Clinical review of 580 ranulas

Yi-Fang Zhao, DDS, MSc,

a
YuLin Jia, DDS, MSc,
b
Xin-Ming Chen, MD,
c
and
Wen-Fen Zhang, DDS, MSc,
a
Wuhan, Peoples Republic of China
WUHAN UNIVERSITY
Objective. The purpose of this paper was to compare clinical features among 3 patterns of ranula and the recurrence rates
of each when treated by different surgical methods.
Methods. A retrospective review of clinical and pathologic records in 580 ranulas was undertaken. Ranulas were
classied into 3 clinical types according to sites of the primary swelling: oral ranula, plunging ranula, and mixed ranula.
Information was collected on age at presentation, sex, history of onset, sites of swelling, surgical methods, histological
ndings, and outcome of treatment.
Results. Ranula was most prevalent in the second decade of life and slightly more common in females (male to female
ratio of 1:1.2), but a distinct male predilection was noted for the plunging ranula (male to female ratio of 1:0.74). Oral
ranula was most commonly involved in the left side (left to right ratio of 1:0.62), while the plunging and mixed ranula
were commonly involved in the right side (left to right ratio of 1:1.38, 1:1.16 respectively). In the plunging ranula group,
there were more patients who had the history more than 6 months. The recurrence rates of ranulas were not related to
swelling patterns and surgical approaches, but intimately related to the methods of surgical procedures. The recurrent
rates for marsupialization, excision of ranula, and excision of the sublingual gland or gland combined with lesion were
66.67%, 57.69%, and 1.20%, respectively.
Conclusion. Three patterns of ranula have similar clinical and histopathologic ndings, although plunging ranula has
some different clinical features. Removal of the sublingual gland via an intraoral approach is necessary in the
management of various clinical patterns of the ranula. Recurrence rates of ranulas of any type are excessive unless the
involved sublingual gland is removed.
(Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;98:281-87)
Ranulas develop from extravasation of mucous after
trauma to the sublingual gland or obstruction of the
ducts.
1,2
It typically has a bluish appearance that is
compared to a frogs belly, hence the term ranula. When
it is a cervical swelling or mass without a prominent oral
swelling, the ranula may be misdiagnosed as thyro-
glossal duct cyst, dermoid or epidermoid cysts, vascular
malformations, and even submandibular sialoadenitis.
Clinicians have used several different methods of treat-
ment for ranulas. These include excision of the ranula via
either an intraoral or cervical approach, marsupiali-
zation, intraoral excision of the sublingual gland and
drainage of the lesion, and excision of the lesion and
sublingual gland. Despite these treatments, many
patients have experienced recurrence and sometimes
larger lesions have occurred, eg, conversion of an oral
ranula into a plunging (cervical) ranula.
3,4
This study
reports clinicopathological features of 3 clinical cat-
egories of ranula and the results of several surgical
methods for management of ranulas.
MATERIALS AND METHODS
This study was based on the relevant clinical details
and pathology reports from the patients records and
follow-up data. Our materials consisted of 606 cysts in
571 patients with ranula treated in the Department of
Oral and Maxillofacial Surgery at the Hospital of
Stomatology, Wuhan University, China, between the
years 1962 and 2002. All patients were treated surgi-
cally. Marsupialization was performed by excising the
superior wall of the lesion and suturing the inner wall to
the mucosa of the oor of the mouth. Some ranulas were
excised via an intraoral or extraoral approach. Excision
of oral ranula was accomplished by making a linear or
elliptical incision along the superior aspect of the ranula,
medial to the plica sublingularis. Excision of plunging
ranula was performed by a routine submandibular
incision. The ranula was freed by blunt dissection from
the adjacent structures including the sublingual gland.
When excision of the sublingual gland was chosen for
a
Professor, Department of Oral and Maxillofacial Surgery, College &
Hospital of Stomatology, Wuhan University Wuhan, Peoples
Republic of China.
b
Oral Surgeon, Department of Oral and Maxillofacial Surgery,
College & Hospital of Stomatology, Wuhan University Wuhan,
Peoples Republic of China.
c
Clinical professor, Department of Oral Pathology, College &
Hospital of Stomatology, Wuhan University Wuhan, Peoples
Republic of China.
