Publications by authors named "Hajime Horiuchi"

66 Publications

Pleural nodule with osteal protrusion anterior to the rib tubercle: a case report.

Radiol Case Rep 2021 Aug 8;16(8):2091-2094. Epub 2021 Jun 8.

Department of Pathology, NTT Medical Center Tokyo, Tokyo, Japan.

Pleural soft tissue density nodules with osteal protrusions are often detected at their characteristic site, the anterior portion of the rib tubercle. Herein, we report a pathologically proven case of this lesion. A 65-year-old man underwent surgery for primary lung cancer in the right middle lobe. Preoperative computed tomography showed a 10-mm pleural soft tissue density nodule with osteal protrusion anterior to the tubercle of the right sixth rib, and this lesion was concomitantly resected. Intraoperative findings showed that this pleural lesion originated from the parietal pleura and was pathologically diagnosed as a benign fibrous tissue. We can avoid unnecessary invasive examinations such as biopsies by recognizing these benign fibrous lesions.
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http://dx.doi.org/10.1016/j.radcr.2021.05.026DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC8203593PMC
August 2021

Histopathological heterogeneity in an atypical type A thymoma variant with pulmonary metastases.

Pathol Int 2021 Jun 3;71(6):438-440. Epub 2021 Mar 3.

Department of Pathology, NTT Medical Center Tokyo, Tokyo, Japan.

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http://dx.doi.org/10.1111/pin.13089DOI Listing
June 2021

Clinicopathologic Characteristics of Esophageal Ectopic Sebaceous Glands: Chronological Changes and Immunohistochemical Analysis.

Int J Surg Pathol 2021 Jun 26;29(4):378-384. Epub 2020 Aug 26.

NTT Medical Center Tokyo, Tokyo, Japan.

Esophageal ectopic sebaceous glands are very rare lesions. A series of 5 cases in a single report has been the maximum number described in the English literature to date. We conducted a clinicopathologic study of 8 cases of esophageal ectopic sebaceous glands. The median patient age at the time of diagnosis was 60 years (range, 50-71 years), and 7 of the 8 patients were male. A focal lesion was observed in 7 cases, whereas 1 case exhibited multiple lesions throughout the esophagus. Four patients had previously undergone upper gastrointestinal endoscopy; in 3 patients, the focal lesion had not been detected. After diagnosis, 3 cases showed spontaneous regression at least once. Immunohistochemically, sebocytes of all 8 cases were negative for the estrogen receptor (ER) and the progesterone receptor (PgR), whereas sebocytes of 5 cases were positive for the androgen receptor (AR). Basal/parabasal cells were positive for AR, ER, and PgR in 5, 7, and 4 cases, respectively. GATA3 was expressed in the sebocytes and basal/parabasal cells of 6 out of 7 available cases, whereas all of 7 available cases were negative for mammaglobin and GCDFP15. Our report provides the basic clinicopathologic characteristics of esophageal ectopic sebaceous glands by the largest case series reported in English literature to date. Furthermore, the chronological changes, particularly spontaneous regression, and immunohistochemical expression of hormone receptors and GATA3 are compatible with lesions resulting from congenital misplacement under hormonal regulation. Therefore, they seem to be congenital misplacements detectable as a result of hormonal stimulated growth.
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http://dx.doi.org/10.1177/1066896920951844DOI Listing
June 2021

Cisplatin plus pemetrexed therapy and subsequent immune checkpoint inhibitor administration for malignant peritoneal mesothelioma without pleural lesions: Case report.

Medicine (Baltimore) 2020 May;99(22):e19956

Division of Respirology.

Rationale: Malignant peritoneal mesothelioma is a rare tumor with a poor prognosis and has no recommended therapy after first-line pemetrexed and platinum-based chemotherapy. Moreover, effects of immune checkpoint inhibitors on peritoneal mesothelioma remains to be elucidated. We herein report the case of a 75-year-old man with peritoneal mesothelioma treated with cisplatin plus pemetrexed and subsequent nivolumab.

Patient Concerns: A 75-year-old man was referred to our hospital due to lower abdominal pain.

Diagnosis: Positron emission tomography-computed tomography (CT) showed the accumulation of fluorodeoxyglucose in an intraperitoneal mass. A histological examination of a laparoscopic biopsy specimen revealed malignant peritoneal mesothelioma.

Interventions: After 4 cycles of cisplatin plus pemetrexed and 13 subsequent cycles of pemetrexed maintenance therapy showed beneficial responses until CT revealed liver metastasis. Nivolumab was then administered as the second-line therapy.

Outcomes: After 3 cycles of biweekly nivolumab administration, he developed severe abdominal distention. CT revealed an intraperitoneal mass growing much more rapidly than ever, indicating hyperprogressive disease after nivolumab treatment. He ultimately died 51 days after the initial nivolumab administration.

