A case of urolithiasis due to vitamin D intoxication in a patient with idiopathic hypoparathyroidism

We report a case of urolithiasis in a patient with idiopathic hypoparathyroidism treated with vitamin D therapy. A 30-year-old woman with idiopathic hypoparathyroidism, who had been treated with vitamin D therapy with 2-4 micrograms/day of alpha-calcidol for 9 years, was admitted for recurrence of bilateral renal stones and progressing left hydronephrosis. Laboratory data revealed normal serum calcium level and remarkable hypercalciuria. The dose of oral administration of alpha-calcidol was reduced to 1 microgram/day and 2 mg/day of trichlormethiazide was started. Now her serum calcium concentration and the total amount of urine calcium was completely under control. Bilateral renal stones are no longer progressive and tetany has not been recognized. We considered it essential to monitor closely not only the serum but also the urine calcium level in the vitamin D therapy for idiopathic hypoparathyroidism.     

Successful treatment of a patient with a 13-year history of post-traumatic rhinorrhea due to malabsorption of cerebrospinal fluid.

Chronic cerebrospinal fluid (CSF) leaks associated with skull base fractures are rare but intractable and patients may be subjected to numerous operations. We present a 30-year-old man with a 13-year history of chronic CSF rhinorrhea following a cranial trauma. Computed tomography (CT) showed a bone defect in the planum sphenoidale. CT cisternography revealed a leak from the defect and CSF malabsorption. The absence of symptoms of CSF malabsorption may be attributable to external leakage of excess CSF. After closing the leak via the extended transsphenoidal approach we placed a ventriculoperitoneal shunt for occult hydrocephalus. We discuss the clinical symptoms of chronic CSF leakage and present therapeutic strategies dictated by the mechanisms underlying the leak.     

Successful surgical treatment of a patient with multiple visceral artery aneurysms due to fibromuscular dysplasia

Multiple visceral artery aneurysms due to fibromuscular dysplasia are rare. A 43-yr-old man with a pulsatile abdominal mass detected by ultrasonography had multiple visceral artery aneurysms diagnosed by angiography. This included a huge superior mesenteric artery aneurysm. Aneurysm resection and arterial reconstruction was performed successfully. Pathologic examination revealed fibromuscular dysplasia of the medial fibroplasia type.     

Successful treatment of a patient with low pressure syndrome associated with gait disturbance

The authors report an unusual case of low intracranial pressure (ICP) syndrome that was successfully treated by the placement of an anti-siphon device (ASD). This 36-year-old male had suffered suprasellar germinoma with hydrocephalus and had had a V-P shunt following radiotherapy. Sixteen years later he developed gait disturbance and somnolence and MRI demonstrated a small lateral ventricle as well as a diffuse dural enhancement. A lumbar tap revealed a low ICP of 12 mmH2O. Because of this, an ASD was placed in the patient. Postoperatively, his symptoms of gait and consciousness disturbance improved. Typical clinical findings of low ICP syndrome such as headache were not observed in this case. To our knowledge, no symptom of gait disturbance with low ICP has been reported previously. We present an interesting case of low ICP syndrome with gait disturbance and discuss the mechanism of the symptoms. Symptoms of this patient were due at first to brain ischemia. After convulsion and consciousness disturbance due to low intracranial pressure, the symptoms increased in strength until gait disturbance occurred. The possibility is suggested that gait disturbance in this patient was due both to brain ischemia and low intracranial pressure.     

Vitamin D-related progressive renal insufficiency in an elderly patient with postsurgical hypoparathyroidism associated with extensive brain calcification

We present a 76-year-old female patient with dementia who has postsurgical hypoparathyroidism associated with extensive brain calcification and progressive renal insufficiency. She had been treated with vitamin D combined with calcium or vitamin D alone due to hypoparathyroidism for 8 years. However, intermittent hypercalcemia including hypercalcemic crisis (serum Ca 15.2 mg/dL) and progressive renal dysfunction had developed. This patient was transferred to our long-term care hospital because of worsening dementia. Since laboratory data at admission revealed hypercalcemia and azotemia, alfacalcidol (1 microg/day) was discontinued. However, severe hypocalcemia (3.9 mg/dL) occurred later, while her azotemia was improved. With a low dose of alfacalcidol(0.25 microg/day), the serum calcium level is now below normal (approximately 7.0 mg/dL). There is neither hypocalcemic symptom nor exacerbation of renal insufficiency. From the clinical history of recurrent hypercalcemic episodes and renal calculi observed on computed tomography, the progression of renal insufficiency was considered to be related to persistent hypercalciuria caused by vitamin D and calcium, especially vitamin D therapy.     

