[A Case of High-Grade Appendiceal Mucinous Neoplasm].

A 70-year-old woman underwent conservative treatment for abscess-forming appendicitis. A contrast-enhanced abdominal computed tomography(CT)revealed a cystic lesion at the appendiceal base while the abscess had disappeared posttreatment. With the diagnosis of a low-grade appendiceal mucinous neoplasm(LAMN), a laparoscopic-assisted ileocolic resection was performed. The appendix was distended with mucus in the lumen. Histopathological examination showed that the tumor cells were more atypical than that of low-grade appendiceal mucinous neoplasm. There were no findings of adenocarcinoma, such as invasive growth. Therefore, high-grade appendiceal mucinous neoplasm(HAMN)was diagnosed. HAMN is a term advocated by Carr et al in 2016 to classify appendiceal mucinous neoplasm and was noted as a new category of mucinous tumors among appendiceal epithelial tumors in the fifth edition of the 2019 revised WHO classification. HAMN is a rare disease and only one case has been reported in Japan. It has not yet been noted in the Japanese Colorectal Cancer Treatment Manual. Here, we report this rare case with a review of the study.

Duplicate appendix complicated by low-grade appendiceal mucinous neoplasm: a rare case report and review of the literature.

Low-grade appendiceal mucinous neoplasms (LAMNs) have been a very controversial tumor, and there is a lack of standardization for the optimal surgical procedure due to the infrequency of this disease. This is the first case report of duplicate appendix complicated by LAMN. The preoperative imaging examinations revealed that the mucinous tumor was shrinking spontaneously, allowing for safe laparoscopic resection. The histopathological findings indicated a dilated common base of the duplicated appendix, suggesting that the mucinous content drained spontaneously to the cecum. Further studies of the various complications of LAMNs are needed to establish the optimal surgical approach for LAMNs.

Intramedullary Nail Fixation with Autologous Bone Marrow Transplantation in an Incomplete Atypical Femoral Fracture Patient: Use of Bone Marrow Extracted from the Hollow Reamer.

The present report describes an incomplete atypical femoral fracture (AFF) patient who underwent simultaneous autogenous bone transplantation to the resected fracture region during intramedullary nail fixation. A 73-year-old female with a history of multiple myeloma had been undergoing treatment with intravenous drip injections of Zoledronic Acid. She was introduced to our department due to the left lateral thigh pain, with no trauma incidence. An anteroposterior radiograph showed a transverse thin fracture line with localized periosteal and endosteal thickening, which is compatible with subtrochanteric incomplete AFF. A biochemical investigation revealed the existence of severely suppressed bone turnover. She underwent intramedullary nail fixation for fear of a complete fracture. After the fixation, the cortical bone at the fracture region was excised as a wedge-shaped block, and bone marrow extracted from the hollow reamer was simultaneously transplanted to the resected fracture region. Histological examination showed few bone formation features at the fracture line in the excised lateral cortical bone. At 7 months after surgery, radiographs demonstrated complete bone repair, and no clinical problems were observed two years postoperatively. To the best of our knowledge, this is the first report in which autogenous bone marrow transplantation, noninvasive to the iliac crest, was performed in an incomplete AFF patient. We believe that this low invasive procedure can be a useful technique for AFF treatment.

[A Case of Adenocarcinoma Arising from Ectopic Gastric Mucosa in Meckel's Diverticulum with Abdominal Wall Abscess].