Lessons: To our knowledge, this is the first report of hyperprogressive disease in a case of peritoneal mesothelioma after nivolumab treatment. While immune checkpoint inhibitors may be promising therapeutic strategies for treating malignant peritoneal mesothelioma, careful monitoring must be practiced with their application.
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http://dx.doi.org/10.1097/MD.0000000000019956DOI Listing
May 2020

A Novel Role of Interleukin 13 Receptor alpha2 in Perineural Invasion and its Association with Poor Prognosis of Patients with Pancreatic Ductal Adenocarcinoma.

Cancers (Basel) 2020 May 20;12(5). Epub 2020 May 20.

Tumor Vaccines and Biotechnology Branch, Division of Cellular and Gene Therapies, Center for Biologics Evaluation and Research, Food and Drug Administration, Silver Spring, MD 20993, USA.

Perineural invasion (PNI) is one of the major pathological characteristics of pancreatic ductal adeno-carcinoma (PDAC), which is mediated by invading cancer cells into nerve cells. Herein, we identify the overexpression of Interleukin-13 Receptor alpha2 (IL-13Rα2) in the PNI from 236 PDAC samples by studying its expression at the protein levels by immunohistochemistry (IHC) and the RNA level by in situ hybridization (ISH). We observe that ≥75% samples overexpressed IL-13Rα2 by IHC and ISH in grade 2 and 3 tumors, while ≥64% stage II and III tumors overexpressed IL-13Rα2 (≥2+). Interestingly, ≥36 % peripancreatic neural plexus (PL) and ≥70% nerve endings (Ne) among PNI in PDAC samples showed higher levels of IL-13Rα2 (≥2+). IL-13Rα2 +ve PL and Ne subjects survived significantly less than IL-13Rα2 -ve subjects, suggesting that IL-13Rα2 may have a unique role as a biomarker of PNI-aggressiveness. Importantly, IL-13Rα2 may be a therapeutic target for intervention, which might not only prolong patient survival but also help alleviate pain attributed to perineural invasion. Our study uncovers a novel role of IL-13Rα2 in PNI as a key factor of the disease severity, thus revealing a therapeutically targetable option for PDAC and to facilitate PNI-associated pain management.
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http://dx.doi.org/10.3390/cancers12051294DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7281570PMC
May 2020

Progressively increasing density of the solid center of a ground-glass nodule in a solitary pulmonary capillary hemangioma: A case report.

Pathol Int 2020 Aug 5;70(8):568-573. Epub 2020 May 5.

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Tokyo, Japan.

Solitary pulmonary capillary hemangiomas (SPCHs) are recently recognized, rare benign lesions that form solitary nodules owing to capillary proliferation. These lesions are usually detected incidentally as small ground-glass nodules (GGNs) on computed tomography (CT), and progressively enlarge over time. The radiological distinction from peripheral lung cancers is particularly challenging. However, to date, there have been no reports on progressive changes in the central density of SPCH on CT. An asymptomatic 49-year-old man was referred to our hospital for an abnormal shadow that was detected on chest CT during medical check-up. He was subsequently followed-up with chest CT. The nodule increased in size, and the central area became progressively denser. He underwent surgery 5 years and 10 months after the first visit owing to suspicion of lung cancer. Despite the collapse of the surgical specimen by artifacts, histopathological examination revealed a diagnosis of SPCH; collagenous fibers were found in the walls of the intralesional capillaries. The patient is presently alive without any recurrence, 6 months after the operation. In this case, the SPCH demonstrated a GGN with progressively increasing density of the central solid area on the CT. This remarkable feature made the preoperative distinction from lung cancer particularly difficult.
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http://dx.doi.org/10.1111/pin.12945DOI Listing
August 2020

Colonic Neurogenic Lesion: An Admixture of Mucosal Neurofibromatous Lesion and Submucosal Ganglioneuromatous Lesion With Transition.

Int J Surg Pathol 2020 Aug 6;28(5):563-568. Epub 2020 Feb 6.

NTT Medical Center Tokyo, Tokyo, Japan.

Benign neural tumors or tumor-like lesions are rarely detected in the gastrointestinal tract. In this article, we present the case of a neural lesion of the sigmoid colon, which was incidentally detected in a 68-year-old man treated with laparoscopic low anterior resection for an advanced carcinoma of the rectosigmoid junction. Within the resected specimen, a submucosal tumor-like protruding lesion was found in the sigmoid colon. Histologically, the growth was composed of mucosal neurofibromatous and submucosal ganglioneuromatous lesions, between which there was transition. Immunohistochemical analysis revealed a rupture of the perineurium in the area of transition, along with a proliferation of Schwann cells and supporting cells extending into the deep mucosa. This transition indicated that the mucosal and submucosal lesions comprised a single lesion, and that a diagnosis of neurofibroma or ganglioneuroma would be inadequate in this case. Because we could not classify it as an established single entity, we diagnosed the mass as an unclassifiable colonic neurogenic lesion. In summary, we report the case of an extremely rare occurrence of an unclassifiable colonic neurogenic lesion comprising an admixture of transitioning mucosal neurofibromatous and submucosal ganglioneuromatous lesions.
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http://dx.doi.org/10.1177/1066896920904158DOI Listing
August 2020

Arteritis and Plexiform Lesion in Intralobar Pulmonary Sequestration: The First Case With Such Two Distinct Complex Lesions Associated With Local Pulmonary Hypertension.

