Unusual gingival epithelial proliferation: Primary pseudoepitheliomatous hyperplasia

Short communications & case reports

Unusual gingival epithelial proliferation: Primary pseudoepitheliomatous hyperplasia

A primary pseudoepitheliomatous histologic features of carcinoma suggested that the histogenesis dental lamina.

hyperplasia arising in the gingiva of a g-year-old boy with was surgically excised and has not recurred after 2 years. It is is atypical proliferation of an aborted gingival cyst arising from

R

ecentlq we happened upon a peculiar gingival lesion presenting clinical and histologic characteristics which led to a diagnostic quandary. As a result, some consultant pathologists classified the lesion as malignant while others considered it benign. Because of the apparent rarity, of this lesion, the diagnostic nosologic problem, the questionable method of treatment. and the prognostic uncertainty. we believe that this case is of special interest CASE REPORT

Fig. 1. Clinical 23, 1976.

Hi\tologlc c,xamination rtz\ealctl ;I hcmisccred triangular plcue of. \ofl tissue co\crcd Mlth alternating uirophic and

appearance

ot’ teslon when tirsr tori

un

Sept.

Primury pseudoepirheliomatous hyperplasia

Volume 47 Number 5

Fig. 3. Low-power photomicrographof incisional biopsy tissue showing numerous keratocystic SIX. (Hematoxylin and eosin stain. Magnification, ~3.)

437

spaces of varying

thickened stratilied squamous epithelium (Fig. 3). At the crest of the specimen there was an inversion of the epithelium into the underlying connective tissue associated with a keratocystic mass which appeared to be exteriorized. The contiguous connective tissue stroma contained islands and cords of stratified squamous epithelial cells exhibiting microcystic formation. individual cell keratinization. acanthosis, mild acantholyais. and cellular disorientation. The lesion was diagnohcd as an aborted gingival cyst, and complete excision uaa recommended. On Oct. 30. 1976. the patient presented for excision of the lesion. At this time the lesion extended from the maxillary leti central incisor to the left first primary molar (Fig. 3). It was removed by hide excision under local anesthetic.

Histologic examination revealed an elongated. hemisected strip of soft tissue covered with stratified squamous epithclium of varying thickness. The surface was papillap with numerous in\,olutionh containing parakeratin and orthokeraIIII.

The rcte ridges were elongated and extended into the

undcrlymg connectice tissue, wherein they appcarcd to be t,rming keratin cysta (Fig. 5). Individual cell keratiniration. keratin pcarlh. and cellular disorientation were noted (Fig. 6). The surrounding connective tissue contained a light infiltrate 01‘ chronic inflammatory cells. The inferior margins of the \pcc!mcn contained normal crevicular epithelium. There wcrc no inicroorganisilIx or other cellular features commonly asso&cd with secondary pseudoepitheliomatous hyperplasia. The diagnosis at this time was aborted, atypical gingival cyst with pseudoepitheliomatous hyperplasia (primary). The patient has been f’ollowed regularly tbr 24 months. Thcrc I\ no clinical evidence of recurrence (Fig. 7).

DISCUSSION

Inasmuch as the histomorphology of the lesion was unusual. representative microscopic sections were sent

Fig. 4. Clinical Oct. 30. 1976.

appearance

at time of surgical

excision

on

to several pathologists for consultation. Not surprisingly. the diagnoses received from the consultants varied considerably. The consultative diagnoses included inverted papilloma, carcinoma. and atypical epithelial hyperplasia, and one consultant mentioned that he believed it was somehow related to dental lamina cysts. In addition to the aforementioned, nevus unis lateralis. verruca vulgaris. and keratoacanthoma were considered as diagnoses. In establishing the tinal diagnosis, an assumption had to be made. We had to either assume that the proliferating epithelium originated from an epithelial cell source

within

the gingival

lamina propria and ultimately

abutted the overlying epithelium ing epithelium originated from and

extended

into

the

gingival

or that the proliferatthe surface epithelium lamina

propria.

Al-

\IIo\\ “,y iiliiii itlu;i/ Fig. 6. Hiphcr-power ~‘ilorc’iilli‘n)gt.~l’ti orientation. (Hzmatox\~lln ,~nd cobin stain .~la~niti~,;~t~cln

CL‘II

hsimtlrr

h~t~~~l~r~~/,,li,~~i.

1>c;11.b,. 2nd ~‘dltiiar

dis-

,-HI 1 though

the hi\tologic

helievc

that the proliferating

an epithclial us

cell

concluded

c\~~cience

\ourcc that

dcrivecl

rests 01‘ the These rest\ or tumor\.

t’rom g~ngl\‘a cmld

0~.

originated

ue from

uithin

the lamina

propria.

the lesion

represented

3 primary

(idiopathic) l~~~t~docpithclit)n~~t(~us rna~ ha\ e dc\ eloped from cpithelial I-N\

uncqulvocal,

IS not

epithelium

the dental

It

hyperplasia which odontopenic cell

lamina.

