Publications by authors named "Masanori Hisaoka"

132 Publications

Fatal hemoperitoneum due to rupture of mesenteric artery in remission state of microscopic polyangiitis, concomitant with severe hypertension and posterior reversible encephalopathy syndrome: an autopsy case report.

CEN Case Rep 2021 May 4. Epub 2021 May 4.

Department of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyushu, Fukuoka, Japan.

Microscopic polyangiitis (MPA) is a type of necrotizing vasculitis associated with high levels of myeloperoxidase-specific antineutrophil cytoplasmic antibody (MPO-ANCA). While generally associated with renal dysfunction, MPA can also cause intraabdominal hemorrhage in rare cases. A 66-year-old man was admitted to our hospital for renal dysfunction, numbness, and weight loss for 3 months. He had no significant medical history. Renal biopsy revealed crescentic glomerulonephritis with necrotizing vasculitis, which was associated with a high serum titer of MPO-ANCA, leading to a diagnosis of MPA. Remission-induction treatment with glucocorticoids and rituximab was initiated, which improved the patient's general condition and renal failure. His blood pressure was elevated and was controlled by amlodipine treatment. Two months after discharge, he visited the emergency department because of chest pain. A diagnosis of acute cardiovascular syndrome was suggested; however, his cardiac artery was not stenotic. The patient's blood pressure was high despite antihypertensive therapy, and he developed posterior reversible encephalopathy syndrome (PRES). Despite intensive treatment, the patient died 3 days later. An autopsy revealed that the cause of death was hypovolemic shock due to massive intra-abdominal hemorrhage from the ruptured mesenteric artery involved in vasculitis. In cases of MPA with sudden-onset chest or abdominal pain, a ruptured intra-abdominal artery should be considered. Secondary hypertension associated with vasculitis should be carefully managed to prevent hemorrhagic complications and PRES.
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http://dx.doi.org/10.1007/s13730-021-00606-wDOI Listing
May 2021

Malignant Transformation of Giant Cell Tumor of Bone 7 years After Initial Surgery: A Case Report and Literature Review.

JBJS Case Connect 2021 04 20;11(2). Epub 2021 Apr 20.

Department of Orthopaedic Surgery, Kyushu Rosai Hospital, Japan.

Case: A 64-year-old man with a history of giant cell tumor of bone (GCTB) in the fibula 7 years earlier developed a recurrence with histologic features of osteosarcoma. Both the primary GCTB and the secondary osteosarcoma were found to have the H3F3A gene mutation. Despite immediate above-the-knee amputation, the patient died of respiratory failure because of lung metastases 3 months later.

Conclusion: This is the first report of proven H3F3A mutation in both the primary GCTB and the secondary osteosarcoma in the same case. Clinicians should consider secondary malignancy in patients presenting with a lesion at the site of a previously treated GCTB after a long interval.
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http://dx.doi.org/10.2106/JBJS.CC.20.00417DOI Listing
April 2021

Cardiac Tamponade as an Unusual Initial Clinical Manifestation of CIC-DUX4 Sarcoma.

Am J Case Rep 2021 Feb 28;22:e929349. Epub 2021 Feb 28.

Department of Orthopedic Surgery, Kyushu Rosai Hospital, Kitakyushu, Fukuoka, Japan.

BACKGROUND CIC-rearranged sarcoma (CRS) is a recently described subset of undifferentiated small-round-cell sarcomas of bone and soft tissue. DUX4 is the most common gene involved in CRS. CRS usually presents in the soft tissue of the trunk and extremities, and is recognized as being clinically aggressive, with poor prognosis. Our case highlights an unusual presentation of CRS with cardiac tamponade. CASE REPORT A 48-year-old man presented with hypotension caused by hemorrhagic cardiac tamponade. ¹⁸F-fluorodeoxyglucose-positron emission tomography showed increased uptake in multiple lesions, including lesions in the left proximal humerus and several lymph nodes. Biopsy specimens of the humerus revealed proliferation of round-shaped cells. In addition, CIC-DUX4 gene rearrangement was detected by polymerase chain reaction and direct sequencing, leading to a diagnosis of cardiac tamponade caused by CRS. Although the patient received systemic chemotherapy as well as radiotherapy to the mediastinal lesion and left humerus, he died of progressive disease 12 months after diagnosis. CONCLUSIONS Because CRS is a recently proposed entity that is distinct from Ewing sarcoma, the clinical presentation and outcome of CRS has not been well documented in the literature. This is the first case report of CRS presenting as cardiac tamponade. Although cardiac tamponade due to metastatic sarcoma is extremely rare, CRS can be included in the differential diagnosis.
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http://dx.doi.org/10.12659/AJCR.929349DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7931773PMC
February 2021

Primary clear cell sarcoma of the femur: a unique case with RT-PCR and direct sequencing confirmation of EWSR1/ATF1 fusion gene.

BMC Musculoskelet Disord 2021 Jan 21;22(1):99. Epub 2021 Jan 21.

Department of Orthopaedic Surgery, Faculty of Medicine, Oita University, 1-1 Idaigaoka Hasama, 879-5593, Yufu City, Oita, Japan.

