A very rare case of spontaneous regression of basaloid squamous cell carcinoma of the lung.

Spontaneous regression of non-small cell lung cancer is relatively rare. Here, we present a very rare case of spontaneous regression of lung cancer which occurred in a patient with basaloid squamous cell lung cancer. To the best of our knowledge, this is the first report of such a case. A 76-year old man was referred to our hospital with nodules in the right upper lobe determined by chest computed tomography. The nodules spontaneously regressed during follow-up. Two years later, the tumor had regrown and the patient subsequently underwent surgery. The pathological findings showed basaloid squamous cell carcinoma. Stimulation of the immune system was considered to be the cause of the spontaneous regression and CD-8 positive and CD-4 positive lymphocytes might play an important role.

Comparative Immunohistochemical Analysis of Macrophage Phenotypes in Cutaneous Sarcoid Granuloma, Suture Granuloma, and Lipogranuloma.

ABSTRACT: Granulomas are composed of a heterogeneous population of resident and recruited macrophages according to the type of lesion, extent of injury, and local tissue environment (eg, involved site and interaction with infiltrating lymphocytes). Although macrophage phenotypes in various types of granulomas have been previously described, the experimental conditions varied across studies, precluding a comparative and comprehensive understanding of granulomas. This study was conducted to comparatively analyze the expression of markers of the M1 and M2 phenotypes in macrophages that compose various types of granulomas, including epithelioid lesions, under strict conditions. Surgical specimens of cutaneous sarcoidosis (11 lesions), suture granuloma (10 lesions), and subcutaneous lipogranuloma (12 lesions) were immunohistochemically stained for CD11c, CD206, CD163, and CD10. The expression of these markers in macrophages composing each type of granuloma was scored and statistically analyzed. Granuloma macrophages were mostly immunoreactive for CD11c and CD206 in all the examined cases, although many intermingling CD206-negative cells were observed in 5 cases of lipogranuloma. CD163 and CD10 were diffusely expressed in macrophages composing suture granuloma and lipogranuloma, whereas they were not expressed in epithelioid cells in cutaneous sarcoidosis. Meanwhile, "interstitial" macrophages around epithelioid granulomas revealed moderate to marked CD163 expression in 7 lesions of cutaneous sarcoidosis. These results indicate significant differences of expression of CD163 and CD10 between cutaneous sarcoidosis and suture granuloma/lipogranuloma; CD163 and CD10 are downregulated after the epithelioid transformation of macrophages in cutaneous sarcoidosis.

Disseminated carcinomatosis of the bone marrow caused by granulocyte colony-stimulating factor: A case report and review of literature.

BACKGROUND: Disseminated carcinomatosis of the bone marrow (DCBM) is a widespread metastasis with a hematologic disorder that is mainly caused by gastric cancer. Although it commonly occurs as a manifestation of recurrence long after curative treatment, the precise mechanism of relapse from dormant status remains unclear. Granulocyte colony-stimulating factor (G-CSF) can promote cancer progression and invasion in various cancers. However, the potential of G-CSF to trigger recurrence from a cured malignancy has not been reported. CASE SUMMARY: A 55-year-old Japanese woman was diagnosed with Ewing sarcoma localized on the fifth lumbar vertebrae 6 years after curative gastrectomy for T1 gastric cancer. After palliative surgery to release nerve compression, pathological diagnosis of the resected specimen was followed by curative radiation and chemotherapy. During treatment, G-CSF was administered 32 times for severe neutropenia prophylaxis. Eight months after completing definitive treatment, she complained of severe back pain and was diagnosed as multiple bone metastases with DCBM from gastric cancer. Despite palliative chemotherapy, she died of disseminated intravascular coagulation 13 d after the diagnosis. Immunohistochemical examination of the autopsied bone marrow confirmed a diffuse positive staining for the G-CSF receptor (G-CSFR) in the relapsed gastric cancer cell cytoplasm, whereas the primary lesion cancer cells showed negative staining for G-CSFR. In this case, G-CSF administration may have been the key trigger for the disseminated relapse of a dormant gastric cancer. CONCLUSION: When administering G-CSF to cancer survivors, recurrence of a preceding cancer should be monitored even after curative treatment.

Primary spindle cell tumor originating from the liver that was difficult to diagnose.

