瘍が緩徐に増大し、摘出術から17ヵ月後にガンマナイフ照射を行った。

Authorship：Lead author   Language：Japanese  

Language：Japanese   Publisher：(株)メディカ出版  

術前後放射線治療を必要とした脳腫瘍手術におけるコラーゲン吸収性人工硬膜の安全性を、髄液漏発生頻度を指標として検討した。脳腫瘍手術のうち、吸収性人工硬膜であるDuraGenを用いて硬膜閉鎖したもののその後の経過において放射線治療を追加せざるを得なかった症例、放射線治療後の手術においてDuraGenを使用せざるを得なかった114例(男性57例、女性57例、中央値51歳)を対象とした。脳脊髄液漏は5例(4.4%)に認められ、42～90歳で男性1名、女性4名であり、原疾患はそれぞれ膠芽腫、退形成性髄膜腫、異型性髄膜腫、軟骨肉腫、頭蓋咽頭腫で、いずれもテント上硬膜の閉鎖にDuraGenが用いられていた。髄液漏を認めた5例のうち2例は感染の関与が示唆されたため、外科的にDuraGen除去および硬膜再建術が施行された。DuraGenを用いて硬膜閉鎖を行っても、放射線治療により追加手技を要するような髄液漏発生リスクは増加しないと考えられた。

Language：Japanese   Publisher：Surgical Neurology International  

Background: We previously found the usefulness of dural sac shrinkage signs (DSSSs), which are the anterior shift of the spinal cord and dura mater behind the cord, detected by magnetic resonance imaging (MRI) at the thoracic level for the diagnosis of spontaneous intracranial hypotension (IH). This is a retrospective survey on the usefulness of DSSSs for the early detection of iatrogenic IH caused by overdrainage through a lumboperitoneal shunt (LPS) for patients with idiopathic normal pressure hydrocephalus (INPH). Methods: Forty-five INPH patients had an LPS using a pressure programmable valve equipped with an anti-siphon device. Results: Nine patients complained of orthostatic headache after the LPS, indicating IH due to overdrainage, which persisted for more than a week in three patients and 2-7days in six patients. The headache was transient/ nonorthostatic in ten patients and absent in 26 patients. The DSSSs and accompanying enlargement of the venous plexus were observed in all three patients with prolonged orthostatic headaches. Only the anterior shift of the dura mater was observed in 1 (4%) among 25 patients who had short-term orthostatic headache, transient/ nonorthostatic headache, or absent headache, and underwent spinal MRI. A patient with prolonged severe orthostatic headache with both DSSSs eventually developed intracranial subdural effusion and underwent tandem valve surgery, which provided a quick improvement of symptoms. The DSSSs on thoracic MRI also disappeared promptly. Conclusion: DSSSs may serve as objective signs for the diagnosis of IH due to overdrainage through an LPS for INPH.

DOI： 10.25259/SNI_291_2022

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PubMed

Language：Japanese  

Language：Japanese   Publisher：Interdisciplinary Neurosurgery: Advanced Techniques and Case Management  

Background: Spinal sagittal malalignment affects a patient's quality of life, and the condition has gained increasing importance. The cranial center of the gravity-sagittal vertical axis (CCG-SVA) helps in assessing sagittal alignment. Two major landmarks of the CCG are used for measuring the CCG-SVA: the midpoint of the nasion-inion line (MNI) and the external auditory canal (EAC). However, the correspondence between these two points has not been proved. In this study, we radiologically examined the positional relationship between them. Methods: We obtained lateral skull radiographs from 87 patients aged between 21 and 91 years. We evaluated the following: the vertical distance between the MNI and the upper margin of the EAC (Distance A), the horizontal distance between the plumb line from the MNI and the anterior margin of the EAC (Distance B), and the horizontal distance between the plumb line from the MNI and the center of the EAC (Distance C). Results: The average values of Distances A, B, and C were 19.6 ± 5.0, 1.65 ± 5.7, and 5.45 ± 5.5 mm, respectively, indicating that the plumb lines dropped from the MNI and the EAC did not match exactly. The measured values showed no sex-specific differences (P > 0.05, Mann–Whitney U test). Conclusions: The present study demonstrated that the MNI and EAC locations had minimal horizontal differences. The CCG-SVA should be measured by considering the differences between the MNI and the EAC.

