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(1)

CAPILLARY

LYMPHANGIOMA

OF

THE

TONGUE

COMPLICATED

BY GLOSSITIS

C. Everett Koop, M.D., Sc.D. (Med.), and Elias A. Moschakis, M.D.

Surgical Clinic, the Children’s Hospital of Philadelphia, and the Harrison Department of

Surgical Research, School of Medicine, University of Pennsylvania

Presented at the Annual Meeting of the American Academy of Pediatrics, October 16, 1960.

ADDRESS: (C.K.) Children’s Hospital, 18th and Bainbridge Streets, Philadelphia 46, Pennsylvania.

Psnwrmcs, May 1961

800

O

NE OF the most perplexing but

fortu-nately rare intra-oral lesions that we

have encountered in children is that of

lingual capillary lymphangioma complicated

by glossitis. The eight cases here reported

and seen in a 13-year period of observation

have in each instance been associated witil

a major lymphangioma. One of these was

confined to the tongue, two were subglottic

and the remainder were large cystic

hy-gromas of the neck and face. During this

same period, we have seen two additional

lymphangiomas of tile tongue unassociated

with the eruptive phenomena on the

sur-face of the tongue.

Medical literature contains numerous

ref-erenees to hereditary or familial

hemor-rhagic telangiectasia, with an excellent

re-view and bibliography by Hodgson et al.1

However, little can be found concerning the

lesion here described which has not had a

familial aspect in a single instance.

Several authors,25 in writing about cystic

hygromas of the neck, have alluded to the

concomitant finding of macroglossia or

lymphangiomatous involvement of tile floor

of the mouth. Lierle3 described three cases

of macroglossia associated with cystic

hy-groma and noted superficial ulceration of

the mucous membrane as well as deformity

of the mandible. Fuller et al. found

macro-glossia to be an infrequent finding with

cystic hygroma of the neck, but one of their

illustrative photographs showed a mild form

of the dynamic pathologic process to be

de-scribed ilere. Perzik reported a single case

of macroglossia and described the tongue

as being superficially ulcerated and

crust-covered and continuously exuding a

sero-sanguinous discharge. He suggested that

excision of tile sublingual extension of tile

cervical hygroma causes regression of the

macroglossia. Our experience would

mdi-eate that although removal of sublingual

ex-tensions of the cervical lymphangioma

might reduce the intra-ora! contents, the

macroglossia per se is not altered.

Goetsch,#{176} in his classic paper on cystic

hygromas, considered them to be benign

true neoplastic tumors of lymphatic origin,

excision of which is usually followed by

cure. Harkins and Sabiston, in discussing

the pathology of lymphangiomas, stated that

the cavernous lymphangiomas occur in the

tongue among several sites. They are

de-scribed as diffuse, spongy, compressible

masses, the microscopic appearance of

which consists chiefly of multiple dilated

lymph channels with abundant or scanty

fibrous stroma. The lesions described here,

although similar in histologic appearance to

photomicrographs of Harkins and Sabiston

had lymphatic channels capillary in size,

and the tongues were certainly neither

spongy nor compressible.

CLINICAL CHARACTERISTICS

The clinical history begins with the

spon-taneous eruption of a variable number of

clear, fluid-filled vesicles on tile dorsum of

the tongue, which apparently are not painful

(Fig. 1). These disappear in several days, to

be replaced by others. Indeed, tile process

is quite dynamic, with the surface of the

tongue changing rapidly. As the vesicles

spontaneously disappear, a relatively small

number of them leave a residual

papilloma-tous lesion that in some early instances

re-sembles a papule of granulation tissue and

(2)

801

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5.- .

. --.:-

-*. -.:.

- .,. ..,,..-, .t

.

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*

Fic. 2. (BA.) Late appearance of tongue after vesicles. Note hypertrophic villi, hemorrhagic

carious teeth.

recurrent eruptions of lymphangiectasia and

Tile former bleed easily and are painful to

touch, while the latter seem to have lost their propensity for bleeding but are

never-theless 1)ainful.

