Table 1.
Overview of clinical studies included in meta-analysis.
| First author (year) | Study design | OI type | Age, y | Sex | n | CG | Markers or parameters | Data | QS |
|---|---|---|---|---|---|---|---|---|---|
| Collagen degradation markers | |||||||||
| Pressac (2002) | CS | I, III, IV | 1 mo-17 | MF | 46 | RWS | uCTx1 | AG | 12 |
| Iwamoto (2002) | CR | I | 58 | M | 1 | AAR | uNTx, uDPD | IPD | 14 |
| Zacharin (2002) | CS | III, IV | 1-14 | MF | 18 | AAR | uDPD | AG | 12.5 |
| Zacharin (2004) | CH | Mild | 2-15 | NR | 18 | AAR | uDPD | AG | 12.5 |
| Kitaoka (2011) | CS | I, IV, I/IV | 9 mo-13 | MF | 13 | AAR | sNTx | AG | 12.5 |
| Asharani (2012) | CS | XIII | 1-5 | NR | 2 | RWS | uDPD | IPD | 12 |
| D’Eufemia (2014) | CS | I | 2-6 | MF | 18 | AAR | sCTx1 | AG | 12 |
| Hryhorovskyi (2015) | CS | I, III | 5-17 | NR | 4 | AMG | sCTx1 | IPD, AG | 12 |
| Hoyer-Kuhn (2016) | CS | I, IV, NA | 5-11 | MF | 10 | AAR | sNTx, uDPD | IPD, AG | 12 |
| D’Eufemia (2017) | CS | I | 4-5 | MF | 18 | AAR | sCTx1 | AG | 13 |
| Uehara (2017) | CS | I | 14-42 | F | 3 | AAR | uNTx | IPD | 12 |
| Hoyer-Kuhn (2019) | RT | I, III | 8.6 | MF | 9 | AAR | uDPD | AG | 12 |
| Xu (2019) | CR | XIII | 15.7 | M | 1 | RWS | sCTx1 | IPD | 14 |
| Zhang (2022) | CS | I, III, IV, V | 14-38 | NR | 149 | RWS | sCTx1 | AG | 13 |
| Bone histomorphometric parameters | |||||||||
| Baron (1983) | CH | NR | 6-15 | MF | 9 | AMG | NOc, OcS | AG | 12.5 |
| Ste-Marie (1984) | CS | NR | 5-69 | MF | 12 | CAR | NOc, ES | IPD, AG | 13 |
| Rauch (2000) | CH | I, III, IV | 3-11 | MF | 70 | CAR | NOc, OcS, ES | AG | 13 |
| Iwamoto (2002) | CR | I | 58 | M | 1 | AAR | NOc, OcS, ES | IPD | 14 |
| Rauch (2002) | CS | I, III, IV | 1-17 | MF | 44 | CAR | OcS, ES | AG | 12.5 |
| Hryhorovskyi (2015) | CS | I, III | 5-17 | NR | 3 | AAR | NOc, OcS | IPD AG | 12 |
| Ward (2016) | CR | VI | 34 wk | M | 1 | CAR | NOc, OcS/BS | IPD | 15 |
| Webb (2017) | CR | XIV | 15-24 | MF | 2 | RWS | NOc, OcS, ES | IPD | 12 |
| Surowiec (2020) | CH | I, III, IV, | 1.3-23 | MF | 8 | AAR | NOc | AG | 12 |
| Stürznickel (2021) | CR | XX | 22 wk | NR | 1 | AMG | NOc, ES | IPD | 13 |
| Lui (2022) | CR | XII | 6 | M | 1 | CAR | ES | IPD | 12 |
The study designs of included clinical studies were case report (CR), case series (CS), retrospective (RT)
or cohort (CH). Sex was reported for male (M) and female (F) patients or for groups of male and female
patients (MF). Control group (CG) was reported as follows: (1) as an age-matched group (AMG), (2) reference values were given but the source unknown (reference without source [RWS]), (3) reference to control is cited, age-matched data extracted (cited age-matched reference [CAR]), (4) control data were not given or cited, age-matched reference assigned (assigned age-matched reference [AAR]). Data types were individual patient data (IPD) or aggregated data (AG). Quality score (QS; out of 17 maximum) was assessed using the checklist (Supplementary method 2).
Abbreviations: ES, eroded surface; NOc, osteoclast numbers; NR, not reported; OcS, osteoclast surface; OI, osteogenesis imperfecta; sCTx, serum CTx; sNTx, serum NTx; uCTx, urinary CTx, uDPD, urinary deoxypyridinoline; uNTx, urinary NTx.