Case Studies PLUNGING NEII. E.I.
RANULA:
IANGLOIS.
The plunging gland. and
in
swelling.
i.5 (1 mucows ratrclz~rc.tntion iu
cyst of the .suhlingual
inJemc~le.s, shoua no .side pwfkwnce.
the .second ccnti third with
cold it mq
be leji to the wporting
The hi.stologic of rpithelium
co~~nectiz~e ti.csl;e wll tiol Jijr the most t$rtiur Hr:v
or endothelium,
contnining
and mar rvphage.s .stuffed with
~ppeur(~nce
P.~~‘HoI. 23: 1 X5-IXM.
ran ula
the clinicctl pathologi.st
I))
to gjvr
the
i.s chnr~L~t~,~-i.stirull~ with
CLvclsruln~inflammctto~~
fihrocel1.c
i.s ~.S.SFII-
i.s rscision
of the .subliugunl
Copyright
0
1992
of
by W.A.
Snu nders Corn/xc n?l
Ranutas are mucous retention cysts of the sublingual gland. Whereas the oral ranuta is relatively common and presents as a cyst in the mouth, the plunging ranula is rare and manifests itself as a mass in the neck with or without an associated oral lesion. If there is no intraoral component, clinical suspicion may be low and the pathologist may he misled by Recognition of the diagnosis of the histologic appearance.’ plunging ranuta is essential for the correct treatment of these lesions. We report a case of a 9-year-old girt and review the English literature to discuss the epidemiology and pathology of these lesions. CASE
REVIEW
where ;I ttissectiou of the cyst re~xdect it 10 lx attached to the suhrn~undit,t~l~ir &d. The cyst watt with the adherent salivar\~ gland W:I~ excised and terra&line powder installed into the cavity to ohtileratc it. At rev’iew 4 months later the swelling had I-rcurrecl agairi.
diqno.sis
Thr correct dmgnosi.s
which
It
ng in the rwk
au intrwrtll
.somr chronic
m rtcin.
treatment.
dec~1e.s oj’ l{fe.
.swlli
If therr is no hi,story oJ an orctl rclnula diqnosis.
AND A LITERATURE
MA, AND PKALHAI) KOI.HE, FRCS
fburofjizw ems i.s ccssociated
o cyst. dezloid
gland.
REPORT
mt~ n{fr.st.c MS o pnin1e.s.s. nonmobile
is di’cultl rorrrrt
MB BCHIK,
more common
is more prel&ent
t~pirull~ and
runula
It i.s .tlightly
A CASE
REPORT
A Svear-old girt complaining of a large cystic swelling in the left side of her neck was referred to a dental surgeon h) her general practitioner in May 1990. No dental pathotoby was identified and the patienr was referred to the ear, nose, and throat department. The swelling had been present fo1 approximately 3 months; it exhibited no change with eating, chewing, or swallowing. There had been no discharge. On examination a nontender, diffuse, cystic mass could he seen and felt in the left submandibular area. The tentative diagnosis of hranchiat cyst or cystic hygroma was made. In October 1990. the patient underwent a left neck exploration, A muttitobutated cystic lesion, deep to the ptatysma and wrapped around the submandibular gland. was discovered. The cyst extended into the floor of the mouth. lateral pharyngeal watt, and tonsils, reaching the skull base, where the internal carotid artery and stytoid process were palpable. The cyst ruptured during dissection, but as much as possible was excised. The diagnosis of plunging ranuta was made on pathologic grounds. By December 1990, the swelling had recurred (Fig 1). The patient was referred to the department of plastic surgery From the Department of Pathology, University of Aberdeen, Aberdeen, Scotland; and the Department of Surgery, Aberdeen Royal Infirmary, Aberdeen, Scotland. Accepted for publication June 16. 1992. Rq words: ranula. plunging. cervical. Address cot-respondence and reprint requests to Neil E.I. Langlois, MB BChir. MA, Department of Pathology, University Medical Buildings, Foresterhill, Aberdeen AB9 2ZD. Scotland. Copyright 0 1992 by W.B. Saunders Compaq 0046~8177/92/2X1 l-0018$5.00/0
1306
PATHO1,0(;1 ‘t‘hc specinien receivect consisted of an irregular portion of tissue, one surface of which !+as smoorh, suggesting it was ai1 otmiecl cyst. Histologic csaniination confirmecl the presence of a cystic structure. The w~dl was colnposect of loose. cascular-, fibroconnec~ive tissue rictl in connective tissue mucin (Fig 2). On the inner surface were niacrophages containing mucin that stained positivetv with mucicarmine and diast;~s~-digeste~t ptlriodic acid-Schift. (Fig 3). Else of i~r~munohisto(.tle~~~isrI-y (XItibodies to CAM 5.2. AE I /AE3. and factor VIII) ctemonsrrated the absence c’f an endothetiat or epithetiat lining. Within the outer cvst watt portions of a mixed (mutinous anct serous) salivary gta~id wel-e presenr. DISCL’SSION I0 ttiv oral randa, the t~tunging rxiiit;i is a11 lesion. To manifest as a IKISS in the neck without ;m oral lesion. AS in this case, is even more I-are.