The latter two lesions tend to remain,

once they have appeared, although they,

too, seem capable on occasion of sloughing

at the base to leave a normal appearing

tongue surface behind. Indeed, some

tongues subject to this phenomenon look

almost normal for days or even weeks, only to have tile lesions reappear in tile same

order but not necessarily with the same

speed or in tile same quantity.

At times there is obvious edema and

pro-trusion of the tongue (which was thought

at birth to he too large in five of the eight

patients ill the present series). After

re-peated episodes of swelling, the tongue fails

to regaul its former size and seems

perma-nently hypertrophied. Acute glossitis with

swelling, excessive salivation, increased

Paffi, fever, and even respiratory distress due to obstruction, may be initiated by

respiratory infection or ingestion of certain

foods, or it may be idiopathic (Fig. 3). In

tile small infant, nursing becomes

impos-sible, fluid intake is difficult and parentera!

fluid therapy must follow. In older children,

Fic. 1. (C.O.) Fluid filled vesicles erupting on surface of tongue. Note smaller hemorrhagic

papillae in lower left corner.

eating is at first difficult, then too painful to

be attempted. Speech, already suffering

from some impediment under the best of

conditions due to the macroglossia, becomes

more difficult because of pain and swelling.

Sleep is possible only with the tongue

ex-truded from the mouth, in which position

(3)

4

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V :4- . ,! #{149}

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Fic. 3 (K.O.) Edematous painful tongue with confluent lesions.

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%4J’ 802

- ‘t -. 4’ ,

extension of tile process just described (Fig.

4).

All patients are not affected in the same

degree by this eruptive phenomenon. Oddly

enough, the three patients of this group

re-quiring least attention to the

!ymphangiec-tasias on the surface of the tongue and now

asymptomatic were those in whom the

ma-jor lymphangioma was confined to the

Fic. 4. (B.A.) Subsiding acute exacerbation 1 year after partial glossectomy. This tongue fits well into mouth, when free of glossitis. Note

prominence of mandible.

tongue or subglottic area without extension

into the submaxillary region or other

tn-angles of the neck. Of the remaining five,

three are yet problems in management.

None has reached puberty.

The major visible tumor masses were all

seen at or shortly after birth, while in only

half the patients were the vesieles over the

dorsum of the tongue prominent enough to

be recorded at the same time. Clinical

prob-lems with the lymphangiectasias arose in

the newborn period in only a single infant.

In the remaining patients there was a

pe-nod of 4% to 33 months between our first

en-counter with the major lymphangioma and

the appearance of tile clinical difficulties

due to the ectasias on tue tongue.

TREATM ENT

Treatment in all patients was directed

first toward excision of the major lesion,

whether cervical or intra-oral. Tile single

patient (L.L.) requiring attention to the

sun-face lesions on the tongue may have gone

for some time without a major eruptive

problem had his tongue not been burned by

prior treatment.

Treatment of the tongue itself consisted

of two parts, once the major lymphangioma

was cared for: electrodesiccation of the

(4)

1 (GO.) (lays 9 mo Neck (left) yr 6 no No No No

(CA.) Syr 6 mo 6 yr S mo SuLgiottic 7 yr 4 mo No No No

S ((.O.) 6 yr S yr 3 mo Tongue .5 yr 1 no Only with lymphangioma No No

4 (LI,.) wk wk Neck (bilateral), submaxillary I I yr 7 mo Yes Yes Ye

5 (MA.) g uk 1 yr .5 mo Neck (bilateral), suhglottic S yr 6 mo Yes Yes Yes

S (K.1).) 7 mo 1 yr Neck (bilateral), suhglottie I I yr S mo Ye Ye, Yes

7 (KR.) 2 yr 6 no S yr 6 mo Neck (right), face, supraclavicular fossa Syr Yes Yes Yes