DOI： 10.1016/j.inat.2021.101299

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Language：English   Publishing type：Research paper (scientific journal)  

Language：Japanese   Publisher：World Neurosurgery  

Objective: Pin-type head frame systems have become a worldwide standard procedure, but they can cause some complications on rare occasions. This study aimed to examine the incidence and associated risk factors of depressed skull fracture and related intracranial hematoma (DSFH) due to the use of head frames in our institute over the past 10 years. Methods: This study included 1749 patients who underwent neurosurgical surgeries using pin-type head frames, including the Mayfield (Integra NeuroSciences, Plainsboro, NJ) skull clamp (721 cases) and the Sugita (Mizuho Ikakogyo Co., Ltd., Tokyo, Japan) head frame (1028 cases). We retrospectively reviewed hospital records of our institute to identify cases of DSFH, and documented the type of head frame used, as well as patient characteristics. Results: The incidence of DSFH was 0.29% (5 of 1749 cases). All 5 cases had an associated epidural hematoma, with a single case having an additional dural laceration (without subdural damage). All perforation sites, located at the parietal bone near the pterion, occurred by the unilateral horizontal screw of the Sugita head frame. None of the patients experienced postoperative neurological decline. Conclusions: Even in the adult population, the DSFH by the pin-type head frame can occur infrequently. Based on our results, we recommend that the following factors should be considered when the pin-type head frame is used for neurosurgical procedures: location of pin application, thickness and fragility of the skull, and adequate control of compressive forces exerted by the head frame.

DOI： 10.1016/j.wneu.2021.08.070

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PubMed

Authorship：Lead author   Language：Japanese  

Language：English   Publishing type：Research paper (scientific journal)  

Authorship：Corresponding author   Language：English  

Language：Japanese   Publisher：Acta Neurochirurgica  

Background: Spontaneous intracranial hypotension (SIH) is secondary to a cerebrospinal fluid leak at the spinal level without obvious causative events. Several signs on brain and cervical spine magnetic resonance (MR) imaging (MRI) have been associated with SIH but can be equivocal or negative. This retrospective study sought to identify characteristic SIH signs on thoracic spinal MRI. Methods: Cranial and spinal MR images of 27 consecutive patients with classic SIH symptoms, who eventually received epidural autologous blood patches (EBPs), were analyzed. Results: The most prevalent findings on T2-weighted MRI at the thoracic level were anterior shift of the spinal cord (96.3%) and dorsal dura mater (81.5%), probably caused by dural sac shrinkage. These dural sac shrinkage signs (DSSS) were frequently accompanied by cerebrospinal fluid collection in the posterior epidural space (77.8%) and a prominent epidural venous plexus (77.8%). These findings disappeared in all six patients who underwent post-EBP spinal MRI. Dural enhancement and brain sagging were minimum or absent on the cranial MR images of seven patients, although DSSS were obvious in these seven patients. For 23 patients with SIH and 28 healthy volunteers, a diagnostic test using thoracic MRI was performed by 13 experts to validate the usefulness of DSSS. The median sensitivity, specificity, positive-predictive value, negative-predictive value, and accuracy of the DSSS were high (range, 0.913–0.931). Conclusions: Detection of DSSS on thoracic MRI facilitates an SIH diagnosis without the use of invasive imaging modalities. The DSSS were positive even in patients in whom classic cranial MRI signs for SIH were equivocal or minimal.