‘The difttrential clinical diagnosis includes I!Illphadenopathy. abscess, thyrogtossat dust cyst, dermoict or epidel-maid cyst, taryngocete, tipoma. hemangioma. cervical thymic cyst, cysts of the paralhyroid or thyroid gland. or Iumor.‘.” 4 review of the case reports of plunging ranutas in the English tirerature revealed a mate to female ses ratio of I : I .S and no significant side preference. Presentation was most co~n~non in the second and third decades of life. with an age range of 3’ IO 61 ’ years. Typically, the history was of a painless mass that did not vaq in size with eating, chewing, or swatlowing. However, pain was an occasional feature.‘.” Unusual presentations included rapid enlargement following infection” and varying size with the valsatva maneuver.’ Asynchronous involvement of both sides also has been reported.8 The majority of patients presented within 3 months of occurrence of the swelling, but the reported range was 24 hours” to I8 years.” The majority of masses were 4 to 10 cm in size and lay within the submandibular area of the neck, but extension to the submental region and both sides of the neck,‘“,” to the base of the skutt,“~” to the retropharynx,” to the clavicle,‘,” and even from both ears to the sternum” has been reported. The reported cases included comments concerning the intraoral appearance of the masses: 45% presented first with an oral swetting or ranula, 34% had an associated oral mass at presentation, and 21% had only a swelling in the neck. The histologic appearance is typically that of a cyst, devoid of epithetiat lining, with the wall composed of vascular fihroconnective tissue resembling granulation tissue with mucin and foamy macrophages. Partial epithetial lining, however, has been recorded.14 Batsakis and McCIatchey” note that the histologic appearance changes with time from a loose. vascutarized connective tissue with an abundance of mucin to a predominantly In cxbntrast
unco1nmon
CASE STUDIES
FIGUR !E 1. Profile of the patient showing the scar from tTer firs?- operation and the recurrence of the plunging ranula
FIGURE 2. A typical area from the plunging ranula showing the cystic space (center and left) bound by vascular connective tissue devoid of epithelial lining.
HUMAN PATHOLOGY
Volume 23, No. 11 (November
1992)
FIGURE 3. Higher-power view to demonstrate the foamy macrophages that line the cyst and stain positively with diastase digested periodic acidschiff
dense, well-\,asclllal-izecl
tissue with .t paucity ofntuciti. Plunging ranulas also have been associaLed with vascular tiialforniatior~s’” and dermoid cvsts.“’ From analysis of the sect&m within the ranula it appears that their origin is due to a extravasation of saliva from the sublingual gland forming a pseudocyst without epithelial lining.” It has been suggested that leakage may either be due to trauma,’ obstruction of the salivary duct,17 or salivary duct anomaly.” The plunging ranula may arise in the neck by three possible mechanisms: the sublingual gland tnay project through the mylohyoid” to provide an origin.‘” the cyst may penetrate through the mylohyoid to join the sublingual gland in the mouth,‘.‘” or a duct from the sublingual gland may join the submaxillary duct”‘~‘” and give rise to ranula in continuity with the submandibular gland or its duct.1’,2’ The latter explanation is favored in this case as the ranula was adherent to the submaxillary gland, but recurred after the gland was excised. Reviews of surgical management of this lesion underline the necessity of removal of the sublingual gland rather than the excision of the cyst itself. Typically, more than one operation is required to effect a cure, the highest success rate being achieved by excision of the sublingual gland.‘,‘2,2’.L’2 This case revealed several interesting features: the presentation was solely of a swelling in the neck, it was found to extend to the base of the skull, and it appeared to take its origin from the submandibular gland. However, the pathologic appearance was typical, which enabled the correct diagnosis. Thus, the plunging ranula is a pseudocyst of the sublingual gland and should be treated as such.