A 47-year-old woman visited a neighboring hospital because of lower abdominal pain and high fever. She was diagnosed with acute pyelonephritis and administered(oral)antibiotics. However, there was no sign of improvement. She was referred to our hospital for closer examination. Computed tomography revealed an 8 cm abdominal wall abscess spreading from the prevesical space to the rectus abdominis. Incisional drainage was performed under local anesthesia. She developed rectus abdominis muscle diastasis, and a laparotomy was performed 2 months later. Intraoperative findings showed that her Meckel's diverticulum was continuous with the abdominal wall abscess. Diagnosed with Meckel's diverticulitis with abdominal wall abscess, the patient underwent surgery(excision)for these. Histopathological findings showed adenocarcinoma cells in the abscess tissue and were continuous with gastric pyloric gland-like tissue in Meckel's diverticulum. Based on these findings, the patient was diagnosed with adenocarcinoma arising from ectopic gastric mucosa in the Meckel's diverticulum. The patient received postoperative adjuvant chemotherapy for a year. The patient is currently alive and has not experienced recurrence for 2 years since surgery. It is difficult to diagnose carcinoma of Meckel's diverticulum preoperatively due to late onset of symptoms. The diagnosis is often made at the advanced stage, when the prognosis is poor. This case is rare due to the incidental finding of an abdominal abscess and the absence of recurrence 2 years after surgery.

Unusual MRI Findings in a Polio Survivor.

A 63-year-old male consulted our institution due to worsening of right hip pain for approximately one month. The patient had no apparent functional disorders besides rigidity of the right ankle secondary to childhood poliomyelitis. Plain radiographs demonstrated narrowing of the right hip joint space. Magnetic resonance imaging (MRI) showed unusual findings in the right gluteus medius muscle, suspecting a malignant musculoskeletal tumor. Further examinations clarified acute inflammation caused by Staphylococcus aureus with no atypia. After treatment, serum inflammatory markers normalized and MRI showed homogeneous fat signal intensity in the muscle, which was consistent with poliomyelitis. Total hip arthroplasty was performed due to progression of osteoarthritis. Intraoperative findings showed flaccidity of the gluteus medius muscle, and histological examination of the specimen also was compatible with poliomyelitis. Postoperatively there was no hip instability and the patient has been able to resume his previous physical activity. To our knowledge, this is the first report regarding polio survivors combined with septic arthritis, and sole MRI examination was unable to lead to the diagnosis. The current patient demonstrates the possibility that the involved muscles in poliomyelitis exist even in asymptomatic regions, which will be helpful for accurate diagnosis and life guidance in polio survivors.

Pseudotumor Caused by Titanium Particles From a Total Hip Prosthesis.

A 77-year-old woman underwent metal-on-polyethylene total hip arthroplasty for osteoarthritis of the right hip at another institution. During surgery, the greater trochanter was broken and internal fixation was performed with a trochanteric cable grip reattachment. Although postoperative recovery was uneventful, approximately 6 years later, the patient had severe right hip pain with apparent swelling, and she was referred to the authors' institution. Plain radiographs showed evidence of severe osteolysis in the proximal femur and cable breakage; however, preoperative aspiration culture findings were negative for bacterial growth. Magnetic resonance imaging showed a well-circumscribed mass, presumed to be a pseudotumor. Serum cobalt and chromium levels were within normal limits, and the serum titanium level was high. During surgery, the mass was excised and removal of the cable system revealed a sharp deficit in the bare femoral stem. Gross surgical findings showed no obvious evidence of infection and no corrosion at the head-neck junction; therefore, all components were retained besides the cable system, which resulted in clinical recovery. All of the cultures from specimens were negative for bacterial growth, and histologic findings were compatible with a pseudotumor, such as histiocytes containing metal particles, abundant plasma cells, and CD8-positive cells. Quantitative analysis by inductively coupled plasma atomic emission spectrometry showed that the main source of metal debris in the pseudotumor was the femoral stem, which was made of titanium alloy, not the broken cable, which was made of cobalt-chromium alloy. The findings suggest that titanium particles can form symptomatic solid pseudotumors.

[A Case of Sigmoid Colon Cancer with Synchronous Liver Metastasis, Treated with Laparoscopic Liver Resection after SOX plus BV Chemotherapy].