Received for publication Oct 17, 2003; returned for revision Dec 2,
2003; accepted for publication Jan 28, 2004.
1079-2104/$ - see front matter
2004 Elsevier Inc. All rights reserved.
doi:10.1016/j.tripleo.2004.01.013
281
management of ranula, the cyst was only evacuated
through the surgical eld with no intention to dissect the
cyst wall. The majority of ranulas were surgically treated
by staff oral surgeons, whereas a few surgeries were
performed by junior surgeons under guidance of senior
oral surgeons. Tissues obtained at surgery were rou-
tinely sectioned and stained with hematoxylin-eosin for
microscopic examination. Sections were reviewed by 2
senior pathologists.
The age at presentation of ranula, course of the lesion,
location of the swelling, methods of surgical treatment,
and pathological ndings were reviewed. Lesions were
classied into 3 clinical types according sites of the
primary swelling: oral ranula (intraoral swelling only),
plunging ranula (submandibular and/or submental
swelling without intraoral swelling), and mixed ranula
(intraoral and extraoral swelling). Clinical features were
compared among 3 patterns of ranula. All patients were
followed up postoperatively and they were contacted by
mail or telephone for failure to return. The follow-up
period of patients who had follow-up data ranged from 6
months to 26 years. Recurrence rates were analyzed and
compared in viewof the site of involvement and methods
of treatment. Clinical observation, diagnosis, and follow-
up were made by staff oral surgeons. The chi-square test
was used to assess the signicance of each variable
(SPSS Inc, Ver. 11.0, Chicago, Ill).
RESULTS
The ages of 571 patients at diagnosis are shown in
Fig 1. The youngest patient was 3 months old and the
oldest 80 years, with a peak frequency within the second
decade. There was a similar percentage among the
patients with 3 clinical patterns of ranulas. The gender
distribution was 260 males (45.53%) and 311 females
(56.47%), with a slight predilection of females. How-
ever, a distinct male predilection was noted for the
plunging ranula, with a 1 to 0.73 ratio (Table I), and the
difference was highly signicant between oral and
plunging ranulas (P\.01). All patients were Chinese.
Five hundred and seventy-one patients had 580 cysts.
Three hundred and twenty-four cysts occurred in the left
and 256 in the right side. The plunging ranula was most
commonly involved in the right side (Table II). The
difference was highly signicant between oral and
plunging ranulas (P\.01) and signicant between oral
and mixed ranulas (P\.05). There were 9 bilateral
ranulas, in which 3 cases of ranula occurred simulta-
neously and 6 lesions occurred in the opposite side 4
months to 2 years and 7 months, respectively, after
a ranula in the involved side was treated.
Most of patients with oral ranula presented with
a gradually enlarging swelling of the oor of the mouth.
The swellings were round or oval and uctuant (Fig 2).
When it was signicantly large, ranula produced
Fig 1. Age distribution of 571 patients with ranula.
OOOOE
282 Zhao et al September 2004
deviation of the tongue and also crossed the midline. The
swelling was painless and did not change in size on
gustatory stimulations except in 13 cases, where there
were intermittent episodes of swelling of the sub-
mandibular gland on the same side during eating. Most
lesions were present for only a few days to 3 weeks, but
some patients had them for months or even years before
seeking treatment. In the plunging ranula group, there
were more patients who had the history more than 6
months (Table III); the difference was highly signi-
cant compared with oral and mixed ranulas (P\.01).
Nineteen patients had a complaint of pain and rapidly
increasedswelling following aspiration of ranula. Twenty-
four oral swellings drained spontaneously at intervals
but at no time completely disappeared. Seventeen pa-
tients were able to relate the development of the cyst
to trauma.
