Publications by authors named "Chihiro Ichikawa"

14 Publications

  • Page 1 of 1

A Rare Case of Immature Sacrococcygeal Teratoma With Lymph Node Metastasis in a Neonate.

J Pediatr Hematol Oncol 2020 Dec 14. Epub 2020 Dec 14.

Departments of Pediatric Surgery.

This is the first report of an immature sacrococcygeal teratoma with inguinal lymph node metastasis, providing the histologic transformation of an immature teratoma in association with chemotherapy. Incomplete tumor resection with coccygectomy was performed, and the histopathologic diagnosis was a grade 3 immature teratoma. Following the initial surgery, the residual tumors enlarged and the tumors metastasized to the inguinal lymph node, demonstrating immature teratoma without yolk sac tumor components. Although the tumor markers normalized after chemotherapy, the residual tumors had enlarged significantly. Therefore, a complete resection of the residual tumors was performed, and they were found to be mature teratomas.
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http://dx.doi.org/10.1097/MPH.0000000000002042DOI Listing
December 2020

Severe course with lethal hepatocellular injury and skeletal muscular dysgenesis in a neonate with infantile liver failure syndrome type 1 caused by novel LARS1 mutations.

Am J Med Genet A 2021 03 10;185(3):866-870. Epub 2020 Dec 10.

Center for Medical Genetics, Keio University School of Medicine, Tokyo, Japan.

Infantile liver failure syndrome type 1 (ILFS1) is a recently recognized autosomal recessive disorder caused by deleterious mutations in the leucyl-tRNA synthetase 1 gene (LARS1). The LARS1 enzyme is responsible for incorporation of the amino acid leucine during protein polypeptide synthesis. Individuals with LARS1 mutations typically show liver failure from infancy to early childhood during periods of illness or other physiological stress. While 25 patients from 15 families with ILFS1 have been reported in the literature, histological reports from autopsy findings are limited. We report here a premature male neonate who presented with severe intrauterine growth retardation, microcytic anemia, and fulminant liver failure, and who was a compound heterozygote for two novel deleterious mutations in LARS1. An autopsy showed fulminant hepatitis-like hepatocellular injury and fibrogenesis in the liver and a lack of uniformity in skeletal muscle, accompanied by the disruption of striated muscle fibers. Striking dysgenesis in skeletal muscle detected in the present case indicates the effect of LARS1 functional deficiency on the musculature. Whole-exome sequencing may be useful for neonates with unexplained early liver failure if extensive genetic and metabolic testing is inconclusive.
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http://dx.doi.org/10.1002/ajmg.a.62012DOI Listing
March 2021

Long-term functional outcome of sacrococcygeal teratoma after resection in neonates and infants: a single-center experience.

Pediatr Surg Int 2020 Nov 29;36(11):1327-1332. Epub 2020 Sep 29.

Department of Pediatric Surgery, Osaka Women's and Children's Hospital, Izumi, Japan.

Purpose: This study aimed to evaluate the incidence and factors associated with long-term functional outcomes of sacrococcygeal teratoma (SCT) after resection in neonates and infants.

Methods: Twenty-nine patients with a minimum of 3 years of follow-up who underwent resection and were histologically diagnosed with SCTs between 1982 and 2017 at our institution were included.

Results: The median age at the time of the study was 10.0 years. Functional disorders occurred after surgery in 6 (20.7%) patients. Anorectal dysfunction, urologic dysfunction, and lower-extremity motor disorders occurred in 6 (20.7%), 4 (13.8%), and 3 (10.3%) patients, respectively. One patient with all three types of functional disorders developed intestinal perforation due to ileus and died of sepsis at 13 years of age. The overall mortality rate after tumor resection was 3.4%. The patients who developed functional disorders presented a low 1-min Apgar score, larger tumors requiring abdominosacral resection, surgical injury to the pelvic organs, and immature or malignant histological findings.

