JBJS | Type-B-IIIa Hip Rotationplasty: An Alternative Operation for the Treatment of Malignant Tumors of the Femur in Early Childhood*

The Journal of Bone & Joint Surgery, Volume 82, Issue 6

Articles | June 01, 2000

Type-B-IIIa Hip Rotationplasty: An Alternative Operation for the Treatment of Malignant Tumors of the Femur in Early Childhood*

Winfried W. Winkelmann, M.D.†

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Investigation performed at the Department of Orthopedics, Westfülische Wilhelms-Universitüt Münster, Münster, Germany
*No benefits in any form have been received or will be received from a commercial party related directly or indirectly to the subject of this article. No funds were received in support of this study.
†Klinik und Poliklinik für Allgemeine Orthopüdie, Westfülische Wilhelms-Universitüt Münster, Albert-Schweitzer-StraÞ¥ 33, 48129 Münster, Germany. E-mail address: fiegeh@uni-muenster.de.

The Journal of Bone & Joint Surgery. 2000; 82:814-814

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Abstract

Background: The biological plasticity of the cartilaginous proximal part of the tibia in children makes it possible to use the tibia to reconstruct the lower extremity after excision of a sarcoma of the thigh. A type-B-IIIa rotationplasty is an alternative to prosthetic replacement in very young children who have a malignant tumor of the femur that requires extensive resection.

Methods: A type-B-IIIa rotationplasty was done in eight patients who had a femoral tumor: four had a Ewing sarcoma; three, an osteosarcoma; and one, a primitive neuroectodermal tumor. The ages ranged from two years and eight months to ten years and six months at the time of the procedure.

Results: All eight patients were able to bear full weight and had a good range of motion of the hip joint at a median of five years and one month (range, two years and four months to eight years) postoperatively. They also were able to participate in sports activities. Radiographs and magnetic resonance imaging studies confirmed that the lateral part of the tibial plateau had remodeled to form a structure that resembled a developing femoral head. Seven patients were operated on only once, and a second hospital stay was not necessary. The remaining patient had a prolonged hospitalization for revision of the wound.

Conclusions: As an alternative to amputation or an extendable tumor prosthesis, a type-B-IIIa rotationplasty offers not only a better functional result but also biological reconstruction. Placement of the cartilaginous head of the tibia into the acetabulum permits development of a new femoral head. Thus, not only is the foot preserved as a functional knee joint but a newly formed hip joint develops as well.

Figures in this Article

Introduction

Introduction | Materials and Methods | Results | Discussion | References

The most common malignant bone tumors in children and adolescents are osteosarcoma and Ewing sarcoma. The femur was the site of a Ewing sarcoma in 24 percent (fifty-seven) of 235 children less than ten years old⁴, and it was the site of an osteosarcoma in 57 percent (110) of 192 children less than ten years old²⁰.

Limb-salvage procedures, particularly in such young patients, include either use of vascularized bone grafts in combination with massive allografts for the treatment of diaphyseal defects^8,9,16,27,37 or, when the hip or knee joint has to be resected, use of an extendable prosthesis^{13,14,17,31,32,42,46,48}. These reconstructive procedures are associated with very high rates of primary and secondary complications and, very often, with multiple hospital stays. Even after a patient has reached skeletal maturity, the prognosis with regard to function and longevity of the salvaged limb is still uncertain. Therefore, in very young patients, an amputation or a rotationplasty is the preferred procedure^{6,22,23,33,49,51,52}.

A hip rotationplasty is performed in patients who have a malignant tumor of the proximal part of the femur either with or without involvement of the hip, in those who have a tumor of the pelvis, and in those who have involvement of the entire femur, necessitating its removal. Both the hip joint and the knee joint are replaced. Either the distal part of the femur or the proximal end of the tibia is rotated 180 degrees and then placed in the acetabulum. In older patients, an endoprosthesis is placed in the acetabulum.

A rotationplasty is a single operation associated with a relatively low rate of primary and secondary complications. Cammisa et al.⁶ performed a rotationplasty in twelve patients and reported delayed wound-healing in three and a deep infection, a nonunion, and compartment syndrome in one patient each. Gottsauner-Wolf et al.²² reported that, of seventy patients who had had a rotationplasty, seven had a vascular complication, eight had delayed wound-healing, two had a deep infection, one had malrotation, six had a delayed union, eight had a fracture, and seven had a nerve palsy (which became permanent in two of them). In my previous report⁵² on 134 patients who had had a rotationplasty, seven patients had a vascular complication, eight had delayed wound-healing, two had a deep infection, four had a pseudarthrosis, five had malrotation, two had a compartment syndrome, twelve had a fracture, and nine had a nerve palsy (which became permanent in two of them). Surgeons who have preferred to use an expandable prosthesis in the past have begun to prefer, in very young children, a rotationplasty because of the lower prevalence of complications³⁴.

I believe that, in young children, the lateral condyle of the tibia, under a variety of tensile forces, can remodel and form a semblance of a femoral head. In support of this hypothesis, I report the results of rotationplasty in eight patients.

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Fig. 1-A:: Figs. 1-A through 1-G: Drawings illustrating the steps of the type-B-IIIa hip rotationplasty (see text for details).
Fig. 1-A: The incisions are made with the patient positioned on the unaffected side.

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Fig. 1-B:: The proximal part of the operation is similar to a hip disarticulation.

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Fig. 1-C:: The distal part of the operation is similar to a knee disarticulation.

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Fig. 1-D:: After the leg is rotated 180 degrees, the lateral aspect of the proximal part of the tibia is placed into the acetabulum and fixed in position.

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Fig. 1-E:: The medial and lateral heads of the gastrocnemius and the tendons of the rotators are attached to the hip capsule, and the tendon of the iliopsoas muscle is attached to the tibial periosteum.

