Hodgkin's lymphoma initially presenting with polymyalgic symptoms: a case report

The association of polymyalgic symptoms and lymphoma is a rare event whose pathogenesis remains to be clarified. Here, we describe a case of a 75-year old man with Hodgkin's lymphoma, who had presented with polymyalgic symptoms suggesting polymyalgia rheumatica. An intensive investigation with respect to malignancy was initially negative. Corticosteroid treatment was administered first and a dramatic clinical improvement was achieved. Four months later, when the corticosteroid treatment was tapered off, the initial manifestations reappeared. After the development of lymph node enlargement, the patient was diagnosed by lymph node biopsy as having Hodgkin's lymphoma. The lymphadenopathy and musculoskeletal manifestations all responded well to chemotherapy. Hodgkin's lymphoma should be considered in the differential diagnosis of PMR. These musculoskeletal syndromes should alert the physician to possible paraneoplastic manifestations of an evolving neoplasm.     

以肺栓塞为首发表现的高嗜酸粒细胞综合征一例并文献复习

目的 分析高嗜酸粒细胞综合征伴多发血栓形成的临床特征,提高临床对嗜酸粒细胞增多导致血栓性疾病的认识.方法 以“嗜酸粒细胞增高、血小板减少、深静脉血栓形成”为中文关键词,“hypereosinophilic,thrombocytopenia,deep vein thrombosis"为英文关键词检索2002年1月-2012年1月万方数据库和Pebmed数据库的相关文献,共获得9篇,其中中文5篇,英文4篇,排除未明确诊断的2篇文献,共纳入7篇,报道了8例患者,其中男5例,女3例,年龄24-75岁,结合文献报道的病例特点及本病例特征进行文献复习.结果 患者男,27岁,以胸痛气短伴痰中带血起病,经胸部强化CT确诊肺栓塞,同时有下肢深静脉血栓形成,实验室检查提示血嗜酸粒细胞增高和血小板下降.结合文献嗜酸粒细胞增高可损伤血管内皮细胞,导致多发动静脉血栓形成,抗凝同时应用糖皮质激素治疗有效.结论 高嗜酸粒细胞综合征是嗜酸粒细胞增多症的一种,临床以多器官受累为表现,伴多发血栓形成,抗凝治疗同时应用皮质激素可降低嗜酸粒细胞计数,减少其血管内皮损伤对血栓形成的影响.     

Malignant lymphoma of spleen presenting as acute pancreatitis： A case report

This is a case report of a patient who presented with acute pancreatitis without the common causes. A pancreatic biopsy revealed large B cell lymphoma. Spleen lymphoma with pancreatic involvement inducing acute pancreatitis, which is a rare disorder, was diagnosed. Here we also review the few similar cases reported in the literature.     

A rare case of multiple myeloma initially presenting with pseudoachalasia

Pseudoachalasia is a rare clinical entity with clinical, radiographic, and manometric features often indistinguishable from achalasia. Primary adenocarcinomas arising at the gastroesophageal junction or a tumor of the distal esophagus are the most frequent causes of pseudoachalasia. Rarely, processes other than esophagogastric cancers including chronic idiopathic intestinal pseudo-obstruction, amyloidosis, sarcoidosis, Chagas' disease, vagotomy, antireflux surgery, pancreatic pseudocysts, von Recklinghausen's neuroinomatosis, gastrointestinal stromal tumor, and other malignancies and rare genetic syndromes, may lead to the development of pseudoachalasia. Secondary achalasia is extremely rare, with less than 100 cases reported in the literature so far. Gastrointestinal manifestations in primary or secondary amyloidosis include abdominal pain, diarrhea, constipation, malabsorption, obstruction, motility disturbance, intestinal infarction, perforation, and hemorrhage; however, gastrointestinal tract involvement is asymptomatic in most instances. We present here a rare case of multiple myeloma initially presenting with dysphagia because of esophageal amyloidosis and manometric findings typical of achalasia.     

