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potential of the parasite, the immune response of the host, and external ('complicating') bacterial and fungal infections. While genital damage (particularly hydroceles) and lymphoedema / elephantiasis are the most recognizable clinical entities associated with lymphatic filarial infections, there are much earlier stages of lymphatic pathology and dysfunction whose recognition has only recently been made possible through ultrasonographic and lymphoscintigraphic techniques. For example, ultrasonography has identified massive lymphatic dilatation around and for several cm beyond adult filarial worms which, though they are in continuous vigorous motion, remain 'fixed' at characteristic sites within lymphatic vessels. Histologically, dilatation and proliferation of lymphatic endothelium can be identified, and the abnormal lymphatic function associated with these changes can be readily documented by lymphoscintigraphy. Interestingly, despite the earlier paradigm that pathology associated with lymphatic filarial diseases was primarily the result of immune-mediated inflammatory responses, all of these changes can occur in the absence of such overt inflammatory responses and, even by themselves, can lead to both lymphoedema and hydrocele formation. The immune system during the development of this 'non-inflammatory pathology' is to keep itself 'down-regulated' through the production of contra-inflammatory immune molecules; specifically, the characteristic mediators of Th2-type T-cell responses (IL-4, IL-5, IL-10) and antibodies of the IgG4 (non-complement-fixing) subclass that serve as "blocking antibodies". Such adaptations do, of course, serve to promote the biological principle of parasitism in which a satisfactory balance between parasite 'aggressiveness' and host responsiveness must evolve to maintain this special relationship. Animated documentation of the fi
ORGANISM 1: Wuchereria bancrofti GENERAL CHARACTERISTICS COMMON NAME: Bancroft's filariasis, elephantiasis worm. GEOGRAPHICAL DISTRIBUTION: Worldwide, prevalent in t h e tropics a n d subtropics. PATHOGENESIS: Elephantiasis, fibrosis, thrombi, lymphatic inflammation, granulomatous infiltration. HABITAT: Lymphatic system. VECTOR: Mosquitoes. RESERVOIR HOST: None. INFECTIVE F O R M : Filariform larva. M O D E OF INFECTION: Inoculation. PERIODICITY: Nocturnal with subperiodic Pacific strains. SPECIMEN OF CHOICE: Blood. SPECIMEN PROCESSING: Giemsa's or Hematoxylin/Eosin stains. Figure 4-30. Life cycle of Wuchereria bancrofti. (continued) MD0842 4-37
timori9. LF is one of the leading causes of permanent and long-term disability in the world and has been targeted for elimination by an important task force24.30. giving rise to drainage of the whitish secretion typical of the disease (Fig. namely Wuchereria bancrofti.8.21. it is estimated that three million individuals live in areas considered to be at risk and approximately 49. This secretion serves as an excellent culturing medium that favors repeated bacterial infections. both because of its social stigma and because of psychosocial damage and economic losses31. in the northeastern region4.26.000 people are infected with W. the advanced stages of the disease7.19. The World Health Organization estimates that close to 20% of the world's population is living at risk of the disease. The largest sample is in the metropolitan area of Recife.11. which affects 120 million people in at least 83 countries throughout the tropics and subtropics33.34 . In Brazil. elephantiasis or hydrocele) caused by these nematodes31. .More than 40 million people suffer from chronic forms of the disease (lymphedema. 1). Filariasis is an important cause of disability. It is believed that around 15 to 20% of infected individuals may evolve to clinical filarial disease14.15. Lymph scrotum or superficial scrotal lymphangiomatosis is a urogenital condition characterized by the presence of lymphatic vesicles on the surface of the scrotal skin that can easily rupture.and approximately 29 million men worldwide present urogenital manifestations of lymphatic filariasis3. Brugia malayi and B. Wuchereria bancrofti is the only lymphatic filarial parasite that induces genital diseases11. bancrofti. It may trigger progression of the condition to lymphedema and scrotal elephantiasis.20.Lymphatic filariasis (LF) is a vector-borne parasitic disease caused by three lymphatic-dwelling nematode parasites.
