Vol.2, No.7, 427-431 (2013) Case Reports in Clinical Medicine http://dx.doi.org/10.4236/crcm.2013.27112 Copyright © 2013 SciRes. OPEN ACCESS Strongyloides stercoralis in an immunocompetent adult: An unexpected cause of weight lost Lia Marques1*, André Rodrigues1, Elisa Vedes2, Dores Marques1 1Serviço de Medicina III, Hospital de Pulido Valente, Centro Hospitalar Lisboa Norte, Lisboa; *Corresponding Author: marques.lia@gmail.com 2Unidade de Cuidados Intensivos do Hospital da Luz, Lisboa Received ************* 2013 Copyright © 2013 Lia Marques et al. This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. ABSTRACT S trongyloides stercoralis infect s at least 100 mil- lion humans worldwide each year, but its preva- lence is underestimated. It is endemic in hot and humid climates as well as resource poor coun- tries with inadequate sanitary conditions. The rise of international travel and immigration has a positive impact in strongyloidiasis. Due to its unique auto infection life-cycle, Strongyloides may lead to chronic infections remaining unde- tected for decades. Strongyloidiasis is most of- ten asymptomatic but it has a wide ra nge o f c lin i- cal presentations. The two most severe forms of strongyloidiasis are hyperinfection and dissemi- nated syndromes. These occur most often in pa- tients with impaired cell mediated immunity. A 42-year-old immunocompetent man presented with chronic watery diarrhea, malaise, upper ab- dominal pain, anorexia and weight lost. Strongy- loides stercoralis was identified in stool sam- ples and duodenal biopsy. The patient was suc- cessfully treated with albendazole. The authors report a case of strongyloidiasis hyperinfection in an immunocompetent host 20 years away from an endemic area and make a literature re- view. Keyw ords: Strongyloides st er corali s; Immunocompetent; Hyperinfection 1. INTRODUCTION Strongyloides stercoralis (S. stercoralis) is an intesti- nal nematode endemic in tropical and subtropical coun- tries and in the Southeastern United States [1-5]. The helminth is also present in some restricted areas of the European Union such as northern Italy, rural Romania or southeastern Spain [4]. S. stercoralis affects 30 - 100 mil- lion people worldwide [4,6,7], but this current estimate dates back to review articles published between 1989 and 1996 [2,8]. Recen t pu blication s state that strongyloidiasis remains an underestimated public health problem [1,4,9, 10]. A 10-year multicenter surveillance program per- formed in Spain and published in 2013, highlights the high prevalence of S. stercoralis cases imported by im- migrants, most being asymptomatic and with eosinophilia [4]. Strongyloidiasis is caused by the female nematode S. stercoralis. It’s life cycle is comprised of 2 parts: a free- living cycle outside of the host as rhabditiform larvae and a parasitic life cycle as infective filariform larvae [5, 11-14]. During the free-living cycle in the soil, Strongy- loides transform from rhabditiform larvae into infective filariform larvae, which penetrate the human skin and proceed into the submucosa, then into the venous circu- lation, and then toward the right heart and lungs [5,11- 14]. During the pulmonary maturation process, Strongy- loides larvae induce alveolar capillary bleeding and po- tent eosinophilic inflammation, resulting in eosinophilic pneumonitis [5,11-14]. From the alveoli, the larvae mi- grate up the pulmonary tree and trachea, reaching the larynx, were they are swallowed and travel to the stom- ach and small bowel [5,11,13]. Inside the GI tract, Stron- gyloides larvae mature into adult female worms that em- bed themselves in the mucosa of the small bowel and produce eggs via parthenogenesis. Within the intestinal lumen, the eggs hatch into non infective rhabditiform larvae, which are excreted, along with stool, into the en- vironment [5,11]. Some larvae, however, re-enter the blood stream through the bowel wall and migrate through lungs, it reenters the host via enteral circulation (endo- autoinfection) or perianal skin (exoautoinfection) by- passing the soil cycle [1,4,5,11-14]. Thus, parasites can remain in the human body for th e remainder of the ho st’s L. Marques et al. / Case Reports in Clinical Medicine 2 (2013) 427-431 Copyright © 2013 SciRes. OPEN ACCESS 428 life. This ability for auto infectio n implies that infestation can be life-long and extremely heavy [1,4,5,15]. Strongyloidiasis is generally benign and asymptomatic, eosinophilia and larvae in stool may be the only indica- tors of infection, but eosinophilia is not obligatory [1,5, 16]. In immunocompromised host, it can cause substan- tial intestinal disease and can disseminate widely to ex- tra-intestinal sites, this clinical form of the infection be- ing called hyperinfection syndrome [1,4,5]. Hyperinfec- tion means accelerated auto infection characterized by development or enhanced GI and pulmonary symptoms with increased numbers of larvae seen in stool or sputum, restricted to the organ of auto infective cycle [1,5]. Typi- cally, hyperinfection syndrome occurs in patients from endemic areas of S. stercoralis who receive immunosup- pressive therapy. The diagnosis in such patients may be difficult because of a lower incidence of eosinophilia. Dis- seminated disease is defined by the presence of parasites outside of the traditional life cycle (i.e. , in organs other than the skin, GI tract, or lungs) [1,5,15,18,19]. Involve- ment of brain, heart, gall bladder, and kidney has been reported [1,6]. Although hyperinfection syndrome can occur in any host, disseminated disease occurs mainly in immunocompromised individuals [5,14,18]. In immunocompetent hosts, Strongyloides typically cause a low-grade chronic infection, which has been seen even up to 40 years after exposure [1,19]. Most patients are completely asymptomatic. Some have mild gastroin-tes- tinal, cutaneous, or pulmonary symptoms with or with- out fever. These symptoms may be acute or they may wax and wane chronically before spontaneous resolution. Gastrointestinal presentations include diarrhea and ab- dominal discomfort, nausea, anorexia, weight loss caused by gastritis, or enteritis with ulceration. Patients also can have occult gastrointestinal blood loss or fat and vitamin B12 malabsorption [20]. Pulmonary symptoms include cough and dyspnea, sometimes associated with wheezing, caused by larval migration through the lungs, or eosino- philic pneumonia [20]. The pathognomonic rash of Strongy- loides infection is larva currens that likely represents an allergic response to migrating larvae [20 ]. The clinical presentation of hyperinfection syndrome is similar to that of classic strongylo idiasis [5]. However, due to increased parasite turnaround and dissemination, patients with hyperinfection syndrome and disseminated disease often have catastrophic clinical manifestations such as shock, disseminated intravascular coagulation, meningitis, renal failure, and /or respiratory failu re [5 ,12 ]. Mortality with hyperinfection can be up to 87% in the immunosu p pressed patients [20]. The diagnosis of S. stercoralis is often delayed due to the presence of sub clinical or poorly symptomatic cases, the usually low parasite load and irregular larvae output and the lack of a gold standard diagnostic test [4]. Diag- nosis is based on detection of larvae in the stool or spu- tum. The sensitivity of a single examination is on ly 25 to 30%, but increases to 70% to 85% with three consecutive stool samples using the agar plate method of larval de- tection [5,20-23]. Detection of larvae in the duodenal aspirates is more sensitive and hence should be consid- ered when there is a clinical suspicion of hyperinfection [24]. In the past years serological and immunological stool tests have been developed in order to improve di- agnostic sensibility in immunocompromised and post transplant patients [24-26]. S. stercoralis serological as- says can simplify the diagnosis and overcome the poor sensitivity of single stoo l exams, but its specificity is less well defined [27]. Problems of cross-reactivity seem to arise especially in areas where other nematodes are also endemic. New and promising tools such as serological methods based on recombinant antigens or PCR are also available in some referral centers. However, the optimal diagnostic strategy, both for epidemiolo gical surveys and for individual diagnosis and screening, has yet to be de- fined [28,29]. Str o ng ylo id e s i nf ec ti on is treated with the aim to erad i- cate strongyloidiasis. In chronic infection, ivermectin (200 mg/kg orally, once daily) for 1 - 2 days or albenda- zole (400 mg orally, twice daily) for 7 days is sufficient. A number of randomized clinical trials have been carried out, showing that ivermectin is the drug of choice, and