A case of relapsing polychondritis initially presenting with bronchial chondritis

Abstract

A 51-year-old woman presented with one-month history of fever, productive cough, dyspnea, hoarseness and polyarthritis. Computed tomography (CT) depicted diffuse bronchotracheal stenosis, which deteriorated in exhalation. Three-dimensional CT revealed airway stenosis from the trachea to the lobar bronchi. We made a diagnosis of relapsing polychondritis (RP). Administration of high-dose glucocorticoid and oral cyclophosphamide resolved the symptoms and the bronchial stenosis. It was noteworthy that all features presented were bronchial chondritis and polyarthritis. RP should be considered when patients have polyarthritis with respiratory symptoms. The dynamic expiratory CT and three-dimensional image reconstructions are useful for detecting and following up bronchotracheal involvement.     

A rare case of Gitelman's syndrome presenting with hypocalcemia and osteopenia

Gitelman's syndrome (GS), an autosomal recessive disorder caused by a defect of the thiazide-sensitive Na-Cl cotransporter (TSC) at the distal tubule, is characterized by hyperreninemic hyperaldosteronism with normal or low blood pressure, hypokalemia, metabolic alkalosis, hypomagnesemia and hypocalciuria. An 18-yr-old Japanese man was admitted to our hospital with a history of muscle weakness and transient tetanic episodes. He showed hypocalcemia in addition to hypokalemia, severe hypomagnesemia, hypocalciuria and hyperreninemic hyperaldosteronism with normal blood pressure. Furthermore, bone mineral density at the lumbar spine revealed osteopenia. A diagnosis of GS was made on the basis of clinical features, laboratory data and renal function test. The electrolyte imbalance was corrected and bone mineral density was slightly increased with chronic treatment of magnesium and potassium salts. Genetic analysis revealed that TSC gene of the patient has a heterozygous C to A nucleotide substitution at position 545 in exon 4, which causes a threonine (Thr) to lysine (Lys) substitution at position 180. This is a rare case of GS with hypocalcemia and osteopenia which could be caused by severe hypomagnesemia.     

Transient infantile hypertriglyceridemia with jaundice: A case report

Rationale:Transient infantile hypertriglyceridemia (HTGTI) is a rare autosomal recessive inherited disease caused by inactivating mutations in the glycerol-3-phosphate dehydrogenase 1 gene. To date, only a few patients have been reported worldwide. The symptoms of the affected individuals present a certain degree of transient hypertriglyceridemia, hepatomegaly, elevated liver enzymes, persistent fatty liver and hepatic fibrosis in early infancy. However, the clinical characteristics and pathogenesis of this disease are remain unclear.Patient concerns:A one month and twenty-five days old girl was admitted to hospital because of persisted jaundice and hepatomegaly for fifty days.Diagnose:The girl was diagnosed with HTGTI coincident with a noval mutation in glycerol-3-phosphate dehydrogenase 1.Intervention:She was advised to take low-fat diet and supplement of medium-chain fatty acids.Outcomes:Her jaundice was gradually normal at the age of 4 months without any treatment, and hypertriglyceridemia were normal at the age of 13 months, but still had elevated transaminases and hepatic steatosis.Lessons:Jaundice may be a novel phenotype in HTGTI. The report contributes to the expansion of HTGTI''s gene mutation spectrum and its clinical manifestations.     

Ectopic pancreas presenting with intractable diarrhea: case report

Ectopic pancreas is an uncommon congenital anomaly, which is usually found incidentally in clinical practice. It presents with nonspecific gastrointestinal symptoms like epigastric pain and dyspepsia and rarely with the clinical findings of obstructive jaundice or intestinal obstruction, or it may mimic gastrointestinal system cancer. Herein, we describe a case of ectopic pancreas in the duodenum, which was the cause of the intractable diarrhea. In our patient, upper gastrointestinal endoscopy and endoscopic ultrasonography revealed a 1.5 cm submucosal lesion, which was umbilicated centrally with a normal in appearance overlying mucosa. Endoscopic biopsy of the lesion was normal. Pathological examination of the lesion after surgical excision was compatible with ectopic pancreas. After total excision of the lesion, the clinical findings of the patient normalized. Ectopic pancreas presenting with diarrhea has not been reported previously in the literature.     

