Does Early Treatment of Paediatric Orbital Fracture Offer Any Advantage in Terms of Post-Operative Clinical Outcomes

Abstract

Background

Trapdoor fractures commonly occur in children below 6 years of age. The high resiliency of bone, pneumatisation of sinuses and other factors lead to entrapment of muscle and/or soft tissue which undergoes ischaemic changes leading to residual diplopia. The timing of intervention in children ranges from 24 h to greater than 2 weeks. Early surgical intervention is particularly indicated in cases of Oculocardiac reflex.

Methods

A Prisma guided systematic review of literature was conducted with no filters on language till September 2020. Studies on paediatric orbital fractures with data on timing of intervention and clinical outcomes were considered eligible for the review. The Oxford Level Of Evidence was used to assess the strength of individual studies.

Results

A total of 19 studies (18 English, 1 French) were selected; except for one study all were retrospective series. The timing of intervention ranged from 24 h to more than 1 month. Most of the studies agreed that orbital fractures in children should receive early intervention preferably within 2 weeks. In case of white-eyed blowout fracture, oculocardiac reflex and trapdoor fractures with muscle entrapment surgical intervention should be carried out within 24–48 h.

Discussion

Children presenting with facial injuries should be thoroughly examined for signs of muscle entrapment, diplopia, nausea, vomiting and bradycardia. If present these should receive early intervention. In cases with no signs of oculocardiac reflex and muscle entrapment a treatment within 2 weeks is recommended. If diplopia is mild or resolving with minimal hypoglobus and enophthalmos a wait and watch policy should be carried out.

Introduction

The orbit acts as a protective shield to the globe and as such is involved in a number of midface fractures [1]. The fracture of the orbital floor is termed as pure blow out fracture and in conjunction with the rim is termed as impure blow out fracture [2]. A trapdoor fracture is a subtype of blow out fracture where because of the resiliency, the orbital floor hinges inferiorly, herniating the muscle and soft tissue and recoiling back entrapping the tissues with it [3, 4]. The inferior oblique and rectus are the most commonly involved muscles causing restriction in extraocular muscle movement in upward gaze and diplopia [3]. White-eyed blowout fracture first described by Jordan is a pure orbital blow out fracture, may be linear or hinged which presents itself in a benign form but requires immediate attention [5, 6]. Persistent diplopia which accounts for > 50% of cases of WEBOF (White-Eyed Blowout Fracture), may resolve ranging from 25 days to 18 months with a longer recovery time in children < 9 years of age [5]. Putterman[7] in 1974 advocated conservative approach towards the management of orbital fractures contrary to Smith and Regan’s operative approach. [8, 9] Controversy also exists on the indications for orbital exploration. Burnstine [10] defined the indications for orbital exploration. An immediate intervention is required in cases of WEBOF and clinical and CT (Computed Tomography) evidence of muscle entrapment with Oculocardiac Reflex (OCR). An intervention within 2 weeks is required in cases of symptomatic diplopia, persistent hypoglobus or enophthalmos and hypoesthesia. In cases of minimal diplopia with no motility restrictions, enophthalmos or hypoglobus an observation usually suffices. Almost similar indications were given by Gart [2] and Dubois [11].

Our present systematic review focuses on the timing of intervention especially in paediatric cases, as these are the case which most frequently requires immediate intervention. Our study would assess the effect of timing of intervention on post-operative functional and aesthetic outcomes, specially diplopia and enophthalmos in addition to nerve paraesthesia, vision and motility restrictions. Finally, we would give a guideline on the timing of intervention for the management of orbital fractures in children.

Methods

Search Strategy

The current systematic review has been prepared according to the equator guidelines (https://www.equator-network.org) and Prisma Statement (http://prisma-statement.org/) [35]. The study has been registered with Prospero: CRD42020203728 (https://www.crd.york.ac.uk/PROSPERO/).

Eligibility Criteria

The PICO questionnaire [36] has been used to assess the eligibility of the studies.

