Massive Bilateral Hydroureteronephrosis and End-Stage Renal Disease Ensuing From Chronic Schistosomiasis: A Case Report.

Globally, schistosomal infections affect over 200 million people resulting in the loss of 70 million disability-adjusted life years. In the sub-Saharan Africa region, where over 85% of the global schistosomal infections are found, it is estimated that about 120 million people become symptomatic, over 20 million have severe disease, and nearly 200 000 die every year. Renal impairment is a severe consequence of schistosomiasis occurring in about 6% of all infected individuals and in 15% of those with the hepatosplenic form. We present a case of massive bilateral hydroureteronephrosis and end-stage renal disease resulting from chronic schistosomiasis in a 38-year-old male of African origin. A 38-year-old male rice farmer of African origin presented with a history of elevated blood pressure, abdominal swelling, and reduced urinary output for about 10 months. Abdominal examination revealed an intraabdominal mass measuring 30 cm × 17 cm extending from the right hypochrondrium region downward to right inguinal outward to umbilicus crossing the midline. He had an estimated glomerular filtration rate of 3.9 mL/min, hemoglobin of 6.78 g/dL, and had multiple electrolyte abnormalities. A computed tomography intravenous urogram scan of the abdomen revealed hepatomegaly (18 cm), bilateral renal enlargement with hydroureteronephrosis, and multiple calcifications on the urinary bladder. A rectal biopsy isolated haematobium eggs and confirmed the diagnosis. Urinary schistosomiasis can have distressing effects on the urinary system in particular and survival prospects in general. In view of this, extensive evaluation of the genitourinary system is pivotal for timely diagnosis and prompt management particularly in residents of schistosoma-endemic communities presenting with obstructive uropathy.

Case Report: Percutaneous Treatment of Multiple Echinococcal Cysts Presenting as Abdominal Palpable Mass.

We report the case of an adolescent Moroccan girl with abdominal pain and palpable mass in the upper right side of the abdomen. In the emergency department, an abdominal ultrasound showed hepatomegaly and eight active liver cysts, compatible with cystic echinococcosis. Serology for Echinococcus granulosus confirmed the diagnosis. Other sites of localization were excluded. Treatment involved albendazole combined with puncture, aspiration, injection, re-aspiration, performed only for the most medial cysts. Periodical follow-up with abdominal ultrasound and with abdominal magnetic resonance imaging showed a progressive involution of all cysts. The treatment with albendazole was stopped after, overall, 6 months, and monthly ultrasound scan were planned as follow-up. In case of hepatic cysts, E. granulosus should be excluded, especially in children from endemic countries. A multidisciplinary approach with pediatric infectious disease specialists, radiologists, and surgeons is fundamental for disease management.

Síndrome de larva migrans visceral y absceso hepático: Reporte de un caso / Visceral larva migrans syndrome and hepatic abscess: A case report

La infección por T oxocara canis o catis es una zoonosis diseminada en el ser humano. La toxocariasis puede coexistir con otras parasitosis endémicas. El hombre actúa como huésped no natural y adquiere la infección a través de la ingesta de huevos del geohelminto. Estos pueden localizarse en la tierra, los patios y los juegos de los niños, y son eliminados, principalmente, por perros o gatos. Existen distintos espectros en la presentación clínica; algunos de ellos son toxocariasis ocular, larva migrans visceral, toxocariasis encubierta y neurotoxocariasis. Se presenta el caso de un paciente de 2 años y 3 meses de edad, con antecedente de síntomas respiratorios, fiebre prolongada y hepatomegalia, con resultados de laboratorio que informa hipereosinofilia, hipergammaglobulinemia y serología positiva para toxocariasis (ensayo por inmunoabsorción ligado a enzimas). Se plantea el diagnóstico de síndrome de larva migrans visceral.

