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Relapsing polychondritis presenting with inflammatory pseudotumor

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Relapsing polychondritis (RP) is an uncommon, chronic multisystem disorder characterized by recurrent episodes of cartilaginous tissue inflammation... He had a history of recurrent inflammatory episodes involving auricular and nasal cartilage, as well as a history of conjunctivitis... Cryptococcus antigen test and mycobacterium tuberculosis polymerase chain reaction test were negative... The final CSF culture results revealed no organisms... He was started on IV methylprednisolone, 1 g/day over 5 days followed by oral prednisolone, 60 mg/day... High dose steroid therapy resulted in full neurologic recovery after 3 weeks... The prednisolone dosage was gradually reduced... Proptosis with chemosis simulating an orbital pseudotumor is the uncommon manifestation, but may be the initial presentation of RP... In this case, a biopsy was not performed for tissue confirmation because of the lesion location... Lichauco et al. reported an orbital mass in a patient with RP that was confirmed by biopsy as mucosa-associated lymphoid tissue type B cell lymphoma... In the reported case by Lichauco et al., biopsy was performed because the initial steroid treatment was only partially effective... We report our experience with a patient diagnosed with RP and pseudotumor who presented with multiple cranial nerve palsies... In RP patients, CNS and ocular manifestations are diverse.

No MeSH data available.


Related in: MedlinePlus

Fat-saturated contrast-enhanced T1-weighted magnetic resonance imaging showing: (A, C) diffuse enhancing thickening involving left orbital apex (arrows); (B) enhancing pachymeningeal thickening, including in left Meckel’s (trigeminal) cave (arrowheads); (D) cavernous enhancement bilaterally. These findings were considered consistent with inflammatory pseudotumor.
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f2-kjim-2015-076: Fat-saturated contrast-enhanced T1-weighted magnetic resonance imaging showing: (A, C) diffuse enhancing thickening involving left orbital apex (arrows); (B) enhancing pachymeningeal thickening, including in left Meckel’s (trigeminal) cave (arrowheads); (D) cavernous enhancement bilaterally. These findings were considered consistent with inflammatory pseudotumor.

Mentions: Magnetic resonance imaging (MRI) of the cranial nerves showed diffuse thickening and enhancement of the bilateral cavernous sinuses, orbital apex, and pachymeninges along with Meckel’s cave (Fig. 2). These findings suggest inflammatory pseudotumor involvement of the cranial nerve pathways. Nerve conduction study, electromyography, and blink test were performed. The results indicated bilateral trigeminal nerve dysfunction. Biopsy of the lesion was not performed because it was difficult to reach and the procedure was considered high risk for complications such as nerve injury.


Relapsing polychondritis presenting with inflammatory pseudotumor
Fat-saturated contrast-enhanced T1-weighted magnetic resonance imaging showing: (A, C) diffuse enhancing thickening involving left orbital apex (arrows); (B) enhancing pachymeningeal thickening, including in left Meckel’s (trigeminal) cave (arrowheads); (D) cavernous enhancement bilaterally. These findings were considered consistent with inflammatory pseudotumor.
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC5016277&req=5

f2-kjim-2015-076: Fat-saturated contrast-enhanced T1-weighted magnetic resonance imaging showing: (A, C) diffuse enhancing thickening involving left orbital apex (arrows); (B) enhancing pachymeningeal thickening, including in left Meckel’s (trigeminal) cave (arrowheads); (D) cavernous enhancement bilaterally. These findings were considered consistent with inflammatory pseudotumor.
Mentions: Magnetic resonance imaging (MRI) of the cranial nerves showed diffuse thickening and enhancement of the bilateral cavernous sinuses, orbital apex, and pachymeninges along with Meckel’s cave (Fig. 2). These findings suggest inflammatory pseudotumor involvement of the cranial nerve pathways. Nerve conduction study, electromyography, and blink test were performed. The results indicated bilateral trigeminal nerve dysfunction. Biopsy of the lesion was not performed because it was difficult to reach and the procedure was considered high risk for complications such as nerve injury.

View Article: PubMed Central - PubMed

AUTOMATICALLY GENERATED EXCERPT
Please rate it.

Relapsing polychondritis (RP) is an uncommon, chronic multisystem disorder characterized by recurrent episodes of cartilaginous tissue inflammation... He had a history of recurrent inflammatory episodes involving auricular and nasal cartilage, as well as a history of conjunctivitis... Cryptococcus antigen test and mycobacterium tuberculosis polymerase chain reaction test were negative... The final CSF culture results revealed no organisms... He was started on IV methylprednisolone, 1 g/day over 5 days followed by oral prednisolone, 60 mg/day... High dose steroid therapy resulted in full neurologic recovery after 3 weeks... The prednisolone dosage was gradually reduced... Proptosis with chemosis simulating an orbital pseudotumor is the uncommon manifestation, but may be the initial presentation of RP... In this case, a biopsy was not performed for tissue confirmation because of the lesion location... Lichauco et al. reported an orbital mass in a patient with RP that was confirmed by biopsy as mucosa-associated lymphoid tissue type B cell lymphoma... In the reported case by Lichauco et al., biopsy was performed because the initial steroid treatment was only partially effective... We report our experience with a patient diagnosed with RP and pseudotumor who presented with multiple cranial nerve palsies... In RP patients, CNS and ocular manifestations are diverse.

No MeSH data available.


Related in: MedlinePlus