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Fig. 1-F:: The femoral artery and vein are anastomosed to the popliteal artery and vein, the adductor muscles are reattached to the tibial periosteum and fasciae, the tendons of the gluteus medius and minimus are fixed to the tibial periosteum and attached to the capsule, and the gluteus maximus muscle is attached along the tibial periosteum.

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Fig. 1-G:: The overlapping skin edges are excised with the greater part of the skin taken from the distal flap.

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Fig. 2-A:: Figs. 2-A and 2-B: Case 6. Photographs showing the functional outcome and sports activity of a nine-year-old girl five years following a rotationplasty.
Fig. 2-A: The patient is shown performing range-of-motion exercises for the hip and knee as well as weight-bearing activities while wearing the final prosthesis.

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Fig. 2-B:: The patient is shown jumping over a hurdle.

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Fig. 3-A:: Figs. 3-A through 3-D: Case 2. Radiographs showing sequential changes that led to conversion of the lateral aspect of the proximal part of the tibia into a new femoral head over a seven-year period.
Fig. 3-A: Five weeks postoperatively, there is atrophy due to inactivity of the proximal part of the tibia. The medial part of the tibia is not well centered in the acetabulum.

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Fig. 3-B: Three years and four months postoperatively, there are signs of remodeling, with formation of a partial femoral head and acetabulum.

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Fig. 3-C: Six years and ten months postoperatively, there is increasing centering of the femoral head, which is starting to have a more normal appearance. The medial part of the tibial physeal plate is oriented toward the vector of force. There is appositional bone growth on the medial side of the proximal part of the tibia, and the compressive trabeculae are merging into the thickened tibial cortex.

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Fig. 3-D: The contralateral hip is shown for comparison.

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Fig. 4-A:: Figs. 4-A and 4-B: Case 6. Magnetic resonance imaging scans showing the newly formed lateral aspect of the femoral head five years and nine months after rotationplasty.
Fig. 4-A: Three sections from the axial T2-weighted fast-spin-echo sequence showing good centering of the newly formed femoral head within the acetabulum. The thick cartilage covering can be seen.

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Fig. 4-B:: Three sections from the coronal T1-weighted fast spin-echo sequence showing the thick cartilage covering the newly formed femoral head that conforms with the acetabulum. The gluteus muscles are attached to the newly developed greater trochanter.

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TABLE I: Data on the Eight Patients Who Had a Type-B-IIIa Rotationplasty

*EICESS/CESS = vincristine, actinomycin D, ifosfamide, and Adriamycin (doxorubicin)^29,30,39; COSS = Adriamycin, cisplatin, ifosfamide, and high-dose methotrexate^21,53; and EVAIA = etoposide, vincristine, actinomycin D, ifosfamide, and Adriamycin.NED = no evidence of disease, and DOD = died of disease.This patient had thoracotomy and insertion of a tumor prosthesis in the proximal part of the right humerus.Â§This patient had necrosis of the wound requiring revision.

Case

Gender, Age at Op. (yrs. + mos.)

Date of Op.

Tumor

Preop. Complications

Neoadjuvant Chemother.*

Radiat. Ther.

Histological Response⁴¹(grade)

Durat. of Hospital Stay (days)

Addit. Hospital Stay for Complications

Durat. of Follow-up (yrs. + mos.)

Status at Latest Follow-up

Range of Motion (degrees)

Use of External Support

Radiographic Result

Hip

Knee Flexion/ Extension

Type

Site

Stage

Flexion/ Extension

Abduction/ Adduction

Ext./Int. Rotation

M, 2 + 8

9/91

Ewing sarcoma

Prox. + mid. parts of femur

IIB

CESS

8 + 0

NED

100/0

35/30

25/20

90/0

Excellent

F, 5 + 1

1/92

Osteo-sarcoma

Mid. part of femur

IIB

COSS

7 + 7

NED

75/0

35/15

20/10

80/0

Cane for walking long distances

Excellent

F, 3 + 5

12/92

Ewing sarcoma

Prox. + mid. parts of femur

IIB

Pathol. fract.

CESS

6 + 8

NED

80/0

30/30

25/10

80/0

Excellent

F, 10 + 0

2/93

Osteosarcoma

Almost entire femur

IIIB

COSS

III

Yes

3 + 5

DOD

100/0

40/10

20/15

90/0

Good

M, 8 + 0

7/94

Ewingsarcoma

Prox. part of femur

IIB

Pathol. fract. Â¥ 2; severe malrotat., femur; partial palsy, sciatic nerve; 8-cm limb-length discrep.

EVAIA

60 Gy at 3 yrs. preop.

46Â§

5 + 1

NED

60/0

20/10

10/15

20/0

Moderate

F, 4 + 0

8/94

Ewing sarcoma

Almost entire femur

IIB

EICESS

III

5 + 0

NED

110/0

35/35

50/40

100/0

Excellent

M, 4 + 3

11/94

Primitive neuroecto- dermal tumor

Prox. + mid. parts of femur

IIB

EVAIA

4 + 10

NED

80/0

20/15

25/30

Excellent

M, 10 + 6

4/97

Osteo- sarcoma

Prox. + mid. parts of femur

IIB

COSS

2 + 4

NED

80/0

20/20

90/0

Good

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TABLE I: Data on the Eight Patients Who Had a Type-B-IIIa Rotationplasty

Case

Gender, Age at Op. (yrs. + mos.)

Date of Op.

Tumor

Preop. Complications

Neoadjuvant Chemother.*

Radiat. Ther.

Histological Response⁴¹(grade)

Durat. of Hospital Stay (days)

Addit. Hospital Stay for Complications

Durat. of Follow-up (yrs. + mos.)

