10.14639/0392-100X-N0759
Objective. Congenital nasolacrimal duct cyst (NLDC) is a rare disorder, which can present with ophthalmological and nasal signs and symptoms. The authors analyse their personal experience to identify diagnostic criteria for NLDC, which were treated by endoscopic transnasal procedure.
Methods. Clinical records of patients with a diagnosis of NLDC were retrospectively reviewed. All patients underwent rhinoscopy and ophthalmologist evaluation before surgery, whereas imaging was performed in selected cases. All neonates underwent transnasal endoscopic marsupialisation after failure of conservative medical therapy. Results. Five patients were included in the study. One patient presented bilateral NLDC. In 3 cases, CT scan of the sinus was carried out. A total of 6 marsupialisation procedures were performed and a bi-canalicular lacrimal stent was positioned in 1 case. Complete remission of symptoms was observed in all cases.
Conclusions. Nasal endoscopy is mandatory to diagnose NLDCs, and, in some cases, it can be complemented by radiological procedures. When symptoms persist after systemic and topical therapy, nasal endoscopic marsupialisation is the treatment of choice. This surgical procedure is effective, safe and can be repeated if needed.
application/xml
1827-675X
759
5
en
Marco Berlucchi
https://creativecommons.org/licenses/by-nc-nd/4.0
ACTA Otorhinolaryngologica Italica
minimally invasive surgical procedures
Treatment of congenital nasolacrimal duct cyst: the role of endoscopic marsupialisation
Text.Serial.Journal
https://www.actaitalica.it/
40
Rhinology
2020-12-03
2020-03-23
2020-12-04
2020-12-04
377-382
1
Marco Berlucchi
2020/12/03
10.14639/0392-100X-N0759
377
Unit of Pediatric Otorhinolaryngology, ASST Spedali Civili of Brescia, Italy
1827-675X
5
minimally invasive surgical procedures
en
382
Treatment of congenital nasolacrimal duct cyst: the role of endoscopic marsupialisation
https://www.actaitalica.it/article/download/759/466
40
ACTA OTORHINOLARYNGOLOGICA ITALICA 2020;40:377-382; doi: 10.14639/0392-100X-N0759




Rhinology


Treatment of congenital nasolacrimal duct cyst:
the role of endoscopic marsupialisation
Il trattamento delle cisti congenite del dotto nasolacrimale:
il ruolo della marsupializzazione endoscopica
Vittorio Rampinelli1, Marco Ferrari1,2, Silvia Zorzi1, Marco Berlucchi3
1
 Unit of Otorhinolaryngology-Head and Neck Surgery, University of Brescia, ASST Spedali Civili of Brescia, Italy; 2 Section
of Otorhinolaryngology-Head and Neck Surgery, Department of Neurosciences; University of Padua, Italy; 3 Unit of Pediatric
Otorhinolaryngology, ASST Spedali Civili of Brescia, Italy


