Abstract
Background:
Tsukamurella species were first isolated in 1941. Since then, 48 cases of Tsukamurella bacteremia have been reported, a majority of which were immunosuppressed patients with central venous catheters.A case is described and previous cases of Tsukamurella bacteremia are reviewed.
Patients and Methods:
A 70-year-old total parenteral nutrition (TPN)-dependent female with recurrent enterocutaneous fistula (ECF), developed leukocytosis one week after a challenging ECF takedown. After starting broad-spectrum antibiotic agents, undergoing percutaneous drainage of intra-abdominal abscess, and subsequent repositioning of the drain, her leukocytosis resolved. Blood and peripherally inserted central catheter (PICC) cultures grew Tsukamurella spp. The patient was discharged to home with 14 days of daily 2 g ceftriaxone, with resolution of bacteremia.
Conclusions:
Tsukamurella spp. are a rare opportunistic pathogen predominantly affecting immunocompromised patients, with central venous catheters present in most cases. However, there have been few reported cases in immunocompetent individuals with predisposing conditions such as end-stage renal disease and uncontrolled diabetes mellitus.
T
In this report, we present a case of Tsukamurella bacteremia in a surgical patient with diabetes mellitus who received total parenteral nutrition (TPN) via a peripherally inserted central catheter (PICC).
Case Report
A 70-year-old female underwent distal pancreatectomy for cystadenocarcinoma in 2003. Her course was complicated by adhesive disease and chronic intestinal obstructive symptoms requiring multiple small and large bowel resections. Her course was further complicated by small bowel perforation after an enteroscopy in 2023. After undergoing two exploratory laparotomies that year, she developed enterocutaneous fistula (ECF) that recurred despite bowel rest with TPN and wound care. This was a high-volume ECF that prohibited oral intake. Her other comorbidities include uncontrolled type 2 diabetes mellitus (HA1c, 8.1), deep vein thrombosis on therapeutic enoxaparin, hypothyroidism, and hyperlipidemia. The patient desired ECF takedown and had been medically optimized, so she was taken to the operating room for ECF takedown.
Intra-operative findings were notable for a frozen abdomen with ECF originating from a prior small bowel–small bowel anastomosis staple line that tracked to a piece of mesh at the midline incision, as well as dense interloop small bowel adhesions, requiring several hours of adhesiolysis. Several enterostomies, including a colotomy in the rectum, were inadvertently made while we were freeing up the visceral block containing the ECF. A stricture at the ileocolonic anastomosis distal to the ECF was found that likely contributed to her fistulization process. To prevent further injuries to the bowels, the decision was to resect small bowels en bloc with the fistula and divert her with an end ileostomy. We measured approximately 65 cm small bowels remaining from the ligament of Treitz. To address the rectal injury, we divided approximately 7 cm of rectum with the colotomy. We were able to close the fascia without tension after raising bilateral myocutaneous flaps. The fascial closure was reinforced with an on-lay synthetic absorbable mesh.
In the post-operative period, the patient was kept nil per os until she demonstrated return of bowel function on post-operative day (POD) 4. She remained in the hospital due to poor appetite and rising leukocytosis. On POD 7, she developed fever with an elevated lactate. A computed tomography (CT) scan of the abdomen and pelvis with intravenous and oral contrast demonstrated new fluid and gas collection along the anterior abdomen with rim enhancement. Re-assuringly there was no definite evidence of enteric contrast extravasation. The patient was started on 3.375 g piperacillin-tazobactam every eight hours and a percutaneous abscess drain was placed.
Surprisingly, cultures from the intra-abdominal fluid collection did not yield any organisms, despite the growth of Tsukamurella spp. in her blood cultures. Because of persistent leukocytosis, the abscess drain was repositioned on POD 10. Her white blood cell (WBC) count finally normalized by POD 12. Concurrently, blood cultures collected from her home PICC became positive for Tsukamurella spp., suggesting a catheter-related blood stream infection (CLABSI).
Because she remained afebrile with normal WBC count and negative repeat blood cultures while on intravenous antibiotic agents, the PICC was left in place and the patient was discharged home on POD 14 with a regular diet, TPN, and total 14 days of intravenous antibiotic agents from the last negative blood cultures. Based on the organism's susceptibility (Table 1), she completed her antibiotic course with daily 2 g ceftriaxone. At her two-week post-operative clinic visit, she was doing well with improved appetite. In the coming month she subsequently stopped TPN and met her oral intake requirements by mouth. The abscess drain was interrogated and removed one month after.
Minimum Inhibitory Concentrations of the Tsukamurella spp. in the Present Case
MIC = minimum inhibitory concentrations.
Literature Review
A comprehensive literature review was conducted, encompassing all relevant case reports published on PubMed prior to December 2023. In total, 24 articles were identified, yielding a total of 48 cases of Tsukamurella spp. bacteremia, as summarized in Table 2. Among these patients, 40 were immunocompromised. Specifically, 20 (50%) had solid organ malignancy requiring chemotherapy, 17 (43%) had hematologic malignancy requiring bone marrow transplantation or chemotherapy, two (5%) had hematologic disorder and received bone marrow transplantation, and one (3%) had solid-organ transplantation. Nearly all patients, with the exception of one, had a central venous catheter. Conversely, only eight patients were immunocompetent. Among these eight patients, four were undergoing hemodialysis, and all but one had a central venous catheter. Notably, the sole patient without a central venous catheter had pre-existing uncontrolled type 2 diabetes mellitus (HA1c > 8%). In cases of CLABSI, treatment involved the removal of the central venous catheter and administration of a course of antibiotic agents.
Clinical Features of Forty-Nine Patients With Bacteremia Caused by Tsukamurella spp. Reported in the Literature
IgA = immunoglobulin A.
Conclusions
Tsukamurella spp. is an uncommon opportunistic pathogen documented in 40 immunocompromised patients, with central venous catheters present in all but one case. Conversely, only eight cases have been reported in immunocompetent individuals with predisposing factors such as end-stage renal disease and uncontrolled diabetes mellitus. In this report, we present the case of an immunocompetent surgical patient who had uncontrolled diabetes mellitus and was on long-term TPN via a PICC. Her bacteremia was treated successfully with targeted antibiotic therapy without removing the PICC.
Footnotes
Acknowledgments
We wish to express our gratitude to the patient.
Authors' Contributions
Conceptualization: Forrester. Methodology: Forrester. Writing—original draft: Wong. Writing—review and editing: Forrester, Wong. Supervision: Forrester. Data curation: Wong.
Funding Information
No funding was received for conducting this study.
Author Disclosure Statement
We have no conflicts of interest to declare.
