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Severe Strongyloidiasis in Corticosteroid-Treated Patients: Case Series and Literature Review

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Severe Strongyloidiasis in Corticosteroid-Treated Patients: Case Series and Literature Review Journal of Infection (2007) 54,18e27 www.elsevierhealth.com/journals/jinf Severe strongyloidiasis in corticosteroid-treated patients: Case series and literature review Laurence Fardet*, Thierry Ge´ne´reau, Jean-Louis Poirot, Bertrand Guidet, Adrien Kettaneh, Jean Cabane Hopital Saint-Antoine, Internal Medicine, Parasitology and Intensive Care Department, 184 rue du Fbg Saint-Antoine, 75012 Paris, France Accepted 22 January 2006 Available online 14 March 2006 KEYWORDS Summary Objective: To describe the main features of severe strongyloidiasis in Strongyloidiasis; corticosteroid-treated patients Strongyloides Methods: We report on 3 cases of corticosteroid-treated patients with severe stron- stercoralis; gyloidiasis and review cases of severe strongyloidiasis in corticosteroid-treated Corticosteroids patients reported in the literature. Results: One hundred and fifty-one cases of severe strongyloidiasis complicated a therapy with corticosteroids were evaluated. The mean age of the patients was 48 Æ 17 years and 71% were men. Corticosteroids were given for hematological ma- lignancies in 34 (23%), systemic lupus erythematosus or vasculitis in 27 (18%), and nephropathy or renal transplantation in 32 (21%). At time of infection, the mean daily dosage of prednisone-equivalent was 52 Æ 42 mg (median: 40 mg) and 84% of patients had received a cumulative dosage of prednisone-equivalent higher than 1000 mg. The total duration of treatment ranged from 4 days to 20 years (6 months or less: 69%). Non-specific gastro-intestinal symptoms were reported in 91% of these patients associated or not with pulmonary complaints. Low-grade fever was present in 54% of patients. Fifty-nine patients (39%) experienced severe bacterial or yeast infection during the course of severe strongyloidiasis. Peripheral eosinophilia was detected at presentation in 32% of patients. Strongyloidiasis was usually confirmed by repeated stool examinations. Thiabendazole was the treatment the more widely used. Eighty-nine patients (59%) deceased during the course of the disease. Conclusions: Severe strongyloidiasis is a risk in every corticosteroid-treated patient who has traveled to a soil-infested country, even if the contact was 30 years prior. This diagnosis should be suspected in patients who either experience unusual gastro-intes- tinal or pulmonary symptoms or suffer from unexplained Gram-negative bacilli sepsis. ª 2006 The British Infection Society. Published by Elsevier Ltd. All rights reserved. * Corresponding author. Tel.: þ33149282373. E-mail address: [email protected] (L. Fardet). 0163-4453/$30 ª 2006 The British Infection Society. Published by Elsevier Ltd. All rights reserved. doi:10.1016/j.jinf.2006.01.016 Severe strongyloidiasis and Corticosteroids 19 Introduction ‘‘strongyloidiasis’’ or ‘‘Strongyloides stercoralis’’. Bibliographies of all articles were scanned for ref- Strongyloides stercoralis (Ss) is an ubiquitous intes- erences not identified in the initial search. For the tinal nematode endemic in sub-Saharan Africa, present analysis we included only the references in Southeast Asia, Latin America as well as in parts of English or French reporting patients treated with south-eastern United States and some European corticosteroids. Data were summarized using countries.1,2 This worm has a complex parasitic a standardized data form, including age, gender, life cycle that causes a long-lived auto-infection origin, current dosage and cumulative dosage of in the host.2 Many patients chronically infected corticosteroids at the time of Ss infection, dura- with Ss are asymptomatic or exhibit only minor tion of corticosteroids therapy at time of cutaneous (larva currens), pulmonary (cough) or infection, symptoms, specimens positive for gastro-intestinal (abdominal bloating and an- Strongyloides larvae, associated infection, eosino- orexia) symptoms. Thus, Ss can persist for decades philia, treatment and evolution. without being diagnosed. Under some conditions, Strongyloidiasis was classified as severe in case Ss can trigger the so-called hyperinfection syn- of (1) severe and persistent gastro-intestinal drome and the life-threatening disseminated forms symptoms with fever and/or marked asthenia, or of strongyloidiasis. The term ‘‘hyperinfection’’ has (2) extra-intestinal symptoms induced by Ss, or (3) been used to denote an increase in the auto-infec- presence of the parasite in extra-intestinal regions. tion cycle resulting in a massive invasion by filari- Inclusion was limited to patients with severe stron- form larvae.3 In these patients, most of whom are gyloidiasis treated by corticosteroids before the symptomatic, worms are