Journal of Tropical Pediatrics, 2015, 61, 304–309 doi: 10.1093/tropej/fmv041 Advance Access Publication Date: 30 June 2015 Case Report

CASE REPORT

Testicular Schistosomiasis Mimicking Malignancy in a Child: A Case Report 1

Sub-Department of Paediatric Surgery, University of Nigeria Teaching Hospital, Enugu 400001, Nigeria 2 Department of Morbid Anatomy, University of Nigeria Teaching Hospital, Enugu 400001, Nigeria Correspondence: Sebastian O. Ekenze, Sub-Department of Paediatric Surgery, University of Nigeria Teaching Hospital, Enugu 400001, Nigeria. Tel: 234-803-777-3831. E-mail .

ABSTRACT Schistosomiasis is an important communicable disease in the developing world. However, testicular schistosomiasis is an extremely rare condition. We report a case of testicular schistosomiasis mimicking testicular tumour in a 13 year old who presented with huge unilateral testicular mass. The dilemma encountered in the diagnosis and treatment of this child is presented to highlight the need for high index of suspicion of this pathology in children with testicular mass presenting from schistosomiasis-endemic areas. K E Y W O R D S : Schistosomiasis, testis, tumour, child

INTRODUCTION Schistosomiasis is a parasitic disease caused by blood-dwelling fluke worms (trematodes) of the genus Schistosoma [1]. It is a devastating tropical disease and a major source of morbidity and mortality for developing countries in Africa, South America, the Caribbean, the Middle East and Asia [1, 2]. The main disease-causing species are Schistosoma haematobium, Schistosoma mansoni and Schistosoma japonicum. Others are Schistosoma mekongi and Schistosoma intercalatum [1, 2]. Schistosomiasis commonly involves urinary bladder, causing haematuria, or large bowel, causing bloody stool. Involvement of the testis is a very rare occurrence [3]. Of the species, only S.mansoni and S.haematobium have been isolated in testicular schistosomiasis [3]. The hypothesized

pathophysiology is a haematogenous spread of schistosomal eggs via venous communication between internal spermatic vein and mesenteric vein [1]. Presentations may include a testicular mass, haematuria, lower urinary symptoms and haemospermia and infertility (in adults) [3, 4]. Diagnosis of this condition is challenged by the lack of index of suspicion of the pathology in the affected patient [1, 3, 4]. Because of the presentation of most of these cases as a painless testicular mass, they tend to mimic malignancy and may cause a diagnostic difficulty [3–5]. We report a case of testicular schistosomiasis mimicking testicular tumour in a 13 year old who presented with huge unilateral testicular mass. The focus is on highlighting the challenges of managing

C The Author [2015]. Published by Oxford University Press. All rights reserved. For Permissions, please email: [email protected] V



304

Downloaded from http://tropej.oxfordjournals.org/ at University of Georgia on September 28, 2015

by Sebastian O. Ekenze,1 Victor O. Modekwe,1 Martin A. Nzegwu,2 Samuel C. Ekpemo,1 and Uchechukwu O. Ezomike1

Testicular Schistosomiasis Mimicking Malignancy in a Child

this case and the need to consider this diagnosis in children with testicular mass presenting from schistosomiasis-endemic areas.

305

siblings and the parents. His source of water was stream. The patient was referred to our unit following a failed herniotomy during which a ‘hard testis’ was discovered. Physical examination was significant for mild pallor, non-tender 8 cm  6 cm, hard, nonreducible left hemiscrotal swelling with an oblique inguinal scar (Fig. 1). The left testis was not distinct from the mass and the inguinal nodes were not palpable. There was reducible right inguinoscrotal hernia. Laboratory investigations revealed haemoglobin level of 8.0 g/dl, serum alpha-feto-protein level of 3.0 ng/ml and absence of ova of Schistosoma in urine and stool specimen. Sonogram showed a left scrotal mass of 7.7 cm (in its widest dimension) with heterogenous echogenicity and intact margin. The right testis measured 3.2 cm  2.1 cm and had homogenous echotexture. There was no enlargement of paraaortic nodes.

Fig. 1. The 13 year old with hard and non-reducible left hemiscrotal swelling. Note the oblique inguinal scar from previous attempt at surgery. The left testis was not distinct from the mass, and the inguinal nodes were not palpable.