Epithelial

cell

emanate from xveral sources. potentially give r&c to cpithelial cyst5 ma)

such bourcc ol‘epithclium

in the gingiva

i\ implantation -&i;l trauma. It should be pointed out that the patient in this i’asc did suffer trauma to the affected ;I~c‘;I mcl ttl:tt the histogencsi\ 1)1‘ the reported lesion Lx~t~l(l

ha\c

;II’IXC~ from

implanted

eplthelium.

Kelln’

Volume 47 Number 5

Fig. 8. LOM-power photomicrograph ot’ keratocysts toxylin and eosin stain. Mqnitication. X4.)

Fig. 9. Lou-power photolnicrogl.3ph aswciatea.’ (Hematoxylin and twin

noted in an infant with chondrocctodcmmal

of developmental cyst:, in posterior stain. .M;qnitic;ltion. ~30.)

has suggested aborted epithelium destined to become salivary glands as a possible source of oral epidermal cysts. Similarly, Burke and associates’ suggested that abortion of predestined salivary epithelium may be ;I source of Epstein’s pearls and f?idpalatal cysts. Finally. rests of epithelium in the gingiva could represent residual epithelial elements of the dental lamina. To us, this is the most seductive theor\ of histogenesis. Kreshove?’ found an unexpected nulnber of gingival

dysplasia.

(Herno

hard palate ;IS reportecl by Burke and

cysts while making serial sections from the maxillas and mandibles of several infants. He suggested that the gingival cyst represented the unusual progression and fate of degenerating dental lamina. Recently Fromm’ suggested that it would be more appropriate to call these cysts dental lamina cysts rather than gingival cysts. gingival

In rcvieuing cysts

previous

of infants.

material

we found

in our

that gingival

files

on

tissue

from an infant with chondroectodermal dysplasia clini-

440 El;u~~ md 0’ Keefk

tally

had

numerous

gingival

these cysts exhibited kcratin

abutting

mous

concentric

the overlying

atrophic

epithelium

these cysts.

(Fig.

\ia

morphology

ing similarity

(Fig.

The in\crtod diagnosis.

Hobcber.

papilloma

reported

sion

\,acuolated

and

in \crruca of anaplasia there

were

heratoc\stic

negated matous

h!,perplasia.

entitled

Hyperplasia.”

either type.

or the nodulovegetating

according

and Abulafia”

in reporting

indicated to

that their

Grinspan

and

culls

rcvicucd

in their

article

Pseudoepitheliomatous

intcrcst

type.

cast.

pseudoepithelio-

is the fact that the!

pseudoepithcliomatous

the verrugoma

thoma.

unis of the

or granular

ot‘ secondary

2

lends

of nevus because

basic

hyperpla\ia

types.

pseudoepitheliomatclus

and Buchanan.!’

that after

spaces noted in the present

into three

primaq

but I‘L‘;I-

of the lesion

organism:,

tht

typicall!

The fact

considered

(It’ particular

(keratoacanthorna)

U;I~

carcinoni;l

The diagnosis

Cutaneous

primary

as a

lesion

diagnoses,

of kcratoacanthoma

“Idiopathic

classitied

present

morphology

lacking.

Grinspan

forms

keratocyhtic

Squamous-cell

of‘ mycotic

the diagnosis

the L arious

the le-

of epitheli-

and did not contain

peg

not seriously

The absence

islands

and

has been no recurrence

was

numerous

keratin the

rete

to its benignity.

latcralis

inverted

exhibited

was also considered

I ulgaris.

years

as a possible

of’ oral

islands

in the differential

turrx

tied

of

\ulgaris than exophqtic

cells

b\, Burke

whereas

contained

histologicall>

u as considered

credence

cases

epithelium.

primarily

more endophytlc found

was considered

cast

SPXCS.” ’ I!erruc;I but

chondroectowe note a .strik-

in the literature

in the present

diagnosis.

lesion,

the three

ucantholytic

composed

UHI

reported

Y).

papilloma

ot‘ xquamous

from

cysts

our present

that

the histo-

cysts

and palatai uith

01 squa-

may terminate

Comparing

of the gingival

and associates’

laminations stratified

pearls.

exteriorization.’

dysplasia

Histologically.

It has been suggested

8).

as h.ell as Epstein’s

ultimately dermal

cysts.

multiple

;I\

sebaceum

In 1960 Helsham

a case of oral lesion

classi-

hyperplasia

the molluscum type.

The)

could

Abulatia’s

keratoacailbe classified system

as a