Toxocariasis canis or catis is a zoonotic infection disseminated in humans. Human beings can act as non-natural hosts in which the parasite can survive for long periods of time and they become infected by the ingestion of geohelminth eggs. These can be located on the ground, playgrounds and children's games, and are mostly eliminated by dogs or cats. There are different spectra in the clinical presentation of this infection, which can vary from an asymptomatic host to the production of serious organic lesions; some of them are ocular toxocariasis, visceral larva migrans, covert toxocariasis and neurotoxocariasis. In this case report a patient who presents with a history of respiratory problems, prolonged fever, and hepatomegaly. Laboratory analyses show hypereosinophilia, hypergammaglobulinemia and serodiagnosis is positive for toxocariasis. Preliminary diagnosis: Visceral Larva Migrans Syndrome.

Disease severity in patients with visceral leishmaniasis is not altered by co-infection with intestinal parasites.

Visceral leishmaniasis (VL) is a neglected tropical disease that affects the poorest communities and can cause substantial morbidity and mortality. Visceral leishmaniasis is characterized by the presence of Leishmania parasites in the spleen, liver and bone marrow, hepatosplenomegaly, pancytopenia, prolonged fever, systemic inflammation and low body mass index (BMI). The factors impacting on the severity of VL are poorly characterized. Here we performed a cross-sectional study to assess whether co-infection of VL patients with intestinal parasites influences disease severity, assessed with clinical and haematological data, inflammation, cytokine profiles and BMI. Data from VL patients was similar to VL patients co-infected with intestinal parasites, suggesting that co-infection of VL patients with intestinal parasites does not alter disease severity.

A RARE PARASITIC DISEASE OF THE LIVER - ALVEOLAR ECHINOCOCCOSIS.

Alveolar echinococcosis is a rare parasitic disease, especially of the liver, caused by the larval stage of the tapeworm Echinococcus multilocularis. At the end of the last century France, Germany, Austria and Switzerland were the regions where this disease most often manifested itself, these days this infection is diagnosed also in our territory. We describe the case of the disease of a twenty-five-year-old male with nonspecific signs and hepatomegaly, who was diagnosed on the basis of imaging and laboratory sampling. Due to inoperability the patient is now in infectologist follow-up on long-term treatment with Albendazole. He is clinically stable, included on the waiting list for liver transplantation.

Capillaria hepatica in man--an overview of hepatic capillariosis and spurious infections.

Capillaria hepatica (syn. for Calodium hepaticum) is a zoonotic nematode parasitizing in the livers of rodents as main hosts and in numerous other mammals including humans. It is the causative agent of the rare conditions of hepatic capillariosis and spurious C. hepatica infections in humans. In this review, 163 reported cases of infestations with this parasite (72 reports of hepatic capillariosis, 13 serologically confirmed infestations and 78 observations of spurious infections) are summarized with an overview on the distribution, symptoms, pathology, diagnosis, serology and therapy of this rare human pathogen.

Hepatosplenomegaly associated with chronic malaria exposure: evidence for a pro-inflammatory mechanism exacerbated by schistosomiasis.

In sub-Saharan Africa, chronic hepatosplenomegaly, with palpable firm/hard organ consistency, is common, particularly among school-aged children. This morbidity can be caused by long-term exposure to malaria, or by Schistosoma mansoni, and it is exacerbated when these two occur together. Although immunological mechanisms probably underlie the pathogenic process, these mechanisms have not been identified, nor is it known whether the two parasites augment the same mechanisms or induce unrelated processes that nonetheless have additive or synergistic effects. Kenyan primary schoolchildren, living in a malaria/schistosomiasis co-transmission area, participated in cross-sectional parasitological and clinical studies in which circulating immune modulator levels were also measured. Plasma IL-12p70, sTNF-RII, IL-10 and IL-13 levels correlated with relative exposure to malaria, and with hepatosplenomegaly. Soluble-TNF-RII and IL-10 were higher in children infected with S. mansoni. Hepatosplenomegaly caused by chronic exposure to malaria was clearly associated with increased circulating levels of pro-inflammatory mediators, with higher levels of regulatory modulators, and with tissue repair cytokines, perhaps being required to control the inflammatory response. The higher levels of regulatory modulators amongst S. mansoni infected children, compared to those without detectable S. mansoni and malarial infections, but exposed to malaria, suggest that S. mansoni infection may augment the underlying inflammatory reaction.