BACKGROUND: It has been reported that hepatocellular carcinoma (HCC) with spindle cell tumor accounts for 1.8% of all HCCs, but spindle cell tumors that do not show an obvious conventional HCC are extremely rare. In this report, we describe a case of resection of a primary spindle cell tumor of the liver that was difficult to diagnose. CASE PRESENTATION: A 75-year-old man presented with fever and right chest pain. He was suspected of a giant primary diaphragmatic tumor of extrahepatic origin by imaging studies. The preoperative differential diagnosis included benign masses such as myxoid sarcoma and schwannoma, and we planned a diaphragmatic resection. Intraoperatively, however, dissection of the tumor from the liver was not possible, requiring an extended right posterior segmentectomy with combined resection of the diaphragm. The patient had a good postoperative course and 1 year has passed since the surgery with no recurrence. The pathology showed that the mass was located just below the hepatic capsule/parenchymal region and was adherent to the diaphragm, but there was no continuity. The morphology suggested a low-grade mesenchymal tumor such as a solitary fibrous tumor and perivascular epithelioid cell tumor, but immunostaining was negative, making the diagnosis difficult. Although some areas of high proliferative activity were observed, finally, the diagnosis of primary spindle cell tumor of the liver with smooth muscle differentiation was made based on the positive results of muscle markers such as αSMA, desmin, and h-caldesmon. CONCLUSIONS: Spindle cell tumor arising from the liver is so rare that preoperative and pathological diagnosis is often difficult to reach. Although further studies are needed to elucidate and better understand this uncommon clinical entity, we consider that complete resection is necessary for the above case, which may contribute to long-term survival.

Phosphoglyceride crystal deposition disease involving adnexa uteri: a case report with histogenetic consideration.

We present a rare case of phosphoglyceride crystal deposition disease (PCDD), as a gynecologic disease, with reference to histogenesis of crystal deposition. An 84-year-old woman, who had undergone simple hysterectomy for uterine leiomyoma 44 years previously, presented with multiple masses in the bilateral adnexa and the pelvic wall. The bilateral adnexal tumors were resected. The masses histologically revealed a foreign-body granuloma composed of numerous tiny, radially arranged needle-like crystal lumps surrounded by multinucleated giant cells and macrophages. The crystals showed birefringence under polarized light and were positive for gold hydroxamic acid stain, and the tumor was thus diagnosed as PCDD. The masses revealed central cystic changes due to old hemorrhage, which contained crystal lumps without foreign-body reaction or birefringence. The present case demonstrated for the first time that phosphoglyceride crystals developed in old hemorrhagic foci, although it was not confirmed whether the old hemorrhagic foci were formed after hysterectomy or due to endometriosis.

[Case of Asymptomatic Multiple Bone and Bone Marrow Metastases in Gastric Signet-Ring Cell Carcinoma].

A 57-year-old man visited our hospital for evaluation of an abnormal shadow identified on chest radiography. Chest computed tomography findings suggested diffuse bone metastases in the thoracic spine and the bilateral ribs. Notably, 18- fluoro-deoxyglucose positron emission tomography revealed no evidence of the primary tumor. Esophagogastroduodenoscopy revealed a small flat depressed lesion in the greater curvature of the gastric angle. Histopathological examination of this specimen revealed a signet-ring cell carcinoma. Histopathological examination of a biopsy obtained from the right iliac bone revealed a signet-ring cell carcinoma similar to that observed in the gastric mucosa. He was diagnosed with a gastric signetring cell carcinoma with multiple bone and bone marrow metastases. Cervical metastases caused gradual worsening of respiratory functions, necessitating artificial ventilation. He died of sudden ventricular tachycardia on the 36th day. Clinicians should be aware of the features of primary gastric cancer with bone and bone marrow metastases for early diagnosis and prompt treatment.

Immunohistochemical Localization of Phosphorylated and Unphosphorylated Form of ß-catenin With Regard to Shadow Cell and Squamous Differentiation in Cutaneous Pilomatricoma.

