Perinatal/Neonatal Case Presentation - Nature

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hemoglobin 9.6 g/dl, hematocrit 28.7%, platelets 346,000x109/l. ... Address correspondence and reprint requests to Daniel S. Knee, MD, Department of Newborn ... Rollins DM, Joseph SW, Actinomyces summary (University of Maryland web.
Perinatal/Neonatal Case Presentation Actinomyces Species and Cerclage Placement in Neonatal Sepsis: A Case Report Daniel S. Knee, MD Michael J. Christ, MD Delores M. Gries, MD Mark W. Thompson, MD

A 26-year-old female with a history of preterm labor and cerclage placement presented at 29 weeks gestation. Twin girls were delivered at 2917 weeks. Twin A presented with clinical sepsis at birth. Twin A’s blood cultures became positive for Actinomyces species on day of life 15. Despite aggressive medical management twin A died at 35 days of life. Journal of Perinatology (2004) 24, 389–391. doi:10.1038/sj.jp.7211097

INTRODUCTION Actinomyces species are normal flora of the upper respiratory, gastrointestinal and female genital tracts and are considered to have a low virulence potential. The association between Actinomyces and the use of intrauterine devices (IUDs) is well known.1 In this case report there is a possible relationship between cervical cerclage placement and invasive disease caused by Actinomyces species. CASE REPORT A 2917 week estimated gestational age (EGA) female infant was twin A of a dichorionic, diamnionic twin pregnancy was born to a 26year-old gravida 3 para 2 mother. Serology of maternal blood for syphilis, HIV and GBS was negative. The pregnancy was complicated by an emergent placement of a McDonald cerclage placed at 1947 weeks EGA due to funneling of the cervical canal. The pregnancy was further complicated by preterm labor (PTL) at 26 weeks gestation treated with steroids, tocolytics and ampicillin for

Department of Newborn Medicine, Tripler Army Medical Center, Tripler AMC, HI 96859-5000, USA. The views expressed in this manuscript are those of the authors and do not reflect the official policy or position of the Department of the Air Force, Department of the Army, Department of Defense, or the US Government. Address correspondence and reprint requests to Daniel S. Knee, MD, Department of Newborn Medicine, MCHK-CI, 1 Jarrett White Road, Tripler Army Medical Center, HI 96859,USA.

group B Streptococcus (GBS) prophylaxis for 7 days. At 28 weeks gestation, the mother was again admitted for PTL and treated with magnesium and terbutaline. On the day of delivery the mother was noted to have vaginal pressure, pain, bleeding and mucopurulent discharge. The cerclage was removed on the morning of delivery and there was spontaneous rupture of membranes shortly thereafter. The twins were born via elective Cesarean section due to breach presentation of twin B. At delivery, twin A had purulent amniotic fluid and a friable umbilical cord. Twin B had clear amniotic fluid. Twin A was intubated in the delivery room secondary to poor respiratory effort. The initial exam was remarkable for severe respiratory distress and generalized edema. Initial laboratory data included: white blood cells 23.1  109/l, hemoglobin 9.6 g/dl, hematocrit 28.7%, platelets 346,000  109/l. The differential included 19% segmental white blood cells, 38% bands, 15% lymphocytes, 16% monocytes, 1% eosinophil, 7% meta, 2% myelo, and 5 nucleated red blood cells per 100 white blood cells. The patient was mechanically ventilated for 3 days and was given one dose of Infasurf due to respiratory distress syndrome. The patient was started on ampicillin and cefotaxime at birth. Gastric, blood and tracheal sputum specimens were sent for culture. On DOL 3 the initial cultures were all reported as Gram positive organisms. A lumbar puncture was performed on DOL 4 which revealed no organisms on Gram stain and the final culture had no growth. On DOL 5 the patient deteriorated and was reintubated. On DOL 6 the culture report indicated an organism that most closely resembled a Corynebacterium species and was sent to a reference lab for identification (Focus Technologies, Cypress, CA). On DOL 15 the reference lab cultures were identified as Actinomyces species (unable to speciate). The Actinomyces was susceptible to Vancomycin and had a minimum inhibitory concentration (MIC) of r0.12 for ampicillin. Prior to and after identification of the Actinomyces species the patient received multiple courses of antibiotics (Figure 1). Despite the antibiotic coverage the patient continued to do poorly. The patient developed hypotension requiring cardiogenic pressors and stress dose hydrocortisone. The patient developed a coagulopathy, renal dysfunction, azotemia, metabolic acidosis, hypoproteinemia, hyponatremia, edema and hyperkalemia. The patient expired on DOL 35 after life support was withdrawn by the mother’s request. Autopsy demonstrated significant pulmonary congestion, intraalveolar hemorrhage, emphysematous change, pneumonitis,

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Figure 1. Antibiotic coverage for each day of life. Bold numbers represent a day in which the antibiotic coverage changed.

microthrombi, and focal thickened alveolar walls. The placenta had acute suppurative chorioamnionitis and acute funisitis. The placenta was green tinged and Gram stain revealed many Grampositive and-negative organisms, no significant hemorrhage, nodules, cysts or other gross lesions were noted. Specific cultures for Actinomyces were not sent.

