International Journal of Otolaryngology and Head & Neck Surgery, 2013, 2, 280-285
Published Online November 2013 (
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Papillary Cystadenoma of Larynx: A Case Report
Tammam Abou Ali1, Basel Al Sabah2, Kavitha Gopalan2
1Department of ENT, Farwaniya Hospital, Al-Farwaniy a h, Kuwait
2Department of ENT, Zain & Al Sabah Hospital, Kuwait City, Kuwait
Received August 28, 2013; revised September 26, 2013; accepted October 15, 2013
Copyright © 2013 Tammam Abou Ali et al. This is an open access article distributed under the Creative Commons Attribution Li-
cense, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
An uncommon clinical entity o f papillary cystadenoma of the larynx in a lady of 37-year-old is presented. Th e diagnosis
after clinical examination was achieved by video-stroboscopy, CT scan of the neck, biopsy and histopathological ex-
amination. This case study emphasizes the importance of recognition of this lesion by the clinician as the differential
diagnosis includes lesions with similar clinical appearance.
Keywords: Papillary Cystadenoma; Larynx; Minor Salivary Gland Tumors
1. Introduction
Cyst adenomas represent between 2.0% and 4.7% of all
minor salivary gland neoplasms [1,2], and papillary cys-
tadenoma of salivary glands is an uncommon benign,
well-circumscribed epithelial neoplasm resembling a
mucous retention cyst. The median age of occurrence is
53.5 and 55 years [1] with a female prevalence. The cys-
tic lesions have papillary projections arising from undif-
ferentiated epithelium of the intercalated ducts of the
glands [3] which could be the minor salivary glands, the
parotid glands, oral cavity, larynx, nasopharynx or the
lacrimal gland.
We herein present a case of multiple papillary cyst
adenomas of the larynx with gross, histological features
and important differential diagnosis of these lesions.
2. Case Report
A 37-year-old wo man with unr emarkable medical histor y,
presented with dysphonia and mild dysphagia for liquids
since one year which were gradually progressive. She did
not give history of dyspnea, odynophagia, otalgia or
cough. Laryngeal examination and videostroboscopy
revealed multiple pinkish sessile cystic lesions of 1 to 1.5
cm diameter, with normal overlying mucosa, distributed
all over the supraglo ttis giving a cobb le-stone appearance.
The true vocal fo lds were normal in appearance and mo-
bility. The sub glottis was free. No cervical lymphade-
nopathy was noted. Serum biochemistry, full blood count
and erythrocyte sedimentation rate were within normal
limits. Computed tomography scan of neck revealed mild
nodular irregularity of entire supraglottis including both
false vocal cords (more appreciated on the left side) with
normal CT appearance of true vocal cords as shown in
Figure 1. Stroboscopy confirmed the CT findings of
multiple cystic lesions in supraglottis as shown in Fig-
ures 2(a)-(c). Direct laryngoscopy and biopsy of the cys-
tic lesions were done. Figures 3(a) and (b) show the cys-
tic lesions in supraglo ttis while biopsied.
Histopathological analysis revealed benign tumors
(cysts), covered my non-ulcerated mucosa of non-
keratinized squamous epithelium with areas of focal hy-
Figure 1. CT neck.
T. A. ALI ET AL. 281
Figure 2. Stroboscopic pictures of larynx with multiple cys-
tic lesions in the entire supraglottis.
perplasia as shown in Figures 4(a)-(c). Submucosa
showed prominent fibrosis and mild inflammatory cellu-
lar infiltrate. The sub epithelial stroma showed multiple
Figure 3. Operative pictures of the supraglottic cysts during
cystic and partially solid nodules. The cystic cavity ex-
hibited papillary growth and finger like processes with
fibro vascular core as seen in Figures 5(a)-(c). The lin-
ing epithelium is mainly columnar with abundant cyto-
plasm and basophilic nucleus. No evidence of mitosis
noted and no evidence of invasion to the surrounding
connective tissue stroma was noted. The established di-
agnosis was papillary cystadenoma.