DOI： 10.1007/s00701-021-04933-w

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PubMed

Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.97.s.hpt_52

Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.97.s.hpt_89

Language：Japanese   Publisher：一般社団法人 日本内分泌学会  

DOI： 10.1507/endocrine.97.s.hpt_110

Language：Japanese   Publisher：(一社)日本内分泌学会  

術後残存腫瘍、再発腫瘍に対してガンマナイフ療法を行った下垂体腺腫40例を対象として、その効果と安全性について検討した。非機能性下垂体腺腫28例の照射線量中央値は単回照射15Gy、分割照射27.5-30Gyで、経過中の腫瘍制御率は100%であり、照射後新たにホルモン補充を要した3例のうち1例は照射半年後に副腎不全に陥った。機能性下垂体腺腫は先端巨大症11例、Cushing病1例で、照射線量中央値は25Gyであり、先端巨大症の腫瘍制御率は100%で、照射後8例でIGF-1が正常化したが、Cushing病は難治性で再手術、再照射を要した。また、機能性下垂体腺腫では内頸動脈狭窄3例と動眼神経麻痺1例を認めた。下垂体腺腫に対するガンマナイフ療法の有効性は明らかであったが、機能性下垂体腺腫では治療適応や照射線量、照射方法を再検討する必要がある。

Language：Japanese   Publisher：(一社)日本内分泌学会  

当院で手術を行った下垂体卒中32例(男性16例、女性16例、平均年齢51.7±18.5歳)の画像所見と臨床経過について検討した。その結果、術前症状は頭痛が71.9%と最多で、病理診断は非機能性下垂体腺腫が71.9%と最も多かった。頭部CTにおけるトルコ鞍内の高吸収域は50%にみられ、MRI T1強調像高信号域は56.3%、鏡面像は28.1%、副鼻腔粘膜肥厚は34.3%で認められた。また、術中所見で血腫は62.5%で認められた。尚、退院時ホルモン補充の要否と上記画像所見、あるいは術中所見との間に有意な関連は認められなかったが、頭痛発症例ではホルモン補充を要する傾向があった。

Language：Japanese   Publisher：(一社)日本内分泌学会  

70歳代、女性。近医にて副腎不全の精査中に鞍上部腫瘍を発見され、当科へ紹介となった。画像所見より下垂体卒中と診断され、ステロイド補充後に副腎不全は軽快したが、初発から2ヵ月後に急速な視力・視野障害、電解質異常、意識障害が出現した。患者は関節リウマチの治療中で、医原性の免疫不全状態であることを踏まえ、治療は下垂体炎や膿瘍など炎症・感染性疾患を鑑別して緊急で経蝶形骨洞的生検術が行われた。その結果、病理組織所見および服薬歴より本症例はメソトレキセート(MTX)関連リンパ増殖性疾患と診断された。対処としてMTX休薬とステロイド投与が行われるも効果がなく、全身状態不良のため緩和治療に移行した。

Language：Japanese   Publisher：Cancers  

Prediction of tumor consistency is valuable for planning transsphenoidal surgery for pituitary adenoma. A prospective study was conducted involving 49 participants with pituitary adenoma to determine whether quantitative pharmacokinetic analysis of dynamic contrast-enhanced magnetic resonance imaging (DCE-MRI) is useful for predicting consistency of adenomas. Phar-macokinetic parameters in the adenomas including volume of extravascular extracellular space (EES) per unit volume of tissue (ve ), blood plasma volume per unit volume of tissue (vp), volume transfer constant between blood plasma and EES (Ktrans ), and rate constant between EES and blood plasma (kep) were obtained. The pharmacokinetic parameters and the histologic percentage of collagen content (PCC) were compared between soft and hard adenomas using Mann–Whitney U test. Pearson’s correlation coefficient was used to correlate pharmacokinetic parameters with PCC. Hard adenomas showed significantly higher PCC (44.08 ± 15.14% vs. 6.62 ± 3.47%, p < 0.01), ve (0.332 ± 0.124% vs. 0.221 ± 0.104%, p < 0.01), and Ktrans (0.775 ± 0.401/min vs. 0.601 ± 0.612/min, p = 0.02) than soft adenomas. Moreover, a significant positive correlation was found between ve and PCC (r = 0.601, p < 0.01). The ve derived using DCE-MRI may have predictive value for consistency of pituitary adenoma.