We performed laparoscopic liver resection in a patient with synchronous liver metastasis from advanced sigmoid colon cancer after induction with S-1 plus oxaliplatin (SOX) plus bevacizumab (BV) chemotherapy. A 61-year-old woman underwent laparoscopy-assisted sigmoidectomy for locally advanced sigmoid colon cancer with synchronous liver metastasis. SOX plus BV chemotherapy was initiated. After 3 courses, the liver tumor was downsized, and metastasectomy was performed laparoscopically with R0 resection. The postoperative course was uneventful and the patient was discharged on the 11th postoperative day. She has been free from recurrence. Induction with SOX plus BV chemotherapy is considered to be not only effective, but also beneficial for maintaining the quality of life (QOL) in patients with advanced colorectal cancer.

Titanium alloy stem as a cause for adverse reaction to metal debris after bipolar hemiarthroplasty.

A 68-year-old male with failure of bipolar hemiarthroplasty consistent with adverse reaction to metal debris (ARMD) who presented with a painful cystic lesion and lower extremity swelling was encountered. However, revision surgical findings showed no apparent cause of ARMD previously described in the literature, such as corrosion at the head-neck junction and articular abrasion. Therefore, it was difficult to make a definite diagnosis of failure secondary to ARMD, which consequently led to the decision to perform two-stage revision procedure, though the stem was firmly fixed. Postoperative analysis in the retrieval tissues showed that the metal debris mainly originated from the titanium alloy stem itself. Although this is a very rare case, one should be aware that even the well-fixed femoral components themselves have the potential to be the cause of ARMD.

Osseous metaplastic meningioma in the thoracic spine mimicking osteosarcoma: a case report.

STUDY DESIGN: Case report. OBJECTIVE: We describe a case of osseous metaplastic meningioma in the thoracic spine that pathologically mimicked osteosarcoma. SUMMARY OF BACKGROUND DATA: As meningioma presents in many pathological forms, it is sometimes difficult to diagnose it pathologically. METHODS: The patient's medical records, imaging results, and pathological findings were reviewed, as was the relevant literature. RESULTS: A 20-year-old woman with a 6-month history of lumbago and right sciatica was referred to our hospital because magnetic resonance imaging (MRI) showed a tumor compressing her spinal cord at the T11 vertebra level. Computed tomography (CT) showed calcification of the tumor, and the preoperative diagnosis was meningioma. Surgery was performed and the tumor was entirely removed. The tumor was very hard, and pathological findings suggested atypical meningioma with massive ossification. Some parts of the tumor seemed malignant, as spindle cells with a high nucleocytoplasmic ratio were highly concentrated, which led to the possibility of osteosarcoma. The tumor was conclusively diagnosed as osseous metaplastic meningioma based not only on the pathology, but also on CT and MRI findings and the postoperative course. CONCLUSION: As meningioma presents in many pathological forms, it is sometimes difficult to diagnose it pathologically. Results of imaging studies including CT and MRI, as well as patients' postoperative course, should be considered when making a final diagnosis of meningioma. LEVEL OF EVIDENCE: N/A.

Dumbbell-shaped nonpsammomatous malignant melanotic schwannoma of the cervical spinal root.

BACKGROUND CONTEXT: Melanotic schwannoma is a very rare tumor of Schwann cell origin, which can develop in various locations, similar to conventional schwannoma. This tumor has a malignant potential and therefore careful therapy is required. PURPOSE: To describe a case of melanotic schwannoma with a histopathologically and clinically malignant behavior. STUDY DESIGN: Case report. METHODS: A 64-year-old man presented with sensory changes in his arm and gait disturbance. Magnetic resonance imaging revealed a dumbbell-shaped tumor at the left C7 spinal root, which was hyperintense on T1-weighted images and generally hypointense on T2-weighted images in comparison with conventional schwannoma; however, the peripheral zone was relatively hyperintense, and the central zone was hypointense like a target sign. RESULTS: The tumor was partially resected and diagnosed to be nonpsammomatous malignant melanotic schwannoma. The patient experienced local recurrence and metastases to the bone and lung and finally developed quadriplegia. Radiation therapy failed to palliate the symptoms. CONCLUSIONS: Some melanotic schwannomas present with an aggressive behavior, which thus leads to poor prognosis. We should therefore be familiar with its characteristic clinical imaging and pathologic findings to provide a correct diagnosis and appropriate treatment for such patients.