8 (BA.) 6wk 6 mo Bilateral, stibmandihular 4 yr Yes Yes Yes

TABLE I

HISTORY OF EIGHT CHILDREN WITh SYNDROME OF LINGUAL CAPILLARY

LYMPH-A NGIOMLYMPH-A COMPLICATED BY GIOSSITIS

Surface Fir.t See,, Leajon., a (a.c (age) P,oblent

(age)

Site of Major Lymphangioma Follow-up (age)

Partial Symplo-Macroglossia Glossec- malic lomy Lale

form, and amputation of a portion of the

tongue in some patients in order to reduce

it to intra-oral size (Table I). No

postopera-tive problems followed partial glosseetomy

but there was variable edema, infection,

pain and inability to eat following

electro-desiccation. Discussion of this aspect of care

would be more profitable after presentation

of the ease material.

Case 1, G.O.

CASE REPORTS

A 2-day-old white male was first seen

with a cystic hygroma involving the entire

left part of the neck from mandible to

clavi-ele and from the angle of the mouth to the

external auditory canal. Within the large

mass, smaller nodular masses were palpable.

On December 31, 1957, the cystic hygroma

was removed.

The child’s second admission in

Septem-ben, 1958, was necessary because of

multi-pie lesions on the surface of the tongue and

an inguinal hernia. The hernia was repaired,

and many of the larger lymphangiectasias

on the dorsal aspect of the tongue were

electrodesiccated.

Only rarely did vesicles appear afterward,

and they were not symptomatic.

Case 2, C.A.

A 3%-year-old white male was first seen

because, 1 month before, the father had

noticed a small lump under the right side

of the child’s tongue. At the time of

admis-sion, the tongue was enlarged so that it

pro-truded from the mouth about 1.5 cm. A

large cystic mass was found on the floor of

the mouth beneath the tongue. On February

11, 1953, the cystic mass was removed and

the anterior 2 cm of the tongue was excised.

Histologic diagnosis was lymphangioma.

The child’s second admission in May,

1956, was necessary because areas of

vesi-des and bleeding papillary lesions had

ap-peared on the surface of the tongue and

were painful. These were electrodesiceated.

The child had no recurrence of the

erup-tive phenomenon, and speech remained

normal, although the tongue was slightly

deformed and all teeth were carious.

Case 3, CO.

A 6-year-old white female was first seen

with swelling of the tongue that had been

present since birth. Three years before she

bit her tongue, which bled profusely. Since

then the lesions had been asymptomatic but

had increased gradually in size. At the time

of her admission to the Children’s Hospital,

a blue nontender mass was present on the

left lateral surface of the tongue anteriorly.

A few vesieles were asymptomatic.

In May, 1955, an elliptical incision was

made about the lesion through normal

tonque tissue, and by sharp dissection the

entire tumor mass was resected. Histologic

diagnosis was lymphangioma. A smaller

le-sion posterior to the first was excised in

January, 1960.

Speech was very poor, but the child is

(5)

804

tongue later occurred only with respiratory

infections and were not painful. The teeth

were very earious.

Case 4, L.L.

A 2-week-old white male infant was

ad-mitted because of respiratory difficulty

as-sociated with swelling of the tongue. At the

time of birth two small firm swellings were

noted beneath the mandible, and the tongue

was thought to be too large. There were

small lesions which, in retrospect, may be

described as typical vesicles treated by local

caustic applications. A burn of the tongue

produced by the medication had resulted in

secondary infection and a cellulitis leading

to swelling of the tongue and difficulty in

respiration. At the time of admission the

child’s mouth was open and from it

pro-truded a purple, swollen tongue.

Surround-ing the bilateral neck mass there was

obvi-ous edema and inflammatory change. The

temperature was 101.6#{176}F (38.7#{176}C).

Following subsidence of tile infection and

edema on September 8, 1948, a

multi!ocu-lated lymphangioma extending back to the

angles of the jaw bilaterally and well

around to the posterior triangle of the neck

was excised except for some small locules

which extended to the base of the tongue.