With regards to the focus question: “what is the ideal timing of intervention for Paediatric orbital fractures?” Electronic (MEDLINE (PubMed), https://www.ncbi.nlm.nih.gov/pubmed/; EMBASE,

https://www.embase.com/; and Cochrane database http://www.cochranelibrary.com, clinicaltrials.gov, ctri.nic.in) and manual literature searches (Hand searches were done where articles or abstracts were not available electronically) were carried out for studies on paediatric orbital fractures evaluating the effect of timing of intervention on post-operative outcomes (functional: Diplopia, vision, nerve paraesthesia, motility restrictions; Aesthetic: Enophthalmos). There was no restriction on study language. Studies with adequate data on timing of intervention and post-operative outcomes until September 2020 were included. Studies on both adult and paediatric age groups were included provided satisfactory data were available on children or adolescents. Individual case reports, review articles, commentaries, cadaveric and animal studies were excluded. The search terms included (("orbital fractures"[MeSH Terms] OR orbital fracture[Text Word]) AND ("child"[MeSH Terms] OR children[Text Word])) OR (("orbital fractures"[MeSH Terms] OR orbital fracture[Text Word]) AND ("paediatrics"[MeSH Terms] OR paediatric[Text Word])) OR (("orbital fractures"[MeSH Terms] OR orbital fracture[Text Word]) AND ("adolescent"[MeSH Terms] OR adolescent[Text Word])) OR ("orbital fractures"[MeSH Terms] OR orbital fracture[Text Word]).

Study Selection

Two reviewers screened all identifiable titles and abstracts independently. In addition, the reference lists of the subsequently selected abstracts and the bibliographies of the systematic reviews, human randomised and non-randomised controlled trials and prospective and retrospective cohort studies were searched manually. For studies appearing to meet the inclusion criteria, or for which insufficient data in the title and abstract was available, the full text was obtained. Disagreements were solved through discussion between the reviewers. The inter-rater reliability was assessed using Cohen’s Kappa; values ≤ 0 indicated no agreement, 0.01–0.20 as none to slight, 0.21–0.40 as fair, 0.41–0.60 as moderate, 0.61–0.80 as substantial and 0.81–1.00 as perfect agreement. Finally, the full-text evaluation of the remaining publications was done using the above-listed inclusion and exclusion criteria. The oxford level of evidence 2011 was used to assess the strength of each study. Quality assessment of the selected studies was executed by Newcastle–Ottawa scale. Scale was applied for cohort studies to judge each included study on selection of studies, comparability of cohorts, and the ascertainment of either the exposure or outcome of interest. Stars were awarded such that the highest quality studies were awarded up to nine stars. The Oxford Level of Evidence was used to assess the strength of the studies. [12, 13]

The Oxford 2011 Levels of Evidence

Level Category of evidence.

I SR (with homogeneity) of RCT.

Individual RCT.

II SR (with homogeneity) of cohort studies.

Individual cohort study (including low-quality RCT. For example.

 < 80% follow-up).

“Outcome” research; ecological studies.

III SR (with homogeneity) of case–control studies.

Individual case–control study.

IV Case series and poor-quality cohort and case–control studies.

V Expert opinion without explicit critical appraisal, or based on.

physiology, bench research or first principles.

SR = systematic review, RCT = randomised controlled trials.

Results

Figure 1 describes the study selection process according to the Prisma Guidelines. Cohen’s Kappa coefficient was applied at each step. Tables 1 and 2 describe the individual study characteristics and surgical outcomes. All the selected studies were of moderate quality. Non-English studies were translated by two independent translators blinded to the outcome. A Kappa value of 0.81–1.00 indicated definitive agreement. A total of 19 studies were included in this systematic review from 1998 to 2018. A total of 883 children and adolescents were evaluated in this study. Except for a single study all were retrospective studies or retrospective case series. The most common surgical approach used was transconjunctival followed by subciliary. Medpor and titanium mesh were the two most commonly used implants in addition to supramid, gelfilm, rapidsorb, PPE (porous polyethylene) and PDS (polydioxanone). The timing of intervention from injury to surgery was varied, ranging from 24 to 48 h for WEBOF or trapdoor fractures to greater than 1 month even a year in a single study. Most of the studies agreed that trapdoor fractures and white-eyed blowout fractures should receive early intervention. However, the timing of early intervention is varied ranging from 6 h to 3 days. Some studies such as Amrith et al. [17], Yang et al. [15] found no difference in outcome when non-trapdoor fractures were operated on a delayed basis compared to early intervention. Road traffic accidents (RTA) were associated with increase in likelihood for extra ocular motility (EOM) restrictions than other aetiologies. Younger children (< 6 years of age) are associated with an increased likelihood of post-operative diplopia and enophthalmos. Coon et al. [27] stated the indications of orbital exploration, early intervention should be sorted for clinical and CT evidence of muscle entrapment. Patients with larger defects can be managed later. There is an amplified probability of residual diplopia if orbital fractures are treated beyond 14 days, Su et al. [28].