Hepatosplenomegaly is associated with low regulatory and Th2 responses to schistosome antigens in childhood schistosomiasis and malaria coinfection.

Hepatosplenomegaly among Kenyan schoolchildren has been shown to be exacerbated where there is transmission of both Schistosoma mansoni and Plasmodium falciparum. This highly prevalent and chronic morbidity often occurs in the absence of ultrasound-detectable periportal fibrosis and may be due to immunological inflammation. For a cohort of school-age children, whole-blood cultures were stimulated with S. mansoni soluble egg antigen (SEA) or soluble worm antigen (SWA). Responses to SWA were found to be predominantly Th2 cytokines; however, they were not significantly associated with either hepatosplenomegaly or infection with S. mansoni or P. falciparum. In comparison, SEA-specific Th2 cytokine responses were low, and the levels were negatively correlated with S. mansoni infection intensities and were lower among children who were coinfected with P. falciparum. Tumor necrosis factor alpha levels in response to stimulation with SEA were high, and a negative association between presentation with hepatomegaly and the levels of the regulatory cytokines interleukin-6 and transforming growth factor beta(1) suggests that a possible mechanism for childhood hepatomegaly in areas where both malaria and schistosomiasis are endemic is poor regulation of an inflammatory response to schistosome eggs.

Granulomatous, hepatolithiasis and hepatomegaly caused by Capillaria hepatica infection: first case report of Thailand.

This is the first case report in Thailand of a Capillaria hepatica infection causing a granulomatous hepatic lesion, bile duct dilatation, hepatolithiasis and hepatomegaly. The patient's chief complaint was abdominal pain with fever and chills. Imaging of the liver revealed a 3-cm mass in the postero-inferior sub-segment of the right lobe of the liver with bile duct dilatation. Right hepatectomy and cholecystectomy were performed. Gross pathology of the right hepatectomy revealed focal intrahepatic duct dilatation with prominent periductal fibrosis. The histopathological section revealed chronic inflammation and some granuloma formation surrounding the bile ducts, generalized portal infiltration, prominence of eosinophils and hepatolithiasis. Histopathotogical section revealed oblique sections of C. hepatica egg (size 35.4+/-6.38 microm in width) and brown amorphous pigment.

Schistosomiasis mansoni: novel chemotherapy using a cysteine protease inhibitor.

BACKGROUND: Schistosomiasis is a chronic, debilitating parasitic disease infecting more than 200 million people and is second only to malaria in terms of public health importance. Due to the lack of a vaccine, patient therapy is heavily reliant on chemotherapy with praziquantel as the World Health Organization-recommended drug, but concerns over drug resistance encourage the search for new drug leads. METHODS AND FINDINGS: The efficacy of the vinyl sulfone cysteine protease inhibitor K11777 was tested in the murine model of schistosomiasis mansoni. Disease parameters measured were worm and egg burdens, and organ pathology including hepato- and splenomegaly, presence of parasite egg-induced granulomas in the liver, and levels of circulating alanine aminotransferase activity as a marker of hepatocellular function. K11777 (25 mg/kg twice daily [BID]), administered intraperitoneally at the time of parasite migration through the skin and lungs (days 1-14 postinfection [p.i.]), resulted in parasitologic cure (elimination of parasite eggs) in five of seven cases and a resolution of other disease parameters. K11777 (50 mg/kg BID), administered at the commencement of egg-laying by mature parasites (days 30-37 p.i.), reduced worm and egg burdens, and ameliorated organ pathology. Using protease class-specific substrates and active-site labeling, one molecular target of K11777 was identified as the gut-associated cathepsin B1 cysteine protease, although other cysteine protease targets are not excluded. In rodents, dogs, and primates, K11777 is nonmutagenic with satisfactory safety and pharmacokinetic profiles. CONCLUSIONS: The significant reduction in parasite burden and pathology by this vinyl sulfone cysteine protease inhibitor validates schistosome cysteine proteases as drug targets and offers the potential of a new direction for chemotherapy of human schistosomiasis.