Pseudohypoparathyroidism presenting with ventricular arrhythmia: a case report

Pseudohypoparathyroidism (PHP) is a rare disorder characterized by varying degrees of unresponsiveness to parathyroid hormone. Patients usually present with hypocalcemia-induced seizures or tetany, whereas no case of hypocalcemia-induced cardiac arrhythmia in PHP has been described to date. In this paper, we report the case of a male adolescent with PHP type 1a who presented with hypocalcemia-induced ventricular extrasystoles (bigeminy, trigeminy) and mild corrected QT interval prolongation. The patient had brachydactyly and his second fingers and toes were longer than the others, a finding consistent with PHP. Laboratory tests detected hypomagnesemia, as well as elevated levels of creatine kinase and lactate dehydrogenase. Ventricular arrhythmia and abnormal laboratory tests improved with calcium supplementation and vitamin D treatment. The findings in this patient suggest that hypomagnesemia may make patients with PHP more susceptible to hypocalcemia and may thus prompt a state of hypocalcemia-induced arrhythmia or other cardiac complications.     

Sarcoidosis presenting with Raynaud's syndrome: a case report

We report a case of association between sarcoidosis and Raynaud's syndrome. A 39 year old female presented fatigue, Raynaud's syndrome, IgG and erithrosedimentation rate (ESR) increase, polyarthralgy in which disseminated micronodular infiltration in the chest X-ray and histological demonstration of non-caseating epitheloid microgranulomas led to a diagnosis of concomitant sarcoidosis. Clues to the diagnosis of sarcoidosis coexisting with autoimmune disease are discussed.     

Case report: hypoparathyroidism with menses-associated hypocalcemia.

For 18 years, a patient with idiopathic hypoparathyroidism has experienced stiffness or tetany chiefly at the time of menses. A significant decline (0.2 mmol/L) in her serum total and ionized calcium values was observed after the withdrawal of oral contraceptive and after spontaneous onset of menses. No increase in urinary calcium excretion or decline in serum magnesium or calcitriol occurred to explain the fall in serum calcium. In two hypoparathyroid women without the history of menses-associated tetany, serum calcium remained stable after withdrawal of oral contraceptive or conjugated estrogen. Serum calcitriol levels unexpectedly increased in these controls, possibly helping sustain serum calcium. The author concludes the following: (1) some hypoparathyroid women show a significant and symptomatic decline in serum calcium at the time of menses; (2) serum calcium values obtained at the time of menses may not be a reliable guide to adjusting medication dosage for hypoparathyroidism; and (3) further investigation will be needed to determine the mechanism of the decline in serum calcium values and whether the effect of estrogens on serum calcitriol might be altered in the absence of the parathyroid glands.     

Disseminated cysticercosis presenting with bilateral proptosis: A case report

Rationale: Disseminated cysticercosis is characterized by presence of cysts in multiple body organs, like brain, skin, eyes, muscles and rarely heart and lungs.Patient concerns: A 22-year-old man presented with bilateral proptosis of 1-year duration. He also had two episodes of cysticercosis encephalitis. In the second episode of encephalopathy, the patient died.Diagnosis: Disseminated cysticercosis.Interventions: Corticosteroids(Initially intravenous dexamethasone 0.4 mg/kg/day for 2 weeks, followed by oral prednisolone 1.0 mg/kg/day).Outcomes: The patient died of cysticercosis encephalitis approximately 2 months later.Lessons: Disseminated cysticercosis in our case presented only with proptosis as he had very heavy infestation of the brain and eyes. Heavy larval infestation in a patient with disseminated cysticercosis can be life-threatening.     