Histologic diagnosis was lymphangioma

in-volving the submaxillary gland.

On November 17, 1948, four radon seeds

were inserted through the tongue into the

areas of remaining lymphangioma.

Addi-tional admissions in March, 1949; April,

1949; November, 1950; January, 1951;

Feb-ruary, 1952; and September, 1959, were

necessitated because of an enlarged tongue

with acute inflammatory change and pain

on eating. On these occasions

electrodesic-cation of vesicles and papillary hemorrhagic

lesions was performed. At times this child

was almost completely free of lesions on the

dorsum of the tongue and at times he was

completely asymptomatic. His exacerbations

were triggered usually by acute

upper-re-spiratory infections, although ingestion of

some foods, especially salty ones, produced

painful episodes of several days.

Case 5, M.A.

A 2-week-old white female was seen with a mass under her tongue and bilateral cystic

hygromas present since birth. At the time

of admission the tongue was pushed

cephalad, causing difficulty in feeding and

excessive salivation that tile patient found

difficult to swallow. On January 7, 1952, a

sublingual incision was made and the mass

between the tongue and tile floor of the

mouth was removed; tnacheostomy was

pen-formed because of tile considerable

respir-atory difficulty. Histologic diagnosis was

cavernous lymphangioma.

The cervical mass on the left was

ne-moved in January, 1952, and the mass on

the right, 14 days later, each by radical

ap-proach to the appropriate side of the neck.

Histologic diagnosis was similar to that of

the subglottic lesion.

The child was admitted again in July,

1953, to undergo direct ianvngoscopy before

the tracheostomy tube was removed. This

examination revealed considerable

involve-ment of tile larynx and epiglottic folds with

lymphangioma. These areas were treated by

electrodesiccation, and tile tracheostomy

tube was left in place to be removed finally

in September, 1953.

In January, 1955, a lymphangiorna was

removed from the submental area. In

Feb-ruary of the same year electrodesiccation

was performed on the dorsum of the tongue

because many of the vesieles had ruptured

spontaneously and had left behind tiny

blood-filled cystic areas which were painful.

In May, 1958, the child was admitted again

because of an acute glossitis, a

tempera-tune of 105#{176}F(40.6#{176}C) and inability to close

the mouth because of swelling of the

tongue.

Final admission took place in June, 1958,

when a partial glossectomy was carried out.

Speech remained difficult to understand;

there was deformity of the tongue from

scarring and prominence of the mandible

in reference to the other structures of the

face.

The youngster had very bad dentition;

(6)

crusty foods were difficult to eat because

they could not be placed properly on the

tongue. Exacerbations of the eruptions on

the surface of the tongue were occasioned

usually by upper-respiratory infections.

Case 6, K.O.

A 7-month-old white female was noted

at the time of delivery to have a swollen

tongue that interfered somewhat with

respi-ration and necessitated the use of oxygen.

From then until time of admission there had

been recurrent swelling of both sides of the

neck involving the region of the angle of the

jaw as well as the floor of the mouth. The

size of the swelling was inconstant, and

before she had been seen by us the

diag-nosis of cavernous hemangioma had been

entertained and three x-ray treatments had

been given without beneficial result.

At the time of admission she had a short,

fat neck and gross swelling over the angle

of the left jaw. A single, large, freely

mov-able mass in the right anterior cervical

tn-angle and a large mass below the ramus of

the right jaw were prominent findings. The

floor of the mouth was irregular, and the

buccal mucosa, swollen. Tile tongue was

covered over its anterior surface with

yes-ides and papillomatous lesions.

In March, 1949, tile tumor mass was

cx-posed in the neck, and the right half of the

cervical lesion was excised. Tile histologic

diagnosis was lymphangioma. Tile second

admission, in April, resulted in excision of

tile mass on tile left side of the neck, and it

was clear from this dissection that the lesion

there was contiguous with tile one in the

floor of the mouth. Tile postoperative course

was stormy, and on the evening of the

op-erative day tile patient became cyanotic and

was in status epilepticus for several hours.

