Rapidly Growing Chest Wall Lipoma: Case Report and Calculating Volume Doubling Time.

Chest wall lipoma is a rare disease that might be asymptomatic and discovered incidentally. Chest wall lipomas are presumed to grow slowly, but no reports have evaluated the tumor volume doubling time (TVDT). The present study herein reports the case of a 35-year-old female patient with a relatively fast-growing chest wall lipoma. Lipomas have their characteristic shape and grow very slowly, so they are rarely completely resected, even though they are monitored and repeated imaging studies are performed. Homogeneous very low density, clear margins, and no invasion to the surrounding structure are characteristic finding on imaging, but some patients without these characteristics here have been reported here. As there has been no report of TVDT for chest wall lipoma, comparison was not possible, but TDVT for lipoma in this patient ranged from 235-412 days. Compared with reports that patients with non-small cell lung cancer showed TVDT of less than 450 days, TVDT in the patient described here did not appear to be slow. Accumulation of knowledge about this rare disease will help to elucidate it further.

Continuous glucose monitoring in a patient with insulinoma presenting with unawareness of postprandial hypoglycemia.

Summary: Unawareness of postprandial hypoglycemia for 5 years was identified in a 66-year-old man at a local clinic. The patient was referred to our hospital because of this first awareness of hypoglycemia (i.e. lightheadedness and impaired consciousness) developing after lunch. In a 75 g oral glucose tolerance test, the plasma glucose concentration was decreased to 32 mg/dL (1.8 mmol/L) at 150 min with relatively high concentrations of insulin (8.1 µU/mL), proinsulin (70.3 pmol/L), and C-peptide (4.63 ng/mL). In a prolonged fasting test, the plasma glucose concentration was decreased to 43 mg/dL (2.4 mmol/L) at 66 h with an insulin concentration of 1.4 µU/mL and a C-peptide concentration of 0.49 ng/mL. Computed tomography showed an 18 mm hyperenhancing tumor in the uncinate process of the pancreas. A selective arterial calcium stimulation test showed an elevated serum insulin concentration in the superior mesenteric artery. The patient was then diagnosed with insulinoma and received pancreaticoduodenectomy. Continuous glucose monitoring (CGM) using the Dexcom G6 system showed unawareness of hypoglycemia mainly during the daytime before surgery. When the sensor glucose value was reduced to 55 mg/dL (3.1 mmol/L), the Dexcom G6 system emitted an urgent low glucose alarm to the patient four times for 10 days. Two months after surgery, an overall increase in daily blood glucose concentrations and resolution of hypoglycemia were shown by CGM. We report a case of insulinoma with unawareness of postprandial hypoglycemia in the patient. The Dexcom G6 system was helpful for assessing preoperative hypoglycemia and for evaluating outcomes of treatment by surgery. Learning points: Insulinoma occasionally leads to postprandial hypoglycemia. The CGM system is useful for revealing the presence of unnoticed hypoglycemia and for evaluating treatment outcomes after surgical resection. The Dexcom G6 system has an urgent low glucose alarm, making it particularly suitable for patients who are unaware of hypoglycemia.

Gastrocolic fistula caused by transverse colon cancer: a case report.

BACKGROUND: A gastrocolic fistula is an unusual communication between the colon and the stomach. Although colon cancer is the most common malignant cause of gastrocolic fistula in the Western world, the incidence of gastrocolic fistula due to colon cancer is 0.3% in operated cases. CASE PRESENTATION: A 68-year-old man presented with anorexia, general malaise, weight loss, and vomiting of fecal matter. Investigations revealed that the patient had a large nonmetastatic splenic flexure tumor that was diagnosed as colon cancer and had invaded the stomach and pancreas. An upper gastrointestinal series confirmed a gastrocolic fistula. Left hemicolectomy, distal gastrectomy, distal pancreatectomy, and splenectomy were performed. Histology revealed transverse colon cancer, which was UICC stage (8th edition) pT4bN1bcM0 pStage IIIC. Adjuvant chemotherapy was not performed. There was no recurrence or metastasis one year after surgery. We reviewed 17 cases including our case of a gastrocolic fistula caused by colon cancer. Neoadjuvant chemotherapy was not given to any of the patients, and en bloc resections were conducted in all cases. Adjuvant chemotherapy was given to almost all of the patients. There was no recurrence or metastasis. CONCLUSIONS: For gastrocolic fistula caused by advanced colon cancer, secure en bloc surgical resection was the initial treatment in all 17 reported cases including the present case, and adjuvant chemotherapy may contribute to a better prognosis.

Recurrent distal cholangiocarcinoma and humoral hypercalcemia of malignancy: report of a rare case and literature review.