psychological and socioeconomic repercussions for individuals who present this condition. Pernambuco State (Brazil) is an outpatient clinic that provides care for individuals who have been identified and referred through research projects coordinated by this clinic. It seems to be the complication of LF that produces the greatest incapacitation among men11. It also provides care for patients referred from various public or private clinics for investigations on filarial disease. with lymphangiectasia as the turning point in the natural history of the disease. However. the pathogenesis of lymph scrotum is not well defined15. MATERIAL AND METHODS The National Reference Service for Filariasis (CPqAM/FIOCRUZ) in Recife. There is scarce information in the literature about this morbid condition. Although lymph scrotum is a less frequent manifestation of LF.17.The pathogenesis of the clinical forms caused by adult worms is known. it has important medical. A survey was conducted on the medical records of all patients attended between 2000 and 2007 and seven men with a diagnosis of lymph scrotum on admission . The aim of the present study was to provide support for diagnosis and therapeutic management of this chronic genital morbid condition and to draw attention to the clinical and surgical approaches towards hydrocele among patients living in an endemic area.
6. Over the study period.280 (67. scrotal ultrasonography was performed by radiologists with extensive experience in identifying the "filarial dance sign". All the patients included in this study underwent clinical and urological assessments and laboratory evaluations. All of the patients reported recurrent episodes of acute dermal lymphangioadenitis.were identified. which is a measure of major importance for managing filarial morbidity.095. The patients received guidance regarding genital hygiene.361 patients were attended. In addition. Olinda and Jaboatão dos Guararapes. In case of successive acute crises. The Research Ethics Committee of CPqAM (CEP/CPqAM/FIOCRUZ) gave its approval for this study (CAAE: 0061.2%) were male.0. antibiotic prophylaxis with benzathine penicillin was also used to better control the condition. The parasitological diagnosis was performed using the technique of filtration on a polycarbonate membrane to investigate microfilariae in nocturnal blood samples (thereby respecting the local periodicity of the microfilariae)10. bancrofti in the lymphatic vessels5. five presented previous histories of filarial infection (positive result from the thick smear test at a time prior to the diagnosis of the present study) and only two of these had undergone previous treatment with DEC. All of them came from urban areas in the metropolitan region of Recife that were recognized to be endemic for Bancroftian filariasis: the cities of Recife. The patients with positive parasitological tests (microfilaremia) and/or the "filarial dance sign" on ultrasound examination were treated with diethylcarbamazine (DEC). . The time that had elapsed between the initial clinical manifestations (scrotal pain. of whom 4. increased scrotal volume and localized signs of inflammation) and the diagnosis ranged from two to 15 years. which consists of movements suggestive of the presence of adult filarial worms of W. according the WHO recommendations (6 mg/kg/day for 12 days). RESULTS Seven patients of ages ranging from 26 to 64 years were identified as presenting lymph scrotum. All the patients were from the metropolitan region of Recife. These seven cases were admitted between April 2002 and April 2005.000-07). Among the seven patients. The serological diagnosis was performed using the antigen investigation techniques of ELISA (Enzyme-Linked Immunosorbent Assay) Og4C322 and ICT-AD12 (Immunochromatographic Test)29. which is an endemic area for this disease. as well as to identify other possible urological abnormalities.
