Intracranial actinomycosis of odontogenic origin masquerading as auto-immune orbital myositis: a fatal case and review of the literature.


Journal

BMC infectious diseases
ISSN: 1471-2334
Titre abrégé: BMC Infect Dis
Pays: England
ID NLM: 100968551

Informations de publication

Date de publication:
02 Sep 2019
Historique:
received: 27 05 2019
accepted: 25 08 2019
entrez: 4 9 2019
pubmed: 4 9 2019
medline: 15 11 2019
Statut: epublish

Résumé

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. 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. 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.

Sections du résumé

BACKGROUND 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 METHODS
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 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.

Identifiants

pubmed: 31477035
doi: 10.1186/s12879-019-4408-2
pii: 10.1186/s12879-019-4408-2
pmc: PMC6720412
doi:

Types de publication

Case Reports Journal Article Review

Langues

eng

Sous-ensembles de citation

IM

Pagination

763

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Auteurs

G J Hötte (GJ)

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.

M J Koudstaal (MJ)

Department of Oral and Maxillofacial Surgery, Erasmus Medical Center, Rotterdam, The Netherlands.

R M Verdijk (RM)

Department of Pathology, Erasmus Medical Center, Rotterdam, The Netherlands.

M J Titulaer (MJ)

Department of Neurology, Erasmus Medical Center, Rotterdam, The Netherlands.

J F H M Claes (JFHM)

Department of Neurology, Franciscus Gasthuis and Vlietland, Rotterdam, The Netherlands.

E M Strabbing (EM)

Department of Oral and Maxillofacial Surgery, Erasmus Medical Center, Rotterdam, The Netherlands.

A van der Lugt (A)

Department of Radiology, Erasmus Medical Center, Rotterdam, The Netherlands.

D Paridaens (D)

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.

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