A 61-year-old Japanese woman presented with epigastric pain and jaundice. Imaging showed the presence of primary distal cholangiocarcinoma (DCC). A subtotal stomach-preserving pancreaticoduodenectomy was performed, followed by chemotherapy using S-1. However, second-line chemotherapy with gemcitabine and cis-diamminedichloroplatinum was required for the treatment of hepatic metastasis of the DCC 3 months following the surgery. Nine months after the surgery, the serum calcium and parathyroid hormone-related peptide concentrations were high, at 16.5 mg/dL and 28.7 pmol/L, respectively, which suggested the presence of humoral hypercalcemia of malignancy (HHM) secondary to the DCC. Moreover, marked leukocytosis, with a white blood cell count of 40,400/µL, was also present. The patient died 11 months after the diagnosis of DCC. Because hypercalcemia of malignancy is associated with a poor prognosis, and HHM and leukocytosis caused by DCC are very rare, we have presented the present case in detail and provide a review of the existing literature.

A Malignant Tumor Developed Seven Years After Small Cell Lung Cancer Treated With Etoposide-containing Chemotherapy.

BACKGROUND/AIM: Long-term survival of patients with small cell lung cancer (SCLC) is rare, and, to the best of our knowledge, there has been no SCLC patient who developed second malignancy after long-term survival. CASE REPORT: A 66-year-old woman with a history of smoking was admitted to our hospital with a nodule in her right lung. She was diagnosed with cT2aN3M0 localized-SCLC. Chest irradiation and chemotherapy including etoposide was performed. A new nodule appeared in the right lung more than 7 years after the end of treatment for SCLC. A specimen obtained by bronchoscopic biopsy was pathologically confirmed to be a non-SCLC malignancy. CONCLUSION: There is a possibility of tumor development associated with etoposide, which is known to be carcinogenic, or residual tumor development from combined type SCLC. We could not confirm whether it was second malignancy or recurrence after long-term interval. The number of long-term survivors of SCLC is likely to increase in the future. The clinical course of this patient is interesting from the perspective of long-term survival of SCLC patients and might have implications for the treatment of patients with similar clinical course in the future.

Rare Presentation of Angiolipoma Affecting an Intact Achilles Tendon.

Musculoskeletal lipomatous lesions are common in soft tissues. However, these are rarely associated with tendon sheaths or tendon compartments. Moreover, angiolipoma of the Achilles tendon is yet to be described. Here, we report an angiolipoma of an intact Achilles tendon, which has not been described previously. A 54-year-old woman presented with a two-year history of a palpable mass in the posterior aspect of the left ankle. The mass caused an intermittent localized pain while walking and a catching phenomenon induced by the plantar dorsiflexion movement of the ankle joint. Magnetic resonance imaging revealed a well-circumscribed, oval lesion on the lateral aspect of the Achilles tendon. The location and shape of the lesion had changed over time, suggesting that the lesion was moving in and out around the Achilles tendon. At the surgery, the tumor was confirmed under the crural fascia. Histopathological examination revealed that the tumor comprised mature adipocytes covered peripherally with a fibrovascular capsule. Based on these features, the tumor was diagnosed as an angiolipoma. Angiolipomas are typically treated surgically by simple excision, and lipomatous lesions of the tendon sheath are not different. From this case report, angiolipomas are rare but should be considered in the differential diagnosis and treatment of Achilles tendon tumors.

Serial assessments of anterior pituitary hormones in a case of mixed histiocytosis representing Langerhans cell histiocytosis overlapping with Erdheim-Chester disease.

SUMMARY: A 61-year-old man developed central diabetes insipidus caused by mixed histiocytosis (MH) representing Langerhans cell histiocytosis overlapping with Erdheim-Chester disease. Bone, skin, vascular, and retroperitoneal involvements were also observed. Dynamic hormonal testing showed normal responses for anterior pituitary hormones, except for impaired secretion of growth hormone (GH). MRI of the brain showed thickening of the pituitary stalk with slightly reduced signal hyperintensity in the posterior pituitary lobe on T1-weighted imaging. During 2 years of follow-up without radical treatment for MH, imaging studies suggested extension of vascular and retroperitoneal involvements. In contrast, brain MRI did not show any particular interval changes, except for the disappearance of hyperintense signalling in the posterior pituitary lobe. Moreover, no other anterior pituitary dysfunctions beyond GH deficiency emerged during the 2 years of follow-up. The natural history of MH in this case is described, focusing on serial assessments of pituitary functions using dynamic tests. LEARNING POINTS: Erdheim-Chester disease and Langerhans cell histiocytosis overlapping as MH was described, focusing on pituitary functions. MH caused both GH deficiency and central diabetes insipidus. Despite a lack of radical therapy for MH, no other anterior pituitary dysfunctions emerged for 2 years. Radiological images showed no particular interval changes in pituitary stalk lesions, while vascular and retroperitoneal involvements extended.