The majority of them were 2 to 3 cm in diameter
but a few mixed or plunging ranulas extended from
the oor of the mouth into the submandibular space,
parapharyngeal space posteriorly, or even into the
carotid triangle inferiorly. Intraoral lesions were blue
and uctuant whereas plunging lesions were the color of
normal mucosa or skin. The plunging ranula typically
manifests as a soft, painless, and nonmobile swelling in
the neck (Fig 3). The mixed ranula had both intraoral and
extraoral swellings (Fig 4), usually intraoral swelling
was found earlier than cervical lesion. Oral and mixed
ranulas gave few diagnostic problems but plunging
lesions could be confused with lymphatic malformations
in 7 cases, venous malformations in 2, and thyroglossal
tract cyst in 1. Preoperative aspiration showed viscous
colorless liquid or viscous amber-colored uid in 63
cases and aspiration got over 50 mL viscous uid in 2
cases.
Surgery was performed under local or general
anesthesia. Five hundred and seventy-one patients had
606 procedures, in which 580 were for primary lesions
and 26 for recurrent cysts. Of 606 procedures, there were
28 excisions of the ranula, 9 marsupializations, 356
excisions of the sublingual gland, and 213 excisions of
the gland in combination with ranula. The majority of
procedures were performed through the mucosa of the
oor of the mouth (intraoral approach), but the access of
surgical explorations was through the submandibular
incision in 58 plunging or mixed ranulas. When the
lesion was operated via the submandibular approach, it
was often found that on elevation of the platysma
muscle, there was a thin-walled cyst intimately associ-
ated with the anterior portion of the submandibular
gland. However, further dissection showed that the lesion
was in connection with the sublingual gland (Fig 5). Of
58 lesions treated via the submandibular incision, the
cyst was resected in 3 cases, in continuity with the
sublingual gland in 36, and with the submandibular and
sublingual glands in 19 cases.
Four hundred and fteen patients (450 cysts) obtained
more than 6 months of follow-up data and recurrence
was found in 21 cases after the rst treatment. When we
considered each procedure as 1 operation, the recurrence
rates for marsupialization, excision of the ranula, and
excision of the sublingual gland or excision of the gland
combined with lesion were 66.67%, 57.69%, and 1.20%,
respectively (Table IV). Ranulas treated by simple
excision or marsupialization had a signicantly higher
recurrence rate than those of ranulas treated by excision
Table I. Sex distribution of 571 patients with ranula
Patterns Case no. Male Female M:F ratio
Oral 387 161 226 1:1.40
Plunging 118 68 50 1:0.74
Mixed 66 31 35 1:1.13
Total 571 260 311 1:1.20
Table II. Site distribution of 580 ranulas
Patterns Case no. Left Right L:R ratio
Oral 394 243 151 1:0.62
Plunging 119 50 69 1:1.38
Mixed 67 31 36 1:1.16
Total 580 324 256 1:0.79 Fig 2. Clinical photograph of oral ranula presented as an oval
swelling of the right oor of mouth.
Table III. Comparison of the history among 3 clinical
patterns of ranulas
Patterns Case no. #6 months >6 months
Oral 394 334(84.77) 60(15.23)
Plunging 119 76(63.87) 43(36.13)
Mixed 67 47(70.15) 20(19.85)
Total 580 457(78.79) 123(21.21)
OOOOE
Volume 98, Number 3 Zhao et al 283
of the sublingual gland or removal of both the gland and
ranula (P\.01). The recurrence rates of ranulas were not
related to swelling patterns (P > .05), but intimately
related to the methods of surgical procedures (Tables IV,
V). Twenty-one patients with recurrent ranula had
a second operation, namely excision of ranula in 8 and
excision of the lesion combined with the sublingual
gland in 13 cases. Of these cases, 5 had a second re-
currence, in which 4 developed from excision of ranula
and 1 from excision of both the lesion and the sublingual
gland. The third operation in all recurrences involved
simultaneous excision of the lesion and the sublingual
gland. This treatment resulted in a cure.