Fig. 1.

study selection for review

Table 1.

Study characteristics of individual studies

Authorref	Type of study	Type of fracture	Surgical technique/reconstruction	No. of patients	Timing of intervention
Jordan et al. 19986	RCS	WEBOF		20	Variable 2 days–20 days Immediate: Early: 2–3 weeks Delayed: > 3 weeks
Egbert et al. 200014	RCS	Orbital fractures	Transcutaneous/transconjunctival Implants: silastic, Teflon, bone graft and gelfilm	34	12 patients < 7 days 13 patients: 8–14 days 5 patients: 15–21 days 3 patients: 3–4 weeks
Yoon et al. 200315	RCS	Orbital fractures/trapdoor	Transconjunctival Implants: silastic sheet, medpor	44	Mean: 12.8 days (1–57)  < 1 week 1–2 weeks  > 2 weeks
Lane et al. 200716	RCS	Orbital fractures (WEBOF)	Subciliary/transconjunctival approach	30 patients	WEBOF: mean 6.4 days Open fracture: 15.6 days
Amrith et al. 201017	RCS	Blowout fractures	Transconjunctival approach ± cantholysis. Implants used: supramid, medpor, titanium mesh. Author prefers medpor	69 Children: 13 Patients with Age < 16 years were considered children	Immediate < 1 week Delayed > 1 week
Gerbino et al. 201018	Retrospectives	Orbital trapdoor fractures, linear fracture (Ia) and hinged (Ib). Linear fractures were more common	Subciliary approach Implants used: dura, heterologous bone graft, lactosorb	24 patients Age (6–16 years)	12 patients: < 24 h.(immediate treatment) 8 patients: 24–96 h (early treatment) 4 patients: > 96 h (late treatment)
Wang et al. 201019	Retrospective	Orbital fractures	Subciliary/Transconjunctival approach Implants: supramid or medpor	41 patients	≤ 2 days  ≤ 2 weeks  ≤ 1 month  > 1 month
Carroll et al. 201020	RCS	Orbital fractures	Transconjunctival approach Implants: Medpor, titanium	20	Mean: 14 days Median: 6 days Range: (0–113 days)
Yvonnet et al. 201221	Retrospective	Orbital fractsures	Subciliary Implant: PDS	34	6 h: ophthalmoplegia  < 48 h: persistent diplopia
Neinstein et al. 201222	Retrospective	Orbital floor fractures	Implants: PPE, titanium mesh, resorbable mesh	18 patients	Mean: 9.7 days(1–45 days)
Gerber et al. 201323	Retrospective	Orbital floor fractures (trapdoor and open)		24 patients	2 days (0–15)
Su et al. 201524	Retrospective	Orbital blowsssout fractures	Transconjunctival approach with lateral canthotomy or cantholysis. Medpor, HA and rapidsorb were used as implants	83	Median time interval between injury to surgery: 20 days (7d–1 year)
Heggie et al. 201525	RCS	Isolated orbital fractures	Transconjunctival Implants: gelfilm	22	< 24 h 4th day 10th day
Yang et al. 201526	Prospective	Orbital wall fractures	Transconjunctival approach Implants: medpor	25	Group 1: < 24 h Group 2: 24–72 h Group 3: > 72 h
Coon et al. 201627	Retrospective	Orbital fractures	Implants: PPE, titanium mesh	150	90% (< 3 weeks) 37% (< 48 h) 10% (> 3 weeks)
Su et al. 201628	RCS	Orbital fractures	Implants: medpor, HA and rapidsorb	135 patients	Early: ≤ 14 days Delayed: > 14 days
Mehta et al. 201729	RCS	Orbital fractures	Implants: supramid, medpor, gelfilm	28	≤ 24hours  > 24 h
Su et al. 201830	Retrospective	Orbital trapdoor fractures	Implants: Medpor, HA, rapidsorb	30	Group A: (5–60 days; mean:16.7 days) Group B: (15–60 days; mean: 24.2 days)
Barh et al. (2018)31	Retrospective	Orbital fractures	Transconjunctival Transcaruncular Implants: PPE, titanium, x ray plate, polycaprolactone	52	Median: 9 days (0–337 days)