Int J Surg Pathol 2020 May 6;28(3):321-324. Epub 2019 Nov 6.

NTT Medical Center Tokyo, Tokyo, Japan.

In intralobar pulmonary sequestrations, vascular changes similar to those in pulmonary hypertension (PH) are generally observed, such as intimal proliferation and plexiform lesions. However, to our knowledge, a sequestrated lung manifesting vascular changes with both arteritis and a plexiform lesion has never been reported. A 25-year-old man was diagnosed with intralobar pulmonary sequestration. Pathologically, both arteritis and a plexiform lesion were observed in the sequestrated lung. Systemic vasculitis syndrome was clinically excluded, and the pathological findings appeared to be associated with local PH. Arteritis is an extremely rare finding; only one case of arteritis associated with local PH has been reported in intralobar sequestration. In this case, the artery near the plexiform lesion had milder inflammation and fibrosis, suggesting that the arteritis formed prior to the plexiform lesion. This is the first case of arteritis and a plexiform lesion co-occurring in intralobar pulmonary sequestration associated with local PH. This case may shed light on the formation of plexiform lesions and their association with arteritis.
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http://dx.doi.org/10.1177/1066896919886663DOI Listing
May 2020

Association Between Helicobacter pylori Infection and Short-segment/Long-segment Barrett's Esophagus in a Japanese Population: A Large Cross-Sectional Study.

J Clin Gastroenterol 2020 May/Jun;54(5):439-444

Gastroenterology.

Goal: The goal of this study was to investigate the relationship between Helicobacter pylori (H. pylori) infection and short-segment and long-segment Barrett's esophagus (SSBE and LSBE).

Background: H. pylori infection is reported to be inversely associated with Barrett's esophagus (BE) in western countries. However, the impact of BE segment length on the association between BE and H. pylori infection has scarcely been investigated.

Materials And Methods: The study subjects were 41,065 asymptomatic Japanese individuals who took medical surveys between October 2010 and September 2017. Using this large database of healthy Japanese subjects, we investigated the association between H. pylori infection and SSBE/LSBE. We used multivariable logistic regression analysis to estimate odds ratios (ORs) and 95% confidence intervals (CIs).

Results: Among the study subjects, 36,615 were eligible for the analysis. H. pylori seropositivity was significantly associated with a lower rate of LSBE (OR: 0.42; 95% CI: 0.16-0.91) and a higher rate of SSBE (OR: 1.66; 95% CI: 1.56-1.78) after multivariate adjustment. In the subgroup analysis, H. pylori seropositivity was significantly associated with a high rate of SSBE in subjects without reflux esophagitis (RE) (OR: 1.73; 95% CI: 1.61-1.85). However, H. pylori seropositivity was not associated with SSBE in subjects with RE (OR: 1.07; 95% CI: 0.84-1.37).

Conclusion: In a Japanese population, H. pylori infection was inversely associated with LSBE but significantly associated with SSBE only in subjects without RE. H. pylori may be a risk factor for SSBE, especially in individuals without RE.
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http://dx.doi.org/10.1097/MCG.0000000000001264DOI Listing
September 2019

Myocardial calcification in a patient with B-lymphoblastic leukemia accompanied by tumor lysis syndrome.

Cardiovasc Pathol 2019 Nov - Dec;43:107146. Epub 2019 Aug 7.

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Tokyo, Japan; Faculty of Healthcare, Tokyo Healthcare University, Tokyo, Japan.

Myocardial calcification, a rare disease that leads to chronic or acute heart failure and with a poor prognosis, occurs in patients with abnormal calcium-phosphorus metabolism. The association between myocardial calcification and tumor lysis syndrome has not been reported to date. A 50-year-old man with hyperthermia and general malaise presented to our hospital and was clinically diagnosed with B-lymphoblastic leukemia (B-ALL) and febrile neutropenia accompanied by septic shock. Prednisolone was administered for tumor reduction. Two to three hours later, electrocardiography demonstrated ST elevation in V4-6, and blood tests showed elevated levels of cardiac enzymes. Transthoracic echocardiogram revealed diffuse severe hypokinesis with decreased left ventricular ejection fraction. Additionally, blood tests showed that serum phosphorus level increased to 8.0 mg/dl, which was likely due to tumor lysis syndrome. Circulatory and respiratory failure due to left heart failure progressed, and he died 3 days after administration of prednisolone. Pathological autopsy revealed diffuse proliferation of atypical B-lymphoblasts in the bone marrow, which led to the pathological diagnosis of B-ALL, accompanied by necrosis. On the cut surface of the heart, the left ventricle was dilated, and patchy yellowish-brown areas were present in the epicardial-side of the myocardium and spread through the circumferential wall of the left ventricle and interventricular septum. Microscopically, myocardial fibers were granularly basophilic in that area and were revealed as calcium deposits by Von Kossa staining. He was diagnosed with myocardial calcification. The drastic increase in the serum phosphorus level caused by tumor lysis syndrome seemed to be associated with myocardial calcification.
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http://dx.doi.org/10.1016/j.carpath.2019.07.005DOI Listing
February 2020

Intrahepatic Carcinosarcoma With Cholangiocarcinoma Elements and Prominent Bile Duct Spread.