t 1. Hitchin AD: Raluta. Br Dent J XfJ::325-327, 1!btfi IL’. van den Akker HP, Bays RA. Becker- AK: Plunging or wrbicat I-anula: A review of the litcratwe and a’report of 4 caws. J Maxillofa Surg 6:286-29X 197X
17. Harrison salivary 18. I!). 20. Anatomy.
1. McClatrhey KI). Haley-AppelhlattN. %arho RJ. ct al: Piu,,gir,g I-arrula. Oral Surg Oral Med Oral Pathol 5i:1OX-4 I”, 198-l
1308
GRL:
11X:215-254.
Mylohyoid
boutoniere
of the
1975 and sublingual
bouton.
J Anat
I!163
RI-idger
rl qort
effrcts 01 ductal liptiun
JD, (;a~-rer JR: Hisrologicat
of the ~1. J Pathot
C:anghran
!l7:5ti5-568, mtl
REFERENCES
glands
AG, Carter
of three
Williams (37th
PL.
P. Br-idgrr
case% Aust Warwick
ed). Edinburgh.
C;P: Plunging
N % J Surg
R, Dyson UK.
rmuta:
59345.948.
Literature
M. et at (eds): Splanchnoto~~,
Churchill
Livingstone.
revieu
1989 19X9.
in Gray’s p I?93
ranuta: A repel-c of 21. Par&h D. Stewart M. Joseph C. er at: Plunging three casts and a review of the literature Rr J Surg 74:307-309. 19X7 22. Yoshimura Y, Sughara T, Matsuura R: Plunging nnuta: A report of two cases and P review of the tierature. Quintessence Int ?0:435-437. 1989
RANULA:
IANGLOIS.
The plunging gland. and
in
swelling.
i.5 (1 mucows ratrclz~rc.tntion iu
cyst of the .suhlingual
inJemc~le.s, shoua no .side pwfkwnce.
the .second ccnti third with
cold it mq
be leji to the wporting
The hi.stologic of rpithelium
co~~nectiz~e ti.csl;e wll tiol Jijr the most t$rtiur Hr:v
or endothelium,
contnining
and mar rvphage.s .stuffed with
~ppeur(~nce
P.~~‘HoI. 23: 1 X5-IXM.
ran ula
the clinicctl pathologi.st
I))
to gjvr
the
i.s chnr~L~t~,~-i.stirull~ with
CLvclsruln~inflammctto~~
fihrocel1.c
i.s ~.S.SFII-
i.s rscision
of the .subliugunl
Copyright
0
1992
of
by W.A.
Snu nders Corn/xc n?l
Ranutas are mucous retention cysts of the sublingual gland. Whereas the oral ranuta is relatively common and presents as a cyst in the mouth, the plunging ranula is rare and manifests itself as a mass in the neck with or without an associated oral lesion. If there is no intraoral component, clinical suspicion may be low and the pathologist may he misled by Recognition of the diagnosis of the histologic appearance.’ plunging ranuta is essential for the correct treatment of these lesions. We report a case of a 9-year-old girt and review the English literature to discuss the epidemiology and pathology of these lesions. CASE
REVIEW
where ;I ttissectiou of the cyst re~xdect it 10 lx attached to the suhrn~undit,t~l~ir &d. The cyst watt with the adherent salivar\~ gland W:I~ excised and terra&line powder installed into the cavity to ohtileratc it. At rev’iew 4 months later the swelling had I-rcurrecl agairi.
diqno.sis
Thr correct dmgnosi.s
which
It
ng in the rwk
au intrwrtll
.somr chronic
m rtcin.
treatment.
dec~1e.s oj’ l{fe.
.swlli
If therr is no hi,story oJ an orctl rclnula diqnosis.