RCS retrospective case series, WEBOF white-eyed blowout fracture, HA hydroxyapatite, PPE porous polyethylene, PDS polydioxanone

Table 2.

Follow-up and final outcome of individual studies

Author	Follow-up	Results/outcome
Jordan et al. [6]	Variable	Discussed a new subset of patients: Young patients with clinically benign presentation but restriction in upward or downward gaze with pain. CT scan showing little or no bony displacement with teardrop soft tissue herniation. These patients underwent early surgical exploration (2–4 days) had better functional and aesthetic outcome as compared to delayed treatment
Egbert et al. [14]	Median: 6 months. (1 day- > 1 year)	Patients with trapdoor fractures had significantly more ocular restrictions than other variants. There was a significant improvement in diplopia in patients who had surgery within 7 days compared to patients who had within 14 days. There was no difference however in patients receiving surgery within 1 month
Yoon et al. [15]	Mean 5.3 months (3–21)	There was a significant improvement in diplopia among patients who underwent surgery within 5 days. However, there was no difference in supraduction limitation amongst the groups
Lane et al. [16]	6 months	Post-operative diplopia was more in the WEBOF group. No patients experienced diplopia in primary gaze
Amrith et al. [17]	> 6 months	Children had undergone earlier treatment (1.9 ± 1.3 weeks) compared to adults (4.6 ± 3.3 weeks). The median pre-operative motility score was 3 and post-operative was 1 (positive values indicate poorer scores). The final score was − 2. 84.6% of paediatric patients had good outcome and no children had poor outcome. There was no difference in delayed versus immediate intervention (p value = 0.23). Orbital floor fractures had higher motility scores than other sites, although insignificant. In general RTA was associated with poorer outcome compared to other aetiologies especially Assault. A significant improvement in enophthalmos was noted following surgery. (p value < 0.001). no difference in motility with regards to the type of implant used. Indications for orbital exploration: Burnstine criteria
Gerbino et al. [18]	36 months	Children who underwent surgery < 24 h had only 1 patient with residual post-operative diplopia compared to 37.5% in the early treatment group and 100% in the late group. Success rate was 91.7% in early group, 62.5% and 0 in the early and late groups, respectively. Diplopia was more frequent In linear fractures. Authors considered paediatric trapdoor fractures as emergency requiring early intervention
Wang et al. [19]	Mean: 6.5 months	A significant improvement in motility tests was seen in the early, immediate and within 1 month of surgical repair. No patient showed complete improvement In diplopia when surgery was performed more than 1 month
Carroll et al. [20]	Median: 2 months	No statistical assessment has been made. In general persistent diplopia was present in cases who received early surgery as well as delayed treatment. Diplopia was only present in extreme gaze
Yvonnet et al. [21]	1 year	Trapdoor fractures were most common. Residual diplopia was related timing of intervention > 12 h, entrapment of muscle or fat. No patients had residual enophthalmos
Neinstein et al. [22]	Mean: 15.4 months (6–36 months)	Early timing of intervention resulted in significantly lower chances of motility restrictions. Median timing for early intervention being 2 days
Gerber et al. [23]	First follow-up: 10 days Longest follow-up: 396 days	Trapdoor fractures had more complications than open fractures. Delay in treatment increased complications. Authors recommended orbital exploration within 12–24 h in trapdoor fractures. open fractures should ideally be treated with 3 days to injury
Su et al. [24]	18.2 months (12–72)	The incidence of trapdoor was significantly higher in children < 6 years. Study favoured early intervention in the young age group to prevent residual post-operative diplopia
Heggie et al. [25]	1–18 months	Except for one case where residual diplopia remained (operated on 10th day), diplopia was resolved in all other cases. Maximum time was 12 weeks for resolution of diplopia and minimum was 7 days. Enophthalmos was either minimal or nil
Yang et al. [26]	Mean: 90 days	Post-operatively there was no difference in EOM movement in either of the groups. The relation between interval to surgery and post-operative EOM motility was also insignificant. The average period of full recovery of EOM was 50.9 days
Coon et al. [27]		Trapdoor fractures be treated within 24 h Indications for exploration: 1. Clinical and CT evidence of muscle entrapment (early treatment) 2. Larger defect with enophthalmos (can be operated later) Patients with neither of the above may require intervention (preserve conjugate gaze and late enophthalmos)
Su et al. [28]	Mean: 13 months	In case of trapdoor fractures, younger age (< 6 years) and muscle swelling were significantly associated with residual diplopia. In non-trapdoor fractures, in addition to age and muscle involvement timing of intervention played a significant role in residual diplopia. There was a significant increase in residual diplopia > 14 days of surgical intervention particularly in non-trapdoor fractures
Mehta et al. [29]		No difference in overall post-operative ocular motility measurements between two groups. Operating within 24 h decreases the chances of residual diplopia, although not significantly differently from the delayed treatment group
Su et al. [30]	Mean: 16.4 months	Group A: muscle entrapment. Group B: soft tissue herniation Patients in Group A had significantly better resolution of diplopia compared to Group B
Barh et al. [31]	Mean: 124.8 days	Patients operated within 15 days had complete resolution of diplopia