Convulsions were finally controlled with

intravenously given sodium pentothal, and

there were no sequelae to this episode.

On the third admission, in May, the

tongue was enlarged and protruded from

the mouth, with painful papillary eruptions

over its inferior surface. The oral cavity

ap-peared to be almost completely filled by the

tongue. A large cystic mass was excised

from the base of the left side of the tongue.

This lesion was vell encapsulated and did

not extend into the adjacent tissues of the

floor of the mouth. Although the procedure

reduced substantially the size of tile tongue,

postoperative edema caused the tongue to

swell remarkably and protrude from the

mouth by the next day. The color became

deeply cyanotic, but there was no

obstrue-tion to the respiratory tract. Edema had

completely subsided in 6 days to permit

taking food by mouth.

In October, the child fell and struck her

chin on the floor. Shortly thereafter the

tongue began to swell, and on admission

the next day tile tongue was protruding

be-yond the tips of tile teeth approximately 4

em. It was dark purple in color and dry.

Final admission to the hospital was for a

partial glossectomy which was carried out

in September, 1950, when the anterior half

of the tongue was excised with wedging

and construction of a reasonably pointed

tongue that fit the oral cavity.

With subsequent scarring the tongue

be-came quite deformed and still had a

num-her of ectasias and papillomatous lesions

over its dorsal surface. However, speech

be-came quite good, except for some difficulty

in tile sounds of “s” and “1”, but the child

progressed well in school. She was not

troubled with pain even when tile eruptive

lesions were prominent.

Case 7, K.R.

This 23-year-old white female was first

seen with Il enlarged tongue wilich had

been asymptomatic until tile time of the

initial consultation. The child had been 6

weeks premature and weighed 5 lb 8 oz

(2,495 gm). At the time of admission,

the

tongue was enlarged so that it protruded

from the mouth about 2.5 cm and was

covered with numerous clear lymphatic

ectasias. There was a cystic hygroma

in-volving tile entire right side of the neck

cx-tending from the angle of the jaw anteriorly

and downward to the supraclavicular space.

(7)

the floor of the mouth or in the buccal

mueosa.

On June 2, 1952, under general

endo-tracheal anesthesia, the hygroma was

re-moved; it was somewhat difficult to

cx-tirpate completely because it extended

be-tween the esophagus and trachea and also

was media! and posterior to the carotid

sheath. Following extirpation of the cervical

mass, the vesieles along the periphery of the

tongue were electrodesiecated. Histologic

diagnosis of the cervical mass was

lym-phangioma.

A second admission, in May, 1953, was

necessitated because many of the vesicles

which had ruptured spontaneously left

be-hind tiny blood-filled cystic areas that were

painful and prevented adequate chewing

and ingestion of food.

Additional admissions for

electrodesicea-tion of painful areas occurred in January,

1954; December, 1954; December, 1955;

October, 1956; and November, 1956. There

were times when the child was completely

free of these hemorrhagic excrescences on

the tongue and was periodically completely

asymptomatic. On other occasions there

would be swelling of the entire tongue with

temperatures up to 102.6#{176}F (39.2#{176}C).

Po-tato chips and certain soft drinks were the

only foods associated with exacerbations.

On the last of these admission nine radon

seeds were implanted into the tongue to

give approximately 500 r throughout the

an-tenor two-thirds of that organ.

By April, 1958, the tongue was so

en-larged that speech was difficult, and the

portion of the tongue that extruded from

the mouth under normal circumstances

be-came so dried out during sleep that it

cracked and bled. A partial glossectomy was

carried out, consisting of excision of the end

of the tongue that protruded beyond the

teeth as well as the excision of a wedge of

tongue in the longitudinal axis. Histologic

diagnosis was lymphangioma of the tongue

with chronic inflammation.

