Technical Note Pediatr Neurosurg 2013;49:380–383 DOI: 10.1159/000369029

Received: January 3, 2014 Accepted after revision: October 12, 2014 Published online: December 9, 2014

Troubleshooting Distraction Osteogenesis for Craniosynostosis Takuya Akai Syunsuke Shiraga Yasuo Sasagawa Hideaki Iizuka Masanobu Yamashita Shigehiko Kawakami Department of Neurosurgery and Plastic and Reconstructive Surgery, Kanazawa Medical University, Uchinada, Japan

Key Words Craniosynostosis · Distraction osteogenesis · Troubleshooting

Abstract Purpose: We previously reported that distraction osteogenesis is less invasive and gives greater skull advancement compared to conventional cranioplasty [Akai et al: Pediatr Neurosurg 2006;42:288–292]. In this study, we analyzed the distraction osteogenesis process and tried to identify and solve various technical problems. Patients and Results: We operated on 22 patients, 5 syndromic and 17 nonsyndromic. During treatment, we encountered several problems: (i) dural laceration during craniotomy (2 cases), solution: repair by suturing with fascia; (ii) skull fracture at sphenofrontal or coronal sutures (2 cases), solution: completed distraction; (iii) device dislocation during distraction (1 case), solution: the device was secured to the skull with stainless wire; (iv) wound issues around shaft and device (3 cases), solution: treated with antibiotic ointment. Discussion: (1) Extra caution is needed to avoid dural damage at frontal bottom burr holes. (2) Completion of craniotomy should be confirmed by checking if the bone flap moves in sync with brain pulsation. The craniotomy line should be placed forward of coronal sutures. (3) For patients younger than 2 years, employ clamp-type devices. (4) Shafts should be cut short enough to prevent their tips from exerting pressure or puncturing the scalp

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from beneath. Conclusions: The distraction osteogenesis technique has complications that may not occur in conventional one-stage cranioplasty. In this study, neither age at operation nor distraction distance were significant causes of complications during distraction osteogenesis. © 2014 S. Karger AG, Basel

Introduction

Distraction osteogenesis was first applied during a craniomaxillofacial surgery by McCarthy et al. [1] in 1992. This technique was later introduced for skull expansion in craniosynostosis treatment [2, 3]. Since 1999, we have used this technique for patients with craniosynostosis. We can treat craniosynostosis by either conventional one-stage cranioplasty or distraction osteogenesis. With distraction osteogenesis, the dissection of the dura mater is limited, which results in a shorter operation time, less bleeding, no extradural fluid collection and good blood flow preservation to the dura mater. Also, stress to the scalp is limited and has not resulted in poor wound healing [4–7]. As such, this method is less invasive compared to the conventional one-stage cranioplasty, gives greater skull advancement and causes fewer wound problems [8– 12]. Distraction osteogenesis would be especially suitable for syndromic craniosynostosis [13, 14]. This technique has complications, however, that may not occur in a conTakuya Akai, MD Department of Neurosurgery Kanazawa Medical University 1.1 Daigaku, Uchinada, Kahokugun, Ishikawa 920-0293 (Japan) E-Mail akai @ kanazawa-med.ac.jp

Fig. 1. a Case 4, a 17-month-old girl with

trigonocephaly. CT performed at 13 mm of advancement showed dissociation of the sphenofrontal suture (arrows) due to incomplete orbital osteotomy (arrowhead). We distracted 1 mm further and finished. b Case 5, a 7-month-old girl with trigonocephaly. We felt loosening at turning of the shaft. CT performed at 14 mm of advancement showed dissociation of the coronal suture (arrow). We finished the distraction.

a

b

Table 1. List of patients with complications

Case

Type

Syndrome

Age at operation, months

Distraction distance, mm

Complications

1 2 3 4 5 6 7 8

oxycephaly turricephaly brachycephaly trigonocephaly trigonocephaly trigonocephaly trigonocephaly pancraniosynostosis

Apert’s Pfeiffer’s none none none none none none

6 15 6 17 7 11 12 26

27 25 22 14 14 12 14 13

frontal dura laceration scalp erosion under shaft fontal dura laceration sphenofrontal dissociation fracture at coronal suture device dislocation device exposure device exposure

ventional one-stage cranioplasty [15–18]. In this study, we analyzed the distraction osteogenesis process and tried to identify and solve technical problems occurring using this process.