Background: It is very rare for clear cell sarcomas (CCS) to arise in the bone. During diagnosis, it is important to distinguish primary CCS of bone from bone metastasis of melanoma because this difference fundamentally changes the therapeutic options. Recently, characteristic fusion genes of CCS have been detected using reverse transcription polymerase chain reaction (RT-PCR) or direct sequencing which allowed to distinguish CCS from melanoma. However, there was no study applying these analyses with positive results. In this case, we describe the use of fusion gene analysis to diagnose a primary CCS of the bone.

Case Presentation: A 36-year-old male presented with a four-months history of left knee pain. Magnetic resonance imaging showed a lesion in the left femoral medial epicondyle. Histological examination of the biopsy specimen revealed proliferating oval or rounded cells. These cells had clear cytoplasm arranged in fascicles or compact nests with frequent deposits of brown pigment. Furthermore, immunohistochemistry analysis revealed that tumor cells were positive for S-100 protein, HMB-45, Melan-A, and SOX10. It stained negative for CD34 and BRAF v600e. Conclusively, detection of the EWSR1/ATF1 fusion gene using RT-PCR and direct sequencing confirmed that the lesion was a primary CCS of the bone. Wide-margin resection and reconstruction with a tumor endoprosthesis were performed.

Conclusions: Herein, we diagnosed a rare case of primary CCS of the bone by detecting EWSR1/ATF1 fusion gene using RT-PCR and direct sequencing. Since fluorescence-in situ hybridization (FISH) and RT-PCR could show false positive by mainly due to technical problems, it is better to perform direct sequencing to confidently diagnose the tumor as a primary CCS especially at very rare site such as bone.
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http://dx.doi.org/10.1186/s12891-021-03969-4DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7819310PMC
January 2021

A Retrospective Study of Superficial Type Atypical Lipomatous Tumor.

Front Med (Lausanne) 2020 17;7:609515. Epub 2020 Dec 17.

Department of Dermatology, University of Occupational and Environmental Health, Kitakyushu, Japan.

Atypical lipomatous tumor (ALT) has been defined as a well-differentiated liposarcoma exhibiting a higher frequency of a local recurrence after surgical resection. ALT is mainly classified into deep type and superficial type. Compared with deep type ALT, superficial type ALT is rarely observed. One of the most important issues is that little has been known about superficial type ALT and it is not easy to predict the presence of superficial type ALT before surgical resection. To clarify the clinical manifestations of superficial type ALT, we examined 15 cases with superficial type ALT and 118 cases with benign lipoma, and analyzed their differences in clinical characteristics and the findings of MRI test. In clinical characteristics, the tumor size of superficial type ALT was significantly greater than that of benign lipoma, and superficial type ALT showed a significantly higher frequency of the tumor size of more than 4 cm. Superficial type ALT exhibited poor tumor mobility and hardness with elastic soft. In addition, a significantly higher frequency of tumor location of superficial type ALT was observed in extremities. Among tumor sites at the trunk, buttocks, and shoulder were high frequent location in superficial type ALT. In an MRI examination, superficial type ALT exhibited a significantly higher frequency of the septal structures compared with benign lipoma. The combinations of clinical characteristics, including physical examinations, MRI, and histological examinations, are helpful for the diagnosis of superficial type ALT.
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http://dx.doi.org/10.3389/fmed.2020.609515DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7774599PMC
December 2020

Primary Site Identification of Soft-Tissue Mass: Things to Know in MRI Assessment.

J Magn Reson Imaging 2020 Sep 18. Epub 2020 Sep 18.

Department of Radiology, University of Occupational and Environmental Health School of Medicine, Kitakyushu, Japan.

The spectrum of soft-tissue mass is varied, including neoplastic and nonneoplastic/inflammatory lesions. However, soft-tissue tumors have similar imaging findings and, therefore, the diagnosis of soft-tissue mass is challenging. Although careful assessment of the internal characteristics on imaging can often narrow the differential diagnoses, the differential diagnosis may be out of the question if identification of the soft-tissue mass origin is missed. The purpose of this article is to review the imaging findings and the essential anatomy to identify the primary site of the soft-tissue mass, and discuss the associated potential pitfalls. In order not to fall into a pitfall, recognition of characteristic imaging findings indicating the origin of the soft-tissue mass and anatomical knowledge of the normal tissue distribution are necessary. LEVEL OF EVIDENCE: 5 TECHNICAL EFFICACY STAGE: 3.
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http://dx.doi.org/10.1002/jmri.27368DOI Listing
September 2020

[Message from the Editor-in-Chief].

Authors:
Masanori Hisaoka

J UOEH 2020;42(3):229-230

Editor-in-Chief, Journal of University of Occupational and Environmental Health, Japan.

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http://dx.doi.org/10.7888/juoeh.42.229DOI Listing
October 2020

Nuclear expression of MDM2 in hibernoma: a potential diagnostic pitfall.

Virchows Arch 2021 Mar 20;478(3):527-534. Epub 2020 Aug 20.

Department of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-Ku, Kitakyushu, 807-8555, Japan.