Status at Latest Follow-up

Range of Motion (degrees)

Use of External Support

Radiographic Result

Hip

Knee Flexion/ Extension

Type

Site

Stage

Flexion/ Extension

Abduction/ Adduction

Ext./Int. Rotation

M, 2 + 8

9/91

Ewing sarcoma

Prox. + mid. parts of femur

IIB

CESS

8 + 0

NED

100/0

35/30

25/20

90/0

Excellent

F, 5 + 1

1/92

Osteo-sarcoma

Mid. part of femur

IIB

COSS

7 + 7

NED

75/0

35/15

20/10

80/0

Cane for walking long distances

Excellent

F, 3 + 5

12/92

Ewing sarcoma

Prox. + mid. parts of femur

IIB

Pathol. fract.

CESS

6 + 8

NED

80/0

30/30

25/10

80/0

Excellent

F, 10 + 0

2/93

Osteosarcoma

Almost entire femur

IIIB

COSS

III

Yes

3 + 5

DOD

100/0

40/10

20/15

90/0

Good

M, 8 + 0

7/94

Ewingsarcoma

Prox. part of femur

IIB

Pathol. fract. Â¥ 2; severe malrotat., femur; partial palsy, sciatic nerve; 8-cm limb-length discrep.

EVAIA

60 Gy at 3 yrs. preop.

46Â§

5 + 1

NED

60/0

20/10

10/15

20/0

Moderate

F, 4 + 0

8/94

Ewing sarcoma

Almost entire femur

IIB

EICESS

III

5 + 0

NED

110/0

35/35

50/40

100/0

Excellent

M, 4 + 3

11/94

Primitive neuroecto- dermal tumor

Prox. + mid. parts of femur

IIB

EVAIA

4 + 10

NED

80/0

20/15

25/30

Excellent

M, 10 + 6

4/97

Osteo- sarcoma

Prox. + mid. parts of femur

IIB

COSS

2 + 4

NED

80/0

20/20

90/0

Good

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TABLE II: Results of Gait Analysisin Four Patients*

*The values are given for the affected limb/contralateral limb.

Case	Trunk Motion (degrees)		Range of Motion (degrees)		1st Force Maximum (percent of body weight)	Force Minimum (percent of body weight)	2nd Force Maximum (percent of body weight)	Impulse (nsec.)	Stride Length (m)	Gait Velocity (m/sec.)
Hip	Knee
1	11.4/4.0	36.2/55.2	39.7/66.8	107/120	89/79	98/117	8.3/11.1	1.20/-	1.04/-
2	10.4/2.3	28.8/51.2	51.3/64.7	122/138	71/53	105/119	6.8/11.2	1.56/-	1.45/-
3	12.5/2.8	43.6/49.0	55.7/66.3	123/124	76/75	113/110	7.3/9.6	1.30/-	1.24/-
4	9.4/2.7	50.0/62.1	56.3/70.6	133/165	67/41	109/141	4.2/5.0	1.36/-	1.40/-
Mean ± stand. dev.	10.9 ± 1.3/ 3.0 ± 0.7	39.7 ± 9.2/54.4 ± 5.8	50.8 ± 7.7/67.1 ± 2.5	121.3 ± 10.7/136.8 ± 20.4	75.8 ± 9.6/ 62.0 ± 18.1	106.3 ± 6.4/121.8 ± 13.4	6.7 ± 1.7/9.2 ± 2.9	1.36 ± 0.15/-	1.28 ± 0.18/-

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TABLE II: Results of Gait Analysisin Four Patients*

*The values are given for the affected limb/contralateral limb.

Case	Trunk Motion (degrees)		Range of Motion (degrees)		1st Force Maximum (percent of body weight)	Force Minimum (percent of body weight)	2nd Force Maximum (percent of body weight)	Impulse (nsec.)	Stride Length (m)	Gait Velocity (m/sec.)
Hip	Knee
1	11.4/4.0	36.2/55.2	39.7/66.8	107/120	89/79	98/117	8.3/11.1	1.20/-	1.04/-
2	10.4/2.3	28.8/51.2	51.3/64.7	122/138	71/53	105/119	6.8/11.2	1.56/-	1.45/-
3	12.5/2.8	43.6/49.0	55.7/66.3	123/124	76/75	113/110	7.3/9.6	1.30/-	1.24/-
4	9.4/2.7	50.0/62.1	56.3/70.6	133/165	67/41	109/141	4.2/5.0	1.36/-	1.40/-
Mean ± stand. dev.	10.9 ± 1.3/ 3.0 ± 0.7	39.7 ± 9.2/54.4 ± 5.8	50.8 ± 7.7/67.1 ± 2.5	121.3 ± 10.7/136.8 ± 20.4	75.8 ± 9.6/ 62.0 ± 18.1	106.3 ± 6.4/121.8 ± 13.4	6.7 ± 1.7/9.2 ± 2.9	1.36 ± 0.15/-	1.28 ± 0.18/-

Materials and Methods

Introduction | Materials and Methods | Results | Discussion | References

Since 1991, a type-B-IIIa rotationplasty was performed in eight children, who had a mean age of five years and eleven months (range, two years and eight months to ten years and six months), at the Westfülische Wilhelms-Universitüt Hospital in Münster. Four patients had a Ewing sarcoma, three had an osteosarcoma (stage IIB in two and stage IIIB in one), and one had a primitive neuroectodermal tumor involving the femur. Two patients (Cases 3 and 5) had a history of pathological fracture of the femur (Table I). One of them (Case 5) had had two fractures secondary to radiation therapy three years earlier. He also had malalignment, axial deviation, shortening of the extremity, and a partial paralysis of the sciatic nerve. Another patient (Case 4) had two metastases to the lungs; these lesions were removed in a second operation after the rotationplasty. All eight patients had removal of the entire thigh followed by a type-B-IIIa rotationplasty. All were treated with neoadjuvant multiple-drug chemotherapy (Table I).

