Unilateral pleural effusion secondary to Takayasu arteritis: a case report and literature review

Takayasu arteritis (TA) is a chronic, nonspecific inflammatory disease of large and medium-sized vessels that primarily involves the aorta and its branches. TA involving the pulmonary arteries has a prevalence ranging from 14% to 86%, which can lead to pulmonary hypertension, a progressive increase in pulmonary artery pressure, and eventually death from right heart failure. The presentation of pulmonary arteritis (PA) is very nonspecific, with a reported misdiagnosis rate of up to 60% and a diagnosis time ranging from 1 month to more than 10 years. The clinical manifestation of pleural effusion is very rare in both TA and PA cases. Based on our literature review, this is the 6^th reported case of TA with pleural effusion, and the specific mechanism of TA with pleural effusion is still unclear. The characteristics of this case and the previously reported cases are summarized in this article to improve the understanding of TA and PA and reduce the misdiagnosis rate.     

变异硬脑膜窦1例

笔者在解剖一老年男性尸体发现其右侧有一变异硬脑膜窦,起自蝶骨小翼下方,向外至蝶骨小翼末端弯曲向后,贴颞骨鳞部水平向后走行注入横窦末端.该硬脑膜窦走行中接受大脑中浅静脉和颞叶桥静脉的注入.     

An autopsy case report of annuloaortic ectasia with cardiac tamponade ruptured from an aneurysm of the right Valsalva sinus

Annuloaortic ectasia (AAE) is a clinicopathologic condition with primary or secondary dilatation of the aortic annulus and aneurysm of the proximal thoracic aorta, leading to aortic regurgitation. We herein report an autopsy case of a Japanese 57-year-old male with AAE who died of a cardiac tamponade rupture from the sinus of the right coronary. The wall of the aortic root, particularly that of the sinus of the right coronary Valsalva, underwent extensive fibrosis with loss or fragmentation of the elastic lamina in the medial layer and perforation directly into the pericardial space. The adventitia of the proximal aorta to the aortic arch was diffusely fibrotic with both acute and chronic hemorrhage and chronic inflammatory infiltrate. However, the ascending aortic media was largely intact, except for focal laminar necrosis at the center of the medial layer; no medial cystic necrosis, laminar necrosis, or mesoaortitis/panaortitis was present in the thoracic or abdominal aorta, nor in the main aortic branches, which was suggestive of Takayasu disease and giant cell arteritis. Thus, this patient was diagnosed to have idiopathic AAE with sustained peri-aortic hemorrhage, and he finally died of a cardiac tamponade resulting from an aneurysmal rupture.     

An autopsy case of aortic dissection due to giant cell arteritis

Giant cell arteritis (GCA) is a systemic vasculitis affecting mainly large and medium-sized arteries. GCA sometimes involves the aorta and its major branches and causes aortic dissection as a rare complication. We have experienced an autopsy case of aortic dissection due to GCA. The patient was an 87-year-old Japanese woman with Stanford type A aortic dissection who died 7 days after admission. Two years previously she had been diagnosed as having abdominal aortic aneurysm and undergone endovascular aneurysm repair (EVAR). Although she had no characteristic symptoms of GCA, autopsy revealed marked granulomatous inflammation in the dissected area and coronary arteries. Active arteritis was evident not only in the arteries of the upper extremity but also those in the lower extremity. Granulomatous inflammation was not evident in the aneurysm. The aortic dissection might have been an initial manifestation of GCA. We report the regions of GCA extension and its histology in detail.     

Endodermal sinus tumour of stomach--a case report

This is a case report of an endodermal sinus tumour arising in the stomach of a 1 1/2 years male child. The tumour occupied full thickness of fundus and part of the body of the stomach. H&E sections of the tumour tissue showed endodermal sinus tumour with predominantly reticular and endodermal sinus patterns. Elevated level of alpha-fetoprotien was detected in the serum of the patient and within the tumour tissue by immunohistochemical technique.     

Deep femoral artery aneurysm presenting as lower limb swelling: a case report

We report a rare case of lower limb swelling due to compression of the superficial femoral vein by a solitary deep femoral artery aneurysm. The patient was a 58-year-old man presenting with acute swelling of the right lower limb caused by deep venous thrombosis. A multi-detector computed tomographic scan (CT) confirmed the diagnosis of a deep femoral artery aneurysm and revealed no evidence of aneurysms or occlusive lesions in the other arteries. Aneurysmectomy and ligation of the deep femoral artery were successfully performed. Preoperative multi-detector CT scanning is a valuable, non-invasive diagnostic tool to delineate not only the state of the aneurysm, but also that of the distal arterial tree.     

Giant intrasellar aneurysm presenting with panhypopituitarism and subarachnoid hemorrhage: case report and literature review

A 52-year-old woman was admitted to our hospital presenting with subarachnoid hemorrhage, left ophthalmoplegia, and right hemiparesis. Previous symptoms and signs suggested the presence of panhypopituitarism. A giant intracranial aneurysm of the internal carotid artery, diagnosed by magnetic resonance imaging, showed the characteristic flow void phenomenon with black appearance. Analysis of endocrine function disclosed panhypopituitarism and hyperprolactinemia. After proximal balloon occlusion of the aneurysm, diabetes insipidus developed. Pituitary function reassessed 6 months after initial admission confirmed anterior and posterior hypopituitarism and hyperprolactinemia. Possible mechanisms are discussed. A review of the literature on pituitary dysfunction caused by carotid artery aneurysms discloses that the pituitary-gonadal axis is the most frequently involved (67.5% of cases), followed by the pituitary-adrenal axis (48.6%) and the pituitary-thyroid axis (40.5%). These frequencies are very similar to those described in other types of hypopituitarism.Abbreviations ICA internal carotid artery - MRI magnetic resonance imaging - SAH subarachnoid hemorrhage Correspondence to: J.M. Fernandez-Real     

