Pediatric Terrible Elbow Tetrad: A Case Report and Systematic Review

doi:10.21203/rs.3.rs-4932409/v1

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Case Report

Pediatric Terrible Elbow Tetrad: A Case Report and Systematic Review

https://doi.org/10.21203/rs.3.rs-4932409/v1

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A 6-year-old girl presented with a complex elbow injury, involving a posterior transolecranon fracture-dislocation and a radial neck fracture. Surgical intervention, performed through a posterior elbow approach, revealed an additional fracture of the coronoid process. Stabilization included a retrograde crossed K-wire for the radial neck fracture and a tension band construct (comprising K-wires and suture wire) for the olecranon fracture extending into the coronoid process. This combination of olecranon, radial neck, and coronoid fractures, coupled with a posterior elbow dislocation, constitutes a rare and intricate entity that may be regarded as a “terrible elbow tetrad”.

Elbow Injuries

Pediatrics

Fracture Dislocations

Olecranon Fractures

Radial Neck Fractures

Coronoid Process

Complex elbow fracture-dislocations are relatively rare and pose a diagnostic and management challenge, particularly in children where the presence of a non-ossified cartilaginous component complicates diagnosis. Despite this challenge, managing these injuries in children may be comparatively simpler than in adults due to lower comminution when lesions are appropriately detected and treated.

In adults, the term "terrible elbow tetrad" denotes a specific combination of fractures involving the olecranon, radial head, and coronoid process, often linked with posterior or posterolateral elbow dislocation [1]. This pattern differs from other elbow injuries, such as the "terrible triad," Monteggia equivalents, and transolecranon fracture-dislocations (TOFD). The latter involves an anterior elbow dislocation with an olecranon fracture [2, 3], while the "terrible triad" typically includes posterior joint dislocation with fractures of the radial head and coronoid process. Monteggia equivalents comprise combined injuries resembling classic Monteggia fractures, involving ulnar fractures and radial head dislocation.

Although the "terrible tetrad" has not been described in pediatric cases, we report a fitting case involving posterior elbow dislocation with fractures of the olecranon, radial neck, and coronoid. This report details the case, covering diagnosis and management, and includes a systematic literature review to identify potential injuries falling within this category, outlining treatments and associated complications.

A 6-year-old girl, initially treated at another medical center following a fall from a swing, received a diagnosis of left elbow fracture dislocation. Upon initial examination, she exhibited moderate swelling, elbow deformity, and an inability to move the elbow actively, with palpable crepitation. Distal neurovascular examination yielded normal results. X-rays indicated a posterior elbow dislocation along with fractures of both the olecranon and radial neck (Fig. 1, A-B). She was initially immobilized with a splint and subsequently referred to our pediatric center for further assessment, where a CT scan of the elbow was performed (Fig. 1, C-D).

Urgent surgery was conducted with the patient in the supine position, her limb on a hand table, and under general anesthesia with an axillary block. Despite an initial gentle attempt at closed reduction, this proved unsuccessful (Fig. 2).

During the surgery, which was performed under an upper limb tourniquet, we utilized a posterior elbow approach. We initially identified the olecranon fracture and observed an intra-articular triceps entrapment (Fig. 3A). Following meticulous dissection and the opening of the lateral tricipital retinaculum, we exposed the olecranon tip attached to the triceps, discovering an extension of the olecranon fracture to the anteromedial facet of the coronoid process (Fig. 3B). Subsequently, we visualized the displaced fractured radial head, directly reduced it, and secured it with a 1.2-mm retrograde crossed Kirschner wire (KW) (Fig. 3, C-D). We then reduced the ulnohumeral joint and the olecranon fracture with an extension to the coronoid, fixing them using a tension band construct using 2x1.5-mm KWs directed toward the anterior ulnar cortex and a cerclage with a double Fiberwire No.2 suture (Fig. 3, E-G). We assessed the elbow's stability across the full range of flexion-extension, ensuring unrestricted complete pronation and supination. The annular ligament was repaired with two sutures, and the lateral extensor retinaculum was secured with various single sutures (Fig. 3, H), followed by a layered closure. Intraoperative fluoroscopy images are visible in Fig. 4, A-B.