Midgut malrotation presenting with hyperemesis gravidarum: A case report

Rationale:Midgut malrotation is a rare congenital abnormality resulting from failure of complete intestinal rotation and subsequent fixation during early fetal development. There appeared to be no obvious symptoms in most patients, and a few patients may exhibit symptoms similar to hyperemesis gravidarum, such as nausea and vomiting. Here, we present a case of midgut malrotation presenting as hyperemesis gravidarum.Patient concerns:A 27-year-old woman with an intrauterine pregnancy of 27 + 6 weeks complained of severe nausea and vomiting for 2 weeks.Diagnosis:Magnetic resonance imaging showed obvious dilatation in the proximal part of the duodenum and gastric cavity and the absence of a duodenal path dorsal to the superior mesenteric artery, which was diagnosed as midgut malrotation.Interventions:Considering that the patient’s vital signs were stable, without manifestation of peritonitis or the risks of surgery to the fetus, conservative treatment was adopted. Unfortunately, the fetus developed severe hydrocephalus at 32 weeks. The patient and her family decided to abandon the fetus, and a mid-trimester-induced abortion was performed.Outcomes:The related symptoms completely disappeared after delivery, and the relevant examination after discharge also confirmed the presence of midgut malrotation without gastrointestinal discomfort within 1 year after delivery.Lessons:Midgut malrotation can be considered as a differential diagnosis of hyperemesis gravidarum. Conservative treatment under close monitoring is desirable in pregnant women diagnosed with midgut malrotation.     

神经纤维瘤1型伴恶性嗜铬细胞瘤一例报道

神经纤维瘤病1型( NF1)伴嗜铬细胞瘤较罕见,国内报道很少[1,2]。国内尚未见NF1伴恶性嗜铬细胞瘤的报道,本院2010年收治1例,现将该病例报道如下。患者男性,40岁,未婚,汉族,因发现血压升高7年、腰痛1周,于2010年10月16日入院,7年前体检发现血压升高,因无不适未重视,半年前因反复发作性头痛就诊,血压180/110mm Hg(1 mm Hg=0.133 kPa),无心慌和出汗,CT显示左肾上腺区近10 cm占位,血浆肾上腺素(E) 99.33 pg/ml（正常值＜100 pg/ml,下同）、去甲肾上腺素(NE)3 606.67 pg/ml ＜600)、多巴胺370.00 pg/ml＜100)。于本院泌尿外科拟诊为“左肾上腺嗜铬细胞瘤”手术治疗。术中未发现肿瘤浸润邻近组织器官,周围淋巴结未见明显肿大。     

Malignant lymphoma with various cardiac manifestations: a case report

A 49-year-old woman presented with malignant lymphoma manifesting as dyspnea, palpitation and leg edema. Chest radiography demonstrated cardiomegaly, electrocardiography(ECG) showed first degree atrioventricular block, and echocardiography showed concentric left ventricular hypertrophy with granular sparkling texture and mild pericardial effusion. The diagnosis was malignant lymphoma(B-cell) based on lymph node biopsy. Chemotherapy(CHOP) was performed under a suspected diagnosis of cardiac invasion of malignant lymphoma. Immediately after chemotherapy, ECG showed complete atrioventricular block with sinus arrest, ventricular fibrillation and atrial fibrillation. ECG was normalized after 2 weeks on chemotherapy. Left ventricular hypertrophy was improved and pericardial effusion disappeared. However, she died of recurrence of malignant lymphoma on day 116.     

Acute pericarditis presenting with sinus bradycardia: A case report

Acute pericarditis is almost invariably associated with sinus tachycardia. Recent-onset chest pain in the presence of (sinus) bradycardia is considered to be associated with an acute ischemic syndrome rather than acute pericarditis. This report describes a patient with acute pericarditis initially presenting with sinus bradycardia, probably due to a vasovagal response to (chest) pain.