Hibernoma is a rare benign adipocytic tumor composed of a proliferation of brown and white fat cells varying in their proportions. The tumor may also contain fat cells resembling lipoblasts, which makes it difficult to distinguish it from atypical lipomatous tumor/well differentiated liposarcoma (ALT/WDLS). Although nuclear expressions of murine double minute 2 (MDM2) and cyclin-dependent kinase 4 (CDK4) are widely used as immunohistochemical surrogate markers for ALT/WDLS, the utility of these proteins in distinguishing between hibernoma and ALT/WDLS still remains to be elucidated. We evaluated immunohistochemical expressions of MDM2 and CDK4 in 10 hibernomas expressing uncoupling protein-1 (UCP-1), a mitochondrial protein transporter consistently expressed in brown fat cells, and lacking MDM2 gene amplification, which was analyzed by fluorescence in situ hybridization (FISH). In contrast to the data previously obtained, nuclear expression of MDM2 was observed in 100% (10/10 cases) of the hibernomas irrespective of the proportion of brown fat cells, whereas no cases were positive for CDK4. The tumors also showed almost concurrent expression of p53 (in 9/10 cases) and ubiquitin-specific-processing protease 7 (USP7) (in 10/10 cases), which deubiquitinates and stabilizes MDM2, potentially resulting in its nuclear expression without MDM2 gene amplification. MDM2 expression may thus be a diagnostic pitfall for hibernoma particularly in differentiating it from ALT/WDLS.
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http://dx.doi.org/10.1007/s00428-020-02914-5DOI Listing
March 2021

Desmoplastic Fibroblastoma Invading the Humerus.

Case Rep Orthop 2020 23;2020:9780263. Epub 2020 May 23.

Department of Orthopaedic Surgery, Kyushu Rosai Hospital, Japan.

Desmoplastic fibroblastoma (DFB) is an uncommon, benign, soft tissue tumor. The tumor most often presents as a slowly growing, painless soft tissue mass and is usually small. There have been only a few reports of patients with DFB who presented with bone invasion. Herein, we report the case of a 66-year-old woman with DFB with bone invasion in her left axilla. A lump under the left axilla was detected and was associated with pain and limited range of motion (ROM) of the shoulder. Computed tomography showed a soft tissue mass with invasion of the adjacent left humerus. Magnetic resonance imaging revealed a lesion with low signal intensity on T1- and T2-weighted images, and weak internal enhancement on postcontrast T1-weighted images with fat suppression. Histologic evaluation of a preoperative needle biopsy revealed DFB with FOSL1 expression. The tumor was marginally excised. Postoperative outpatient follow-up demonstrated a significant improvement in pain and ROM of the shoulder and no recurrence after 1 year. Even though DFB with bone invasion can cause pronounced clinical symptoms with pain and limited ROM, we conclude that simple excision is an adequate treatment.
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http://dx.doi.org/10.1155/2020/9780263DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7271290PMC
May 2020

Infantile Lipofibromatosis-like Neural Tumour Investigated by a Fusion Gene Detection Assay.

Acta Derm Venereol 2020 Jun 11;100(13):adv00180. Epub 2020 Jun 11.

Division of Dermatology, Department of Medicine of Sensory and Motor Organs, Faculty of Medicine, Tottori University, 36-1 Nishicho, Yonago, Tottori 683-8504, Japan. E-mail:

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http://dx.doi.org/10.2340/00015555-3542DOI Listing
June 2020

Peritumoral Fat Content Correlates with Histological Prognostic Factors in Breast Carcinoma: A Study Using Iterative Decomposition of Water and Fat with Echo Asymmetry and Least-Squares Estimation (IDEAL).

Magn Reson Med Sci 2021 Mar 6;20(1):28-33. Epub 2020 Mar 6.

Department of Radiology, University of Occupational and Environmental Health School of Medicine.

Purpose: To correlate peritumoral fat content using iterative decomposition of water and fat with echo asymmetry and least-squares estimation (IDEAL) with histologic prognostic factors in breast carcinoma.

Materials And Methods: This study consisted of 100 patients who were diagnosed with invasive carcinoma of breast and underwent breast MRI including IDEAL before surgery. The scan time of IDEAL fat fraction (FF) map imaging was 33 s. Four regions of interests (ROIs), which are a distance of 5 mm from the tumor edge, and one ROI in the mammary fat of the healthy side were set on the FF map. Then average peritumoral FF values (FFt), average FF values in the healthy side (FFh), and peritumoral fat ratio (pTFR: defined as FFt/FFh) were calculated. Histologically, the presence of lymph node metastasis and the MIB-1 index were evaluated.

Results: FFt and pTFR for breast carcinoma with lymph node metastasis (79.27 ± 10.36 and 0.897 ± 0.078) were significantly lower than those without (86.23 ± 4.53 and 0.945 ± 0.032) (P < 0.001 and P = 0.005). Spearman rank correlation suggested that the FFt correlated with the MIB-1 index (r = -340, P = 0.001).

Conclusion: Quantification of peritumoral fat using IDEAL-iron quantification is associated with the histologic prognostic factors, and may be a practical tool for therapeutic strategy of breast carcinoma.
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http://dx.doi.org/10.2463/mrms.mp.2019-0201DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7952210PMC
March 2021

Lanthanum Deposition in the Gastroduodenal Mucosa of Dialysis Patients.

J UOEH 2019 ;41(4):387-395

Department of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, Japan.

