Accepted Manuscript

Title: The Outcomes of Endoscopic Dacryocystorhinostomy

in Children: A Systematic Review

Author: Federico Maria Gioacchini Matteo Alicandri-Ciufelli

Shaniko Kaleci Massimo Re

PII: S0165-5876(15)00182-2
DOI: http://dx.doi.org/doi:10.1016/j.ijporl.2015.04.023
Reference: PEDOT 7553

To appear in: International Journal of Pediatric Otorhinolaryngology

Received date: 29-1-2015

Revised date: 12-4-2015
Accepted date: 13-4-2015

Please cite this article as: F.M. Gioacchini, M. Alicandri-Ciufelli, S. Kaleci,

M. Re, The Outcomes of Endoscopic Dacryocystorhinostomy in Children: A
Systematic Review, International Journal of Pediatric Otorhinolaryngology (2015),
http://dx.doi.org/10.1016/j.ijporl.2015.04.023

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THE OUTCOMES OF ENDOSCOPIC DACRYOCYSTORHINOSTOMY IN CHILDREN: A

SYSTEMATIC REVIEW

Federico Maria Gioacchini, MD*; Matteo Alicandri-Ciufelli, MD**; Shaniko Kaleci, PhD°; Massimo Re,
MD*

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*Otolaryngology Department, Marche Polytechnic University, Ancona, Italy

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**Otolaryngology Department, University Hospital of Modena, Modena, Italy
°Department of Diagnostic Medicine, Clinical and Public Health University Hospital of Modena Italy

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Corresponding author:

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Dr Federico Maria Gioacchini, MD; Otolaryngology Department, Marche Polytechnic University,
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of Marche: Ospedali Riuniti of Ancona, Via Conca 71, 60020 Torrette, AN, Italy.

Email address: giox83@hotmail.com

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Work phone: 3771525135

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Mobile phone: 3490069191

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Word count: 2495

All of the authors have read and approved the manuscript. None have any financial relationships to
disclose.
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Page 1 of 21
ABSTRACT

OBJECTIVES: To systematically review and discuss the published results about the application of

endoscopic dacryocystorhinostomy in treating children with nasolacrimal duct obstruction.

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METHODS: In October 2014 an appropriate string was run on PubMed to retrieve all relevant

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articles. A cross-check was performed by two of the authors on abstracts and full-text articles found

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using the selected inclusion and exclusion criteria. A non-comparative meta-analysis concerning the

procedures’ rate of success and failure was performed.

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RESULTS: Fourteen studies were identified comprising a total of 346 subjects affected by

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nasolacrimal duct obstruction (unilateral or bilateral). Overall there were 393 surgical procedures,
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all performed with an exclusive endoscopic approach. The average length of follow-up was reported

in twelve studies resulting 15.2 months and ranging from 3 to 27.1 months. On the basis of our
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statistical analysis the mean (95% CI) rate of failure was 0.14 (0.09-0.21). The mean (95% CI) rate
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of success resulted 0.87 (0.80-0.91).

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CONCLUSIONS: Although in young patients the nasal anatomy is more complex and narrow than
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in adults our review showed as the endoscopic dacryocystorhinostomy allows similar results in

terms of success compared to the external approach.

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KEYWORDS: children; lacrimal drainage; epiphora; nasolacrimal duct obstruction; probing;

endoscopic dacryocystorhinostomy.

Page 2 of 21
INTRODUCTION

Nasolacrimal duct obstruction (NLDO) is the most common cause of persistent tearing and ocular

discharge in children occurring in up to 20% of all normal newborns and causing symptoms in up to

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6% of children during the first year of life. [1]

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Congenital NLDO usually results from a failure of canalisation of the distal end of the nasolacrimal

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duct with persistence of a membranous web at the level of the Hasner valve. [2]

The acquired type of NLDO obstruction accounts for a relatively small number of cases of this

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condition in children, and its etiologies have not been investigated completely.