Int J Surg Pathol 2019 Dec 17;27(8):900-906. Epub 2019 Jun 17.

NTT Medical Center Tokyo, Tokyo, Japan.

Carcinosarcomas with elements of cholangiocarcinoma and sarcoma are rare and have a poor prognosis. The spreading pattern and radiological findings of these lesions remain unclear. A 74-year-old man presented with a high γ-glutamyl transferase level. Magnetic resonance imaging revealed dilation of the right intrahepatic and common bile ducts, consistent with an intraductal papillary neoplasm of the bile duct (IPNB), and diffusion-weighted imaging (DWI) indicated an area of high signal intensity in the intrahepatic bile duct. Bile duct biopsy yielded a small amount of atypical spindle cells, and the patient underwent a right hepatectomy. Microscopically, the tumor contained cholangiocarcinoma and sarcomatous components, including osteosarcoma and leiomyosarcoma, leading to a diagnosis of intrahepatic carcinosarcoma. The tumor spread primarily through the intrahepatic bile duct. An accurate radiological diagnosis of carcinosarcoma was challenging, given the apparent similarities with IPNB. The findings from DWI and pathology of a bile duct biopsy may assist with preoperative diagnosis.
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http://dx.doi.org/10.1177/1066896919855766DOI Listing
December 2019

Aortogenic Embolic Stroke Diagnosed by a Pathological Examination of Endovascularly Removed Thrombus: An Autopsy Report.

Intern Med 2019 Oct 7;58(19):2851-2855. Epub 2019 Jun 7.

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Japan.

Complex aortic atheroma is a high-risk factor for recurrent embolic stroke. An accurate identification of stroke etiology is clinically important; however, it can be challenging. A 91-year-old man with atrial fibrillation was diagnosed with cardioembolic stroke and treated with mechanical thrombectomy. The removed thrombus microscopically contained foamy cells, suggesting an atheroembolism. An autopsy revealed an atherosclerotic lesion with ulceration, located in the aortic arch. At the lesion, the plaque had microscopically ruptured into the lumen. We therefore concluded that the aortic atherosclerotic lesion was the embolic source. Removed thrombi should be pathologically examined even if a cardioembolic stroke is clinically suspected.
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http://dx.doi.org/10.2169/internalmedicine.2857-19DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6815896PMC
October 2019

Enterocolic lymphocytic phlebitis with marked myointimal hyperplasia and perivenous concentric fibrosis.

Cardiovasc Pathol 2019 May - Jun;40:68-71. Epub 2019 Mar 6.

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Tokyo, Japan; Faculty of Healthcare, Tokyo Healthcare University, Tokyo, Japan.

Enterocolic lymphocytic phlebitis (ELP) is a rare enteropathy characterized by lymphocytic phlebitis of the mesenteric veins without arteritis. Idiopathic myointimal hyperplasia of mesenteric veins (IMHMV) is a rare disease similar to ELP, characterized by myointimal hyperplasia that constricts the lumen of veins, causing mucosal injury. A 62-year-old man with chief complaint of abdominal pain was treated by partial resection of the ileum after 3 months of conservative therapy. The pathologic diagnosis was ELP with prominent myointimal hyperplasia. Histologically, the lesion consisted of lymphocytic infiltration into the vein accompanied by prominent myointimal hyperplasia and perivenous concentric fibrosis, which are characteristics shared by ELP and IMHMV. The observations in this case suggest that some of ELP and IMHMV may belong to the same disease spectrum. Furthermore, perivascular concentric fibrosis was a remarkable observation that may contribute to differential diagnosis between ELP and "true" IMHMV.
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http://dx.doi.org/10.1016/j.carpath.2019.02.006DOI Listing
July 2019

Intramucosal colorectal carcinoma with lymphovascular invasion: clinicopathological characteristics of nine cases.

Histopathology 2019 Jun;74(7):1055-1066

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Tokyo, Japan.

Aims: Recent studies have provided the concept of invasive intramucosal colorectal carcinoma (CRC), and a case of intramucosal CRC with lymphatic invasion has been reported; however, the characteristics of such cases and the risk of lymph node metastasis have never been investigated. Therefore, we aimed to assess the pathological characteristics of intramucosal CRCs with lymphovascular invasion as well as the possibility of lymph node metastasis as an indication for additional surgery.