easily detectable in bowels onset of strongyloidiasis symptoms, with larvae of Ss and often in lungs. The term ‘‘disseminated’’ has evidenced on biological samples (feces, gastric been used to describe a form of the disease in aspiration, bronchoalveolar lavage, etc.). which worms are also located in extra-intestinal and extra-pulmonary sites. However, these two Case reports forms are clinically difficult to distinguish. These severe forms of the disease are usually associated Case 1 with immunosuppressive or debilitating disorders. In October 2000 an 80-year-old white woman was Long-term corticosteroid therapy, leading to some hospitalized for abdominal pain, vomiting and suppression of inflammatory and immunologic re- diarrhea of 1 month duration. She had been treated ÿ1 sponses and altering the hosteparasite equilibrium, with prednisone (80 mg d ) since August for giant is a well-known condition that predisposes the cell arteritis. The striking findings were fever patient to the occurrence of severe strongyloidiasis. (37.9 C) and a distended painful abdomen with To clarify the spectrum of clinical and biological predominant epigastric tenderness. No cutaneous features of Ss infection in patients receiving lesions were observed. Peripheral blood count ÿ1 ÿ1 corticosteroids, we report herein 3 patients and showed 730 mL eosinophils and 12 000 mL reviewed the English and French language medical neutrophils. A blood culture was positive for literatures. Escherichia coli. Stool examinations showed numer- ous Strongyloides larvae. Upper gastro-intestinal endoscopy showed marked gastritis and duodenitis. Patients and methods Biopsies revealed an acute inflammatory infiltrate and the presence of many Ss ova and larvae, mainly We reviewed data for all patients with severe in the gastric glands. Chest X-rays were normal. She strongyloidiasis associated with corticosteroids was successively treated with antibiotics and thia- therapy admitted to our department between bendazole (2 g dÿ1, 5 days). All symptoms improved January 2000 and January 2005. and repeated stool examinations were assessed We also performed a MEDLINE search, using the without evidence of any parasite. The patient was following terms, for cases reported in the litera- discharged 1 month after her admission. Ss was ture during 1966e2004: ‘‘immunocompromised’’, thought to be imported from Vietnam where the pa- ‘‘immunodepression’’, ‘‘corticosteroids’’, ‘‘gluco- tient lived between 1950 and 1960, without any corticoids’’, ‘‘steroids’’, ‘‘prednisone’’, ‘‘prednis- tropical journey thereafter. olone’’, ‘‘methylprednisolone’’, ‘‘asthma’’, ‘‘leukaemia’’, ‘‘lymphoma’’, ‘‘graft’’, ‘‘vasculi- Case 2 tis’’, ‘‘lupus erythematosus’’, ‘‘rheumatoid arthri- An 85-year-old man who had immigrated from tis’’, ‘‘sarcoidosis’’, ‘‘giant cell arteritis’’, ‘‘crohn Vietnam 40 years previously was diagnosed in March disease’’ or ‘‘ulcerative colitis’’ in addition to 2003 with giant cell arteritis. A treatment with 20 L. Fardet et al. 35 mg dÿ1 of prednisone was initiated. Moreover, be- Literature cases cause of the patient’s origins (and without stool ex- amination), a systematic prophylaxis of Ss infection A total of 163 corticosteroid-treated patients with was prescribed (2 courses of thiabendazole, total: severe Ss infection were found in literature. 1 4 g). While on prednisone 30 mg dÿ , the patient Fifteen of them were not included because of e came back to our department in June 2003 because poorly documented clinical history (n ¼ 5),4 8 on- of persistent abdominal pain of 1 month duration set of strongyloidiasis symptoms prior to steroid e associated with diarrhea, weight loss and the recur- therapy (n ¼ 8),9 17 duplicated case (n ¼ 1)18 or rence of biological inflammatory syndrome. Blood unavailable paper (n ¼ 1)19 leaving 148 patients pressure and heart rate were normal and tempera- corresponding to 108 papers.20e128 ture was 37.7 C. Examination of chest, abdomen The 3 cases mentioned above were merged with and central nervous system was also normal. Hemo- 148 cases retrieved in the literature for an overall 1 globin concentration was 11 g dLÿ , white blood analysis. ÿ1 ÿ1 cell count was 12 500 mL with 500 eosinophils mL . The general characteristics of these 151 pa- ÿ1 C-reactive protein was higher than 100 mg L , tients are summarized in Table 1. All patients but 3 ÿ1 fibrinogen was 7 g L . Chest roentgenogram was received oral corticosteroids (parenteral pulse: 2 normal as well as films of the abdomen. Fresh stool patients, subconjunctival injections of dexameth- examination showed numerous larvae of Ss. Oral asone: 1 patient122). The total duration of cortico- ÿ1 thiabendazole (2 g d , 10 days) was started immedi- steroid therapy at time of infection was available ately and the patient condition improved remark- in 137 patients and the daily dosage of corticoste- ably. He was discharged
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