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Page 1 of 4 Case report Licensee OA Publishing London 2012. Creative Commons Attribution License (CC-BY) Competing interests: none declared. Conflict of interests: none declared. All authors contributed to the conception, design, and preparation of the manuscript, as well as read and approved the final manuscript. All authors abide by the Association for Medical Ethics (AME) ethical rules of disclosure. F��: Al-Rabadi H, Zoubi Z, Al-Khawalde M, Kretschmer W, Wangerin K. Unilateral progressive condylar resorption post-orthognathic surgery: a case report. Hard Tissue. 2012 Nov 10;1(1):2. Unilateral progressive condylar resorption post-orthognathic surgery: a case report H Al-Rabadi 1 *, Z Zoubi 1 , M Al-Khawalde 1 *, W Kretschmer 2 , K Wangerin 2 Abstract Introduction Progressive condylar resorption is a rare complication following orthog- nathic surgery. Its aetiology and pathogenesis remains unclear, al- though several theories have been suggested to explain it. Management modalities for progressive condylar resorption range from conservative follow-up to a second corrective or- thognathic surgery. This is a report of a unilateral case of severe progressive condylar resorption that occurred in a female patient with class II skeletal deformity after bimaxillary osteotomy. The patient was followed-up for 9 years post- operatively. Case Report A 17 year old female patient referred to the Department of Oral and Maxillofacial Surgery by her ortho- dontist presented with mandibular hypoplasia with 5-mm overjet, high mandibular plane angle with ante- rior open bite, and mandibular asymmetry with deviation to the right side. Conclusion The 9-year follow-up of this case suggests that the treatment policy for future patients with PCR should be based on stabilization of the occlu- sion with splint followed by a long period of followup. Introduction Progressive condylar resorption (PC- R), a major complication of ortho- gnathic surgery, is a process of destructive remodelling of the condylar process of the mandible that leads to progressive alteration of shape and mass decrease 1 . The occurrence of PCR has been identified in many studies 2–5 as being associated with orthodontic treat- ment and orthognathic surgery; however, the real aetiology and patho- genesis of condylar resorption fol- lowing orthognathic surgery remains unclear. There is a predilection for females with pre-existing temporo- mandibular joint (TMJ) dysfunction in whom large mandibular advance- ment is performed 4 . Some investigators claim that a high mandibular plane angle is also a predisposing factor 4,6 . Several theories have been suggest- ed to explain this phenomenon: one suggests the concept of avascular necrosis of the condyle due to com- promised blood supply 3,4,7 ; another theory is based on genetic preference and is supported by its association with a high mandibular plane angle 3,4 ; and other theories suggest hormonal influence supported by predilection for young females 3,4,7 . The management of PCR remains a controversial issue. Distraction osteo- genesis correction has been advocated as a less stressful treatment modality compared with the classical orthog- nathic procedure, as there is less TMJ loading due to the gradual increase in mandibular ramus length 8,9 . We report a case of severe pro- gressive condylar resorption that was refractory to several treat- ment modalities in a female patient with class II skeletal deformity who underwent orthognathic surgery. Case report A 17-year-old female patient referred to the Department of Oral and Maxil- lofacial Surgery at Marienhospital- Stuttgart (Germany) by her orthodontist presented with mandibular hypo- plasia (class II skeletal malocclusion) with 5-mm overjet, high mandibular plane angle with anterior open bite, and mandibular asymmetry with deviation to the right side (Figure 1). Bimaxillary osteotomy was per- formed with Le Fort I osteotomy and bilateral sagittal split osteotomy and genioplasty fixed with rigid internal fixation. The initial results were good with class I occlusion. However, 9 months after the operation, the first changes of the right condyle appeared, as as- sessed radiographically, and 3 months later, condylar resorption became progressive with signs and symptoms of relapse, which included shortening of the right ascending ramus and pro- gressive class II skeletal malocclusion with deviation of the mandible to the right side (Figure2). A second operation was performed for the removal of plates and inser- tion of a distraction apparatus to lengthen the right ascending ramus according to astrict protocol, with the vector of distraction parallel to the mandibular plane (Figure 3). However, no satisfactory results were obtained and condylar resorption with relapse proceeded. The third operation was performed 6 months later, wherein the mandibu-lar distractor was removed and a * Corresponding authors Email: [email protected]; khawalde@ gmail.com 1 Department of Oral and Maxillofacial Surgery, King Hussein Medical Center, Royal Medical Services, Amman, Jordan 2 Department of Maxillofacial and Reconstructive Surgery, Paracelsus Hospital, Ruit, Germany dalities in a female patient with class II skeletal deformity who underwent orthognathic surgery. Oral & Cranio-Maxillofacial Surgery