Methods And Results: To delineate the histological features of intramucosal CRCs with lymphovascular invasion, we analysed several histological features and compared their incidence among nine such cases, as well as 20 other cases of intramucosal CRCs without lymphovascular invasion. High-grade tumour budding and a pattern of 'eosinophilic cytoplasm and round nuclei with inflammatory reaction (ERI)' were morphological characteristics of intramucosal CRCs with lymphovascular invasion, compared with those without lymphovascular invasion (both P < 0.05). Among the seven lymph node-dissected cases of intramucosal CRCs with lymphovascular invasion, none showed lymph node metastasis.

Conclusions: In intramucosal CRCs with lymphovascular invasion, high-grade tumour budding and the 'ERI' pattern are morphological characteristics that are distinct from those of non-invasive CRC, which is synonymous with high-grade dysplasia. Further studies using a larger number of cases by focusing on the above-mentioned histological pattern are expected to clarify the potential of lymph node metastasis of such cases.
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http://dx.doi.org/10.1111/his.13826DOI Listing
June 2019

Colon Metastasis From Microscopic Serous Carcinoma of the Fallopian Tube Fimbria Mimicking a Primary Colon Cancer.

Int J Surg Pathol 2019 Jun 20;27(4):390-395. Epub 2019 Jan 20.

1 NTT Medical Center Tokyo, Tokyo, Japan.

Metastatic diseases rarely develop in the colorectum, and diagnosing colorectal metastasis by biopsy without history of a malignant tumor or clinical information of a primary tumor is challenging. A 65-year-old woman with a 6-month history of constipation and diarrhea was admitted to our hospital and diagnosed with rectosigmoid colonic micropapillary carcinoma. Low anterior resection was performed after neoadjuvant chemotherapy. Because the lipoleiomyoma in the uterus obstructed the operator's vision, total hysterectomy and bilateral salpingo-oophorectomy were performed. Examination of the colon and adnexa, together with immunohistochemical studies, revealed that the colonic tumor was actually serous carcinoma that had metastasized from the left fimbria of the fallopian tube. Retrospective immunohistochemical examination of the colon biopsy specimen suggested carcinoma with a Müllerian immunophenotype. When a colon biopsy reveals carcinoma with an invasive micropapillary pattern without a component of conventional tubular adenocarcinoma, immunohistochemical examination should be performed to rule out the possibility of metastasis.
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http://dx.doi.org/10.1177/1066896918824028DOI Listing
June 2019

Cerebral Embolism Caused by Thrombus in the Pulmonary Vein Stump after Left Lower Lobectomy: A Case Report and Literature Review.

Intern Med 2019 May 18;58(9):1349-1354. Epub 2018 Dec 18.

Department of Cerebrovascular Medicine, NTT Medical Center Tokyo, Japan.

Cerebral embolism after left upper lobectomy caused by a thrombus in the pulmonary vein stump (PVS) is a serious complication. However, it is unclear if cerebral embolism can develop after other types of lobectomy. We present a case of a 68-year-old man with cerebral embolism after left lower lobectomy with a longer PVS than normal. There were no clinically suspected sources for the thrombus except for the PVS. This thrombus seemed to have formed in the PVS. The endovascularly removed thrombus contained scattered nuclear debris around neutrophils, suggesting a physiological response caused by tissue injury.
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http://dx.doi.org/10.2169/internalmedicine.1962-18DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6543232PMC
May 2019

An estrogen receptor-positive locally aggressive smooth muscle neoplasm of the transverse colon: A case report.

Medicine (Baltimore) 2018 Nov;97(46):e13250

Department of Diagnostic Pathology, NTT Medical Center Tokyo.

Rationale: Extrauterine leiomyomas (LMs) in women are often positive for the estrogen receptor (ER); however, almost all extrauterine leiomyosarcomas (LMSs) are negative for ER. Invasive smooth muscle neoplasms (SMNs) of the gastrointestinal tract walls are very rare and those ER statuses have not been well studied.

Patient Concerns: A 48-year-old woman presented to our hospital with a 10 years history of recurrent severe abdominal pain and diarrhea lasting about an hour, with frequency of about twice per year. She was clinically diagnosed with a submucosal tumor (SMT) of the transverse colon and underwent a partial transverse colectomy.

Diagnosis: A colonoscopy revealed a 30-mm SMT in the transverse colon. A contrast abdominal computed tomography detected a 21-mm mass with significant late phase enhancement in the transverse colon and the lesion was clinically diagnosed as an SMT. Post-operative pathology confirmed a diagnosis of ER-positive locally aggressive SMN.

Interventions: The patient underwent laparoscopic partial transverse colectomy.

Outcomes: The patient received no adjuvant therapy postoperatively. The patient has remained disease-free without recurrence 13 months after the surgery.

Lessons: This is the first case of an ER-positive invasive SMN in the gastrointestinal tract. It highlights the difficulty in classifying some gastrointestinal SMNs as either LMs or LMSs and the importance of ER status in SMNs.
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http://dx.doi.org/10.1097/MD.0000000000013250DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6257419PMC
November 2018

Extended thoracotomy with subcostal incision for giant solitary fibrous tumor of the diaphragm.