CT computed tomography, WEBOF white-eyed blowout fracture, RTA road traffic accident, EOM extraocular muscle

Discussion

The key results of individual studies are tabulated in Table no: 2. Liner trapdoor orbital floor fractures are characterised by minimal bony displacement and tissue herniation. The entrapment of the musculature particularly occurs behind the globe due the close proximity of the musculature to the bony orbit, Gerbino et al. [18]. This linear variant of trapdoor fracture is age related, more common in children < 12 years of age.[32]. However, recent evidence suggests that this variant of fracture is not exclusively restricted to children. [33] Trapdoor fractures in children are likely to present with nausea, vomiting and bradycardia. Delayed treatment may be associated with likelihood of ischaemic changes leading to residual diplopia. Trapdoor fractures with evidence of muscle entrapment should be treated in an emergency basis. Open type blow out fractures should also be treated within 3 days to injury in contrast to the established Burnstine criteria, Gerber et al. [23]. Blaisdell stated that irreversible ischaemic damage to skeletal muscles starts within 3 h of damage and completes after 6 h [34]. Similar situations may also arise in extraocular muscles, leading to irreversible damage, fibrosis and persistent diplopia. This led to the recommendation of an early surgical intervention, Mehta et al. [29].

Limitations

• Of the total 19 included studies except one, all the other studies are either retrospective studies or case series. The Oxford Level of Evidence for our selected studies were either of categories III or IV. None of the studies are clinical trials.

• There is considerable variation in surgical approaches and implants used. A uniform comparison of the effect of surgical approach and implants used on post-operative clinical outcomes could not be made.

• Lack of uniformity in the timing of intervention. The time defined as immediate, also lacks uniformity. Some studies defined early intervention as within < 6 h, some within < 24 h and some within < 48 h. The timing of early intervention is also variable; early intervention varies from < 7 days to < 3 weeks. The term delayed intervention also lacks homogeneousness. Some studies considered delayed treatment > 45 days, some > 3 weeks and in certain studies the treatment in delayed group has been deferred for months.

