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PubMed1992

Relapsing thymic carcinoma during pregnancy.

A 23 year old woman, who was known to be in remission from thymic carcinoma, presented in her first trimester. She had previously been treated with radiation and chemotherapy. Her pregnancy was complicated by fever and contractions at 30 weeks and ended with the birth of a live preterm male infant. Subsequent investigation revealed a massive relapse of her mediastinal tumor. This case reinforces the devastating consequences that pregnancy has on thymic hyperplasia. Of nine previously reported cases, five of the women died either during pregnancy or within six months and only one was alive longer than five years. Therapeutic abortion should be considered in the pregnant woman with known thymic neoplasia.

Adult↗
PubMedDate not supplied

[Congenital dilatation of the common bile duct (congenital choledochal cyst)].

INTRODUCTION: Choledochal cyst, although a rare disease, is the most frequent malformation of the extrahepatic biliary ducts. The review of the world medical literature gives the overall number of 2500 cases of choledochal cyst. The incidence of this disease is about 1 in 2000000, but it is about four times more frequent in the Yellow race, and 3 times more frequent in females. Congenital choledochal cyst is a disease of paediatric population, as 60% of all patients are younger than 10 years. The basic anomaly of the disease is the embryological arrest of the normal migration of the pancreaticobiliary junction towards the duodenal wall. The result of the disturbance is the extraduodenal location of the pancreaticobiliary junction and the abnormally long common biliopancreatic channel. The flow of bile and pancreatic juice in the duodenal luminary space is insufficient, so the accumulation of bile and pancreatic juice in the common channel and the reflux of this activated mixture towards biliary ducts occur. Choledochal cyst is formed with all its complications: cholangitis, liver abscess, biliary stone formation, biliary cirrhosis, acute and chronic relapsing pancreatitis, and after many years, biliary cancer. Our results in the treatment of this disease are given. We operated on 31 paediatric patients for choledochal cyst over the period 1978-1998. The study is both retrospective and prospective. RESULTS: There were 24 girls and 7 boys, from neonates to teenagers. In 83 percent of patients the complaints started during the first 3 years of life. Unfortunately, only in 22% of patients the complaints were lasting less than 3 months untill the time of operation. The basic complaints were: abdominal pain and jaundice in 95% of patients, vomiting in 74%, high temperature and fever in 56% and palpabile tumour in 48%. About 30% of children had a relapsing cholangitis and 35% of patients an acute or chronic relapsing pancreatitis. Biliary stones were found in 18% of patients. High level of bilirubins was found in 52% of patients, serum transaminase (SGOT and SGPT) in 69%, amylase level was elevated in blood and urine in about half of the patients, and in choledochal cyst in 91%. Ultrasonography was carried out in 78% of patients and has proved to be a very good screening diagnostic tool. Endoscopic retrograde cholangiopancreaticography (ERCP) was performed in most of the patients. DISCUSSION: The only treatment of choledochal cyst is surgical. The conservative treatment is reserved only for complications (pancreatitis, cholangitis). The only adequate surgical treatment is the radical treatment, which consists of the resection of the whole anomalous choledochus and definitive separation of bile ducts from the pancreatic duct. This is achieved by the technique of Roux-en-Y hepaticojejunostomy. The paliative method of internal drainage of choledochal cyst consists of cystoduodeno or cystojejunostomy, but is now abandoned, as the disease continues with all its manifestations and complications. The internal drainage was performed in 6 our patients, but one of them died (a neonate with sepsis at admitance). Three patients were excluded from the study, and 2 girls were later radically reoperated on. In all other 25 patients resection of the whole choledohus and Roux-Y hepaticojejunostomy were carried out. There were neither operative, nor postoperative deaths. Except the temporary bile leaking, spontaneously healed, there were no significant intraoperative complications. All patients were regularly followed-up, they had no complaints, and all their laboratory and ultrasonographic findings were normal. CONCLUSION: Congenital dilatation of biliary ducts is a rare disease, but it must be considered in differential diagnosis of unclear upper abdominal pains, jaundice and pancreatitis in children. In the diagnosis of this disease the ultrasound imaging is of utmost importance as a screening method, and endoscopic retrograde cholangio-pancreaticography as a method of complete visualization. Resection of the diseased common bile duct and Roux-Y hepatico-jejunostomy are the method of choice in the treatment of children with choledochal cyst.

Adolescent↗
PubMed2000

Staphylococcus aureus bacteremia as a cause of early relapse of thrombotic thrombocytopenic purpura.