Negative-pressure pulmonary Hemorrhaging Due to Severe Obstructive Sleep Apnea.

A 24-year-old man with a history of bloody sputum for 6 months was referred to our hospital with suspected alveolar hemorrhaging due to vasculitis. Chest computed tomography showed ground-glass opacities in both lungs, and an examination of his bronchoalveolar lavage fluid showed alveolar hemorrhaging. However, no evidence of vasculitis was found, and subsequent polysomnographic testing confirmed that he had severe obstructive sleep apnea (OSA). Since the alveolar hemorrhaging improved after the initiation of continuous positive airway pressure treatment, the diagnosis was negative-pressure alveolar hemorrhaging due to severe OSA.

Pancreatic panniculitis in a patient with pancreatic adenosquamous carcinoma.

Pancreatic panniculitis is a rare complication of pancreatic diseases. We aimed to evaluate a case of pancreatic panniculitis. A 58-year-old woman was referred to our hospital with complaints of painful cutaneous nodules on her limbs. Various diagnostic tests confirmed pancreatic panniculitis and pancreatic adenosquamous carcinoma. We diagnosed pancreatic panniculitis by a skin nodule biopsy that revealed fine basophilic material within anucleate cells and neutrophil infiltration. Abdominal imaging detected a tumor with necrosis on the pancreas and endoscopic ultrasound-guided fine-needle aspiration revealed it as an adenocarcinoma. The patient underwent pancreatoduodenectomy after neoadjuvant chemotherapy. The tumor was composed of differentiated adenocarcinoma and squamous cell carcinoma and diagnosed as adenosquamous carcinoma. This is the first report of pancreatic panniculitis in a patient with adenosquamous cell carcinoma of the pancreas.

Metachronous Isolated Contralateral Lung Metastasis from Pulmonary Adenosquamous Carcinoma with EGFR Mutation.

Lung metastasis and metachronous double primary lung cancer are both common and often present diagnostic challenges. We present a case of metachronous isolated contralateral lung metastasis from pulmonary adenosquamous carcinoma with EGFR mutation. A 75-yearold woman presented with left lung nodule on a routine follow-up chest radiograph. She had had surgery for pulmonary adenocarcinoma with EGFR Ex21 L858R mutation 6 years ago. She underwent surgical resection, and histologic findings revealed adenosquamous carcinoma with the same EGFR mutation. Re-assessment of the resected specimen of the primary tumor resected 6 years ago revealed the morphologically similarity to the left lung tumor. Based on morphological and genetic identity, final diagnosis was adenosquamous cell carcinoma and metachronous isolated contralateral lung metastasis. The diagnosis of metachronous isolated metastasis is difficult but important for appropriate management and prediction of prognosis. A careful pathological examination and evaluation of genetic abnormality are needed to make the correct diagnosis.

Combination of Pembrolizumab With Platinum-containing Chemotherapy for Pleomorphic Carcinoma of the Lung.

BACKGROUND/AIM: Pleomorphic carcinoma of the lung is a rare, highly malignant subtype of lung cancer, with a more aggressive clinical course compared with other types of non-small-cell lung cancer (NSCLC). Platinum-containing chemotherapy has been the standard therapy for patients with NSCLC and pembrolizumab is one of the novel and reliable agents for these patients. CASE REPORT: We herein report the case of a 60-year-old man with advanced chemo-naïve pleomorphic carcinoma of the lung who was successfully treated with a combination of pembrolizumab with platinum-containing chemotherapy. CONCLUSION: In the absence of definitive clinical trials, which are unlikely to be performed due to the rarity of this tumor, our case demonstrates the potential utility of the combination of pembrolizumab with platinum-containing chemotherapy. Our result also suggest that this combination of therapy may be key to the treatment of pleomorphic carcinoma of the lung.

Challenges in treatment of disseminated nocardiosis in an elderly patient with renal failure on corticosteroids: a case report.

We report the case of a 71-year-old Japanese man with a history of chronic kidney disease and sarcoidosis receiving chronic corticosteroids who presented with disseminated Nocardia brasiliensis infection. He initially showed improvement with empiric antimicrobial therapy including trimethoprim-sulfamethoxazole. However, he deteriorated after modifying the empiric regimen due to complicated hyperkalemia and ultimately died. In general, elderly patients have decreased renal function. Standard therapy for nocardiosis with trimethoprim-sulfamethoxazole may not be used for a prolonged period of time. This case emphasizes the challenges and importance of prudent selection of empiric antimicrobial therapy for disseminated nocardiosis in elderly patients with underlying kidney disease.

Pleural fluid due to papillary thyroid cancer.

