Publications by authors named "Tokuhide Oyama"

12 Publications

  • Page 1 of 1

Clinical features and symptoms of IgG4-related ophthalmic disease: a multicenter study.

Jpn J Ophthalmol 2021 Sep 19;65(5):651-656. Epub 2021 Jun 19.

Kanazawa University, Kanazawa, Japan.

Purpose: The aim of this study was to elucidate the clinical features and symptoms of IgG4-related ophthalmic disease (IgG4-ROD).

Study Design: Retrospective, multicenter study.

Methods: The medical charts of 378 patients with IgG4-ROD diagnosed at 9 hospitals in Japan were reviewed. The demographic profiles, clinical findings, and ocular symptoms of the patients were analyzed.

Results: On the basis of the diagnostic criteria for IgG4-ROD, the diagnosis was definite in 261 patients (69%), probable in 45 patients (12%), and possible in 72 patients (19%). The patients' mean age at the time of diagnosis was 60.6 ± 13.9 years; 195 (52%) were male. The mean IgG4 serum level at the time of the initial diagnosis was 578.9 mg/dL. Imaging studies showed pathologic lesions as follows: lesions in the lacrimal glands (86%), extraocular muscles (21%), trigeminal nerve (20%), and eyelids (12%); isolated orbital mass (11%); diffuse orbital lesion (8%); lesion in the perioptic nerve (8%); and lesion in the sclera (1%). The ophthalmic symptoms included dry eye (22%), diplopia (20%), decreased vision (8%), and visual field defects (5%). IgG4-ROD with extraocular lesions was observed in 182 patients (48%).

Conclusion: Although the lacrimal glands are well known to be the major pathologic site of IgG4-ROD, various ocular tissues can be affected and cause ophthalmic symptoms including visual loss.
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http://dx.doi.org/10.1007/s10384-021-00847-3DOI Listing
September 2021

The natural course of IgG4-related ophthalmic disease after debulking surgery: a single-centre retrospective study.

BMJ Open Ophthalmol 2019 9;4(1):e000295. Epub 2019 Aug 9.

Division of Ophthalmology and Visual Science, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan.

Objective: This study aimed to examine the natural course and relapse rate of IgG4-related ophthalmic disease (IgG4-ROD) after debulking surgery in Japanese patients.

Methods And Analysis: This retrospective review included patients with IgG4-ROD who did not undergo further treatment following debulking surgery. The patients were diagnosed between January 2009 and December 2018 at the Department of Ophthalmology and Pathology, Niigata University Medical and Dental Hospital. The main outcome measures included postoperative IgG4-ROD recurrence rate and differences between patients with and without recurrent disease.

Results: Fifteen patients (six male, 9 female; 61.8±16.2 years) were included. Twelve patients (80.0%) had dacryoadenitis disease and three patients (20.0%) had orbital fat tissue disease. About 70%-100% of the lesion was resected in the debulking surgery and the pathological diagnosis was rendered. A definitive diagnosis was made in 13 cases (86.7%) and a probable diagnosis in 2 cases (13.3%). Patients were followed up for 39.0±25.5 months following operation. All patients had lesion volume reduction and patients with dacryoadenitis had eyelid swelling improvement after surgery. Two patients (13.3%) had disease recurrence and six patients (40.0%) had extraophthalmic lesions. There was no statistically significant difference in clinical features between relapsed and non-recurring cases.

Conclusion: We observed a 13.3% relapse rate following debulking surgery in patients with IgG4-ROD who did not undergo further treatment. This rate is lower than the documented relapse rate of 30%-70% following oral prednisolone therapy. Therefore, debulking surgery may be a treatment option for IgG4-ROD.
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http://dx.doi.org/10.1136/bmjophth-2019-000295DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6711462PMC
August 2019

The Role of Mutation Rates of GNAQ or GNA11 in Cases of Uveal Melanoma in Japan.

