Publications by authors named "Koh-Ichi Ohshima"

17 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

Upregulated Expression of Activation-Induced Cytidine Deaminase in Ocular Adnexal Marginal Zone Lymphoma with IgG4-Positive Cells.

Int J Mol Sci 2021 Apr 15;22(8). Epub 2021 Apr 15.

Division of Pathophysiology, Okayama University Graduate School of Health Sciences, Okayama 700-8558, Japan.

Immunoglobulin G4-related disease (IgG4-RD) is a systemic disorder characterized by tissue fibrosis and intense lymphoplasmacytic infiltration, causing progressive organ dysfunction. Activation-induced cytidine deaminase (AID), a deaminase normally expressed in activated B-cells in germinal centers, edits ribonucleotides to induce somatic hypermutation and class switching of immunoglobulin. While AID expression is strictly controlled under physiological conditions, chronic inflammation has been noted to induce its upregulation to propel oncogenesis. We examined AID expression in IgG4-related ophthalmic disease (IgG4-ROD; = 16), marginal zone lymphoma with IgG4-positive cells (IgG4+ MZL; = 11), and marginal zone lymphoma without IgG4-positive cells (IgG4- MZL; = 12) of ocular adnexa using immunohistochemical staining. Immunohistochemistry revealed significantly higher AID-intensity index in IgG4-ROD and IgG4+ MZL than IgG4- MZL ( < 0.001 and = 0.001, respectively). The present results suggest that IgG4-RD has several specific causes of AID up-regulation in addition to inflammation, and AID may be a driver of oncogenesis in IgG4-ROD to IgG4+ MZL.
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http://dx.doi.org/10.3390/ijms22084083DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8071226PMC
April 2021

Treatment outcomes of IgG4-producing marginal zone B-cell lymphoma: a retrospective case series.

Int J Hematol 2020 Dec 12;112(6):780-786. Epub 2020 Aug 12.

Department of Hematology and Oncology, Okayama University Hospital, 2-5-1, Shikata-cho Kita-ku, Okayama, 700-8558, Japan.

IgG4-producing marginal zone B-cell lymphomas (MZLs) have been recently proposed as a subtype of MZLs. Despite the abundant literature on pathophysiological features of this type of lymphoma, only a few retrospective studies pertaining to the treatment outcomes have been reported, and its prognosis remains unclear. We retrospectively analyzed seven patients with IgG4-producing MZLs diagnosed at our institute, with specific reference to treatment and outcomes. The median age was 69.0 years (55-79), and all were males. The median follow-up period was 66.6 months (8-121). All patients had localized disease; four patients had tumors of the ocular adnexa, whereas two had retroperitoneal tumors. Five patients were treated with irradiation (30 Gy/15 fr) (n = 4) or surgery (n = 1), resulting in tumor reduction. Two patients were treated by chemotherapy or irradiation. Among them, one commenced rituximab monotherapy, which led to an inadequate reduction of the tumor. Subsequent irradiation induced complete response (CR). The other patient experienced repeated relapses during follow-up and finally achieved CR by combination chemotherapy. Treatment was well tolerated in all cases, and none of the patients showed disease progression at the last follow-up visit. Our results indicate that the standard treatments for MZLs are generally appropriate for IgG4-producing MZL.
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http://dx.doi.org/10.1007/s12185-020-02968-wDOI Listing
December 2020

A subset of ocular adnexal marginal zone lymphomas may arise in association with IgG4-related disease.

Sci Rep 2015 Aug 27;5:13539. Epub 2015 Aug 27.

Department of Pathology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan.