Pilomatricoma usually contains a mutation in CTNNB1 that encodes ß-catenin (BC). It also shows nuclear accumulation of BC protein, which plays an important role in tumorigenesis of pilomatricoma. In vitro studies have indicated that mutant BC protein is unphosphorylated and shows nuclear accumulation, but this theory has not been confirmed in various tumors with CTNNB1 mutation. We examined immunohistochemical localization of phosphorylated BC (pBC) and unphosphorylated BC (npBC) with regard to the modes of cell death or differentiation in 25 cases of pilomatricoma. As for the component showing shadow cell differentiation, BC was detected in cytoplasm/nucleus and along cell membrane in basaloid cells, whereas only in the latter in transitional cells in all cases. Meanwhile, npBC was localized along cell membrane of transitional cells, but not in basaloid cells, nor in nucleus of any components. The components with squamous differentiation also revealed the staining patterns similar to those seen in shadow cell differentiation in some cases. pBC was found in some cell fragments in the amorphous debris containing apoptotic bodies among shadow cell nests. These results suggested that npBC plays an important role in cell adhesion during differentiation and that pBC expression is associated with apoptosis of basaloid cells in pilomatricoma. BC accumulated in the nucleus was not immunoreactive for npBC possibly due to post-translational modification or conformational changes that resulted in loss of or masked antigenicity when BC is assumed to be unphosphorylated.

Sebaceous carcinoma of the breast predominantly characterized by intraductal growth: a case report.

BACKGROUND: Sebaceous carcinoma (SC) is frequently classified as periocular or extraocular. Extraocular SC is rare and mainly occurs in the head and neck, the major salivary glands, or oral mucosa. SC of the breast, lung, and ovary is particularly rare, and the few cases of SC of the breast predominantly exhibit intraductal growth. CASE PRESENTATION: A 47-year-old Japanese woman was referred to our hospital with accumulated polymorphic calcification in the left breast which was detected using mammography. Ultrasonography revealed an irregular 13-mm mass in the left breast, and analysis of a core needle biopsy revealed noninvasive ductal carcinoma. Total mastectomy and sentinel lymph node biopsy were performed. Histopathology demonstrated that carcinoma in situ (CIS) represented a significant lesion, and the cytoplasm of tumor cells was clear with numerous minute vacuoles. Immunohistochemical analysis demonstrated that most tumor cells expressed adipophilin. Together, these findings led to a diagnosis of SC, mainly comprising CIS. CONCLUSIONS: We encountered a rare case of SC of the breast with predominant CIS.

[A Case of Stromal Sarcoma of the Breast Showing Rapid Increase].

An 83-year-old woman visited our hospital with a mass on her right breast mass. Breast ultrasonography revealed a tumorous lesion approximately 30mm in diameter in the right E region. Fibrous tumor was suspected from core needle biopsy. Since surgery for definite diagnosis was not desired, she was followed. Seven months after the first visit, skin redness, pain, and rapid increase in tumor size was consciously recognized, which prompted a reconsultation. The tumor diameter rapidly increased to 74 mm, and a total right mastectomy was performed as a semi-emergency procedure. Necrotic and hemorrhagic lesions were mixed in a mottled state in the cleaved surface of the excised specimen, and the pathological diagnosis was stromal sarcoma mainly comprising of fibrosarcoma components. Stromal sarcoma of the breast lacks the epithelial component of a malignant phyllodes tumor, and effective systemic therapy has not been established; therefore, cases with local recurrence or distant metastasis result in poor prognosis. Since we encountered a case of stromal sarcoma of the breast that showed a rapid increase after several months, we will provide a report with some literature consideration.

Clinical significance of serum PSA in breast cancer patients.

BACKGROUND: Recent preclinical data suggest that androgen receptor (AR) signaling plays a significant role in subsets of breast cancer. Clinical trials testing AR-targeting therapies in breast cancer have been conducted. Assessment of AR-signal in breast cancer tissue maybe useful for treatment selections. Prostate specific antigen (PSA) is the product of an androgen-responsive gene. Serum PSA (sPSA) can be detected in women by a highly sensitive assay although the concentration is much lower than that observed in males. We investigated if sPSA reflects tumor biology, including AR signaling in breast cancer patients. METHODS: In this study, 132 healthy controls and 144 breast cancer patients were enrolled. sPSA was evaluated by the chemiluminescent enzyme immunoassay (CLEIA) method. Correlations between sPSA and the various clinicopathological factors were analyzed. RESULTS: In post-menopausal state, sPSA detection rate was significantly higher in breast cancer patients compared with controls (27.4% vs 11.3%: p = 0.0090), but not in the whole cohort (29.2% vs 25.8%: p = 0.5265) or pre-menopausal subgroup (37.0% vs 42.6%: p = 0.6231). In post-menopausal breast cancer cases, higher sPSA value was associated with clinic-pathological factors including the expression of AR protein in primary legion. In a correlation analysis of quantitative data limited to post-menopausal metastatic breast cancer (MBC), sPSA was positively, albeit weakly correlated with clinic-pathological features including serum testosterone levels and AR positivity. CONCLUSIONS: Our data suggest that sPSA may reflect tumor biological properties including AR activity in post-menopausal breast cancer.