Lanthanum (La) carbonate (LC) is one of the most popular phosphate binders used in dialysis patients with end-stage renal disease. Only a small amount of LC is believed to be absorbed from the gastrointestinal (GI) tract because LC strongly binds to dietary phosphate and forms insoluble complexes. La deposition in the gastroduodenal mucosa has been recently identified. Endoscopically, La deposition is demonstrated as whitish lesions of varying sizes and shapes in the gastroduodenal mucosa. Microscopically, La deposition is characterized by histiocytic reaction or small foreign body granulomas containing gray or brown materials mainly in the lamina propria of the GI mucosa. Some histiocytes containing La can migrate into regional lymph nodes via the lymphatic flow. The amount of La deposition in the gastroduodenal mucosa is correlated with the total dose of LC administration, and La deposition is almost consistently observed in LC-treated dialysis patients. Although the detailed mechanism of La deposition in the GI tract is still unclear, several factors, such as gastric pH and metaplastic change of the mucosal epithelium, may be involved in the La deposition in the gastroduodenal mucosa. Here we present an overview of the feature of La deposition in the GI tract.
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http://dx.doi.org/10.7888/juoeh.41.387DOI Listing
August 2020

Alteration in tumoural PD-L1 expression and stromal CD8-positive tumour-infiltrating lymphocytes after concurrent chemo-radiotherapy for non-small cell lung cancer.

Br J Cancer 2019 09 7;121(6):490-496. Epub 2019 Aug 7.

Second Department of Surgery (Chest Surgery), University of Occupational and Environmental Health Japan, Kitakyushu, Japan.

Background: Consolidation treatment with an anti-PD-L1 antibody, durvalumab, following concurrent chemo-radiotherapy (cCRT) has become a new standard of care for locally advanced non-small cell lung cancer (NSCLC). The rationale of PD-L1 blockade after cCRT is based on preclinical evidence suggesting that chemotherapy and radiotherapy up-regulate tumoural PD-L1 expression, which has not been shown in clinical studies.

Methods: To examine alteration in tumoural PD-L1 expression (tumour proportion score, TPS) and density of stromal CD8-positive tumour-infiltrating lymphocytes (CD8 + TILs) after cCRT, paired NSCLC samples obtained before and after cCRT were reviewed in comparison with those obtained before and after drug therapy.

Results: PD-L1 expression was significantly up-regulated after cCRT (median TPS, 1.0 at baseline versus 48.0 after cCRT; P < 0.001), but not after drug therapy. There was no significant correlation between baseline TPS and post-cCRT TPS. CD8 + TIL density was significantly increased after cCRT (median, 10.6 versus 39.1; P < 0.001), and higher post-cCRT CD8 + TIL density was associated with a higher pathologic response and with a favourable survival (P = 0.019).

Conclusion: Tumoural PD-L1 expression was up-regulated after cCRT, which provides pathologic rationale for PD-L1 blockade following cCRT to improve prognosis. Stromal CD8 + TIL density was also increased after cCRT, and higher post-cCRT CD8 + TIL density was a favourable prognostic indicator.
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http://dx.doi.org/10.1038/s41416-019-0541-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6738061PMC
September 2019

Adenocarcinoma of intestinal type of the vulva.

Int Cancer Conf J 2019 Apr 14;8(2):89-93. Epub 2019 Feb 14.

Department of Obstetrics and Gynecology, Steel Memorial Yahata Hospital, Kitakyushu, Japan.

Primary adenocarcinoma of the vulva is a rare disease, and usually arises in a Bartholin gland or occurs in association with Paget's disease. Furthermore, adenocarcinoma of intestinal type of the vulva is an extremely rare neoplasm, and few cases have been reported. The appropriate treatment for optimal prognosis is unclear. We report a case of adenocarcinoma of intestinal type of the vulva occurring in a 63-year-old female. The tumor was found near the urethra, and biopsy specimen showed a proliferation of signet ring cells embedded in an abundant myxoid stroma and irregular tubular structures of atypical columnar epithelium. An extensive workup showed no metastases. Local excision with a 2-cm lesion in the vulva side, bilateral superficial inguinal lymph node dissection and Cloquet lymph node biopsy were performed. Cancer cells contained mucinous materials in the cytoplasm, which exhibited diffuse positive staining for cytokeratin 20 and CDX2. The final pathologic diagnosis was adenocarcinoma of intestinal type of the vulva (pT1bN0M0). The patient received adjuvant external irradiation because of positive urethral surgical margin. She is well 1 year after therapy. Immunohistochemical staining for cytokeratin 20 and polyclonal CDX2 is helpful with investigation of adenocarcinoma of intestinal type, but long-term prognosis remains unclear.
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http://dx.doi.org/10.1007/s13691-019-00361-xDOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6498270PMC
April 2019

Bone Involvement Mimicking an Aggressive Bone Lesion in a Diffuse-type Tenosynovial Giant Cell Tumor in the Thoracic Vertebral Lamina: A Case Report.

J Orthop Case Rep 2018 May-Jun;8(3):14-17

Department of Orthopaedic Surgery, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Introduction: Diffuse-type tenosynovial giant cell tumor (D-TGCT), or pigmented villonodular synovitis, is a benign, but aggressive lesion, primarily involving large joints. The spine is rarely affected, with the involvement of the thoracic spine being particularly rare. Massive bone involvement associated with facet joints is a characteristic of spinal D-TGCT.

