Giant Scrotal Hydrocele and Bilateral Leg Lymphedema as Clinical Manifestation of Chronic Lymphatic Filariasis.

A 51-year-old male came with the complaint of recurrent swelling in the scrotum and legs. Swelling of the scrotum first appeared 17 years ago in the left scrotum approximately the same size as an apple and underwent surgery. However, 2 years after surgery, the swelling reemerged and gradually increase in size in both scrotums. Left leg swelling began to emerge 5 years ago followed by right leg 3 years after. The patient lives in Sarmi regency Papua province (endemic).

Clinical case estimates of lymphatic filariasis in an endemic district of Bangladesh after a decade of mass drug administration.

BACKGROUND: Bangladesh has a high burden of lymphatic filariasis (LF) disease. This survey estimated the current number of clinical cases in a historically endemic district after a decade of mass drug administration (MDA). METHODS: A cluster survey was conducted in 30 villages of Nilphamari District. Ten households per village were randomly selected, and individuals with LF clinical conditions identified. Hydrocoele and lymphoedema (leg, arm, breast) cases were quantified and mapped, and differences by sex, age, severity of disease and episodes of acute dermatolymphangioadenitis attacks (ADLA) examined. RESULTS: From 1242 surveyed >10 years old 55 individuals (4.4%) with LF clinical conditions were identified. Males were mostly affected by hydrocoeles, and females by leg lymphoedema. Higher prevalence and more advanced stages of disease were found in older age groups. CONCLUSIONS: Overall low prevalence of severe clinical disease was found, which has positive implications for the elimination goal. Future planning of MMDP strategies will need to address the varying clinical conditions, severity of disease and impact of ADLAs affecting different sub-groups of the population. It will also be important to monitor for new cases, and examine the shift in age distribution of cases over time.

Role of persistent processus vaginalis in hydroceles found in a tropical population.

BACKGROUND: Lymphatic obstruction by Wuchereria bancrofti is thought to be the mechanism for development of tropical hydrocele in men and for elephantiasis, mostly in women. Hydrocele prevalence is used to determine the effectiveness of para site eradication programs. METHODS: We maintained a prospective log of operations performed at 1 Canadian Field Hospital during its relief mission to Léogâne, Haiti. Information regarding duration of symptoms, type of previous surgery (if any), surgical approach, associated inguinal hernia and volume and appearance of hydrocele fluid in patients with tropical hydroceles were recorded. RESULTS: From January to March 2010, 4922 patients were seen, none of whom had elephantiasis. Of the 64 patients who collectively underwent 69 inguino-scrotal procedures, 5 patients had inguinal hernia repair several years after hydrocele excision via the scrotum, 19 patients with bilateral hydroceles underwent a scrotum-only approach, and 45 patients had an inguinal approach (33 unilateral and 12 bilateral) to repair 57 hydroceles. A patent processus vaginalis was present in 50 of 57 (88%) hydroceles where the groin was explored. CONCLUSION: Hydroceles remain common in Léogâne despite successful eradication of filariasis with mass drug administration using diethylcarbamazine-fortified cooking salt. Persistent patent processus vaginalis is a more likely cause than persistent filariasis. There is probably little difference between hydrocele in developed countries and tropical hydrocele other than neglect. Hydrocele prevalence is not a measure of the effectiveness of parasite eradication programs.

Lymphatic filariasis in western Ethiopia with special emphasis on prevalence of Wuchereria bancrofti antigenaemia in and around onchocerciasis endemic areas.

