Tropical Medicine and Infectious Disease Case Report Percutaneous Emergence of Gnathostoma spinigerum Following Praziquantel Treatment Sarah G. H. Sapp 1,*, Monica Kaminski 2, Marie Abdallah 2 , Henry S. Bishop 1, Mark Fox 1,3, MacKevin Ndubuisi 1 and Richard S. Bradbury 1 1 Parasitic Diseases Branch, Division of Parasitic Diseases and Malaria, Center for Global Health, Centers for Disease Control and Prevention, Atlanta, GA 30029, USA; [email protected] (H.S.B.); [email protected] (M.F.); [email protected] (M.N.); [email protected] (R.S.B.) 2 New York City Health and Hospitals Corporation, New York, NY 10013, USA; [email protected] (M.K.); [email protected] (M.A.) 3 Oak Ridge Institute for Science and Education, Oak Ridge Associated Universities, Oak Ridge, TN 37830, USA * Correspondence: [email protected] Received: 4 November 2019; Accepted: 11 December 2019; Published: 14 December 2019 Abstract: A Bangladeshi patient with prior travel to Saudi Arabia was hospitalized in the United States for a presumptive liver abscess. Praziquantel was administered following a positive Schistosoma antibody test. Ten days later, a subadult worm migrated to the skin surface and was identified morphologically as Gnathostoma spinigerum. This case highlights the challenges of gnathostomiasis diagnosis, raising questions on potential serologic cross-reactivity and the possible role of praziquantel in stimulating outward migration of Gnathostoma larvae/subadults. Keywords: gnathostomiasis; schistosomiasis; imported helminthiasis; praziquantel 1. Introduction Gnathostomiasis is a foodborne zoonosis with diverse and sometimes serious clinical outcomes. Transmission occurs via consumption of advanced third-stage (AL3) larvae encysted in undercooked meat of intermediate or paratenic hosts, commonly freshwater fish, frogs, snakes, and fowl. Larvae undergo an invasive course of migration in the aberrant human host after penetrating the gastrointestinal wall, first migrating to the liver parenchyma and then other organs [1]. The usual presentation is cutaneous gnathostomiasis, frequently preceded by a nonspecific prodrome due to larval migration. Other presentations include urogenital, visceral, ocular, and neurological gnathostomiasis [1,2]. Management can be challenging with frequent relapses or treatment failures [3]. The known zoonotic species are Gnathostoma spinigerum, G. hispidum, G. doloresi, G. nipponicum, G. binucleatum, and possibly G. malaysiae [1,2]. Among these, G. spinigerum is the best-studied with the broadest occurrence; this species is endemic across Southeast Asia, East Asia, India, Africa, and possibly Australia [1]. Gnathostomiasis has been recognized as a parasitic infection of travelers and refugees from endemic regions [2,4], however, awareness among general practitioners in nonendemic countries may be limited. 2. Case Report In June 2018, a 46-year-old female originally from Bangladesh (emigrated to the USA in 2017) presented to her primary care physician with complaints of diarrhea. The patient had traveled to Saudi Arabia for ten days and developed symptoms one week after returning to the USA. She was prescribed a 7-day course of metronidazole, though she only complied for 2 days due to religious fasting. Two Trop. Med. Infect. Dis. 2019, 4, 145; doi:10.3390/tropicalmed4040145 www.mdpi.com/journal/tropicalmed Trop. Med. Infect. Dis. 2019, 4, 145 2 of 6 Trop. Med. Infect. Dis. 2019, 4, x FOR PEER REVIEW 2 of 6 weeks following symptom onset, the patient was admitted to the hospital with diffuse intermittent abdominalfasting. Two pain weeks that beganfollowing two symptom days earlier onset, and thecontinuing patient was diarrhea.admitted to She the reported hospital epigastricwith diffuse and rightintermittent upper abdominal abdominal quadrant pain that (RUQ) began pain two and days increased earlier belching.and continuing No other diarrhea. signs/ symptomsShe reported were reportedepigastric or observed.and right upper abdominal quadrant (RUQ) pain and increased belching. No other signs/symptomsUpon admission were she reported was afebrile or observed. and had stable vital signs. Physical examination revealed mild discomfortUpon to admission deep palpation she was of afebrile the epigastrium and had stable and vital RUQ. signs. Her Physical white bloodexamination cell (WBC) revealed count mild was elevateddiscomfort at 20.64 to deep10 palpation9/L (normal of the (4.70–10.30) epigastrium 10and9/ L)RUQ. with Her 57.8% white eosinophils, blood cell (WBC) 18.8% count neutrophils, was × × 19.7%elevated lymphocytes, at 20.64 × and109/L 0.5% (normal basophils. (4.70–10.30 A CT) × scan109/L) of with the 57.8% abdomen eosinophils, showed 18.8% diffuse neutrophils, gastric wall thickening19.7% lymphocytes, with mild adjacent and 0.5% inflammatory basophils. A change,CT scan suggestive of the abdomen of gastritis, showed and diffuse a hypodensity gastric wall in the leftthickening lower liver with measuring mild adjacent 2.2 cm inflammatory with a rim enhancing change, suggestive wall, suspicious of gastritis, for abscess. and a hypodensity Metronidazole in the and ceftriaxoneleft lower treatment liver measuring was initiated. 