AND A LITERATURE
MA, AND PKALHAI) KOI.HE, FRCS
fburofjizw ems i.s ccssociated
o cyst. dezloid
gland.
REPORT
mt~ n{fr.st.c MS o pnin1e.s.s. nonmobile
is di’cultl rorrrrt
MB BCHIK,
more common
is more prel&ent
t~pirull~ and
runula
It i.s .tlightly
A CASE
REPORT
A Svear-old girt complaining of a large cystic swelling in the left side of her neck was referred to a dental surgeon h) her general practitioner in May 1990. No dental pathotoby was identified and the patienr was referred to the ear, nose, and throat department. The swelling had been present fo1 approximately 3 months; it exhibited no change with eating, chewing, or swallowing. There had been no discharge. On examination a nontender, diffuse, cystic mass could he seen and felt in the left submandibular area. The tentative diagnosis of hranchiat cyst or cystic hygroma was made. In October 1990. the patient underwent a left neck exploration, A muttitobutated cystic lesion, deep to the ptatysma and wrapped around the submandibular gland. was discovered. The cyst extended into the floor of the mouth. lateral pharyngeal watt, and tonsils, reaching the skull base, where the internal carotid artery and stytoid process were palpable. The cyst ruptured during dissection, but as much as possible was excised. The diagnosis of plunging ranuta was made on pathologic grounds. By December 1990, the swelling had recurred (Fig 1). The patient was referred to the department of plastic surgery From the Department of Pathology, University of Aberdeen, Aberdeen, Scotland; and the Department of Surgery, Aberdeen Royal Infirmary, Aberdeen, Scotland. Accepted for publication June 16. 1992. Rq words: ranula. plunging. cervical. Address cot-respondence and reprint requests to Neil E.I. Langlois, MB BChir. MA, Department of Pathology, University Medical Buildings, Foresterhill, Aberdeen AB9 2ZD. Scotland. Copyright 0 1992 by W.B. Saunders Compaq 0046~8177/92/2X1 l-0018$5.00/0
1306
PATHO1,0(;1 ‘t‘hc specinien receivect consisted of an irregular portion of tissue, one surface of which !+as smoorh, suggesting it was ai1 otmiecl cyst. Histologic csaniination confirmecl the presence of a cystic structure. The w~dl was colnposect of loose. cascular-, fibroconnec~ive tissue rictl in connective tissue mucin (Fig 2). On the inner surface were niacrophages containing mucin that stained positivetv with mucicarmine and diast;~s~-digeste~t ptlriodic acid-Schift. (Fig 3). Else of i~r~munohisto(.tle~~~isrI-y (XItibodies to CAM 5.2. AE I /AE3. and factor VIII) ctemonsrrated the absence c’f an endothetiat or epithetiat lining. Within the outer cvst watt portions of a mixed (mutinous anct serous) salivary gta~id wel-e presenr. DISCL’SSION I0 ttiv oral randa, the t~tunging rxiiit;i is a11 lesion. To manifest as a IKISS in the neck without ;m oral lesion. AS in this case, is even more I-are.‘The difttrential clinical diagnosis includes I!Illphadenopathy. abscess, thyrogtossat dust cyst, dermoict or epidel-maid cyst, taryngocete, tipoma. hemangioma. cervical thymic cyst, cysts of the paralhyroid or thyroid gland. or Iumor.‘.” 4 review of the case reports of plunging ranutas in the English tirerature revealed a mate to female ses ratio of I : I .S and no significant side preference. Presentation was most co~n~non in the second and third decades of life. with an age range of 3’ IO 61 ’ years. Typically, the history was of a painless mass that did not vaq in size with eating, chewing, or swatlowing. However, pain was an occasional feature.‘.” Unusual presentations included rapid enlargement following infection” and varying size with the valsatva maneuver.’ Asynchronous involvement of both sides also has been reported.8 The majority of patients presented within 3 months of occurrence of the swelling, but the reported range was 24 hours” to I8 years.” The majority of masses were 4 to 10 cm in size and lay within the submandibular area of the neck, but extension to the submental region and both sides of the neck,‘“,” to the base of the skutt,“~” to the retropharynx,” to the clavicle,‘,” and even from both ears to the sternum” has been reported. The reported cases included comments concerning the intraoral appearance of the masses: 45% presented first with an oral swetting or ranula, 34% had an associated oral mass at presentation, and 21% had only a swelling in the neck. The histologic appearance is typically that of a cyst, devoid of epithetiat lining, with the wall composed of vascular fihroconnective tissue resembling granulation tissue with mucin and foamy macrophages. Partial epithetial lining, however, has been recorded.14 Batsakis and McCIatchey” note that the histologic appearance changes with time from a loose. vascutarized connective tissue with an abundance of mucin to a predominantly In cxbntrast
unco1nmon
CASE STUDIES
FIGUR !E 1. Profile of the patient showing the scar from tTer firs?- operation and the recurrence of the plunging ranula
FIGURE 2. A typical area from the plunging ranula showing the cystic space (center and left) bound by vascular connective tissue devoid of epithelial lining.