The patient was initially immobilized with a long arm splint, maintaining the forearm in a neutral position for 3 weeks. At this juncture, the radial neck Kirschner wire (KW) was removed. Following this, the patient utilized a sling for an additional 2 weeks, during which passive-assisted and active mobility exercises were introduced. By the 3.5-month mark, the patient had resumed normal sports activities without reporting any pain or discomfort. Olecranon KWs were removed at 4 months (Fig. 4, C-D). Comprehensive clinical and radiological outcomes are presented in Figs. 5 and 6, with functional scores detailed in Table 1.

Table 1

Scores on functional scales at last follow-up
Pain measured with the Visual Analogue Scale (VAS)	No pain, 0
Mayo Elbow Performance Score (MEPS) · Pain · Motion · Stability · Function TOTAL	· None, 45 points ·Arc > 100, 20 points · Stable, 10 points · Comb hair, 5 points · Feed, 5 points · Hygiene, 5 points · Shirt, 5 points · Shoe, 5 points 100 points
Quick DASH (Disabilities of the Arm, Shoulder and Hand	2.3 points
Upper Extremity Functional Scale (UEFS) · Sleeping · Writing Opening jars · Picking up small objects with fingers · Driving a car more than 30 minutes · Opening a door · Carrying milk jug from the refrigerator · Washing dishes TOTAL	· 0, No problem · 0, No problem · 0, No problem · 10, Not applicable · 0, No problem · 0, No problem · 0, No problem 10/80 = 12.5%

Methods

The systematic literature review adhered to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines [4]. The search spanned three electronic databases from inception to June 5, 2023. Additionally, supplemental searches were conducted by checking the bibliographies of the identified studies to find any further relevant publications. No restrictions were placed on language or publication period. Updated searches were performed on July 23, 2023, and August 7, 2023. The study selection process was performed independently by 3 reviewers. If a study passed the initial screening based on title, abstract, and keywords review, the full text was retrieved for a detailed review against the eligibility criteria (Table 2). Discrepancies were resolved through consensus with a fourth investigator.

Table 2

Eligibility criteria for the systematic review
Inclusion criteria	Exclusion criteria
(1) Case reports or case series (2) Presenting transolecranon fracture dislocation or terrible triad of the elbow (3) In pediatric patients (0–18 years) (4) With fully documented case and imaging studies	(1) Conference abstracts (2) Insufficient information to classify the injury (3) Chronic or neglected injuries (4) Studies that did not meet the inclusion criteria.

The initial database search yielded 145 studies, with 30 duplicates removed, leaving 115 studies for inclusion screening (Fig. 7). Ultimately, 25 articles were included in the review—23 related to TOFD and 3 to the terrible triad. These studies collectively detailed 54 cases (51 TOFD, 3 terrible triad). Characteristics of these studies are outlined in Table 3.