Clinical presentation and management of visceral leishmaniasis.

BACKGROUND: Febrile illnesses like malaria, typhoid, and tuberculosis are the commonest problems in our area, but visceral leishmaniasis (VL) is also one of the diseases presenting with fever in this part of the country. This study was conducted to evaluate its clinical spectrum and way of management. METHODS: This study was conducted in Paediatric Department of Women and Children Hospital and Ayub Teaching Hospital, Abbottabad from October 1985 to August 1999 during which 70 cases of VL were diagnosed and managed. RESULTS: All patients were below 10 years of age and were from Hazara division. Majority of them were from two specific localities, one in Abbottabad District (43%) and the other in Mansehra District (14%). Common clinical features were Fever 99%, Splenomegaly (99%), Anaemia (96%), Hepatomegaly (86%), distension of abdomen (47%) and bleeding diathesis 14%. Haemoglobin was below 7.9 gm in 82.86%, white cell count was below 4000/mm3 in 42.85%, Platelet count was below 100000/mm3 in 67.14% and ESR was >50 mm at the end of first hour in 86% of the patients. All the patients showed Leishmania Donovani bodies in the bone marrow smears except one, where tap was dry and then trephine biopsy was performed to confirm the diagnosis. In 67 cases amastigote form was found and only in 3 patients the promastigotes were found. Fifty two patients had received meglumine antimoniate (glucantime) and 18 received sodium stibogluconate (pentostam) along with supportive therapy. Mortality was 11.43%. CONCLUSIONS: The disease is gradually spreading southwards in the country. Children below 5 years are mainly affected. Bone marrow examination is the most reliable and simple method of diagnosis. A high index of suspicion must he kept in mind for all febrile cases coming from Hazara division, Northern areas, Azad Kashmir.

Accelerated neutrophil apoptosis in neutropenic patients with hepatosplenic schistosomiasis is induced by serum Fas ligand.

Neutropenia in patients with hepatosplenic (HS) schistosomiasis may stem from enhanced neutrophil apoptosis. However, the molecular mechanism of neutrophil apoptosis has not been clearly defined. Neutrophils harvested from neutropenic patients with HS schistosomiasis (n = 25), non-neutropenic patients with hepatointestinal (HI) schistosomiasis (n = 10), and age- and sex-matched healthy control subjects (n = 10) were examined for the degree of apoptosis after incubation with autologous sera. Neutrophil apoptosis was quantified by flow cytometry through determination of propidium iodide nuclear staining and confirmed by DNA gel electrophoresis at 0 time (fresh neutrophil), 4 and 24 h culture. Neutrophils from healthy subjects were also incubated with either 10% heterologous normal or neutropenic serum, with and without anti-Fas ligand antibody. Serum Fas ligand levels were assessed in sera of patient groups and healthy controls by ELISA. Compared with normal controls and HI, HS group demonstrated greater neutrophil apoptosis in the presence of autologous serum (P < 0.01, < 0.05, respectively). Furthermore, compared with normal neutrophils exposed to heterologous normal serum, those exposed to heterologous neutropenic serum exhibited higher apoptosis rates (P < 0.01). The apoptotic effect of neutropenic sera is attenuated by anti-Fas ligand. Fas expression was significantly higher in HS group as compared to both HI and normal healthy controls (P < 0.05). Serum Fas ligand levels were significantly higher among HS group as compared to both HI and control groups (P < 0.01 for both). Neutrophil apoptosis was not correlated to the size of spleen in HS group. In conclusion, the rate of neutrophil apoptosis is accelerated in neutropenic HS schistosomiasis. These findings suggest that enhanced neutrophil apoptosis demonstrated in HS patients is triggered by soluble Fas ligand, which is mostly derived from spleen.