HUMAN PATHOLOGY
Volume 23, No. 11 (November
1992)
FIGURE 3. Higher-power view to demonstrate the foamy macrophages that line the cyst and stain positively with diastase digested periodic acidschiff
dense, well-\,asclllal-izecl
tissue with .t paucity ofntuciti. Plunging ranulas also have been associaLed with vascular tiialforniatior~s’” and dermoid cvsts.“’ From analysis of the sect&m within the ranula it appears that their origin is due to a extravasation of saliva from the sublingual gland forming a pseudocyst without epithelial lining.” It has been suggested that leakage may either be due to trauma,’ obstruction of the salivary duct,17 or salivary duct anomaly.” The plunging ranula may arise in the neck by three possible mechanisms: the sublingual gland tnay project through the mylohyoid” to provide an origin.‘” the cyst may penetrate through the mylohyoid to join the sublingual gland in the mouth,‘.‘” or a duct from the sublingual gland may join the submaxillary duct”‘~‘” and give rise to ranula in continuity with the submandibular gland or its duct.1’,2’ The latter explanation is favored in this case as the ranula was adherent to the submaxillary gland, but recurred after the gland was excised. Reviews of surgical management of this lesion underline the necessity of removal of the sublingual gland rather than the excision of the cyst itself. Typically, more than one operation is required to effect a cure, the highest success rate being achieved by excision of the sublingual gland.‘,‘2,2’.L’2 This case revealed several interesting features: the presentation was solely of a swelling in the neck, it was found to extend to the base of the skull, and it appeared to take its origin from the submandibular gland. However, the pathologic appearance was typical, which enabled the correct diagnosis. Thus, the plunging ranula is a pseudocyst of the sublingual gland and should be treated as such.
t 1. Hitchin AD: Raluta. Br Dent J XfJ::325-327, 1!btfi IL’. van den Akker HP, Bays RA. Becker- AK: Plunging or wrbicat I-anula: A review of the litcratwe and a’report of 4 caws. J Maxillofa Surg 6:286-29X 197X
17. Harrison salivary 18. I!). 20. Anatomy.
1. McClatrhey KI). Haley-AppelhlattN. %arho RJ. ct al: Piu,,gir,g I-arrula. Oral Surg Oral Med Oral Pathol 5i:1OX-4 I”, 198-l
1308
GRL:
11X:215-254.
Mylohyoid
boutoniere
of the
1975 and sublingual
bouton.
J Anat
I!163
RI-idger
rl qort
effrcts 01 ductal liptiun
JD, (;a~-rer JR: Hisrologicat
of the ~1. J Pathot
C:anghran
!l7:5ti5-568, mtl
REFERENCES
glands
AG, Carter
of three
Williams (37th
PL.
P. Br-idgrr
case% Aust Warwick
ed). Edinburgh.
C;P: Plunging
N % J Surg
R, Dyson UK.
rmuta:
59345.948.
Literature
M. et at (eds): Splanchnoto~~,
Churchill
Livingstone.
revieu
1989 19X9.
in Gray’s p I?93
ranuta: A repel-c of 21. Par&h D. Stewart M. Joseph C. er at: Plunging three casts and a review of the literature Rr J Surg 74:307-309. 19X7 22. Yoshimura Y, Sughara T, Matsuura R: Plunging nnuta: A report of two cases and P review of the tierature. Quintessence Int ?0:435-437. 1989