NLDO in children has a high rate of resolution without surgery. Medical treatments consist of

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compression or massage of the nasolacrimal sac and topical antibiotics when a discharge is present.
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However after 12 months of age, the likelihood of spontaneous resolution decreases and most

patients are treated with probing or intubation of the nasolacrimal drainage system. [3, 4, 5, 6, 7]
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Refractory cases of sachal and post sachal obstructions have been historically managed by external
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DCR. This procedure was first described by Toti in 1904 [8] and represents an highly effective

procedure for the correction of a common NLDO in children unresponsive to medical therapy,
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probing(s) and intubation. [9, 10, 11]

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With the advent of rigid nasal endoscopes and fibreoptic light carrier systems, surgical access

through the nasal cavity had been greatly enhanced because of better illumination and
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magnification. [12] Overall, the endonasal approach presents many advantages over the external

one. [13, 14, 15] Unlike external DCR, endoscopic DCR allows the drainage of an obstructed

lacrimal sac and system without a facial incision and subsequent scar. Endoscopic DCR also causes

less surgical trauma to medial canthal and orbital tissue and causes less bleeding than is observed in

conventional surgery. [16] For these reasons during recent years, DCRs in adults have increasingly

been performed endoscopically and studies have shown that the success rates of external and

Page 3 of 21
endoscopic DCRs have been comparable. [17, 18] Nevertheless, the data on endoscopic DCR in

children are limited and the issue about the advantages of its application is still open.

Analyzing the reports published about this topic, our aim was to investigate the outcomes of

endocopic DCR for the treatment of pediatric NLDO.

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MATERIALS AND METHODS

In October 2014, a literature search was performed using the following search string on PubMed:

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 ("Child"[Mesh]) AND "Dacryocystorhinostomy"[Mesh]

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The initial search returned a total of 307 results. Abstracts and titles obtained were screened

independently by two of the authors (FMG and MR) who subsequently met and discussed
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disagreements on citation inclusion.

Inclusion criteria for citations were:

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 Reports presenting cohorts of patients with diagnosis of NLDO

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Exclusion criteria for citations were:

 Studies concerning others techniques than endoscopic DCR

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 Studies describing clearly unrelated pathologies

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Between the 307 articles, 23 met the initial inclusion criteria according to both authors (FMG and

MR), so they were obtained and reviewed in detail by the same two authors, who met and discussed
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disagreements on article inclusion. Inclusion criteria for full text articles and single patients

identified were:

 Sufficient and accurate description of surgical procedure

 Sufficient and accurate presentation of post surgical outcomes

Exclusion criteria were:

 Analysis including groups of adult patients

 Analysis including patients treated with others techniques than endoscopic DCR

Page 4 of 21
A total of eleven studies were excluded because of insufficient data about the surgical procedure

(one study) and post surgical outcomes (three studies) while seven studies were ruled out because

patients treated with others techniques were comprised. A further manual check was performed on

the references included in the articles and two additional studies were identified that met the

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inclusion criteria. The final number of articles included in the present review was identified, and the

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main information was extracted and summarized.

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We performed a non-comparative meta-analysis and the heterogeneity between studies was

assessed by the χ2-based Cochran’s Q statistic test and I2 metric. Heterogeneity was considered

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significant at P<0.01 for the Q statistic (to assess whether observed variance exceeds expected

variance). And for the I2 metric (I2=100% x(Q-df)/Q), the following cut-off points were used: I2=0-

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25%, no heterogeneity; I2=25-50%, moderate heterogeneity; I2= 50-75%, large heterogeneity;

I2=75-100%, extreme heterogeneity. All analyzes were performed using Comprehensive Meta-
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Analysis statistical software, version 2.0 (Biostat, Englewood, NJ, USA).
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RESULTS

After an initial check, full-text retrieval, and manual cross-checking of references included in the
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articles, 14 studies comprising a total of 346 subjects and 393 surgical procedures were chosen for
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analysis (Figure 1). The characteristics of these selected studies are showed in Table 1.