We used the functional scoring system of Enneking et al.¹⁹ to evaluate orthopaedic sequelae. The score is an observer score and is based on factors that affect the overall lifestyle of the patient (pain, emotional acceptance, and restriction of activities of daily living and work) as well as factors specific to the affected extremity (use of external supports, walking ability, and gait). Each of these six factors was assigned a value of 0 to 5 points (maximum overall score, 30 points) on the basis of established criteria.

Quality of life was assessed with use of the quality-of-life questionnaire (EORTC QLQ-C30) developed by the European Organization for Research and Treatment of Cancer¹. The questionnaire comprises nine multiple-item scales: five functional scales (physical, role, emotional, social, and cognitive); three symptom scales (fatigue, pain, and nausea and vomiting); and a global health-and-quality-of-life scale. On the functional scales, a score of 100 points indicates that there is no restriction of any parameter, whereas on the symptom scales 100 points indicates maximum severity of the symptoms (for example, 100 points on the pain scale refers to maximum restriction due to pain). Other single-item measures of symptoms also are included in the questionnaire.

Four patients had a gait analysis, performed with use of a passive-marker system (Motion Analysis, Palo Alto, California). Reflective markers were applied to the shoulder, the thoracic and lumbar spine, the posterior superior iliac spine, the greater trochanter, and the lateral aspects of the knee and ankle. Marker movements were recorded with four infrared cameras at a frequency of sixty hertz. Simultaneously, the ground-reaction force was measured with two three-dimensional strain-gauge force-plates (Hentschel System, Hannover, Germany), which were embedded in a twelve-meter-long walkway. The patients were asked to walk at a freely chosen, comfortable speed. As soon as a consistent cadence of stride was reached, data from six trials was recorded and subsequently analyzed. The active range of motion of the hip and knee joints as well as movements of the trunk and pelvis were estimated. The cinematic information from the motion analysis in combination with the ground-reaction-force data was used to evaluate the movements acting on the hip joint⁴⁰.

During the follow-up period, all patients had magnetic resonance imaging of the hip joint, performed with use of a 1.5-tesla magnet (Magnetom Vision; Siemens, Erlangen, Germany) every six months. T1-weighted spin-echo and T2-weighted fast-spin-echo sequences were used to assess the articular cartilage in the sagittal, axial, and coronal planes.

Operative Technique

The patient is positioned on the unaffected side. A longitudinal incision is made on the posterior aspect of the thigh and is joined with proximal and distal circumferential oval incisions. A longitudinal lateral skin incision is also made from the distal circumferential incision of the leg (Fig. 1-A).

The proximal part of the operation is similar to a hip disarticulation. It is recommended that, if possible, the hip-joint capsule and its surrounding muscles be preserved. The sciatic nerve is identified along the posterior incision in the thigh and is dissected and preserved in its entire length. The femoral nerve is dissected immediately after it has passed beneath the inguinal ligament. The femoral artery and vein are dissected to the point where the deep femoral artery and vein branch off, and they are cut at this point (Fig. 1-B).

The distal part of the operation is similar to a knee disarticulation. The popliteal artery and vein are dissected to the adductor canal, where they are later cut. The medial and lateral heads of the gastrocnemius muscle are detached at their tendinous insertion. The knee-joint capsule is incised circumferentially, with retention of as much of its tendinous part as possible. In order to guarantee good placement of the lateral tibial condyle into the acetabulum, it is necessary to shorten the fibula by five centimeters and to resect the fibular head (Fig. 1-C).

After the leg has been rotated 180 degrees, the lateral aspect of the proximal part of the tibia is placed into the acetabulum and is fixed in position by performing a capsulorrhaphy between the remaining hip and knee-joint capsules and attaching the cruciate ligaments to the round ligament of the femoral head (Fig. 1-D). The medial and lateral heads of the gastrocnemius are attached to the newly formed capsule of the hip joint. The tendon of the iliopsoas muscle is attached to the periosteum of the tibia as distally as possible. The tendons of the rotators are attached posteriorly to the capsule (Fig. 1-E).

A tension-free end-to-end anastomosis of the femoral artery and vein is performed with use of the popliteal artery and vein, respectively. Medially, the diagonal edge of the adductor muscles is reattached to the periosteum of the tibia and its fasciae. Laterally, the tendons of the gluteus medius and minimus muscles are fixed to the periosteum of the tibia and are attached to the new hip capsule as distally as possible. Finally, the gluteus maximus is fixed to the periosteum of the tibia. The anastomoses of the great vessels as well as the sciatic nerve are placed subcutaneously (Fig. 1-F). The overlapping skin edges are excised in order to obtain a smooth, tension-free closure (Fig. 1-G).

The closure of the incision is a very important step in the operation, and it should be taken into consideration during the planning of the procedure. The overlapping skin edges are placed circumferentially one on top of the other, and the redundant skin is carefully resected within these boundaries so that a tension-free subcutaneous and cutaneous closure can be achieved. As a rule, only the skin attached to the distal part of the extremity is excised, as the skin over the buttocks is better vascularized and more robust, allowing it to withstand the pressure exerted by the prosthesis. Because of the possibility that a hip disarticulation may be performed in the future, the skin of the buttocks should be preserved. We used metal clips instead of sutures for closure of the skin, as they can be removed easily. Absorbable sutures also can be used for the closure.

Postoperative Regimen

Postoperatively, a hip-spica cast is used to immobilize the extremity. A window is cut in the cast to permit the extremity to be lifted out and visualized circumferentially in order to assess the vascularity of the rotated leg. Vascularity also is measured with use of a pulse oximeter over the great toe.

All patients were given an age-appropriate dose of 100 international units of standard heparin per kilogram of body weight each day for one week, for prophylaxis against thrombosis.