By endolaryngeal surgery using Kleinsasser’s micro
laryngoscopy technique, multiple cysts in entire suprag-
lottis and false vocal cords were completely excised us-
ing micro instruments as shown in Figure 6. Laser or
powered instruments were avoided for fear of fibrosis
latter. Patient had uneventful post-operative recovery.
Histopathology of the excised lesions confirmed the di-
agnosis of papillary cyst adenoma and no stromal inva-
sion were identified.
The patient is currently being followed up and shows
no signs of recurrence.
3. Discussion
The classification of salivary gland tumors by WHO
(1991) has distinguished two variants of cystadenoma:
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Figure 4. (a), (b) and (c) (LP): shows pieces of mucosa and
sub mucosa, covered by non-keratinized squamous epithe-
lium with no ulceration of the covering mucosa.
papillary cystadenoma and mucinous cystadenoma [4].
Figure 5. (a) and (b) (H&E × 100): cystic cavity exhibiting
papillary projections supported by fibrous connective tissue;
(c) (H&E × 200): papillary projections lined by tall colum-
nar cells and surrounding tissue free of invasion.
Histologically, it consists of a multilocular proliferation
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T. A. ALI ET AL. 283
Figure 6. Operative picture of endoscopic excision of the
lesions in the supraglottis.
of glandular epithelium. Epith elial cells may b e co lumnar,
cuboidal, squamous or oncocytic-like. Papillary forma-
tions are frequently seen. Stroma is fibrous, rarely with
prominent lymphocytic elements. Papillary cystadenoma
closely resembles Warthin’s tumour, but is devoid of
lymphoid stroma. Mucinous cystadenoma, like papillary
cystadenoma, is a multicystictumour, but is lined by
mucous-producing goblet cells. There are no atypia, nor
any invasion, in either of the two types of cystadenoma.
Papillary cystadenoma, although rare in salivary glands
outside the larynx, can only occasionally be found in
major salivary glands, the nasopharynx, buccal mucosa,
the lacrimal gland the palate and the tonsillar region.
Laryngeal papillary cystadenoma often show oncocytic
metaplasia and are reactive cystic lesions with hyperpla-
sia rather than true neoplasia [5]. This cellular oncocyto-
sis appears to be age related and is accelerated by smok-
The rarity of this tumour is reported by many authors.
The incidence of papillary cystadenoma in a review of
426 cases of intraoral minor salivary gland tumours by
Waldron et al. [6] is 4.7% of all minor salivary gland
neoplasms and 8.1% of all benign epithelial minor sali-
vary gland neoplasms. Similarly, Chaudry et al. [7] re-
ported only 2% of occurrence of papillary cystadenoma
in a large study of 800 intraoral minor salivary gland
benign tumours.
Previous reports have shown an age range from 12 to
89 years, and an average age at diagnosis of 57 years [8].
Also, previous series have shown a female predominance
In our case, laryngeal examination along with vide-
ostroboscopy and CT scan of neck guided the clinical
diagnosis of multiple cystic lesions in the supraglottis.
Excision biopsy and histopathology ascribed the definite
diagnosis of papillary cystadenoma. As special stains and
immunohisto chemistry are non-contributory for the di-
agnosis [14], they were not done.
The histologic features of this case of papillary cysta-
denoma were characteristic of the lesion rendering the
diagnosis easier. The lesions were well circumscribed
having cystic spaces with intracystic papillary projec-
tions lined by tall co lumnar cells with b asoph ilic nu cleus.
Oncocytes, squamous epithelium or mucous cells were
not seen in these lesions which are common variants re-
ported. No cellular atypia or mitotic figures were noted
and adjacent stroma was free of invasion ruling out ma-
Clinically, the differential diagnosis encountering cys-
tic lesions of the larynx includes mucous retention cysts,
extra nodal lymphomas, lipomas, papillomas, schwan-
nomas and neurofibromas, adenomatoid hyperplasia of
minor salivary glands etc.