Downloaded from http://tropej.oxfordjournals.org/ at University of Georgia on September 28, 2015

CASE REPORT A 13 year old male presented with left scrotal swelling that dates back to infancy, and intermittent haematuria of 5 years. He has been domiciled with his parents at Ishielu in Ebonyi state of Nigeria (one of the schistosomiasis-endemic areas in Nigeria). The scrotal swelling has been progressive and spontaneously reducible up until 2 years before presentation, when it became irreducible. There were no associated gastrointestinal obstructive symptoms. The haematuria was terminal and intermittent, but has not warranted blood transfusion. There was no associated weight loss, no history of undescended testis and no history of haematuria among the



306



Testicular Schistosomiasis Mimicking Malignancy in a Child

by numerous non-caseating granulomas centred on viable and degenerating schistosomal ova; some of the latter were calcified. Numerous eosinophils and fibrous tissue were seen around the granulomas. No atypical cells were seen (Figs 3 and 4). He was commenced on praziquantel following the histopathological confirmation of schistosomiasis. The patient has been followed up for 3 months after surgery and is scheduled for regular clinical and ultrasound evaluation of the scrotum. DISCUSSION Schistosomiasis commonly involves the urinary bladder or the large bowel [1, 3]. However, unusual presentation of schistosomiasis has been described in the appendix, prostate, ovary, fallopian tube and cervix [3, 6–8]. In these cases, the lesions are not clinically suspicious of the disease and the diagnosis is made

Fig. 2. Finding at inguinoscrotal exploration. Irregular hard mass without distinct testis and epidydimis.

Downloaded from http://tropej.oxfordjournals.org/ at University of Georgia on September 28, 2015

The patient was optimized for operative treatment. He had exploration of the left scrotal mass via the inguinal approach. The finding at the operation was a 10 cm  6 cm, hard and irregular mass with a scar from a previous surgery and significant fibrous tissues (Fig. 2). The scrotal mass was completely excised. Remarkably, inspection of the right testis during right herniotomy showed a suspicious nodule of 0.5 cm. The nodule was excised. Post-operative period was uneventful, and he was discharged on post-operative day three. Gross anatomy of the cut scrotal mass showed an outer surface that is well encapsulated and rough. The internal surface is greyish white with predominantly solid areas and few cystic areas that contain jelly-like substance in their loci. Histology sections of the left scrotal mass and the nodule on the right testis showed total replacement of the normal testicular architecture

Testicular Schistosomiasis Mimicking Malignancy in a Child



307

Fig. 4. At lower magnification (10) showing extensive areas containing schistosomal ova with accompaying granulomatous orchitis.

Downloaded from http://tropej.oxfordjournals.org/ at University of Georgia on September 28, 2015

Fig. 3. Histopathological examination of the testicular tissue. At 600 magnification showing Schistosoma ova with terminal spine and accompanying granulomatous orchitis destroying the surrounding seminiferous tubules with fibrosis.

308



Testicular Schistosomiasis Mimicking Malignancy in a Child

Imaging with ultrasound to demonstrate testicular lesion with hypoechoic or heterogenous echotexture or with magnetic resonant imaging (MRI) to show irregular tunica have been reported to raise suspicion for the granulomatous process [3, 5, 10, 12, 13]. These findings are non-specific and can be produced by other scrotal infestations like filariasis, paracoccidioidomycosis and tuberculosis and can create more diagnostic challenges [3, 5, 9, 14]. Of more ominous significance is that similar findings have been demonstrated in testicular cancer, and because neither an ultrasound nor an MRI allows differentiation between benign and malignant disorders, tissue diagnosis may be imperative [9, 13, 14]. Testicular schistosomiasis is potentially treatable with medical therapy [1, 3, 9]. The recognition of testicular schistosomiasis as different from testicular tumours is crucial, and the use of frozen biopsy in suspicious testicular lesion has been useful in diagnosis and this avoids unnecessary orchidectomy [13]. However, in a setting like ours, where frozen section is not available, therapeutic dilemma may arise. In such a situation, regular biopsy of the testicular lesion can be undertaken via an inguinal approach as an initial step. If the histology result indicates schistosomiasis, the patient will be managed with medical therapy, i.e. praziquantel, 400 mg at the beginning. On the other hand, if the histology indicates testicular cancer, the patient will then need orchidectomy and other adjuvant therapy. In this way, unnecessary orchidectomy will also be avoided. It may be necessary to evaluate the contralateral testis to exclude its involvement with the disease. The incidental finding during herniotomy on the apparently unaffected testis has shown that both testes may be affected despite absence of clinical and radiological evidence of the disease. Follow-up of the patients is imperative to ensure eradication of the disease. CONCLUSION Testicular schistosomiasis is a rare sequelae of human schistosomiasis. The clinical manifestations of the disease may not raise suspicion for schistosomiasis and might mimic testicular tumour. It is imperative to have a high index of suspicion for testicular schistosomiasis in boys or adult males presenting with testicular mass from schistosomiasis-