Thoracoscopic resection of a cystic seminoma of the mediastinum.

Mediastinal seminoma is an uncommon tumor that accounts for 25% of primary mediastinal germ cell tumors, which in turn comprise fewer than 5% of all germ cell tumors. Although CT normally shows a solid, lobulated tumor, mediastinal cystic seminoma has rarely been described. Here, we report a 24-year-old man who presented with a mediastinal cystic tumor that was resected after an 18-month delay via video-assisted thoracoscopic surgery while in the supine position; the procedure involved lifting the chest wall with a subcutaneous Kirschner wire. Pathological examination revealed a mediastinal cystic seminoma. No evidence of recurrence has been noted during 25 months of follow-up. Mediastinal cystic seminoma should be considered in the differential diagnosis of cystic lesions of the mediastinum. Moreover, video-assisted thoracoscopic resection may be an appropriate option for the diagnosis and treatment of such lesions.

Shadow Cell Differentiation: A Comparative Analysis of Modes of Cell Death with Apoptosis and Epidermal/Trichilemmal Keratinization.

Shadow cells are characterized by an eosinophilic cytoplasm and a ghost-like nuclear contour; the cell shape is preserved, in spite of nuclear disappearance. Shadow cell nests (SCNs) are frequently observed in pilomatricoma (PMX), where the transitional cells immediately adjacent to SCNs often have a crescent-shaped nucleus showing fragmentation similar to that of apoptotic bodies. They show nuclear accumulation of beta-catenin and DNA double strand breaks (as revealed by in situ 3'-tailing reaction or immunohistochemistry for single-stranded DNA [ssDNA]), while they are negative for cleaved caspase-3 or cleaved lamin A, suggesting that shadow cell differentiation (SCD) is a caspase-independent programmed cell death. SCD can be differentiated from epidermal keratinization (EK) and trichilemmal keratinization (TK) based on the expression pattern of beta-catenin, ssDNA, and caspase-14/CD138. SCD is observed not only in PMX, but also sometimes in basal cell carcinomas, gonadal teratomas, and various extra-cutaneous carcinomas. In particular, SCNs are found in 24$ of endometrial adenoacanthoma and are derived from squamoid morules. This establishes a link between basaloid cells in PMX and squamoid morules in endometrial adenoacanthomas as common precursors of shadow cells. Overall, it is suggested that SCD is different from, but partly similar to, apoptosis and that SCD and EK/TK should be differentiated from the standpoint of cell death/differentiation.

Promoter hypomethylation of SKI in autoimmune pancreatitis.

The relationship between methylation abnormality and autoimmune pancreatitis (AIP)-a representative IgG4-related disease-has not yet been elucidated. We identified SKI might have a significant methylation abnormality in AIP through methylation array analysis using the Illumina Infinium Human Methylation 450K BeadChip array, and investigated the relationship of SKI with AIP clinicopathological features. The methylation rate of SKI was assessed by quantitative SYBR green methylation-specific PCR, and the degree of SKI expression in tissue specimens was assessed by immunohistochemistry in 10 AIP cases, 14 cases of obstructive pancreatitis area in pancreatic ductal adenocarcinoma (PDA) without a history of AIP, and 9 normal pancreas (NP) cases. The SKI methylation ratio was significantly lower in AIP than in PDA and NP. Additionally, the immunohistochemical staining-index (SI) score for SKI was significantly higher in AIP than NP, although there was no significant difference between AIP and PDA. There was a strong negative correlation between SI score and SKI methylation ratio, and between the serum concentrations of IgG4 and the SKI methylation ratio. There was a moderate positive correlation between the serum concentrations of IgG4 and SI. SKI is thought to be an oncogene indicating that SKI hypomethylation and carcinogenesis might be linked to AIP. Furthermore, the correlation between serum concentrations of IgG4 and SKI methylation levels suggest SKI might be involved in the pathogenesis of AIP. However, the role of SKI has not been clearly elucidated. Further studies are needed to understand further the function of SKI.