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Page 1: Unilateral progressive condylar resorption post ... · plane angle with anterior open bite, and mandibular asymmetry with deviation to the right side (Figure 1). Bimaxillary osteotomy

Page 1 of 4

Case report

Licensee OA Publishing London 2012. Creative Commons Attribution License (CC-BY)

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F�� �������� ��������: Al-Rabadi H, Zoubi Z, Al-Khawalde M, Kretschmer W, Wangerin K. Unilateral progressive condylar resorption post-orthognathic surgery: a case report. Hard Tissue. 2012 Nov 10;1(1):2.

Unilateral progressive condylar resorption post-orthognathic

surgery: a case reportH Al-Rabadi1*, Z Zoubi1, M Al-Khawalde1*, W Kretschmer2, K Wangerin2

AbstractIntroductionProgressive condylar resorption is a rare complication following orthog- nathic surgery. Its aetiology and pathogenesis remains unclear, al- though several theories have been suggested to explain it. Management modalities for progressive condylar resorption range from conservative follow-up to a second corrective or- thognathic surgery. This is a report of a unilateral case of severe progressive condylar resorption that occurred in a female patient with class II skeletal deformity after bimaxillary osteotomy. The patient was followed-up for 9 years post-operatively.Case ReportA 17 year old female patient referred to the Department of Oral and Maxillofacial Surgery by her ortho-dontist presented with mandibular hypoplasia with 5-mm overjet, high mandibular plane angle with ante-rior open bite, and mandibular asymmetry with deviation to the right side. ConclusionThe 9-year follow-up of this case suggests that the treatment policy for future patients with PCR should be based on stabilization of the occlu- sion with splint followed by a long period of followup.

IntroductionProgressive condylar resorption (PC-R), a major complication of ortho-gnathic surgery, is a process of destructive remodelling of the condylar process of the mandible that leads to progressive alteration of shape and mass decrease1.

The occurrence of PCR has been identified in many studies2–5 as being associated with orthodontic treat-ment and orthognathic surgery; however, the real aetiology and patho-genesis of condylar resorption fol-lowing orthognathic surgery remains unclear. There is a predilection for females with pre-existing temporo-mandibular joint (TMJ) dysfunction in whom large mandibular advance-ment is performed4. Some investigators claim that a high mandibular plane angle is also a predisposing factor4,6.

Several theories have been suggest-ed to explain this phenomenon: one suggests the concept of avascular necrosis of the condyle due to com-promised blood supply3,4,7; another theory is based on genetic preference and is supported by its association with a high mandibular plane angle3,4; and other theories suggest hormonal influence supported by predilection for young females3,4,7.

The management of PCR remains a controversial issue. Distraction osteo-genesis correction has been advocated as a less stressful treatment modality compared with the classical orthog-nathic procedure, as there is less TMJ loading due to the gradual increase in mandibular ramus length8,9.

We report a case of severe pro-gressive condylar resorption that was refractory to several treat-ment modalities in a female patient with class II skeletal deformity who underwent orthognathic surgery.

Case reportA 17-year-old female patient referred to the Department of Oral and Maxil-lofacial Surgery at Marienhospital-Stuttgart (Germany) by her orthodontist presented with mandibular hypo-plasia (class II skeletal malocclusion) with 5-mm overjet, high mandibular plane angle with anterior open bite, and mandibular asymmetry with deviation to the right side (Figure 1).

Bimaxillary osteotomy was per-formed with Le Fort I osteotomy and bilateral sagittal split osteotomy and genioplasty fixed with rigid internal fixation.

The initial results were good with class I occlusion. However, 9 months after the operation, the first changes of the right condyle appeared, as as-sessed radiographically, and 3 months later, condylar resorption became progressive with signs and symptoms of relapse, which included shortening of the right ascending ramus and pro-gressive class II skeletal malocclusion with deviation of the mandible to the right side (Figure2).

A second operation was performed for the removal of plates and inser-tion of a distraction apparatus to lengthen the right ascending ramus according to astrict protocol, with the vector of distraction parallel to the mandibular plane (Figure 3). However, no satisfactory results were obtained and condylar resorption with relapse proceeded.