We previously suggested a relationship between ocular immunoglobulin (Ig)G4-related disease (IgG4-RD) and marginal zone lymphomas (MZLs). However, the cytokine background associated with these disorders and whether it differs between ocular adnexal MZLs with (IgG4-associated MZL) and without (IgG4-negative MZL) numerous IgG4(+) plasma cells are unknown. In this study, we identified the mRNA expression pattern of Th2 and regulatory T-cell (Treg) cytokines in IgG4-RD and in IgG4-associated MZL and IgG4-negative MZL using real-time polymerase chain reaction analysis. Ocular IgG4-RD and IgG4-associated MZL exhibited significantly higher expression ratios of interleukin (IL)-4/β-actin, IL-10/β-actin, IL-13/β-actin, transforming growth factor (TGF) β1/β-actin, and FOXP3/β-actin than did IgG4-negative MZL (p < 0.05). This finding further supports our prior observations that a significant subset of ocular MZLs arises in the setting of IgG4-RD. Furthermore, the presence of a different inflammatory background in IgG4-negative MZLs suggests that IgG4-associated MZLs may have a different pathogenesis.
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http://dx.doi.org/10.1038/srep13539DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC4550912PMC
August 2015

Location and frequency of lesions in patients with IgG4-related ophthalmic diseases.

Graefes Arch Clin Exp Ophthalmol 2014 Mar 3;252(3):531-8. Epub 2014 Jan 3.

Department of Ophthalmology, Mitoyo General Hospital, 708, Himehama, Toyohama-cho, Kanonji, Kagawa, 7691695, Japan,

Background: It is well-known that the lacrimal gland (LG) may be affected in IgG4-related ophthalmic disease (IgG4ROD). Recently, IgG4-related ophthalmic lesions other than those of the lacrimal gland have been reported. However, no study to date has revealed the details of these lesions. This study was conducted to evaluate the location and frequency of lesions found in conjunction with IgG4ROD using radiological imaging.

Methods: Radiological images and clinical records of 65 patients collected from seven institutions in Japan were reviewed retrospectively. All patients had been pathologically diagnosed with IgG4ROD. Patients of mucosa-associated lymphoid tissue lymphoma associated with IgG4-related lesions were excluded. Orbital magnetic resonance imaging or computed tomography findings were evaluated.

Results: Of the 65 patients, 31 (47.7 %) had lesions involving the LG alone, whereas 34 (52.3 %) had lesions involving the areas other than LG, including eight patients who had lesions without any LG involvement. IgG4-related ophthalmic lesions included LG enlargement in 57 patients (87.7 %), trigeminal nerve branch enlargement in 25 (38.5 %), extraocular muscle enlargement in 16 (24.6 %), diffuse orbital fat lesions in 15 (23.1 %), orbital mass lesions in 11 (16.9 %), eyelid lesions in eight (12.3 %), and nasolacrimal duct lesion in one (1.5 %). Six patients (9.2 %) presented with visual disturbance due to optic nerve disturbance, eight (12.3 %) with a restriction of ocular movement, and 19 (29.2 %) with exophthalmos.

Conclusions: Thirty-four (52.3 %) of the 65 IgG4ROD patients had lesions in areas other than LG. Lesions were found in the trigeminal nerve branch including pterygopalatine fossa, extraocular muscles, orbital fat, eyelid, and nasolacrimal duct.
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http://dx.doi.org/10.1007/s00417-013-2548-4DOI Listing
March 2014

IgG4-related disease involving the sclera.

Mod Rheumatol 2014 Jan;24(1):195-8

Department of Pathology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Science , 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558 , Japan.

A 49-year-old female patient previously treated for scleritis and uveitis-induced cataract in the right eye presented with a subretinal white lesion in the same eye. With a preliminary diagnosis of choroidal tumor, enucleation of the eyeball was performed in accordance with the patient's request. Histologic and immunohistologic examinations were consistent with immunoglobulin G4-related disease. The case demonstrates that it is important to consider IgG4-related disease in the differential diagnosis of an intraocular tumor.
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http://dx.doi.org/10.3109/14397595.2013.852842DOI Listing
January 2014

A case of IgG4-related dacryoadenitis that regressed without systemic steroid administration.

J Clin Exp Hematop 2013 ;53(1):53-6

Division of Ophthalmology, National Hospital Organization Okayama Medical Center, Okayama, Japan.