Appl Immunohistochem Mol Morphol 2018 10;26(9):658-663

Division of Molecular and Diagnostic Pathology.

GNAQ and GNA11 mutations are thought to be important for the tumorigenesis of uveal melanoma. Although previous studies have reported on mutation rates in cases of uveal melanoma, presently, no such report for the Japanese population exists. In this study, we examined the frequency of GNAQ and GNA11 somatic mutations in cases of uveal melanoma in Japan and their relationship with clinicopathologic features or Ki-67-positive cell rates (Ki-67 labeling index: Ki-67 LI) using immunofluorescence methods. The study involved 19 cases of uveal melanoma. We extracted the template DNA from formalin-fixed, paraffin-embedded specimens using a DNA extraction kit. We amplified the DNA sequences of GNAQ and GNA11 using polymerase chain reaction and analyzed mutations by direct sequencing. We evaluated Ki-67 LI using immunofluorescence methods. The frequencies of GNAQ and GNA11 somatic mutations were 26.3% (5/19) and 31.6% (6/19), respectively. The GNAQ and GNA11 mutations were mutually exclusive, as indicated in previous reports. The frequency of GNA11 mutations was significantly higher in epithelioid cells; however, no significant association between GNAQ mutations and cell type was evident, and there was no significant difference in Ki-67 LI between the mutation-positive and mutation-negative tumors. GNAQ and GNA11 mutations were identified in cases of uveal melanoma in Japan, although at lower frequencies than in white counterparts. The mutation frequency of GNA11 was significantly higher in epithelioid cells.
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http://dx.doi.org/10.1097/PAI.0000000000000505DOI Listing
October 2018

Secondary glaucoma associated with bilateral complete ring cysts of the ciliary body.

J Glaucoma 2014 Sep;23(7):477-81

*Division of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Niigata University †Seki Eye Clinic ‡Division of Ophthalmology, Nagaoka Chuo General Hospital, Niigata, Japan.

Purpose: To report a case of acute glaucoma due to complete ring cyst of the ciliary body.

Patients And Methods: A 33-year-old woman experienced blurred vision in her left eye. Intraocular pressure of the left eye was elevated accompanied with a very shallow anterior chamber. Anterior segment-optical coherence tomography and ultrasound biomicroscopy detected ring-shaped ciliary masses in the both eyes. Myopic change and contact between the ciliary mass and lens in the left eye suggested the presence of aqueous misdirection resulting in forward displacement of the lens-iris diaphragm. Because elevated intraocular pressure was refractory to conservative management, the left eye underwent clear lens aspiration and implantation of intraocular lens.

Results: Although the anterior chamber became deep in the left eye initially after lens extraction in conjunction with core vitrectomy, possible aqueous misdirection recurred. Second vitrectomy in the left eye together with posterior and anterior capsulotomies, to establish humoral communication between the anterior and posterior chambers, deepened the anterior chamber and lowered the intraocular pressure. The anterior chamber in the fellow right eye remained deep a year after the episode.

Conclusions: This is the first reported case of bilateral ring cysts of the ciliary body. The manifestation caused secondary glaucoma, which was resolved by lensectomy and vitrectomy.
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http://dx.doi.org/10.1097/IJG.0b013e31827a088cDOI Listing
September 2014

A case of MPO- and PR3-ANCA-positive hypertrophic cranial pachymeningitis with elevated serum IgG4.

Mod Rheumatol 2013 Jan 15;23(1):151-5. Epub 2012 Feb 15.

Division of Clinical Nephrology and Rheumatology, Department of Medicine II, Niigata University Graduate School of Medical and Dental Sciences, 1-757 Asahimachi-dori, Chuo-ku, Niigata, Niigata, 951-8510, Japan.