DISCUSSION Actinomyces species are normal flora of the upper respiratory, gastrointestinal and female genital tracts and are considered to have a low virulence potential. With invasive disease Actinomyces species cause acute pyogenic infection or more commonly chronic infection that is both suppurative and granulomatous. These infections are characterized by multiple abscesses and sinus tracts. However, Actinomyces species are fastidious, and may take up to 2 weeks or more to culture.2 Infection caused by Actinomyces species is rare. The genital organs have the highest rate of infection caused by Actinomyces species, followed by skin-related infections, and then urinary tract infections.3 The association between Actinomyces and the use of intrauterine devices (IUDs) is well known.1 Actinomyces species can often be found in the cervical smears of asymptomatic patients with IUDs.4 The occurrence of colonization increases with the length of time that the IUD is in place.5 Sabbe et al.3 reported two cases of invasive pelvic Actinomyces in women who had IUDs in place and had Actinomyces on cervical smears 3 and 7 years prior to the invasive infection. No prior case reports of Actinomyces species associated with cerclage placement have been published. In this case, the only history of a uterine foreign body is the MacDonald cerclage placed in the mother at 19 weeks estimated gestational age. Since IUDs have been shown to be associated with invasive Actinomyces disease, we suspect that other intrauterine foreign bodies such as a cerclage may also be associated with invasive Actinomyces disease. The consequences of Actinomyces in preterm delivery, chorioamnionitis, and funisitis have been reported.6,7 This is the second identified case report of neonatal sepsis caused by 390

Actinomyces species.8 In both cases, the infant was premature (27 weeks EGA vs. 29 weeks EGA). In the previous case report, the mother had copious white vaginal discharge at 16 weeks but the amniotic fluid is not described at the time of rupture. In our case, there was mucopurulent vaginal discharge on the day of delivery and purulent amniotic fluid. In this case the infant was symptomatic with respiratory distress requiring mechanical ventilation, anemia, elevated band count, generalized edema and renal dysfunction. The infant in the original case report had only mild respiratory distress and an elevated band count. The original case report infant was treated with ampicillin, gentamicin initially and then switched to parenteral penicillin G for 2 weeks and oral penicillin G for the following 4 weeks,8 while our patient was treated with multiple antibiotics, see Figure 1. This case of Actinomyces was complicated by the prolonged time (15 days) between the onset of symptoms (birth) and identification of Actinomyces species. The original case report identified the Actinomyces neuii early in the hospital course.8 The mother presented with PTL at 26 weeks EGA and was treated with tocolytics, steroids and ampicillin for GBS prophylaxis. We postulate that ampicillin may have partially treated the chorionic/amniotic Actinomyces infection, only for the infection to be clinically symptomatic 2 weeks later. Abadi and Abadi,9 in their case report of a pregnancy complicated by Actinomyces, suggested that anaerobic cultures should be performed on amniotic fluid and placentas from women who are seen in preterm labor, particularly those who have a history of miscarriages, pelvic infections, IUD use, or vaginal discharge. Our case would tend to support this suggestion, particularly in those women who have an intrauterine (IUD) or cervical (cerclage) foreign body.

References 1. Arend SM, Oosterhof H, Van Dissel JT. Actinomyces and the intrauterine device. Arch Intern Med 1998;158:1270. 2. Rollins DM, Joseph SW, Actinomyces summary (University of Maryland web site) August 2000. Available at http://www.life.umd.edu/classroom/bsci424/ index.html. Accessed November 21, 2002. Journal of Perinatology 2004; 24:389–391

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3. Sabbe LF, Van De Merwe D, Schouls L, Bergmans A, Vaneechoutte M, Vandamme P. Clinical spectrum of infections due to the newly described Actinomyces species A. turicensis, A. radingae, and A. europaeus. J Clin Microbiol 1999;37(1):8–13. 4. Valicenti JF, Pappas AA, Graber CD, Williamson HO, Fowler Willis N. Detection and prevalence of IUD-associated Actinomyces colonization and related morbidity: a prospective study of 69,925 cervical smears. JAMA 1982;247:1149–52. 5. Evans DTP. Actinomyces israelii in the female genital tract: a review. Gentourin Med 1993;69:54–9.

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6. Wright Jr. JR, Stinson D, Wade A, Haldane D, Heifetz SA. Necrotizing funisitis associated with Actinomyces meyeri infection: a case report. Pediatr Pathol 1994;14(6):927–34. 7. Zakut H, Achiron R, Treschan O, Kutin E. Actinomyces invasion of placenta as a possible cause of preterm delivery. Clin Exp Obstet Gynecol 1987;14(2):89–91. 8. Mann C, Dertinger S, Hartmann G, Schurz R, Simma B. Actinomyces neuii and neonatal sepsis. Infection 2002;30(3):178–80. 9. Abadi MA, Abadi J. Actinomyces chorioamnionitis and preterm labor in a twin pregnancy: a case report. Am J Obstet Gynecol 1996;175(5):1391–92.

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