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      Myiasis-induced sepsis: a rare case report of Wohlfahrtiimonas chitiniclastica and Ignatzschineria indica bacteremia in the continental United States

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          Abstract

          Rationale:

          The presentation of sepsis and bacteremia in cutaneous and cavitary myiasis is uncommon. We present a patient, residing in a temperate region of the United States, with myiasis and sepsis from the emerging human pathogens Wohlfahrtiimonas chitiniclastica and Ignatzschineria indica.

          Patient concerns:

          A 37-year-old male patient with an 8-month history of chronic lymphedema and ulcers of the lower left extremity presented with myiasis of the left foot and leg. The patient was initially seen by his family practitioner many times and was prescribed antibiotics which he could not afford. Debridement of the myiasis was not conducted by the family practitioner due to the belief that the patient's current state of myiasis would effectively debride and eventually heal the chronic ulcers along with multiple antibiotic regimens. Over the 8-month period, the patient developed a progressive, painful, necrotizing infection of his lower left extremity.

          Diagnoses:

          Physical examination clearly showed myiasis of the patient's lower left extremity, believed to be caused by Lucilia sericata (green bottle fly). Blood cultures revealed the presence of Providencia stuartii, W chitiniclastica, and I indica to be the underlying cause of sepsis and bacteremia.

          Interventions:

          All visible maggots were extracted, debridement of devitalized tissue was performed, and the leg ulcers were wrapped in pH neutral bleach. The patient was initially treated with a broad-spectrum antibiotic regimen of vancomycin, clindamycin, piperacillin, and tazobactam which, following clinical improvement, was de-escalated to cefepime.

          Outcomes:

          The fly larvae and maggots were removed from the extremity by scrubbing, pulse lavage, and filing away the callused tissue. Additionally, the patient's sepsis and bacteremia, caused by W chitiniclastica and I indica, were successfully treated through antibiotic intervention. Amputation was avoided.

          Lessons:

          The use of pulse lavage and chlorhexidine-soaked brushes for the removal of cavitary myiasis is an effective and minimally invasive procedure which does not cause additional damage to surrounding tissue. W chitiniclastica and I indica are emerging bacteria that have known association to parasitic fly myiasis in humans and are capable of causing sepsis and/or bacteremia if not accurately identified and treated promptly.

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          Most cited references21

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          Wohlfahrtiimonas chitiniclastica gen. nov., sp. nov., a new gammaproteobacterium isolated from Wohlfahrtia magnifica (Diptera: Sarcophagidae).

          New Gammaproteobacteria were isolated from 3rd stage fly larvae of the parasitic fly Wohlfahrtia magnifica. Phylogenetic analysis of the new isolates showed that these bacteria belong to a distinct lineage close to Ignatzschineria larvae, which was originally isolated from the same species of fly. The low similarity values in 16S rRNA gene sequences (93.8-94.8 %), and differences in fatty acid profiles, RiboPrint patterns, MALDI-TOF mass spectra of cell extracts, and physiological and biochemical characteristics differentiate the isolates from the type strain of Ignatzschineria larvae (DSM 13226T), and indicate that our isolates represent a new genus within the Gammaproteobacteria. The major isoprenoid quinone of the strains is Q8, the major fatty acids are C18 : 1 and C14 : 0, and the predominant polar lipids are phosphatidylglycerol, phosphatidylethanolamine and phosphatidylserine. The G+C content of the DNA of the type strain is 44.3 mol%. The name Wohlfahrtiimonas chitiniclastica gen. nov., sp. nov., is proposed for this novel genus and species. The type strain is S5T (=DSM 18708T=CCM 7401T).
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            First case of fulminant sepsis due to Wohlfahrtiimonas chitiniclastica.

            We report the first case of fulminant sepsis due to Wohlfahrtiimonas chitiniclastica. This case is also the first one reported in South America. We emphasize the importance of recognizing bacteria that live in the larvae of a parasitic fly as the causative agent of severe infections in homeless patients.
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              Wohlfahrtiimonas chitiniclastica Bacteremia in Homeless Woman