The majority of the included studies were performed with a retrospective cohort design (only two
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reports being prospective). The average length of follow-up was reported in thirteen studies

resulting 15.2 months and ranging from 3 to 27.1 months.

Overall, the number of patients in each study included in this analysis varied from 6 to 71.

Patients' mean age was reported in ten studies varying from 3.9 years to 11.2 years.

Overall 161 NLDOs were congenital while acquired NLDOs resulted 12, but it must be noted that

the majority of reports did not specified the origin of NLDO.

Page 5 of 21
The 2.7 mm and 4 mm nasal endoscopes were equally used by different surgeons. To perform

osteotomy many authors reported the application of a powered diamond burr while others preferred

a Kerrison rongeur or a punch. In two studies (Cakmak et al. and Uysal et al.) a laser technique was

applied.

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Concerning the placement of silicone tube, between eleven articles (comprising overall 297

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procedures) specifying this detail, there were 240 (80.8%) placements.

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No major complications were reported in the articles analyzed. Among minor complications were

described four cases of nasal synechiae, three cases of nasal granulomas, one abscess, one case of

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mild postoperative epistaxis and one important intraoperative bleeding.

In the majority of studies the success was defined as a “complete resolution of the symptoms of

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tearing and discharge”. In four articles the success was considered as a “symptomatic relief” while
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one study defined the surgical outcome on the basis of “ fluorescein dye disappearance test

(FDT)”.
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Statistical analysis
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The rate of total failure in the 14 included studies is illustrate in Figure 2. The total number of
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included procedures was 393. Results were moderate heterogeneity (Q=30.5, I2=57.4%, p=0.004)

and statistically significant. The mean (95% CI) rate of failure was 0.14 (0.09-0.21).
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The rate of total success in the 14 included studies in illustrate in Figure 3. The total number of

included procedures number was 393. Results were moderate heterogeneity (Q=30.7, I2=57.6%,

p=0.004) were statistically significant. The mean (95% CI) rate of success was 0.87 (0.80-0.91).

DISCUSSION

Most of articles reporting outcomes in paediatric lacrimal surgery describe cohorts of patients

affected by refractory congenital NLDO. [13, 19, 20] Nevertheless, other authors reported a

Page 6 of 21
significant percentage of patients affected by acquired paediatric NLDO including cases of

lacrimal fistula and post-traumatic NLDO. [11, 21] Overall between the articles presented in this

review there were eight papers (comprising 173 NLDOs) that specified the type of NLDO. These

had a congenital origin in 161 (93%) cases while resulted acquired in 12 (7%).

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Pediatric NLDO has a high rate of resolution without surgery. In their observational study Paul et

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al. [22] reported as the rate of healing with only medical management by 1 year of age was 80% at

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3 months, 70% at 6 months old, and 52% at 9 months of age. Some studies suggest that delaying

this operation, is associated with higher failure rates.[23, 24, 25] The fibrosis caused by the

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prolonged inflammation in lacrimal drainage system could represent the principal explanation [26,

27] and many studies have shown success rates to be higher at younger ages, especially among

children <2 years of age. [28, 29, 30]

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Outcomes of probing procedure vary between different authors on the basis of patients’ cohorts.

Arora et al. [31] reported an overall success rate of 72% while Honavar et al. [32] reported similar
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results with a success rate of 73%, in a cohorts of patients with 33 months of median age. The
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Pediatric Eye Disease Investigator Group (PEDIG)’s prospective study in 2008 showed a higher

success rate (78%) in a study population with a lower mean age (13.6 months). [25]
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In case of persistent epiphora following probing a nasolacrimal intubation is often performed.