BACKGROUND: Thrombotic thrombocytopenic purpura (TTP) is a multisystem disease characterized by the sudden onset of hemolytic anemia, thrombocytopenia, fever, renal failure, and neurologic dysfunction, occurring in various combinations. The most effective treatment of TTP is plasma exchange and the administration of corticosteroids, which reduces mortality from the 90 percent seen in untreated patients to 10 percent. CASE REPORTS: Two patients responded favorably to plasma exchange and corticosteroid therapy, but their TTP relapsed during treatment. In both cases, the early relapse of TTP was associated with Staphylococcus aureus bacteremia secondary to central line infection. Treatment of the infection and intensification of the plasma exchange regimen resulted in a sustained remission. CONCLUSION: Infection should be actively sought and treated in TTP patients who are refractory to treatment or in whom an exacerbation of the disease occurs while they are undergoing plasma exchange.

Adult↗
PubMed1999

Disseminated histoplasmosis in infants.

BACKGROUND: Disseminated histoplasmosis usually occurs in immunocompromised patients who reside in Histoplasma capsulatum-endemic regions. It has also been described in immunocompetent infants after exposure to a large inoculum of the pathogen resulting in case fatality rates of 40 to 50%. METHODS: From 1983 through 1996 all infants with documented disseminated histoplasmosis were treated with amphotericin B followed by daily ketoconazole for 3 months. Immunologic workups were performed at the time of diagnosis and at 4 to 6 weeks of therapy. Surviving patients were followed for at least 1 year. Time to resolution of signs and symptoms was recorded, as were complications. RESULTS: We managed 40 patients with disseminated histoplasmosis. The age in months at diagnosis was 15.3+/-10.2 (mean +/- SD), and 24 were male. All patients were from endemic regions and they presented with fever, spleen and/or liver enlargement and hematologic abnormalities. Diagnosis was made by histology and culture of bone marrow, spleen, lymph node, bronchoalveolar or liver samples. Twenty patients presented with T cell deficiency that resolved at 4 to 6 weeks of therapy in all of the retested patients, and 10 of 12 tested patients had hyperglobulinemia that resolved. Thirty-five (88%) patients were cured by treatment; 4 died and 1 relapsed. CONCLUSIONS: Disseminated histoplasmosis should be considered in infants from endemic areas who present with fever, hepatosplenomegaly and hematologic abnormalities. These patients develop transient hyperglobulinemia and T cell deficiency that resolve with treatment. Treatment with amphotericin B followed by an oral azole for 3 months is effective in most patients.

Amphotericin B↗
PubMed2001

[A case of relapsing polychondritis monitored by daily peak flow measurement].

A 55-year-old man was admitted to our hospital with dyspnea and stridor. At presentation, he also had a low grade fever, arthralgia and nasal obstruction. His chest roentgenogram showed diffuse tracheal narrowing, and chest CT revealed a thickened tracheal wall and fractured cricoid and thyroid cartilages. A diagnosis of relapsing polychondritis was made on the basis of his symptoms and in accordance with Damiani's criteria and CT findings. Daily peak flow (PEF) monitoring was carried out to assess the disease condition of the trachea. Methylprednisolone pulse therapy was started, and was followed by oral prednisolone, which improved his condition immediately, but reduction of oral prednisolone led to recurrence. The clinical course was clearly evaluated by PEF monitoring, but the change was undetectable on chest CT. Dapson, cyclophosphamide, and inhaled steroid were administered with oral prednisolone and the patient's condition has since been fair and stable. PEF monitoring may be useful for the early detection of recurrence in cases of relapsing polychondritis.

Humans↗
PubMed2006

Impact of alternate definitions of fever resolution on the composite endpoint in clinical trials of empirical antifungal therapy for neutropenic patients with persistent fever: analysis of results from the Caspofungin Empirical Therapy Study.

BACKGROUND: Sensitivity analyses were incorporated in a Phase III study of caspofungin vs. liposomal amphotericin B as empirical antifungal therapy for febrile neutropenic patients to determine the impact of varying definitions of fever resolution on response rates. METHODS: The primary analysis used a 5-part composite endpoint: resolution of any baseline invasive fungal infection, no breakthrough invasive fungal infection, survival, no premature discontinuation of study drug, and fever resolution for 48 h during the period of neutropenia. Pre-specified analyses used 3 other definitions for fever resolution: afebrile for 24 h during the period of neutropenia, afebrile at 7 days post therapy, and eliminating fever resolution altogether from the composite endpoint. Patients were stratified on entry by use of antifungal prophylaxis and risk of infection. Allogeneic hematopoietic stem cell transplants or relapsed acute leukemia defined high-risk patients. RESULTS: In the primary analysis, 41% of patients in each treatment group met the fever-resolution criteria. Low-risk patients had shorter durations of neutropenia but failed fever-resolution criteria more often than high-risk patients. In each exploratory analysis, response rates increased in both treatment groups compared to the primary analysis, particularly in low-risk patients. CONCLUSIONS: Response rates for the primary composite endpoint for both treatment groups in this study were driven by low rates of fever resolution. Requiring fever resolution during neutropenia in a composite endpoint can mask more clinically relevant outcomes.