SUMMARY
Objective. Congenital nasolacrimal duct cyst (NLDC) is a rare disorder, which can present
with ophthalmological and nasal signs and symptoms. The authors analyse their personal
experience to identify diagnostic criteria for NLDC, which were treated by endoscopic
transnasal procedure.
Methods. Clinical records of patients with a diagnosis of NLDC were retrospectively re-
viewed. All patients underwent rhinoscopy and ophthalmologist evaluation before surgery,
whereas imaging was performed in selected cases. All neonates underwent transnasal endo-
scopic marsupialisation after failure of conservative medical therapy.
Results. Five patients were included in the study. One patient presented bilateral NLDC. In     Received: March 23, 2020
3 cases, CT scan of the sinus was carried out. A total of 6 marsupialisation procedures were    Accepted: May 21, 2020
performed and a bi-canalicular lacrimal stent was positioned in 1 case. Complete remission
of symptoms was observed in all cases.                                                          Correspondence
Conclusions. Nasal endoscopy is mandatory to diagnose NLDCs, and, in some cases, it can         Vittorio Rampinelli
be complemented by radiological procedures. When symptoms persist after systemic and            Unit of Otorhinolaryngology-Head and Neck
topical therapy, nasal endoscopic marsupialisation is the treatment of choice. This surgical    Surgery, Department of Medical and Surgical
procedure is effective, safe and can be repeated if needed.                                     Specialties, Radiological Sciences, and Public
                                                                                                Health, University of Brescia, ASST Spedali Civili
KEY WORDS: nasolacrimal duct, children, nasal surgical procedures, minimally invasive           di Brescia, 25121 Brescia, Italy
surgical procedures                                                                             Tel. +39 030 3995319. Fax +39 030 3995212
                                                                                                E-mail: vittorio.rampinelli@gmail.com
RIASSUNTO
                                                                                                Funding
Obiettivo. La cisti congenita del dotto nasolacrimale (CDNL) è una condizione rara, che
                                                                                                None.
si manifesta con segni e sintomi oftalmologici e nasali. Gli autori analizzano la propria
esperienza al fine di identificare criteri diagnostici per la CDNL, trattata con procedura
                                                                                                Conflict of interest
endoscopica transnasale (ET).
                                                                                                The Authors declare no conflict of interest.
Metodi. È stata eseguita un’analisi retrospettiva dei dati clinici dei pazienti affetti da
CDNL. I pazienti sono stati sottoposti ad endoscopia nasale (EN) e valutazione oculistica
prima dell’intervento, l’imaging è stato eseguito in casi selezionati. Tutti i neonati sono       How to cite this article: Rampinelli V, Ferrari M,
stati sottoposti a marsupializzazione ET dopo fallimento di terapia medica.                       Zorzi S, et al. Treatment of congenital nasolacri-
Risultati. Cinque pazienti sono stati inclusi nello studio. Un paziente ha presentato CDNL        mal duct cyst: the role of endoscopic marsupiali-
bilaterale. In 3 casi, è stata eseguita TC del massiccio facciale. Sono state eseguite 6 pro-     sation. Acta Otorhinolaryngol Ital 2020;40:377-
                                                                                                  382. https://doi.org/10.14639/0392-100X-N0759
cedure di marsupializzazione; uno stent lacrimale bi-canalicolare è stato posizionato in 1
caso. La remissione completa dei sintomi è avvenuta in tutti i casi.
                                                                                                © Società Italiana di Otorinolaringoiatria
Conclusioni. L’EN è chiave per la diagnosi di CDNL e può essere integrata con procedure         e Chirurgia Cervico-Facciale
radiologiche. Quando i sintomi persistono dopo terapia sistemica e topica, la marsupia-
lizzazione ET rappresenta il trattamento di scelta. Questa procedura chirurgica è efficace,                    OPEN ACCESS
sicura e può essere se necessario ripetuta.
                                                                                                This is an open access article distributed in accordance with
PAROLE CHIAVE: dotto nasolacrimale, pediatrico, procedure chirurgiche nasali,                   the CC-BY-NC-ND (Creative Commons Attribution-Non-
procedure chirurgiche mininvasive                                                               Commercial-NoDerivatives 4.0 International) license. The
                                                                                                article can be used by giving appropriate credit and mentio-
                                                                                                ning the license, but only for non-commercial purposes and
                                                                                                only in the original version. For further information: https://
                                                                                                creativecommons.org/licenses/by-nc-nd/4.0/deed.en




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V. Rampinelli et al.