Overexpression and altered subcellular localization of autophagy-related 16-like 1 in human oral squamous-cell carcinoma: correlation with lymphovascular invasion and lymph-node metastasis.

Autophagy is a dynamic process of subcellular degradation, which has recently sparked great interest because it is involved in various developmental processes and various diseases including cancer. Autophagy-related 16-like 1 is a component of a large protein complex essential for autophagosome formation. We previously applied proteomic methods to characterize differentially expressed proteins in oral squamous cell carcinoma cells and detected significantly high expression levels of autophagy-related 16-like 1 in oral squamous cell carcinoma-derived cell lines compared to human normal oral keratinocytes. In the current study, to further determine the potential involvement of autophagy-related 16-like 1 in oral squamous cell carcinoma, we evaluated the state of autophagy-related 16-like 1 protein expression in human oral premalignant lesions and primary oral squamous cell carcinomas, and correlated the results with clinicopathologic variables. Autophagy-related 16-like 1 immunoreaction was predominant in a variety of subcellular components of oral squamous cell carcinoma tissues, including the cytoplasm and plasma membrane of malignant cells (45% and 39%, respectively) and peritumoral and intratumoral stroma (52%), whereas all of the components in normal tissues had no or faint autophagy-related 16-like 1 expression. In addition, high stromal expression of autophagy-related 16-like 1 was associated significantly with lymphovascular invasion of tumor cells (P = .037) and positive lymph node status (P = .015). Furthermore, cytoplasmic and plasma membranous autophagy-related 16-like 1 were also expressed in abundance in the oral premalignant lesion cells (74% and 32%, respectively). Our finding suggests that dysregulation of autophagy-related 16-like 1 protein expression is a frequent and early event during oral carcinogenesis and could affect the malignant behavior of oral squamous cell carcinoma cells.

Clinical significance of gelsolin-like actin-capping protein expression in oral carcinogenesis: an immunohistochemical study of premalignant and malignant lesions of the oral cavity.

BACKGROUND: Gelsolin-like actin-capping protein (CapG) is a ubiquitous gelsolin-family actin-modulating protein involved in cell signalling, receptor-mediated membrane ruffling, phagocytosis, and motility. CapG has generated great interest due to its oncogenic function in the control of cell migration or invasion in a variety of cancer cells. We previously applied proteomic methods to characterize differentially expressed proteins in oral squamous-cell carcinoma (OSCC) cells and detected significantly high expression levels of CapG in OSCC-derived cell lines compared to human normal oral keratinocytes. In the current study, to further determine the potential involvement of CapG in OSCC, we evaluated the status of CapG protein and mRNA expression in human oral premalignant lesions (OPLs) and primary OSCCs and correlated the results with clinicopathologic variables. METHODS: Matched normal and tumour tissue sections of 79 human primary OSCCs and 28 OPLs were analyzed for CapG expression by immunohistochemistry (IHC). Correlations between CapG-immunohistochemical staining scores of OSCCs and clinicopathologic features were evaluated by Fisher's exact test. Real-time quantitative reverse transcriptase-polymerase chain reaction (qRT-PCR) was used to estimate CapG expression at the mRNA level. RESULTS: In IHC, substantial up-regulation of CapG protein was observed in primary OSCCs (52%) and OPLs (64%), whereas corresponding normal tissues showed consistently weak or absent immunoreactivity of CapG. qRT-PCR data were consistent with the protein expression status. Moreover, CapG expression was correlated with the TNM stage grading of OSCCs. CONCLUSION: Our finding of frequent dysregulated expression of CapG in premalignant and malignant lesions together with an association with an advanced clinical disease stage suggests that CapG could contribute to cancer development and progression and that CapG may have potential as a biomarker and a therapeutic target for OSCC.