Adolescent↗
PubMed1975

Roentgenographic findings in typhoid fever.

Barium meal examinations have been performed on 20 patients with proved or clinically suspected typhoid fever. There were distinct abnormalities in the terminal ileum early in the febrile stage of the disease. The abnormality was different in a single case of relapse of typhoid. The study is easily and safely performed and we feel that it is a valuable adjunct in the diagnosis of typhoid fever, particularly in those cases where a positive blood culture is not available to confirm the clinical impression. The result of the roentgenographic examination is obtainable immediately and treatment can be initiated early.

Barium Sulfate↗
PubMed2001

Zoledronic acid in the treatment of hypercalcemia of malignancy: results of the international clinical development program.

This report summarizes results of the clinical development program evaluating zoledronic acid (Zometa; Novartis Pharmaceuticals Corp, East Hanover, NJ) in the treatment of hypercalcemia of malignancy (HCM). In addition to a phase I dose escalation trial, two randomized, double-blind, double-dummy studies were conducted in parallel to investigate the clinical efficacy and safety of 4 mg and 8 mg zoledronic acid in patients with moderate to severe HCM. Patients were treated with a single dose of zoledronic acid (4 or 8 mg) via 5-minute infusion or a control treatment, 90 mg pamidronate via 2-hour infusion. Patients who relapsed or had refractory HCM after initial treatment could be re-treated with 8 mg zoledronic acid. End points included rate of complete response, defined as normalization of corrected serum calcium by day 10, change in corrected serum calcium, time to relapse, duration of response, and bone biochemical markers. Doses of > or =0.02 mg/kg were effective and nontoxic in the phase I study. In the controlled studies, 287 patients were randomized and evaluated for safety and 275 patients were evaluable for efficacy. The proportions of patients with a complete response by day 10 were 88.4% and 86.7% in the 4 mg and 8 mg zoledronic acid groups, respectively, compared with 69.7% in the 90 mg pamidronate group. Corrected serum calcium normalization occurred by day 4 in 45.3% of patients treated with 4 mg zoledronic acid, 55.6% of patients treated with 8 mg zoledronic acid, and 33.3% of patients treated with pamidronate. Mean change from baseline in corrected serum calcium also was greater with zoledronic acid than with pamidronate. Median times to relapse were significantly longer in both the zoledronic acid 4 mg and 8 mg groups compared with the pamidronate group. There were no significant differences in efficacy between the 4 mg and 8 mg zoledronic acid doses. Retreatment in 69 patients with relapsing or refractory hypercalcemia with 8 mg zoledronic acid resulted in a 52% complete response rate. Fever, hypophosphatemia, and asymptomatic hypocalcemia were the most common drug-related adverse events. These studies have shown that a short single intravenous dose of 4 mg or 8 mg zoledronic acid is effective in treating moderate to severe HCM. Zoledronic acid produced a higher rate of calcium normalization, faster onset of action, and longer time to relapse than pamidronate, while maintaining an excellent safety profile. The lower dose of 4 mg is recommended as initial therapy, with the 8 mg dose reserved for patients requiring retreatment.

Bone Neoplasms↗
PubMed1999

Consecutive epidemics of Q fever in a residential facility for drug abusers: impact on persons with human immunodeficiency virus infection.

Two large outbreaks of Q fever occurred in 1987 and 1988 in an agricultural community for the rehabilitation of drug users. Approximately 40% of the residents were human immunodeficiency virus (HIV)-positive. Two hundred thirty-five residents presented with clinical evidence of a flulike syndrome that was confirmed to be Q fever; moreover, a large proportion of residents developed an asymptomatic infection. Clinical signs and symptoms were rather nonspecific: fever, malaise, and muscle pain that were often associated with pulmonary symptoms. Single or multiple opacities were detected, with mild interstitial inflammation evident on chest roentgenograms. The source of infection was the sheepfold, which is part of the stock-farming activity of the community. Both outbreaks occurred just after lambing had begun. Residents who were exposed during the first epidemic were protected in the second one. The attack rate among HIV-positive residents was significantly higher than that among HIV-negative residents in the first outbreak, whereas only a slight, marginally significant difference was observed in the second outbreak. The clinical features of Q fever did not differ between HIV-positive and HIV-negative individuals. No cases of relapse or chronic disease were observed.