The third operation was performed 6 months later, wherein the mandibu-lar distractor was removed and a

* Corresponding authorsEmail: [email protected]; [email protected] Department of Oral and Maxillofacial Surgery,

King Hussein Medical Center, Royal Medical Services, Amman, Jordan

2 Department of Maxillofacial and Reconstructive Surgery, Paracelsus Hospital, Ruit, Germany

dalities in a female patient with class II skeletal deformity who underwent orthognathic surgery.

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Page 2: Unilateral progressive condylar resorption post ... · plane angle with anterior open bite, and mandibular asymmetry with deviation to the right side (Figure 1). Bimaxillary osteotomy

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Case report

Licensee OA Publishing London 2012. Creative Commons Attribution License (CC-BY)

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F�� �������� ��������: Al-Rabadi H, Zoubi Z, Al-Khawalde M, Kretschmer W, Wangerin K. Unilateral progressive condylar resorption post-orthognathic surgery: a case report. Hard Tissue. 2012 Nov 10;1(1):2.

DiscussionIt seems that PCR is not an isolated event; it is clearly amulti-factorial phenomenon based on interaction between two groups of factors: me-chanical stimuli and host adapting capacity2,7. Crawford et al.6 assumed that one possible explanation for the ongoing resorption could be the adaptability of the TMJ complex; and those who had ongoing PCR may not have had adaptation of the TMJ dur-ing the initial episodes to resist fur-ther PCR or had not reached the point of adaptation to accept new vectors of force applied and thus resist further episodes of PCR.

Based on the diagnosis and the patient’s chief complaint, we believe that we had good reasons for our treatment planning of bimaxillary osteotomy, although there were some predisposing factors such as female gender, high mandibular plane angle, signs of osteoarthritis seen in the right condyle pre-operative radio-graphs and a relatively large amount of mandibular advancement (about 7 mm) during operation with counter-clockwise rotation.Late skeletal relapse caused by PCR

Figure 1: Lateral view for the patient pre-operatively.

Figure 2: OPG radiograph of the patient 1 year after operation.

Figure 3: OPG radiograph of the patient after removal of plates and insertion of distraction apparatus.

right side sagittal split osteotomy of the mandible was performed to cor-rect facial asymmetry. This resulted in ongoing osteolysis with approxi-mately 15 mm shortening of the man-dibular ascending ramus compared with the first preoperative radiograph (Figure 4).

A conservative protocol of treatment was decided for this patient by stabiliz-ing the occlusion with occlusal splint and supportive treatment with non- steroidal anti-inflammatory drugs followed by long-term follow-up.

The last follow-up for the patient was performed in October 2011 (9 years after the first operation). The patient had a stable mild class II occlusion with mandibular facial asymmetry but with acceptable facial profile (Figure 5).

Page 3: Unilateral progressive condylar resorption post ... · plane angle with anterior open bite, and mandibular asymmetry with deviation to the right side (Figure 1). Bimaxillary osteotomy

Page 3 of 4

Case report

Licensee OA Publishing London 2012. Creative Commons Attribution License (CC-BY)

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F�� �������� ��������: Al-Rabadi H, Zoubi Z, Al-Khawalde M, Kretschmer W, Wangerin K. Unilateral progressive condylar resorption post-orthognathic surgery: a case report. Hard Tissue. 2012 Nov 10;1(1):2.

upon a second time, while the other four underwent occlusal rehabilita-tion. The results for the patients who underwent revisional surgery were unsatisfactory. The patients treated with an occlusal splint (with or with-out orthodontics and/or prosthetic therapy) had a functional occlusion and tolerable TMJ complaints.

Distraction osteogenesis is consid-ered especially beneficial over classi-cal orthognathic procedures because it is thought to reduce the incidence of PCR8,9. However, some cases of condylar resorption after distraction osteogenesis have been published8. In our case, distraction osteogenesis was a good option with less loading on the condyle, although this has not been scientifically proven. However, there is always a risk of reactivation of condylar resorption or making it even worse as seen in our case.

There is an increasing trend in the literature to limit a second correc-tive orthognathic surgery to cases of condylar resorption of small magni-tude1,6. Huang et al.1 suggest that orthognathic surgery should only be performed when the condyles are stable as determined by clinical ex-amination, radiographs or bone scans as appropriate, and it is best limited to maxillary surgery.