a single dose is highly effective (over 90%) [9,28,29]. However, drug efficacy may have been overestimated, as faecal-based methods alone have been used to assess cure in almost all studies. Multiple doses may be neces- sary to obtain the goal of eradication in a patient, and the current indications by Wold Health Organization refer to a schedule of two consecutive d ays as a possible alterna- tive to the single dose [28]. In recent years there has been an increasing number of papers concerning strongyloidiasis in medical literature, mainly case reports in immunocompromised and post transplant patients, but also some reports of strongyloi- diasis in immunocompetent patients and with atypical presentations [1,17,20]. The actuality of this re emerg- ing infection justifies the present case report of stron- gyloidiasis in an immunocompetent otherwise healthy patient. 2. CASE REPORT A 42-year-old man was admitted to our hospital with chief complaints of chronic diarrhea for the previous three months. He had a history of passing watery and foul smelling stool. He complained of upper abdominal pain, anorexia and weight lost (4 kilograms over three months). He was natural from Guinea Bissau, and had lived in Portugal for the past 20 years, during this time L. Marques et al. / Case Reports in Clinical Medicine 2 (2013) 427-431 Copyright © 2013 SciRes. OPEN ACCESS 429 he hadn’t return Guinea Bissau. He had been recently medicated with proton pump inhibitor (omeprazole) after a normal upper endoscopy that was performed for upper abdominal pain in the previous mouth. From the past medical history only to point alcoholic habits of 46 g/ ethanol/day. The patient denied other symptoms namely vomits or fever. He had no history of tick exposure, con- tact with sickness, or recent travel. Physical examination revealed a weight of 59.6 kg (body mass index of 18 Kg/m2), dehydration, abdominal examination was notable for diffuse pain and tenderness. Anal exam was not possible due to painful anal fissure and systemic examination findings were normal. There was no lymphadenopathy, clubbing, skin lesions and/or pedal edema. His laboratory investigations were as follows: Total leucocyte count: 13,050 × 109/L with normal differential, reactive C protein mildly elevated (2.8 mg/dL), no throm- bocytopenia and no anemia, mild renal impairment (urea 30.70 mmol/L; Creatinine 141.44 mmol/L), hyponatre- mia (127 mmol/L), hypokalemia (2.7 mmol/L). Chest roen- togram (Figure 1) has shown left hilar opacity sugges- tive of lymph node enlargement. Abdomen ultrasound was normal. During the hospital stating the patient remained apy- retic, with liquid diarrhea (5 to 6 passages of liquid brown stools without blood or mucus). The investiga- tions revealed: Serology for human immunodeficiency virus and acute h ep atitis A, B, and C infection were nega- tive, thyroid function was in the normal range, erythro- cyte sedimentation rate 103 mm in 1 h. Stool fat content was in the normal range. Colonoscopy wasn’t performed due to painf ul ana l fissure. The pa tie nt u nd erw ent upp er endoscopy revealing mild esophagitis and duodenitis with nematodes most sugges- tive of S. stercoralis visualized within the crypts. To in- vestigate consumptive symptoms and clarify the x-ray abn ormality a computer tomography (CT) was performed and showed (Figure 2): pulmonary micro nodules the Figure 1. Chest roentogram—left hilar opacity suggestive of lymph node enlargement. left inferior lobe and the right superior lobe suggesting inflammatory or infectious focus; lymph node enlarge- ment in the left hilar area; intestinal distension without enlargement of abdominal lymph nodes. Bronchial Fi- broscopy revealed generalized inflammatory signs. Bron- choalveolar lavage analysis was negative for mycobacte- rias, bacteria and neoplastic cells. Bacteriological and mycobacterium stool and blood analysis were negative. Serial stool samples were collected and subsequently re- vealed many S. stercoralis larvae. Treatment with oral albendazole (400 mg twice daily) was initiated and the patient completed a 7 day regime with good clinical and laboratory responses. Three months after hospital dis- charge the patient was reevaluated and remained asymp- tomatic, diarrhea subsided and he had a 10 Kilograms weight gain. Serial stool analysis was obtained and consis- tently negative for ova and parasites. 