Patients and Results

We operated on 22 patients between the years 1999 and 2012. Patient types included 5 syndromic (including 3 brachycephaly, 2 turricephaly) and 17 nonsyndromic (5 trigonocephaly, 4 scaphocephaly, 4 plagiocephaly, 3 brachycephaly and 1 pancraniosynostosis). The ages at operation were 6 months to 11 years in syndromic patients and 5 months to 4 years in nonsyndromic patients. Distraction distances were 18–27 and 12–25 mm, respectively. The details of this technique were previously published [1]. In short, distraction osteogenesis involved three surgeries. In operation 1, we performed a craniotoDistraction Osteogenesis

my and created one piece of frontal bone flap with an orbital rim for fronto-orbital advancement and placed the distraction device. We began distraction 3 days after device placement. In operation 2, performed 7–14 days later, we cut the device shaft and closed the wound. In operation 3, performed 3 months after surgery 1, patients were readmitted to have the device removed. During the course of treatment, we encountered several technical problems: (1) dural laceration during craniotomy, (2) skull fracture at sphenofrontal suture or coronal sutures, (3) device dislocation, and (4) wound issues (table 1). Dural Laceration during Craniotomy. We had 2 patients who experienced dural laceration during craniotomy. Both patients had these troubles at frontal bottom burr holes. The lacerations were repaired by suturing with fascia. Skull Fracture at Sphenofrontal Suture or Coronal Suture. The dissociation of the sphenofrontal suture was induced during the distraction in patient 4 (fig. 1a). We Pediatr Neurosurg 2013;49:380–383 DOI: 10.1159/000369029

381

ment. Patient 2 developed scalp erosion during distraction beneath the shaft. The erosion was mitigated by removing the shaft.

Discussion

Color version available online

Fig. 2. Case 6, an 11-month-old boy with trigonocephaly at 16 mm of advancement, showing left device dislocation (arrow). We secured the device to the skull with stainless wire at the shaft cutting, and advanced 3 mm further during that operation.

Fig. 3. Case 8, a 26-month-old boy with pancraniosynostosis. The

tip of the cut shaft (arrow) is exposed.

finished the distraction at 14 mm of advancement. In patient 5, a part of the parietal bone attached to the device broke at the coronal suture during the distraction (fig. 1b). We could only advance the frontal bone flap to 14 mm. Device Dislocation. A distraction plate was dislocated during the distraction in patient 6 (fig. 2). We secured the device to the skull with stainless wire. Wound Issues. Patients 7 and 8 developed device exposure at the tip of the cut shaft (fig. 3). The device was kept in place until the scheduled device removal date by treating the exposed surrounding area with antibiotic oint382

Pediatr Neurosurg 2013;49:380–383 DOI: 10.1159/000369029

The distraction osteogenesis method has several advantages, but we see many areas still needing improvement [15, 16, 18, 19]. Regarding the dural laceration during craniotomy: to avoid cerebrospinal fluid leak during distraction, extreme care is needed at craniotomy. To cut the orbital roof for fronto-orbital advancement through frontal and lateral burr holes without a frontal craniectomy, we have to dissect the dura mater from the frontal base bone completely. However, the frontal base is very thin and fibers are running in longitudinal directions, therefore we must handle the dura mater gently. Regarding skull fractures: in patients who had dissociation of the sphenofrontal suture, orbital craniotomy may be incomplete for fronto-orbital advancement. To address this, we confirmed complete craniotomy by checking if the bone flap moves in sync with brain pulsation, or by passing a thread along the orbital craniotomy. We found that using a thread-wire saw was safer and more effective than using a chisel [20]. For the patient with parietal bone tip breakage, we found that the device might be too close to the coronal suture to maintain extension pressure. To avoid fracture, the craniotomy line was placed forward of coronal sutures, or the extension plates were put forward and back of coronal sutures. Regarding device dislocation: the skull may not be thick enough to keep the device in place. We overcame this by using one or more clamp-type devices on each side for patients younger than 2 years [10, 16]. A cautionary note: if devices are left implanted for too long, they may become embedded in the skull and may be difficult to remove at the final operation. Regarding scalp wounds in the area of the shaft tip: we addressed this by cutting the shaft short enough to avert the tip from exerting pressure upward towards the scalp. To avoid skin erosion by the shaft at distraction, we needed to push the shaft through the scalp far from the device to create space between the shaft and the scalp. We found that the scalp wound around the shaft would be treatable without device removal. Among the syndromic craniosynostosis group, age at operation was 7–133 months (average 50 months) in the Akai/Shiraga/Sasagawa/Iizuka/Yamashita/ Kawakami