Case Report: We report the case of a 26-year-old woman with D-TGCT in her first thoracic vertebral lamina. Computed tomography (CT) showed an osteolytic expansive lesion without the involvement of the facet joint. Magnetic resonance imaging (MRI)revealed a lesion with intermediate signal intensity on T1- and T2-weighted images. After computed tomographically guided biopsy, curettage was performed, and D-TGCTwas diagnosed.

Conclusions: Features of CT and MRI suggested an aggressive bone lesion, and it was challenging to assume D-TGCT, particularly without the involvement of the facet joint.
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http://dx.doi.org/10.13107/jocr.2250-0685.1088DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6298722PMC
December 2018

Unusual Congenital Multiple Clustered Dermatofibroma: First Reported Case on the Face.

Acta Derm Venereol 2019 Mar;99(3):341-342

Division of Dermatology, Department of Medicine of Sensory and Motor Organs, Faculty of Medicine, Tottori University, 36-1 Nishicho, Yonago, Tottori 683-8504, Japan.

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http://dx.doi.org/10.2340/00015555-3109DOI Listing
March 2019

[A Case of Carcinoma Showing Thymus-Like Differentiation (CASTLE) of the Thyroid].

J UOEH 2018;40(3):259-266

Department of Surgery 1, School of Medicine, University of Occupational and Environmental Health, Japan.

The case presented herein was a 70-year-old woman who had no compliant, but had a mass in the lower part of the right lobe of the thyroid detected by ultrasound (US). The US image of the tumor, measuring 13 mm in diameter, showed a low and heterogeneous internal echo level with calcification and an irregular margin. The tumor appeared to extend to the adjacent sternothyroid muscle, and cervical lymph node swelling was detected in a computer tomography (CT) image, but no metastatic lesion was found by positron emission tomography (PET)-CT. In a fine needle aspiration cytology of the tumor, papillary thyroid carcinoma was suggested because of the atypical epithelial cells having some changes other than intranuclear inclusion bodies. A subtotal thyroidectomy and central neck lymph node dissection were performed. The excised tumor was histologically composed of irregular nests or sheets of atypical squamoid epithelial cells with some ductal structures that leached to the sternothyroid muscle and involved the right lower parathyroid gland. Carcinoma showing thymus-like differentiation (CASTLE) was diagnosed histopathologically and immunohistochemically with the following immunohistochemical results: Cluster of differentiation 5 (CD5) (+), tumor protein p63 (p63) (+), KIT proto-oncogene receptor tyrosine kinase (c-KIT(CD117)) (+), thyroglobulin (-), and thyroid transcription factor-1 (TTF-1) (-). CASTLE is a rare carcinoma of the thyroid that architecturally resembles thymic epithelial tumors. Many CASTLE patients have been misdiagnosed as other carcinomas, such as anaplastic carcinoma, poorly differentiated carcinoma or squamous cell carcinoma of the thyroid. Immunohistochemical examination, including CD5 played an important role in the final diagnosis of CASTLE, although the distinction from diagnosis as squamous cell carcinoma or mucoepidermoid carcinoma in Hematoxylin-Eosin staining was challenging in our case. Nodal metastasis and perithyroidal tumor extension of CASTLE can predict its worse prognosis. Thus, at least careful follow-up studies are mandatory in cases of CASTLE.
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http://dx.doi.org/10.7888/juoeh.40.259DOI Listing
March 2019

Dedifferentiated parosteal osteosarcoma of the maxilla: a case report and review of the literature.

J Med Case Rep 2018 Aug 17;12(1):235. Epub 2018 Aug 17.

Department of Dentistry and Oral Surgery, Division of Oral and Maxillofacial Surgery, Keio University, School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo, 160-8582, Japan.

Background: Parosteal osteosarcomas are usually low-grade tumors, however, sometimes they transform to high-grade tumors, which is named dedifferentiation. This phenomenon has been reported in long bones. Recently, we encountered a patient with dedifferentiated parosteal osteosarcoma occurring in the maxilla. Here, we report a first case of dedifferentiated parosteal osteosarcoma of the head and neck region.

Case Presentation: A 45-year-old Japanese woman with a refractory bone lesion in the maxilla presented to our hospital. A biopsy showed atypical spindle cell proliferation involving dedifferentiated high-grade component, which was diagnosed as dedifferentiated parosteal osteosarcoma. Three cycles of neoadjuvant chemotherapy using ifosfamide and pirarubicin were performed followed by sub-total maxillectomy. Histopathological results showed that neoadjuvant chemotherapy was effective for high-grade component. The decision to perform adjuvant chemotherapy (cisplatin and pirarubicin) was made because distant metastasis has been reported, even in cases with dedifferentiated parosteal osteosarcoma in which complete necrosis of high-grade component was achieved due to neoadjuvant chemotherapy. There was no recurrence 15 months after surgery.

Conclusions: Dedifferentiated parosteal osteosarcoma can occur in the head and neck region. Chemotherapy including anthracycline anticancer agent could be effective for high-grade component of dedifferentiated parosteal osteosarcoma.
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http://dx.doi.org/10.1186/s13256-018-1747-3DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6097278PMC
August 2018

MUC4 expression in meningiomas: under-recognized immunophenotype particularly in meningothelial and angiomatous subtypes.