Table 3

Characteristics of the systematic review studies on transolecranon fracture- dislocation and terrible triad of the elbow in children.
Search: ("Transolecranon" OR "olecranon") AND ("Fracture") AND ("dislocation") AND ("pediatric" OR "children") NOT (“supracondylar”)
Study	Age (yr) / Sex	Description of fracture	Treatment
Wilkerson 1993 [5]	7 / M	Anterior TOFD	ORIF with TB construct (2x1.6 mm KW + wire)
Ruchelsman et al. 2005 [6]	7 / M	Anterior TOFD + radial neck fr	OR radial head + 2x1.2 mm KW + annular ligament reconstruction
Guitton et al. 2009 [2]	11 (8–13) / M	Anterior TOFD	ORIF with 4-hole one-third tubular dynamic compression plate
		Anterior TOFD
		Anterior TOFD	ORIF with 6-hole one-third tubular dynamic compression plate
		Anterior TOFD	ORIF with TB construct with screw
Butler et al. 2012 [3]	9 / M	Anterior TOFD + avulsion of the medial epicondyle	ORIF with TB construct (2x1.6 mm KW + No. 2 Ethibonds suture) + ORIF epicondyle with partially threaded, 4.0-mm screw
	4 / M	Open anterior TOFD	ORIF with TB (2x1.6 mm KW + suture)
	8 / F	Anterior TOFD + radial neck fr	ORIF with TB construct (2x1.6 mm KW + wire)
Charter et al. 2014 [7]	8 / F	Anterior TOFD	ORIF with TB construct (2KW + wire)
Tiemdjo et al. 2014 [8]	9 / M	Anterior TOFD + radial neck fr	CR + casting
Wu et al. 2016 [9]	10 / F	Posterior TOFD + coronoid fr + PRUJ dislocation	CR of dislocation + ORIF with TB construct
Bouaziz et al. 2018 [10]	7 / M	Anterolateral TOFD	OR + casting
Patel et al. 2018 [11]	6 / M	Anterolateral TOFD	CR + casting
Miravete-Galvez et al. 2018 [12]	4 / M	Anterior TOFD	CRPF with 2x1.6 mm KW
Arain et al. 2019 [13]	7 / F	Anterolateral TOFD	CR + casting
Ashar et al. 2020 [14]	6 / F	Anterior TOFD + ulnar nerve palsy	ORIF + KW
Batheja et al. 2021 [15]	5 / F	Anterior TOFD	ORIF with TB construct (2x1.5 mm KW + wire)
Yamaura et al. 2021 [16]	7 / M	Anterior TOFD + coronoid avulsion fr	CRPF with KW
Massetti et al. 2021 [17]	7 / F	Posterior TOFD + radial head fr	ORIF with 2 bicortical 1.5 mm KW + ESIN radial head
Karashi et al. 2021 [18]	12 / M	Posterior TOFD + radial neck fr	ORIF with TB construct (2 KW 1.5 mm + wire) + KW fixation for radial head
Yang et al. 2021 [19]	11 / M	Anterior TOFD	ORIF with TB construct
	6 / M	Anterior TOFD + coronoid fr + ulnar nerve injury	ORIF with TB construct
	12 / M	Anterior TOFD	ORIF with KW
	8 / M	Anterior TOFD + coronoid fr + ulnar nerve injury	ORIF with KW
	13 / M	Anterior TOFD	1º ORIF with TB; 2º ORIF with locking plate
	4 / M	Anterior TOFD + coronoid fr + distal ulna & radius fr	ORIF with locking plate
	11 / M	Anterior TOFD	ORIF with locking plate
	9 / M	Anterior TOFD	ORIF with screws
	4 / F	Anterior TOFD	ORIF with locking plate
	8 / F	Anterior TOFD + distal ulna & radius fr	ORIF with locking plate
	13 / M	Anterior TOFD	ORIF with screws
Tomer et al. 2022 [20]	9 / F	Anterior TOFD	CR + casting
Jlidi et al. 2022 [21]	6 / M	Anterior TOFD	ORIF with TB construct (2 KW 1.5 mm + suture)
Zheng et al. 2022 [22]	7 / F	Anterior TOFD + osteochondral coronoid fr	CR + KW fixation + ORIF coronoid and screw fixation
Su et al. 2022 [23]	9 / M	Anterior TOFD	ORIF with KW and TB construct
	7 / M	Anterior TOFD	ORIF with KW and TB construct
	5 / M	Anterior TOFD	ORIF with KW and TB construct
	14 / M	Anterior TOFD + Medial epicondyle fr	ORIF with KW and TB construct
	8 / F	Anterior TOFD	ORIF with KW and TB construct
	6 / M	Anterior TOFD	ORIF with KW and TB construct
	9 / F	Anterior TOFD	ORIF with KW and TB construct
	11 / M	Anterior TOFD	ORIF with KW and reconstruction plate
	13 / M	Anterior TOFD + radial neck fr + coronoid fr	ORIF with KW and reconstruction plate
	14 / M	Anterior TOFD + coronoid fr	ORIF with KW and reconstruction plate
	12 / F	Anterior TOFD + coronoid fr	ORIF with KW and reconstruction plate
	14 / F	Anterior TOFD	ORIF with KW and TB construct
	7 / M	Anterior TOFD	ORIF with KW and TB construct
	8 / M	Anterior TOFD	ORIF with KW and TB construct
	10 / M	Anterior TOFD	ORIF with reconstruction plate
Cateriniet al. 2002 [24]	10 / M	Posterior TOFD + possible coronoid fr	CR and casting
Search: ("terrible triad" OR "elbow triad") AND ("pediatric" OR "children")
Dailiana et al. 2013 [25]	4 / F	Posterior TOFD + radial head fr + coronoid fr	ORIF with nonabsorbable sutures for coronoid and 3 KW
Rooke et al. 2018 [26]	12 / M	Posterior elbow dislocation + radial neck fr + coronoid chondral fr	OR, coronoid fragment removal, anterior capsule reattachment with sutures
Kraft et al. 2022 [27]	10 / F	Posterior elbow dislocation + radial neck fr + possible coronoid fr	ORIF with anterograde KW
TOFD, Transolecranon Fracture Dislocation; Yr, years; M, Male; F, female, ORIF, Open Reduction and Internal Fixation; TB, tension band construct; KW, Kirschner wires; OR, Open reduction; fr, fracture; wks, weeks; PRUJ, Proximal radioulnar joint