We report a case of orbital tumor and hypertrophic cranial pachymeningitis in a 64-year-old woman, who was initially suspected to have IgG4-related disease because of an elevated level of serum IgG4 at onset. However, her condition was resistant to glucocorticoid therapy, and additional cyclophosphamide was necessary to control the disease activity. Additional features included elevated levels of serum myeloperoxidase (MPO) and proteinase-3-anti-neutrophil cytoplasmic antibody (PR3-ANCA), and biopsy specimens from the orbital mass revealed very few infiltrating IgG4-positive cells. Instead, rupture of the elastic layer of the arterial walls with neovascularization and a small number of giant cells were observed. Considering these findings and the clinical course, the disease was considered more likely to be ANCA-associated pachymeningitis with elevation of the serum IgG4 level.
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http://dx.doi.org/10.1007/s10165-012-0613-1DOI Listing
January 2013

[Case of unifocal orbital langerhans cell histiocytosis in an adult].

No Shinkei Geka 2011 Dec;39(12):1183-8

Department of Neurosurgery, University of Niigata, Niigata, Japan.

A 34-year-old man had a 3-month history of left upper eyelid swelling and pain. Computed tomography showed osteolytic lesion of the left frontal bone. Magnetic resonance imaging (MRI) revealed a mass lesion with heterogeneously gadolinium enhancement and extension to both the lateral orbit and the frontal base. We performed mass resection via left frontotemporal craniotomy to diagnose the lesion. Pathological findings of the specimen were Langerhans cell histiocytosis (LCH). Systemic investigations revealed no sign of the disease elsewhere. The patient was diagnosed with unifocal orbital LCH and received neither chemotherapy nor radiotherapy. Follow-up MRI has shown no recurrences 2 years after surgery. Although orbital LCH rarely occurs in adults, it should be considered in differential diagnosis of orbital tumor. The patients with unifocal orbital LCH required no treatment in addition to mass resection.
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December 2011

Multifocal mucosa-associated lymphoid tissue lymphoma associated with IgG4-related disease: a case report.

Jpn J Ophthalmol 2011 May 17;55(3):304-306. Epub 2011 May 17.

Department of Ophthalmology and Visual Sciences, Niigata University Graduate School of Medical and Dental Sciences, 1-757 Asahimachi, Niigata, 951-8510, Japan.

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http://dx.doi.org/10.1007/s10384-011-0003-9DOI Listing
May 2011

Neuroblastoma amplified sequence gene is associated with a novel short stature syndrome characterised by optic nerve atrophy and Pelger-Huët anomaly.

J Med Genet 2010 Aug 24;47(8):538-48. Epub 2010 Jun 24.

Department of Molecular Genetics, Yakut Scientific Center of Complex Medical Problems, Siberian Department of Russian Academy of Medical Science, Yakutsk, Russia.

Background: Hereditary short stature syndromes are clinically and genetically heterogeneous disorders and the cause have not been fully identified. Yakuts are a population isolated in Asia; they live in the far east of the Russian Federation and have a high prevalence of hereditary short stature syndrome including 3-M syndrome. A novel short stature syndrome in Yakuts is reported here, which is characterised by autosomal recessive inheritance, severe postnatal growth retardation, facial dysmorphism with senile face, small hands and feet, normal intelligence, Pelger-Huët anomaly of leucocytes, and optic atrophy with loss of visual acuity and colour vision. This new syndrome is designated as short stature with optic atrophy and Pelger-Huët anomaly (SOPH) syndrome.

Aims: To identify a causative gene for SOPH syndrome.

Methods: Genomewide homozygosity mapping was conducted in 33 patients in 30 families.

Results: The disease locus was mapped to the 1.1 Mb region on chromosome 2p24.3, including the neuroblastoma amplified sequence (NBAS) gene. Subsequently, 33 of 34 patients were identified with SOPH syndrome and had a 5741G/A nucleotide substitution (resulting in the amino acid substitution R1914H) in the NBAS gene in the homozygous state. None of the 203 normal Yakuts individuals had this substitution in the homozygous state. Immunohistochemical analysis revealed that the NBAS protein is well expressed in retinal ganglion cells, epidermal skin cells, and leucocyte cytoplasm in controls as well as a patient with SOPH syndrome.