DOI： 10.3390/cancers13153914

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PubMed

Language：English   Publisher：(一社)日本内分泌学会  

甲状腺刺激ホルモン産生下垂体腫瘍(TSHoma)に対するランレオチドオートゲル(LAN-ATG)療法の有効性と安全性を多施設共同試験により評価した。日本人TSHoma患者13名(男性5名、女性8名)に対してLAN-ATG 90mgを4週間毎に投与し、60mgまたは120mgに用量調整した。解析は、術前治療を受けた患者6名の24週までのデータと一次治療または術後治療を受けた患者7名の52週までのデータを解析した。有効性の主要評価項目はTSH、遊離トリヨードサイロニン(FT3)、遊離サイロキシン(FT4)の血清中濃度、副次的評価項目は下垂体腫瘍の大きさおよび臨床症状とした。その結果、治療によりTSH、FT3、FT4の血清中濃度は低下し、最終評価時まで甲状腺機能は維持された。最終評価時のFT4は10名が基準範囲内であった。最終評価時の下垂体腫瘍サイズのベースライン時からの変化率中央値(四分位範囲)は-23.8%(-38.1～-19.8)であった。臨床症状も改善した。術前治療を受けた患者は、周術期に甲状腺クリーゼを発症しなかった。安全性について、12名に有害事象が認められたが、治療中止例はなかった。主な有害事象は胃腸障害(12名)と投与部位反応(5名)であった。これらの結果から、LAN-ATGは、TSHoma患者の甲状腺機能の制御および下垂体腫瘍の縮小に有効であり、忍容性があることが示唆された。

Language：English   Publisher：(一社)日本脳神経外科学会  

頭蓋咽頭腫患者42名(男性17名、女性25名、診断時年齢中央値44歳)を対象に、Telomerase Reverse Transcriptase promoter(TERTp)、BRAF、及びCTNNB1 hotspot変異を調べ、それらの変異やメチル化が、腫瘍再発に関係するかどうか調べた。その結果、初発及び再発腫瘍においてBRAF V600E変異12名(28.6%)、CTNNB1変異21名(50%)を認めた。TERTp変異を有する患者は認めなかった。原発検体でTERTpメチル化は24名中14名(58.3%)に認めた。TERTpメチル化と、病理学的亜型、遺伝子型、あるいは腫瘍侵襲性との関連は認めなかった。結論として、頭蓋咽頭腫の腫瘍形成において、その臨床経過にかかわらず、TERTp変異の獲得を介したtelomerase活性増加は稀な経路であることが示された。

Authorship：Lead author   Language：English  

Language：Japanese   Publisher：Innovative Surgical Sciences  

Objectives: Total surgical resection is the gold standard in the treatment of craniopharyngioma. However, there is concern that aggressive surgical resection might result in high rates of endocrinologic, metabolic, and behavioral morbidities. Subtotal resection (SR) with subsequent radiation therapy (RT) may reduce surgical complications, but it may also increase the risk of tumor recurrence and radiation-induced side effects. Therefore, the optimal surgical strategy remains debatable. Methods: To determine the optimal surgical strategy, we assessed the clinical courses of 39 patients (19 male patients and 20 female patients) with newly diagnosed craniopharyngioma who were treated at our institute. The median age at diagnosis was 34 years (range: 0-76 years). The median follow-up period was 8.5 years (range: 3-160 months). Our treatment strategy comprised gross total resection (GTR) for craniopharyngioma in patients that were not at surgical risk. Conversely, after adequate tumor decompression, we used RT, mainly Gamma Knife radiosurgery, in patients at risk. We divided the patients into the following three groups depending on the treatment course: GTR, SR with RT, and SR with staged surgery. We compared tumor characteristics, as well as patients' conditions at the preoperative stage and last follow-up, among the three groups. Results: There were 8, 21, and 10 patients in the GTR, SR with RT, and SR with staged surgery groups, respectively. There were no differences in the maximum tumor diameter, tumor volume, composition, and presence of calcification among the groups. Among the 39 patients, 24 underwent transcranial microsurgery and 15 underwent trans-sphenoidal surgery as the initial treatment. No cases involving surgical mortality, cerebrospinal fluid leakage, severely deteriorated visual function, or severe hypothalamic damage were observed. No tumor recurrence was noted in the GTR group. One patient required additional RT, and one patient underwent second surgery for tumor recurrence in the SR with RT group. In the SR with staged surgery group, 8 of the 10 patients eventually underwent RT, but tumor control was achieved in all patients at the latest follow-up. In this group, the third trans-sphenoidal surgery caused a severe vascular injury in one patient. At the final follow-up, 33 (85%) patients were undergoing anterior pituitary hormone replacement, and the rate of diabetes insipidus was 51%. There was no significant difference in the pituitary dysfunction rate among the groups. Conclusions: We observed a low rate of surgical complications and a sufficient tumor control rate in response to our treatment strategy. Despite attempting preservation of the pituitary stalk, we found it difficult to rescue anterior pituitary function.

