糖皮质激素联合硫唑嘌呤治疗复发性淋巴细胞性垂体炎三例报道

报道3例复发性淋巴细胞性垂体炎(LYH)患者均为女性,其中病例2为老年女性.病例1主要累及垂体前叶,并侵犯海绵窦和视交叉,病例2则主要累及下丘脑和漏斗部,病例3则是垂体前叶和垂体柄均受累.病例1手术后4个月第一次复发,给予大剂量甲基泼尼龙治疗(HDMPT)有效,但停药后14个月再次复发;病例2中等剂量强的松治疗停药16个月后复发,而病例3则在HDMPT治疗,糖皮质激素减量的过程中复发.3例患者复发后均以糖皮质激素联合硫唑嘌呤治疗16周,疗效良好,停药后随访3例患者,MRI显示LYH均无复发,而且病例1和3垂体功能正常.

Abstract：

All three cases of recurrent lymphocytic hypophysitis were female, one of them being 70-yearold postmenopausal woman.Adenohypophysis, cavernous sinus, and optic chiasm were involved in case 1,hypothalamus and neurohypophysis were invaded in case 2, and adenohypophysis and hypophyseal stalk were involved in case 3.Relapse occured 4 months after operation in case 1, then high dosage methylprednisolone pulse therapy (HDMPT) brought about a remission lasting for 14 months before second relapse set in.Relapse occurred in case 2 at 16 months after prednisone treatment was discontinued, and case 3 recurred during the period of dose reduction.All three patients were then treated with glucocorticoid plus azathioprine for 16 weeks, and good response was seen in 3 cases.During follow-up, the symptoms were relieved and significant reduction of lesion was revealed by MRI in all thee patients, and the pituitary function was resumed in case1and 3.     

Hemorrhagic pseudocyst and pseudocyst with pseudoaneurysm successfully treated by pancreatectomy: report of three cases

Hemorrhagic pseudoaneurysm of pancreatic pseudocyst is one of the serious complications of acute pancreatitis. We successfully treated three patients who had hemorrhagic pseudocyst and pseudocyst with pseudoaneurysm by pancreatectomy. Case 1 was 43-year-old Japanese man who had had several episodes of acute pancreatitis and was diagnosed with hemorrhagic pseudoaneurysm of the splenic artery in a pseudocyst in the pancreatic tail, shown on computed tomography (CT) and angiography. Transarterial embolization (TAE) yielded hemostasis of the pseudoaneurysm, but rebleeding occurred 2 weeks after the TAE. Distal pancreatectomy and splenectomy was successfully performed. Case 2 was a 64-year-old Japanese man who presented to us with several attacks of acute pancreatitis. Imagings showed bleeding pseudoaneurysm of the transverse pancreatic artery in a pseudocyst in the pancreatic body. Because of marked stenosis in the proximal portion of the transverse pancreatic artery, TAE was unsuccessful. Distal pancreatectomy and splenectomy was performed successfully. Case 3 was a 40-year-old Japanese woman who had a history of abdominal trauma. Imagings showed bleeding pseudoaneurysm of the splenic artery in a posttraumatic pseudocyst in the pancreas. TAE of the pseudoaneurysm was unsuccessful because of the proximity of the pseudoaneurysm and the splenic artery. Distal pancreatectomy and splenectomy was successfully performed and her postoperative outcome was satisfactory. Whenever interventional radiology (IVR) is not indicated or has failed, aggressive and immediate surgical intervention should be considered for early and definitive recovery in these patients.     

糖皮质激素成功治疗原发性肉芽肿性垂体炎一例报道并文献分析

原发性肉芽肿性垂体炎( primary granulomatous hypophysitis,PGH)是垂体炎的一种,极其罕见,约占手术确诊蝶鞍病变的1％[1],以弥漫分布多核巨细胞为特征,临床表现为垂体功能减退和蝶鞍占位所致的症状,明确诊断需要病理学检查.大多数报道对糖皮质激素治疗无明显疗效[2,3],本文报道1例经手术活检病理确诊的PGH患者用糖皮质激素成功治疗前后的激素和垂体影像学变化,并结合文献探讨PGH的诊断和治疗.     

Pulmonary hyalinizing granuloma with laryngeal and subcutaneous involvement: report of a case successfully treated with glucocorticoids

We report a case of pulmonary hyalinizing granuloma (PHG) with laryngeal and subcutaneous involvement. A 43-year-old man was admitted to our hospital for assessment of hoarseness. Cervical and chest computed tomography, respectively, revealed a laryngeal tumor and two pulmonary masses. Specimens obtained from the pulmonary masses were compatible with PHG. The histopathology of biopsy specimens from both the laryngeal tumor and a subcutaneous tumor resembled that of the resected lung masses. Although there is no established treatment for PHG, the laryngeal tumor was diminished and all other lesions disappeared with glucocorticoid treatment.     