              To the Editor: In May 2006, a 60-year-old homeless woman with a history of alcoholism was admitted to the emergency department of the Conception Hospital, Marseille, France. Firefighters had just found her in an abandoned container in the outskirts of the city, beside the body of her companion, who had died several days earlier. She described no symptoms other than fatigue. On examination, she was found to be dirty and covered with thousands of body and hair lice; dozens of insect larvae were in her hair. She was mildly febrile (38°C) and had widespread excoriations but no sign of localized bacterial infection. Head shaving exposed superficial ulcers on her scalp but no maggots. Blood analysis showed marked neutropenia (0.44 ×109/L), thrombocytopenia (28 × 109/L), a marked but well-tolerated iron deficiency anemia (hemoglobin 6.8g/dL), and a C-reactive protein level of 182 mg/L. Louse infestation was treated with a single dose of ivermectin (12 mg), and the woman was hospitalized. On day 3, she was still febrile. Louse-borne borreliosis had been ruled out by a negative blood smear, and results of serologic testing and molecular screening of lice for the other 2 louse-transmitted bacteria, Rickettsia prowazekii and Bartonella quintana ( 1 ), were negative. In contrast, 2 cultures of blood taken at the time of admission grew gram-negative rods susceptible to amoxicillin, ceftriaxone, imipenem, ciprofloxacin, amikacin, and trimethoprim/sulfamethoxazole. However, phenotypic tests failed to identify this bacterium with accuracy. Intravenous therapy with ceftriaxone at 2 g/d was initiated, and the patient’s fever, neutropenia, and thrombocytopenia improved. Scalp wounds healed with local care. Using 16S rRNA gene amplification and sequencing as previously described ( 2 ), we identified the bacilli as Wohlfahrtiimonas chitiniclastica and determined its similarity to be 99.5% with strain E43 (GenBank accession no. AJ517825). The 16S rRNA sequence obtained from the patient’s strain was deposited in GenBank under no. EU484335. The strain was deposited in the Collection de Souches de l’Unité des Rickettsies (CSUR; World Data Center for Microorganisms 875, http://ifr48.timone.univ-mrs.fr/portail2/index.php?option = com_content&task = view&id = 96&Itemid = 52) under reference CSUR P16. W. chitiniclastica is a recently described γ-proteobacterium isolated from larvae of the parasitic fly Wohlfahrtia magnifica ( 3 ). Although the pathogenicity of this new species for humans is as yet undescribed, it is phylogenetically close to Ignatzschineria larvae, another bacterium associated with W. magnifica larvae ( 4 ), which cause severe wound myiasis in cattle ( 5 ). Because of its strong chitinase activity, I. larvae may play a role in the metamorphosis of its host fly, as has been observed for other fly symbionts, and thus may be a symbiont of W. magnifica flies ( 6 ). The bacterium was later discovered in swine waste in Quebec ( 7 ). In 2007, three publications renewed researchers’ interest in I. larvae. First it was reclassified as the only species within the genus Ignatzschineria ( 4 ). Then 2 case reports demonstrated that it plays a role as a human pathogen ( 8 , 9 ). Both described an I. larvae bacteremia in adults with myiasis in southeastern France. The first patient was an elderly farmer with diabetes and myiasis of the leg, scrotum, and anus ( 8 ). The second patient was a middle-aged homeless man with a history of alcoholism who also had foot wound myiasis ( 9 ). We report W. chitiniclastica bacteremia also in a homeless woman from southeastern France. Although we did not test body lice for W. chitiniclastica, we believe that the bacteremia originated from the patient’s scalp maggots. Unfortunately, as previously reported for cases of I. larvae bacteremia, the maggots had been rapidly discarded, permitting neither bacterial analysis nor entomologic identification. However, these larvae may have been from W. magnifica flies. These flies are present in southern France, and although they are not typically found at low altitude and in a semiurban environment, their distribution is known to be progressively expanding, in part because of their broad adaptation capacities. Animal hosts for W. magnifica flies are numerous, but humans can also be infected; >10 cases of this myiasis in humans have been reported in Europe, Asia, Morocco, and Egypt. The scalp was affected in 2 of these patients ( 10 ). Among homeless persons, ectoparasitism is very common; body lice (Pediculus humanus humanus) are of particular interest because they transmit 3 bacterial bloodstream infections: trench fever (B. quintana), epidemic typhus (R. prowazekii), and louse-borne relapsing fever (Borrelia recurrentis) ( 1 ). Myiasis should also be considered as a relevant type of ectoparasitism in homeless and hygiene-deficient persons. In addition, like body lice, ticks, and fleas, fly larvae should also be regarded as another potential source of specific arthropod-borne bacterial systemic infections.
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                Author and article information

                Journal
                Medicine (Baltimore)
                Medicine (Baltimore)
                MEDI
                Medicine
                Wolters Kluwer Health
                0025-7974
                1536-5964
                December 2018
                28 December 2018
                : 97
                : 52
                : e13627
                Affiliations
                [a ]Trauma and Acute Care Surgery, OhioHealth Grant Medical Center, Columbus, Ohio
                [b ]General Surgery, OhioHealth Doctors Hospital, Lincoln Village, Columbus
                [c ]Trauma and Acute Care Surgery, Indiana University Health Methodist Hospital, Indianapolis, Indiana.
                Author notes
                []Correspondence: Meghan E. Wooster, General Surgery, OhioHealth Doctors Hospital, 5100 W. Broad St., Columbus, OH 43228 (e-mail: MeghanWoosterDO@ 123456gmail.com ).
                Article
                MD-D-18-05010 13627
                10.1097/MD.0000000000013627
                6314694
                30593131
                896471bb-97f3-4f0f-a199-fad7b74d5253
                Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc.

                This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0

                History
                : 7 August 2018
                : 19 November 2018
                Categories
                7100
                Research Article
                Clinical Case Report
                Custom metadata
                TRUE

                bacteremia,green bottle fly,ignatzschineria indica,lucilia sericata,sepsis,treatment of cavitary and cutaneous myiasis,wohlfahrtiimonas chitiniclastica

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