Aust. N . Z . J . Surg. 1989.59.
945-948
PLUNGING RANULA: LITERATURE REVIEW AND REPORT OF
THREECASES
A. G . BRIDGER,' P.CARTER*
Department of Otolaryngology, Prince of Wales Hospital. Sydney, New South Wules
Three cases of plunging ranula are described and the literature is reviewed. In many cases, a plunging ranula is
iatrogenic and follows surgery to an oral ranula. In the cases presented, the cervical swelling was associated
with prolongations of sublingual gland into or through the mylohyoid muscle. All patients were cured by
partial or total excision of the sublingual gland.
Key words: extravasationpscudocyst, mylohyoid boutonnKre, ranula, retention cyst, sublingual gland.
Introduction
A ranula is commonly regarded as a simple reten-
tion cyst which occurs in the anterior floor of the
mouth. The name 'ranula' comes from the Latin
rana (frog) because the cystic swelling has a char-
acteristic distended translucent blue appearance,
somewhat akin to the underbelly of a frog. The
recommended treatment is either surgical excision
or marsupialization and is regarded as uncomplicated.
Rarely, the intra-oral lesion is accompanied by a
smooth, fluctuant cervical swelling in the submen-
tal or submaxillary areas, referred to as a plunging
ranula. This is usually caused by the extravasation
of mucus from the intra-oral lesion into the cervical
tissues to form a pseudocyst. This pseudocyst lacks
the epithelial lining, and its wall consists of an
encapsulating thin layer of fibrous tissue which
forms as a reaction to the extravasated mucus.
Roediger and Kay have postulated that a duct
anomaly may predispose to stasis of saliva in the
deep portion of the sublingual gland.' From this
region, extravasation of saliva into the contiguous
tissues is possible. In addition, Parekh et al. have
described ranula formation in association with
trauma and infection of the sublingual gland.' For
the treatment of this condition, it is now accepted
that the sublingual gland must be completely
removed.
Although the standard surgical textbooks
describe these treatments as straightforward, a
review of the literature reveals that complete ex-
cision of the sublingual gland can be difficult.
' Final year medical student. Visiting medical officer.
Correspondence: Dr G. P. Bridger. 1\21 Kitchener Parade,
Bankstown. NSW 2200. Australia.
Accepted for publication 17 M a y 1989.
945
A N D G . P. B R I m E R 2
Furthermore, the patterns of clinical presentation
and behaviour clearly demonstrate that argument
still exists regarding the aetiology of both the
simple oral and more complicted plunging ranulae.
Research and experience suggests that the typical
oral ranula may be either a retention cyst or an
extravasation pseudocyst, and that simple excision
or marsupialization might be not only unsuccessful
but could predispose to the formation of a plunging
ranula. This complication can, in some cases, be
directly connected to an anatomical anomaly where
sublingual glandular tissue herniates through dehis-
cences of the underlying mylohyoid muscle. On
very rare occasions, the cervical pseudocyst can be
the only presenting symptom.
The following three cases are presented to illustrate
the varied presentations and treatment difficulties.
Case histories
C A SE I
A 3 1 year old female presented in November 1984
with a typical blue dome cyst approximately
2cm X 1 cm in the left floor of the mouth. At
surgery, the distended mucosa and underlying
myxomatous matrix were excised widely. These
tissues contained no identifiable submucosal epith-
elial lining. The wound was left open to granulate,
but within 3 months the oral ranula had recurred. At
this stage, a sialogram of the left submandibular
gland was normal. The patient was reassessed by a
colleague who admitted her to hospital for a wide
excision of the ranula as well as the underlying
sublingual gland. The pathology showed chronic
low-grade sublingual sialadenitis and a granulating
cavity containing mucinous material. Within 3
weeks, the oral swelting had recurred. In August,
946
1985 the patient presented with an enlargement of
the oral ranula and an extensive cystic cervical
swelling extending into the left submandibular and
parapharyngeal tissues accompanied by severe ear-
ache and distress. which was relieved only by
incising the oral component of the ranula. Copious
amounts of mucus exuded from the incision. The
relief was temporary. lasting less than 24h. In
October 1985. a wide extirpation of the pseudocyst
and submandibular gland was carried out via a
cervical approach. Within I month the oral ranula
had returned. A further wide intra-oral excision of
all content5 down t o the mylohyoid muscle was
perforined. The pathology revealed a recurrent
pseudtxyst but no salivary tissue was identified.
