Case Reports
Chronic Wuchereriasis Presenting as a Vaginoperineal Fistula Report of a Case with Aspiration Cytologic Diagnosis
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ferential diagnosis of filarial species on cytologic specimens. Chronic wuchereriasis presenting as a vaginoperineal fistula has not been reported previously. (Acta Cytol 2005; 49:335–338)
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Filariasis is often an occult disease with myriad presentations. Cytology has an established role in diagnosing clinically unsuspected cases.
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Background
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Prashant Sharma, M.B.,B.S., Neeta Kumar, M.D., Parul Jain, M.B.,B.S., Renu Gur, M.D., and Shyama Jain, M.D.
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Keywords: Wuchereria banA 20-year-old female present- The privotal role of FNA in establishing crofti, vaginal fistula, aspiraed with recurring perineal the clinically unanticipated diagnosis tion biopsy. ulcers, vaginal discharge and ilariasis is often an ocinguinal lymphadenopathy. underscores the success achieved with cult disease, as lymphRadiology revealed a vaginothis rapid, minimally invasive and edema is the only visible perineal fistula. Fine needle cost-effective modality in tropical manifestation.1 Most asympaspiration (FNA) of the ulcer tomatic patients have subbed and smears prepared from countries. clinical lymphatic damage.2 the ulcer discharge showed an The specific diagnosis in a adult, gravid, female filarial clinically suspected case is achieved principally by deworm and unsheathed larvae. Similar microfilariae were tection of microfilariae in peripheral blood or by seroalso seen in FNA smears from inguinal lymph nodes. The logic methods.3 However, detection of microfilariae atypical clinical presentation and unusual parasite moror a gravid/nongravid female or male worm in cytophology posed initial difficulties in characterization of the logic samples from various body sites has been reportmicrofilarial species. ed frequently in clinically asymptomatic cases.4-6 Conclusion A case of chronic lymphatic filariasis with an unusual presentation and clinically unsuspected diagnosis is This case report highlights the morphologic clues to the difFrom the Departments of Pathology and Microbiology, Maulana Azad Medical College and Lok Nayak Hospital, New Delhi, India. Drs. Sharma and P. Jain are Residents, Department of Pathology. Drs. Kumar and S. Jain are Professors, Department of Pathology. Dr. Gur is Senior Specialist, Department of Microbiology. Presented as a poster at the XVIIIth Annual Conference of the Indian Association of Pathologists and Microbiologists (Delhi Chapter), Delhi, April 6, 2003. Address correspondence to: Prashant Sharma, M.B.,B.S., Flat 188, Block C2A, Pocket 16, Janak Puri, New Delhi—110058, India (
[email protected]). Financial Disclosure: The authors have no connection to any companies or products mentioned in this article. Received for publication December 15, 2003. Accepted for publication May 29, 2004.
0001-5547/05/4903-0335/$19.00/0 © The International Academy of Cytology
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Discussion
presented. Fine needle aspiration (FNA) established the diagnosis, although unusual parasite morphology caused initial difficulties in species characterization.
Nearly 1,100 million people live in areas endemic for lymphatic filariasis, and there are an estimated 120 million cases of the disease, either with patent microfilaremia or with chronic filarial disease.7 India contributes about 40% of the global burden of disease and accounts for 50% of the population at risk of infection by this mosquito-borne nematode. Wuchereria bancrofti, the predominant species, accounts for about 98% of cases in India, while Brugia malayi is restricted in distribution.1,8 Other species of filaria, of which there are > 200, have only rarely been reported from the Indian subcontinent.9 The spectrum of clinical illness is large and varied,
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A 20-year-old female living in Bihar, India (known to be a zone endemic for filariasis) presented with recurring ulcers in the perineal region. The ulcers initially developed as furuncles in the skin; they then ruptured to form open sores, intermittently discharging pus. These healed slowly over the next 2–3 months, only to be followed by another furuncle in a few months. The whole process had been repeating itself for nearly 10 years. The perianal lesions had occasionally been incised and drained in the past, but this had not prevented their recurrence. There was a history of vaginal discharge in the patient, who was unmarried and denied having sexual contact. She had been treated on different occasions for pelvic inflammatory disease and perianal “sinuses,” including a course of antitubercular therapy empirically, with no response. On examination, the perineal skin showed a single ulcer surrounded by pouting granulation tissue. The lesion was nontender, and a purulent discharge could be expressed on applying pressure. The ulcer base was indurated and fixed to the underlying structures. Multiple puckered scars were present around the lesion. Two small lymph nodes, each measuring 0.5 × 0.5 cm, were palpable in the right inguinal region. Digital rectal as well as proctoscopic examinations did not reveal an inner opening of the fistula. The patient did not consent to vaginal examination. The clinical differential diagnosis included fistula-in-ano secondary to recurrent anorectal abscesses of unknown etiology, scrofuloderma and hidradenitis suppurativa. Routine blood counts and biochemistry were within normal limits. Radiography of the pelvis showed a small, calcified nodule in the pelvis (Figure 1A). A fistulogram established that the perianal sinus was actually a high vaginoperineal fistula (Figure 1B). Meanwhile, FNA of the base of the anal ulcer and smears prepared from the purulent discharge revealed numerous microfilariae and embryonated ova within body parts of a gravid, female worm (Figure 2A). FNA of a right inguinal lymph node also revealed mostly degenerated and a few well-preserved microfilariae (Figure 2B). Noteworthy features in the microfilariae were absence of a sheath, a round cephalic end and a tail tip free of nuclei (Figure 2C). Peripheral blood film showed 12% eosinophils, but no hemoparasites were seen. Serologic testing for antifilarial IgG by enzyme-linked immunosorbent assay (ELISA) was strongly positive. The final diagnosis was recurring vaginoperineal fistula due to Wuchereria bancrofti infection.