Lymphatic filariasis is known to be endemic in Gambella Region, western Ethiopia, but the full extent of its endemicity in other regions is unknown. A national mapping program for Ethiopia was initiated in 2008. This report summarizes initial data on the prevalence of Wuchereria bancrofti antigenaemia based on surveys carried out in a sampled population of 11685 individuals living in 125 villages (112 districts) of western Ethiopia. The overall prevalence rate was 3.7%, but high geographical clustering and variation in prevalence (ranging from 0% to more than 50%) was found. The prevalence of hydrocele (in males) and lymphoedema of limbs was 0.8% and 3.6% respectively. Significantly higher (χ(2)=49.6; P<0.01) prevalence of antigenaemia was noted in known onchocerciasis endemic districts (4.7%) compared to non-onchocerciasis endemic districts (2.3%). Thirty-four of the 112 districts, with a population of 1547685 in 2007, were found to be endemic. Of these, the numbers of districts with prevalence rates of >20%, 10-20% and 5-9% were nine, 14 and 20 respectively. Twenty-nine of these 34 endemic districts were found in three regions: Gambella Region (seven districts), Beneshangul-Gumuz Region (13 districts), and Southern Nations, Nationalities and Peoples' Region (SNNPR) (nine districts). The other five were from Amhara (two districts) and Oromia (three districts) regions. A tentative distribution map has been drawn to facilitate the launching of the Ethiopia LF elimination program.

Capacity building for the modified filarial hydrocelectomy technique in West Africa.

To harmonise the technique of filarial hydrocelectomy throughout the West African subregion, training workshops were organized in 10 endemic West African countries involving surgeries on Filarial hydrocele patients (Filariceles). Countries included Ghana, Nigeria, Niger, Burkina Faso, Senegal, Mali, Liberia, Gambia, Sierra Leone and Togo. Patients were recruited by the Filariasis programme managers in all these countries and workshops were done to train surgeons from teaching, government and district hospitals with funding mainly from the West African Filariasis Project. A total of 16 workshops were held in 10 West African Countries, and a total of 500 filarial hydrocele/chylocele cases were operated using the technique of total tunical resection without drainage with excellent postoperative results. The average operating time for a unilateral hydrocele was 45 min to 1h and for bilateral hydrocele was 1½ h to 2h depending on the size of the hydrocele .All patients were admitted for a minimum of 5 days postoperative. Training workshops were successfully organized in 10 out of the envisaged 12 west African Countries with 214 surgeons trained. Since 2004 till date the trained surgeons have also operated 2500 filaricele cases bringing it to a total of 3000 cases operated so far. Evaluation studies revealed that the infection rates in one country was initially 60% and then it later dropped to 20% and this was actually attributed to poor follow up by the selected National Coordinator. However complication rates in all the Countries where workshops were done ranged between 5% and 7% made up of infection, haematoma and or delayed wound healing. Recurrence rates have been very low about 3-5% per Country.

The role of ultrasonography in the differentiation of the various types of filaricele due to bancroftian filariasis.

OBJECTIVE: (i) To determine the frequencies of urogenital pathologies in men infected with bancroftian filariasis, and (ii) to evaluate the role of ultrasonography (USG) as a diagnostic tool to differentiate between diverse pathologies with different clinical implications. To date, all types of scrotal enlargement resulting from lymphatic filariasis (LF) have been summarized under one term: "filaricele". PATIENTS AND METHODS: Data were compiled from recruitment phases for field trials in an endemic area for LF in Ghana. 1453 men aged 18 years and above underwent ultrasound examinations of the scrotum. Observation parameters were: Filaria Dance Sign (FDS), dilation of supratesticular lymphatic vessels, thickness of scrotal skin, occurrence and amount of fluid accumulation, echogenicity of the fluid between the layers of the tunica vaginalis, as well as position and homogenicity of testis, epididymis and spermatic cord. In 1132 men, blood samples were taken for parasitological analysis. RESULTS: In 56% of examined patients, fluid accumulation around the testis was detected (38% subclinical-, 18% clinical stages). Differentiation of the echogenicity of the fluid revealed echo-free hydrocele (EFH) in 47% and echo-dense hydrocele (EDH) in 9%. Patients without hydrocele and subclinical stages had a thinner scrotal skin than those in clinical stages or with lymphscrotum (P < 0.001). In the EDH group the scrotal skin was thicker than in the EFH group (P < 0.001). 1.4% had a lymphscrotum. FDS was detected in 24% of all 1453 volunteers who underwent USG. The number of worm nests correlated with microfilarial load and levels of circulating filarial antigen (P < 0.001; 20% microfilaremic, 48% antigen positive). CONCLUSION: In an unexpected high number of men (56%) fluid accumulation around the testis was detected by USG of which more than one third (38%) presented with subclinical stages. The study showed that echo-dense and echo-free fluid could be differentiated and that a considerable number of cases had EDH (9%) posing a risk to develop necrotic testis and infertility and therefore requiring immediate surgical intervention. USG thus turned out to be a useful diagnostic technique to differentiate between those cases requiring immediate surgical intervention from those that can be treated with (anti-wolbachial and hyperpermeability reducing) drugs that ameliorate or halt progression of the disease.