Xanthogranulomatous gastritis with pseudosarcomatous changes.

Reported herein is a rare case of xanthogranulomatous inflammation of the gastric wall occurring in a 77-year-old man. Two submucosal lesions presented as rapidly enlarging nodules, and biopsy showed interweaving bundles of spindle cells with numerous atypical cells with marked nuclear pleomorphism. The differential diagnosis from mesenchymal malignancies, particularly from a malignant gastrointestinal stromal tumor, was difficult and immunohistochemical investigations could not improve the diagnostic accuracy of HE histology alone. Thus, an erroneous diagnosis of malignancy was made and a partial gastrectomy was performed. On macroscopic examination of the resected material, spontaneous regression of the lesions was observed and microscopic examination showed characteristic features of xanthogranulomatous inflammation; large numbers of foamy histiocytes including multinucleated giant cells were admixed with chronic inflammatory cells and fibrous reaction. Although the precise pathogenesis could not be elucidated, recognition of this unusual morphological appearance is of importance to avoid an overdiagnosis of malignancy.

The importance of bacterial superantigens produced by Staphylococcus aureus in the treatment of atopic dermatitis using povidone-iodine.

Atopic dermatitis (AD) is frequently associated with intestinal and cervical lesions. Staphylococcus aureus produces many kinds of toxins, the bacterial superantigens. The detection rate of toxins was 80.1% from 196 S. aureus strains. Neurological examinations revealed abnormalities in 59 out of 81 AD patients. Cervical magnetic resonance imaging (MRI) was performed in 46 patients randomly and showed abnormal findings in 38 of these patients. In 23 patients who underwent MRI and duodenal biopsy, 3 were found to be normal neurologically and 2 patients showed normal duodenal tissue. However, 18 patients had abnormal findings both on neurological examination and in duodenal tissue. Serial duodenal biopsy tests were performed in 10 AD patients. In 5 patients, the findings of chronic duodenitis disappeared after the therapy with povidone-iodine. These data indicate that the therapy was effective not only for the skin lesions, but improved gastrointestinal tract lesions and cervical myelopathy, by eradicating bacterial superantigens.

Adenocarcinoma with peritoneal dissemination secondary to multiple mature teratomas of the omentum.

BACKGROUND: Teratomas of the greater omentum are rare. In most cases, they are composed of mature tissue elements and behave clinically in a benign fashion. CASE: A 62-year-old woman with a history of bilateral salpingo-oophorectomy due to ectopic pregnancy was found to have multiple cystic teratomas of the omentum. There was a histologic evidence of a sequential transition from benign through borderline to obviously invasive adenocarcinoma within the lining of the cysts as well as peritoneal dissemination. CONCLUSION: We defined the present case as malignant transformation of a mature teratoma, but have no evidence whatsoever for the origin of multiple mature teratomas. Further detailed analyses with more elaborated procedures on many relevant reports would be required to establish the origin and biologic behavior of teratomas of the omentum.

Intraductal papillary-mucinous tumors of the pancreas.

