Case Report

A Fatal Case of Strongyloidiasis in Pregnancy Arin M. Buresch, MD, Nancy E. Judge, MD, Ashlesha K. Dayal, MD, and David J. Garry, DO BACKGROUND: Strongyloides stercoralis is a common human parasite worldwide and has been associated with severe infection in immunosuppressed patients. High mortality rates have accompanied this severe disseminated infection. There is a scarcity of literature surrounding severe Strongyloides infection in pregnancy. CASE: A 30-year-old primigravid Haitian woman at 25 weeks of gestation presented with acute abdominal pain and an abnormal fetal heart tracing. Mild anemia and eosinophilia were laboratory abnormalities on admission. She received corticosteroids for the fetus and subsequently developed septic shock. Sputum and stool were positive for S stercoralis larvae. Hyperinfection was diagnosed, stillbirth occurred, and the patient died. CONCLUSION: A more global awareness and education surrounding helminth infection during pregnancy may improve response, reduce delay in diagnosis, and potentially improve outcome. (Obstet Gynecol 2015;126:87–9) DOI: 10.1097/AOG.0000000000000676

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trongyloides stercoralis is the most common human parasitic nematode that can proliferate and complete a full life cycle within its host.1 There is a paucity of literature regarding the maternal and fetal effects of Strongyloides infection during pregnancy, especially when care is rendered in developed countries. Presenting symptoms can be extremely vague, and severe infection, especially in the immunosuppressed

From the Montefiore Medical Center, Einstein College of Medicine, Department of Obstetrics & Gynecology and Women’s Health, Division of Maternal-Fetal Medicine, Bronx, New York. Corresponding author: David J. Garry, DO, Montefiore Medical Center, Department of Obstetrics & Gynecology and Women’s Health, 600 East 233rd Street, Bronx, NY 10466; e-mail: [email protected]. Financial Disclosure The authors did not report any potential conflicts of interest. © 2015 by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. All rights reserved. ISSN: 0029-7844/15

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Teaching Points 1. When caring for women who have arrived in the United States from low-resource countries, possible regional illnesses should be considered. 2. Eosinophilia and anemia and gastrointestinal and pulmonary complaints can be associated with Strongyloides infection, but asymptomatic infections are common. Patients can remain infected even years after leaving an endemic area. 3. Ivermectin is recommended for treatment of Strongyloides infection, with albendazole as an alternative.

individual, can lead to maternal death.2 We present a maternal mortality case associated with disseminated S stercoralis infection.

CASE A 30-year-old primigravid Haitian woman presented at 25 weeks of gestation with acute abdominal and epigastric pain. On arrival, the fetal heart rate tracing had a prolonged deceleration that recovered and was followed by minimal variability with occasional variable decelerations and irregular uterine contractions. Initially, in triage, there was preparation for emergent cesarean delivery owing to the fetal heart tracing; however, it improved. The patient then was admitted for continuous fetal monitoring and further evaluation of her abdominal pain. Her pregnancy was complicated by an outpatient Escherichia coli urinary tract infection that had an unrecognized resistance to the antibiotic used in treatment and poor maternal weight gain, with a recent 3.3-lb (1.5-kg) weight loss. Her body mass index (calculated as weight (kg)/[height (m)]2) was 20.1. Her past gynecologic history was significant for a 6-cm anterior fundal leiomyoma recently treated with indomethacin for pain attributed to degeneration. She had no travel outside the United States for more than 1 year. Her prenatal laboratory values, obtained from her obstetrician on hospital day 5, were remarkable for normocytic, normochromic anemia with hemoglobin of 9.7 g/dL, human immunodeficiency virus (HIV)–negative, and eosinophilia 790 cells/microliter (10%). The initial working diagnosis was placental abruption based on the clinical scenario of abdominal pain, contractions, and an abnormal fetal heart tracing. Bedside ultrasound examination was nondiagnostic, and the fetal heart tracing improved; however, continued abdominal pain and contractions prompted preparation for a preterm birth. A course of betamethasone 12 mg (two doses 24 hours apart)