The pathological examination revealed that the cyst
wall consisted of broconnective or granulation tissue,
usually with a scanty or minimal degree of chronic in-
ammatory inltration. The cyst-like space contained
mucus, histocytes, polymorphs, and lymphocytes. The
cystic cavity was occasionally lined with a small area of
ductal epithelium (Fig 6), which was observed in 11
lesions (1.82%). The adjacent salivary gland acini
showed some chronic inammatory changes and part
of their ducts were dilated. In a few cases, the sur-
rounding loose edematous stroma showed numerous
dilated, blood-lled vascular channels (Fig 7). The
histologic ndings were not signicantly different
between the oral and plunging or mixed ranula. In 19
submandibular glands excised, 13 had chronic inam-
matory features under microscope.
DISCUSSION
Obstruction of excretory ducts or extravasation and
subsequent accumulation of saliva from the sublingual
gland in the tissue are responsible for the formation of
ranulas.
1,2,5,6
Ranulas or mucoceles of the oor of the
mouth occur in approximately 5% of patients un-
dergoing submandibular duct relocation for the man-
agement of uncontrollable sialorrhea.
7
In our study, 17
patients (2.98%) demonstrated the history of trauma or
surgery in the oor of the mouth.
Clinically, the oral ranula, though they are generally
small to medium in size, displaces the tongue, and
interferes with oral function. Very large oral ranulas or
ranulas located in the area of the caruncula sublingualis
Fig 3. Plunging ranula showing the swelling in the right
submandibular region without evidence of intraoral involve-
ment.
Fig 4. Mixed ranula originating from the right sublingual
gland in 13-year-old boy, showing obvious swelling of oor of
mouth crossing midline (A) with involvement of the submental
and right submandibular regions (B).
OOOOE
284 Zhao et al September 2004
may lead to partial obstruction of the Wharton duct
resulting in submandibular swelling during eating. In
this study, obstructive symptoms were observed pre-
operatively in 16 patients, in whom 13 postoperative
specimens showed chronic inammation in the sub-
mandibular gland parenchyma. The formation of the
plunging ranula may originate from sublingual gland
mucus leakage in the deeper areas of the gland, and the
uid drainage inferior into the submandibular space as
a result of gravity. Therefore, the lesion less interferes
with function, and patients with the plunging ranula may
seek treatment later than the patients with oral ranula. It
is unclear why the plunging ranula more commonly
affects males more than females and the sublingual gland
in the right side more than in the left side.
The diagnosis of ranula is based principally on the
clinical examination and sometimes on computerized
tomographic or magnetic resonance imaging ndings for
the plunging lesion. When it is an isolated oral lesion, the
diagnosis is generally easily accomplished. The suspi-
cion of the mixed ranula is denitely increased if
evidence of a ranula has been seen intraorally with
cervical swelling. In our study, it was demonstrated that
there was a spectrum of clinical changes in the mixed
ranula over time and this ranula, in part at least,
developed from the oral ranula that was a lesion with
a longer history or recurrent lesion. However, when
ranulas present as a cervical swelling without an oral
component, differential diagnosis may be more difcult.
Other lesions that should be considered include
thyroglossal duct cysts, branchial cleft cysts, parathyroid
cysts, cervical thymic cysts, dermoid cysts, cystic
hygroma, and benign teratoma.
8
If there is doubt about
the diagnosis, aspiration of mucous from the lesion and
a laboratory determination of amylase content should
make the diagnosis of ranula obvious.
3
The plunging
ranula should be distinguished from a mucocle resulting
from the submandibular gland because of different
surgical treatment for them. In the case of the sub-
mandibular gland mucocele it is essential to excise the
lesion with the submandibular gland, and this is best
accomplished through use of a cervical approach.
9
In the
case of the plunging ranula, however, excision of the
sublingual gland and drainage of the cyst via an intraoral
approach is the approach of choice. It is impossible to
distinguish from them clinically. However, mucoceles
originating from the submandibular gland are extremely
rare. Anastassov et al
9
reviewed the English literature
and found only 5 such cases. Computerized tomography
and specically the presence of a so-called tail sign
are pathognomonic for the plunging ranula.
