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Intracranial actinomycosis of odontogenic origin masquerading as auto-immune orbital myositis: a fatal case and review of the literature.
BMC Infect Dis 2019; 19(1):763BI

Abstract

BACKGROUND

Actinomycetes can rarely cause intracranial infection and may cause a variety of complications. We describe a fatal case of intracranial and intra-orbital actinomycosis of odontogenic origin with a unique presentation and route of dissemination. Also, we provide a review of the current literature.

CASE PRESENTATION

A 58-year-old man presented with diplopia and progressive pain behind his left eye. Six weeks earlier he had undergone a dental extraction, followed by clindamycin treatment for a presumed maxillary infection. The diplopia responded to steroids but recurred after cessation. The diplopia was thought to result from myositis of the left medial rectus muscle, possibly related to a defect in the lamina papyracea. During exploration there was no abnormal tissue for biopsy. The medial wall was reconstructed and the myositis responded again to steroids. Within weeks a myositis on the right side occurred, with CT evidence of muscle swelling. Several months later he presented with right hemiparesis and dysarthria. Despite treatment the patient deteriorated, developed extensive intracranial hemorrhage, and died. Autopsy showed bacterial aggregates suggestive of actinomycotic meningoencephalitis with septic thromboembolism. Retrospectively, imaging studies showed abnormalities in the left infratemporal fossa and skull base and bilateral cavernous sinus.

CONCLUSIONS

In conclusion, intracranial actinomycosis is difficult to diagnose, with potentially fatal outcome. An accurate diagnosis can often only be established by means of histology and biopsy should be performed whenever feasible. This is the first report of actinomycotic orbital involvement of odontogenic origin, presenting initially as bilateral orbital myositis rather than as orbital abscess. Infection from the upper left jaw extended to the left infratemporal fossa, skull base and meninges and subsequently to the cavernous sinus and the orbits.

Authors+Show Affiliations

Department of Ophthalmology, Erasmus Medical Center, Rotterdam, The Netherlands. g.hotte@oogziekenhuis.nl. Department of Orbital Oculoplastic and Lacrimal Surgery, The Rotterdam Eye Hospital, PO box 70030, 3000 LM, Rotterdam, The Netherlands. g.hotte@oogziekenhuis.nl.Department of Oral and Maxillofacial Surgery, Erasmus Medical Center, Rotterdam, The Netherlands.Department of Pathology, Erasmus Medical Center, Rotterdam, The Netherlands.Department of Neurology, Erasmus Medical Center, Rotterdam, The Netherlands.Department of Neurology, Franciscus Gasthuis and Vlietland, Rotterdam, The Netherlands.Department of Oral and Maxillofacial Surgery, Erasmus Medical Center, Rotterdam, The Netherlands.Department of Radiology, Erasmus Medical Center, Rotterdam, The Netherlands.Department of Ophthalmology, Erasmus Medical Center, Rotterdam, The Netherlands. Department of Orbital Oculoplastic and Lacrimal Surgery, The Rotterdam Eye Hospital, PO box 70030, 3000 LM, Rotterdam, The Netherlands.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

31477035

Citation

Hötte, G J., et al. "Intracranial Actinomycosis of Odontogenic Origin Masquerading as Auto-immune Orbital Myositis: a Fatal Case and Review of the Literature." BMC Infectious Diseases, vol. 19, no. 1, 2019, p. 763.
Hötte GJ, Koudstaal MJ, Verdijk RM, et al. Intracranial actinomycosis of odontogenic origin masquerading as auto-immune orbital myositis: a fatal case and review of the literature. BMC Infect Dis. 2019;19(1):763.
Hötte, G. J., Koudstaal, M. J., Verdijk, R. M., Titulaer, M. J., Claes, J. F. H. M., Strabbing, E. M., ... Paridaens, D. (2019). Intracranial actinomycosis of odontogenic origin masquerading as auto-immune orbital myositis: a fatal case and review of the literature. BMC Infectious Diseases, 19(1), p. 763. doi:10.1186/s12879-019-4408-2.
Hötte GJ, et al. Intracranial Actinomycosis of Odontogenic Origin Masquerading as Auto-immune Orbital Myositis: a Fatal Case and Review of the Literature. BMC Infect Dis. 2019 Sep 2;19(1):763. PubMed PMID: 31477035.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Intracranial actinomycosis of odontogenic origin masquerading as auto-immune orbital myositis: a fatal case and review of the literature. AU - Hötte,G J, AU - Koudstaal,M J, AU - Verdijk,R M, AU - Titulaer,M J, AU - Claes,J F H M, AU - Strabbing,E M, AU - van der Lugt,A, AU - Paridaens,D, Y1 - 2019/09/02/ PY - 2019/05/27/received PY - 2019/08/25/accepted PY - 2019/9/4/entrez PY - 2019/9/4/pubmed PY - 2019/9/4/medline KW - Actinomycosis KW - Intracranial infection KW - Intraorbital infection KW - Odontogenic origin KW - Orbital myositis SP - 763 EP - 763 JF - BMC infectious diseases JO - BMC Infect. Dis. VL - 19 IS - 1 N2 - BACKGROUND: Actinomycetes can rarely cause intracranial infection and may cause a variety of complications. We describe a fatal case of intracranial and intra-orbital actinomycosis of odontogenic origin with a unique presentation and route of dissemination. Also, we provide a review of the current literature. CASE PRESENTATION: A 58-year-old man presented with diplopia and progressive pain behind his left eye. Six weeks earlier he had undergone a dental extraction, followed by clindamycin treatment for a presumed maxillary infection. The diplopia responded to steroids but recurred after cessation. The diplopia was thought to result from myositis of the left medial rectus muscle, possibly related to a defect in the lamina papyracea. During exploration there was no abnormal tissue for biopsy. The medial wall was reconstructed and the myositis responded again to steroids. Within weeks a myositis on the right side occurred, with CT evidence of muscle swelling. Several months later he presented with right hemiparesis and dysarthria. Despite treatment the patient deteriorated, developed extensive intracranial hemorrhage, and died. Autopsy showed bacterial aggregates suggestive of actinomycotic meningoencephalitis with septic thromboembolism. Retrospectively, imaging studies showed abnormalities in the left infratemporal fossa and skull base and bilateral cavernous sinus. CONCLUSIONS: In conclusion, intracranial actinomycosis is difficult to diagnose, with potentially fatal outcome. An accurate diagnosis can often only be established by means of histology and biopsy should be performed whenever feasible. This is the first report of actinomycotic orbital involvement of odontogenic origin, presenting initially as bilateral orbital myositis rather than as orbital abscess. Infection from the upper left jaw extended to the left infratemporal fossa, skull base and meninges and subsequently to the cavernous sinus and the orbits. SN - 1471-2334 UR - https://www.unboundmedicine.com/medline/citation/31477035/Intracranial_actinomycosis_of_odontogenic_origin_masquerading_as_auto-immune_orbital_myositis:_a_fatal_case_and_review_of_the_literature L2 - https://bmcinfectdis.biomedcentral.com/articles/10.1186/s12879-019-4408-2 DB - PRIME DP - Unbound Medicine ER -