A case of pituitary mass suggestive of lymphocytic adenohypophysitis

A 29-year-old woman suffered from frontalgia with vomiting and visual field defects in the 9th month of pregnancy. Two weeks after delivery, a pituitary mass with suprasellar extension was demonstrated on computed tomography (CT). Angiogram revealed elevation of bilateral A1 segments and right persistent primitive trigeminal artery. Repeated CT performed three months after delivery showed that the pituitary mass had undergone a spontaneous regression in size, and her visual impairment improved. The pituitary function tests revealed no response to ACTH, GH and PRL stimulation performed about a year after delivery. Laboratory data showed high titer of antinuclear antibody and low titer of complement. This case suggests that some patients with postpartum hypopituitarism suggestive of lymphocytic adenohypophysitis, and with pituitary mass need not have early surgical intervention but may be closely observed and treated by hormone replacement alone.     

A case of sarcoidosis associated with membranous nephropathy

A 59-year-old woman was admitted to the hospital with a complaint of right visual disturbance. At admission, she had proteinuria and microscopic hematuria. The pulmonary X-ray showed bilateral hilar lymphadenopathy (BHL) but tuberculin test were negative. Also, bronchoalveolar lavage (BAL) and renal biopsy were performed. Bronchoalveolar lavage fluid (BALF) contained a high percentage of OKT 4 positive cells. In renal biopsy, light microscopic study showed thickening glomerular basement membrane (GBM) with tubulo-interstitial legion. Immuno-fluorescent study revealed granular deposits of IgG along glomerular capillary wall. Electron microscopic study showed subepithelial electron dense deposits of GBM, which are characteristic for membranous nephropathy (MN). It is well known that both sarcoidosis and MN are associated with immunological abnormality. This case might be significant for the study of pathogenesis of sarcoidosis.     

A case of thoracic vertebral tuberculosis associated with pulmonary sarcoidosis

A 56-year-old woman was admitted to our hospital, on October 10, 1999 because of tingling numbness of the Th3 area and paraparesis. Her medical history included the diagnosis of pulmonary sarcoidosis which had responded to steroid therapy. On radiographic examination, a Th2 compression fracture and gadolinium enhanced areas were shown in MRI. We diagnosed vertebral sarcoidosis and restarted steroid therapy. Next day, she had no paresis and she was discharged after 20 days. However, the patient returned to our hospital with recurrence of the same pain and paraparesis on January 1, 2000, and spinal fusion of the Th2-3 was performed by a transpleural anterolateral approach using a block of iliac bone as a graft. Histology and cultural studies of tissue revealed tuberculosis. There is a diagnostic dilemma, "sarcoidosis or tuberculosis?", but it is necessary to be aware of this at the time of the initial diagnosis.     

A case of mitral stenosis associated with cardiac sarcoidosis and left ventricular aneurysm

We report a rare case of mitral stenosis associated with cardiac sarcoidosis and left ventricular aneurysm. A 60-year-old female was admitted with complaint of palpitation and dyspnea on exertion. Chest X-ray revealed cardiomegaly and pericardial calcification, but no bilateral hilar lymphadenopathy. Electrocardiogram showed various arrhythmia. Coronary angiography showed no stenosis in any coronary arteries. Cardioangiogram showed left ventricular aneurysm at the apex and mitral stenosis. Laboratory findings showed no evidence of sarcoidosis. Open mitral commissurotomy and left ventricular aneurysmectomy was performed. Pathological findings of the myocardium showed a remarkable degenerative change and a granulomatous inflammation. Postoperative biopsied specimens of right scalene lymph nodes revealed sarcoid reaction. The postoperative course was satisfactory, but arrhythmia remained. The patient was treated with steroids and pacemaker implantation. She has been doing well for 2 year postoperatively. An operative case of mitral stenosis associated with cardiac sarcoidosis and ventricular aneurysm due to sarcoidosis is very rare and the prognosis in patient is very poor usually.     

Preoperative diagnosis of lymphocytic hypophysitis (adenohypophysitis) unresponsive to short course dexamethasone: case report.

