CAN OBSTRUCTIVE INTRALYMPHATIC GRANULOMAS BE THE CAUSE OF CHEILITIS GRANULOMATOSA?

A Szuba, M Nittner-Marszalska, C Kobierzycki, K Solarewicz-Madejek, A Wolanczyk-Medrala, P Dziegiel

Abstract


Cheilitis granulomatosa (ChG), also
known as Miescher’s cheilitis, is an
uncommon, immunologically mediated nonnecrotizing
granulomatous inflammatory
disease characterized by recurrent, painless
swelling of the lips. The aim of the study was
a pathomorphological and immunohistochemical
assessment of cases clinically
classified as ChG to investigate potential
pathological mechanisms of ChG symptoms
and to verify the hypothesis of intravascular
granulomas as a cause of lymphatic vessel
obstruction and localized edema in ChG.
We report 6 patients with ChG who clinically
presented localized edema of the lips. Lip
biopsy with pathomorphological and immunohistochemical
examination was performed in
all cases. We found discrete, non-necrotizing
granulomas which were adjacent to numerous
blood and lymphatic vessels. The lumen of
lymphatic channels was dilated and was
either empty or contained lymph and few
macrophages or was completely occluded by
nearby granulomas. All patients demonstrated
a characteristic pattern of lymphangiectasia
and perivascular lymphatic aggregates with
evidence of non-necrotizing granulomas. None
manifested intralymphatic granulomas. These
results do not support the view that lymphatic
vessel obstruction is caused by intravascular
histiocytic granulomas described as the main
part in the etiology of lymphatic edema in
ChG. However, perivascular granulomas and
dilation of lymphatic vessels confirm presence
of inflammatory lymphostasis in all studied
cases of ChG.


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