The occurrence of PCR in this case seems to support the hypothesis of predisposing risk factors mentioned above. However, this is the only case of refractory PCR that has occurred so far in our series of more than 1000 patients who have undergone bimaxillary osteotomies in the past 12 years, starting from 2000. In ret-rospect, it is very difficult to find out what happened at what moment in our case. We are fortunate not to have seen other patients with this problem, but if we do encounter such a patient, it would be a real problem to solve.

ConclusionFrom our experience of the 9-year follow-up of this case of PCR, we suggest that the treatment policy for future patients with PCR should be based on stabilization of the occlu-sion with splint followed by a long period of follow-up. Subsequently, the next step can be determined either with non-invasive treatment, such as orthodontics or prosthodon-tic rehabilitation with acceptance of suboptimal occlusion, or with more invasive treatment, such as distrac-tion osteogenesis and maxillary and mandibular segmental osteotomies after explaining the risks to the patient.

ConsentWritten informed consent was ob-tained from the patient for publication of this case study and accompanying images. A copy of the written consent is available for review with the Editor-in-Chief of this journal.

Figure 5: PA and OPG views for the patient 9 years after the operation.

Figure 4: OPG radiograph of the patient 4 years after operation.

after orthognathic surgery is a rare complication, but when it happens, it is a real problem to solve. There is a lot of controversy in the literature regarding the results of distraction osteogenesis or a second osteotomy to correct malocclusion resulting from skeletal relapse, subsequent to PCR in particular, following orthog-nathic surgery6,10. Crawford et al.6 reported seven patients who under-went a second osteotomy to correct relapse resulting from PCR after the first osteotomy. Only two patients were skeletally stable with good oc-clusion at 41 months and 19 months, respectively. The remaining five pa-tients had skeletal relapse of varying degrees secondary to PCR. Merkx and Van Damme10 evaluated the treat-ment in eight patients with condylar resorption after orthognathic sur-gery. Four patients were operated

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Licensee OA Publishing London 2012. Creative Commons Attribution License (CC-BY)

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F�� �������� ��������: Al-Rabadi H, Zoubi Z, Al-Khawalde M, Kretschmer W, Wangerin K. Unilateral progressive condylar resorption post-orthognathic surgery: a case report. Hard Tissue. 2012 Nov 10;1(1):2.

(2):103–11. Hwang SJ, Haers PE, Zimmermann A, Oechslin C, Seifert B, Sailer HF. Surgical risk factors for condylar resorption after orthognathic surgery. Oral Surg Oral Med.

May; 89(5):542–52. Crawford JG, Stoelina PJW, Blijdorp PA, Brouns JJA. Stability after reoperation for progressive condylar resorption after or-thognathic surgery: report of seven cases. J Oral Maxillofac Surg. 1994 May;52(5): 460–6.7. Bouletreau P, Frey R, Breton P, Freidel M. Focus on the effect of orthognathic surgery on condylar remodeling. Rev Stomatol Chir Maxillofac. 2004 Nov;105(5):283–8.8. van Strijen PJ, Breuning KH, Becking AG,

Tuinzing DB. Condylar resorption follow-ing distraction osteogenesis: a case report. J Oral Maxillofac Surg. 2001 Sep;59(9):1104–7.9. Ow A, Cheung LK. Bilateral sagittalsplit osteotomies versus mandibular distraction osteogenesis: a prospective clinical trial comparing inferior alveolar nerve function and complications. Int J Oral Maxillofac Surg. 2010 Aug;39(8):756–60.10. Merkx MAW, Van Damme PA. Condylar resorption after orthognathic surgery. Evaluation of treatment in 8 patients. J Cranio maxillofac Surg. 1994 Feb;22(1):53–8.

References 1. Huang YL, Pogrel MA, Kaban LB. Diagnosis and management of condylar resorption. J Oral Maxillofac Surg. 1997 Feb;55(2):114–9.2. Arnett GW, Milam SB, Gottesman L. Pro-gressive mandibular retrusion—idiopathic condylar resorption. Part II. Am J Orthod Dentofacial Orthop. 1996 Aug;110(2):117–27.3. Moore KE, Gooris PJJ, Stoelinga PJW. The contributing role of condylar resorp-tion to skeletal relapse following mandib-ular advancement surgery: report of five cases. J Oral Maxillofac Surg. 1991 May;49(5):448–60.4. Hwang SJ, Haers PE, Seifert B, Sailer HF. Non-surgical risk factors for condylar resorption after orthognathic surgery. 5. J. Cranio maxillofac Surg. 2004 Apr;32

6. Oral Pathol Oral Radiol Endod. 2000