Lymphocytic hypophysitis (adenohypophysitis) is a rare lymphocytic infiltration of the pituitary gland that usually occurs during pregnancy. Because of its rarity, it has seldom been diagnosed preoperatively, and no trials of therapeutic treatment have been reported to date. We describe a 29-year-old woman with a pituitary mass and visual-field defects during pregnancy. The patient's pituitary profile revealed an abnormal thyroid axis and relatively low prolactin for her stage of pregnancy. This finding suggested adenohypophysitis, and the patient was given a trial course of dexamethasone. The progression of the visual-field defects, however, indicated that the steroids, both in dosage and duration, were not effective. Thus, the patient underwent a partial hypophysectomy for decompression. The pathology report confirmed adenohypophysitis, and steroids were continued for the remainder of the pregnancy, with slow resolution of the visual-field defects to normal. This report is the first case of adenohypophysitis where the diagnosis was suspected preoperatively, and a trial course of steroids was given. The steroids at the dosage and duration used were not effective. Further evaluation of the use of steroids in this disease is warranted.     

A case of primary lung cancer associated with diffuse panbronchiolitis

Pulmonary resection was performed for primary lung cancer of a 72-year-old patient which was associated with diffuse panbrochiolitis that required home oxygen therapy. The preoperative total pulmonary vascular resistance at rest was 564 dyne.sec.cm-5/m2, but thoracotomy and reduction surgery were possible by strict respiratory and circulatory control. The troublesome postoperative pulmonary phthisis was dealt with through extended control by means of total parenteral nutrition and use of the Heimlich valve.     

Carcinoma of the renal pelvis and bladder associated with sarcoidosis: a case report

A 75-year-old woman with transitional cell carcinoma of the renal pelvis was revealed to have sarcoid granulomas within the kidney as well as in the renal pedicle lymph nodes. Furthermore, noncaseating granulomas had been found in the pelvic lymph nodes in a histological study following previous total cystectomy for bladder cancer. The association of sarcoidosis with genitourinary malignancies is briefly discussed.     

A case of sarcoidosis overlapping with rheumatoid arthritis

We report the case of a 54-year-old woman who presented on May 28, 2001 with sarcoidosis overlapping with rheumatoid arthritis. She had experienced morning stiffness 2 years previously and was diagnosed as having rheumatoid arthritis. She had been treated with bucillamine and loxoprofen for 3 months. In October 2000, she developed proteinurea. The patient discontinued treatment with bucillamine and loxoprofen. Proteinurea persisted, and the patient's renal function declined. On admission, subcutaneous nodules were palpable in the patient's legs. The patient's serum creatinine and calcium levels were 2.49 mg/dl and 11.6 mg/dl, respectively. Intact-PTH was suppressed, and PTHrP was not elevated. Despite the presence of hypercalcemia, the patient's serum 1 alpha 25(OH)2D3 was not suppressed. Serum ACE and lysozyme levels were elevated beyond the normal ranges. A renal biopsy was performed, and non-caseous epithelioid granuloma was found in the renal interstitium. Based on the histological findings, the patient was diagnosed as having sarcoidosis. Following treatment with prednisolone, the patient's serum calcium levels returned to normal and her renal function improved.     

A case of epididymal sarcoidosis

Sarcoidosis is a systemic inflammatory condition that primarily affects the lungs, lymphatic system and skin. Extra thoracic manifestations occur in about 50% of cases; however, it rarely affects the male reproductive tract. The first documented case of epididymal and testicular sarcoid in the Irish literature is of a 25-year-old male who presented with a painful 1-cm extra-testicular scrotal mass. While the involvement of the male reproductive tract is rare, it should be included in the differential of testicular pathology. It can be diagnosed on frozen section and may be managed conservatively.     