淋巴细胞性垂体炎三例并文献复习

目的提高临床医师对淋巴细胞性垂体炎的认识。方法分析3例淋巴细胞性垂体炎的临床特点和病理结果。结果3例患者均为青年女性,例1产后起病,例2和例3在非妊娠或产后期间发病;影像学特点:病变广泛,不仅局限于鞍区和垂体柄,并向下丘脑扩展呈“舌状”改变侵犯海绵窦;增强后病变均匀或环状强化;主要临床表现:(1)垂体功能受损和尿崩症;(2)有局部受压症状,如头痛,视交叉受压;(3)病变侵犯海绵窦,引起动眼、外展神经麻痹;2例经病理证实为淋巴细胞性垂体炎,垂体组织见大量淋巴细胞、浆细胞、组织细胞浸润;2例大剂量甲泼尼龙治疗后,症状明显好转。结论淋巴细胞性垂体炎亦可发生在非妊娠或产后期间的年轻女性,大剂量甲泼尼龙冲击治疗能有效缩小病变和改善垂体功能。     

Adult autoimmune enteropathy treated successfully with tacrolimus

BACKGROUND: Autoimmune enteropathy is a life-threatening, chronic disease of the small bowel mucosa, which generally responds well to steroids. Treatment requires long-term immunosuppression, and steroid-sparing treatment strategies are desirable. Azathioprine and cyclosporine A have limitations, however alternatives have not been described in adults. CASE REPORT: We present the case of a 54-year-old male patient with autoimmune enteropathy who responded initially to a standard treatment with steroids, but was dependent on 30 mg prednisolone. Medical treatment was changed to tacrolimus after renal function deteriorated under treatment with cyclosporine A. Under this regimen, small bowel histology normalized and the clinical condition is stable after 2 years of introduction of tacrolimus. CONCLUSION: This constitutes the first report of effective treatment of adult autoimmune enteropathy with tacrolimus, a substance with a similar mode of action to cyclosporine, but with fewer side effects and improved bioavailability.     

Primary chronic autoimmune neutropenia successfully treated with sirolimus

We report a 74-year-old man with severe chronic primary neutropenia (neutrophil count: 390 per millimeter cube) uncovered following surgery for perianal abscess collection. Clinical, laboratory and roentgenographic findings revealed no abnormality. Antineutrophil antibodies were positive in two consecutive serum samples. Under cyclosporine, neutrophil count reached 1970 per millimeter cube. However, this therapy was discontinued due to new onset of severe renal failure. After six weeks, neutrophil count was 950 per millimeter cube and sirolimus was started, resulting in renal function improvement and resolution of neutropenia.     

Idiopathic autoimmune hemolytic anemia successfully treated with danazol

A 50 year old female suffered from idiopathic autoimmune hemolytic anemia. She was initially treated with prednisolone, and anemia was improved moderately. However, maintenance of Hb value was difficult with the decreased dosage of prednisolone. She was subsequently administered danazol, and anemia was completely improved. It is worth to use danazol for the treatment of the patient of autoimmune hemolytic anemia who is refractory to prednisolone therapy.     

Chronic cytopenia associated with T8 lymphocytosis successfully treated with glucocorticoids

We reported a prednisolone effective case of T 8 lymphocytosis. The patient was 83 years old male, who had suffered from hypoplastic anemia for more than 5 years before he admitted to our hospital. His blood picture was peculiar because adding to granulocytopenia, CD8 (+) large granular lymphocytes were dominant among his lymphocytes. A few days after administration of bolus methylprednisolone, prominent reticulocytosis and rapid improvement of anemia occurred and the latter continued for more than 1 year. We performed in vitro study on the mechanism of his cytopenias. The result of Southern blot analysis showed that his blood lymphocytes had a unique rearranged pattern of T cell receptor beta chain gene. Mononuclear cells had an inhibitory effect on autologous and allogenic CFU-E colony formation of bone marrow cells. These facts suggest that colony proliferating CD8 (+) lymphocytes had an inhibitory effect on erythropoiesis in vivo, and prednisolone blocked this inhibitory pathway in this patient.     