Introduction                                                      Pediatric Otorhinolaryngology, Spedali Civili of Brescia,
                                                                  Brescia - Italy, from January 2010 to January 2019 were
Congenital nasolacrimal duct (NLD) obstruction is a com-
                                                                  retrospectively reviewed.
mon disorder. Cassady reported incomplete canalisation of
                                                                  Informed consent was systematically acquired at time of
NLD in more than 70% of newborns 1, with Hasner’s valve
                                                                  hospitalisation. Ethical approval was not required due to
being the most frequent site of stenosis 2,3. The majority of
                                                                  the retrospective nature of the study and anonymisation of
cases resolve spontaneously during the first weeks of life 4,5,
                                                                  all data.
while persistent epiphora is observed in 6-20% of infants 5,6.
                                                                  All patients underwent preoperative flexible nasal endosco-
Several malformations have been attributed to persistent
                                                                  py and ophthalmologist evaluation. Before January 2013,
epiphora in young patients. The term “inferior mucocele”
                                                                  all patients underwent CT scan of sinuses to confirm diag-
or “NLD cyst” (NLDC) refers to an unperforated Hasner’s
                                                                  nosis; thereafter, our diagnostic strategy changed, in order
valve showing an inferior prolapse and therefore occupying
                                                                  to avoid redundant radiological exams. We utilised the fol-
the inferior meatus and nasal cavity 7. NLDC associated
                                                                  lowing diagnostic criteria for NLDC:
with dacryocystocele, which consists of a cystic enlarge-
                                                                  • nasal obstruction;
ment of lacrimal sac 8, is reported in 0.1% of infants dur-
                                                                  • epiphora;
ing the first year of life 5. The dacryocystocele may resolve
                                                                  • nasal endoscopy revealing a translucent, cystic lesion
spontaneously, but frequently becomes infected and rapidly
                                                                     centered on the anterior portion of the inferior turbinate
progresses to acute dacryocystitis, lacrimal sac empyema
                                                                     and obstructing the nasal fossa;
and preseptal/orbital cellulitis 9.
                                                                  • no suspicion of orbital complications at ophthalmologist
Diagnosis of NLDC is usually based on clinical symptoms,
                                                                     evaluation;
endoscopic appearance and imaging. The typical clinical
                                                                  • no suspicion of another pathological condition (i.e., cho-
presentation includes epiphora, history of relapsing dacryo-
                                                                     anal atresia, pyriform aperture stenosis, nasopharyngeal
cystitis, and unilateral nasal obstruction. When the malfor-
                                                                     teratoma, and meningoencephalocele) at flexible nasal
mation is bilateral, clinical presentation emulates adenoid
                                                                     endoscopy.
hypertrophy, with different respiratory issues consisting
of chronic snoring respiration, difficulties in sleeping and      If all the above criteria were satisfied, a clinical diagno-
feeding, and severe respiratory distress associated with          sis was made and CT and/or magnetic resonance imaging
cyanosis and/or intercostal retraction. At nasal endoscopic       (MRI) were deemed unnecessary. All patients underwent
evaluation, NLDC appears as a mucosal translucent protru-         surgery after failure of conservative treatments (i.e., mas-
sion stemming from the inferior meatus, in proximity to the       sage of the medial cantus, antibiotic and steroid eye drops,
area of Hasner’s valve. If associated with dacryocystocele,       systemic antibiotic therapy) for at least 3 weeks. Signs of
a blue swelling at the level of the ipsilateral medial cantus     acute inflammation of the nasolacrimal duct system were
is usually noticeable. CT scan of the sinuses confirms diag-      considered a temporary contraindication to surgery, which
nosis, above all when the nasal fossa is not clearly explor-      was postponed after resolution of inflammation-related
able and/or other associated conditions are suspected 10.         symptoms.
Management of NLDC includes conservative and non-
conservative treatments. The former comprises massage of          Surgical technique and postoperative management
the lacrimal sac, topical antibiotics and steroids and sys-       The surgical procedure was performed under general anaes-
temic antibiotic therapy. The latter, which usually follows       thesia. After mucosal decongestion with pledgets soaked
an unsuccessful conservative strategy, consists of a surgical     with adrenalin (1:100,000), a rigid nasal 0° endoscope 2.7
procedure, ranging from nasolacrimal probing to intrana-          diameter, 11 cm long (Karl Storz, Tuttlingen, Germany)
sal endoscopic marsupialisation 2. The aim of the present         connected with high definition camera and xenon-175-watt
single-institution study is to analyse clinical outcomes of       cold light illumination source (HD 3 CCD camera, Karl
children who underwent transnasal endoscopic marsupi-             Storz, Tuttlingen, Germany) was placed intranasally to
alisation of NLDC and to identify clinical and endoscopic         identify and analyze the extent of NLDC. The NLDC was
diagnostic criteria to diagnose congenital NLDC.                  marsupialised by removing the anteromedial wall of the
                                                                  cyst by cold sharp instruments that are commonly em-
                                                                  ployed for otosurgery. In one case, a microdebrider was
Materials and methods                                             used to perform partial marsupialisation of NLDC. Once
Patients and preoperative workup                                  valve patency was checked with a 30° scope (Karl Storz,
Clinical records of patients who underwent transnasal             Tuttlingen, Germany), antibiotic ointment (Mupirocin) was
endoscopic marsupialisation of a NLDC at the Unit of              applied in the inferior meatus. Bilateral NLDCs were oper-