A case of pneumonectomy for lung cancer associated with scleroderma]

A 60-year-old female, diagnosed as scleroderma, was referred to our hospital because of symptoms of common cold and abnormal findings on a chest X-ray. The chest X-ray and CT scan revealed a mass in the left upper field, suspected to invade left anterior chest wall. Moreover, fibrotic changes were observed in the lung field. Cytology by bronchofiberscopy showed squamous cell carcinoma. Evaluation of pulmonary function, including unilateral pulmonary artery occlusion test (UPAO), revealed possibility of lung resection. Subsequently, pneumonectomy with combined resection of left 1st rib was performed. Postoperative course was uneventful and she was discharged. She was admitted again four months after the operation with appetite loss and body weight loss. Further examinations revealed arrhythmia, renal failure, pancreatitis and liver metastasis. Her general conditions grew worse and she died five months after the operation. In conclusion, UPAO was a useful method to determine the functional lung resectability for the case with scleroderma. However, effects of surgical stress for the development of scleroderma remain to be elucidated.     

Lymphocytic adenohypophysitis: MRI findings of a suspected case

We report a case of a 26-year-old woman who had developed decrease of visual acuity, and restriction of the temporal visual field of the left eye in the 30th week of gestation. A skull roentgenogram showed no abnormality, but a pituitary mass was visualized by plain computed tomographic scan. Magnetic resonance imaging (MRI) demonstrated a symmetric sellar mass which had homogeneous signal intensity in all pulse sequences. T1 relaxation time of the mass was elongated as compared with that of a normal pituitary gland. The height of the mass was 11mm, and optic chiasm appeared compressed by the mass. Her visual disturbance improved before delivery, and the size of the pituitary mass regressed spontaneously. Although no histological examination was carried out, the most likely explanation for this phenomenon is lymphocytic adenohypophysitis. Early surgical intervention is not required for lymphocytic adenohypophysitis, because this disorder may be self-limiting, and may resolve itself. Most of these cases have been reported in women, often coincident with pregnancy. So we consider that MRI is the most useful and safest method for diagnosis and follow-up of this disease.     

Lung cancer associated with sarcoidosis

Sarcoidosis is often identified as swollen hilar lymph nodes found in chest radiography during routine physical checkups. We report a patient with concomitant sarcoidosis and lung cancer necessitating thoracoscopic lymph node biopsy to differentiate between sarcoidosis and lung cancer as the cause of mediastinal/hilar lymph node swelling prior to conducting pulmonary lobectomy. Thoracoscopic biopsy of left mediastinal lymph nodes and sampling of frozen sections of right lymph nodes during intraoperative diagnosis did not detect metastasis. A permanent pathological slide, however, indicated that right interlobar (#11s) lymph nodes involved both sarcoidosis and lung cancer metastasis. Despite careful preparation, it thus remains difficult to distinguish between these conditions even when lymph nodes are strictly evaluated.     

Hepatocellular carcinoma associated with sarcoidosis

INTRODUCTION

The association of hepatic sarcoidosis with hepatocellular carcinoma (HCC) is considerably rare. Here we report a rare case of HCC associated with sarcoidosis.

PRESENTATION OF CASE

A 75-year-old male with no history of alcohol addiction or viral hepatitis was referred to our hospital because of an abnormal liver mass. Subsegmentectomy of the liver was performed for the diagnosis of HCC. A histopathological examination revealed small non-necrotizing granulomas with a tendency to coalesce that were scattered in and around the carcinoma. No features of cirrhosis, steatohepatitis, and any other liver diseases were observed. Furthermore, swelling of the bilateral lung hilar lymph nodes with uptake of 18F-fluorodeoxyglucose was found on positron emission tomography/computed tomography and the tuberculin reaction test results were negative. On the basis of these findings, the final diagnosis of HCC associated with sarcoidosis was confirmed.

DISCUSSION

By reviewing previous cases, we found only five cases that described patients diagnosed with HCC associated with sarcoidosis. Of these, four patients died within two years after diagnosis because of ruptures or inoperable huge tumors. In contrast, radical hepatectomy was performed at an earlier stage of disease in two patients, including ours, and both these patients have remained healthy with no recurrences or metastases at the latest follow-up visit.

CONCLUSION

Periodic checkups of the liver should be conducted for patients with systemic sarcoidosis, regardless of the presence of liver cirrhosis.     