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B Figure 1 (A) Pelvic radiograph showing small, calcific nodule. (B) Fistulogram. Radiopaque dye delineating the vaginoperineal fistula.
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space in both is larger than the diameter of head but was equal in our case.12 Ulcerative skin lesions are seen in onchocerciasis, but the morphologic trait of multiple columns of small nuclei in our case virtually ruled out that species, which has characteristic large nuclei in a single column.10,12 In view of these features, a species diagnosis of W bancrofti, the most common species in India, was considered next. W bancrofti larvae are normally sheathed, but the sheath may be lost during smearing or go unnoticed due to weak staining in Giemsa preparations.13 The rounded tail tip, cephalic space equal to the diameter of the head, multiple columns of small nuclei and peripheral blood eosinophilia helped consolidate the diagnosis.12 Serology for antifilarial IgG, usually by ELISA utilizes a crude, heterologous, antigenic preparation. A positive result does not differentiate between species, and false positives may occur with other helminthic infections, including strongyloidiasis.10 In conclusion, the present case highlights a clinically and cytologically uncharacteristic presentation of filariasis. The pivotal role of FNA in establishing the clinically unanticipated diagnosis underscores the success achieved with this rapid, minimally invasive and cost-effective modality in tropical countries. To the
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but asymptomatic microfilaremia is the most common manifestation of bancroftian filariasis.10 Much less frequent are recurrent inflammatory lesions. Localized inflammatory nodules in the breast, scrotum and subcutaneous tissue are recognized.4-6,10 Infected superficial nodes, usually inguinal, may suppurate and form sterile abscesses that often leak lymph before rapidly healing with a scar.10 In the present case, a long-standing anorectal abscess possibly repeatedly burst through the perineal skin or into the vagina. The presence of adult worm and microfilariae in the abscess (as confirmed on cytology of the discharge) and superimposed bacterial infections due to the location may have caused delayed healing and recurrence of the fistula. The presence of a parasite or associated granulomatous response is a known cause of nonhealing of sinuses and fistulae.11 Morphologic appearances of various microfilarial species are well characterized. Variations from the usual, however, can cause difficulty, as in the present case. The differential diagnosis of unsheathed microfilariae with a tail tip free of nuclei includes Onchocerca volvulus and Mansonella ozzardi. The tail tip in both of them, however, is curved and pointed, while it was rounded in the present case. Moreover, the cephalic
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Figure 2 (A) Gravid female worm with numerous embryonated ova. Many coiled larvae are visible (Giemsa stain, × 400). (B) Lymph node smear showing a microfilaria in a chronic inflammatory background (Giemsa stain, × 480). (C) Unsheathed microfilaria with cephalic space equal to the diameter of the head and multiple columns of small nuclei (Giemsa stain, × 700).
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best of our knowledge, this is the first report of bancroftian filariasis presenting as nonhealing perineal ulcers and complicated by a vaginoperineal fistula.
phatic filariasis. Acta Cytol 1996;40:948–952 6. Kapila K, Verma K: Gravid adult female worms of Wuchereria bancrofti in fine needle aspirates of soft tissue swellings: Report of three cases. Acta Cytol 1989;33:390–392
Acknowledgments
7. Michael E, Bundy DA, Grenfell BT: Re-assessing the global prevalence and distribution of lymphatic filariasis. Parasitology 1996;112:409–428
The authors gratefully acknowledge Dr. Lawrence R. Ash, Professor Emeritus of Infection and Tropical Medicine, Department of Epidemiology, School of Public Health, University of California, Los Angeles, for his expert opinion and help with the species diagnosis.
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8. Sabesan S, Palaniyandi M, Das PK, Michael E: Mapping of lymphatic filariasis in India. Ann Trop Med Parasitol 2000; 94:591–606 9. Dissanaike AS, Abeyewickreme W, Wijesundera MD, Weerasooriya MV, Ismail MM: Human dirofilariasis caused by Dirofilaria (Nochtiella) repens in Sri Lanka. Parasitologia 1997;39: 375–382
References
10. King CL, Freedman DO: Filariasis. In Hunter’s Tropical Medicine and Emerging Infectious Diseases. Eighth edition. Edited by GT Strickland. Philadelphia, WB Saunders, 2000, pp 740– 754
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