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Volume 20, Number 6—June 2014
Letter

Streptococcus suis Infection and Malignancy in Man, Spain

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To the Editor: Streptococcus suis is an emerging zoonotic agent. Human infection is associated with occupational exposure to swine. Affected persons are usually, but not always, healthy (1,2). Immunosuppressive conditions can predispose persons to S. suis infection, and cancer has classically been associated as a risk factor for S. suis infection (1,2). Nevertheless, the actual number of reported cases is low (27). We describe a severe case of S. suis infection in a man who had not been exposed to swine but for whom disseminated cancer was diagnosed 5 months after the infection.

In 2012, a 57-year-old alcoholic man from Spain, who had no other medical conditions and no contact with animals sought care for headache and vomiting for 24 hours. He reported a 4-day history of fever and a painful right shoulder. At admission, temperature was 38.9°C, blood pressure 180/100 mm Hg, heart rate 68 beats/min, and respiratory rate 24 breaths/min. Neck stiffness and lethargic mental status were noted.

Laboratory tests revealed the following values: leukocytosis of 14 × 109 (reference range 3.9–10 × 109) cells/L with 90.4% neutrophils, platelets 100×109 (reference 135–333 × 109) cells/L, hemoglobin 16 (reference 12.6–16.6) g/dL, creatinine 131 (reference 0–111) μmol/L, and C-reactive protein 243 (reference 0–5) mg/L. Lumbar puncture yielded turbid cerebrospinal fluid (CSF), with high opening pressure (>32 cm H2O), pleocytosis (0.4 × 109 leukocytes/L; 88% neutrophils), high protein level (70 [reference range 15–45] mg/dL) and a low glucose level (<0.3 [reference 2.2–4.1] mmol/L). CSF showed gram-positive cocci in chains. Cefotaxime, dexamethasone, and mannitol were administered. After septic shock and respiratory insufficiency developed, the patient was transferred to the intensive care unit.

Streptococcus spp. grew in blood and CSF cultures. Although initially misidentified as S. bovis, the pathogen was confirmed as S. suis by sequence analysis of the 16S rRNA gene. Multilocus sequence typing (http://ssuis.mlst.net) identified this isolate as sequence type (ST) 3.

The patient was transferred to the medical ward 18 days after admission. Neurologic examination demonstrated vestibular ataxia, hearing loss, and diplopia resulting from cranial nerve VI palsy. Furthermore, a diagnosis of subacromial/subdeltoid bursitis led to arthroscopic debridement. Ceftriaxone was administered for 4 weeks. Results of abdominal computed tomography and echocardiogram were within normal limits. Because the Streptococcus organism was initially identified as S. bovis, colonoscopy and assessment of tumor markers were also requested; results were within normal limits.

After the patient was discharged (4 weeks after admission), diplopia and the shoulder mobility limitation completely resolved, but bilateral deafness and ataxia persisted. Five months later, the patient was readmitted for severe hypercalcemia. Positron-emission and computed tomography revealed liver, lung, and bone metastases. Tumor markers were elevated (carcinoembryonic antigen 4,152 [reference range 0–4.3] μg/L; monoclonal antibody CA-19–1 9,233 [0–39] U/mL). The patient died of multiorgan failure 21 days after admission. Necropsy revealed a disseminated esophageal adenocarcinoma.

S. suis is an encapsulated gram-positive, catalase-negative facultative anaerobe coccus, positive for Lancefield group antigens R-S or T. This pathogen of swine is infrequently transmitted to humans (13,6); recently, however, the number of S. suis cases in humans has increased substantially. Most cases have been reported in Europe and Southeast Asia, where pig farming is intensive (1). Although cases are usually sporadic, 2 outbreaks in China (1998 and 2005) caused a substantial number of deaths. Exposure to infected pigs was demonstrated for almost all patients. However, some patients had not been exposed to animals (1,2,4). S. suis can be an opportunistic pathogen in immunocompromised persons (1,2). Splenectomy is a well-established risk factor. Other predisposing factors are alcoholism, heart disease, and diabetes (1,4,5).

Although cancer is accepted as a risk factor (1,2), the reported number of cases with associated malignancy is quite low (Table). For all cases except one, cancer was diagnosed before or during the episode of infection. A primary adrenal lymphoma was diagnosed 1 year after S. suis meningitis (6), but probably an underlying defect in humoral immunity was already present. The patient reported here probably had subclinical malignancy at the time of infection. Although we cannot rule out a spurious relationship between cancer and infection, we believe that malignancy, in combination with the patient’s alcoholism, led to an immunosuppressed condition that facilitated the development of infection.