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for improved fetal outcome was initiated, along with magnesium sulfate for fetal neuroprotection. Cefazolin was started for treatment of the patient’s urinary tract infection. Her abdominal pain improved with acetaminophen and morphine. Hospital admission laboratory results demonstrated a hemoglobin of 9.7 g/dL, white blood cell count 9.9 K/microliter, eosinophil count 1,100 cells/microliter (11%), and platelet count 829 K/microliter. The thrombocytosis was attributed to an acute phase reactant. Maternal pain symptoms improved, the fetal heart tracing normalized, and continuous fetal monitoring was discontinued. On hospital day 4, the patient reported chest pain and shortness of breath. There was an episode of copious bilious vomiting followed by oxygen desaturation requiring supplemental oxygen with a nonrebreather mask. An electrocardiogram showed nonspecific T-wave changes, troponin results were negative, and a maternal echocardiogram was normal. Chest X-ray and chest computed tomography angiography demonstrated multilobar pneumonia. The patient’s antibiotics were changed to ceftriaxone and azithromycin. On hospital day 5, the patient developed septic shock and was admitted to the intensive care unit. Differential diagnosis at the time included aspiration pneumonia or urosepsis. A repeat urine culture, which was obtained on admission, returned positive for E coli, and an infectious disease consultant changed antibiotics from ceftriaxone to cefepime. The patient developed a fever (39.4˚C), and her respiratory status worsened, requiring intubation. Once intubated, sputum for culture, gram stain, ova, and parasite evaluation was performed. Respiratory Gram stain followed by culture results were positive for E coli, and sputum demonstrated S stercoralis rhabditiform larvae. Stool evaluation also demonstrated Strongyloides larvae. After larvae were seen, human T-lymphotropic virus type 1 (HTLV-1) testing was performed and returned a positive result. The antiparasitic ivermectin was initiated, with vancomycin and metronidazole added to cefepime for antibiotic coverage. On hospital day 7, while the patient remained intubated in the intensive care unit on pressor agents, fetal demise was diagnosed and followed by spontaneous onset of labor and subsequent vaginal delivery of a stillborn male fetus weighing 705 g; the fetus appeared morphologically normal for gestational age. The following day, albendazole and oseltamivir were initiated and metronidazole and azithromycin discontinued. The patient received vasopressor support but then suffered cardiopulmonary arrest and died. After autopsy, the cause of death was reported as S stercoralis superinfection, septic shock due to S stercoralis and E coli, systemic inflammatory response syndrome, and diffuse bronchopneumonia.

DISCUSSION S stercoralis is a round worm that is estimated to infect 50 to 100 million people worldwide, with a high prevalence in tropical regions of Africa, Asia, and South America.1 There are two life cycle stages: the

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rhabditiform stage and the filariform stage. The filariform larvae penetrate human host skin and initiate the parasitic cycle.3 Rhabditiform larvae are produced from eggs that are laid by female adult worms living within the host’s small intestine. They are either passed in the stool to become free-living or cause an autoinfection within the host (parasitic cycle).3 Symptoms of Strongyloides infection are vague, including intestinal bloating and abdominal pain. This parasitic infection, which does not cross the placenta, has been reported to have no direct effect on maternal or fetal outcomes.4 Our patient was immunosuppressed owing to both her pregnancy and infection with HTLV-1. In this setting, the corticosteroid therapy likely triggered disseminated infection, leading to her death. The majority of Strongyloides infections are asymptomatic autoinfections, but there is potential for severe hyperinfection leading to disseminated infection, especially in immunosuppressed hosts.3 During disseminated infection, larvae can be found anywhere but most commonly are present in sputum and skin. The larvae penetrate the gut wall, leading to a secondary gramnegative sepsis, which adds to the potential for mortality.5 Severe disseminated infection has a mortality rate of up to 87% in the nonpregnant state.5 Risk factors for severe hyperinfection include immunosuppression, corticosteroid therapy, HTLV-1 positivity, collagen vascular disease, neoplasms, malnutrition, severe diabetes mellitus, end-stage renal disease, advanced age, HIV-1 infection, travel to endemic areas, incarceration, and gastrointestinal anatomic abnormalities (diverticulosis).6 Our patient received corticosteroids to improve outcome for a potential delivery of the very premature fetus. Corticosteroid therapy in Strongyloides infection has been problematic and associated with life-threatening hyperinfection irrespective of dosage and route of delivery.6,7 Our patient received betamethasone in preparation for preterm birth. Treatment is complicated by the need for complete parasitic eradication; however, this is difficult to ascertain in many cases given low worm loads.3 Ivermectin is recommended, and albendazole is an alternative treatment. Owing to the risk of gram-negative septicemia, administration of broad spectrum antibiotics is advisable in cases of hyperinfection and disseminated infection.3 In the case of Strongyloides infection, autoinfection may explain the possibility of persistent subclinical infection for many years in persons who have not recently traveled to endemic areas. In these individuals, hyperinfection can occur when they are immunocompromised.5 The World

