Paper
Design
Number patients
Details/results
LoE
Chandran et al. [9]
Retrospective comparative study
12
2 no treatment
10 patients underwent operative treatment (median age in both groups was 5 years):
Group A (five patients): excision + iliac BG + fully threaded pin
Group B (five patients – one with bilateral pseudarthroses): excision + iliac BG + reconstruction plate
2 failures of surgery – both in group A: one with non-union, one with wound breakdown and infection
III
Elliot and Richards [10]
Case series
2
Both patients (both aged 5 years) were treated surgically with resection, Tutobone (bovine cancellous xenograft) and IF (with 3.5 mm reconstruction plate)
Both failed with ‘significant osteolysis and failure of incorporation of the graft’
IV
Persiani et al. [11]
Retrospective comparative study
17
All 17 were treated surgically; 12 with BG (iliac crest); 9 with plate fixation, 8 with Kirschner wire fixation
Operative age {mean (range)} was 5.8 (4–7.5) years and 6.4 (5.25–7.6) in the plate and K-wire groups respectively
Five patients (4 with plate, 1 with K-wire) required a secondary procedure in their grading system, 11 patients achieved a good result (4 with plate, 7 with K-wires), 3 patients achieved a fair result (2 with plates, 1 with K-wire), 3 patients had a poor result (all with plate)
III
Ullot Font et al. [12]
Case series
9
4 no treatment
5 treated with resection + iliac BG + IF (4 with plate and one with threaded pin); mean operative age, 8.4 years; range 5–14 years
One patient (the one with threaded pin) required early removal metalwork because of protrusion
IV
Ettl et al. [13]
Case series
3
All 3 were treated surgically (operative ages: 4, 6, 8 years) by resection + BG + reconstruction plate; mean follow-up 44 months
No failures of surgery
IV
Lorente Molto et al. [14]
Case series
6
1 no treatment
5 treated surgically (1 bilateral; ages 18 month to 4 years) with BG + IF with K-wire
All healed by 6–8 weeks
IV
Cadhilac et al. [15]
Retrospective comparative study
25
8 treated non-operatively
17 treated surgically (at mean age 6 years 4 months; mean age at end follow-up 11.5 years) with resection and internal fixation with wire or plate: BG [6], no BG [1]
Non-union: 0/9 in BG group, 3/8 in no BG group
III
Koster et al. [16]
Case series
2
2 treated operatively by resection + BG + IF with K-wire
Both successful, with union at 8 weeks
IV
Price and Price [7]
Case series
2 (father and daughter)
Father previously operated on aged 10 years – unsuccessfully
IV
Schoenecker et al. [17]
Case series
5
All 5 treated surgically: resection, BG and IF with plate for all (BG: 4 from iliac crest, 1 local)
Consolidation at average 3 months for all; MW removed at average 16 months post-op; average FU 4 years; all full function
IV
Grogan et al. [18]
Case series
8
All 8 were treated operatively [of note, at operation, 6 of the 8 were <2.5 years age; one was 5 years; one was 6 years]:
Resection with maintenance of periosteal sleeve [there was no BG (though resected bone was placed around the approximated bon ends); no IF was used except for a loop of absorbable suture, threaded through bicortical drill holes on either side of the defect, to ‘loosely bring the bone ends togther’].
All were fully healed by 14 weeks
IV
Schnall et al. [19]
Case series
6
All 6 were treated surgically by resection, BG and IF [5 with plate (mean operative age 10 years, range 4–15.5 years), 1 with wire (4.5 years)]
All healed; no surgical failures
IV
Quinlan et al. [20]
Case series
4
1 no treatment
No surgical failures
IV
Toledo and MacEwen [26]
Case series
10
6 no treatment
4 treated surgically (ages not ascertainable from manuscript): excision + BG + Steinmann pin
1 acute neurological compromise (termed ‘massive neuropraxia of the brachial plexus’) necessitating immediate removal of internal fixation
IV
Ahmadi and Steel [22]
Case series
5
3 no treatment
2 treated surgically (operative ages 5 and 6 years) with excision, BG + IF (IM pin)
No surgical failures
IV
Gibson and Carroll [6]
Case series
27
13 no treatment
14 treated surgically (mean operative age, 10 years.; range, 2–19 years): 11 BG, 8 with K-wires and 1 with compression plate (one unspecified type of surgery)
3/14 failure of surgery
IV
Wall [23]
Case series
5
Conservative treatment recommended
IV
Owen [24]
Case series
33
13 no treatment
20 treated surgically:
(16 BG, 4 excision; 10 with wire or Rush pin)
1/20 non-union
IV
Alldred [3]
Case series
9
4 no treatment
5 treated surgically (age range 2–16 years): resection [9] + IF (IM pin (3), encircling wire [22]) + BG (4; iliac (1), tibial [22], rib [22], local chips [22])
1 treatment failure in 2 year-old; and 1 further non-union (presumably intentional, in 16 year-old managed by resection alone)
IV
What Is the Natural History of Congenital Clavicular Pseudarthrosis?
The natural history of this condition remains unclear in absolute terms, though some lesions enlarge with time and/or become painful. Others remain painless throughout life with no functional deficit [25]. Once identified, there is no record of spontaneous fusion/resolution [14, 16]. Pooling the numbers from the 19 studies above (which excludes single case reports) yields a total of 190 patients, of whom 61 were treated conservatively. Given that this data set largely represents ‘surgical’ series, and even when the studies identify patients treated non-operatively there is the potential for selection bias, it seems likely that the proportion identified as being treated non-operatively (61/190) is an underestimate. Moreover, given the limitations on follow-up, there is little information as to the ‘success’, or otherwise, of non-operative treatment. Therefore there is little evidence per se to guide the surgeon or parents of a child with an asymptomatic congenital clavicular pseudarthrosis.
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