Eur J Orthop Surg Traumatol DOI 10.1007/s00590-015-1659-5

GENERAL REVIEW • SPINE - PAGET DISEASE

Surgical treatment in spine Paget’s disease: a systematic review Alberto Jorge-Mora1 • Samer Amhaz-Escanlar1 • Ana Lois-Iglesias2 Susana Leborans-Eiris1 • Jesu´s Pino-Minguez1



Received: 12 February 2015 / Accepted: 16 June 2015  Springer-Verlag France 2015

Abstract Paget’s disease of bone (PDB) is a disease characterized by a disorder in the bone metabolism. The spine is the second region affected after the pelvis. Surgical treatment is reserved for cases refractory to medical treatment. We performed a systematic review of patients with Paget disease of bone affecting the spine, treated surgically in the last 30 years. The main objective of the review is to find out indications for surgery, outcomes of these patients and also the standard perioperative management. Keywords

Paget’s disease  Spine  Surgery

Introduction Paget’s disease of bone (PDB) is a disease characterized by a disorder in the bone metabolism, first described by Sir John Paget in 1877 [1]. This ‘‘osteitis deformans’’ as first described can affect any bone. The origin of the disease is unknown, but we have seen a decrease in the number of patients affected and the severity of the disease in the last years [2]. Nowadays the main treatment is the administration of bisphosphonate, but even with new medications we can find cases refractory to treatment or local complications that predispose for surgery [2–7]. & Alberto Jorge-Mora [email protected] 1

Complexo Hospitalario Universitario de Santiago, Universidad de Santiago de Compostela, Santiago de Compostela, Spain

2

Hospital Nuestra Sen˜ora de La Esperanza, Universidade da Corun˜a, A Corun˜a, Spain

Spine implication accounts in 15 % of the patients with PDB [8, 9]. Pain and spinal stenosis were the main symptoms. We made a systematic review of patients with PDB affecting spine, treated surgically in the last 30 years, to find out indications for surgery, outcomes and also the perioperative management.

Materials and methods Literature search We performed a systematic review of articles indexed in MEDLINE from January 1984 until December of 2014 (last 30 years), searching articles which met the following criteria: Articles should be in English or Spanish, should be original, should include cases of Paget’s disease involving the spine treated surgically. Exclusion criterion was articles including malignancy (Paget’s disease with sarcomatous degeneration). We searched in PubMed using the following Boolean operators: ‘‘(spine OR spinal) AND paget AND surgery,’’ ‘‘(spine OR spinal) AND paget AND treatment,’’ ‘‘(spine OR spinal) AND paget AND operative,’’ ‘‘(spine OR spinal) AND paget AND surgical’’ and ‘‘(spine OR spinal) AND paget AND stenosis.’’ The principal author (A. J. M.) reviewed the title and abstract from all the articles. The initial search retrieved 117 articles. After the first screening, ten articles were fully read, and finally six articles were included in the review. We also read titles and abstracts from the references of the articles included, and three more articles were added for the final analysis. Articles included are listed in Table 1 [6, 10–17].

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Data extraction

We consider a complication in the same way as the authors of the original articles describe it.

In every article, we searched for the number of patients treated, the administration of any concomitant medical treatment, the vertebra affected, the cause for surgery and the improvement after surgery and postoperative medication. Table 1 Articles included in the review Article

Year

Author

Journal

Patients

1

1988

Hadjpavlov et al.

Spine (Phila Pa 1976)

1

2

1991

Hepgul et al.

Surg Neurol

1

3

1994

Smidt et al.

Orthop Rev

9

4

1998

Hepple et al.

Injury

1

5

2003

Kremer et al.

J Neurosurg

1

6

2006

Tancioni et al.

Surg Neurol

1

7

2007

Oikonomou et al.

Spine (Phila Pa 1976)

1

8

2008

Tessitore et al.

Spine (Phila Pa 1976)

1

9

2011

Pedicelli et al.

