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Giant Aneurysmal Bone Cyst of the Anterior Cranial Fossa and Paranasal Sinuses Presenting in Pregnancy: Case Report and Literature Review.

ABSTRACT: Background and Purpose Aneurysmal bone cysts (ABCs) rarely involve the cranium and have seldom been reported in pregnancy. Clinical Presentation We describe a case of a 28-year-old woman who presented at 37 weeks of gestation with 3 months of gradually worsening vision, 10 months of proptosis, and restricted ocular motility on the left. Brain imaging revealed a multicystic enhancing mass measuring 5.9 × 5.3 × 3.7 cm, centered on the cribriform plate on the left, extending into the anterior cranial fossa superiorly as well as the left nasal cavity, maxillary, sphenoid, and frontal sinuses. Her clinical course is described in detail; 3-month postoperative imaging demonstrated no residual mass. Conclusion A literature review revealed five previous cases of ABCs associated with pregnancy. We report a rare case of a giant ABC of fibrous dysplasia involving the paranasal sinuses and anterior cranial fossa. We postulate on the possible influence of pregnancy on the clinical course.

SUBMITTER: Hnenny L

PROVIDER: S-EPMC4648731 | biostudies-other | 2015 Nov

REPOSITORIES: biostudies-other

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Project description:Rhinosinusitis (RS) is an acute (ARS) or chronic (CRS) inflammatory disease of the nasal and paranasal sinus mucosa. CRS is a heterogeneous condition characterized by distinct inflammatory patterns (endotypes) and phenotypes associated with the presence (CRSwNP) or absence (CRSsNP) of nasal polyps. Mucosal barrier and mucociliary clearance dysfunction, inflammatory cell infiltration, mucus hypersecretion, and tissue remodeling are the hallmarks of CRS. However, the underlying factors, their priority, and the mechanisms of inflammatory responses remain unclear. Several hypotheses have been proposed that link CRS etiology and pathogenesis with host (eg, "immune barrier") and exogenous factors (eg, bacterial/fungal pathogens, dysbiotic microbiota/biofilms, or staphylococcal superantigens). The abnormal interplay between these factors is likely central to the pathophysiology of CRS by triggering compensatory immune responses. Here, we discuss the role of the sinonasal microbiota in CRS and its biofilms in the context of mucosal zinc (Zn) deficiency, serving as a possible unifying link between five host and "bacterial" hypotheses of CRS that lead to sinus mucosa remodeling. To date, no clear correlation between sinonasal microbiota and CRS has been established. However, the predominance of Corynebacteria and Staphylococci and their interspecies relationships likely play a vital role in the formation of the CRS-associated microbiota. Zn-mediated "nutritional immunity", exerted via calprotectin, alongside the dysregulation of Zn-dependent cellular processes, could be a crucial microbiota-shaping factor in CRS. Similar to cystic fibrosis (CF), the role of SPLUNC1-mediated regulation of mucus volume and pH in CRS has been considered. We complement the biofilms' "mechanistic" and "mucin" hypotheses behind CRS pathogenesis with the "structural" one - associated with bacterial "corncob" structures. Finally, microbiota restoration approaches for CRS prevention and treatment are reviewed, including pre- and probiotics, as well as Nasal Microbiota Transplantation (NMT).

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Giant Aneurysmal Bone Cyst of the Anterior Cranial Fossa and Paranasal Sinuses Presenting in Pregnancy: Case Report and Literature Review.

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