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Multiple aseptic splenic abscesses in a 15 year old patient
© Jordan et al.; licensee BioMed Central Ltd. 2014
Received: 4 October 2013
Accepted: 3 February 2014
Published: 6 February 2014
Splenic abscesses in children are rare. In recent years aseptic abscesses have been recognized as a new disease entity, especially in adults.
We present a rare case of a 15 year old girl with aseptic abscesses, in whom antibiotic therapy comprising metronidazole and meropenem was partly beneficial in improving the patient’s clinical condition and inflammatory parameters. Eventually corticosteroid therapy led to complete and long lasting resolution of symptoms. Further diagnostic work-up revealed autoimmune thyroiditis, but no signs of inflammatory bowel disease.
Aseptic splenic abscesses should always prompt clinicians to initiate further diagnostics to determine a potential underlying condition and a regular follow-up. Anaerobic bacteria may play a role in the pathogenesis of the disease and besides corticosteroid treatment antibiotics covering anaerobes may be beneficial.
Splenic abscesses represent a rare disease entity which is even rarer in the paediatric age group. In most cases, these abscesses are caused by an infectious agent like Staphylococcus aureus, streptococcus spp., Klebsiella pneumoniae, Escherichia coli, Mycobacterium tuberculosis, or Salmonella spp. Risk factors constitute infections of the abdominal cavity, endocarditis, and immunosupression [1, 2]. Malignant splenic lesions can also mimic abscesses if they are of the hypoechoic texture in ultrasound examination .
Serologic studies for acute infection with Epstein-Barr virus, cytomegalovirus, Herpes simplex-virus, Human immunodeficiency virus, Mycoplasma pneumoniae, Yersinia enterica/pseudotuberculosis, Treponema pallidum, Coxiella burnetii, Brucella spp., Bartonella henselae, Franciscella tularensis, and Echinococcus, as well as blood cultures were negative. A throat swab from the aphthous lesions showed normal resident flora. Urine analysis, chest X-ray and echocardiography as well as Mantoux test and interferon-gamma release assay were negative.
Aseptic abscesses represent a new disease entity that was only recently recognized [4, 5]. The spleen is the most frequently involved organ, but aseptic abscesses can also be found in abdominal lymph nodes, liver, lung, pancreas or the brain. Moreover, they can occur uni- or multifocal. Their aetiology is idiopathic or can be associated with other conditions such as relapsing polychondritis or inflammatory bowel disease, especially Crohn’s disease. The bowel inflammation can occur concomitantly, manifest prior or after the appearance of the abscesses [4, 5]. Therefore, it is still possible that our patient may develop inflammatory bowel disease in the future. However, during the follow-up she has not developed any other gastrointestinal symptoms.
In the paediatric population aseptic abscesses are very rare with only six reported patients younger than 18 years at diagnosis (age range 6 to 17) [4, 6, 7]. The clinical presentation seems to be similar to those in adults with fever, increased ESR and elevated white blood cell count. In four of the six patients the abscesses were primarily found in the spleen, in one patient only the lung and in another one the site of involvement was not mentioned. Two thirds of patients were reported to have a concomitant disease such as Crohn’s disease, pyoderma gangrenosum and/or relapsing polychondritis. To date, there are no reports of aseptic abscesses in children under the age of 5 years.
To our knowledge, our case is the first patient described with aseptic splenic abscesses in the absence of an overt accompanying clinical disorder, in whom the antibiotic regimen including meropenem and metronidazole improved the patient’s clinical condition, led to the healing of the aphthous ulcers of the throat and decreased systemic inflammatory markers. After stopping this antibiotic therapy inflammatory markers rose again, and could be decreased repeatedly after reintroduction of the same regimen. However, the antibiotic treatment did not have any beneficial effect on the number and size of the splenic abscesses. Of note, in one case of Crohn’s disease with multiple aseptic liver abscesses antibiotic therapy with meropenem and metronidazole also improved the patient’s clinical condition and inflammatory parameters . One possible explanation for this effect could be gram-negative anaerobic bacteria playing an aetiological factor. The effects of the antibiotic treatment could be exerted by the reduction of the luminal bacterial content in the gut that somehow triggers the inflammatory processes like in inflammatory bowel disease. This mode of action would fit into the hypothesis of gut derived chemokines that maintain a persistent local (splenic) inflammatory neutrophil infiltrate. Additionally, direct immunomodulatory effects of metronidazole may play a role [9–11].
Our observations in the presented case are in accordance with previously published data where the diagnosis of aseptic abscesses relies on a combination of typical clinical, laboratory and radiological findings and exclusion of infectious diseases and other causes . Involvement of the pharynx as in our case has once been described in a patient with aseptic abscesses . Of note, autoimmune thyreoiditis has so far not been described as a concomitant autoimmune mediated disease in patients with aseptic abscesses. Improvement and finally complete resolution of the splenic lesions after the patient receives an immunosuppressive therapy is another characteristic feature for aseptic abscesses, although the relapse rate is high with 32 to 66% when the immunosuppressive therapy is stopped .