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Health Organization advises preventive strategies, including the provision of sanitation and hygiene in endemic areas.1,4 There is limited literature related to Strongyloides infection during pregnancy. Heaton et al8 report a case of successful treatment for Strongyloides in early pregnancy. Villar et al9 found an increased incidence of fetal growth restriction among women infected with a number of different parasites and helminths. The incidence of fetal growth restriction increased with the number of parasitic species detected and with increasing levels of infection. High levels of infection with S stercoralis, however, were not associated with an increased incidence of fetal growth restriction, differing from the associations noted with protozoas and helminthic Trichuris trichiura.9 Ndibazza et al10 studied low-intensity helminthic infections in a highprevalence region during pregnancy. They found no overall effect of antihelminthic treatment during pregnancy on maternal anemia, birth weight, perinatal mortality, or congenital anomalies, although there was a possible benefit to albendazole treatment in prevention of severe anemia for women with hookworm infection.10 Women in low-resource countries have limited antenatal care and exposure to steroids in preterm labor management. Helminthic infections affect many pregnant and nonpregnant women worldwide each year. The consequences of these infections in pregnant women can vary from mild anemia to fetal death, maternal death, or both. Obstetrician–gynecologists need to have a more global approach to the diagnosis and management of parasitic infections during pregnancy. Special attention to those women who travel from endemic areas to developed countries is becoming essential for a variety of infections. Strongyloidiasis should be considered when a patient presents from an endemic area with nonspecific pulmonary complaints and eosinophilia (more than 5% relative or more than 400 eosinophils/microliter).2 Roman-Sanchez et al report that patients with a finding of eosinophilia (more than 400 cells/microliter) had 73-fold higher odds of having strongyloidiasis and that presence of eosinophilia was the only reliable predictor of infection.11 Our patient had evidence of eosinophilia but lacked recent travel history and pulmonary and gastrointestinal

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complaints on initial presentation. Steroid treatment for fetal benefit was completed before the etiology of the patient’s eosinophilia was established. Delays in making a diagnosis, errors in empiric treatment, and facilitating the development of hyperinfection by initiating steroids occurs more often in treatment of suspected asthma, when health care providers are not familiar with immigrant or travel medicine.2 This patient’s course serves as a cautionary reminder for obstetric providers caring for global citizens: awareness that disseminated Strongyloides infection can be associated with antepartum steroids given for fetal indications, with serious consequences. REFERENCES 1. Carvalho E, Da Fonseca Porto A. Epidemiological and clinical interaction between HTLV-1 and Strongyloides stercoralis. Parasite Immunol 2004;26:487–97. 2. Boulware DR, Stauffer WM, Hendel-Paterson BR, Rocha JL, Seet RC, Summer AP, et al. Maltreatment of Strongyloides infection: case series and worldwide physicians-in-training survey. Am J Med 2007;120:545–51. 3. Grove DI. Strongyloidiasis: a conundrum for gastroenterologists. Gut 1994;35:437–40. 4. Dotters-Katz S, Jeffrey K, Philips H. Parasitic infections in pregnancy. Obstet Gynecol Surv 2011;66:189–92. 5. Marcos LA, Terashima A, Dupont HL, Gotuzzo E. Strongyloides hyperinfection syndrome: an emerging global infectious disease. Trans R Soc Trop Med Hyg 2008;102:314–8. 6. Keiser PB, Nutman TB. Strongyloides stercoralis in the immunocompromised population. Clin Microbiol Rev 2004; 17:208–17. 7. Wurtz R, Mirot M, Fronda G, Peters C, Kocka F. Short report: gastric infection by Strongyloides stercoralis. Am J Trop Med Hyg 1994;51:339–40. 8. Heaton J, Shippey S, Macri C, Macedonia C. Intestinal helminthes infestation in pregnancy: a case report and literature review. Mil Med 2002;167:954–5. 9. Villar J, Klebanoff M, Kestler E. The effect on fetal growth of Protozoan and helminthic infection during pregnancy. Obstet Gynecol 1989;74:915–20. 10. Ndibazza J, Muhangi L, Akishule M, Kiggundu M, Ameke C, Oweka J, et al. Effects of deworming during pregnancy on maternal and perinatal outcomes in Entebbe, Uganda: a randomized controlled trial. Clin Infect Dis 2010;50:531–40. 11. Román-Sánchez P, Pastor-Guzmán A, Moreno-Guillén S, IgualAdell R, Suñer-Generoso S, Tornero-Estébanez C. High prevalence of Strongyloides stercoralis among farm workers on the Spanish Mediterranean coast. Analysis of the predictive factors of infection in developed countries. Am J Trop Med Hyg 2003; 69:336–40.

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Copyright ª by The American College of Obstetricians and Gynecologists. Published by Wolters Kluwer Health, Inc. Unauthorized reproduction of this article is prohibited.

A Fatal Case of Strongyloidiasis in Pregnancy.

Strongyloides stercoralis is a common human parasite worldwide and has been associated with severe infection in immunosuppressed patients. High mortal...
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