9-11
The sign
is absent in mucoceles originating in the submandibular
glands. The ndings during surgery are very important
for determining the origin of the lesion. When the
Fig 5. Recurrent plunging ranula. A, Recurrence occurred
after excision of the lesion via the left submandibular approach
eight months ago. B, Multiple-cystic lesion under the
platysmal muscle. The cyst connected with the deep portion
of the sublingual gland by a duct-like structure. No recurrence
ve years after excision of the ranula and the sublingual gland.
Table IV. Surgical methods and recurrence rates
Operations
Cyst
no.
Recurrence
no.
Recurrence
rate
Excision of ranula 26 15 57.69
Marsupialization 9 6 66.67
Excision of sublingual
gland
286 3 1.05
Excision of sublingual
gland and ranula
129 2 1.55
Total 450 26 5.78
Table V. Swelling patterns of ranulas and recurrence
rates
Patterns Cyst no. Recurrence no. Recurrence rate
Oral 306 21 6.86
Plunging 89 5 5.62
Mixed 55 0 0
Total 450 26 5.78
OOOOE
Volume 98, Number 3 Zhao et al 285
plunging or mixed ranula was operated via the extraoral
approach, we observed a ductlike extension between the
lesion and the sublingual gland at the posterior margin of
the mylohyoid muscle, suggesting the origin of the lesion
from the sublingual gland.
Surgery is the mainstay for management of ranula.
Simple marsupialization has fallen into disfavor primar-
ily because the failure rate has been anywhere from
61% to 89%.
7
Baurmash
12
believes that simple mar-
supialization of relatively large lesions ([1.5 cm), which
usually have their origin from the deeper portion of the
sublingual gland, is a procedure without a sound
foundation from an anatomic, mechanical, or histolog-
ical standpoint. A new ranula generally will develop
within 6 to 8 weeks. Marsupialization with packing the
cyst cavity may reduce recurrence of ranula.
12
Crysdale
et al
7
reported that the recurrence rate was 100% in cases
with incision and drainage, 61% in cases of simple
marsupialization, and 0% in case of excision of the
ranula with or without sublingual gland excision. Parekh
et al
3
reviewed 139 procedures in 89 cases of plunging
ranula; a recurrence rate of 70.5% was observed after
incision and drainage, 52.6% after marsupialization,
84.8% after excision of the lesion in the neck, 3.8% after
cervical excision of the lesion combined with excision
of the sublingual gland, and 0% after intraoral excision
of the sublingual gland and drainage of the cyst. Our
ndings also clearly conrm that marsupialization or
excision of the ranula has a high recurrence rate and are
not suitable treatments.
As ranulas are usually extravasation pseudocysts
developing after disruption of sublingual gland ele-
ments, many authors advocate that excision of the
ipsilateral sublingual gland is the management approach
of choice.
3,7,13,14
In the present study, it is demonstrated
that incomplete excision of the sublingual gland may
lead to recurrence in 5 cases. Four oral ranulas received
excision of ranula and the sublingual gland through
a transoral approach, and another plunging ranula
underwent excision of both through a submandibular
incision, but recurrence occurred 4 to 19 months after the
operation, respectively. The second operation found that
the sublingual gland incompletely removed was associ-
ated with the cyst, suggesting that the residual secretory
acini remain active after partial excision of the sublingual
gland.
Ichimura et al
15
treated 7 patients with a plunging
ranula. All patients underwent surgery via a cervical
approach. Although total sublingual gland excision was
not performed in 2 patients, no recurrence was observed
in any patient. They suggest that a cervical approach may
still be the method of choice for the rst operation or for
salvage surgery after recurrence subsequent to intraoral
procedure if there is no swelling of the oral oor. Mizuno
and Yamaguchi
16
suggest since a plunging ranula is due
to extravasation from the sublingual gland herniating
through the mylohyoid muscle, excision of the sub-
lingual gland followed by transoral drainage of the
plunging ranula is regarded as the best treatment. Our
results show that the intraoral excision of the offending
sublingual gland is a simple and curable procedure with
minimal potential complications for all plunging ranulas,
whereas the extraoral approach is a relatively destructive
procedure, which may result in skin scarring, and is
unacceptable.