• There is lack of uniform reporting and reporting tools for post-operative clinical outcomes like diplopia, enophthalmos, hypoesthesia, vision. The different grading tools and their gradations are different. All studies did not report all the parameters. This could lead to bias in the inference of our review.

• Finally, the indications of orbital explorations have been different in many studies. There have been no universal indications for orbital explorations.

Interpretation

Jordan et al. [6] divided the management of orbital fractures according to two schools of thought., one advocating early surgical intervention < 2 weeks and the other, wait and watch. The indications of early intervention are symptomatic diplopia with positive FDT (forced duction test) and CT evidence of muscle entrapment, diplopia which does not improve over 2 weeks, enophthalmos > 3 mm, > 50% orbital defect, associated fractures of the midface and significant hypoophthalmos. The conservative approach should be undertaken in cases of resolving or minimal diplopia, minimal enophthalmos or hypoophthalmos, etc. The compartment syndrome that results from the recoil of bone in case of trapdoor fractures and resultant ischaemia may be detrimental to the orbital tissues if early release is not planned. Putterman et al. [7] stated that most of the time it is the orbital fat that gets entrapped instead of the muscle per se and residual diplopia can be managed adequately even after 4 months. Lane et al. [16] stated that there is an increased incidence of orbital roof fractures compared to floor in young children; incomplete pneumatisation of frontal sinuses, greater midfacial adipose and a larger cranial to orbital ratio are a few contributors. With increase in age the fracture pattern tends to mimic the adult outline. In children the increased elasticity of the bone entrapment is more common leading to compartment syndrome along with oculocardiac reflex clinically manifesting as nausea, vomiting and bradycardia with motility restrictions requiring emergent intervention. Studies by Jordan [6], Egbert [14] and Grant [3] stated the single most important factor contributing to surgical delay was referral to an Ophthalmologist competent to perform the repair. Heggie et al. [25] stated that CT can have false negative results and hence muscle entrapment should always be checked clinically in addition to CT. MRI has proved to be effective than CT in cases where CT scan is non-confirmatory.

Conclusion

In children consideration should be given for early repair. Ideally paediatric orbital fractures should be repaired within 2 weeks to prevent residual Diplopia and Enophthalmos. Children presenting with any facial injury should be specifically examined on the below-mentioned points:

1. Thorough ocular examination in all directions of gaze and specifically on any sort of Diplopia.

2. Forced Duction test (EOM motility examination) and a CT scan to rule out soft tissue and muscle entrapment.

3. Rule out clinical signs of Oculocardiac reflex (nausea, vomiting and bradycardia).

If any of the above-mentioned points turn out be positive an immediate consultation with an Ophthalmologist is necessary for evaluation and opinion following which surgical intervention may be considered. WEBOF or Linear Trapdoor fractures when diagnosed, necessitates definitive surgical intervention ideally within 24 h or maximum within 3 days. Our study finally concludes that further prospective studies and clinical trials are necessary with a standardised timing protocol to compare immediate, early and delayed reconstruction of orbital floor fractures. We tabulate our guidelines in Table 3.

Table 3.

Indications of Paediatric orbital explorations based on timing of intervention

< 24 h	< 72 h	< 2 weeks	> 2 weeks/wait and watch
1. Retrobulbar haemorrhage 2. White-eyed blowout fracture 3. Linear trapdoor fractures 4. Fractures with clinically motility restrictions and CT evidence of muscle entrapment 5. Oculocardiac reflex	1. Open door fractures 2. Hinged type trapdoor fractures	1. Significant diplopia 2. Hypoglobus 3. Enophthalmos > 3 mm 4. Floor fractures > 2cm2/ 50% 5. Blow in fractures	1. Resolving diplopia/minimal Diplopia 2. Linear fractures without clinical or CT evidence of muscle entrapment 3. Mild or minimal Hypoglobus or Enophthalmos

Funding

Study and authors did not receive any form of funding.

Compliance with Ethical Standards

Conflict of interest

None declared.

Patient Consent

Not applicable.

Ethical Clearance

Not applicable.