Histopathology 2019 Jan 30;74(2):276-283. Epub 2018 Oct 30.

Department of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, Kitakyushu, Japan.

Aims: MUC4 is a transmembrane glycoprotein that plays a role in cell growth signalling and is expressed in various epithelial tissues. Gene expression profiling and immunohistochemical analyses revealed that MUC4 is also constantly and specifically expressed in low-grade fibromyxoid sarcomas and sclerosing epithelioid fibrosarcomas among the mesenchymal tumours, and immunohistochemical detection of MUC4 is extremely useful for their diagnoses. In our routine pathological practice, we noticed that meningiomas are also often positive for MUC4, which has not yet been reported previously, despite the extensive scrutiny of its expression in soft tissue tumours.

Methods And Results: We examined immunohistochemically the expression of MUC4, progesterone receptor (PgR) and somatostatin receptor 2A (SSTR2A) in 140 meningiomas of various histological subtypes and 123 other mesenchymal tumours, including intracranial or sinonasal tumours and peripheral nerve sheath tumours. MUC4 was expressed in 130 meningiomas (92.9%). MUC4 expression was constant and almost diffuse in meningothelial and angiomatous subtypes, whereas it was limited in 5% or fewer tumour cells or absent in 26 of 28 fibrous meningiomas. All other mesenchymal tumours examined were negative for MUC4. PgR and SSTR2A were expressed in 94 (67.1%) and 134 (95.7%) meningiomas, respectively. Five of six SSTR2A-negative meningiomas focally expressed MUC4.

Conclusions: MUC4 is expressed variably but almost consistently in meningiomas, particularly in meningothelial or angiomatous subtypes. Its immunohistochemical detection is useful to distinguish meningiomas from other intracranial or head and neck mesenchymal tumours, particularly those with epithelioid features. Our study could expand a variety of MUC4-positive mesenchymal tumours.
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http://dx.doi.org/10.1111/his.13730DOI Listing
January 2019

Giant Adrenal Myelolipoma in a Patient without Endocrine Disorder: A Case Report and a Review of the Literature.

Case Rep Surg 2018 11;2018:4854368. Epub 2018 Jun 11.

Department of Surgery 1, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahata-nishi-ku, Kitakyushu 807-8555, Japan.

We herein present a surgically treated case of huge adrenal myelolipoma. A 62-year-old woman presented to our surgical outpatient clinic with a retroperitoneal tumor. A clinical examination revealed an elastic soft, smooth-surfaced, painless, child-head-sized tumor with poor mobility, which was located in the left upper abdomen. Computed tomography (CT) and magnetic resonance imaging (MRI) of the abdomen revealed an uneven tumor surrounding the stomach, spleen, pancreas, and left kidney, which was 20 × 18 × 10 cm in size. The retroperitoneal tumor was resected. The tumor was attached to the surrounding organs, including the pancreas, spleen, and left kidney, but had not directly invaded these organs. The tumor was yellow and elastic soft and covered with a thin film. The origin of the tumor was suggested to be the left adrenal gland. The weight of the excised tumor was 1500 g. The histopathological diagnosis was adrenal myelolipoma. The patient had an uneventful recovery and was discharged from the hospital on the thirteenth day after the operation. She has been followed up in our outpatient clinic.
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http://dx.doi.org/10.1155/2018/4854368DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6016169PMC
June 2018

A unique case of a huge mixed squamous cell and glandular papilloma of non-endobronchial origin with a peripheral growth.

Respir Med Case Rep 2018 4;24:108-112. Epub 2018 May 4.

Departments of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyushu, 807-8555, Japan.

We report a case of a huge solitary non-endobronchial pulmonary tumor in a 76-year-old male smoker. The tumor measured 11 × 10 × 8 cm. It was ill-defined, and it was located periphery of the right lower lobe with the subpleural cystic spaces. He underwent right lower lobectomy with mediastinal lymph node dissection and is free from tumor 30 months after surgery. Microscopically, it was composed of a proliferation of squamous and ciliated columnar epithelial cells with a few mucous cells. These cells were arranged in a papillary growth fashion extending along the fibrously thickened alveolar septa together with metaplastic bronchiolar and squamous epithelia displaying an usual interstitial pneumonia-pattern. Although the histologic features of the tumor were that of a mixed squamous cell and glandular papilloma (MSCGP), it was peripherally located and showed a lepidic growth, and it was much larger than previously reported MSCGPs. It is possible that the tumor developed in association with bronchial metaplasia in the periphery of the lung, and then extended along the surface of the reconstructed air spaces, which resulted in its unique histologic appearance. Further investigations of respiratory papilloma are needed to clarify the pathogenesis of these lesions.
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http://dx.doi.org/10.1016/j.rmcr.2018.05.001DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6010628PMC
May 2018

Presacral myelolipoma as a possible parasymptom of cancer: A case report.

Rare Tumors 2018 23;10:2036361318772124. Epub 2018 Apr 23.

Department of Orthopaedic Surgery, Graduate School of Medicine, Kyoto University, Kyoto, Japan.