There are no reports on the effect of anti-allergic agents against IgG4-related disease. We herein report a case of IgG4-related dacryoadenitis that is believed to have regressed due to the administration of anti-allergic agents. A 57-year-old woman consulted us because of bilateral temporal upper eyelid swelling and induration. She had also been suffering from allergic rhinitis and allergic conjunctivitis for 20 years. We performed an incisional biopsy of the lesion. With respect to the pathology, extranodal marginal zone lymphoma of mucosa-associated lymphoid tissue type was strongly suspected. On obtaining consent from the patient, follow-up alone was to be continued without radiation therapy. In addition to the observation of lacrimal gland lesions, the administration of epinastine hydrochloride at a dosage of 20 mg/day and 0.01% betamethasone eye drops twice a day to both eyes was commenced in order to treat both allergic rhinitis and allergic conjunctivitis. The lacrimal gland lesion decreased in size over time, becoming predominantly normal 7 years after the commencement of agent administration. We therefore re-examined the blood and pathology specimens. As a result, the serum IgG4 level was found to have increased to 540 mg/dl, while IgG4/IgG was 36.2%. The pathological diagnosis was revised to IgG4-related dacryoadenitis. The hypotheses of spontaneous remission and/or the effect of epinastine hydrochloride administration can be proposed regarding the mechanism by which the lacrimal gland lesion decreased in size. [J Clin Exp Hematop 53(1): 53-56, 2013].
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http://dx.doi.org/10.3960/jslrt.53.53DOI Listing
May 2014

A case of conjunctival follicular lymphoma mimicking mucosa-associated lymphoid tissue lymphoma.

J Clin Exp Hematop 2013 ;53(1):49-52

Department of Pathology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan.

Ocular adnexal lymphoma may involve the eyelids, conjunctiva, orbital tissue, or lacrimal structures. The majority are non-Hodgkin's B-cell lymphomas of mucosa-associated lymphoid tissue (MALT) lymphoma type. Follicular lymphomas represent a small percentage of ocular adnexa lymphomas, particularly in Japan. We report a 68-year-old female patient who presented with a salmon pink patch-like lesion of the left conjunctiva, suspected of being (MALT) lymphoma. However, histologic and immunohistologic examinations were consistent with follicular lymphoma. This case demonstrates the importance of considering such rare lymphomas when making a diagnosis of ocular adnexal lymphoid neoplasms. [J Clin Exp Hematop 53(1): 49-52, 2013].
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http://dx.doi.org/10.3960/jslrt.53.49DOI Listing
May 2014

Pathological findings of infraorbital nerve enlargement in IgG4-related ophthalmic disease.

Jpn J Ophthalmol 2012 Sep 27;56(5):511-4. Epub 2012 Jul 27.

Department of Ophthalmology, Mitoyo General Hospital, 708 Himehama, Toyohama-cho, Kanonji, Kagawa 769-1695, Japan.

Purpose: We report a case of bilateral infraorbital nerve enlargement (IONE) associated with immunoglobulin (Ig)G4-related ophthalmic disease and describe the associated histopathologic findings.

Case: An otherwise healthy 59-year-old man presented with bilateral exophthalmos and right visual disturbance. Orbital magnetic resonance imaging showed bilateral IONE and a soft tissue mass in the right orbit. Excisional biopsy in the left infraorbital canal was performed. Histopathologic assessment revealed IgG4-related disease involving the epineurium of the infraorbital nerve. The patient received systemic steroid therapy, to which he responded well.

Conclusion: IONE in IgG4-related ophthalmic disease is due to IgG4-related disease involving the epineurium.
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http://dx.doi.org/10.1007/s10384-012-0170-3DOI Listing
September 2012

Ocular adnexal IgG4-producing mucosa-associated lymphoid tissue lymphoma mimicking IgG4-related disease.