Five patients presented microfilaremia. antigenemia positive for Og4C3 and adult Wuchereria bancrofti worms ("filarial dance sign" on scrotal ultrasonography). at least one characteristic relating to the etiology of filariasis could be shown in all the patients. and who present chronic lymphangiectasia13. All of these six patients had undergone hydrocelectomy. The following epididymal and testicular abnormalities were found: textural abnormalities seen on ultrasound. testicular abnormalities (n = 2). to detect late complications. while one had microfilaremia. one had undergone hernioplasty and one had had surgery to remove a nodule from inside the scrotum. nodules in the scrotal region (n = 3). two had undergone another surgical procedure in addition to hydrocelectomy. epididymal abnormalities (n = 2). This condition may be identified following surgery for chylocele or hydrocele and it is a dermatological manifestation of pathological lymphatic drainage11. One patient was only positive for Og4C3 (Table 1). adenopathy (n = 1) corresponding to bilateral inguinal adenomegaly and scrotal lymphangitis (n = 1).27. three presented microfilaremia and tested positive for antigens (Og4C3 and ICT). Of these five. three underwent transversal incisions and two had longitudinal incisions (one unilaterally and the other bilaterally). presence of calcifications. DISCUSSION Lymph scrotum almost always occurs in patients with a previous history of acute episodes of filariasis that are characterized by lymphangitis due to bacterial and/or fungal infection. hydrocele (n = 3). varicocele (n = 2). Among the three patients who presented nodules in the scrotum on urological examination. while in one case the orientation was unknown. only one presented microfilaremia alone. two had already undergone treatment with DEC. increased or decreased testicular volume and presence of a cyst in the head of the epididymis. Among these six. the following are described in Table 2: lymphangiectasia (n = 5). Among the associated findings detected by means of scrotal ultrasonography and/or urological examination. Its prevalence in endemic .Through complementary investigations. but none of them had been followed up from a urological point of view prior to their admission to the National Reference Service. Six of the seven patients had undergone some type of urological treatment prior to the appearance of lymph scrotum. One patient with adult filarial worms who was negative for microfilariae was also serologically positive using the ICT and ELISA Og4C3 techniques. With regard to the orientation of the surgical incision among the six patients with previous hydrocelectomy.
The constant crises caused by repeated infections .3. was not observed in all of the seven individuals enrolled in this study.7. patients with hydrocele from endemic areas for LF who have undergone surgery should have clinical follow-up. Nevertheless. It needs to be borne in mind that there is no "simple hydrocele" in patients living in endemic areas. The occurrence of acute episodes of dermal lymphangioadenitis due to secondary bacterial infection in lymphatic vessels that are more superficial is the greatest risk factor for the appearance of lymph scrotum11. particularly in cases of hydrocele secondary to urogenital filariasis. Furthermore. the transversal orientation should not be defined as the only causal or predisposing factor for lymph scrotum. developed lymph scrotum over the course of their filarial disease. However. The repercussions of lymph scrotum for our patients were dramatic. Nonetheless. The surgical technique appears to be an important factor. ACTON & RAO1 suggested that the appearance of this condition was associated with previous surgery for hydrocele. Some urologists have chosen to make a transversal incision in the scrotum for esthetic reasons. which is the preponderant factor in the appearance of other clinical presentations of filariasis.areas is unknown: only isolated cases have been reported in the literature1. the data from the present study suggest that the transversal orientation for the surgical incision may contribute as a predisposing factor towards establishing lymph scrotum. Although surgeons have recommended partial excision of the sac by means of "bottle neck" inversion of the tunica vaginalis. The pathogenesis of lymph scrotum is still not fully defined15. The presence of lymphangiectasia. the orientation of the scrotal incision seems to have some importance in the appearance of lymph scrotum. In view of the possibility of late complications such as lymph scrotum or genital lymphedema. Prophylaxis and adequate treatment for episodes of acute dermal lymphangioadenitis in damaged lymphatic systems are essential for reducing the risk factors for lymph scrotum.32. the technical difficulty in investigating this anatomopathological substrate needs to be taken into account.17. before deeming that the presence of lymphangiectasia is not a factor required for lymph scrotum to appear. thereby preventing predisposition towards lymph scrotum11. current thinking is that complete excision with meticulous attention to cauterization of the edges and oversuturing offers the best cosmetic and functional result. other surgeons have made the incision longitudinally to avoid sectioning the superficial lymphatic vessels in the skin and subcutaneous tissue. with less risk of lymph scrotum11. Despite the small number of cases evaluated. since two patients who underwent longitudinal incisions. and even one patient without any history of surgery.