Presacral myelolipomas are rare, benign, asymptomatic tumors composed of mature adipose tissue and hematopoietic elements. Presacral myelolipomas can occur in patients with a medical history of malignancy, steroid use, and/or endocrine disorders including diabetes mellitus. A 65-year-old man with no specific medical history experienced temporal abdominal pain without bowel symptoms that lasted a few hours. By the time he visited a hospital, the pain had diminished. Computed tomography failed to detect any abnormality in the abdominal or pelvic organs that would have caused the abdominal pain but revealed a lesion 4 cm in diameter in the frontal sacrum. Magnetic resonance imaging showed that the lesion contained fat elements with a high signal intensity on T1- and T2-weighted images, which was decreased on fat-suppression T2-weighted images. Computed tomography-guided biopsy and imaging allowed a diagnosis of presacral myelolipoma. After 3 months, hematochezia was observed, and follow-up examination revealed rectal carcinoma with multiple lung metastases. He died due to spread of the cancer despite chemotherapy, 6 months after the cancer was found. Considering the possible association between presacral myelolipoma and cancer, presacral myelolipoma might be a cancer parasymptom. Checking for possible malignancy may therefore be warranted in patients with presacral myelolipoma, especially in those without diabetes mellitus.
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http://dx.doi.org/10.1177/2036361318772124DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5946595PMC
April 2018

Major pathologic response to alectinib in ALK-rearranged adenocarcinoma of the lung.

Surg Case Rep 2018 Mar 9;4(1):19. Epub 2018 Mar 9.

Second Department of Surgery (Chest Surgery), University of Occupational and Environmental Health Japan, Kitakyusyu, Japan.

Background: Alectinib is a highly selective tyrosine kinase inhibitor of anaplastic lymphoma kinase (ALK) and provided a significantly prolonged progression-free survival compared with chemotherapy in patients with advanced non-small cell lung cancer (NSCLC) harboring rearrangements of the ALK gene. Here, we present the first surgical case of ALK-rearranged lung adenocarcinoma with major pathological response in resected specimens after treatment with alectinib.

Case Presentation: A 65-year-old female with clinical stage IIIA-N2 ALK-rearranged adenocarcinoma originating from the left lower lobe presented. Involvement of lower para-tracheal node was pathologically confirmed by endobronchial ultrasound-guided biopsy. Alectinib was prescribed, as the patient may not tolerate radiotherapy due to a mental illness. After 3 months' treatment with alectinib, a remarkable radiological and metabolic response was achieved. The patient did not tolerate further continuation of alectinib treatment, and surgery was performed without any morbidity. Only < 10% tumor cells were viable in all resected specimens, indicating major pathological response to alectinib.

Conclusions: Salvage surgery after alectinib treatment may be safe and effective for initially unresectable NSCLC harboring ALK-rearrangements.
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http://dx.doi.org/10.1186/s40792-018-0430-7DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5845085PMC
March 2018

Inhibition of WNT/β-catenin signaling under serum starvation and hypoxia induces adipocytic transdifferentiation in human leiomyoma cells.

Lab Invest 2018 04 18;98(4):439-448. Epub 2018 Jan 18.

Department of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, Kitakyushu, Japan.

Fatty metamorphosis is an uncommon alteration in uterine leiomyoma (i.e., lipoleiomyoma), and the pathogenetic mechanisms underlying this phenomenon remain poorly understood. Because a conditional deletion of β-catenin, a major transducer of the canonical Wingless/integrated (WNT) pathway, in the developing mouse uterus can induce adipogenesis in the myometrium, it is hypothesized that inhibition of the WNT/β-catenin signaling may be also involved in the development of fat cells within uterine leiomyoma. In the current study, which was performed to address this point, intracytoplasmic lipid droplets were detectable in cultured human leiomyoma cells by treatment with a potent tankyrase inhibitor, XAV939, which antagonizes β-catenin, in a serum-starved culture medium without additional adipogenesis-inducing agents or supplements, and showed increasing accumulation in a time-dependent manner. In addition, the induction of fat cells was greatly enhanced under hypoxic conditions (i.e., 2.5% O)-recapitulating the local in vivo situation of uterine leiomyoma-in comparison to that under normoxic conditions (i.e., 21% O). The marker genes of differentiated fat cells such as ADIPOQ and PLIN were highly expressed in leiomyoma cells that were treated with XAV939 under hypoxia and serum starvation, whereas the immunohistochemical expression of desmin-a cytoskeletal protein representing smooth muscle differentiation-was downregulated, which appears in line with the switch in differentiation. The results of our study suggest that the inhibition of canonical WNT/β-catenin signaling under the stress due to hypoxia and serum starvation can initiate adipocytic transdifferentiation or metaplasia in human uterine leiomyoma cells, which is potentially related to the development of lipoleiomyoma.
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http://dx.doi.org/10.1038/s41374-017-0020-5DOI Listing
April 2018

Lanthanum-Induced Mucosal Alterations in the Stomach (Lanthanum Gastropathy): a Comparative Study Using an Animal Model.

Biol Trace Elem Res 2018 Sep 4;185(1):36-47. Epub 2018 Jan 4.

Department of Pathology and Oncology, School of Medicine, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyushu, 807-8555, Japan.