Downloaded from http://tropej.oxfordjournals.org/ at University of Georgia on September 28, 2015

histologically as incidental finding. Involvement of the testis is rare. Some cases have been reported in adults and children [1, 3, 4, 5, 9]. The pathogenesis of testicular schistosomiasis may be through larva migration from the lungs to the veins and subsequent lodgement in the testicular veins via either the mesenteric veins (S.mansoni) or vesical plexus (S.haematobium) [1, 3]. Excretion of the eggs in the testis causes granulomatous inflammation. Previous reports have shown that testicular schistosomiasis does not manifest peculiar diagnostic clinical features [3, 4, 5, 9]. In the present report, the patient had chronic unilateral scrotal swelling, which was initially reducible but later became irreducible. Scrotal swelling with or without pain or the subsequent changes noticed in the index patient, clinically, will not raise suspicion for schistosomiasis. Rather the physical finding of painless and hard testicular mass in the patient and testicular nodules in some previous cases may point to tuberculous orchitis, or testicular tumour, which may be prevalent at this age [3, 5]. What would have raised suspicion of schistosomiasis was residence in an endemic area and the presence of intermittent haematuria. However, as revealed in this patient and in previous reports [3, 5], the absence of common symptoms of schistosomiasis and the inability to demonstrate Schistosoma ova in the urine challenged the diagnosis of schistosomiasis. Other presentations reported in older patients include hydrocele from lymphatic obstruction in the tunica, infertility, scrotal pain from epididymitis, chronic scrotal dermatitis and testicular atrophy [3, 5, 9]. These clinical features are also not typical of schistosomiasis. Definitive diagnosis of testicular schistosomiasis involves histological demonstration of chronic granulomatous inflammation containing Schistosoma eggs in the specimen of testicular mass [3, 4, 5, 9, 10]. Demonstration of the ova in urine and stool specimen can be disappointing, as our report and the previous reports [3, 5, 10] have shown. Some authors [1, 4] have suggested that in the presence of risk factors, Schistosoma serology testing might be performed in individuals with unilateral testicular swelling. More recently, diagnosis of active S.haematobium infection has been enhanced by detection of specific DNA fragments from filtered urine samples [11]. This has been shown to be more sensitive than egg detection.

Testicular Schistosomiasis Mimicking Malignancy in a Child

endemic area. This will engender focused management and avoid unnecessary orchidectomy. FUNDING The research was funded by the authors.

REFERENCES

8.

9.

10.

11.

12.

13. 14.

309

Mazigo HD, Zinga M, Heukelbach J, et al. Case series of adenocarcinoma of the prostate associated with Schistosoma haematobium infection in Tanzania. J Glob Infect Dis 2010;2:307–9. Batista TP, deAndrade IJR, daFonte Filho LAM. Schistosoma mansoni: an unusual cause of ovarian pseudotumor. Arch Gynecol Obstet 2010;281:141–3. Adisa J, Egbujo EM, Yahaya BA, et al. Primary infertility associated with Schitosoma mansoni: a case report from the Jos plateau, north central Nigeria. Afri Health Sci 2012;12:563–5. Lopes RI, Leite KR, Prando D, et al. Testicular schistosomiasis caused by Schistosoma mansoni: a case report from Brazil. Braz J Infect Dis 2007;11:523–4 Ibironke OA, Phillips AE, Garba A, et al. Diagnosis of Schistosoma haematobium by detection of specific DNA fragments from filtered urine samples. Am J Trop Med Hyg 2011;84:998–1001 Wedel PG, Jess P. Testicular schistosomiasis simulating malignancy. Case report. Scand J Urol Nephrol 1991;25: 237–8 Al-Qahtani SM, Droupy SJ. Testicular schistosomiasis. Saudi Med J 2010;31:325–7 de Cassio Saito O, de Barros N, Chammas MC, et al. Ultrasound of tropical and infectious diseases that affect the scrotum. Ultrasound Q 2004;20:12–8

Downloaded from http://tropej.oxfordjournals.org/ at University of Georgia on September 28, 2015

1. Gryseels B, Polman K, Clerinx J, Kestens L. Human schistosomiasis. Lancet 2006;368:1106–18 2. World Health Organization. Schistosomiasis. Wkly Epidemiol Rec 2010;85:158–64. 3. Rambau PF, Chandika A, Chalya PL, et al. Scrotal swelling and testicular atrophy due to schistosomiasis in a 9-yearold: a case report. Case Rep Infect Dis 2011;2011:787961. doi: 10.1155/2011/787961 4. Periyasamy P, Subramaniam SR, Rajalingham S. An increasingly notorious mimicker of testicular tumours; crossing borders. BMJ Case Rep 2011. doi:10.1136/ bcr.09.2011.4816. 5. Ondo CZ, Sarr A, Sow Y, et al. Testicular bilharzioma by Schistosomia haematobium: about two cases. Prog Urol 2014;24:67–9. 6. Mazigo HD, Giiti GC, Zinga M, et al. Schistosomal peritonitis secondary to perforated appendicitis. Braz J Infect Dis 2010;14:628–30.

7.



Testicular Schistosomiasis Mimicking Malignancy in a Child: A Case Report.

Schistosomiasis is an important communicable disease in the developing world. However, testicular schistosomiasis is an extremely rare condition. We r...
639KB Sizes 0 Downloads 11 Views