Aseptic splenic abscesses should prompt the clinician to initiate a work-up of a potential underlying condition. Anaerobic bacteria may be an aetiological factor and metronidazole may have direct immunomodulatory effects to improve signs and symptoms.
Written informed consent was obtained from the parent of the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
- Llenas-García J, Fernández-Ruiz M, Caurcel L, Enguita-Valls A, Vila-Santos J, Guerra-Vales JM: Splenic abscess: a review of 22 cases in a single institution. Eur J Intern Med. 2009, 20: 537-539. 10.1016/j.ejim.2009.04.009.View ArticlePubMedGoogle Scholar
- Chang KC, Chuah SK, Changchien CS, Tsai TL, Lu SN, Chiu YC, Chen YS, Wang CC, Lin JW, Lee CM, Hu TH: Clinical characteristics and prognostic factors of splenic abscess: a review of 67 cases in a single medical center of Taiwan. World J Gastroenterol. 2006, 12: 460-464.View ArticlePubMedPubMed CentralGoogle Scholar
- Goerg C, Schwerk WB, Goerg K: Splenic lesions: sonographic patterns, follow-up, differential diagnosis. Eur J Radiol. 1991, 13: 59-66. 10.1016/0720-048X(91)90058-4.View ArticlePubMedGoogle Scholar
- André MF, Piette JC, Kémény JL, Ninet J, Jego P, Delèvaux I, Wechsler B, Weiller PJ, Francès C, Blétry O, Wismans PJ, Rousset H, Colombel JF, Aumaître O: French study group on Aseptic Abscesses: Aseptic Abscesses. A study of 30 patients with or without inflammatory bowel disease and review of the literature. Medicine. 2007, 86: 145-161. 10.1097/md.0b013e18064f9f3.View ArticlePubMedGoogle Scholar
- André M, Aumaître O: Aseptic abscesses syndrome. Rev Med Interne. 2011, 32: 678-688. 10.1016/j.revmed.2011.02.001.View ArticlePubMedGoogle Scholar
- Johnson JL, West DA, Haggstrom AN: Pyoderma Gangrenosum associated with an aseptic Splenic Abscess in a patient with Neurofibromatosis. Pediatr Dermatol. 2013, doi:10.1111/pde.12129 [Epub ahead of print]Google Scholar
- André MFJ, Aumaître O, Grateau G, Chamaillard M, Costedoat-Chalumeau N, Cardoso MC, Henry-Berger J, Ramakrishna BS, Delpech M, Piette JC, Creveaux I: Longest form of CCTG microsatellite repeat in the promoter of the CD2BP1/PSTPIP1 gene is associated with aseptic abscesses and with Crohn disease in French patients. Dig Dis Sci. 2010, 55: 1681-1688. 10.1007/s10620-009-0929-7.View ArticlePubMedGoogle Scholar
- Zakout R, Fonseca M, Santos JM, Marques A, Távora I, Oliveira E, Ferreira C, Victorino RM: Multiple aseptic liver abscesses as the initial manifestation of Crohn’s disease: report of a case. Dis Colon Rectum. 2009, 52: 343-345. 10.1007/DCR.0b013e318199db60.View ArticlePubMedGoogle Scholar
- Rizzo A, Paolillo R, Guida L, Annunziata M, Bevilacqua N, Tufano MA: Effect of metronidazole and modulation of cytokine production on human periodontal ligament cells. Int Immunopharmacol. 2010, 10: 744-750. 10.1016/j.intimp.2010.04.004.View ArticlePubMedGoogle Scholar
- Shakir L, Javeed A, Ashraf M, Riaz A: Metronidazole and the immune system. Pharmazie. 2011, 66: 393-398.PubMedGoogle Scholar
- Fararjeh M, Mohammad MK, Bustanji Y, Alkhatib H, Abdalla S: Evaluation of immunosuppression induced by metronidazole in Balb/c mice and human peripheral blood lymphocytes. Int Immunopharm. 2008, 8: 341-350. 10.1016/j.intimp.2007.10.018.View ArticleGoogle Scholar
- André M, Piette JC, Frances C, Wechsler B, Delevaux I, Aumaître O: Retropharyngeal and splenic aseptic abscesses treated with prednisone and cyclophosphamide in a patient with ulcerative colitis. Dig Dis Sci. 2003, 48: 1193-1195. 10.1023/A:1023745619294.View ArticlePubMedGoogle Scholar
- The pre-publication history for this paper can be accessed here:http://www.biomedcentral.com/1471-230X/14/20/prepub
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