Conclusion: Although the mortality rate was low, the long-term rate of functional disorders after SCT resection was approximately 20%. SCT patients with large tumors, surgical injury to the pelvic organs, and immature or malignant histological findings require thorough follow-up.
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http://dx.doi.org/10.1007/s00383-020-04752-7DOI Listing
November 2020

Placental histopathological features of fetoscopic laser photocoagulation for monoaminotic diamniotic twin pregnancies.

Placenta 2020 10 12;100:159-163. Epub 2020 Jul 12.

Department of Maternal Fetal Medicine, Osaka Women's and Children's Hospital, 840, Murodo, Izumi, Osaka, 594-1101, Japan. Electronic address:

Introduction: This study aimed to compare the histopathological placental features of monochorionic diamniotic (MCDA) twins who did and did not undergo fetoscopic laser photocoagulation (FLP).

Methods: This was a retrospective single-institution cohort study on MCDA twins who underwent FLP between October 2010 and December 2018. The control group included MCDA twins who did not undergo FLP and were delivered during the same period in the institute. The incidence of chorioamnionitis (CAM), funisitis, and other pathological findings was compared between the FLP and control groups after matching by gestational age at delivery.

Results: In total, 292 MCDA pregnant women who underwent FLP and 356 controls gave birth during the study period. After matching the two groups in the ratio 1:1 by gestational age at delivery, each group comprised 194 subjects. The incidence of histological CAM with Blanc association (stage I, 6.2% vs. 3.1%, crude odds ratio (cOR) = 3.1, P = 0.052; stage II, 7.2% vs. 5.7%, cOR = 1.6, P = 0.30; stage III, 2.1% vs. 2.6%, cOR = 0.66, P = 0.52) and funisitis (artery, 5.2% vs. 3.6%, cOR = 1.3, P = 0.63; vein 7.2% vs. 4.1%, cOR = 1.6, P = 0.29) was not statistically significant difference between the FLP and control groups. The FLP group demonstrated a higher incidence of partial placental infarction than the control group (10.3% vs. 3.1%, cOR = 4.3, P = 0.004, adjusted OR = 2.8, P = 0.031).

Discussion: FLP did not appear to increase the incidence of histological CAM or funisitis in subjects matched by gestational age at delivery. The FLP group demonstrated a higher incidence of partial placental infarction than the control group.
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http://dx.doi.org/10.1016/j.placenta.2020.07.002DOI Listing
October 2020

Successful catheter ablation of premature ventricular contractions triggering torsade de pointes in a small infant with histiocytoid cardiomyopathy: a case report.

Eur Heart J Case Rep 2019 Jun;3(2)

Department of Pediatric Cardiology, Osaka Women's and Children's Hospital, 840 Murodo-cho, Izumi, Osaka, Japan.

Background: A short-coupled variant of torsade de pointes (ScTdP) is rare and resistant to medical treatment. There has not been a reported catheter ablation (CA) of a short-coupled premature ventricular contraction (PVC) triggering ScTdP in an infant.

Case Summary: A neonate was referred to our hospital on the day of birth for Wolff-Parkinson-White syndrome, repeated episodes of supraventricular tachycardia, and a left ventricular non-compaction. She underwent CA of an accessory pathway at 72 days of age. On the 5th day after ablation, she had recurrent TdP episodes resistant to various antiarrhythmic drugs and received extracorporeal membrane oxygenation at 86 days of age. She underwent CA of PVCs triggering TdP at 122 days of age and a weight of 3.4 kg. Two types of PVCs triggering TdP were successfully ablated, which originated from the right ventricle (RV). Pre-potentials were recorded at the earliest ventricular activation sites of the targeted PVCs. After the ablation, she had no TdP episodes and the cardiac assist device was removed. However, she died of uncontrolled heart failure at 6 months of age. The histological findings were compatible with histiocytoid cardiomyopathy and abnormal cells were distributed throughout both ventricles. At the ablation site, fibrotic transmural lesions were noted in the RV wall.

Discussion: The PVCs triggering TdP were successfully ablated in a 4-month-old girl with histiocytoid cardiomyopathy. The PVCs were likely caused by triggered activity and associated with abnormal Purkinje cells.
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http://dx.doi.org/10.1093/ehjcr/ytz091DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6601165PMC
June 2019

A Toxoplasma gondii strain isolated in Okinawa, Japan shows high virulence in Microminipigs.