Successful treatment in a patient with Takayasu's arteritis and Marfan syndrome

We report an unusual case of concomitant Takayasu's arteritis and Marfan syndrome manifesting left main coronary ostial obstruction and annuloaortic ectasia. Simultaneous surgical treatment consisting of left coronary ostium endarterectomy, coronary artery bypass grafting, and Bentall operation was performed. This case is unique in that the cardiovascular manifestations of Takayasu's arteritis and Marfan syndrome were both simultaneously presented and surgically treated.     

Successful treatment by cyclooxyenase-2 inhibitor of refractory hypokalemia in a patient with Gitelman's syndrome

Gitelman's syndrome is manifested by hypokalemic alkalosis, hypomagnesemia, hypocalciuria, normotensive hyperreninemia and hyperaldosteronism. Hypokalemia can at times be refractory to treatment. We present a patient refractory to a variety of drugs including indomethacin, the nonspecific COX inhibitor. Rofecoxib, a specific COX 2 inhibitor, promptly elevated serum potassium concentration with normalization of plasma aldosterone and near normalization of renin without a change in serum magnesium. Our patient also had rhabdomyolysis, a rarely reported complication, which was also ameliorated by COX 2 inhibition.     

肌注维生素D2治疗维持性血液透析患者25羟维生素D缺乏的临床观察

目的分析维持性血液透析患者的血清25-羟维生素D[25(OH)D]水平,探讨肌注维生素D2注射液治疗25羟维生素D缺乏的作用。 方法检测我院2020年12月至2021年2月170例维持性血液透析患者空腹血清25(OH)D水平,根据血清25(OH)D水平将患者分为严重缺乏组、缺乏组、不足组和正常组。分析血清25(OH)D水平与患者性别、年龄、透析龄、血红蛋白、白蛋白、血钙、血磷、全段甲状旁腺激素(iPTH)的相关性。其中86例维生素D缺乏或不足的血透患者,随机分为对照组(n＝43)和治疗组(n＝43),对照组给予常规治疗,治疗组在对照组基础上给予肌注维生素D2 (20万单位,1次/2周),连续3个月。比较患者治疗前后血清25(OH)D、血红蛋白、白蛋白、钙、磷、iPTH水平的变化,并观察不良反应情况。 结果170例患者中,维生素D严重缺乏组33例(19.4%),缺乏组33例(19.4%),不足组70例(41.2%),正常组34例(20%)。男性25(OH)D水平显著高于女性(P<0.05)。正常组与维生素D缺乏组和不足组在透析龄、血红蛋白、白蛋白差异方面均具有统计学意义(P<0.05)。血清25(OH)D与年龄、血钙、血磷、甲状旁腺激素无显著相关性(P>0.05)。多元线性逐步回归分析显示,血清25(OH)D与血红蛋白、白蛋白具有相关性(P<0.05)。肌注维生素D2 3月后可使血液透析患者的25(OH)D水平显著上升(P<0.05),同时血红蛋白、血清白蛋白、磷上升(P<0.05),对血钙、iPTH无显著影响(P>0.1)。 结论维持性血液透析患者维生素D缺乏发生率高,血清25(OH)D水平与性别、贫血、营养不良存在密切关系,肌注维生素D2可改善血透患者贫血、营养不良情况,但可能带来血磷升高。     

腹膜后巨大血肿伴腹腔高压综合征及多器官功能衰竭抢救成功1例

患者男,30岁.被汽车撞伤后2h出现烦躁,腹痛、腹胀.体检:血压90/60mmHg,心率120次/min.双肺呼吸音粗,未闻干湿啰音.全腹压痛反跳痛,肠鸣音弱.诊断性腹穿阴性.     

Successful steroid treatment in a patient with membranoproliferative glomerulonephritis associated with hepatitis C virus

Thirteen years ago, a 65-year-old woman was diagnosed to have chronic active hepatitis with hepatitis C virus. After starting interferonα administration, she noticed edema and hypoalbuminemia. Renal biopsy revealed mesangial proliferation with focal endocapillary proliferation, and double contour of the glomerular basement membrane due to mesangial interposition. Interferonα was discontinued, and proteinuria and edema gradually decreased. She was re-admitted due to a relapse of proteinuria 8 years later. Biopsy revealed moderate mesangial and endcapillary proliferation presenting a lobular pattern, in addition to the presence of hyaline thrombi. Granular staining of immunoglobulin M and of C3 in capillary walls were detected. Since cryoglobulinemia was positive, a final diagnosis of cryoglobulinemic membranoproliferative glomerulonephritis was made. Prednisolone was started with an initial dose of 20 mg/day. Proteinuria and hypoalbuminemia improved, and prednisolone was tapered to 5 mg/day 9 months after the 2nd renal biopsy. The hepatitis C virus-RNA titer fluctuated.