J Vasc Interv Radiol

1

Results We have reviewed nine articles and obtained 17 patients for review (Table 2). Paget affected the cervical spine in 5 of 17 (29 %) patients, thoracic spine in 4 of the 17 (23 %) patients and the lumbar spine in the remaining 8 patients (48 %). Only 7 of the 17 (41 %) patients had preoperative treatment with bisphosphonates. The main indications for surgery were neurological deficit in 15 (88 %) patients and pain in 2 (12 %) patients. In all cases with neurological compromise, medullar decompression was performed, with a wide excision of bone and soft tissue affected. None of the patients with lumbar affection required fusion (0/8, 0 %). Fusion was required in 3 of the 5 (60 %) patients with cervical Paget and 1 of the 4 (25 %) patients with thoracic Paget (in one

Table 2 Patients included in the review Patient

Level

1

Cervical

2

Cervical

3

Cervical

4

Cervical

5

Main symptom

Surgical technique

Improve

Postoperative medication

Tetraparesia

Laminectomy, instrumentation

Yes

Bisphosphonate

Bisphosphonate

Myelopathy

Vertebrectomy, fusion and instrumentation

Yes

Bisphosphonate

Bisphosphonate

Compression

Laminectomy

No

Mithramycin

Yes

Paraparesia

Laminectomy

Partial

Bisphosphonate

Yes

Cervical

Bisphosphonate

Pain, instability

Fusion

Yes

6

Lumbar

Bisphosphonate

Low back pain

Vertebroplasty

Yes

7

Lumbar

Low back pain

Vertebroplasty

Yes

8

Lumbar

Stenosis

Laminectomy

Yes

9

Lumbar

Stenosis

Laminectomy

Partial

Calcitonin

Yes

Bleeding

10 11

Lumbar Lumbar

Stenosis Stenosis

Laminectomy Laminectomy

Partial No

Mithramycin Mithramycin

Yes

Bleeding Bleeding

12

Lumbar

Bisphosphonate

Stenosis

Laminectomy

Yes

Bisphosphonate

13

Lumbar

Bisphosphonate

Stenosis

Laminectomy

Yes

14

Thoracic

Paraparesia

Wide decompressive laminectomy

Yes

15

Thoracic

Paraparesia

Laminectomy

Yes

Bisphosphonate

16

Thoracic

Paraparesia

Vertebrectomy, fusion and instrumentation

No

Calcitonin

No

Bleeding, death

17

Thoracic

Paraparesia

Laminectomy and vertebroplasty

Yes

Bifosfonatos

123

Preoperative

Bisphosphonate

Improvement with medication

Complication

Calcitonin

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case, a vertebroplasty was added to improve stability but not instrumentation). In patients with partial or no improvement of the neurological symptoms after surgery, the use of postoperative medication for Paget (bisphosphonates, mithramycin or calcitonin) increased the clinical outcome in the next months in all cases, except one who died 2 weeks after the procedure. One (6 %) of the seventeen patients died 2 weeks after surgery because of a cardiorespiratory failure. This patient had a thoracic affection of the spine and was treated with an anterior decompression. He experienced a profuse bleeding during surgery. Bleeding was a frequent complication in patients who were not treated with preoperative bisphosphonates (4/9, 44 %). In all cases with pain as the main indication, a vertebroplasty was performed with clinical improvement maintained in time. Both cases describe a percutaneous bilateral transpedicular approach with no postoperative complications. Seven and eight milliliters of PMMA were used, respectively.

Discussion The extended use of vertebroplasty has changed the management of mechanical pain in Paget disease in patients with resistance to medical treatment [10, 14]. This option has demonstrated good results, with no complications in the patients reviewed. As many authors recommend, the use of preoperative medication for PDB, especially bisphosphonates, is indicated in every case [6, 18, 19]. This medication can prevent the need for surgery and in cases where surgery is unavoidable; it diminishes complications and postoperative hematoma formation. In the same way, the administration of postoperative bisphosphonates is highly recommended. Postoperative medication can improve outcomes in terms of neurological recovery and function, in patients with partial or marginal recovery. We cannot recommend systematic lumbar fusion added to spine decompression in cases of PDB with lumbar stenosis, because there are no complications described in the eight patients who sustained an isolated lumbar posterior laminectomy. Open surgery in Paget’s disease of the spine is a challenge, with an overall complication rate of more than 25 % in cases of neurological deficit (4/15, 27 %), with variable degree of improvement in preoperative status. There are also cases of early postoperative deaths, and severe

neurological complications described that can darken the prognosis of the disease [6]. Compliance with Ethical Standards Conflict of interest

None of the authors have conflict of interest.

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Surgical treatment in spine Paget's disease: a systematic review.

Paget's disease of bone (PDB) is a disease characterized by a disorder in the bone metabolism. The spine is the second region affected after the pelvi...
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