Parasitol Int 2019 Oct 31;72:101935. Epub 2019 May 31.

The United Graduate School of Veterinary Sciences, Gifu University, 1-1 Yanagido, Gifu 501-1193, Japan; Department of Veterinary Medicine, Faculty of Applied Biological Sciences, Gifu University, 1-1 Yanagido, Gifu 501-1193, Japan; Center for Highly Advanced Integration of Nano and Life Sciences, Gifu University (G-CHAIN), 1-1 Yanagido, Gifu 501-1193, Japan. Electronic address:

Toxoplasma gondii strains have been isolated all over the world and their virulence has been examined mainly using laboratory mice. However, T. gondii differs in virulence depending on the host animal species. Therefore, to evaluate the virulence of each strain in domestic animals, it is necessary to examine using not only mice but also the concerned animals. We have shown that TgCatJpOk4, a T. gondii strain recently isolated in Okinawa, Japan, has a high virulence against laboratory mice, comparable to highest virulent RH strain in mice; however, the virulence to domestic animals remains unknown. In this study, we examined the virulence using the Microminipig. After infection, four out of five infected pigs showed severe clinical symptoms: inappetence, hypoactivity and tachypnea. Eventually, three out of the five infected pigs succumbed before the end of the observation. Among the three dead pigs, histological analysis revealed that interstitial pneumonia and spotty necrosis in the liver indicating that the TgCatJpOk4 strain has a high virulence not only in laboratory mice, but in pigs as well.
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http://dx.doi.org/10.1016/j.parint.2019.101935DOI Listing
October 2019

Pleuritis and Pericarditis Following Silicone Breast Implants as Part of Autoimmune Syndrome Induced by Adjuvants.

J Clin Rheumatol 2018 Oct;24(7):404-406

Departments of General Internal Medicine Medical Center General Hospital Kobe, Japan Department of Plastic and Reconstruction Surgery Kobe City Medical Center General Hospital Kobe, Japan Department of Pathology Kobe City Medical Center General Hospital Kobe, Japan. Department of General Internal Medicine Kobe City Medical Center General Hospital Kobe, Japan

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http://dx.doi.org/10.1097/RHU.0000000000000708DOI Listing
October 2018

Phase I study of neoadjuvant chemotherapy with S-1 and oxaliplatin for locally advanced gastric cancer (Neo G-SOX PI).

ESMO Open 2017 9;2(1):e000130. Epub 2017 Mar 9.

Department of Medical Oncology, Kobe City Medical Center General Hospital, Kobe, Japan.

Background: The prognosis of locally advanced gastric cancer, such as clinical T4 disease, bulky nodal involvement, type 4 and large type 3 gastric cancer, remains unsatisfactory, even with D2 gastrectomy followed by adjuvant chemotherapy. One promising approach is neoadjuvant chemotherapy. Combination chemotherapy with S-1 and oxaliplatin (SOX) is recognised as a potentially promising regimen for gastric cancer. However, the use of neoadjuvant chemotherapy consisting of SOX for locally advanced gastric cancer has not been reported. The aim of this study was to determine the maximum tolerated dose (MTD) and recommended dose of preoperative chemotherapy combined with SOX for locally advanced gastric cancer.

Methods: Patients received two cycles of neoadjuvant chemotherapy with oxaliplatin on day 1, as well as S-1 (80 mg/m/day, twice daily) for 14 days, repeated every 3 weeks. They then underwent gastrectomy with curative D2/3 lymph node dissection followed by adjuvant S-1 (80 mg/m/day, twice daily) for 1 year. Escalation of oxaliplatin dose was planned (starting at level 0, oxaliplatin 100 mg/m; level 1, 130 mg/m).

Results: Six patients were enrolled. MTD was not reached at level 1. Oxaliplatin 130 mg/m in combination with S-1 80 mg/m/day twice daily could be administered with acceptable toxicity. Peripheral neuropathy was observed in all patients but with no functional disorders. No treatment-related death was observed and the incidence of operative morbidity was tolerable. Resection with curative intent was undertaken in all patients with R0 resection performed in five (83%) and R1 in one. Two of the six patients had a pathological complete response (33%).