DOI： 10.1515/iss-2019-1004

Scopus

PubMed

Language：English   Publishing type：Research paper (scientific journal)  

Language：Japanese   Publisher：Journal of Oceanography  

Vertical mixing in the ocean is an important physical process that brings deep nutrients to the upper layer. The Tokara Strait, where the Kuroshio flows out to the North Pacific from the East China Sea, is known for a mixing hotspot, but the associated vertical nutrient transport has not been well quantified yet, especially in the intermediate layer below 200 m depth. This study involved flow, turbulence, and nitrate observations to quantify the vertical turbulent nitrate flux in the Tokara Strait and discuss its impact on the Kuroshio nutrient transport. Temporal variation of current velocity, derived from a 3.5-day mooring observation, captured semi-diurnal internal waves propagating vertically at depths between 200 and 400 m. Vertical shear associated with the semi-diurnal flow was elevated at this depth range. Vertical shear due to the mean and the diurnal flow was also elevated at about 220 m and 400 m depths, respectively. Mean vertical eddy diffusivity was elevated to O(10–3–10–2 m2 s−1) at the density layers of 25.2–26.4 σθ (200–450 m depth). Associated vertical turbulent nitrate flux reached O(10 mmol m−2 day−1), especially at around 25.6 σθ (250 m depth) and 26.2–26.4 σθ (400–450 m depth). The enhanced vertical turbulent nitrate flux suggests a large amount of nitrate is injected vertically to the nutrient stream from the layer just above the North Pacific Intermediate Water and is transported throughout the core of the Kuroshio nutrient stream situated at 25.0–26.2 σθ.

DOI： 10.1007/s10872-020-00581-3

Scopus

Authorship：Lead author   Language：Japanese   Publishing type：Research paper (scientific journal)   Publisher：The Japanese Congress of Neurological Surgeons  

<p>  In 2017, the World Health Organization re-classified pituitary adenomas based on hormone-producing ability and pituitary adenohypophyseal cell lineage. Non-functioning pituitary adenomas account for approximately half of all said tumors, often presenting as visual acuity and visual field disturbances. Currently, no effective pharmacologic therapy has been established for non-functioning pituitary adenomas. Therefore, surgical treatment is the first line of treatment, with endoscopic surgery becoming the mainstream choice. The simultaneous combined supra- and infrasellar approach is effective in treating giant pituitary adenomas, while stereotactic radiotherapy is effective in residual and recurrent tumors. Non-functioning pituitary adenomas are benign tumors with a promising long-term prognosis, and appropriate hormone replacement is required to maintain the patient's quality of life.</p>

DOI： 10.7887/jcns.30.11

Scopus

Authorship：Lead author   Language：Japanese   Publishing type：Research paper (scientific journal)   Publisher：日本脳神経外科コングレス  

近年、内視鏡手術の手術記録は動画をキャプチャーする方法で作成することが多いが、キャプチャー画像では実際の解剖を認識し難いことがある。今回、内視鏡下経蝶形骨洞手術の手術記録をイラストで作成し、若手教育におけるイラストの意義を検討した。教育的な意味合いを込めて、構造物を立体的に描出できるよう積極的に陰影をつけ、実際の画面では血餅などで見えにくくなっている部分はクリアにし、小さくて見にくい部分は少し強調して描くことで、構造物をわかりやすく表現した。術者の意図を表現しやすい手術イラストで手術を振り返ることは、若手医師の技術、知識の向上につながると考える。(著者抄録)