Following glossectomy the child’s speech

improved, there were fewer surface

erup-tions, the occasions of swelling and pain in

eating were fewer, and the remissions

be-tween such painful episodes were longer.

However, the youngster sti!! had some very

difficult times, but, with the knowledge that

subsidence follows, eieetrodesieeation has

been avoided.

Case 8, BA.

A 6-week-old white male was first seen

with asymptomatic bilateral submandibular

masses and macroglossia present since birth.

At the time of admission the tongue was not

only enlarged but covered with numerous

clear vesieles. On December 10, 1955, the

hygroma was removed and biopsy of the

tongue was performed. Histologic diagnosis

was lymphangioma involving tile

submaxil-lary glands and a diffuse capillary angioma

involving the tongue. A second admission,

in July, 1956, was an elective one for the

insertion of eight radon seeds. Additional

admissions in November, 1956; December, 1956; and May, 1957, were necessitated by

acute inflammatory episodes with

tempera-tures as high as 105#{176}F(40.6#{176}C), painful

eat-ing and swelling of the tongue, sufficient on

one occasion to embarrass respiration.

On August 3, 1957, fulguration of the

tongue was carried out; on November 27,

under general endotraeheai anesthesia,

par-tial glosseetomy was performed.

In April, 1958, the tongue was improved

and excision of a lymphangioma from the

left neck and marsupialization of a left

buc-cal cyst were carried out.

This child had episodes of complete

free-dom from lesions on the surface of the

tongue; most exacerbations were produced

by

respiratory infections, which required

hospitalization on several occasions.

PATHOLOGY

The basic histologic lesion is a collection

of vascular spaces (Fig. 5), dilated and

bloodless, lying not only between muscle

fibers but extending to the mucosal surface.

The stroma surrounding some of these

spaces is quite fibrous (Fig. 6).

An inflammatory exudate is seen (Fig. 7)

(8)

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Fic. 5. Vascular spaces, dilated and bloodless, with extension to mucosal

surface (upper left). (x25)

Frc. 6. Fibrous stroma surrounding dilated lymphatics. (x 100)

of fungiform papilla is involved by the

lymphangiomatous lesion associated with

in-flammation and hemorrhage (Fig. 8). Focal

hemorrhage and inflammatory exudate is

seen in other areas (Fig. 9).

Thrombosis in small vessels is common,

and occasionally an organized thrombus

with focal calcification and recanalization

may be identified (Fig. 10).

It was the consensus of the several

pathol-ogists who worked with this material that

all lesions were pure lymphangioma and

that they did not represent tumors of mixed

cellular origin.

COMMENT

The children whose tongue difficulties

have been described here have other

stig-mata seen elsewhere with extensive

!ym-phangioma: overgrowth of adjacent

struc-tures and destruction of tissue. The four

(9)

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mandibles visibly out of proportion to their

maxillary development (Fig. 4). All the

children have carious teeth (Fig. 2), four

having almost complete destruction of the

first teeth and multiple cavities in their

permanent teeth. One child has had partial

tipper and lower dentures since the age of

8. Presumably the canious teeth result from

changes produced in the tooth buds by the

iymphangiomata.

The effects of partial glosseetomy have

brought an extension of time intervals

be-tween painful exacerbations that seem

greater than that to be expected from

re-moving the affected tissue alone. The

an-tenor portion of the tongue is most altered

by the eruptive phenomena, and anterior

resection would, therefore, be expected to

be helpful. However, one wonders whether

postoperative scarring might not also play a

#{182}5

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. . , .,

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-Fic. 8 Lymphangiomatous involvement of the lamina propria of

(10)

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Several of tile children had radon seed

implantation in the early years of our

cx-perienee in hope that scarring might

pre-vent the continued eruption of the vesicles

on the surface of the tongue. Unfortunately

no beneficial effect can be attributed to this

therapy.

Electrodesiccation of the hemorrhagic

lymphangiectasias seem to be followed on

each occasion by a progressively more

seni-- 9

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Fic. 9. Focal hemorrhage and inflammatory infiltrate associated with

numerous lymphangiectatic spaces. (x 100)

role in the apparent improvement.