Adult↗
PubMed1988

Relapsing polychondritis: an unusual cause of PUO in an Asian lady.

A case of pyrexia of unknown origin (PUO) in a 54-year-old Asian lady is described. She subsequently developed ocular and aural inflammation suggestive of relapsing polychondritis (RP) with immediate clinical improvement following steroid therapy. PUO is an unusual presenting feature of RP. The literature is reviewed and the clinical features of RP are discussed.

Female↗
PubMedDate not supplied

Glucocorticoid-unresponsive fever in a patient with Weber-Christian disease.

Weber-Christian disease, a disease of unknown aetiology, is characterised by relapsing febrile episodes and systemic panniculitis. Glucocorticoid therapy is often useful during acute phases of the disease. This report describes a patient in whom hyperpyrexia did not respond to high-dose glucocorticoid treatment, yet did respond to a non-steroidal anti-inflammatory drug (NSAID).

Adult↗
PubMed2000

Inhalation provocation tests in chronic bird fancier's lung.

BACKGROUND: Patients with chronic bird fancier's lung (BFL) can be classified into two types. One group of patients develops chronic disease with fluctuating acute episodes, including low-grade fever, mild exertional dyspnea, and cough (fluctuating chronic BFL; formerly termed recurrent and relapsing chronic BFL). The other group of patients shows no history of acute episodes (insidious chronic BFL). The diagnosis of chronic BFL is difficult, since the onset of chronic BFL may be insidious, with few if any symptoms during the early stages of the disease process. STUDY OBJECTIVE: To attempt to diagnose the conditions of these patients more precisely, inhalation provocation tests were conducted using avian dropping extracts. DESIGN: Retrospective chart review. SETTING: The Tokyo Medical and Dental University Hospital in Japan. PATIENTS: Eleven patients with chronic BFL (6 with fluctuating chronic BFL and 5 with insidious chronic BFL) and 6 control subjects (4 asymptomatic bird owners and 2 idiopathic pulmonary fibrosis patients) were evaluated. MEASUREMENTS AND RESULTS: Inhalation provocation tests using avian dropping extracts were conducted. All BFL patients were evaluated as positive or probable by inhalation challenge, whereas control subjects were evaluated as negative. A peripheral leukocytosis, an increase of alveolar-arterial oxygen pressure difference, an increase of body temperature, and the development of respiratory symptoms including cough and dyspnea were more frequently observed in chronic BFL patients than in control subjects. All the BFL patients had an increase in neutrophils in BAL fluids following inhalation challenge. CONCLUSIONS: We validated the utility of inhalation challenge for the diagnosis of chronic BFL, including fluctuating and insidious BFL. We also demonstrated that neutrophilia in BAL fluids following inhalation challenge could be added to the diagnostic criteria for chronic BFL.

Acute Disease↗
PubMed1994

[Fluconazole therapy for pediatric patients with severe candidal infections].

Three children with severe candidal infections were treated with fluconazole (FLCZ). An excellent effect or a partial effect was obtained in each case. Case 1 was a boy with candida esophagitis. He had been treated for relapsed ALL and became febrile with severe swallowing pain. FLCZ was administered intravenously and he became afebrile after 10 days of the therapy. Case 2 was a boy with candida pneumonitis. He had been treated for relapsed ALL and became febrile with diffuse infiltration in chest X-ray. FLCZ was administered intravenously and he became afebrile transiently, though fever and abnormal X-ray shadows reappeared soon and died of pneumonia and leukemic infiltrations. Case 3 was a girl with candida pneumonitis. She had been treated for relapsed neuroblastoma and became febrile and dyspneic. She was treated with intravenous FLCZ with no effects and died of pneumonia. All three patients showed positive results in 2 of 3 markers for candida infection, including, Cand-Tec, D-arabinitol and fungal index. FLCZ was well tolerated and no adverse effects or abnormal laboratory test results were observed. FLCZ was considered as an effective and safe antifungal agent in the treatment for fungal infections in children.

Adolescent↗
PubMed1993

[A case of vivax malaria in which remission was achieved by primaquine at 1.5 times the standard dose after two earlier relapses].