The study group comprised 31.5% (17) female and 68.5% (37) male patients, with a mean patient age of 8.7 ± 2.9 years (ranging from 4 years to 14 years). Among cases of TODF, 5 (9.4%) were posterior, 44 (83%) were anterior, and 3 (5.7%) were anterolateral. Of the 3 cases classified as elbow triad, 2 were posterior elbow dislocations, and 1 was a posterior TOFD. Two of these cases reported associated radial neck and coronoid fractures, while the third did not describe the coronoid fracture, though images suggested a potential osteochondral lesion. Non-operative management with closed reduction and casting was employed in four cases (7.4%) while the remaining 49 cases (90.7%) underwent surgical treatment.

The presented case involves a school-age girl with a complex elbow injury, encompassing a posterior elbow dislocation coupled with fractures of the olecranon, radial neck, and coronoid processes (Fig. 8). It is noteworthy that this injury should not strictly be categorized as a TOFD as by definition these are characterized by an anterior elbow dislocation [2, 3, 25]. Furthermore, it does not strictly align with the conventional concept of a terrible triad due to the involvement of the olecranon. Unlike a Monteggia equivalent injury, which typically involves an ulnar fracture with a radial head or neck fracture, this case is distinguished by the presence of humero-ulnar joint dislocation. Consequently, it does not neatly fit into any of these three established categories and necessitates consideration as a distinct entity: a "terrible elbow tetrad".

Upon a detailed review of the included studies in the systematic review and their radiological images, we identified three pediatric cases that could be classified as a terrible elbow tetrad, in addition to the present case (Table 4). Although one of them satisfies all the characteristics of this injury, it was categorized as a terrible triad [25]. We argue that the involvement of the olecranon precludes strict classification as a terrible triad. In the other two cases, despite being considered TOFD with a radial head fracture, rather than the typical anterior elbow dislocation we observed a posterior elbow dislocation that is usually associated with a coronoid fracture [17, 18]. The lack of recognition of the coronoid fracture, despite the posterior elbow dislocation, is likely due to the cartilaginous nature of the coronoid. In this context, these cases resemble our case, where neither the X-ray nor the CT scan revealed the coronoid fracture, only becoming evident intraoperatively. This is considered an occult fracture and aligns with the criteria for osteochondral injuries, referred to as TRASH injuries of the elbow in children, standing for "The Radiographic Appearance Seemed Harmless" [28]. These injuries represent osteochondral lesions not readily apparent on imaging tests and, if not adequately treated, can lead to long-term chronic consequences, as outlined by Zheng et al [22].