J Clin Exp Hematop 2012 ;52(1):51-5

Department of Pathology, Okayama University Graduate School of Medicine, Dentistry, and Pharmaceutical Sciences, Okayama, Japan.

IgG4-related disease is a recently proposed clinical entity with several unique clinicopathological features. A chronic inflammatory state with marked fibrosis, which can often be mistaken for malignancy, especially by clinical imaging analyses, unifies these features. In the present report, we describe a case of IgG4-producing mucosa-associated lymphoid tissue lymphoma mimicking IgG4-related disease. The patient was a 55-year-old male who was being followed for right orbital tumor over 1.5 years. The lesion had recently increased in size, so a biopsy was performed. Histologically, the lesion was consistent with IgG4-related disease ; however, IgG4+ plasma cells showed immunoglobulin light-chain restriction and immunoglobulin heavy chain gene rearrangement was detected in the lesion. Therefore, the lesion was diagnosed as IgG4-producing mucosa-associated lymphoid tissue lymphoma. In conclusion, in histological diagnosis of IgG4-related disease, it is important to examine not only IgG4-immunostain but also immunoglobulin light-chain restriction.
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http://dx.doi.org/10.3960/jslrt.52.51DOI Listing
October 2012

The usefulness of infraorbital nerve enlargement on MRI imaging in clinical diagnosis of IgG4-related orbital disease.

Jpn J Ophthalmol 2012 Jul 30;56(4):380-2. Epub 2012 May 30.

Section of Ophthalmology, National Hospital Organization Okayama Medical Center, 1711-1 Tamasu, Kitaku, Okayama 701-1154, Japan.

Purpose: To investigate the frequency of infraorbital nerve enlargement (IONE) in orbital lymphoproliferative disorders, and to show that IONE can contribute to the clinical diagnosis of IgG4-related orbital diseases (IgG4-ROD).

Subjects And Methods: 71 cases in which orbital lymphoproliferative disorders were diagnosed at Okayama Medical Center and Mitoyo General Hospital from April, 2004 to March, 2011 were investigated. The male-to-female ratio was 39:32, and the age range 27-87 years old (average age 64.1 years). Whenever the coronal section of the infraorbital nerve was larger than that of the optic nerve on MRI, it was defined as IONE.

Results: The breakdown of the 71 cases was: 45 cases of non-Hodgkin lymphoma, 16 cases of IgG4-ROD, 5 cases of reactive lymphoid hyperplasia, and 5 cases of idiopathic orbital inflammation. Of these, a total of 9 cases had IONE. The incidence of IONE was compared between the IgG4-ROD patient group and the non-IgG4-ROD patient group and was significantly higher in the IgG4-ROD patient group (p < 0.0001).

Conclusion: If IONE is observed in a case of orbital lymphoproliferative disorders on MRI, then it is highly possible that such a case is IgG4-ROD.
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http://dx.doi.org/10.1007/s10384-012-0151-6DOI Listing
July 2012

Diffuse infiltrating retinoblastoma invading subarachnoid space.

Clin Ophthalmol 2011 23;5:861-3. Epub 2011 Jun 23.

Department of Ophthalmology, Hokkaido University Graduate School of Medicine, Sapporo;

We report herein an unusual case of diffuse infiltrating retinoblastoma involving the brain, which caused a patient's death 27 months after enucleation. An eight-year-old boy complained of blurred vision in his right eye (OD) in October 2006. Funduscopic examination showed optic disc swelling, dense whitish vitreous opacity, and an orange-colored subretinal elevated lesion adjacent to the optic disc. Fluorescein angiography revealed hyperfluorescence in the peripapillary region at an early-phase OD. Because the size of the subretinal lesion and vitreous opacity gradually increased, he was referred to us. His visual acuity was 20/1000 OD on June 20, 2007. Slit-lamp biomicroscopy showed a dense anterior vitreous opacity. Ophthalmoscopically, the subretinal orange-colored area spread out until reaching the mid peripheral region. A B-mode sonogram and computed tomography showed a thick homogeneous lesion without calcification. Gadolinium-enhanced magnetic resonance imaging showed a markedly enhanced appearance of the underlying posterior retina. Enucleation of the right eye was performed nine months after the initial presentation. Histopathology demonstrated retinal detachment and a huge choroidal mass invading the optic nerve head. The tumor was consistent with diffuse infiltrating retinoblastoma. The patient died due to brain involvement 27 months after enucleation. Ophthalmologists should be aware that diffuse infiltrating retinoblastoma may show an unfavorable course if its diagnosis is delayed.
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http://dx.doi.org/10.2147/OPTH.S20913DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3133001PMC
November 2011