CONCLUSION
Comparisons of clinical features and recurrence rates
among 3 patterns of ranula were documented, and the
results were analyzed. The plunging ranula has some
different clinical features from the oral or mixed ranula.
Fig 6. The part of the cyst lining was formed by a single or
double layer of ductal epithelial cells (hematoxylin-eosin stain,
original magnication 3 33).
Fig 7. A mucus-containing space lined brous connective
tissue or granulation tissue with various sizes of vascular
lumen (hematoxylin-eosin stain, original magnication 333).
OOOOE
286 Zhao et al September 2004
The recurrence rates of ranula were not related to
swelling patterns and surgical approaches, but intimately
related to the methods of surgical procedure. We advise
the removal of the sublingual gland combined with
intraoral evacuation of the ranula be used regardless of
the clinical patterns of the lesion (oral, plunging, or
mixed ranula).
REFERENCES
1. Catone GA, Merrill RG, Henny FA. Sublingual gland mucus-
escape phenomenontreatment by excision of sublingual gland.
J Oral Surg 1969;27:774-86.
2. Regezi JA, Sciubba JJ, editors. Oral pathology, clinical
pathologic correlations. 3rd ed. Philadelphia: WB Saunders
Company; 1999. p. 220-2.
3. Parekh D, Stewart M, Joseph C, Lawson HH. Plunging ranula: A
report of three cases and review of the literature. Br J Surg 1987;
74:307-9.
4. Yoshimura Y, Obara S, Kondoh T, Naitoh SI. A comparison of
three methods used for treatment of ranula. J Oral Maxillofac
Surg 1995;53:280-2.
5. Lida S, Kogo M, Tominaga G, Matsuya T. Plunging ranula as
a complication of intraoral removal of a submandibular sialolith.
Brit J Oral Maxillofac Surg 2001;39:214-6.
6. Balakrishnan A, Ford GR, Bailey CM. Plunging ranula following
bilateral submandibular duct transposition. J Laryngol Otol 1991;
105:667-9.
7. Crysdale WS, Mendelsohn JD, Conley S. Ranulas-mucoceles of
the oral cavity: experience in 26 children. Laryngoscope 1988;98:
296-8.
8. Batsakis JG, McClatchey KD. Cervical ranulas. Ann Otol Rhinol
Laryngol 1988;97(5 pt 1):561-2.
9. Anastassov GE, Haiavy J, Solodnik P, Lee H, Lumerman H.
Submandibular gland mucocele: diagnosis and management. Oral
Surg Oral Med Oral Pathol Oral Radiol Endod 2000;89:59-63.
10. Coit WE, Hamsberger RH, Osborn AG, Smoker WR, Stevens
MH, Lufkin RB. Ranula and their mimics: CT evaluation.
Radiology 1987;163:211-6.
11. Charnoff SK, Carter BL. Plunging ranula: CT diagnosis.
Radiology 1986;158:467-8.
12. Baurmash HD. Marsupialization for treatment of oral ranula:
a second look at the procedure. J Oral Maxillofac Surg 1992;50:
1274-9.
13. Bridger AG, Carter P, Bridger GP. Plunging ranula: literature
review and report of three cases. Aust NZ J Surg 1989;59:945-8.
14. de Visscher JG, van der Wal KG, de Vogel PL. The plunging
ranula: pathogenesis, diagnosis and management. J Craniomax-
illofac Surg 1989;17:82-5.
15. Ichimura K, Ohta Y, Tayama N. Surgical management of the
plunging ranula: a review of seven cases. J Laryngol Otol 1996;
110:554-6.
16. Mizuno A, Yamaguchi K. The plunging ranula. Int J Oral
Maxillofac Surg 1993;22:113-5.
Reprint requests:
Yi-Fang Zhao, DDS, MSc
Department of Oral and Maxillofacial Surgery
College & Hospital of Stomatology
Wuhan University
237 LuoYu Road
Wuhan, Peoples Republic of China 430079
yifang@public.wh.hb.cn
OOOOE
Volume 98, Number 3 Zhao et al 287