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                                                                                             Endoscopic marsupialisation of congenital nasolacrimal duct cyst




Table I. Relevant data of the present series (Pt: patient; CT: computed tomography; DCC: dacryocystocele).

 Patient        Age/            Unilateral/         Imaging           Associated           Breastfeeding            Breathing            Hospitalisation days
               Gender            bilateral                             findings             difficulties            difficulties
 1           5 months/F          Unilateral            CT                DCC                      No                     No                          3
 2            18 days/M          Unilateral            CT                None                     No                     No                          2
 3            12 days/F          Unilateral            CT                None                     Yes                    No                          2
 4            45 days/F          Bilateral             No                None                     Yes                    No                          2
 5           2 months/M          Unilateral            No                None                     No                     No                          1



ated on concurrently with a bilateral procedure. No nasal
packing was applied after surgery.
Patients underwent systemic antibiotic therapy (amoxi-
cillin) for 1 week and nasal irrigation twice per day for
1 month after surgery. The first postoperative outpatient
control with nasal flexible endoscopy was scheduled at 1
week. Patients were closely followed for 3 months after
surgery, until appropriate healing of the surgical site was
endoscopically demonstrated. The last control was sched-
uled at 12 months after surgery. According to Lueder 9, the
procedure was considered successful if both symptoms and
signs attributable to lacrimal obstruction or inflammation
completely recovered.

Results
Five patients were included in the study. Transnasal endo-
scopic marsupialisation of the NLDC was the primary sur-
gical treatment for all patients. Relevant data of the series
                                                                                 Figure 1. Endoscopic appearance of congenital nasolacrimal duct cyst (as-
are summarised in Table I. Two patients were male and 3                          terisk) in left nasal fossa. Inferior turbinate (IT) is displaced laterally and supe-
were females, with a mean age of 26 days (range: 12 days                         riorly. NS – Nasal septum.
to 4 months). All children had normal development with
no other relevant comorbidities. No case of acute respira-
tory distress was observed. In all patients, ophthalmologic                      bital canthus was observed. A concomitant dacryocystocele
evaluation documented the presence of epiphora associated                        was identified in a single case. No other malformations or
with at least one episode of dacryocystitis, presenting with                     associated conditions were detected.
eyelid oedema and purulent secretions from lacrimal cana-                        Following the abovementioned diagnostic criteria, 2 cases
liculi. Medial canthal swelling was observed in 1 patient,                       were treated based on clinical and endoscopic diagnosis af-
who had a concomitant dacryocystocele.                                           ter January 2013.
Nasal endoscopy showed a cystic lesion centered on the                           Six endoscopic marsupialisation procedures were per-
area of Hasner’s valve in all patients. The cyst filled the                      formed (4 unilateral and 1 bilateral surgeries). Intraopera-
inferior meatus and nasal fossa, occluding the ipsilateral                       tive probing of the nasolacrimal system was avoided. In the
nasal respiratory space (Fig. 1). The middle meatus was                          patient with a dacryocystocele, a bi-canalicular lacrimal
usually patent or partially narrowed due to cranial displace-                    stent was positioned and removed 6 months later under
ment of the inferior turbinate.                                                  general anaesthesia.
CT scan of sinuses was performed in the 3 cases treated                          No perioperative complication was observed. All patients
before January 2013, showing an enlarged NLD that was                            were discharged within the first and third postoperative
in continuity with a cystic lesion within the nasal cavity.                      day. Endoscopic outpatient medications and follow-up
In all these cases, the NLDC was occupied by hypodense                           evaluations were performed at 1 week, and 1, 2, 3, and 12
material and hypertrophy of soft tissues of the medial or-                       months after surgery. Simple local anaesthesia without se-