S. suis leads to a wide spectrum of clinical manifestations, meningitis being the most common (13,68). A higher frequency of sensorineural hearing loss is characteristic of S. suis meningitis (1). In the patient reported here, meningitis was complicated by permanent deafness, ataxia, and transient diplopia; to our knowledge, only 2 other cases complicated by diplopia have been reported (8,9).

S. suis ST3 belongs to ST clonal complex 1 and is associated with serotype 2 (http://ssuis.mlst.net). Although clonal complex 1 accounts for most S. suis infections in humans (1,10), genotype ST3 is extremely rare. To our knowledge, only 1 other human case of S. suis ST3 infection has been reported, also in Spain (10).

The patient reported here had severe S. suis infection with no prior exposure to swine but with undiagnosed neoplasia. In patients with no exposure to swine, we recommend searching for other predisposing factors, such as malignancy or other immunodeficiencies.

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Acknowledgments

We thank Michael Maudsley for revising the English-language manuscript.

S.G.-Z. is supported by a grant from the Instituto de Salud Carlos III (FIS PI11/00164). J.L.-T. is supported by a grant from the Instituto de Salud Carlos III (FI09/00943).

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Silvia Gómez-ZorrillaComments to Author , Carmen Ardanuy, Jaime Lora-Tamayo, Jordi Cámara, Dolors García-Somoza, Carmen Peña, and Javier Ariza
Author affiliations: University of Barcelona, Barcelona, Spain

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References

  1. Wertheim  HF, Nghia  HD, Taylor  W, Schultsz  C. Streptococcus suis: an emerging human pathogen. Clin Infect Dis. 2009;48:61725. DOIPubMedGoogle Scholar
  2. Manzin  A, Palmieri  C, Serra  C, Saddi  B, Princivalli  MS, Loi  G, Streptococcus suis meningitis without history of animal contact, Italy. Emerg Infect Dis. 2008;14:19468. DOIPubMedGoogle Scholar
  3. Voutsadakis  IA. Streptococcus suis endocarditis and colon carcinoma: a case report. Clin Colorectal Cancer. 2006;6:2268 . DOIPubMedGoogle Scholar
  4. Ma  E, Chung  PH, So  T, Wong  L, Choi  KM, Cheung  DT, Streptococcus suis infection in Hong Kong: an emerging infectious disease? Epidemiol Infect. 2008;136:16917. DOIPubMedGoogle Scholar
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  6. Yen  MY, Liu  YC, Wang  JH, Chen  YS, Wang  YH, Cheng  DL. Streptococcus suis meningitis complicated with permanent perceptive deafness: report of a case. J Formos Med Assoc. 1994;93:34951 .PubMedGoogle Scholar
  7. Arends  JP, Zanen  HC. Meningitis caused by Streptococcus suis in humans. Rev Infect Dis. 1988;10:1317 . DOIPubMedGoogle Scholar
  8. Geffner Sclarsky  DE, Moreno Muñoz  R, Campillo Alpera  MS, Pardo Serrano  FJ, Gómez Gómez  A, Martínez-Lozano  MD. Streptococcus suis meningitis. [in Spanish]. Med Interna. 2001;18:3178.PubMedGoogle Scholar
  9. Meecham  JS, Worth  RC. Persistent diplopia following Streptococcus suis type 2 meningitis. J R Soc Med. 1992;85:57980 .PubMedGoogle Scholar
  10. Vela  AI, Aspiroz  C, Fortuño  B, Tirado  G, Sierra  J, Martinez  R, Meningitis caused by an unusual genotype (ST3) of Streptococcus suis. Infection. 2013;41:7013 .DOIPubMedGoogle Scholar

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DOI: 10.3201/eid2006.131167

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Silvia Gómez-Zorrilla, Infectious Diseases Service, Hospital Universitario de Bellvitge, Bellvitge Biomedical Research Institute, Feixa Llarga s/n 08907, Hospitalet de Llobregat, Barcelona, Spain

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Page created: May 19, 2014
Page updated: May 19, 2014
Page reviewed: May 19, 2014
The conclusions, findings, and opinions expressed by authors contributing to this journal do not necessarily reflect the official position of the U.S. Department of Health and Human Services, the Public Health Service, the Centers for Disease Control and Prevention, or the authors' affiliated institutions. Use of trade names is for identification only and does not imply endorsement by any of the groups named above.
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