AME Case Rep 2017 30;1. Epub 2017 Nov 30.

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Tokyo, Japan.

Solitary fibrous tumors of the pleura (SFTPs) are rare spindle cell neoplasms. The standard treatment is complete resection, but this may be challenging if the tumor is extremely large and originates from the diaphragm. We experienced a case of a giant solitary fibrous tumor originating from the diaphragm. A 74-year-old man presented with an asymptomatic giant mass on the right diaphragm suspicious of a solitary fibrous tumor. We performed a subcostal incision following posterolateral thoracotomy for complete resection. This surgical approach provided a better field around the diaphragm and facilitated radical and safe complete resection. The pathological diagnosis was a malignant solitary fibrous tumor. The patient survived for 1 year postoperatively without recurrence. We conclude that extended thoracotomy combined with a subcostal incision is a useful approach for surgical removal of giant tumors of the diaphragm.
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http://dx.doi.org/10.21037/acr.2017.11.02DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6155638PMC
November 2017

Thrombus Reformation in the Pulmonary Vein Stump Confirmed 16 Months After Cerebral Embolism on the Day After Left Upper Lobectomy for Lung Cancer.

J Stroke Cerebrovasc Dis 2018 Oct 2;27(10):e225-e227. Epub 2018 Aug 2.

Department of Cerebrovascular Medicine, NTT Medical Center Tokyo, Tokyo, Japan. Electronic address:

Cerebral embolism is a very serious complication after lung cancer surgery. In such cases, cerebral embolism is caused by a thrombus formed in the pulmonary vein stump. Most such cases have been reported to occur within 10 days after left upper lobectomy. The patients were treated with anticoagulation therapy to prevent the recurrence of cerebral embolism, and recurrence or thrombus reformation has not been reported to the best of our knowledge. We present a 68-year-old man with a cerebral embolism detected on the day after left upper pulmonary lobectomy for lung cancer. The patient was treated with unfractionated heparin and his neurological symptoms improved. Heparin treatment was subsequently changed to aspirin for the prevention of recurrence; however, thrombus formation in the vein stump was asymptomatically confirmed 16 months after the surgery by contrast-enhanced computed tomography. This is the first case to our knowledge of thrombus reformation in the pulmonary vein stump after a cerebral embolism associated with lung cancer surgery. In our case, anticoagulation therapy was not continued to prevent recurrence, and antiplatelet therapy was performed instead, which might be associated with the thrombus reformation.
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http://dx.doi.org/10.1016/j.jstrokecerebrovasdis.2018.05.019DOI Listing
October 2018

Good's syndrome with clinical manifestation after thymectomy: A case report.

Respir Med Case Rep 2018 27;24:89-91. Epub 2018 Apr 27.

Division of Respirology, NTT Medical Center Tokyo, 5-9-22 Higashigotanda, Shinagawa-ku, Tokyo, Japan.

Good's syndrome is a rare condition of immunodeficiency that is characterized by thymoma and hypogammaglobulinemia. A 74-year-old Japanese woman underwent total thymothymectomy for type AB thymoma (2015 WHO classification). She developed recurrent infectious diseases caused by (bacteremia), (pneumonia and bacteremia) and (bacteremia) in the year after thymectomy. The serum levels of immunoglobulin were significantly low (IgG 157mg/dL), which suggested that her infectious diseases were associated with Good's syndrome. Although she began receiving intravenous immunoglobulin every four weeks, she died of pneumonia a week after the second administration of immunoglobulin. When physicians encounter patients with recurrent infection who have a medical history of thymoma, the detection of hypogammaglubulinemia can be a key clue to the diagnosis of Good's syndrome.
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http://dx.doi.org/10.1016/j.rmcr.2018.04.014DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6010670PMC
April 2018

Small-Cell Carcinoma Transformation of Pulmonary Adenocarcinoma after Osimertinib Treatment: A Case Report.

Case Rep Oncol 2018 May-Aug;11(2):323-329. Epub 2018 May 29.

Division of Respirology, NTT Medical Center Tokyo, Tokyo, Japan.

There are various mechanisms underlying the resistance of EGFR-mutant lung adenocarcinoma to epidermal growth factor receptor tyrosine kinase inhibitors (EGFR-TKIs). We herein report a case of pulmonary adenocarcinoma with EGFR mutation (exon 19 deletion and T790M) that acquired resistance to osimertinib treatment because of transformation into small-cell lung carcinoma (SCLC). A 67-year-old ex-smoking woman was diagnosed with left upper lobe adenocarcinoma of clinical stage IIIA (cT2bN2M0). She was treated with chemoradiotherapy (cisplatin and vinorelbine plus radiation), gefitinib, cisplatin, and pemetrexed followed by pemetrexed maintenance therapy and erlotinib. Since a sample extracted from the metastatic lung tumor taken obtained via a transbronchial lung biopsy was found to be positive for the T790M mutation at the time of disease progression during erlotinib treatment, she received osimertinib treatment for 15 months until progressive disease. She developed resistance to osimertinib due to the histologic transformation to SCLC. Although the standard chemotherapy of carboplatin and etoposide for SCLC was administered, she died due to metastatic liver failure.
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http://dx.doi.org/10.1159/000489603DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6006630PMC
May 2018

Cytomegalovirus Hemorrhagic Cystitis in a Malignant Glioma Patient Treated with Temozolomide.