Orbital inflammation caused by a spirurid larva type X showing small speckles on magnetic resonance imaging.

Jpn J Ophthalmol 2009 Nov 18;53(6):667-668. Epub 2009 Dec 18.

Japan Association of Parasite Control, Tokyo, Japan.

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http://dx.doi.org/10.1007/s10384-009-0734-zDOI Listing
November 2009

Ocular adnexal IgG4-related disease has uniform clinicopathology.

Pathol Int 2008 Aug;58(8):465-70

Department of Pathology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan.

IgG4-related disease is a recently proposed clinical entity with several unique clinicopathological features. Ocular adnexal IgG4-related disease, however, has not well been clarified. The purpose of the present study was to examine 21 patients (10 men, 11 women; age range, 39-86 years) with ocular adnexal IgG4-related disease. In 17 out of 21 patients (81%), the lacrimal glands were involved and bilateral lacrimal gland swelling was frequently observed (n = 12; 70.6%). In contrast, the conjunctiva was not involved in any of the patient. Histology was uniform with marked lymphoplasmacytic infiltration admixed with dense fibrosis, similar to previous reports of IgG4-related disease. Immunostaining detected numerous aggregates of IgG4-positive plasma cells. Serum IgG4 was higher than normal in 10 of the 13 patients tested, although it was measured after treatment in almost all cases. Interestingly, immunoglobulin heavy chain gene rearrangement was detected in two of 17 patients (12%) examined. The present results show that ocular adnexal IgG4-related disease has uniform clinicopathology: that is, disease involving the bilateral lacrimal glands with lymphoid hyperplasia and fibrosis, but not the conjunctiva. And presence of immunoglobulin heavy chain gene rearrangement suggests the possibility of B-cell lymphoma arising in a background of IgG4-related chronic inflammation.
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http://dx.doi.org/10.1111/j.1440-1827.2008.02257.xDOI Listing
August 2008

Neurilemoma of the bulbar conjunctiva.

Jpn J Ophthalmol 2007 Jan-Feb;51(1):68-9. Epub 2007 Feb 9.

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http://dx.doi.org/10.1007/s10384-006-0393-2DOI Listing
April 2007

Primary sebaceous carcinoma of the lacrimal gland treated by carbon ion radiotherapy.

Int J Clin Oncol 2003 Dec;8(6):386-90

Hospital, Research Center for Charged Particle Therapy, National Institute of Radiological Sciences, 4-9-1 Anagawa, Inage-ku, Chiba 263-8555, Japan.

Sebaceous carcinoma is a rare primary neoplasm of the lacrimal gland and, to the best of our knowledge, only six cases have been reported previously. Sebaceous carcinoma of the orbit more commonly occurs as secondary invasion from the eyelid, but may occur by way of metastatic spread from elsewhere in the body. We describe a patient who presented with a rapidly growing neoplasm of the lacrimal gland which, histologically, was a sebaceous carcinoma. The eyelid was entirely normal on examination. In this patient we found a single tumor cell line within the normal lacrimal gland. In this article we describe the diagnosis and treatment with carbon ion radiotherapy of primary sebaceous carcinoma of the lacrimal gland, a condition not previously reported in Japan.
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http://dx.doi.org/10.1007/s10147-003-0348-2DOI Listing
December 2003
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