Lanthanum (La) carbonate (LC) is one of the most potent phosphate binders that prevents the elevation of serum phosphate levels in patients with end-stage renal diseases undergoing dialysis. LC binds strongly to dietary phosphate and forms insoluble complexes that pass through the gastrointestinal tract. La deposition in patients treated with LC is a recently documented finding particularly observed in gastric mucosa. We herein describe the detailed gastric mucosal lesions in 45 LC-treated patients and address the potential underlying pathologic mechanism using oral LC administration in rats. Microscopically, La deposition, as shown by subepithelial collections of plump eosinophilic histiocytes or small foreign body granulomas containing coarse granular or amorphous inclusion bodies, was found in the gastric mucosa of 44 (97.8%) of the 45 dialysis patients in the study cohort, which was most frequently associated with foveolar hyperplasia (37.8%). Using oral administration of rats with 1000 mg/day LC for 2 or more weeks, La deposition was consistently detectable in the gastric mucosa but not in other organs examined. In addition, various histologic alterations such as glandular atrophy, stromal fibrosis, proliferation of mucous neck cells, intestinal metaplasia, squamous cell papilloma, erosion, and ulcer were demonstrated in the rat model. Thus, orally administered LC can induce mucosal injury, designated here as La gastropathy, which may alter the local environment and result in La deposition in the gastric mucosa, thereby potentially inducing abnormal cell proliferation or neoplastic lesions.
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http://dx.doi.org/10.1007/s12011-017-1235-4DOI Listing
September 2018

Sclerosing Mucoepidermoid Carcinoma in the Parotid Gland With CRTC1-MAML2 Fusion: A Case Report.

Int J Surg Pathol 2018 May 23;26(3):250-255. Epub 2017 Nov 23.

1 University of Occupational and Environmental Health, Kitakyushu, Japan.

In this article, we report a case of sclerosing mucoepidermoid carcinoma (MEC) arising in the parotid gland, with CRTC1-MAML2 gene fusion. A 73-year-old woman with a mass in the right parotid region was referred to our hospital. Radiological imaging tests revealed a well-defined mass, measuring 25 mm in diameter, with foci of calcification in the deep lobe of the parotid gland, extending to the parapharyngeal space. Microscopically, the tumor was composed of a proliferation of atypical glandular epithelial cells having intracytoplasmic mucin, squamoid cells, and intermediate cells arranged in nests embedded in a fibrosclerotic stroma, associated with a dense chronic inflammatory infiltrate containing immunoglobulin G4-immunoreactive plasma cells. Reverse transcription-polymerase chain reaction analysis using a formalin-fixed, paraffin-embedded tumor tissue specimen revealed the CRTC1-MAML2 fusion gene transcript. This is the first report of sclerosing MEC with the detection of the MEC-associated fusion gene, reinforcing a common genetic association between MEC and sclerosing MEC.
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http://dx.doi.org/10.1177/1066896917742721DOI Listing
May 2018

Demographics, management and treatment outcomes of benign and malignant retroperitoneal tumors in Japan.

Int J Urol 2018 01 9;25(1):61-67. Epub 2017 Oct 9.

Department of Urology, University of Occupational and Environmental Health, Kitakyushu, Japan.

Objectives: To show the demographics, type of treatment and clinical outcomes of patients with retroperitoneal tumors in Japan.

Methods: We carried out a retrospective analysis of patients with retroperitoneal tumors treated between 2000 and 2012 at 12 university hospitals in Japan. Histology was re-evaluated using the 2013 World Health Organization classification.

Results: A total of 167 patients were included in the analysis. The number of diagnosed patients increased over the 12-year study period. Liposarcoma and schwannoma were the most common histological types among intermediate/malignant and benign tumors, respectively. The intermediate/malignant tumors were larger and were more frequently found in older people. Surgical resection was the primary treatment for 151 patients. The median survival duration for patients with malignant tumors was 91 months, and was significantly shorter than that for patients with benign and intermediate tumors (P < 0.01). R2 resection was associated with significantly shorter survival than R0/R1 resection for malignant tumors (P < 0.01), but not for intermediate. Grossly complete resection of the recurrent tumors improved survival.

Conclusion: The number of patients diagnosed with retroperitoneal tumors increased over time. R2 resection of primary tumors was found to be associated with poor prognosis in malignant tumors, but not in intermediate tumors. Complete surgical resection of recurrent tumors was associated with a better oncological outcome.
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http://dx.doi.org/10.1111/iju.13469DOI Listing
January 2018

Fibroblastic connective tissue nevus: the role of histopathological and molecular techniques in differential diagnosis.

Eur J Dermatol 2017 10;27(5):547-548

Department of Medicine of Sensory and Motor Organs, Division of Dermatology, Faculty of Medicine, Tottori University, 86 Nishi-Cho, Yonago, Tottori 683-8503, Japan.

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http://dx.doi.org/10.1684/ejd.2017.3093DOI Listing
October 2017

Clear cell sarcoma-like tumor of the gastrointestinal tract: A clinicopathological review.

Pathol Int 2017 10 9;67(10):534-536. Epub 2017 Sep 9.

Department of Pathology, Kanagawa Cancer Center, 2-3-2 Nakao, Asahi-ku, Yokohama-shi, Kanagawa, 241-0815, Japan.

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http://dx.doi.org/10.1111/pin.12573DOI Listing
October 2017