Table 2. Age and distraction distance in groups with complications and without complications Cases Age at operation, Distance, months mm Syndromic cases Without complications With complications

3 2

Nonsyndromic cases Without complications With complications

12 5

7–133 (avg. 50.0) 18–19 (avg. 18.7) 6–15 (avg. 10.5) 25–27 (avg. 26.0) 5–57 (avg. 20.1) 6–29 (avg. 15.0)

12–25 (avg. 16.7) 14–22 (avg. 16.4)

Distance = Distraction distance; avg. = average.

cations in either group. Although we expected more complications among patients with longer distraction distances, the actual number turned out to be very low. We treated 5 patients with trigonocephaly by distraction osteogenesis and 4 patients who had problems such as skull fractures, device dislocation and device exposure (table 1). We do not think these problems occurred due to the skull shape and stopped treating trigonocephaly through distraction. We had expected the forehead to self-remodel after dissociation of the frontal bone from the skull base, but the forehead remained flat even after distraction.

Conclusions

group without complications and 6–15 months (average 10.5 months) in the group with complications. Among the nonsyndromic group, age at operation was 5–57 months (average 20.1 months) in the group without complications and 6–29 months (average 15 months) in the group with complications (table 2). Younger children had more complications in both syndromic and nonsyndromic groups, but age was not a statistically significant factor for compli-

Distraction osteogenesis has several advantages as a treatment modality for craniosynostosis. However, this technique has complications that may not occur in conventional one-stage cranioplasty. Special attention must be paid during the entire process until device removal. Problem resolution and development of advanced procedures are essential.

References 1 McCarthy JG, Schreiber J, Karp N, Thorne CH, Grayson BH: Lengthening the human mandible by gradual distraction. Plast Reconstr Surg 1992;89:1–8. 2 Hirabayashi S, Sugawara Y, Sakurai A, Harii K, Park S: Front orbital advancement by gradual distraction. J Neurosurg 1998; 89: 1058– 1061. 3 Sugawara Y, Hirabayashi S, Sakurai A, Harii K: Gradual cranial vault expansion for the treatment of craniofacial synostosis: a preliminary report. Ann Plast Surg 1998; 40: 544– 565. 4 Whitaker LA, Munro IR, Salyer KE, Jackson IT, Ortiz-Monasterio F, Marchac D: Combined report of problems and complications in 793 craniofacial operations. Plast Reconstr Surg 1979;64:198–203. 5 Munro IR, Sabatier RE: An analysis of 12 years of craniomaxillofacial surgery in Toronto. Plast Reconstr Surg 1985;76:29–35. 6 Rachmiel A, Potparic Z, Jackson IT, Fukuta K, Audet B, Tysell B: Extradural dead space following cranial bone advancement. Ann Plast Surg 1994;32:148–155. 7 Spinelli HM, Irizarry D, McCarthy JG, Cutting CB, Noz ME: An analysis of extradural dead space after fronto-orbital surgery. Plast Reconstr Surg 1994;93:1372–1377.

Distraction Osteogenesis

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15 Nishimoto S, Oyama T, Nagashima T, Shimizu F, Tsugawa T, Takeda M, Toda N: Gradual distraction fronto-orbital advancement with ‘floating forehead’ for patients with syndromic craniosynostosis. J Craniofac Surg 2006;17:497–505. 16 Lee JA, Park DH, Yoon SH, Chung J: Distractor breakage in cranial distraction osteogenesis for children with craniosynostosis. Pediatr Neurosurg 2008; 44: 216– 220. 17 Park DH, Chung J, Yoon SH: Rotating distraction osteogenesis in 23 cases of craniosynostosis: comparison with the classical method of craniotomy and remodeling. Pediatr Neurosurg 2010;46:89–100. 18 Winston KR, Ketch LL, Dowlati D: Cranial vault expansion by distraction osteogenesis. J Neurosurg Pediatr 2011;7:351–361. 19 Yano H, Tanaka K, Sueyoshi O, Takahashi K, Hirata R, Hirano A: Cranial vault distraction: its illusionary effect and limitation. Plast Reconstr Surg 2006;117:193–200. 20 Tomita K, Kawahara N, Toribatake Y, Heller JG: Expansive midline T-saw laminoplasty (modified spinous process-splitting) for the management of cervical myelopathy. Spine 1998;23:32–37.

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Troubleshooting distraction osteogenesis for craniosynostosis.

We previously reported that distraction osteogenesis is less invasive and gives greater skull advancement compared to conventional cranioplasty [Akai ...
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