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This procedure involves probing the nasolacrimal duct followed by placement of a silicone tube

stent in one or both canaliculi. The reported success rate is good, ranging from 79% to 97% [5, 26,
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33] and has been reported as being higher for children who undergo surgery between 1 and 4 years

of age, for patients with no acute dacryocystitis, and for cases of mild obstruction. [34, 35]

In case of failure some surgeons may prefer a second or third probing and silicone intubation, while

others opt for a DCR. Described for the first time by Toti in 1904 [8], this procedure is generally

avoided in patients younger than 5 years; however, with an overall success rate of about 90% in

adults [16, 36] and 80% [11, 21, 37, 38, 39] in children, external DCR is considered to be the gold

standard for definitive treatment. The main advantage of the external route is the good anatomical

Page 7 of 21
visualization, allowing precise removal of bone in the lacrimal sac fossa, and accurate suture-

fixated anastomosis of the nasal mucosa and lacrimal sac.

Nevertheless during the last years the endoscopic DCR is gaining popularity in treatment of

children affected by congenital and acquired NLDO because the endoscopic approach ensures

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important advantages compared to the external technique. Firstly it avoids a skin incision, then it

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conserves the medial canthal structures so preserving the lacrimal pump mechanisms, finally it

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allows the apposition of the lacrimal sac and nasal flaps favoring the primary healing process.

A recent meta-analyis performed by Huang et al. [40] analyzed 19 studies investigating the

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differences in terms of success and complications between external and endoscopic DCR in an adult

population. The authors concluded that endoscopic technique presents similar rate of success in

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comparison to external DCR. Nevertheless they noted as the endoscopic approach avoids the risk of
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cosmetically unacceptable scars.

On the other hand pediatric endoscopic DCR presents some disadvantages related to the specific
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type of patients. In fact in paediatric subjects where bony facial abnormalities are common, the
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nasal space might be narrow for endoscopic procedures. [41] Furthermore the Agger nasi cell is not

as well pneumatised as in adults and the identification of the lacrimal eminence and the uncinate
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process can be more difficult in such a narrow space. The inferior turbinates are more bulky and the
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nasal septum can be deviated making more difficult the access to the lacrimal eminence.

Interestingly, despite the technical difficulty to perform this surgery in children, on the basis of data
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obtained through our statistical analysis we may affirm that endoscopic DCR represent a valid

alternative to the external procedure. In fact a success rate of 87% appears completely similar to the

rates of success reported in literature for the external technique (80-90%).

Interestingly the results presented by Jones et al. [42] appear to be much poorer in comparison to

the data presented in the other papers. However this difference may be explained with the presence

of seven patients presenting craniofacial anomaly/syndrome who were treated for uni or bilateral

Page 8 of 21
stenosis (overall eleven procedures). In fact after these eleven operations only one child (9%)

achieved a complete symptoms' resolution.

We also noted a high prevalence of tubing placement (80.8%) in patients of the eleven studies

reporting this information. Silicone tubing is a controversial issue in the literature and appears

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common both for external and endoscopic DCR. Some authors place it in all their cases when others

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use it only in situations in which canalicular stenosis is suspected or in case of revision surgery [13,

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14, 15, 43, 44]. The purpose of nasolacrimal stanting is to maintain DCR ostium patency but there

is evidence that it may rather increase the changes of failure by inciting granulomatous

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inflammation at nasolacrimal fistula site. [45]

To prevent ostium obstruction by healing or granulation tissue the use of antimetabolites agents

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(Mitomycin C; 5-flluorouracil) is considered a possible alternative to tubing. In their recent meta-
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analysis Cheng et al. [46] investigated the real efficacy of Mitomycin C application during

endoscopic DCR. The authors analyzed the outcomes reported in 11 comparative studies (including
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a total of 574 eyes) and concluded that Mitomycin C represents a safe adjuvant in reducing the
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closure rate of the osteotomy. Among the studies grouped in our review we could not find any

application of some chemotherapy during the surgical procedures.