Conclusion: Neoadjuvant chemotherapy with an SOX regimen was feasible in patients with locally advanced gastric cancer. The recommended phase II dose was determined to be oxaliplatin 130 mg/m in combination with S-1 80 mg/m/day, twice daily.
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http://dx.doi.org/10.1136/esmoopen-2016-000130DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5519803PMC
March 2017

Phase I Study of Neoadjuvant Chemotherapy with Capecitabine and Oxaliplatin for Locally Advanced Gastric Cancer.

Anticancer Res 2017 07;37(7):3703-3710

Department of Clinical Oncology, Kagawa University Hospital, Kita, Japan.

Aim: To determine the recommended dose of neoadjuvant chemotherapy of combined capecitabine and oxalipatin (G-XELOX) for locally advanced gastric cancer.

Patients And Methods: Patients received two cycles of neoadjuvant chemotherapy with oxaliplatin on day 1 and capecitabine (2,000 mg/m/day, b.i.d.) on days 1-14, repeated every 3 weeks. They then underwent gastrectomy with curative D2/3 lymph-node dissection followed by adjuvant therapy with S-1 for 1 year. De-escalation of oxaliplatin dose was planned (starting at level 1, oxalipatin 130 mg/m).

Results: Six patients were enrolled. The maximum tolerated dose was not reached at level 1. Oxaliplatin at 130 mg/m combined with capecitabine at 2,000 mg/m/day, b.i.d. had acceptable toxicity. No treatment-related death occurred. Most frequent drug-related adverse events during neoadjuvant G-XELOX were nausea and peripheral sensory neuropathy. One patient declined surgical resection, leaving five undergoing resection with curative intent, of whom four achieved pathological down-staging after neoadjuvant G-XELOX.

Conclusion: Neoadjuvant G-XELOX was feasible in patients with locally advanced gastric cancer.
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http://dx.doi.org/10.21873/anticanres.11742DOI Listing
July 2017

[Two Cases of Oncocytic Papillary Renal Cell Carcinoma].

Hinyokika Kiyo 2016 Apr;62(4):187-91

The Department of Urology, Kobe City Medical Center General Hospital.

Oncocytic papillary renal cell carcinoma isa variant of papillary renal cell carcinoma (PRCC). We herein report two cases treated with retroperitoneoscopic partial nephrectomy. Histologically, tumor cells of both cases exhibit round and regular nuclei with CK7 positive areas in the cytoplasm typical of TYPE1 PRCC and eosinophilic granular cytoplasm with E-cadherin positive areas in the cytoplasmic membrane, which indicates TYPE2 PRCC. Out of 46 cases reported in the literature, only one died of disease, which reveals its low malignant potential.
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April 2016

Infective endocarditis caused by Scedosporium prolificans infection in a patient with acute myeloid leukemia undergoing induction chemotherapy.

Int J Hematol 2015 Jun 29;101(6):620-5. Epub 2015 Jan 29.

Department of Hematology, Kobe City Medical Center General Hospital, 2-2, Minatojima-Minamimachi, Chuo-ku, Kobe, 650-0047, Japan.

Disseminated Scedosporium prolificans infection occurs mainly in immunocompromised patients. The mortality rate is high, as the fungus is resistant to most antifungal agents. Here, we present the case of a 66-year-old female with acute myeloid leukemia who developed infective endocarditis caused by S. prolificans infection during induction chemotherapy. Her 1,3-β-D-glucan levels were elevated and computed tomography revealed bilateral sinusitis and disseminated small nodular masses within the lungs and spleen; it nonetheless took 6 days to identify S. prolificans by blood culture. The patient died of multi-organ failure despite the combined use of voriconazole and terbinafine. Autopsy revealed numerous mycotic emboli within multiple organs (caused by mitral valve vegetation) and endocarditis (caused by S. prolificans). The geographic distribution of this infection is limited to Australia, the United States, and southern Europe, particularly Spain. The first Japanese case was reported in 2011, and four cases have been reported to date, including this one. Recently, the incidence of S. prolificans-disseminated infection in immunocompromised patients has increased in Japan. Therefore, clinicians should consider S. prolificans infection as a differential diagnosis when immunocompromised patients suffer disseminated infections with elevated 1,3-β-D-glucan levels.
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http://dx.doi.org/10.1007/s12185-015-1752-xDOI Listing
June 2015

[Long-term survival following resection of a leiomyosarcoma originating from the inferior vena cava].

