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Is pneumosinus dilatans an osteogenic disease that mimics the formation of a paranasal sinus?

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Abstract

Purpose

Pneumosinus dilatans is a disease that produces an abnormal expansion of a paranasal sinus cavity, which contains only air and is lined by normal mucosa, and whose bony walls are displaced outwardly to cause facial embossing or intracranial, orbital or ethmoidal encroachment. Objective was to evaluate the hypothesis that pneumosinus dilatans is primarily an osteogenic disease.

Methods

A detailed clinical history of three consecutive patients with pneumosinus dilatans was taken. Each patient also underwent computed tomography (CT), fluorine-deoxyglucose positron emission tomography-CT (FDG PET-CT), fluorine 18-labeled sodium fluoride PET-CT (NaF PET-CT), and bone pathology.

Results

The FDG PET-CT and pathology confirmed that the mucosa inside the pneumosinus dilatans was normal and devoid of inflammatory cell infiltrate. Significant uptake of 18F-NaF on PET-CT images correlated well with bone pathology, showing intense and diffuse bone remodeling. At a 1-year follow-up, following a frontotomy for case #1 and a middle antrostomy for case #2, there was a marked resolution of the patients’ clinical symptoms and deformities, new bone formation on the walls, stabilization of the new sinus shape and volume, and persistence of significant uptake of 18F-NaF.

Conclusions

Pneumosinus dilatans is a rare disease. Its diagnosis is based on CT scan images. This study has shown that 18F-NaF PET-CT and bone pathology are useful modalities for the positive diagnosis of difficult cases. Pneumosinus dilatans appears to be an osteogenic disease. Further research is needed to investigate a possible link between mechanisms involved in paranasal sinus formation and those involved in pneumosinus dilatans.

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Conflict of interest

The authors declare they have no conflict of interest.

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Authors and Affiliations

  1. Service ORL et Chirurgie Cervico-Faciale, Faculté de Médecine, Hôpital Central, Université de Lorraine, 54000, Nancy, France

    Roger Jankowski, Sébastien Kuntzler & Nicolas Boulanger

  2. Service de Médecine Nucléaire, Faculté de Médecine, Hôpital de Brabois, Université de Lorraine, Nancy, France

    Olivier Morel

  3. Cabinet ORL, Nancy, France

    Jean Tisserant

  4. Service ORL et Chirurgie Cervico-Faciale, Centre Hospitalier, Remiremont, France

    Nouredin Benterkia

  5. Service d’Anatomie et Cytologie Pathologiques, Faculté de Médecine, Hôpital Central, Université de Lorraine, Nancy, France

    Jean-Michel Vignaud

Authors
  1. Roger Jankowski
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  2. Sébastien Kuntzler
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  3. Nicolas Boulanger
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  4. Olivier Morel
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  5. Jean Tisserant
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  6. Nouredin Benterkia
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  7. Jean-Michel Vignaud
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Corresponding author

Correspondence to Roger Jankowski.

Additional information

This study has been approved by the Institutional Review Board of the Centre Hospitalo-Universitaire de Nancy, France.

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Jankowski, R., Kuntzler, S., Boulanger, N. et al. Is pneumosinus dilatans an osteogenic disease that mimics the formation of a paranasal sinus?. Surg Radiol Anat 36, 429–437 (2014). https://doi.org/10.1007/s00276-013-1222-1

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  • DOI: https://doi.org/10.1007/s00276-013-1222-1

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