BACKGROUND/AIMS: To elucidate the risk of malignancy and the morphological alterations associated with malignancy. METHODOLOGY: Thirty cases of intraductal papillary-mucinous tumors and 5 papillary-mucinous carcinomas (invasive intraductal papillary-mucinous tumors) of the pancreas were clinicopathologically and histopathologically analyzed. RESULTS: The invasive carcinoma developed on the basis of severe dysplasia-carcinoma in situ changes and never from mild or moderate dysplasia changes. However, tumor cell projections of intraductal papillary-mucinous tumors encroached into the duct wall and/or the stroma introduced just beneath the epithelium and "intraductal" tumor cells sometimes came in direct contact with the "extraductal" connective tissues even in adenomas. The frankly invasive adenocarcinoma components of invasive intraductal papillary-mucinous tumors were characterized by the lack or poor formation of their own basement membrane and were usually surrounded by the extensive collagenous proliferation, desmoplastic reaction. Such stromal alterations never developed around the "extraductal" components of non-invasive intraductal papillary-mucinous tumors. CONCLUSIONS: The risk of malignancy for an individual intraductal papillary-mucinous tumor was increased with the degree of cellular and/or structural atypia. The desmoplastic reaction with poor formation of the basement membrane is the sine qua non of the "true invasion".

Localized necrotizing angiitis of the ileum.

A 20-year-old woman was hospitalized repeatedly because of intermittent bouts of intestinal obstruction and the symptoms usually improved with conservative treatments. One year after the first admission the patient was hospitalized in emergency and a laparotomy revealed a circular stricture with a pinhole perforation in the ileum. Histological sections of the stricture showed the characteristic features of microscopic polyangiitis varying from active to resolving stages, which were localized in the ileum. Fibrinoid necrosis, fibroblastic and fibrous proliferation of the intima and fibrous replacement of the media with a variable pan- and perivascular inflammatory cell infiltrate were characteristic in the muscular arteries and arterioles. Vascular occlusion by pale eosinophilic, fibrillar-like materials resembling livedo racemosa of the skin, was noticed in the small arterioles and capillaries. Under no prophylaxis, the postoperative course was uneventful with no recurrence of the illness at an 18-month follow up. The pathological alterations were distributed focally, occasionally segmentally, and haphazardly, and required detailed examination by stepwise sections for the histological diagnosis.

Expression and regulation of the gene for arginase I in mouse salivary glands: requirement of CCAAT/enhancer-binding protein alpha for the expression in the parotid gland.

Arginase in salivary glands is potentially involved in the synthesis of proline, glutamate, and polyamines that play specific physiological roles in the glands, and also in depletion of arginine in the oral cavity to protect teeth from microorganisms. We detected protein and mRNA for the type I isoform of arginase in mouse salivary glands. Enzymes of the arginine-biosynthetic pathway were also detected. Immunohistochemical analysis revealed that arginase I was enriched in the striated duct, and was also present in the acinus, demilune and granulated duct. Mice with targeted disruption of the gene for C/EBPalpha, which is a transcription factor essential for expression of the arginase I gene in the liver, showed dramatically reduced immunoreactivity for arginase I in the parotid gland but not in the submandibular and sublingual glands. Therefore, C/EBPalpha is specifically required for expression of the arginase I gene in the parotid gland.

The importance of disinfection therapy using povidone-iodine solution in atopic dermatitis.

The combination of the local disinfection therapy against Staphylococcus aureus with the conventional therapy for atopic dermatitis has been widely used, and the improvement in skin lesions has been reported to be associated with a remarkable decrease in IgE levels and reagin antibody titers. We have already reported that affected organs were not only the skin but also the gastrointestinal tract in a case with atopic dermatitis. In the present study, the duodenal tissues were examined by biopsy in 32 patients with atopic dermatitis, and mild or chronic duodenitis was observed in all samples. Toxins were examined by PCR from 180 Staphylococcus aureus strains obtained from our patients. The detection rate of toxins was 82.8%. In many patients, antitoxin IgE antibody titers corresponding to their types of toxin and IgE levels were decreased in a parallel manner as time passed. We found 1 patient who complained of paresthesia in all four limbs, and her neurological and radiological examinations showed moderate cervical spondylosis. Neurological examinations revealed some abnormalities in 43 out of 50 patients with atopic dermatitis, such as hyperreflexia of the legs. Cervical MRI was carried out randomly and showed abnormal findings in 21 of 25 patients, in whom 18 duodenal tissues were examined by biopsies.