Hypoplastic leukemia successfully treated with low-dose aclarubicin: a case report

Low-dose aclarubicin (LDACR) therapy is one of the differentiation induction therapy, such as low-dose cytosine arabinoside therapy, 1 alpha, 25 dihydroxy-vitamin D3 or retinoic acid therapy, for myelodysplastic syndrome and atypical leukemias. A 36-year-old female with hypoplastic acute myelogenous leukemia was treated with this atypical leukemias. A 36-year-old female with hypoplastic acute myelogenous leukemia was treated with this LDACR therapy. On admission, she was suffered from general fatigue and her peripheral blood smear showed pancytopenia with 23% of myeloblasts. Bone marrow examination revealed a moderately hypoplastic marrow with 35.4% of myeloblasts. She was diagnosed as having hypoplastic acute myelogenous leukemia. Thereafter, 20 mg of aclarubicin was given daily by one-shot intravenous injection for 10 days. After this LDACR therapy, myeloblasts disappeared from her peripheral blood and pancytopenia improved. Bone marrow examination showed increase in nuclear cell counts and she achieved complete remission. In this article, we report the clinical course of this patient and discuss the effect of LDACR therapy as useful chemotherapy for this patient.     

Ipilimumab: a novel immunomodulating therapy causing autoimmune hypophysitis: a case report and review

Ipilimumab (Yervoy; Medarex and Bristol-Myers Squibb) is a human MAB against cytotoxic T-lymphocyte antigen 4, which enhances co-stimulation of cytotoxic T-lymphocytes, resulting in their proliferation and an anti-tumour response. It is licensed for the treatment of unresectable or metastatic malignant melanoma, while multiple clinical trials using this medication in the treatment of other malignancies are ongoing. As a clinical response to ipilimumab results from immunostimulation, predictably it generates autoimmunity as well, causing immune-related adverse events in the majority of patients. Of those, endocrinopathies are frequently seen, and in particular, autoimmune lymphocytic hypophysitis with anterior panhypopituitarism has been reported a number of times in North America. We present a case of a male referred to our department with manifestations of anterior panhypopituitarism after his third dose of ipilimumab for metastatic malignant melanoma, and we discuss the management of his case in the light of previous reports. We also review the published literature on the presenting symptoms, time to presentation, investigations, imaging, treatment and follow-up of ipilimumab-induced autoimmune lymphocytic hypophysitis.     

甲基强的松龙联合硫唑嘌呤治疗复发性自身免疫性下丘脑炎

自身免疫性下丘脑炎是一种罕见的自身免疫性疾病.患者主要临床表现为垂体前叶功能减退和尿崩症,影像学检查显示下丘脑占位性病变,糖皮质激素治疗有效.近年来国外只有1例报道[1].我们最近收治1例老年女性自身免疫性下丘脑炎,报告如下.     

Recurrent refractory Clostridium difficile colitis treated successfully with rifaximin and tigecycline: a case report and review of the literature

Clostridium difficile colitis infection is on the rise and is considerably increasing the duration of hospital stay, as well as healthcare costs. The management of C. difficile colitis has become more challenging with the increasing failure of therapeutic response to metronidazole and oral vancomycin. Tigecycline is a new glycylcycline that has shown in vitro activity against C. difficile. We report herein a case of C. difficile colitis that failed to improve on a combination of metronidazole and oral vancomycin. The patient subsequently developed a surgical abdomen secondary to refractory C. difficile colitis, but was successfully treated with a combination of rifaximin and tigecycline after she refused to undergo surgical treatment.     

Recurrent hepatocellular carcinoma successfully treated with radiofrequency thermal ablation

We report a patient with hepatocellular carcinoma (HCC) who was successfully treated with radiofrequency thermal ablation (RFA). A 71-year-old man was admitted to our hospital in August 1996 with recurrence of HCC. Partial hepatic resection had been performed in January 1993 for HCC that had measured 1.3 cm in segment VIII, and subsequently he had received six sessions of percutaneous ethanol injection (PEI) for treatment of recurrence. Dynamic computed tomography (CT) performed in August 1996 showed two recurrent tumors, one measuring 3.8 cm in segment VIII adjacent to the right hepatic vein, and one measuring 2.0 cm in segment V. Three sessions of percutaneous RFA were performed. After this treatment, most of the tumor in segment VIII and all the tumor in segment V showed low density on dynamic CT, and the right hepatic vein was preserved. However, a remnant of the mass appeared near the right hepatic vein 2 months after the treatment. An additional two sessions of RFA were performed. After the end of treatment, serum alpha-fetoprotein level dropped to the normal range, and no sign of recurrence has been observed until September 1998.