3. DISCUSSION The presented case represents a S. stercoralis infection in an immunocompetent adult. In our patient, stool sam- ples were positive possibly because of a high parasitic burden. The chest roentogram and CT findings may be attributed to the pulmonary phase of the parasite life cir- cle. In fact, chest radiography can reveal diffuse alveolar or diffuse interstitial infiltrates, segmental alveolar infil- trates, or pleural effusions [20]. Endoscopic findings also support the diagnosis. The endoscopic manifestations of strongyloidiasis are broad, ranging from normal appear- ing mucosa to ulcerative and catarrhal duodenitis [5]. Patients with a clinical and histopathologic diagnosis of “idiopathic” eosinophilic gastroenteritis should also be thoroughly evaluated for Strongyloides because larvae may not always be apparent on initial evaluation, and therapy with corticosteroids may lead to fatal hyperin- Figure 2. Computerized tomography imagery with pulmonary micro nodules the left inferior lobe and the right superior lobe suggesting inflammatory or infectious focus; lymph node en- largement in the left hilar area; intestinal distension without enlargement of abdominal lymph nodes. L. Marques et al. / Case Reports in Clinical Medicine 2 (2013) 427-431 Copyright © 2013 SciRes. OPEN ACCESS 430 fection if the diagnosis of strongyloidiasis is missed [5]. Thus, evaluation should include several stool examina- tions and upper endoscopy procedures with duodenal biopsies. Multiple biopsy specimens should be taken to increase the histopathologic yield, even if the duodenal mucosa does not manifest any major abnormalities [5]. In this patient there was no eosinophilia, but it is pre- sent in 50% to 80% of patients with mild infection. In contrast, a low eosinophil count occurs in patients with hyperinfection and disseminated disease [20,30], as seems to be the present case. Low socioeconomic status, alcoholism, white race, and male gender have been associated with higher preva- lence of Strongyloides stool positivity [31]. The patient had all of these factors and was original from an endemic region (Guinea Bissau) and was probably a chronic host for S. stercoralis acquired before his immigration to Por- tugal, 20 years earlier. It is postulated that the steady immigrant influx to the European Union has increased the number of infected carriers with S. stercoralis [4]. But presently there is no data on portuguese epidemiol- ogy for S. stercoralis, thus epidemiological studies are needed. To support this need there is widespread agree- ment in the scientific community that its prevalence is largely underesti mated [1,10,19]. The clinical manifestation of hyperinfection syndrome varies widely and the onset may be acute or insidious. There is no quantitative definition, but it is characterized by an increase in gastrointestinal or pulmonary symp- toms with an increased larval load in th e stool or sputum. GI syndrome manifestations include watery diarrhea, weight loss, vomiting, and occasional bleeding. Usually symptoms of pulmonary strongyloidiasis include cough, dyspnea, wheezing, pulmonary hemorrhage, and pleural effusion [30]. A recent report was however able to iden- tify through questionnaires and stool evaluations that individuals infected by S. stercora lis were more likely to complain of stomach ache [32,33]. This patient presented with an insidious form of disease, but his clinical mani- festations fit the hyperinfection syndrome diagnosis. Strongyloidiasis is commonly reported in immuno- compromised patients; however, our patient was immu- nocompetent as his HIV status was negative, the history of alcoholism may have induced some imunosupression contributing to susceptibility to hyperinfection. The pa- tient responded well to albendazole therapy and there were no Strongyloides larv ae in the stool wh en examined after therapy. That and his favorab le clinical evo lu tion all support the diagnosis. The present patient was unusual in that he was other- wise healthy and immunocompetent who presented with pulmonary and gastrointestinal symptoms of strongy- loidiasis hyperinfection syndrome. He had risk factors for strongyloidiasis: he was natural from an endemic area and had heavy alcohol consumption. The authors high- light the importance of correctly diagnose and treat strongy- loidiasis. 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