Conclusion: These findings suggest that function of NBAS may associate with the pathogenesis of short stature syndrome as well as optic atrophy and Pelger-Huët anomaly.
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http://dx.doi.org/10.1136/jmg.2009.074815DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2921285PMC
August 2010

[A variety of orbital lymphangioma treatments: one case treated with orbital decompression therapy and the other case with intralesional injection of OK-432 therapy].

Nippon Ganka Gakkai Zasshi 2009 Jul;113(7):732-40

Division of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Niigata University, 1-757 Asahi-machi, Niigata-shi 951-8510, Japan.

Background: Orbital lymphangioma can result in ocular emergencies due to acute enlargement, leading to painful proptosis, compressive optic neuropathy, exposure keratopathy, restrictive ocular motility and increased intraocular pressure. Treatment methods and clinical courses of two cases with orbital lymphangioma are reported, one with orbital decompression therapy and the other with intralesional injection of OK-432 therapy. CASE 1: An 8-year-old girl had multilocular cystic orbital intracornal lymphangioma. Emergency operation was deemed necessary because the patient presented with the painful proptosis, the compressive optic neuropathy, and increased intraocular pressure due to acute enlargement, we put the orbital inferior wall was decompressed by inserting an intact inframedial orbital strut. The proptosis vanished completely and visual acuity loss improved. CASE 2: A 2-year-old girl had multilocular cystic orbital intra and extracornal lymphangioma. She had severe proptosis and exposure keratopathy. The extracornal cystic lesion was treated with an intralesional injection of OK-432. On the 7th day after injection, the patient presented with painful severe proptosis. Aspiration of the fluid in the cystic lesion was performed, and the drain was preserved. After the treatment, both the severe proptosis and the exposure keratopathy improved.

Conclusions: Inferior wall decompression was performed on case 1 while keeping the inframedial orbital strut. Although the eye movement disorder was low at the time, the medial and lateral wall decompression improved. In case 2, the extracornal cystic lesion was treated with an intralesional injection of OK-432 Judging from the results of that clinical course, the injection of OK-432 to the intracornal lesion could prove dangerous to the visual performance. Although OK-432 is predictably-effective against lymphangioma, it should not perform be applied without careful consideration.
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July 2009

Changes of scleral sulfated proteoglycans in three cases of nanophthalmos.

Jpn J Ophthalmol 2009 Mar 31;53(2):171-175. Epub 2009 Mar 31.

Division of Ophthalmology and Visual Science, Niigata University, Niigata, Japan.

Purpose: To examine the composition of scleral sulfated proteoglycans in three nanophthalmic eyes by electron microscopic histochemistry.

Methods: Scleral tissues from three cases of nanophthalmos were collected from the Niigata University Hospital for transmission electron microscopic examination. Sulfated proteoglycans were studied using the cuprolinic blue (CB) dye binding method in conjunction with enzymatic digestion.

Results: Compared with the controls, the nanophthalmic sclerae were much thicker, and the collagen fibrils showed a less orderly arrangement. Twisting or fraying of collagen fibrils was noted in some areas, and fine fibrous materials surrounded the abnormal fibrils. CB-positive proteoglycan filaments were found around the collagen fibrils and in the fine fibrous materials in the nanophthalmic specimens. The number of proteoglycan filaments was smaller than that in the normal specimens. After enzymatic digestion, the CB-positive filaments were identified as dermatan/chondroitin sulfate proteoglycans. Although the number of dermatan sulfate filaments in nanophthalmos sclerae was similar to that in the controls, there were many fewer chondroitin sulfate filaments.

Conclusion: A loss of chondroitin sulfate proteoglycan was observed in nanophthalmic sclerae. The alteration may be related to the abnormal organization of collagen fibrils and the onset of uveal effusion.
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http://dx.doi.org/10.1007/s10384-008-0641-8DOI Listing
March 2009

The connective tissue and glial framework in the optic nerve head of the normal human eye: light and scanning electron microscopic studies.

