Recurrent recalcitrant gingival hyperplasia and plasminogen deficiency: a case report.
BACKGROUND
Recurrent gingival hyperplasia due to plasminogen deficiency is a rare condition due to fibrin deposition in the connective tissue. Only eight cases have previously been reported in the English literature, and all cases were diagnosed before the age of 35 years. This paper presents an older patient with recurrent gingival hyperplasia due to plasminogen deficiency (hypoplasminogenemia).


METHODS
A 59-year-old woman presented with recurrent gingival swelling of 6 years' duration. Multiple biopsies performed at various time periods were histologically reported to be gingival hyperplasia with chronic inflammation. Routine hematoxylin and eosin (H & E) staining and direct immunofluorescence were performed.


RESULTS
H & E-stained sections showed subepithelial, eosinophilic, amorphous, acellular deposits. Direct immunofluorescence showed positive staining for fibrin, immunoglobulin (Ig) G, IgA, and IgM. Functional plasminogen and plasminogen activator inhibitor-1 assays were done and found to be deficient. A diagnosis of gingival hyperplasia due to plasminogen deficiency (hypoplasminogenemia) was rendered.


CONCLUSIONS
Recurrent gingival hyperplasia due to plasminogen deficiency (hypoplasminogenemia) is a newly recognized and rare condition. H & E staining, direct immunofluorescence, and assessment of functional plasminogen levels are essential to differentiate this condition from other conditions in which subepithelial, eosinophilic, amorphous materials are deposited.
Suresh
Lakshmanan
L
Department of Oral Diagnostic Sciences, School of Dental Medicine, University at Buffalo, Buffalo, NY 14214, USA.
Aguirre
Alfredo
A
Kumar
Vijay
V
Solomon
Lynn W
LW
Sielski
Edward A
EA
Neiders
Mirdza E
ME
D003677:Deficiency Diseases / Q000150:complications; D005260:Female; D005885:Gingival Hyperplasia / Q000209:etiology*; D006402:Hematologic Diseases / Q000150:complications; D006801:Humans; D008875:Middle Aged; D010958:Plasminogen / Q000172:deficiency*; D012008:Recurrence
D010958:Plasminogen