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rev bras hematol hemoter. 2016;38(3):274–275

w w w . r b h h . o r g

Revista

Brasileira

de

Hematologia

e

Hemoterapia

Brazilian

Journal

of

Hematology

and

Hemotherapy

Images

in

Clinical

Hematology

Gingival

swelling

associated

with

hypoplasminogenemia

Eric

T.

Stoopler

,

Faizan

Alawi

UniversityofPennsylvania,SchoolofDentalMedicine,Philadelphia,UnitedStates

a

r

t

i

c

l

e

i

n

f

o

Articlehistory:

Received24March2016 Accepted14April2016 Availableonline3May2016

Amiddle-agedwomanpresentedforevaluationofgingival swelling.Shewaspreviouslydiagnosedwith hypoplasmino-genemiaandcurrent plasminogenlevelwas29%(reference value:78–130%). Intraoralexaminationrevealed swelling of the right maxillary gingiva (Figure 1). Biopsy with routine histopathologic analysis revealed fibrinoid deposits and a mixedinflammatoryinfiltratewithinthelaminapropria, con-sistentwithhypoplasminogenemia(Figure2).

Hypoplasminogenemia (type 1 plasminogen deficiency) is commonly associated with ligneous conjunctivitis and ligneous gingivitis.1–3 An oral mucosal biopsy can aid in the diagnosis of this rare condition as histopatho-logic evidence of fibrin deposition is highly suggestive of hypoplasminogenemia.2,3

Correspondingauthorat:DepartmentofOralMedicine,UniversityofPennsylvaniaSchoolofDentalMedicine,240South40thStreet,

Philadelphia,PA19104,USA.

E-mailaddress:[email protected](E.T.Stoopler).

Figure1–Swellingoftherightanteriormaxillarygingiva.

http://dx.doi.org/10.1016/j.bjhh.2016.04.006

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revbrashematolhemoter.2016;38(3):274–275

275

A

B

*

*

*

*

*

Figure2–Amorphousfibrinoiddeposits(*)inthegingivallaminapropria.(A)Hematoxylin–eosin(magnification×100),(B)

trichrome(magnification×40).

Conflicts

of

interest

Theauthorsdeclarenoconflictsofinterest.

Acknowledgements

TheauthorsthankDr.SophiaElmuradi forassistancewith clinicalphotography.

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1.SchusterV,HugleB,TefsK.Plasminogendeficiency.JThromb

Haemost.2007;5(12):2315–22.

2.ScullyC,GokbugetAY,AllenC,BaganJV,EfeogluA,ErsevenG,

etal.Orallesionsindicativeofplasminogendeficiency

(hypoplasminogenemia).OralSurgOralMedOralPatholOral

RadiolEndod.2001;91(3):334–7.

3.ScullyC,GokbugetA,KurtulusI.Hypoplasminogenaemia,

Imagem

Figure 1 – Swelling of the right anterior maxillary gingiva.
Figure 2 – Amorphous fibrinoid deposits (*) in the gingival lamina propria. (A) Hematoxylin–eosin (magnification ×100), (B) trichrome (magnification ×40).

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