A case of subdural hematoma associated with dural metastasis of lung cancer

A 62-year-old male developed headache, restlessness and left hemiparesis three months after being diagnosed with advanced lung cancer. Computed tomography on admission revealed a crescent-shaped, mixed intensity area in the right fronto-parietal subdural region and multiple tumors in the brain parenchyma. Under a diagnosis of chronic subdural hematoma and multiple brain metastases due to lung carcinoma, burr hole irrigation was performed. Adenocarcinoma cells were found in the dura matter and hematoma. Nontraumatic chronic subdural hematoma secondary to dural metastasis is a very rare condition. Only 52 cases of such spontaneous subdural hematoma have been reported. We describe the clinical features and discuss the mechanism referring to the pertinent literature.     

A case of left lung cancer associated with an aneurysm of the thoracic aorta

A 61-year-old man was admitted to our hospital because of a left lung cancer. The chest x-ray film showed an irregular mass in the left upper lung field and the ill-defined left upper mediastinal border. A large portion of the aorta seen in the CT section above the aortic arch was understood to be aortic elongation. When a left pneumonectomy was performed, a saccular aneurysm of the distal aortic arch was found and resected under partial aortic clamping. Following the aneurysmectomy mediastinal dissection was performed in the normal way. The patient recovered uneventfully. The pathological specimens showed a pT2N1M0 squamous cell carcinoma with obstructive pneumonia and an arteriosclerotic aneurysm. There was no report of lung cancer associated with aneurysm of the thoracic aorta. In a patient with left lung cancer obliterating the left upper mediastinal border (the "silhouette sign") the aortic arch should be closely examined by MRI and/or angiography.     

A case of dural sarcoidosis]

A 26 year-old man, who was treated for meningitis in our hospital previously, was rehospitalized 1 year later because he developed disturbance of consciousness, gait disturbance and urinary incontinence. Blood examination revealed accelerated ESR, elevated GPT, slight elevation of serum Ca, strong positive CRP, and a decrease in PHA and Con A. ACE was within normal range and tuberculin reaction was negative. Lumbar puncture revealed that the initial pressure was 310 mmH2O, cells were 152/3, and protein was 343 mg/dl. Bilateral hilar lymphadenopathy was absent in chest X ray film. Head CT revealed enlarged lateral ventricles and irregularly enhancing nodular lesions in the anterior half of the falx cerebri, and abnormally strong enhancement of the choroid plexus. Ventriculoperitoneal shunt was performed. As a diagnosis was difficult to obtain from the clinical data, biopsy of the nodular lesions was performed. The histopathologic diagnosis was sarcoidosis. Steroid hormone was administered thereafter, and the nodular lesions of the falx disappeared in the follow-up. In the literature, only 8 cases of sarcoidosis of the dura mater have been reported. Since intracranial sarcoidosis is a part of systemic sarcoidosis, its diagnosis is not difficult in most cases. However, in cases difficult to diagnose as in our case, biopsy may be necessary. When nodular lesion occurs in the dura mater, sarcoidosis must be included as a possibility in the differential diagnosis in addition to the usual meningioma, lymphoma, and metastatic brain tumor.     

Lymphoid sarcoidosis with axillary localization. A clinical case

Following the case of a patient affected by bilateral axillary sarcoidosis of the lymph nodes the Authors of this paper have carried out a study with particular attention to the histological diagnosis, and making use of both optic and electron microscopy. The case here presented did not have the typical pattern of sarcoidosis. We underline that it was sarcoidosis of the lymph nodes in a patient with no evidence of lung alteration (Chest X-Rays were negative). It is rare case on the basis of world surveys.     

A case for steroids in acute lung injury associated with the retinoic acid syndrome

A 62-year-old woman with acute promyelocytic leukaemia was treated with all-trans retinoic acid. On day 2 she suffered with dyspnoea and general fatigue. Marked hypoxia suggested the occurrence of retinoic acid syndrome. She underwent endotracheal intubation and mechanical ventilation with the administration of dexamethasone. Her symptoms promptly abated. She was subsequently treated with conventional chemotherapy and achieved complete remission.