Ultrastructural analysis of Wuchereria bancrofti (Nematoda: Filarioidea) body wall.

Bancroftian filariasis constitutes the principal mosquito-borne nematode infection of humans and the surface of adult of Wuchereria bancrofti seems to be especially important in the intricate interplay between host and parasite. The study of the parasite's surface structure might help to understand the localization and function of various organelles. W. bancrofti adult worms were recovered from untreated patients during hydrocele repair surgery and studied by transmission electron microscopy. The body wall of adult parasite is composed of cuticle, hypodermis and muscular layer. Cuticle is the external layer and shows transverse cuticular striation. It is composed by an epicuticle, cortical layers, median layer, fibrous layers and basal layer. The epicuticle is the most external cuticular layer and appears as a single laminar electron-dense layer. The cortical external region is more electron-dense and granular in appearance than the inner cortical layer. Electron-dense structures, called bosses are randomly distributed filling the cuticular striation. The median layer is formed by an electron-dense and continuous thick line. The fibrous layer is subdivided in inner and external layers connected by projections. The basal layer includes a large quantity of membranous projections directed toward the hypodermis. The hypodermis is a syncytium where some cellular organelles are observed. The somatic musculature is meromyarian. The muscle fibers consist of contractile and non-contractile regions and the contractile region is composed of myofilaments separated by dense body. This is the first study of W. bancrofti adult worms obtained from untreated patients and studied by transmission electron microscopy.

A pilot program of mass surgery weeks for treatment of hydrocele due to lymphatic filariasis in central Nigeria.

In a pilot program of mass surgery weeks (MSW) to provide hydrocelectomy services to men with filarial scrotal hydrocele, local general practitioners performed 425 surgical repairs in 301 men in five MSW in three rural Nigerian community hospitals between 2002 and 2005. The most common (94%) procedure used was the eversion technique, which was most familiar to the practitioners. Postoperative complications included hematoma (3.7%) and infection (3%), and there was one death from infection in an elderly man with previously unrecognized diabetes. In 115 patients (38%) followed for 1 to 3 years, the hydrocele recurrence rate was 7%. The eversion technique gives an acceptable outcome, and MSW are safe and effective if strict attention is paid to preoperative screening of candidates and asepsis.

Circulating filarial antigen in serum and hydrocele fluid from individuals living in an endemic area for bancroftian filariasis.

This study examined circulating filarial antigen by monoclonal antibody Og4C3-enzyme-linked immunosorbent assay (ELISA) from 114 men with hydrocele, living in an endemic area. Nocturnal blood and hydrocele fluid were collected and examined for microfilaria. ELISA was performed on serum and hydrocele fluid for detection of antigen. Amongst 114 cases, 5(4.4%) showed microfilaria in blood but none in fluid. ELISA was positive in 13(11.40%) serum and 5 (4.4%) fluid samples. All five fluid antigen positive cases were positive for antibodies and showed microfilaria in blood. These findings emphasize the use of circulating filarial antigen detection and alternative usage of hydrocele fluid for diagnosis of filariasis.