Slight swelling related to the lymphatic circulation is often present for six to eight weeks after the operation. Intermittent elevation of the extremity for one to two hours, during the period when the child is receiving physical therapy and chemotherapy, will decrease the swelling.

After the wound has healed completely, a plastic splint consisting of two sections is used so that gentle passive motion of the extremity can be provided by the physiotherapist. The patient remains on the orthopaedic service for two weeks and is then transferred for chemotherapy.

At four weeks, the patient is allowed to begin physical therapy and to practice sitting, transfers, and walking with crutches. A temporary prosthesis is made at six weeks, primarily to allow the patient to exercise the hip and knee joints and not for weight-bearing. This makes it easier for the patient to practice walking with crutches. The final prosthesis is provided after the chemotherapy is completed, as until that time the patient is unable to participate in all of the required prosthetic training.

Results

Introduction | Materials and Methods | Results | Discussion | References

On clinical examination of the active range of motion of the hip, flexion averaged 86 degrees (range, 60 to 110 degrees); extension, 0 degrees; abduction, 29 degrees (range, 20 to 40 degrees); adduction, 21 degrees (range, 10 to 30 degrees); external rotation, 24 degrees (range, 20 to 50 degrees); and internal rotation, 20 degrees (range, 10 to 40 degrees). Passive flexion averaged 90 degrees. An upright sitting position with fully extended legs was always possible (Fig. 2-A and Table I).

With the foot acting as a new knee joint, active knee flexion (achieved with ankle dorsiflexion) averaged 80 degrees (range, 20 to 100 degrees) and active knee extension (achieved with ankle plantar flexion) was 0 degrees in seven patients and -10 degrees in one patient. There was no loss of deep sensation or joint-position sense of the extremity in seven patients. One patient (Case 5) had had a partial paralysis of the sciatic nerve preoperatively that was due to irradiation for the treatment of a Ewing sarcoma three years earlier. The paralysis worsened immediately after the procedure, but there was improvement later.

The median functional Enneking score¹⁹ was 75 percent (range, 60 to 82 percent) of a maximum total score of 100 percent. All patients wore the exoprosthesis every day. Only one patient needed a cane, for walking long distances. The quality-of-life analysis¹ revealed a good result for physical functioning (median score, 60 percent), social functioning (median score, 75 percent), and role functioning (median score, 67 points). No patient had restrictions in daily activities. All patients participated in school sports (Fig. 2-B). One patient complained of mild pain; however, no patient required pain medication.

Radiographs made at the two-year follow-up evaluation showed conversion of the lateral aspect of the proximal part of the tibia into a new femoral head in all eight patients. The criteria that were used to assess the radiographic findings included remodeling of the femoral head, orientation of the medial part of the tibial physis in the direction of the vector of force, appositional bone growth, and the presence of compressive trabeculae in the thickened medial tibial cortex. The acetabulum also showed remodeling, which conformed to the shape of the new femoral head (Fig. 3-A, Fig. 3-B, and Fig. 3-C). The lateral part of the epiphyseal line adapted to the axial load. The structure of the trabeculae of the lateral aspect of the proximal part of the tibia was similar to that of the femoral neck (Fig. 3-C and Fig. 3-D).

The radiographic result was rated as excellent, good, or moderate. Five patients had an excellent result; two, a good result; and one, a moderate result. Both patients who had a good result had had the operation when they were ten years old. The patient who had the moderate result was the one who had a partial paralysis of the sciatic nerve; he did not regain good active or passive motion of the hip.

Seven of the eight patients were alive at the time of this writing. One patient (Case 4), who had had a stage-IIIB osteosarcoma, died three years and five months after the procedure. In addition to having had a thoracotomy because of lung metastases, she had had placement of a prosthesis in the proximal part of the right humerus because of bone metastases.

Magnetic resonance imaging demonstrated continuous rounding of the cartilaginous surface of the lateral aspect of the proximal part of the tibia and corresponding remodeling of the cartilage of the acetabulum in all eight patients (Fig. 4-A and Fig. 4-B).

In general, the patients had a functional gait. However, there was evidence of attempts to unload the hip, as indicated by a limp and a reduced range of motion of the hip and knee joints. The ground-reaction forces indicated a reduced loading response and terminal stance peak as well as pronounced unloading in midstance. The impulse (force-time integral) also indicated a slight unloading of the affected limb. The stride length and gait velocity were acceptable for this age-group (Table II).

Complications

One patient (Case 5) had necrosis of the wound that required revision and a prolonged hospital stay. (This was the same patient who had had a partial nerve palsy preoperatively that worsened after the procedure and was still present at the time of writing.) Two patients (Cases 4 and 8) sustained a greenstick fracture of the tibia, at twelve and eighteen months, respectively; the fracture healed after treatment in a cast. No patient had a vascular complication or a deep infection.

Discussion

Introduction | Materials and Methods | Results | Discussion | References

When children who have a malignant tumor of the femur requiring a total resection of the thigh are treated with a total femoral endoprosthesis, they often need multiple revision procedures to extend the prosthesis until the completion of growth^{14,15,17,32,34,42}. These operations are usually not minor; generally, they involve soft-tissue releases or scar resection, and an even more extensive operation, such as repair of an implant or a reconstructive procedure, may be necessary when there is a dysplastic acetabulum^34,42. Even when a patient receives a modular prosthesis, there may be late complications, such as aseptic loosening of the prosthesis, implant failure, or deep infection. For these reasons, and also because it does not take advantage of the childhood ability for biological reconstruction, use of an extendable femoral prosthesis is no longer indicated in young children⁵⁴.

