Unusual presentation of congenital radioulnar synostosis with osteoporosis, fragility fracture and nonunion: A case report and review of literature

Table 1 Cleary and Omer classification of congenital proximal radioulnar synostosis

Type (proportion)	Description
I (19%)	Fibrous synostosis with a reduced normal-appearing radial head
II (8%)	Visible bony synostosis with a reduced radial head
III (56%)	Visible bony synostosis with a hypoplastic and posteriorly dislocated radial head
IV (17%)	Short bony synostosis with an anteriorly dislocated mushroom-shaped radial head

Table 2 The evaluation results before and after surgery

Outcome assessment	Before surgery		At 1 yr after surgery
	Nondominant, fractured side	Dominant, non-fractured side	Nondominant, fractured side	Dominant, non-fractured side
ROM of elbow E/F (°)	0/140	0/140	0/140	0/140
ROM of forearm P/S (°)	0/5	-30/0	-45/45	-45/30
ADL1
Hygiene and self-care items	3.5	4.0	4.2	4.5
Feeding-related items	3.3	4.6	4.5	4.9
Environmental interaction	3.3	4.3	4.3	4.7
Average	3.4	4.3	4.3	4.7
LES2
Clinical assessment	12	15	17	17
Patient-answered questions	11	30	31	34
Final scores	4.4	8.2	8.7	9.2
DASH3
Function	48		32
Symptoms	14		7
DASH value	26.7		7.5
Failla classification system	Fair	Good	Good	Excellent

¹Scores range from 1 to 5. The lower scores represent greater limitation on activities of daily living.

²Scores range from 0 to 10. The lower scores represent greater symptom and functional severity.

³Value ranges from 1 to 100. Greater scores indicate more extremely limited upper limb function. ROM: Range of motion; E/F: Extension/flexion; P/S: Pronation/supination; ADL: Assessment of activities of daily living; LES: Liverpool Elbow Score; DASH: Disabilities of the arm, shoulder and hand.

Table 3 Different surgical methods used in the treatment of congenital radioulnar synostosis

Ref.	Post-op follow-up in mo	Surgical methods	Preoperative fixed forearm position (°)1	Final forearm position (°)4	Complications
Simcock et al[31], 2015	46 (range, 6-148)	Derotational osteotomy	85 (range, 60-100)	-8 (range, -30-0)	12%
VanHeest et al[35], 2013	33 (range, 24-42)2	Radial head excision3	27.5 (range, 0-60)	Mainly improve elbow extension movement	25% (one with transient radial nerve neuropraxia)
Shingade et al[46], 2014	54 (range, 36-84)	Radial osteotomy	56.3 (range, 30-86)	27.2 (range, 20-30)	Not available
Hung et al[39], 2008	64 (range, 30-129)	Derotational osteotomy	78 (range, 65-85)	8 (range, 0-30)	0%
Bishay et al[40], 2016	30.4 (range, 24-36)	single-session double-level rotational osteotomy	70.7 (range, 60-85)	-15.5 (range, -30-20)	0%
Garg et al[36], 2015	32.1 (range, 16-65)	Radial head excision with a tensor fascia lata graft	51.6 (range, 30-70)	15 (range, 5-32)	50% (superficial infection, posterior interosseous nerve palsy and a persistent extension lag)
Horii et al[41], 2014	60 (range, 12-156)	Derotational osteotomy	72 (range, 40-100)	0	0%
Pei et al[42], 2019	55.19 (range, 24-123)	Derotational osteotomy	62.92 (55-80)	7.94 (range, -10-20)	9.7% (transient nerve palsies and compartment syndrome)
Satake et al[47], 2018	163.2 (range, 120-228)	Derotational osteotomy	51.3 (range, 30-90)	4 (range, -20-30)	0%
Hwang et al[38], 2015	33 (range, 12-72)	Derotational osteotomy	47 (range, 30-65)	27 (range, 25-30)	0%
Kanaya et al[37], 2016	120 (range, 96-144)	Radial osteotomy with a free vascularized adipofascial graft	14.2 (range, 0-25)	6.7 (range, 0-25)	66.7% (flap congestion, transient radial nerve palsy, synovial fold and posterior or anterior radial head dislocation)

¹Degree of pronation.

²The fourth case without specific follow-up time.

³A method of only treating the anteriorly dislocated radial head in type IV radioulnar synostoses with radial head impingement.

⁴Degrees of supination: -8, 8° of pronation; -10, 10° of pronation; -15.5, 15.5° of pronation; -20, 20° of pronation; -30, 30° of pronation.