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Ignatzschineria spp. bacteremia from maggot infestation

Ignatzschineria spp. bacteremia associated with maggot infestation is extremely rare in humans. There are only a few cases worldwide ever reported in the literature. We described a clinical case with a male patient who presented with maggot manifestation at his lower extremity, was found with bacteremia, and subsequently identified as Ignatzschineria spp by 16S rRNA sequencing.


Introduction
Ignatzschineria is a genus of aerobic, gram-negative, non-sporeforming, non-hemolytic rod-shaped bacteria that belongs to the class Gammaproteobacteria [1]. Three recognized species: I. indica, I. larvae, and I. ureiclastica which are commonly recovered from the larvae of parasitic spotted flesh fly Wohlfahrtia magnifica found in Europe, Asia, and North Africa [2]. The Ignatzschineria spp. thrive in the digestive tract of the larvae with Providencia [2]. It is important to note that medical-grade maggots commonly used to promote debridement and wound healing are free of pathogenic bacteria as they are grown in sterile, cultured environments [3]. On the other hand, myiasis, the larval infestation of vertebrate animals and humans, occurs naturally in the environment and may cause uncontrolled infestation as well as bacterial infection [4].

Case study
A 63-year-old man with a history of hypertension and hyperlipidemia presented by EMS with a one-week history of malaise and left foot pain. The patient had his socks on for a week which he had trouble getting off as they seemed stuck to his skin. In the presentation, vital signs were significant with a temperature 101.8 F, blood pressure 87/76, heart rate 113 beats/min, respiratory rate 21 breaths/min, and oxygen saturation >95 % on room air. Physical examination was unremarkable, except a 7 cm open wound on the anterior left ankle, deep down to the muscle layer, covered with numerous maggots (Fig. 1). The patient underwent irrigation and debridement with the wound vac placement. Blood culture on admission grew group C/G beta Streptococcus, Providencia stuartii, and finally, Ignatzschineria species were identified by 16S rRNA sequencing, PCR, and MALDI-TOF tests. Magnetic resonance imaging with the contrast of the left ankle revealed a large anterior deep ankle ulceration that transects the tibialis anterior tendon, with no evidence of osteomyelitis (Fig. 2). The patient was initially started on vancomycin and cefepime, later changed to piperacillin/tazobactam for a total of 4 weeks of the regimen. Upon follow-up visit, the wound completely healed up (Fig. 3).

Discussion
A common cause of myiasis in animals, Ignatzschineria spp. rarely can cause human diseases, like myiasis and septicemia [2]. Wounds infected with maggots in the setting of poor hygiene were strongly associated with invasive Ignatzschineria spp. diseasewith Ignatzschineria indica being the most common species identified. 16S rRNA amplification and sequencing, and mass spectrometry Matrix-Assisted Laser Desorption/Ionization-Time of Flight MALDI-TOF have been used in the identification of the bacteria, which are not typically included in standard microbiological identification systems [1]. However, some cases were described where MALDI-TOF was unsuccessful to identify the Ignatzschineria spp. [  -all had maggot-infested wounds in common [1,3,4,7]. Three cases of Ignatzschineria indica bacteremia in France were described in the setting of wounds infected with a large number of maggots [2]. A case of Ignatzschineria indica bacteremia was reported in the Netherlands in a patient with chronic alcoholism and maggotinfested wounds of the right foot [6]Á Similarly, a case of Ignatzschineria indica septicemia associated with myiasis was described in Argentina and Spain, both patients were impoverished and chronic alcoholics, the former case led to the amputation of the patient's left lower extremity [5,8]. A case of Ignatzschineria indica urinary tract infection was described in the United States in a paraplegic with chronic decubitus ulcers and urethrocutaneous fistulas with no observed maggot infestation. It is recommended that in the absence of maggots, culture isolation of Ignatzschineria indica or any organism commonly found in flies prompt investigation for occult myiasis [4]. Most of the cases were susceptible to   beta-lactam antimicrobials, aminoglycosides and fluoroquinolones, however, there is currently no standardized antimicrobial susceptibility testing due to the rarity of this infection [4,8].
Subsequently, most of the cases were treated with beta-lactam antimicrobials based on sensitivity patterns, bacterial co-infections, and clinical response. (Table 1) There is limited literature regarding the association of maggot species to bacteria causing invasive infection. Some studies raised the concern regarding the potential migration of Wohlfahrtia magnifica parasitic flies or if Ignatzschineria indica can potentially colonize other parasitic flies that cause myiasis [5,7]. A case of myiasisinduced sepsis from maggot infestation was described in the United States where Ignatzschineria indica was isolated from blood culture and was believed to be caused by Lucilia sericata (green bottle fly) based on the appearance of the larvae [3]. Similarly, one of the Ignatzschineria indica bacteremia cases reported in the United States sent larvae for entomologic identification and was identified as the blowfly, Phaenicia sericata [1].

Conclusion
Maggot infestation is uncommon in the United States; therefore, diagnosis and management can be challenging. Cultures should be obtained for prompt Ignatzschineria spp. identification by 16S rRNA amplification and sequencing in patients at high risk for getting the infection, particularly if maggots present. Infection with Ignatzschineria is also considered with high-risk patients without obvious maggot infestation and should prompt investigation for occult myiasis.

Funding
No sources of funding for this manuscript.

Consent
Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request