Sebaceous carcinoma of the breast: a case report.

BACKGROUND: Sebaceous carcinoma of the breast is a distinct variant of invasive ductal carcinoma. It is rare and only several cases have been reported. CASE PRESENTATION: An 80-year-old woman noted bloody discharge from her left nipple and palpated a lump in her left breast. Ultrasonography revealed a 19-mm mass in the left breast. Fine-needle aspiration suggested invasive ductal carcinoma. Partial mastectomy and sentinel lymph node biopsy were performed. On histological examination, the tumor revealed solid growth of small, round uniform cells with clear cytoplasm, partially intermingled with vacuolated cells indicative of sebaceous differentiation. The tumor cells contained abundant Sudan Black B-positive lipid droplets in the cytoplasm, and they were immunohistochemically positive for adipophilin. They were negative for estrogen receptor, progesterone receptor, and androgen receptor; positive for cytokeratin 7 and Ber-EP4; and partially positive for epithelial membrane antigen. Based on these findings, the patient was diagnosed with sebaceous carcinoma of the breast. CONCLUSIONS: We diagnosed a rare case of sebaceous carcinoma of the breast.

[A Case of Chordoma of the Sacrum Treated with Pazopanib].

A-70-year-old man with chordoma of the sacrum was treated with pazopanib (initially 400 mg/day, upto 800 mg/day). After 2 months of treatment, a significant tumor reduction was achieved and the patient was able to sit down easily. Therefore, the pazopanib therapy was continued. He had 14 months of progression-free survival.

Primary mediastinal dedifferentiated liposarcoma resected by lateral thoracotomy with video-assisted thoracoscopic surgery.

Primary dedifferentiated liposarcoma originating in the mediastinum is extremely uncommon. Here, we report a case of mediastinal dedifferentiated liposarcoma. A 74-year-old man was admitted to our hospital with a left intrathoracic tumor ∼10 cm in diameter. Computed tomography that was taken in another hospital 2 years before the initial visit to our hospital had shown a lipomatous tumor ∼3 cm in diameter adjacent to the left lower pulmonary vein. The tumor was resected by left lateral thoracotomy with video-assisted thoracoscopic surgery. Pathological examination revealed primary mediastinal dedifferentiated liposarcoma. No evidence of recurrence has been seen as of 8 months postoperatively.

Shadow cell differentiation from squamoid morule in endometrial adenoacanthoma.

Shadow cell differentiation (SCD), commonly found in cutaneous pilomatricoma (PMX), has been said to be extremely rare in extracutaneous tumors and its morphogenesis has not been clarified yet. In the present study, 25 cases of endometrial adenoacanthoma were examined with special reference to SCD and with immunohistochemistry for beta-catenin and CD10. Shadow cell nests (SCNs) were observed in 2 out of 5 cases of adenocarcinoma with squamoid morules and all of 4 cases of adenocarcinoma with squamous differentiation and morules, but not in any cases of adenocarcinoma with squamous differentiation. SCNs were just adjacent to morules with or without a mutual transition. Immunohistochemical examination revealed nuclear accumulation of beta-catenin and expression of CD10 in the squamoid morules around SCNs. These results indicate that SCNs are derived from squamoid morules in endometrial adenoacanthoma, and established a link between matrical basaloid cells in PMX and squamoid morules in endometrial adenoacanthoma, as common original tissues, showing nuclear accumulation of beta-catenin and expression of CD10, of SCNs. It seems that SCD is not so uncommon as previously estimated in endometrial adenoacanthoma.

Bladder carcinoma with shadow cell differentiation: a case report with immunohistochemical analyses.

A peculiar case of bladder carcinoma showing shadow cell differentiation (SCD) in a 72-year-old man is presented. The tumor histologically revealed high grade urothelial carcinoma (UC) and partially contained squamous component with a transition to shadow cell nests, similar to those seen in cutaneous pilomatricoma (PMX). Immunohistochemically, the modes of cell death in the component of SCD were identical to those in PMX. The present case as well as 10 cases of cutaneous PMX showed nuclear expression of beta-catenin, whereas 10 cases of bladder UC with squamous differentiation revealed membranous localization without nuclear expression. These results suggest that nuclear accumulation of beta-catenin may play an important role for SCD in the present case. SCD in extracutaneous tumor is extremely rare and, in the literature, the present case is the second one as for bladder carcinoma.