The important histological differential diagnosis of
papillary cystadenoma includes papillary cystadenoma
lymphomatosum, oncocytic cyst adenomas, oncocytomas,
low-grade mucoepidermoid carcinoma, the papillary—
cystic variant of acinic cell carcinoma and cystadenocar-
cinoma [10].
Both papillary cystadenoma and papillary cystade-
noma lymphomatosum (Warthin’s tumour) are cystic
tumors but are differentiated by the presence of numer-
ous oncocytes, p apillary formatio ns and lympho id stroma
with lymph follicles which repr esent the main features of
Warthin’stumour [1]. Oncocytic variants of papillary
cystadenoma exist which could be confused with
Warthin’s tumour or oncocytoma.
Oncocytic cystadenoma is characterized by a smooth
cyst wall composed of eosinophyllic cells with granular
cytoplasm (due to numerous mitochondria). The cyst is
adjacent to normal respiratory epithelium.
Oncocytomas are true adenomas and are composed of
solid Oncocytes without cystic component.
Mucoepidermoid carcinoma is characterized his-
tologically by solid nests composed of a mixture of mu-
cous forming cells, epidermoid cells and sometimes of
intermediate cells which are absent in cystadenoma.
In general, four different cell types constitute acinic
cell carcinoma which includes large acinic cells, vacuo-
lated cells, clear cells and non-specific glandular cells
and in the papillary cystic variant of acinic cell carci-
noma, a configuration with papillae in cystic spaces is
seen which resembles papillary cystadenoma [11]. A
microcystic growth pattern in the intra luminar fronds
which is a characteristic feature of acinic cell carcinoma
is not seen in papillary cystadenoma and helps to differ-
The most important differential diagnosis of papillary
cystadenoma is cystadenocarcinoma as both neoplasms
have similar architecture with papillary proliferation of
the epithelial lining and are composed of cells that pos-
sess bland cytomorphologic features [12]. Moreover,
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cystadenocarcinoma often shows little atypia [13]. Dif-
ferentiation of tumou r types depends largely on the id en-
tification of actual infiltration of salivary gland paren-
chyma or surrounding connective tissue by either cystic
or solid epithelium in cystadenocarcino mas.
The suggested management for these benign lesions is
conservative and consists of complete local excision
[5,14]. Open laryngeal surgery like Lateral/Median Thy-
rotomy and pharyngotomy approach or transoral ap-
proach is required for the cartilaginous structures of the
larynx; if the position of larynx is to be altered, or if the
anatomical situation of lesion prevents adequate endo-
scopic visualization .
Laryngeal lesions located in the mucosa or sub muco-
sal space is easily accessible to endoscopic approach
with advantages of avoidance of skin incisions, division
of the thyroid cartilage and tracheotomy.
The surgical approach and excision depend on the tu-
mor size and location; but in general, preservation of
laryngeal function is the primary aim of treatment in the
form of complete excision with tumor-free margins. With
improvements in imaging, histopathology, and surgical
techniques—endoscopy, microscopic magnification, ap-
plication of lasers—there has been marked improvement
in the accuracy of diagnosis and the ability to perform
precision excision surgery for benign tumors. This means
more preservation and restoration of laryngeal function
than was previously possible [15]. As per the recom-
mendations, we completely excised the entire cysts in
supraglottis by endoscopic micro laryngeal surgery and
histopathology reconfirmed the diagnosis as benign pap-
illary cystadenoma. Although papillary cystadenoma are
benign lesions, follow-up is recommended, as recom-
mended for oncocytic cysts since recurrence is possible,
especially in the case of patients with multiple involve-
ments, with a tendency to develop new cysts [16]. In our
case, considering the benign nature of the lesions, annual
revisions were decided for a minimum period of five
4. Conclusion
We present this case of multiple laryngeal papillary cys-
tadenomas, a rare benign lesion presenting with hoarse-
ness and dysphagia as this clinical en tity h as to be kept in
mind while evaluating patients with above mentioned
symptoms. Moreover, the histo-pathological diagnosis is
crucial as many tumours are with similar features and
management of each varies.
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