2.2 cm with Interventional a rim enhancing radiology wall, was suspicious consulted for forabscess. liver abscessMetronidazole drainage, butand the ceftriaxone procedure wastreatment deferred was in initiated. view of theInterventional small size. radiology The abdominal was consulted pain resolved for liver on theabscess second daydrainage, of hospitalization, but the procedure but the was patient deferred remained in view admitted of the small for continuation size. The abd ofominal IV antibiotic pain resolved treatment. on theOn second the thirdday of day hospitalization, of hospitalization, but the patient serum remained and stool admitted specimens for continuation were collected of IV and antibiotic sent for treatment. testing for various parasitic etiologies, including schistosomiasis, to determine the cause of peripheral On the third day of hospitalization, serum and stool specimens were collected and sent for eosinophilia and liver abscess. Antibody tests for Entamoeba histolytica, Strongyloides, and Toxocara testing for various parasitic etiologies, including schistosomiasis, to determine the cause of peripheral and a pan-filarial assay were negative. Three of four stool examinations were negative (one positive eosinophilia and liver abscess. Antibody tests for Entamoeba histolytica, Strongyloides, and Toxocara and for Blastocystis). The patient was discharged on day 8 following placement of a peripherally-inserted a pan-filarial assay were negative. Three of four stool examinations were negative (one positive for central catheter for the continuation of intravenous ceftriaxone (6-week course) and oral metronidazole Blastocystis). The patient was discharged on day 8 following placement of a peripherally-inserted forcentral liver abscess catheter treatment. for the continuation of intravenous ceftriaxone (6-week course) and oral metronidazoleFive weeks for following liver abscess discharge, treatment. a RUQ sonogram showed abscess resolution, however, blood work revealed persistent WBC elevation (17.66 109/L) with 46.1% eosinophils, 29.0% Five weeks following discharge, a RUQ sonogram showed× abscess resolution, however, blood neutrophils,work revealed 20.6% persistent lymphocytes, WBC elevation 2.9% monocytes,(17.66 × 109/L) and with 0.5% 46.1% basophils. eosinophils, She 29.0% was neutrophils, advised to continue20.6% ceftriaxonelymphocytes,/metronidazole 2.9% monocytes, for another and 0.5% week. basophils. A few days She later, was a lowadvised positive to continue result was returnedceftriaxone/metronidazole for the previously-ordered for anotherSchistosoma week. A fewantibody days later, test (FAST-ELISA a low positive value result 13.0 was (0–10returned normal)); for ceftriaxonethe previously/metronidazole-ordered wasSchistosoma ceased andantibody praziquantel test (FAST (40 mg-ELISA/kg in value 2 doses 13.0 taken (0– in10 one normal)); day) was prescribed.ceftriaxone/metronidazole Ten days after praziquantel was ceased and treatment, praziquantel the patient (40 mg/kg reported in 2 doses epigastric taken pain in one with day localized) was rash,prescribed. pruritus, Ten and days hyperesthesia. after praziquantel Clinical treatment, examination the patient identified reported a serpiginous epigastric track pain with with an localized emerging wormrash, over pruritus, the upper and abdomenhyperesthesia. (Figure Clinical1) which examination was extracted. identified Photos a of serpiginous the parasite track were submittedwith an foremerging telediagnosis worm toover the the Centers upper abdomen for Disease (Figure Control 1) which and was Prevention extracted. (Atlanta, Photos of Georgia, the parasite USA). were The specimensubmitted was for stored telediagnosis in 70% ethanol to the Centers and shipped for Disease for morphologic Control and examination. Prevention (Atlanta, Georgia, USA). The specimen was stored in 70% ethanol and shipped for morphologic examination. Figure 1. Gnathostoma spinigerum Figure 1.Serpiginous Serpiginous track trackshowing showing thethe emergingemerging subcutaneoussubcutaneous Gnathostoma spinigerum. .(Bar (Bar = = approximatelyapproximately 1 cm).1 cm). Trop. Med. Infect. Dis. 2019, 4, x FOR PEER REVIEW 3 of 6 By microscopy, the gross appearance and the presence of a cephalic bulb and broad caudal alae (Figure 2) were characteristic of a subadult male Gnathostoma.