We report herein a rare case of massive pleural effusion caused by papillary thyroid cancer, which was accompanied by multiple pulmonary metastasis. A 91-year-old male patient presented with shortness of breath due to massive right pleural fluid. Cytological specimens, which were obtained from pleural fluid by thoracentesis, and was consistent with that observed in surgically resected thyroid cancer 6-year previously. Immunocytochemical staining of the cells was positive for cytokeratin (CK)-7, CK-19, and positive for thyroglobulin. Massive pleural fluid due to a metastatic from papillary thyroid cancer is very rare but may develop in long-term survivors with this disease as observed in this case.

Meningeal carcinomatosis presenting with leukoencephalopathy-like imaging findings.

Meningeal carcinomatosis is a unique and rare form of metastasis observed in patients with malignant tumours. Diagnosis is simple when the primary lesion of the malignant tumour is clear, and when multiple miliary lesions are confirmed via cranial contrast MRI; however, many patients exhibit atypical imaging findings. In the present report, we discuss the case of a 72-year-old man who presented with subacute consciousness impairment and MRI findings suggestive of progressive, bilateral leukoencephalopathy-like lesions around the ventricles. Idiopathic hydrocephalus was initially suspected due to increased cerebrospinal fluid (CSF) pressure accompanied by normal cell counts. Although the patient underwent a ventriculoperitoneal shunt operation, his symptoms did not improve. Whole-body CT revealed findings suggestive of adenocarcinoma in the left lung. Paraneoplastic syndrome was suspected, and he was treated with three courses of high-dose intravenous methylprednisolone. However, his neurological symptoms did not improve, and he died 2 months after admission. The patient was ultimately diagnosed with meningeal carcinomatosis due to lung adenocarcinoma upon autopsy. In this case, we suspected that the white matter lesions observed on MRI resulted from secondary hydrocephalus due to obstruction of the CSF circulation. This is the first reported case of progressive leukoencephalopathy-like imaging findings in a patient with meningeal carcinomatosis.

Chronic expanding hematoma in the chest: A case report.

Chronic expanding hematoma (CEH) is a rare disease that is usually present as a large solitary pulmonary nodule. CEHs are slow growing, but processes underlying their development remain unknown. The present study herein reports the case of a 76-year-old male patient with CEH and discusses a number of CEH cases published in the literature. The majority of these previously described patients were Asians. The CEH in the present case was not a successfully resected one, but the patient's clinical course provided information concerning the natural history of the disease. During the clinical course, the patient underwent several chest computed tomography scans. For the present case report, the doubling time and volume change of the mass was calculated, which revealed that the lesion had an inconstant growth rate and that its onset was between 8.2-11.0 years before the patient succumbed to this disease. Accumulation of knowledge about this rare disease will help to elucidate it further.

A case of microscopic, multiple sclerosing pneumocytoma.

Sclerosing pneumocytoma is a rare tumor of the lung, commonly affecting middle-aged women, and is mostly isolated. Although this tumor is thought to be derived from primitive respiratory epithelial cells, the characteristics of the precursor cells are still unknown. A 19-year-old woman presented with multiple nodules in the right lung. Partial resection of the right middle lobe was performed, and seven sclerosing pneumocytomas, including four that were microscopic, were detected. The latter showed a simple papillary pattern, and three of them consisted of only round cell-like cells (single population). Interestingly, these round cell-like cells were positive for both p63 and TTF-1, but totally negative for SP-A. On the other hand, the tumor cells of the other four sclerosing pneumocytomas showing a papillary pattern with a dual population, were diffusely positive for TTF-1 and focally positive for SP-A (only in surface cells), but negative or very focally positive for p63. It has been reported that p63-positive stem cell-like cells are present in the distal airway and have potential to differentiate into type II pneumocytes. The immunohistochemical features of these multiple microscopic lesions suggest that the p63-TTF-1 double-positive cells are candidate precursor cells of sclerosing pneumocytoma.

Pulmonary metastasis from uterine leiomyosarcoma in a patient with limited cutaneous systemic scleroderma.

A 51-year-old woman, who was diagnosed as having limited cutaneous systemic scleroderma, presented with pulmonary nodules incidentally detected in a chest radiograph. The patient had surgical biopsy of the nodules. In microscopic examination of the specimens, proliferation, mitotic activity, and cellular anaplasia of spindle cells were present. Fluorodeoxyglucose-positron emission tomography showed tumors in lungs as well as uterus. The diagnosis of the tumor was pulmonary metastases from uterine leiomyosarcoma. We should be on alert the possibility of developing malignant disease in patient with this autoimmune disease. If it is certain that there is metastasis, we believe that therapy for the primary lesion will be preceded by biopsy and surgery for the metastatic lesions.