Chronic clinical manifestations related to Wuchereria bancrofti infection in a highly endemic area in Kenya.

Clinical examinations were conducted in an effort to provide baseline data for a pilot filariasis elimination programme implemented in a Wuchereria bancrofti-endemic focus in Malindi district, Kenya. Of 186 males aged 15 years and above examined, 64 individuals (34.4%) had hydrocele, and the prevalence of the manifestation in those above 40 years old was 55.3%. The prevalence of leg lymphoedema in persons aged 15 years and above was 8.5%, with a higher rate in males (12.6%) than in females (5.7%). The overall prevalence of inguinal adenopathy was 8.6%, and males had a significantly higher (12.9%) prevalence of adenopathy than females (5.1%) (P<0.001). The data in the present study provided support for consideration of filarial infection as a possible cause of inguinal lymphadenopathy in bancroftian filariasis-endemic areas. The results of this study also indicate that lymphatic filariasis is a serious public health problem in the northern coastal areas and morbidity control programmes should be implemented to alleviate the suffering of those affected.

Health status of lymphatic filariasis assessed from patients using seven domains five levels (7D5L) instrument.

Health status is the standardized description of health condition of individuals either normal or ill due to diseases and is presented as multidimentional profiles. In an effort to develop an indicator to assess the impact of morbidity intervention against lymphatic filariasis (LF), we assessed the health status of seven different clinical manifestations (health states) of LF from the patient's perspective. One hundred and seventy-four filarial patients either with hydrocele or lymphoedema were involved in the study. Acute episode of adenolymphangitis (ADL) among chronic patients was considered as co-morbidity. Severity levels, focusing on physical, mental and social dimensions of health were defined and quantified, using seven domains and five levels (7D5L) instrument, an extended form of EuroQol (5D3L). All the seven domains of health are affected by filarial disease, with the levels of severity varying with health states. The mean severity score of ADL (25.8) was significantly higher compared to lymphoedema (10.7) and hydrocele (6.9) (P<0.05). In males, the mean score of lymphoedema (11.6) was significantly higher in comparison to hydrocele (P<0.05). The severity scores increase with the progression of filarial disease but independent of gender. The health states of LF were further classified based on the percentage of severity according to International Classification of Functioning, Disability and Health (ICF) on impairments. Lymphoedema grade 4 (oedema with skin changes) and ADL were ranked as severe. The importance of these findings is discussed in view of priority setting and evaluating the morbidity management under Global Programme for Elimination of Lymphatic Filariasis (GPELF).

Doxycycline reduces plasma VEGF-C/sVEGFR-3 and improves pathology in lymphatic filariasis.

Lymphatic filariasis is a disease of considerable socioeconomic burden in the tropics. Presently used antifilarial drugs are able to strongly reduce transmission and will thus ultimately lower the burden of morbidity associated with the infection, however, a chemotherapeutic principle that directly induces a halt or improvement in the progression of the morbidity in already infected individuals would constitute a major lead. In search of such a more-effective drug to complement the existing ones, in an area endemic for bancroftian filariasis in Ghana, 33 microfilaremic and 18 lymphedema patients took part in a double-blind, placebo-controlled trial of a 6-wk regimen of 200 mg/day doxycycline. Four months after doxycycline treatment, all patients received 150-200 microg/kg ivermectin and 400 mg albendazole. Patients were monitored for Wolbachia and microfilaria loads, antigenemia, filarial dance sign (FDS), dilation of supratesticular lymphatic vessels, and plasma levels of lymphangiogenic factors (vascular endothelial growth factor-C [VEGF-C] and soluble vascular endothelial growth factor receptor-3 [(s)VEGFR-3]). Lymphedema patients were additionally monitored for stage (grade) of lymphedema and the circumferences of affected legs. Wolbachia load, microfilaremia, antigenemia, and frequency of FDS were significantly reduced in microfilaremic patients up to 24 mo in the doxycycline group compared to the placebo group. The mean dilation of supratesticular lymphatic vessels in doxycycline-treated patients was reduced significantly at 24 mo, whereas there was no improvement in the placebo group. Preceding clinical improvement, at 12 mo, the mean plasma levels of VEGF-C and sVEGFR-3 decreased significantly in the doxycycline-treated patients to a level close to that of endemic normal values, whereas there was no significant reduction in the placebo patients. The extent of disease in lymphedema patients significantly improved following doxycycline, with the mean stage of lymphedema in the doxycycline-treated patients being significantly lower compared to placebo patients 12 mo after treatment. The reduction in the stages manifested as better skin texture, a reduction of deep folds, and fewer deep skin folds. In conclusion, a 6-wk regimen of antifilarial treatment with doxycycline against W. bancrofti showed a strong macrofilaricidal activity and reduction in plasma levels of VEGF-C/sVEGFR-3, the latter being associated with amelioration of supratesticular dilated lymphatic vessels and with an improvement of pathology in lymphatic filariasis patients.