Kotz and Salzer³³ introduced rotationplasty, which was originally performed in patients who had infectious or congenital malformations^5,44, for the treatment of osteosarcoma of the distal part of the femur. The procedure also was performed for the treatment of tumors of the proximal part of the tibia. After resection of the tumor, the distal aspect of the extremity, including the foot, was rotated 180 degrees and attached proximally to the remaining femur with osteosynthesis. The hip joint was preserved, and the ankle joint functioned as a knee joint. I previously described 134 patients who had had different types of rotationplasty⁵².

Major reconstructive procedures are associated with a higher prevalence of primary and secondary complications. In a study of the results of reconstruction with use of a vascularized fibular graft in fifty patients, Capanna et al.¹⁰ reported a stress fracture of the fibula in nine patients (18 percent), a nonunion in four (8 percent), and a deep infection in two (4 percent). After reconstruction with an allograft in thirty-two patients, Capanna et al.⁹ reported a nonunion in six patients (19 percent), a fracture in three patients (9 percent), and a deep infection in one patient (3 percent). In a study in which massive allografts had been used in forty-one patients, Dubousset and Missenard¹⁶ reported an 83 percent rate of revision (thirty-four patients) because of major complications such as fracture, nonunion, infection, and necrosis. Mankin et al.³⁷ reported that, of 219 patients treated with allograft transplantation, thirty-one (14 percent) had a deep infection; thirty-six (16 percent), a fracture of the allograft; and thirty-one (14 percent), a nonunion. Isler et al.²⁷ noted six nonunions (20 percent), nine fractures (30 percent), and ten infections (33 percent) in thirty patients who had had insertion of a distal femoral osteoarticular allograft.

The highest rate of complications has been reported after reconstruction with use of an expandable prosthesis. Kenan and Lewis^31,32 reported on thirty children in 1991 and forty children in 1995 who were followed for two to ten years after insertion of an expandable tumor prosthesis. Combination of the data from the two groups revealed that twenty-three (33 percent) of the seventy patients had a revision procedure, seventeen (24 percent) had aseptic loosening or stem migration, four (6 percent) had a late deep infection, and one (1 percent) had a broken prosthesis. Eighteen of twenty-one patients who were followed for more than five years had a revision procedure. The overall mean duration of survival for the cemented prostheses was five years and eight months compared with four years and two months for the press-fit prostheses. Eckardt et al.¹⁷ reported that a revision procedure had been performed on seven of twelve skeletally immature patients who had been followed for a minimum of two years after insertion of an expandable endoprosthesis. Delepine et al.¹⁴ reported four deep infections and two instances of aseptic loosening in thirty-two children who had been treated with an expandable prosthesis.

In 1995, Schiller et al.⁴² reported on twenty children who were managed with an expandable prosthesis, and in 1997, Dominikus et al.¹⁵, from the same department, reported on twenty-three children who were managed with such a prosthesis. Three of six children with long-term follow-up had a deep infection. The authors concluded that the high rate of infection was related to the multiple operations performed to lengthen the prosthesis.

The higher rate of primary and secondary complications after use of an expandable prosthesis is associated with multiple hospital admissions. The total number of operations required for revision of a prosthesis in order to preserve the extremity, as well as the number of procedures required to lengthen the extremity, depends on the age of the patient. Kenan and Lewis³² estimated that ten to fifteen procedures per patient would be necessary during a ten-year follow-up period. Schiller et al.⁴² reported a median of eleven operations (range, seven to eighteen operations) in six patients who were between nine and eleven years old at the time of the first operation.

Even after a child has reached skeletal maturity, there is still an uncertain prognosis for the affected limb. Wirganowicz et al.⁵⁴, in a survivorship analysis of 278 patients, reported a mean failure rate (and standard deviation) of 30 ± 4 percent after the index procedure and 31 ± 12 percent after the first revision procedure in thirty-eight patients. Adult patients who had been followed for seven years had a failure rate of 31 percent after the index procedure and 34 percent after revision⁵⁴. Very young patients who originally had had insertion of an expandable prosthesis had bone atrophy or partial bone resorption near the prosthesis as a result of load-shielding, necessitating revision with a longer prosthesis. Cobb et al.¹¹ reported an 83 percent rate of survival of the implant (in forty-nine of fifty-nine patients) at ten years in a series consisting of adults who had had a cemented distal femoral replacement for the treatment of a low-grade tumor. The rate of implant survival in younger patients (66 percent; seventy-two of 109 patients) was less satisfactory. In another study, of 493 patients with a distal femoral replacement, Cobb et al.¹² reported that the ten-year survival rate dropped from 94 to 49 percent when the resected segment was more than 40 percent of the distal part of the femur.

In 1993, I reported on a specific type of hip rotationplasty that can be used in very young children when a total thigh resection is indicated⁵¹. After complete resection of the thigh, the remainder of the extremity was rotated 180 degrees and the lateral tibial condyle was placed in the acetabulum. The basis of this procedure, and particularly the influence of tensile forces such as extension and flexion on the growth of the epiphysis, was reported earlier^25,44, as was the influence of growth and bone-remodeling^18,55,56. Experimental investigations involving the tibial physis have confirmed the principles of bone-remodeling and have demonstrated the plasticity of growing bone^2,3,18,47. The plasticity of growing bone under the influence of tensile forces was first reported by Hueter²⁵ and Volkmann⁴⁵. Major adaptation of growing bone, particularly in the proximal epiphysis of the tibia, was found in experimental investigations^2,3. This plasticity of bone is used in the treatment of Perthes disease and other epiphyseal and metaphyseal disturbances of growth. The childhood ability for biological reconstruction also has been utilized in reconstructive tumor surgery. When the proximal part of the fibula with an epiphyseal plate is transplanted as a vascularized graft to replace the distal part of the radius, the proximal part of the humerus, or the proximal part of the femur in a child, it remodels within a few years secondary to the new weight-bearing and functional forces of the anatomically re-formed distal part of the radius, humeral head, or femoral head^7,26,36,38. If the vascularized clavicle is used as a replacement for the proximal part of the humerus, it remodels within a few years and becomes a robust straight bone⁵⁰.