Authorship：Lead author,　Corresponding author   Language：English  

Language：Japanese   Publisher：日本脳神経外科コングレス  

<p> 近年, 内視鏡手術の手術記録は動画をキャプチャーする方法で作成することが多いが, キャプチャー画像では実際の解剖を認識し難いことがある. 今回, 内視鏡下経蝶形骨洞手術の手術記録をイラストで作成し, 若手教育におけるイラストの意義を検討した. 教育的な意味合いを込めて, 構造物を立体的に描出できるよう積極的に陰影をつけ, 実際の画面では血餅などで見えにくくなっている部分はクリアにし, 小さくて見にくい部分は少し強調して描くことで, 構造物をわかりやすく表現した. 術者の意図を表現しやすい手術イラストで手術を振り返ることは, 若手医師の技術, 知識の向上につながると考える.</p>

DOI： 10.7887/jcns.30.153

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Solitary fibrous tumor (SFT) or hemangiopericytoma (HPC) is a rare fibroblastic tumor of mesenchymal origin. SFT or HPC comprises <1% of all primary central nervous system tumors. SFT or HPC of the sellar or suprasellar region is even more unusual. We herein report a sellar SFT or HPC in an octogenarian who achieved favorable progress with partial removal followed by fractionated gamma knife radiosurgery. An 87-year-old woman presented with occasional headache and visual field defects. A rapidly growing tumor of the sella turcica was diagnosed. The patient underwent endoscopic transnasal transsphenoidal surgery; however, only partial resection of the tumor was possible, as it was fibrous and hard with increased vascularity. A histological examination confirmed the tumor to be grade II SFT or HPC. Two months after the resection, the residual tumor grew rapidly. Given the patient’s advanced age, re-surgery was not the preferred option; thus, fractionated gamma knife radiosurgery (marginal dose, 30 Gy in five fractions) was performed. MRI and visual field examination performed 3 months after irradiation revealed tumor shrinkage and improvement in the visual field, respectively. One year and three months after irradiation, the tumor continued to shrink and her visual field had improved. Taking age into consideration, partial resection with fractionated gamma knife radiosurgery was the more appropriate choice for both local tumor control and the safety of the optic apparatus.</p>

DOI： 10.2176/nmccrj.cr.2021-0103

PubMed

Language：English   Publisher：The Japan Endocrine Society  

<p>Somatostatin analogs are recommended for pharmacotherapy of TSH-secreting pituitary adenoma (TSHoma). A multicenter clinical trial was conducted to evaluate the efficacy and safety of lanreotide autogel treatment for TSHoma. A total of 13 Japanese patients with TSHoma were enrolled from February to December 2018 and treated with lanreotide autogel 90 mg every 4 weeks, with dose adjustments to 60 mg or 120 mg. Analysis was performed on data from patients receiving preoperative treatment (<i>n</i> = 6) up to 24 weeks and from those receiving primary or postoperative treatment (<i>n</i> = 7) up to 52 weeks. The primary efficacy endpoints were serum concentrations of TSH, free triiodothyronine (FT3), and free thyroxine (FT4). The secondary efficacy endpoints were pituitary tumor size and clinical symptoms. The serum concentrations of TSH, FT3, and FT4 decreased with treatment, and euthyroid status was maintained until final assessment. FT4 at final assessment was within reference ranges in 10/13 patients. The median (interquartile range) percent change in pituitary tumor size from baseline at final assessment was –23.8% (–38.1, –19.8). The clinical symptoms were also improved. The patients receiving preoperative treatment did not develop perioperative thyroid storm. Regarding safety, adverse events were observed in 12/13 patients, but none discontinued treatment. The common adverse events were gastrointestinal disorders (12/13 patients) and administration site reactions (5/13 patients). Lanreotide autogel may be effective for controlling thyroid function and reducing the pituitary tumor size, and is tolerable in patients with TSHoma (Japic Clinical Trials Information; JapicCTI-173772).</p>