A 22-year-old male student, who had been to Indonesia, was admitted to our hospital because of a high grade fever on October 12, 1990. On admission, vivax malaria parasites were detected in his blood smears and the antibody titers against Plasmodium vivax and P. falciparum antigen were respectively 1:1024 and 1:64 by the indirect fluorescent antibody test. Therefore, he was diagnosed as having vivax malaria and was treated with sulfadoxine/pyrimethamine (Fansidar), followed by primaquine at 15 mg/day for 14 days. However, relapse occurred twice after treatment with the above-mentioned standard dose of primaquine. After his second relapse, he was treated with primaquine at 22.5 mg/day for 14 days and remission was finally achieved without any adverse reactions. The antibody titer against P. vivax did not re-increase during the first and second relapses.

Adult↗
PubMed1996

Two or three days of ofloxacin treatment for uncomplicated multidrug-resistant typhoid fever in children.

An open randomized comparison of 2 days (Ofx2) versus 3 days (Ofx3) of oral ofloxacin treatment (15 mg/kg/day) was conducted with Vietnamese children between 1 and 15 years of age with suspected typhoid fever. Of 108 children enrolled, 100 were blood culture positive for Salmonella typhi, and 86% of the isolates were multidrug resistant. There were no significant adverse effects. The therapeutic responses were similar in both groups, with mean (+/- standard deviation) fever clearances of 107 +/- 60 h in the Ofx3 group and 100 +/- 64 h in the Ofx2 group (P > 0.2). There were six "clinical" failures in the Ofx2 group and two clinical failures in the Ofx3 group (P > 0.2), in which fever and symptoms persisted for more than 1 week after the start of treatment, but only one of these was culture positive (Ofx3). There was one suspected relapse, and one carrier was identified. Short courses of ofloxacin are simple, inexpensive, safe, and effective for the treatment of uncomplicated multidrug-resistant typhoid fever.

Adolescent↗
PubMed1982

Nonseminomatous germ-cell cancer of the testis.

Fourteen previously untreated patients with metastatic nonseminomatous germ-cell cancer of the testis (NSGC) were treated with a modified VAB-4 regimens that was designed to reduce treatment-related morbidity. Nine of ten patients with minimal disease and the only patient with advanced pulmonary disease achieved a complete response (CR) with chemotherapy alone. Two of three partial responders with advanced abdominal disease were converted to CR status with radiotherapy and/or surgery. None of the 12 CRs has had a relapse (median duration of follow-up, 28+ months). We observed no granulocytopenic fever or permanent renal insufficiency. These results indicate that NSGC patients with a low tumor burden can be spared substantial toxicity without the complete response rates being adversely affected.

Antineoplastic Agents↗
PubMed1982

Nonseminomatous germ cell cancer of the testis. Reducing treatment-related morbidity in patients with disseminated disease.

Fourteen previously untreated patients with metastatic nonseminomatous germ cell cancer of the testis (NSGC) were treated with a modified VAB-4 regimen which was designed to reduce treatment-related morbidity. Nine of 10 patients with minimal disease and the only patient with advanced pulmonary disease achieved a complete response (CR) with chemotherapy alone. Two of three partial responders with advanced abdominal disease were converted to CR status with radiotherapy and/or surgery. None of the 12 CRs have relapsed (median duration of follow-up, 28+ months). We observed no granulocytopenic fever or permanent renal insufficiency. These results indicate that NSGC patients with a low tumor burden can be spared substantial toxicity without adversely affecting complete response rates.

Antineoplastic Agents↗
PubMed1991

Hemolytic uremic syndrome/thrombotic thrombocytopenic purpura: outcome with plasma exchange.

A retrospective analysis was made of 14 consecutive adults with hemolytic uremic syndrome/thrombotic thrombocytopenic purpura (HUS/TTP) who were treated with plasma exchange (PE). In six patients the disease was primary, whilst in eight HUS/TTP was considered secondary to an associated condition. Thirteen patients had renal involvement, six had central nervous system symptoms or signs, three had fever, and one had myocardial damage. Twelve patients (86%) recovered. Four of these relapsed, but responded to further treatment. Two patients failed to respond and died. Hematological response occurred rapidly in survivors: thrombocytopenia resolved after a median of four exchanges, and hemolysis after a median of six. Four patients had a complete recovery, seven had residual mild end organ damage, and one had severe renal impairment. PE was an effective treatment for both primary and secondary HUS/TTP. The platelet count proved to be the earliest indicator of clinical outcome. Continuing follow-up of survivors is required because of the risk of relapse and the high incidence of end organ damage.

Adolescent↗