Detection of filarial antigen and antibody in serum and hydrocele fluid of 100 patients of hydrocele.

AIM: The present study was carried out to detect an association between isolated non-communicable hydrocoele and filariasis and to provide awareness to positive patients regarding sequel and advising methods for the reduction of morbidity. METHODS: Blood samples and hydrocoele fluids were used to detect filarial antigen and antibody by ICT Kit, Trop-bio kit and Sevafilachek Kit. These were followed by statistical evaluation by chi2 test. RESULTS: 14% of cases were positive for filarial antigen and antibody in hydrocoele patient serum, while 15% of cases were positive for filarial antigen and antibody in the serum of non-hydrocoele patients. CONCLUSION: Probability is less than 0.05, which is statistically significant.

Circulating filarial antigen in the hydrocele fluid from individuals living in a bancroftian filariasis area - Recife, Brazil: detected by the monoclonal antibody Og4C3-assay.

The purpose of this study was to examine the circulating filarial antigen (CFA) detected by the monoclonal antibody (mAb) Og4C3-ELISA in paired samples of serum and hydrocele fluid from 104 men with hydrocele, living in an endemic area of Wuchereria bancrofti. Nocturnal blood specimens were filtered and examined for microfilariae (MF) and ultrasound was used in order to identify the presence of adult worms (the filaria dance sign - FDS) in the lymphatic vessels of the scrotal area. Four groups were selected according to their parasitological status: group I - 71 MF- and FDS-; group II - 21 MF+ and FDS+; group III - 10 MF- and FDS+ and group IV- 2 MF+ and FDS-. CFA was identified simultaneously (fluid and serum) in 11 (15.5%), 21 (100%), 3 (30%), and 1 (50%) in groups I, II, III, and IV, respectively. In despite of high CFA+ level (antigen Og4C3) units/ml, the Geometrical Mean (GM) = 2696) in the sera of these 36/104 paired samples, when compared to the hydrocele fluid, (GM = 1079), showed a very good correlation between the CFA level in the serum and CFA level in the fluid (r = 0.731). CFA level in the serum of the 23 microfilaremics (groups II and IV) was extremely high (GM = 4189) and was correlated with MF density (r = 0.442). These findings report for the first time the potential alternative use of the hydrocele fluid to investigate CFA using the mAb Og4C3-ELISA.

Microfilaria in hydrocele fluid cytology.

Filariasis, a parasitic infection endemic in parts of India, Myanmar, islands of the South Pacific, West and East Africa and Saudi Arabia can be diagnosed from various types of cytopathological specimens. This case documents the detection of filarial infection from hydrocele fluid cytology in a 30-year-old Myanmar migrant worker in Malaysia.

Characterization of antibody responses to Wolbachia surface protein in humans with lymphatic filariasis.