Nihon Shokakibyo Gakkai Zasshi 2014 Aug;111(8):1624-31

Department of Surgery, Kobe City Medical Center General Hospital.

We describe the case of a 46-year-old woman in which a large intra-abdominal tumor was detected using computed tomography. It was a low-density, homogeneous, 7 cm tumor, adjacent to the inferior vena cava (IVC). The tumor, along with a portion of the anterior wall of the IVC, was surgically resected. The tumor originated from the IVC wall, and histopathological examination revealed a diagnosis of leiomyosarcoma. The patient is alive without recurrence 10 years after surgery. Although this disease is rare and typically has a poor prognosis, complete resection with long-term survival is achievable.
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August 2014

[A case of gastric large cell neuroendocrine carcinoma (LCNEC) for whom chemotherapy of CDDP+CPT-11 proved very effective].

Gan To Kagaku Ryoho 2010 May;37(5):895-8

Dept. of Surgery, Hata General Hospital.

Large cell neuroendocrine carcinoma(LCNEC)is a relatively new category with biological behavior similar to small cell carcinoma. Thus, it is reportedly well treated by the same chemotherapy as for small cell carcinoma. We experienced a case of gastric large cell neuroendocrine carcinoma, treated very effectively by CDDP+CPT-11.60 mg/m(2) of CDDP was administered on day 1, and 60 mg/m(2) of CPT-11 on day 1, 8 and 15. An intermission after administration for 14 days made for one course. Four courses were carried out. A complete response was obtained for a primary gastric lesion and liver metastasis, and a partial response was obtained for lymph node metastases. So far it has showed no change for 6 months after chemotherapy. Both CDDP and CPT-11 are key drugs in the chemotherapy for common gastric cancer, and it is sometimes difficult to distinguish large cell neuroendocrine carcinoma from poorly-differentiated gastric cancer. We found this combination chemotherapy is a suitable regimen for the assumed existence of large cell neuroendocrine carcinoma at the gastric lesion.
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May 2010

Depigmenting mechanisms of all-trans retinoic acid and retinol on B16 melanoma cells.

Biosci Biotechnol Biochem 2008 Oct 7;72(10):2589-97. Epub 2008 Oct 7.

Department of Applied Biological Chemistry, Faculty of Agriculture, Shizuoka University, Shizuoka, Japan.

We assessed the effects of ATRA and retinol on melanogenesis in murine B16 melanoma cells. In the present study, ATRA and retinol inhibited melanin synthesis in melanoma cells stimulated by alpha-melanocyte stimulating hormone (alpha-MSH) or 3-isobutyl-1-methylxanthine (IBMX). To elucidate the target points of ATRA and retinol on melanogenesis, we performed western blotting for melanogenic proteins, such as tyrosinase, tyrosinase-related protein (TRP)-1, and TRP-2. ATRA inhibited the expression of tyrosinase and TRP-1, and retinol inhibited the expression of tyrosinase, in a dose-dependent manner. Neither ATRA nor retinol inhibited TRP-2 expression. There were no differences in melanogenic protein expression between the two stimulants tested, alpha-MSH and IBMX. Therefore, the depigmenting effect of ATRA and retinol might be due to inhibition of the signaling pathway between cAMP and tyrosinase transcription bound to tyrosinase expression. These results indicate that ATRA and retinol are candidate anti-melanogenic agents that they might be effective in hyperpigmentation disorders.
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http://dx.doi.org/10.1271/bbb.80279DOI Listing
October 2008