No explanation is obvious for the severity

of the pain experienced by some and not by

others. Although emotional differences in

personality may be partially responsible, it

did

not seem to us that it was a major factor

in these eilildnen who learned early to live

with their difficulties and impressed all who

eared for them with their rather stoical

ac-ceptance of tile situation.

(11)

FIG. 11. (L.L.) Pre-adolescent boy, several years following routine glossectomy, well adjusted, and at this time asymptomatic in spite of peripheral

lesions.

oils reaction of the tongue as exemplified by

the edema, pain and systemic evidence of

inflammatory change. For this reason such

treatment is withheld as long as possible,

even though the end-result after the

im-mediate postoperative reaction has

sub-sided is very good. As the patients have

grown older, they have tended to select the

timing of desiccation, weighing the eventual

fresh start they have against the immediate

postoperative pain and discomfort.

The initiating role of a respiratory

infec-tion producing a painful exacerbation has

been uniform in all patients. Potato chips,

salted nuts and other salty foods were not

only very painful to two of the children

but produced a painful reaction for several

days after the insult. One child on several

occasions experienced a reaction similar to

that produced by salt when she ingested an

artificially flavored and colored grape

bever-age.

Obviously we have not found an

cmi-nently satisfactory method of management

in four of the patients. The other four are

quite well and suffer no impairment of usual

behavior. In the present state of our

know!-edge, partial glossectomy followed by

pen-odie eleetrodesiceation of the painful

hem-orrhagic lymphangiectasias provides an

ac-ceptable means of keeping these children

socially acceptable, speaking, attending

school and at times completely free of pain

(Fig.

11).

SUMMARY

The eight children in this series had a

syndrome of lingual capillary

lymphangi-oma complicated by glossitis. Three with

major lymphangiomas of the tongue and

subglottie areas became symptom-free

fol-lowing surgical removal of the major lesion

and electrodesiccation of the hemorrhagic

lymphangiectasias on the tongue. Four of

the remaining five, all of whom had major

cystic hygromas of the neck, had partial

glossectomy; one of these became

asympto-matic. The four symptomatic children

pre-sented a recurrent syndrome of a painful

edematous tongue characterized by the

eruption on its surface of vesieles and

hemorrhagic papillary lesions that could be

relieved by electrodesiccation, which would

be immediately followed by glossitis, edema

and pain. Changes in the growth of the

mandible and dentition also occurred.

REFERENCES

1. Hodgson, C. H., et a!.: Hereditary hemorrhagic telangiectasia and pulmonary arteriovellous

fistula. New Engl.

J.

Mccl., 261:626, 1959.

2. Singleton, A. 0.: Congenital lymphatic diseases: lymphangiomata. Ann. Surg., 105:952, 1937. 3. Lierle, D. M. : Congenital lymphangiomatous

macroglossia vith cystic hygroma of the neck. Ann. Otol., 53:574, 1944.

4. Fuller, F. W., and Conway, H. : Cystic livgrorna.

Surg., Gynec. Obstet., 108:457, 1959. 5. Perzik, S. L. : Early management in extensive

cervical cystic hvgroma and macroglossia. Arch. Surg., 80:460, 1960.

6. Goetsch, E. : Hygroma colli cysticum and hygroma axillare. Arch. Surg., 36:394, 1938.

7. Harkins, C. A., and Sabiston, D. C. : Lympban-gioma in infancy and childhood. Surgery, 47:

(12)

1961;27;800

Pediatrics

C. Everett Koop and Elias A. Moschakis

GLOSSITIS

CAPILLARY LYMPHANGIOMA OF THE TONGUE COMPLICATED BY

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1961;27;800

Pediatrics

C. Everett Koop and Elias A. Moschakis

GLOSSITIS

CAPILLARY LYMPHANGIOMA OF THE TONGUE COMPLICATED BY

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