[Experimental Encephalitozoon cuniculi infection in dexamethasone-immunosuppressed mice]. / Infecção experimental pelo Encephalitozoon cuniculi em camundongos imunossuprimidos com dexametasona.

OBJECTIVE: Microsporidian Encephalitozoon cuniculi has been recognized as an opportunistic pathogen in immunosuppressed individuals, such as AIDS patients. The objective of the study was to develop pharmacologically immunosuppressed animals as a model of the natural occurring E. cuniculi infection. METHODS: Distinct groups of adult Balb-C mice were immunosuppressed with different doses of dexamethasone (Dx, 3 or 5 mg/kg/day, intraperitoneal route - IP) and inoculated with E. cuniculi spores by IP route intraperitoneally. Control groups (inoculated animals but non-immunosuppressed and non- inoculated animals but immunosuppressed) were also used. The spores of E. cuniculi were previously cultivated in MDCK cells. The animals were sacrificed and necropsied at 7, 14, 21, 28 and 35 days post-inoculation. Tissue fragments were collected and processed for light microscopy studies, using Gram-chromotrope and hematoxylin-eosin staining techniques. RESULTS: In all immunosuppressed and inoculated inoculated immunosuppressed mice,specially in those that received 5 mg/kg/day of dexamethasone, the most prominent necropsy findings were hepatomegaly and splenomegaly. The experimental inoculation resulted in a disseminated non-lethal infection, characterized by granulomatous lesions in several organs (liver, lungs, kidneys, gut and brain) but notably in the hepatic tissue. Spores of E. cuniculi were only seen in few animals treated with 5 mg/kg/day of Dx at 35 days post-infection. CONCLUSIONS: Microsporidiosis in Dx-immunosuppressed mice provides a useful model for studies of the microsporidial infection, resembling that one naturally occurring in immunodeficient individuals with AIDS.

[Moroccan experience in the surgical treatment of multiple hydatid cysts in the liver]. / L'expérience marocaine dans le traitement chirurgical des kystes hydatiques multiple du foie: à propos de 94 cas.

We studied 94 cases of multiple hydatid cysts in the liver, over a period of ten years. These cases accounted for 31.3% of all cases of hydatid cysts treated surgically in the Visceral Surgery Department of Avicenne Military Hospital in Marrakech. In these patients, who were often young and male, the principal symptoms were pain in the right hypochondrium (71.3%) and hepatomegaly (24.5%). In about 10% of cases, the cysts were discovered by chance. Ultrasound and CT scans facilitated diagnosis and determination of the position of the cysts, with reliability reaching 100% for CT scans. The cysts had burst in the bile ducts in 26.6% of cases and were infected in 8 cases. They were multivesicular in 77.5% of cases. Association with hydatidosis at another site was observed in 28 cases: in the peritoneum in 15, the thorax in 7, the diaphragm in 4, the spleen in 2 and the kidney in 1 case. Surgically, the route most frequently used was double incision below the rib cage (49.5%). It is not possible to recommend one particular way to treat cysts and the most appropriate approach to treatment depends on the site, type and number of cysts. Resection of the prominent dome is the technique most frequently used (57.25%). However, in recent years, the use of cystectomy has been increasing (20.2%) due to the considerable decreases in post-operative morbidity and duration of hospital stay that it affords. The principal post-operative complications observed were abscesses under the diaphragm (6 cases), biliary leakage (5 cases), pleurisy (6 cases) and the formation of abscesses in the vestigial cavity (4 cases). The rate of morbidity in the RDS appeared high, accounting for 75% of total morbidity. Only one patient died. This patient died from severe hepatic insufficiency due to the near destruction of the liver by the hydatosis. We observed two recurrences during follow up. Both underwent further surgery and neither suffered complications.