DOI： 10.1507/endocrj.ej20-0707

Scopus

PubMed

Language：English   Publisher：The Japan Neurosurgical Society  

<p>Adamantinomatous craniopharyngiomas (ACP) are characterized by alterations in the <i>CTNNB1</i> gene while almost all papillary craniopharyngiomas (PCP) harbor a canonical <i>V600E</i> mutation in the BRAF gene. Although other recurrent driver genes have not been described to date in craniopharyngiomas, the heterogeneous clinical course of these tumors might be associated with the acquisition of further genomic alterations. It is well known that telomerase reverse transcriptase (<i>TERT</i>) promoter (<i>TERT</i>p) alterations, including mutations or methylation, upregulate the expression of <i>TERT</i> and increase telomerase activity, promoting tumorigenesis. We investigated whether <i>TERT</i>p mutations or methylation are associated with tumor relapse in a subset of craniopharyngiomas. Samples from 42 patients with histologically confirmed craniopharyngioma were retrieved. We determined <i>TERT</i>p, <i>BRAF</i>, and <i>CTNNB1</i> hotspot mutations in all samples using targeted sequencing and the <i>TERT</i>p methylation status by methylation-specific polymerase chain reaction (PCR) in 30 samples. While <i>BRAF</i> V600E mutations and <i>CTNNB1</i> mutations were detected in 12 (28.6%) and 21 patients (50%) in the initial tumors and subsequent recurrences, respectively, none of the patients in our cohort, including those with multiple relapses, harbored a <i>TERT</i>p mutation. Furthermore, <i>TERT</i>p methylation was detected in 14 out of 24 cases (58.3%) with available primary samples; however, no correlation between <i>TERT</i>p methylation with the pathological subtype, genotype, or tumor aggressiveness was detected. These data suggest that elevated telomerase activity via acquisition of <i>TERT</i>p mutations is an infrequent pathway in the tumorigenesis of craniopharyngiomas, regardless of their clinical course.</p>

DOI： 10.2176/nmc.rc.2020-0339

Scopus

PubMed

Language：English   Publisher：(一社)日本脳神経外科学会  

症例は87歳女性で、下垂体窩の腫瘤を指摘され当院に紹介された。数年前から間欠性の頭痛を自覚しており、Humphrey視野検査では左側優位の視野欠損を認めた。頭部CTを施行したところ、軽度高吸収を呈する下垂体腫瘤を認め、石灰化やトルコ鞍の肥大はみられなかった。MRIではトルコ鞍および鞍上に進展する固形腫瘍が検出され、T1強調像で等信号、T2強調像で等信号～高信号を示していた。1年半前に撮像されたMRI像と比較して腫瘍サイズの増大が認められ、内分泌検査ではコルチゾール低値、遊離T4低値、プロラクチン軽度高値がみられた。全身状態から手術可能と判断し、視神経減圧と組織病理サンプルの採取目的に内視鏡下経鼻経蝶形骨洞手術を施行した。術中所見では腫瘍は下垂体背側に被包化しており、吸引除去が困難であったため粉砕掻爬術を行った。凍結切片生検の結果、孤発性線維性腫瘍(SFT)/血管周囲細胞腫(HPC)が疑われ、免疫組織化学染色の結果と合わせてSFT/HPCグレードIIと診断した。術後経過は良好でプロラクチン値とコルチゾール値は正常域まで回復したが、視野欠損に改善は得られず、2ヵ月後に遺残腫瘍の増殖を認めたため分割ガンマナイフ放射線療法を開始した。治療開始3ヵ月後には腫瘍退縮が得られ、視野欠損にも改善がみられた。

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Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Venue：名古屋  

Event date： 2021.10

Language：Japanese   Presentation type：Oral presentation (general)  

Venue：Web  

Event date： 2021.9

Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Venue：Web  

Event date： 2021.9

Language：Japanese   Presentation type：Symposium, workshop panel (public)  

Event date： 2021.7

Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Event date： 2021.7

Language：Japanese   Presentation type：Symposium, workshop panel (nominated)  

Venue：東京  

Event date： 2021.6

Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Event date： 2021.6

Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Event date： 2021.5

Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Venue：Web   Country：Japan  

Event date： 2021.4

Language：Japanese   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Venue：Web   Country：Japan  

Event date： 2021.4

Language：English   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Venue：Web   Country：Indonesia  