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It must be noted that our study presents some weakness. In our opinion the more important issue is
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related to the scarce number of patients grouped in this review. Secondly between all papers

included in this review there were not studies reporting a comparison group of patients treated with
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different techniques. Another problem regards the mean follow-up time resulting lower than one

year in four articles and totally absent in one study. Also concerning stenting we noted a wide

variability, even within singular studies, about the decision of tube positioning and time of

removing. Moreover in the vast majority of articles the data about outcomes were calculated for all

patients together. So it was impossible to know the specific rate of success for each sub-group of

patients (with or without tube’s placement) and we could not perform any statistical analysis about

the real efficacy of tubing placement.

Page 9 of 21
CONCLUSIONS

In conclusion the endonasal DCR is a valid surgical procedure for children with NLDO resistant to

probing, irrigation and intubation. This review confirmed that, as previously established for adults,

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also when performed in children the endoscopic DCR may allow similar results in terms of

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outcomes compared to the external technique. The possibility to preserve the pump mechanism of

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tearing and the low incidence of complications represents the main advantages of this technique.

It must be noted that endoscopic DCR requires a wide experience in endoscopic sinus surgery and

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obviously the otolaryngologist appear to be the specialists mostly indicated to perform it.

Nevertheless a multidisciplinary team approach comprising also a pediatric ophthalmologist is

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mandatory to obtain the optimal preoperative evaluation and postoperative care.
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[54] Celenk F, Mumbuc S, Durucu C, Karatas ZA, Aytaç I, Baysal E, Kanlikama M.

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Pediatric endonasal endoscopic dacryocystorhinostomy. Int J Pediatr Otorhinolaryngol. 2013

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Aug;77(8):1259-62.

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FIGURE LEGENDS

Figure 1. Flowchart demonstrating study selection.

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Figure 2. Result of meta-analysis with all evaluable studies for failure rate procedure. The point in

the horizontal line represent the effect size of result of each study and horizontal lines represent the
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95% confidence interval of each effect size. The last horizontal line represents the aggregate effect
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size and 95% confidence interval.

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Figure 3. Result of meta-analysis with all evaluable studies for success rate procedure. The point in

the horizontal line represent the effect size of result of each study and horizontal lines represent the
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95% confidence interval of each effect size. The last horizontal line represents the aggregate effect

size and 95% confidence interval.

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Table(s)

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Table 1. Main characteristics and outcomes of the different studies.
Mean
Type n. of n. of Silicone
n. of Mean follow-

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Authors Country Year of performed successful NLDO etiology stent
patients age (yr) up
study procedures procedures placement
(mo)
Congenital Acquired

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VanderVeen [13] USA 2001 R 17 3.9 22 19 20 22 0 22
Berlucchi et al. [14] Italy 2003 R 6 6 7 7 20 7 0 7
Knijnik [47] Brazil 2005 R 24 5.7 27 21 3 n/a n/a 4
UK 2005 R 16 n/a 16 14 n/a n/a n/a n/a

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Marr et al. [48]
Leibovitch et al. [43] Australia 2006 R 21 6 26 24 18 n/a n/a 26
Gupta and Bansal [49] India 2006 P 18 n/a 18 17 8.2 12 6 0
Jones et al. [42] USA 2007 R 34 n/a 43 25 21 40 3 n/a
Eloy et al. [2] Belgium 2009 R 8 4.3 11 11 10.5 n/a n/a 2

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Cakmak and Yildirim [50] Turkey 2010 P 8 11.2 8 7 9 7 1 8
Kominek et al. [51] Czech Republic 2010 R 52 4.1 58 51 17 58 0 54
Al-Nuaimi et al. [52]
Uysal et al. [12]
de Souza et al. [53]
UK
Turkey
India
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2011
2012
R
R
R
16
18
37
6.5
6.1
n/a
17
20
37
14
17
34
12.1
20.5
12
15
n/a
n/a
2
n/a
n/a
14
20
n/a
ep
Celenk et al. [54] Turkey 2013 R 71 8.9 83 77 27.1 n/a n/a 83

NLDO: nasolacrimal duct obstruction; R: retrospective study; P: prospective study; yr: years; mo: months; n/a: not available.
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Figure(s)

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Figure(s)

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Figure(s)

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