                                                                                                                                                                379
V. Rampinelli et al.




dation was employed during outpatient evaluation. Com-              served in our series, anterior rhinoscopic examination should
plete recovery of symptoms was observed in all patients.            be included in routine newborn examination, especially when
                                                                    newborns or neonates present obstructive symptoms 14.
Discussion                                                          The lacrimal system connects the orbital and nasal com-
                                                                    plexes; therefore, multidisciplinary management of its
The present retrospective observational study reviewed 5            pathological conditions is recommended. Hence, all pa-
patients undergoing endoscopic transnasal marsupialisa-             tients in our series underwent ophthalmologist evalua-
tion for NLDC at a single institution between 2010 and              tion to exclude proximal stenosis, orbital complications,
2019. The aim of the study was to identify clinical and en-         or other malformations. The role of imaging in diagnostic
doscopic features that allow diagnosis of NLDC. Moreo-              work-up is ill-defined. CT and dacryocystographic findings
ver, an overview of the management strategy for this ex-            of patients affected by dacryocystocele and NLDCs have
ceedingly rare clinical entity, focusing on postoperative           been described by several authors 15,16. The presence of a
results of endoscopic treatment, was performed. To the              cystic lesion of the anterior-inferior-lateral portion of the lat-
best of our knowledge, only a few case reports or limited           eral nasal wall, medial displacement of the inferior turbinate,
case series on the transnasal endoscopic marsupialisation           and pronounced distention of NLD possibly associated with
of NLDC can be found in the literature. Moreover, some              lacrimal sac dilation are the main signs raising suspicion for
of confusion has emerged around this pathological entity,           NLDC. Castillo et al. 16 diagnosed NLDC using CT to evalu-
especially concerning its distinction from similar diseases         ate 2 newborns who presented with acute respiratory distress
such as dacryocystocele.                                            immediately after birth. Both were found to have bilateral
The pathogenesis of both NLDC and dacryocystocele con-              soft-tissue masses beneath the inferior turbinates. MRI has
sists of mucosal obstruction of the NLD, which leads to in-         also been used in evaluation of dacryocystoceles 17, by virtue
creasing pressure and subsequent dilatation of the lacrimal         of its optimal contrast resolution on soft tissue, which allows
system in areas that are not completely surrounded by bone          characterisation of the cyst content. The primary advantage
(i.e. Hasner’s valve and the lacrimal sac, respectively).           of MRI over CT is the avoidance of exposure to ionising
The level of obstruction on a craniocaudal axis determines          radiation. On the other hand, MRI requires sedation of the
whether the cystic swelling appears as a dacryocystocele            newborn and provides poorer depiction of bony surfaces.
or a NLDC: cysts located in proximity to Hasner’s valve             We performed CT scan in the first 3 cases we handled to
exhibit an intranasal cystic mass with possible retrograde          study the anatomical region and confirm diagnosis. Based
dilation of the NLD, while more cranial obstruction does            on our experience and the literature 2, we thought that clini-
not show relevant nasal changes 11.                                 cal and endoscopic evaluation could be sufficient for di-
Infantile dacryocystocele and many cases of paediatric              agnosis. Thus, after January 2013, we introduced clinical
acute dacryocystitis are associated with NLDC 9. Barham             diagnostic criteria in our practice. If satisfied, an exclusive-
et al. 4 even hypothesised that the coexistence of the two          ly clinical diagnosis was deemed reliable to proceed with
entities (dacryocystocele and NLDC) is unvarying. On the            treatment, and imaging was avoided. Unfortunately, the
other hand, many cases of NLDCs are not associated with a           small number of patients due to rarity of the disease does
congenital dacryocystocele, nor to epiphora or other distur-        not permit to perform statistical analysis. Consequently, we
bances referable to the lacrimal sac 2,12. In the present series,   hope that present study could stimulate a multicentre trial
which includes 4 monolateral and 1 bilateral NLDCs, all             that confirms this hypothesis.
patients experienced at least 1 episode of acute dacryocyst-        However, CT and/or MRI should be considered to corrobo-
itis, whereas a dacryocystocele was present in a single case.       rate diagnosis when the nasal fossa is not clearly explorable
The slight female predisposition reported by Levin et al. 13        or other/associated conditions are suspected: choanal atre-
and Brachlow et al. 14 was confirmed in our series, with a          sia, pyriform aperture stenosis, nasopharyngeal teratoma
male-to-female ratio of 2:3.                                        and meningoencephalocele 2,10.
Diagnosis is based on history, clinical examination and nasal       Sundry treatment approaches have been described for man-
endoscopy: nasal obstruction, epiphora, medial canthal swell-       agement of NLDCs: massage, warm compresses, topic, en-
ing, eyelid oedema and purulent secretions from lacrimal            teral or parenteral antibiotics, nasolacrimal probing, laser mu-
canaliculi are the most frequent clues, with nasal endoscopy        cosal vaporisation, silastic stenting, and intranasal endoscopic
revealing a translucent, cystic lesion centered on the anterior     cyst marsupialisation. Our management consisted first of
portion of the inferior turbinate and obstructing the nasal fos-    medical therapy associated with delicate massage of the me-
sa. As bilateral NLDCs can be associated with complete nasal        dial canthus. If these therapeutic procedures failed, transnasal
obstruction and severe respiratory distress, although never ob-     endoscopic marsupialisation of the cyst was performed.