Arch Histol Cytol 2006 Dec;69(5):341-56

Division of Microscopic Anatomy and Bio-imaging, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan.

The arrangement of connective tissue components (i.e., collagen, reticular, and elastic fibers) and glial elements in the optic nerve head of the human eye was investigated by the combined use of light microscopy and scanning electron microscopy (SEM). Light-microscopically, the optic nerve head could be subdivided into four parts from the different arrangements of the connective tissue framework: a surface nerve fiber layer, and prelaminar, laminar, and postlaminar regions. The surface nerve fiber layer only possessed connective tissue elements around blood vessels. In the prelaminar region, collagen fibrils, together with delicate elastic fibers, formed thin interrupted sheaths for accommodating small nerve bundles. Immunohistochemistry for the glial fibrillary acidic protein (GFAP) showed that GFAP-positive cells formed columnar structures (i.e., glial columns), with round cell bodies piled up into layers. These glial columns were located in the fibrous sheaths of collagen fibrils and elastic fibers. In the laminar region, collagen fibrils and elastic fibers ran transversely to the optic nerve axis to form a thick membranous layer - the lamina cribrosa - which had numerous round openings for accommodating optic nerve fiber bundles. GFAP-positive cellular processes also ran transversely in association with collagen and elastin components. The postlaminar region had connective tissues which linked the lamina cribrosa with fibrous sheaths for accommodating nerve bundles in the extraocular optic nerve, where GFAP-positive cells acquired characteristics typical of fibrous astrocytes. These findings indicate that collagen fibrils, as a whole, form a continuous network which serves as a skeletal framework of the optic nerve head for protecting optic nerve fibers from mechanical stress as well as for sustaining blood vessels in the optic nerve. The lamina cribrosa containing elastic fibers are considered to be plastic against the mechanical force affected by elevation of the intraocular pressure. The present study has also indicated that glial cells with an astrocytic character play an important role in constructing the connective tissue framework characteristic of the optic nerve head.
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http://dx.doi.org/10.1679/aohc.69.341DOI Listing
December 2006

[Two cases of Senior-Loken syndrome].

Nippon Ganka Gakkai Zasshi 2004 Jan;108(1):29-37

Division of Ophthalmology and Visual Science, Graduate School of Medical and Dental Sciences, Niigata University, 1-757 Asahi-machi, Niigata 951-8510, Japan.

Background: We report two rare cases of Senior-Loken syndrome, one with fundus lesions simulating Coats disease. CASE 1: A 14-year-old boy had juvenile nephronophthisis and bilateral retinitis pigmentosa with retionochoroidal atrophy and salt and pepper fundus. Electroretinogram (ERG) showed non-recordable changes and the Goldmann perimeter showed ring scotomas. CASE 2: A 14-year-old boy had bilateral retinitis pigmentosa and juvenile nephronophthisis with continuous ambulatory peritoneal dialysis. Additionally, both eyes showed massive exudates seen in Coats disease in the entire periphery, exudative retinal detachment, proliferative changes in the inferior periphery, yellow opacitas corporis vitrei, keratoconus, and cataract. The left eye had vitreous hemorrhages. Both eyes received vitreous surgery and endophotocoagulation. After the surgery the left eye showed neovascular glaucoma and a cyclophotcoagulation was performed. The massive edema in the superior retina of both eyes disappeared and intra-ocular pressure in the left eye was normalized. However, despite these treatments, both eyes finally had no light perception.

Conclusion: Case 1 was a typical SLS, and case 2 was a rare one with fundus lesions simulating Coats disease, keratoconus, and cataract. We suspect the exudative changes resulted from various vessel changes and choroidal circulatory disturbance of renal retinopathy, retinal vascular damage, and destruction of blood-retinal barrier with retinitis pigmentosa, the inflammatory changes of vessels, and other things.
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January 2004
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