A unique autopsy case of ascending aortic dissection caused by giant cell arteritis without drug therapy

Giant cell arteritis is a granulomatous inflammation of large and medium‐sized arteries, occurring predominantly in older women. In this case, a 76‐year‐old woman was hospitalized for examination because of a high C‐reactive protein (CRP) level, but nothing remarkable was found on thoracicoabdominal computed tomography (CT) or head magnetic resonanse imaging (MRI). On the 46th day from the first visit, she died suddenly due to cardiac tamponade. On pathological autopsy, we found the cause of death to be acute aortic dissection (Stanford type A) due to giant cell arteritis occurred in the ascending aorta. Histologically, granulomatous vasculitis with giant cells was recognized in the ascending aorta, thoracic descending aorta and abdominal aorta and their branches. Interestingly, similar granulomatous vasculitis was also found in the medium and small vessels of other plural organs, including the heart, liver, uterine corpus, and its appendages. To our knowledge, giant cell arteritis with multiple‐organ granulomatous changes has not been reported before. We herein reported a unique autopsy case of giant cell arteritis in a patient not treated with medication.     

Myxoma of the renal sinus: case report and literature review

Myxoma is a rare mesenchymal tumor and it is mainly seen in heart and skin. Renal myxoma is extremely rare. To date, eleven cases of kidney myxomas have been reported in the literature. One of them is myxoma of the renal sinus. Our case was an 82-year-old man admitted to our hospital symptoms related to the urinary tract obstruction. Abdominal computerized tomography revealed a solid, hypodense mass 9 cm in diameter infiltrating the renal parenchyma in the renal pelvis. The patient underwent nephrectomy. The resected kidney contained gelatinous tumor with indistinct borders. The tumor was composed of slender, bland, spindle-shaped cells with large amounts of mucoid material. Tumor cells were positively stained with vimentin, focally stained positive for smooth muscle actin and had negative reactivity for S-100 protein, epithelial membrane antigen and pancytokeratin. Herein we report the second case of renal myxoma arising from the renal sinus.     

Clear cell adenocarcinoma of the maxillary sinus: a case report

A variety of salivary gland tumours histologically show the presence of clear cells. Clear cell adenocarcinoma of salivary glands (CCASG) is a distinct clinico-pathologic entity arising in the minor salivary glands of mainly the oral cavity in elderly women and having a low grade malignant potential. A case occurring in the maxillary sinus and extending to the nasal cavity is reported.     

Rothia dentocariosa , endocarditis and mycotic aneurysms: case report and review of the literature

Rothia dentocariosa is a rare cause of endocarditis. It occurs most frequently in patients with prior heart conditions. Although the clinical course is typically subacute, it has a high rate of complications. In particular, the reported incidence of mycotic aneurysms is as high as 25%. Penicillin is the treatment of choice, but additional complications may necessitate prompt surgical intervention. As far as we know, this paper reports the first case of repeated subarachnoid hemorrhages due to R. dentocariosa endocarditis.     

上腔静脉伴冠状窦异常1例

在解剖一具成人男性尸体标本时，观察其心脏，见上腔静脉位置与走行及冠状窭异常,报道如下。     

Familial hypercholesterolemia with unusual foamy histiocytes. Report of a case with myelophthisic anemia and xanthoma of the maxillary sinus

Accumulation of phagocytic histiocytes with a foam cell morphology has been described in a number of diseases. Familial hypercholesterolemia, one such disease, is characterized by foamy histiocytic accumulation in cutaneous or tendinous xanthomas and within atheromatous lesions. This report describes a patient with familial hypercholesterolemia who had two unusual manifestations of foamy histiocytic accumulation: a maxillary sinus xanthoma, which presented as an expansile mass, and diffuse bone marrow replacement with foamy histiocytes, which was associated with myelophthisic anemia. The accumulation of foamy cells in both locations resembled that seen in many of the storage diseases. The possibility of foamy histiocytic accumulation should be considered in the differential diagnosis of patients with these disease entities who present with space-occupying lesions.     

Fine-needle aspiration cytology of sinus histiocytosis with massive lymphadenopathy: A case report

Cytological findings of sinus histiocytosis with massive lymphadenopathy (SHML), involving bilateral cervical lymph nodes of a 12-yr-old boy, are presented here. A striking feature was the presence of large histiocytes with abundant pale cytoplasm containing well-preserved lymphocytes, plasma cells, and occasional neutrophils, scattered in a background of reactive lymphoid cells. Subsequent open biopsy of the lymph node mass confirmed the cytologic findings. Cytologic features of SHML are virtually diagnostic and can negate the need for an open biopsy in most cases. Diagn Cytopathol 1996;15:221–223. © 1996 Wiley-Liss, Inc.     

Sick sinus syndrome associated with hypopituitarism：a case report and literature review

Though an association between autoimmune diseases and sick sinus syndrome has been reported,there has been no report on the association of hypopituitarism and sick sinus syndrome.Herein,we provide the first case report of hypopituitarism accompanying sick sinus syndrome in a 51-year-old woman presented to our hospital with syncope due to cardiac arrest.The patient was successfully managed by pacemaker installation and hormone replacement therapy.