Intern Med 2018 Oct 18;57(20):3047-3050. Epub 2018 May 18.

Division of Respirology, NTT Medical Center Tokyo, Japan.

Temozolomide, a key drug in the treatment of malignant glioma, can cause profound lymphopenia and various opportunistic infectious diseases. A 79-year-old woman with anaplastic oligodendroglioma developed a fever and gross hematuria after 8 weeks of standard radiotherapy with concomitant temozolomide treatment. A cytomegalovirus (CMV) antigen test for pp65 antigenemia was positive (137 cells per 75,800 leukocytes), and the findings from a urine cytology test were consistent with CMV-induced hemorrhagic cystitis. She was treated with ganciclovir, and her condition improved. CMV monitoring is needed when patients develop symptoms related to opportunistic infections during temozolomide treatment for malignant glioma.
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http://dx.doi.org/10.2169/internalmedicine.1005-18DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC6232021PMC
October 2018

Eosinophilic Myocarditis With Hypersegmented Granulocytes and Neutrophilia Without Eosinophilia: A Case Mimicking Neutrophilic Myocarditis.

Int J Surg Pathol 2018 Oct 6;26(7):635-636. Epub 2018 Apr 6.

1 NTT Medical Center Tokyo, Tokyo, Japan.

We report a case of a 78-year-old woman with neutrophilia without eosinophilia who was pathologically diagnosed with eosinophilic myocarditis by myocardial biopsy. The biopsy specimen showed infiltrating granulocytes with hypersegmentation, mimicking neutrophils; however, they were confirmed to be eosinophils by Giemsa staining.
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http://dx.doi.org/10.1177/1066896918766669DOI Listing
October 2018

Hamartomatous inverted polyp of the sigmoid colon: A case demonstrating its immunohistochemical characteristics.

Pathol Int 2018 Jul 23;68(7):439-441. Epub 2018 Mar 23.

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Tokyo, Japan.

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http://dx.doi.org/10.1111/pin.12658DOI Listing
July 2018

Efficacy of an over-the-scope clip for preventing adverse events after duodenal endoscopic submucosal dissection: a prospective interventional study.

Endoscopy 2018 05 2;50(5):487-496. Epub 2018 Mar 2.

Department of Gastroenterology, Saitama Medical University International Medical Center, Saitama, Japan.

Background: Duodenal endoscopic submucosal dissection (ESD) remains technically challenging, with a high risk of severe adverse events. Because exposure of the duodenal post-ESD mucosal defect to pancreatic juice and bile acid reportedly induces delayed perforation and bleeding, we examined whether defect closure using an over-the-scope clip (OTSC) system was useful for preventing postoperative adverse events.

Methods: From April 2016 to February 2017, a total of 50 consecutive patients with superficial non-ampullary duodenal epithelial tumors (SNADETs) larger than 10 mm, with no more than semi-circumferential spread, were prospectively enrolled in this study. All of the lesions were treated by experienced ESD operators and the post-ESD mucosal defect was closed using OTSCs.

Results: All of the SNADETs were completely removed by ESD, with an R0 resection rate of 88.0 %. The mean procedure and closure times were 67.3 ± 58.8 minutes and 9.8 ± 7.2 minutes, respectively. Although complete defect closure was achieved in 94.0 % of the patients (47/50), two patients required surgical conversion. Delayed perforation occurred in only one patient (2.1 %), who did not have successful closure of the defect, as misplacement of the OTSC exposed the muscle layer. Meanwhile, delayed bleeding occurred in three patients (6.3 %); however, the bleeding was easily controlled using endoscopic coagulation. The mean duration of postoperative hospitalization was 5.5 ± 7.2 days.

Conclusions: Prophylactic defect closure using OTSCs may be effective in reducing severe adverse events after duodenal ESD.
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http://dx.doi.org/10.1055/s-0044-102255DOI Listing
May 2018

[A CASE REPORT OF PRIMARY MUCOSA ASSOCIATED LYMPHOID TISSUE LYMPHOMA OF THE PROSTATE].

Nihon Hinyokika Gakkai Zasshi 2018 ;109(3):150-155

Department of Pathology, NTT Medical Center Tokyo.