Event date： 2021.4

Language：Japanese   Presentation type：Poster presentation  

Venue：Web  

Event date： 2020.12

Language：English   Presentation type：Public lecture, seminar, tutorial, course, or other speech  

Venue：Web   Country：Indonesia  

Event date： 2013.4

Language：Japanese  

Venue：宮城  

国内学会

Event date： 2013.3

Language：Japanese  

Venue：鹿児島  

国内学会

Event date： 2012.11

Language：Japanese  

Venue：東京  

国内学会

Event date： 2012.10

Language：Japanese  

Venue：大阪  

国内学会

Event date： 2012.10

Language：Japanese  

Venue：大阪  

国内学会

Event date： 2012.9

Language：Japanese  

Venue：横浜  

国内学会

Event date： 2012.9

Language：Japanese  

Venue：横浜  

国内学会

Event date： 2012.8

Language：Japanese  

Venue：福岡(久留米)  

国内学会

Event date： 2012.6

Language：Japanese  

Venue：鹿児島  

国内学会

Event date： 2012.5

Language：English  

Venue：Italy Firenze  

国際学会

Event date： 2012.5

Language：English  

Venue：Italy Firenze  

国際学会

Event date： 2012.4

Language：Japanese  

Venue：名古屋  

国内学会

Event date： 2012.4

Language：Japanese  

Venue：名古屋  

国内学会

Event date： 2012.2

Language：Japanese  

Venue：福岡  

国内学会

Event date： 2012.2

Language：Japanese  

Venue：東京  

国内学会

Event date： 2012.2

Language：Japanese  

Venue：東京  

国内学会

Event date： 2012.1

Language：Japanese  

Venue：浜松  

国内学会

Event date： 2012.1

Language：Japanese  

Venue：東京  

国内学会

Event date： 2011.11

Language：Japanese  

Venue：東京  

国内学会

Event date： 2011.11

Language：Japanese  

Venue：岡山  

国内学会

Event date： 2011.10

Language：Japanese  

Venue：横浜市  

国内学会

Event date： 2011.9

Language：Japanese  

Venue：横浜  

国内学会

Event date： 2011.8

Language：Japanese  

Venue：福岡  

国内学会

Event date： 2011.5

Language：Japanese  

Venue：鹿児島市  

研究会

Event date： 2011.4

Language：Japanese  

Venue：神戸市  

国内学会

Event date： 2011.4

Language：Japanese  

Venue：神戸市  

国内学会

Event date： 2011.4

Language：Japanese  

Venue：神戸市  

国内学会

Event date： 2011.3

Language：Japanese  

Venue：東京  

国内学会

Event date： 2011.3

Language：Japanese  

Venue：東京  

国内学会

Event date： 2011.1

Language：Japanese  

Venue：東京  

国内学会

Event date： 2011.1

Language：Japanese  

Venue：札幌  

国内学会

Event date： 2010.12

Language：Japanese  

Venue：千葉  

国内学会

Event date： 2010.10

Language：English  

Venue：サンフランシスコ  

国際学会

Event date： 2010.10

Language：Japanese  

Venue：大阪  

国内学会

Event date： 2010.10

Language：Japanese  

Venue：福岡  

国内学会

Event date： 2010.10

Language：English  

Venue：サンフランシスコ  

国際学会

Event date： 2010.9

Language：Japanese  

Venue：大阪  

国内学会

Event date： 2010.8

Language：Japanese  

Venue：鹿児島  

国内学会

Event date： 2010.4

Language：Japanese  

Venue：鹿児島  

研究会

Event date： 2010.3

Language：Japanese  

Venue：京都  

国内学会

Event date： 2010.3

Language：Japanese  

Venue：京都  

国内学会

Event date： 2010.2

Language：Japanese  

Venue：兵庫  

国内学会

Event date： 2010.2

Language：Japanese  

Venue：兵庫  

国内学会

Event date： 2010.1

Language：Japanese  

Venue：東京  

国内学会

Language：English  

Language：English  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：Japanese  

Language：English  

Language：Japanese  

Language：Japanese  

Language：English