In this case series.with continual drainage of milky secretion prevent them from carrying out their work or leisure activities. This may be useful in relation to monitoring the macrofilaricidal effect of drugs5. The importance of the treatment in these patients is reflected in two ways. the repeated infections could evolve into a condition of scrotal elephantiasis. This concerns the finding that five patients presented filarial morbidity and microfilaremia. Moreover. and may even have harmed their social interactions. At the times of their crises. either in mass drug treatment or in routine individual investigation. The treatment for this morbid condition is based on . the recommended treatment in these regions is a single dose of ivermectin and albendazole18. with the risk of reinfection. Furthermore. Thus. Thus. With regard to the clinical evolution of the disease. Therefore. It has been recognized in endemic areas that the formation of intrascrotal nodules following treatment with antifilarial drugs (or spontaneously) expresses the death of adult worms. it is important to highlight the fact that these individuals continue to live in endemic areas. All these circumstances together caused psychological stress that could lead these patients to depression. However.28. it was observed that this took place over a long period ranging from two to 15 years. only two had undergone treatment with DEC. even knowing that the group of patients with chronic manifestations of filariasis is not the principal source involved in the transmission chain. together with other information. toilet paper or cloth.23. treatment for these individuals when they present microfilaremia may be of fundamental importance for controlling transmission. these patients also complained that it was impossible to have sexual intercourse. but their spread and future cases can be prevented through delivery of drug combinations to populations where the disease is prevalent. it brings out a particular feature observed in relation to the disease transmission chain. this presentation was characterized as chronic. lack of treatment for individuals with microfilaremia may have an influence on the time taken for the disease to evolve and thus make it possible for greater morbidity to occur. Among the five patients with a previous history of filariasis. just like lymphedema3. In order to carry out their routine activities. Thus. In clinical practice.16. Secondly. the presence of nodules was detected in three patients. may be taken to be an indirect sign of previous filarial infection. Firstly. Since DEC must not be given because of potential side effects during coinfection with onchocerciasis. they must be included within the local filariasis control program. the finding of these nodules. it has commonly been observed that patients with chronic disease do not present microfilaremia5. of whom two had made use of DEC. at the individual level. Chronic filarial manifestations cannot be cured.12. this possibility cannot be ignored. some patients used inventive precautions such as covering the scrotal region with plastic.
and surgical guidelines are available from WHO for endemic filariasis areas32. 55/2004CPqAM/FIOCRUZ). These measures consist of local hygiene care. Knowledge of the profile of lymph scrotum patients highlights the importance of adequate surgical approaches towards hydrocele cases in endemic areas. However. Among the urogenital surgical approaches. There is no consensus on the management methods for different urogenital manifestations. REFERENCES . The need for laboratory investigation and specific treatment is emphasized in this management. lymph scrotum and other urogenital manifestations such as scrotal elephantiasis and chyluria have received less attention from the global program for eradication of lymphatic filariasis2. Furthermore. 25380. with the aim of minimizing the incapacitation produced by the effects of this disease in man11. Urogenital surgery is considered by WHO to be a critical point in controlling filarial morbidity. with the aim of impeding progression within the clinical evolution of the disease and breaking the transmission chain. the importance of knowing these patients' profile is emphasized with regard to discussing the viability of reparative surgical approaches for the condition of lymph scrotum. It also highlights the importance of appropriate surgical and clinical management of this morbid condition. by means of reviews and expert group discussions25. and antibiotic prophylaxis using benzathine penicillin3. in the same way as proposed for lymphedema in the limbs. There is an urgent need to develop such a consensus. in some cases. 814/2006 FIOTEC/SVS) and FACEPE (process no. in accordance with the guidelines of the global program for elimination of lymphatic filariasis.004212/2004-00. considering that this chronic disease is of irreversible nature. surgery for hydrocele has been the key point within control programs for this morbid condition. since even patients with the morbid condition may present microfilaremia. surgical reconstruction has been shown to be efficient 11. However. ACKNOWLEDGEMENTS This work was supported by Centro de Pesquisas Aggeu Magalhães/Departamento de Parasitologia/SRNF (project # 573-17. accord no. with the aim of eliminating a possible risk factor for the appearance of this condition. The points made here have had the aim of contributing towards improving the filariasis control actions.conservative measures that avoid its progression.
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