Symbiotic Wolbachia organisms of filarial nematodes have received much attention as possible chemotherapy targets and disease-causing organisms. In order to further investigate the association between anti-Wolbachia immune responses and chronic filarial disease in humans, antibody responses to Wolbachia surface protein (WSP) were assayed in serum samples collected from 232 individuals living in Leogane, Haiti, an area where Wuchereria bancrofti infection is endemic, and from 67 North Americans with no history of lymphatic filariasis. As opposed to antifilarial antibody responses, which were largely influenced by the patient's infection status, the prevalence and levels of anti-WSP immunoglobulin G (IgG) antibodies among individuals with lymphedema or hydrocele were significantly greater than those in gender- and infection-matched individuals without disease. In at least one case, the anti-WSP IgG response was coincident with the onset of lymphedema development, and among anti-WSP-positive women with lymphedema, anti-WSP IgG levels were negatively correlated with the duration of lymphedema. The presence of anti-WSP IgG was also associated with the severity of inguinal adenopathy among men with hydrocele. In addition to the presence of anti-WSP antibodies among Haitians, 15 of 67 (22%) serum samples collected from individuals from North America, where filariasis is not endemic, were also positive for anti-WSP antibodies. In comparison to those from Haitians, anti-WSP antibodies from North Americans primarily recognized a distinct region of WSP located within the highly conserved second transmembrane domain. The results of this study demonstrate that anti-WSP antibody responses are associated with the presence of chronic filarial morbidity and not filarial infection status in humans and suggest that WSP should be further studied as a potential trigger for the development of filarial disease.

Sensitivity and specificity of ultrasound detection and risk factors for filarial-associated hydroceles.

To better understand risk factors for hydrocele as a consequence of Wuchereria bancrofti infection, 342 men more than 15 years of age in an endemic area in Papua New Guinea were evaluated. Thirty-four subjects (9.9%) had hydrocele by physical examination. Ultrasound examination detected hydroceles in 57 men (16.7%). Compared with ultrasonography, the sensitivity of physical examination was 44.3%, the specificity was 98.2%, and the positive predictive value was 73.5%. Hydrocele was independently associated with age (odds ratio [OR] = 3.3, P < 0.01) and intensity of infection as determined by filarial antigenemia (OR = 2.3, P = 0.07). Dilation of spermatic cord lymphatics detectable by ultrasound did not correlate with hydrocele, but was associated with the presence of infection. These observations suggest that filarial pathology of the male genitalia is under-reported when evaluated by physical examination alone and that duration and intensity of infection are risk factors for hydrocele.

Rapid assessment for lymphatic filariasis in central Nigeria: a comparison of the immunochromatographic card test and hydrocele rates in an area of high endemicity.

The rapid immunochromatographic card test (ICT) for Wuchereria bancrofti circulating filarial antigen is being used to map areas endemic for lymphatic filariasis. However, the ICT is expensive; thus, surveys based on this test must be relatively limited. Our study was conducted to determine if village-based hydrocele surveys could be used to supplement the ICT surveys in the mapping activities. We compared in 144 Nigerian villages the two assessment methods, ICT and examination for clinical hydrocele, in random samples of 30 adults selected using a procedure that obtained 15 younger males (reported age = 16-39 years old) and 15 older males (> or = 40 years), based on the assumption that hydrocele rates may be more prevalent in older age groups. The men were asked if they had scrotal swelling, then examined and tested by the ICT. We found a weakly positive correlation between village prevalence determined by the ICT and hydrocele (r = 0.041, P < 0.001). Only villages with hydrocele rates of 20% or greater were also consistently classified as having endemic filariasis by the ICT. There was no correlation between an individual's ICT positivity and clinical presence of hydrocele, and questioning about scrotal swelling was not predictive for presence of hydrocele. More research is needed to determine if community level hydrocele prevalence surveys can offer an economical and broadly applicable supplement to the ICT for determining the endemicity of filariasis.