Table 4

Cases published in the literature that can be considered as a terrible elbow tetrad in children
Study	Age / Sex	Description of fracture	Treatment	Postoperative treatment	Implant removal
Dailiana et al. 2013 [25]	4 / F	Posterior TOFD + radial head fr + coronoid fr	ORIF with nonabsorbable sutures for coronoid and 3 KW	Long arm splint 6 wks	At 5 wks
Massetti et al. 2021 [17]	7 / F	Posterior TOFD + radial head fr + possible coronoid fr	ORIF with 2 bicortical 1.5 mm KW + ESIN radial head	Long-arm cast 4 wks	At 4 wks
Karashi et al. 2021 [18]	12 / M	Posterior TOFD + radial neck fr + possible coronoid fr	ORIF with TB construct (2 KW 1.5 mm + wire) + KW fixation for radial head	Long arm splint 3 wks	At 6 months
Current case report 2024	6 / F	Posterior TOFD + radial neck fr + coronoid fr	ORIF with tension band construct (2 KW 1.6 mm + suture) + KW fixation for radial head	Long arm splint 3 wks	4 months
TOFD, Transolecranon Fracture Dislocation; Yr, years; M, Male; F, female, ORIF, Open Reduction and Internal Fixation; TB, tension band construct; KW, Kirschner wires; fr, fracture; wks, weeks

Comparing the treatment of our case with the other three published cases, it is observed that all of them underwent open reduction through a posterior elbow approach. Olecranon fixation was performed with KW in one case [25], and with a tension band construct or a variation of it in the other two [17, 18], which aligns with our case. Regarding the radial head fracture, one case was fixed with elastic intramedullary nailing according to Metaizeau [17], while another case used an antegrade KW [18], and non-operatively in another case. In contrast, in our case we employed direct reduction and retrograde KW. The coronoid fracture was addressed through open reduction and fixation with nonabsorbable sutures in the only case where it was mentioned [25]. In our case, due to its attachment to the proximal olecranon fragment, when the olecranon was reduced and fixed, the coronoid was also reduced and remained stable. Immobilization duration ranged from 3 to 6 weeks in these cases, and the KWs were removed at different times: 4 and 5 weeks in two cases and at 6 months in another. In our case, KW removal was performed at 4 months. While no complications were reported in these cases, our patient experienced a slight extension limitation of -10º and a pronation limitation of -5º, which did not impair function.

We propose that a distinct entity, the "terrible elbow tetrad," should be considered when managing pediatric elbow fracture dislocations, given its unique implications for treatment. In contrast to the elbow terrible triad, we contend that this entity can be addressed through a posterior approach, utilizing transolecranian access. However, unlike TOFD, usually linked with anterior elbow dislocation, the presence of a posterior dislocation should prompt consideration of a potential occult coronoid fracture, warranting treatment for elbow stabilization.

Funding: None of the authors have received financial payments or other benefits from any commercial entity related to the subject of this article.

Conflict of interest: The authors declare that they have no conflicts of interest relevant to the content of this article.

Ethics approval: This study was conducted in accordance with the principles of the Declaration of Helsinki. Institutional Review Board (IRB) approval was not required for this case report.

Consent to participate: Informed written consent was obtained from the patient’s family before submission.

Consent for publication: The patient and his parents were informed that data concerning the case would be submitted for publication, and they provided consent.

Availability of data and material: This manuscript does not report data generation or analysis.

Code availability: Not applicable.

Authors' contributions: All authors, M.G.O, H.G.D, S.M.A, and I.M.C made substantial contributions to the conception or design of the work; J.P.B, M.S.B, R.M.O, I.V.P made substantial contributions to the acquisition, analysis, or interpretation of data; .G.O, H.G.D, S.M.A, and I.M.C drafted the work or revised it critically for important intellectual content; M.G.O, H.G.D, S.M.A, J.P.B, M.S.B, R.M.O, I.V.P AND I.M.C approved the version to be published; M.G.O, H.G.D, S.M.A, J.P.B, M.S.B, R.M.O, I.V.P AND I.M.C agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Reporting guidelines:

Authors have adhered to the recommendations for the accurate reporting of information in case reports that resulted in the CARE (CAse REport) Statement and Checklist.
This systematic review was performed in accordance with the Cochrane Collaboration and the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) statement and the Cochrane Handbook for Systematic Reviews of Interventions latest methodological guidance.

Acknowledgments: None

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