The functional results of a type-B-IIIa rotationplasty followed by fitting of an exoprosthesis are similar to those of other rotationplasties such as the type-A-I procedure. McClenaghan et al.³⁵ and van der Windt et al.⁴³, in long-term follow-up studies, found that walking efficiency after a type-A-I rotationplasty was better than that after an above-the-knee amputation, a hip disarticulation, or an arthrodesis, and it was comparable with that after endoprosthetic replacement. A follow-up study from my department, on functional outcomes and quality-of-life measurements for patients who were managed with a femoral endoprosthesis or a rotationplasty, revealed good results with regard to gait and walking in both groups²³. According to the scoring system of the Musculoskeletal Tumor Society¹⁹, the thirty-three patients who had had a rotationplasty had a mean functional score of 24 points and the thirty-four patients who had had an endoprosthetic replacement had a mean score of 25 points; this difference was not significant (p < 0.001). Only one patient who had had a rotationplasty used a cane as an external support for long-distance walking, whereas an external support was necessary for the six patients who had had an endoprosthetic replacement²³.

Cammisa et al.⁶ found that the net energy expended during walking was less after a rotationplasty than it was after an above-the-knee amputation or an endoprosthetic replacement. Hillmann et al.²³ and Hoffmann et al.²⁴ noted that a quality-of-life questionnaire showed that patients who had had a rotationplasty could participate in daily activities such as carpentry and sports to a significantly greater degree than could those who had had an endoprosthetic replacement (p = 0.001). Restriction in daily activities due to pain was significantly less common in the group that had had a rotationplasty than it was in the group that had had an endoprosthetic replacement (p = 0.047). Gottsauner-Wolf et al.²² believed that patients who had had a rotationplasty could take an active part in sports activities such as downhill and cross-country skiing, jogging, and soccer; however, they advised patients who had an endoprosthesis to avoid strenuous activities and encouraged them to adopt a sedentary lifestyle and occupation.

The better role functioning and the absence of differences in social and emotional functioning suggest that there are no psychological disadvantages associated with rotationplasty compared with a tumor prosthesis²⁴. Other authors have reported that patients are not disturbed by the appearance of the rotationplasty and that they consider it to be more of a limb-salvage procedure than a special type of amputation^28,33.

In conclusion, for children less than ten years old, a type-B-IIIa rotationplasty should be recommended as an alternative to a tumor prosthesis because of the high risk of complications associated with the latter form of treatment, which can result in additional hospital stays and eventually can lead to an amputation^7,15,37. Compared with a tumor prosthesis, a type-B-IIIa rotationplasty seems to have no disadvantages relative to complications, mobility, or psychosocial readjustment, and it has been associated with excellent biological conversion and adaptation of the growing skeleton. It is definitely valuable for patients to take advantage of this biological potential.

References

Introduction | Materials and Methods | Results | Discussion | References

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Fig. 1-B:: The proximal part of the operation is similar to a hip disarticulation.

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Fig. 1-C:: The distal part of the operation is similar to a knee disarticulation.

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Fig. 1-D:: After the leg is rotated 180 degrees, the lateral aspect of the proximal part of the tibia is placed into the acetabulum and fixed in position.

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Fig. 1-G:: The overlapping skin edges are excised with the greater part of the skin taken from the distal flap.

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Fig. 2-B:: The patient is shown jumping over a hurdle.

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Fig. 3-B: Three years and four months postoperatively, there are signs of remodeling, with formation of a partial femoral head and acetabulum.

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Fig. 3-D: The contralateral hip is shown for comparison.

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TABLE I: Data on the Eight Patients Who Had a Type-B-IIIa Rotationplasty

Case

Gender, Age at Op. (yrs. + mos.)

Date of Op.

Tumor

Preop. Complications

Neoadjuvant Chemother.*

Radiat. Ther.

Histological Response⁴¹(grade)

Durat. of Hospital Stay (days)

Addit. Hospital Stay for Complications

Durat. of Follow-up (yrs. + mos.)

Status at Latest Follow-up

Range of Motion (degrees)

Use of External Support

Radiographic Result

Hip

Knee Flexion/ Extension

Type

Site

Stage

Flexion/ Extension

Abduction/ Adduction

Ext./Int. Rotation

M, 2 + 8

9/91

Ewing sarcoma

Prox. + mid. parts of femur

IIB

CESS

8 + 0

NED

100/0

35/30

25/20

90/0

Excellent

F, 5 + 1

1/92

Osteo-sarcoma

Mid. part of femur

IIB

COSS

7 + 7

NED

75/0

35/15

20/10

80/0

Cane for walking long distances

Excellent

F, 3 + 5

12/92

Ewing sarcoma

Prox. + mid. parts of femur

IIB

Pathol. fract.

CESS

6 + 8

NED

80/0

30/30

25/10

80/0

Excellent

F, 10 + 0

2/93

Osteosarcoma

Almost entire femur

IIIB

COSS

III

Yes

3 + 5

DOD

100/0

40/10

20/15

90/0

Good

M, 8 + 0

7/94

Ewingsarcoma

Prox. part of femur

IIB

Pathol. fract. Â¥ 2; severe malrotat., femur; partial palsy, sciatic nerve; 8-cm limb-length discrep.