The patient had complete remission of symptoms
until June 1986 when the oral swelling returned. A
similar intraoral re-excision was performed but. on
this occasion. il discrete mass of secreting salivary
tissue was identified. perforating the mylohyoid
muscle. This was removed and the wound allowed
to granulate. The patient has been symptom-free for
morc than 2 years.
CASE 2
A 28 year old female presented to the Prince of
Wales Hospital in October 1987 with a diffuse
swelling in the right upper cervical area. In August
of I986 and I987 she had undergone neck surgery
including excision of the right submandibular
gland. but her cystic swelling persisted. AI no stage
was there any intra-oral swelling. The patient was
diagnosed as having a plunging ranula without the
oral component. The cervical swelling was ex-
plored in January 1988. The pseudocyst was identi-
fied as arising frorn a projection of sublingual tissue
situated in a dehiscence of the mylohyoid muscle.
Only this caudal portion of the gland was excised
and no intra-oral surgery was performed. The
pathology showed a granulomatous pseudocyst and
sniall amounts of norinal salivary tissue. The
patient has remained symptom-free.
cask 3
A S9 gear old female in March 1987 noticed a non-
tender swelling in the right floor of the mouth. This
would rupture spontaneously and exude thick sali-
va. It was surgically drained in August and Septem-
ber. 1987. After this. the oral swelling resolved but
the patient developed a cystic mass in the right
submandibular and submental rcgians (Fig. 1 ). She
was then referred to the Prince of Wales Hospital
where a diagnosis of plunging ranula was made. In
February 1988. using a cervical approach. the
pscudocyst was exposed and found to be continu-
ous with a dcep portion o f the sublingual gland
which had herniated through a defect in the mylo-
BRIDGER ET AL.
hyoid muscle. Excision of the cyst, together with
this portion of the sublingual gland, successfully
relieved the patient's symptoms. Pathology showed
salivary gland tissue with a moderate degree of
chronic sialadenitis and a cyst lined by fibrous
tissue and loose vascular tissue. No epithelial lining
was present over the surface of the cyst.
Fig. 1. Patient with plunging ranula in right neck.
Discussion
A plunging ranula is a rare condition. Only 89
patients with this condition have been reported in
the English literature and there have been no cases
reported from Australia.' In a review of 12 inde-
pendent reports covering 29 detailed case histories,
no author has had a series of more than four
cases.'-'' The present series of three cases must
therefore be regarded as significant.
This condition occurs in young people. In the 32
cases reviewed (including those described here),
the average age was 24 years: four were under 10
years and 22 were aged 10-30 years. Infection and
trauma were not obvious causal features in most of
the histories. In only two cases, the oral ranula may
have been caused by previous unrelated surgery to
the floor of the m ~ u t h . ~ . ~
In contrast to the pathogenesis of the oral ranula,
trauma plays a significant part in the formation of
the plunging ranula. In the review cases, 44% were
iatrogenic. occurring after attempts at removing the
oral ranula. Surgeons must be aware that an incom-
plete oral operation often results, not only in a
recurrence of the oral cyst, but also may predispose
to the formation of the cervical pseudocyst.
Batsakis described two varieties of ranula: the
'simple ranula' or true retention cyst which is
epithelially lined and results from obstruction of the
salivary ducts, and the 'plunging ranula' which is
an extravasation pseudocyst and which may appear
as a classical sublingual ranula or as a subman-
dibular mass without visible intra-oral connec-
tion.13 The literature review has shown that one-
PLUNGING RANULA
third of the authors found that the oral ranula did
not possess an epithelial lining. Parekh et al. sug-
gested that the continuous extravasation of saliva
from an injured sublingual gland results in the
formation of an oral ranula.* This is in keeping with
the first case described in the present report, where
no discrete retention cyst was present at operation.