The patient was a 53-year-old man who was referred to the department of urology of our hospital after screening results indicated elevated prostate-specific antigen (PSA) levels. The PSA level was 5.33 ng/ml, and rectal examination revealed that the prostate was elastic and hard with mild prostatic hyperplasia. Because magnetic resonance imaging revealed abnormal signals in the prostatic transition area, prostate cancer was suspected and the patient underwent transrectal prostate needle biopsy. The pathological diagnosis was adenocarcinoma (Gleason score 5+5 = 10). After using thoracic, abdominal, and pelvic computed tomography (CT) and bone scintigraphy to confirm that metastasis had not occurred, robot-assisted radical prostatectomy (RARP) was performed. Prostate cancer was not detected during pathologic diagnosis of the surgical specimen, and on the basis of the results of re-examination with immunostaining, a diagnosis of mucosa-associated lymphoid tissue (MALT) lymphoma was made. In addition, an upper and lower endoscopy examination, positron emission tomography (PET) -CT, and bone marrow biopsy confirmed that generalized tumor lesions and lymph node swelling were not present, and the patient was diagnosed with primary MALT lymphoma of the prostate. Currently, 12 months since surgery, the patient continues to undergo follow-up as an outpatient and no recurrence has been observed. There have been only a few reports of primary MALT lymphoma of the prostate, in English or Japanese, and herein, we present our experience with a patient for whom a definitive diagnosis was difficult, along with a review of the literature.
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http://dx.doi.org/10.5980/jpnjurol.109.150DOI Listing
January 2018

The Successful Treatment of Pulmonary Pleomorphic Carcinoma with Pembrolizumab: A Case Report.

Case Rep Oncol 2017 May-Aug;10(2):752-757. Epub 2017 Aug 10.

Division of Respirology, NTT Medical Center Tokyo, Tokyo, Japan.

Pulmonary pleomorphic carcinomas are rare malignant tumors, and no standard treatments have been established. We herein report the successful treatment of a patient with pulmonary pleomorphic carcinoma with pembrolizumab. A 51-year-old man who was a current smoker presented to our hospital due to dyspnea and hemosputum. Chest X-ray showed right-sided pneumothorax with pleural effusion; chest tube drainage was therefore performed. Computed tomography after chest tube drainage showed a cavitary nodule in the right upper lobe and right hilar and bilateral mediastinal lymphadenopathy. Surgery was performed for the diagnosis and treatment. He was eventually diagnosed with pulmonary pleomorphic carcinoma corresponding to clinical stage IVB (cT2aN2M1c [PLE, ADR, HEP]). The giant cells strongly expressed programmed death ligand-1, and the tumor proportion score was more than 50%. Therefore, pembrolizumab was introduced as the first-line therapy. After 3 cycles of pembrolizumab, his right hilar and bilateral mediastinal lymphadenopathy and pleural dissemination notably decreased. Pembrolizumab might be an effective therapy for pulmonary pleomorphic carcinoma.
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http://dx.doi.org/10.1159/000479552DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5582494PMC
August 2017

Pulmonary Metastasis of Undifferentiated Uterine Sarcoma Forming Fluid-Containing Cyst.

Int J Surg Pathol 2018 Apr 1;26(2):180-184. Epub 2017 Sep 1.

1 NTT Medical Center Tokyo, Tokyo, Japan.

Undifferentiated uterine sarcoma (UUS) is a high-grade sarcoma with no specific differentiation; however, it exhibits variable positivity for CD10 as an immunohistochemical marker of endometrial stroma, suggesting immunohistochemical differentiation into endometrial stroma. It has been reported that some low-grade endometrial stromal sarcoma pulmonary metastatic foci show cystic changes; however, whether pulmonary metastatic UUS foci are solid or cystic remains undescribed. A 63-year-old woman underwent a hysterectomy. The subsequent pathologic diagnosis was UUS. After the operation, she was treated with chemotherapy; however, pulmonary solitary shadow was radiologically detected. Eight months after the gynecologic operation, her pulmonary lesion was surgically resected. Pathologically, the lesion consisted of a cyst containing serous fluid with thick whitish wall composed of tumor cells, which led to the diagnosis of metastatsis of UUS. The cyst seemed to be formed via secondary degeneration. She remains healthy without recurrence 28 months after her first visit to our hospital.
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http://dx.doi.org/10.1177/1066896917728795DOI Listing
April 2018

Surgery of Thymic Tumor with Persistent Left Superior Vena Cava.

Ann Thorac Cardiovasc Surg 2017 Dec 22;23(6):316-319. Epub 2017 Jun 22.

Department of Diagnostic Pathology, NTT Medical Center Tokyo, Tokyo, Japan.

Because a persistent left superior vena cava (PLSVC) is a rare congenital malformation in the thoracic venous system, surgery of the thymus in such patients has rarely been reported. We herein present a case involving a 68-year-old woman who was treated for a thymic tumor adhering to a PLSVC. She underwent complete resection of the thymic tumor through median sternotomy with left-sided video-assisted thoracic surgery. Although the tumor was close to the left phrenic nerve, this nerve was safely preserved. The pathological diagnosis was mucosa-associated lymphoid tissue (MALT) lymphoma of the thymus. Thoracoscopy combined with median sternotomy enabled us to perform a safe surgical procedure for this patient.
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http://dx.doi.org/10.5761/atcs.cr.16-00273DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5738455PMC
December 2017