EVAIA

60 Gy at 3 yrs. preop.

46Â§

5 + 1

NED

60/0

20/10

10/15

20/0

Moderate

F, 4 + 0

8/94

Ewing sarcoma

Almost entire femur

IIB

EICESS

III

5 + 0

NED

110/0

35/35

50/40

100/0

Excellent

M, 4 + 3

11/94

Primitive neuroecto- dermal tumor

Prox. + mid. parts of femur

IIB

EVAIA

4 + 10

NED

80/0

20/15

25/30

Excellent

M, 10 + 6

4/97

Osteo- sarcoma

Prox. + mid. parts of femur

IIB

COSS

2 + 4

NED

80/0

20/20

90/0

Good

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TABLE I: Data on the Eight Patients Who Had a Type-B-IIIa Rotationplasty

Case

Gender, Age at Op. (yrs. + mos.)

Date of Op.

Tumor

Preop. Complications

Neoadjuvant Chemother.*

Radiat. Ther.

Histological Response⁴¹(grade)

Durat. of Hospital Stay (days)

Addit. Hospital Stay for Complications

Durat. of Follow-up (yrs. + mos.)

Status at Latest Follow-up

Range of Motion (degrees)

Use of External Support

Radiographic Result

Hip

Knee Flexion/ Extension

Type

Site

Stage

Flexion/ Extension

Abduction/ Adduction

Ext./Int. Rotation

M, 2 + 8

9/91

Ewing sarcoma

Prox. + mid. parts of femur

IIB

CESS

8 + 0

NED

100/0

35/30

25/20

90/0

Excellent

F, 5 + 1

1/92

Osteo-sarcoma

Mid. part of femur

IIB

COSS

7 + 7

NED

75/0

35/15

20/10

80/0

Cane for walking long distances

Excellent

F, 3 + 5

12/92

Ewing sarcoma

Prox. + mid. parts of femur

IIB

Pathol. fract.

CESS

6 + 8

NED

80/0

30/30

25/10

80/0

Excellent

F, 10 + 0

2/93

Osteosarcoma

Almost entire femur

IIIB

COSS

III

Yes

3 + 5

DOD

100/0

40/10

20/15

90/0

Good

M, 8 + 0

7/94

Ewingsarcoma

Prox. part of femur

IIB

Pathol. fract. Â¥ 2; severe malrotat., femur; partial palsy, sciatic nerve; 8-cm limb-length discrep.

EVAIA

60 Gy at 3 yrs. preop.

46Â§

5 + 1

NED

60/0

20/10

10/15

20/0

Moderate

F, 4 + 0

8/94

Ewing sarcoma

Almost entire femur

IIB

EICESS

III

5 + 0

NED

110/0

35/35

50/40

100/0

Excellent

M, 4 + 3

11/94

Primitive neuroecto- dermal tumor

Prox. + mid. parts of femur

IIB

EVAIA

4 + 10

NED

80/0

20/15

25/30

Excellent

M, 10 + 6

4/97

Osteo- sarcoma

Prox. + mid. parts of femur

IIB

COSS

2 + 4

NED

80/0

20/20

90/0

Good

View Larger

TABLE II: Results of Gait Analysisin Four Patients*

*The values are given for the affected limb/contralateral limb.

Case	Trunk Motion (degrees)		Range of Motion (degrees)		1st Force Maximum (percent of body weight)	Force Minimum (percent of body weight)	2nd Force Maximum (percent of body weight)	Impulse (nsec.)	Stride Length (m)	Gait Velocity (m/sec.)
Hip	Knee
1	11.4/4.0	36.2/55.2	39.7/66.8	107/120	89/79	98/117	8.3/11.1	1.20/-	1.04/-
2	10.4/2.3	28.8/51.2	51.3/64.7	122/138	71/53	105/119	6.8/11.2	1.56/-	1.45/-
3	12.5/2.8	43.6/49.0	55.7/66.3	123/124	76/75	113/110	7.3/9.6	1.30/-	1.24/-
4	9.4/2.7	50.0/62.1	56.3/70.6	133/165	67/41	109/141	4.2/5.0	1.36/-	1.40/-
Mean ± stand. dev.	10.9 ± 1.3/ 3.0 ± 0.7	39.7 ± 9.2/54.4 ± 5.8	50.8 ± 7.7/67.1 ± 2.5	121.3 ± 10.7/136.8 ± 20.4	75.8 ± 9.6/ 62.0 ± 18.1	106.3 ± 6.4/121.8 ± 13.4	6.7 ± 1.7/9.2 ± 2.9	1.36 ± 0.15/-	1.28 ± 0.18/-

Add to My JBJS

TABLE II: Results of Gait Analysisin Four Patients*

*The values are given for the affected limb/contralateral limb.

Case	Trunk Motion (degrees)		Range of Motion (degrees)		1st Force Maximum (percent of body weight)	Force Minimum (percent of body weight)	2nd Force Maximum (percent of body weight)	Impulse (nsec.)	Stride Length (m)	Gait Velocity (m/sec.)
Hip	Knee
1	11.4/4.0	36.2/55.2	39.7/66.8	107/120	89/79	98/117	8.3/11.1	1.20/-	1.04/-
2	10.4/2.3	28.8/51.2	51.3/64.7	122/138	71/53	105/119	6.8/11.2	1.56/-	1.45/-
3	12.5/2.8	43.6/49.0	55.7/66.3	123/124	76/75	113/110	7.3/9.6	1.30/-	1.24/-
4	9.4/2.7	50.0/62.1	56.3/70.6	133/165	67/41	109/141	4.2/5.0	1.36/-	1.40/-
Mean ± stand. dev.	10.9 ± 1.3/ 3.0 ± 0.7	39.7 ± 9.2/54.4 ± 5.8	50.8 ± 7.7/67.1 ± 2.5	121.3 ± 10.7/136.8 ± 20.4	75.8 ± 9.6/ 62.0 ± 18.1	106.3 ± 6.4/121.8 ± 13.4	6.7 ± 1.7/9.2 ± 2.9	1.36 ± 0.15/-	1.28 ± 0.18/-