An extravasation pseudocyst may evolve when the
simple retention cyst is inadequately excised or
marsupialized. The surgeon must therefore recog-
nize the need to perform complete intra-oral ex-
cision of the sublingual gland when the clinical
findings suggest a pseudocyst rather than a simple
retention cyst, thus eliminating the possibility of
the later formation of the plunging ranula.
The cervical swelling is produced by mucus
extravasation from the oral cavity into the neck.
The pathway may be either along the deep lobe of
the submandibular gland to exit between the
hyoglossus and mylohyoid muscles, or directly
through a dehiscence in the mylohyoid muscle
itself. Gaughran described the mylohyoid bouton-
nitre and sublingual bouton.'4 He examined 324
cadaver half heads in 1 17 (36%) found one or more
distinct masses of sublingual gland tissue resting on
the inferior surface of the mylohyoid muscle. The
mylohyoid boutonnitre was a cleft in the mylo-
hyoid muscle through which the sublingual gland
penetrated. It is not certain, however, that in the
normal population the incidence is as high as this.
An examination of 14 cadaver dissections from two
university anatomy departments revealed only one
case of a sublingual boutonnitre (Fig. 2). There is
little doubt, however, that this anatomical defect is
implicated in the pathogenesis of the plunging
ranula.
Fig. 2. Cadaver specimen with sublingualbouton pen-
etrating mylohyoid muscle.
Nine of the 16 authors found that, in at least one
case, the mucus had extravasated through defects in
the mvlohvoid muscle. In the three patients de-
scribei here, the pseudocyst was conneited directly
to glandular tissue within or deep to the mylohyoid
947
muscle. This readily explains the occurrence of the
cervical swelling in the absence of an intra-oral
ranula. Only five cases of this condition have
previously been reported and our third case is the
sixth. In every case, the pseudocyst was connected
to a mylohyoid defect. This possibility must be
considered in the diagnosis of the isolated upper
cervical neck cyst.
Once a diagnosis of plunging ranula has been
established, a complete excision of the offending
sublingual gland is mandatory. This can be accom-
plished through the mouth. The surgeon must
identify and preserve the lingual nerve and the
submandibular duct. The possibility of extention
into or through the mylohyoid muscle should not be
overlooked. although a significant cervical exten-
sion may be difficult to mobilize through a limited
oral incision, and the surgeon may prefer to a p
proach this via a neck incision, and at the same time
dissect out the pseudocyst. Oral excision of the
sublingual gland followed by simple aspiration of
the cervical pseudocyst is also successful.' Avoid-
ance of a neck operation reduces the possibility of
an orocervical fistula complicating cervical
surgery.
In those rare cases where the cervical cyst is the
only manifestation of the plunging ranula, surgical
exploration is necessary, not only to exclude other
pathology, but also to treat the glandular perfora-
tion of the mylohyoid muscle. In our second and
third patients, partial excision of only this portion
of the gland was sufficient to cure the patient. This
more limited approach has not been published else-
where. In these cases the pseudocyst was easily
mobilized off the submandibular gland, which was
not resected.
Conclusions
An oral ranula may be a simple retention cyst or an
extravasation pseudocyst. Inadequate treatment of
either lesion may lead to recurrence and can predis-
pose to the formation of a plunging ranula. If the
oral cyst recurs, the sublingual gland should be
removed. The oral and cervical components of the
plunging ranula often communicate through defects
in the mylohyoid muscle, through which the mucus
tracks. Boutons of sublingual tissue may occupy
these defects. In any operation for plunging ranula,
exploration of the mylohyoid and excision of this
ectopic glandular tissue is essential.
Acknowledgement
Figure 2 was provided by courtesy of the Depart-
ment of Anatomy. University of Melbourne.
948
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