380
                                                                             Endoscopic marsupialisation of congenital nasolacrimal duct cyst




Though limited to a small sample size, endoscopic transna-       diagnosis of nasal obstruction associated with epiphora in
sal marsupialisation showed optimal results in the present       newborns and infants. Multidisciplinary management by
series: all 5 patients, for a total of 6 procedures, experi-     both an otorhinolaryngologist and ophthalmologist is rec-
enced complete remission of symptoms after surgery, in           ommended. The suggestive clinical picture associated with
line with what reported in the literature 2,9,13,18. Some au-    a complete nasal endoscopy could be sufficient for diag-
thors have suggested probing the NLD to verify its patency       nosis. On the other hand, cross-sectional imaging should
after marsupialisation 2,18. In our experience, lavage of the    be considered in case of uncertain diagnosis or suspect for
lacrimal pathway is sufficient to demonstrate the patency        concomitant malformations. Our experience, even if lim-
of NLD. With the intent of avoiding inflammation-related         ited to 5 NLDC cases, suggests that endoscopic marsu-
complications, an endoscopic procedure was performed             pialisation without NLD stenting is an effective and safe
when signs of acute inflammation resolved with medical           procedure. However, a multicentre, prospective study is
therapy. However, other authors highlighted that in cases of     needed to establish whether this treatment is significantly
acute infection resistant to antibiotic therapy prompt surgi-    preferable over other surgical techniques alongside more
cal management should be considered to prevent disease           precisely quantify the morbidity of surgery.
progression 9. In line with Natesh et al. 7, we consider the
use of powered instrumentation (e.g. microdebrider) a wise       References
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