Correction of Rotational Deformity Following Proximal Humeral Epiphysiolysis in a Newborn: A Case Report with Twenty-Year Follow-Up

Ogawa, Kiyohisa MD; Yamazaki, Satoshi MD; Kobayashi, Shuzo MD; Ikegami, Hiroyasu MD

Journal of Bone & Joint Surgery - American Volume:
doi: 10.2106/JBJS.E.00280
Case Reports
Author Information

1 Department of Orthopedic Surgery (K.O., S.K., and H.I.) and Sports Medicine (S.Y.), School of Medicine, Keio University, 35 Shinanomachi, Shinjuku-Ku, Tokyo 160-8582, Japan. E-mail address for K. Ogawa: ogawa51@beige.plala.or.jp

Article Outline

The remodeling potential of a long bone to correct posttraumatic angular deformities during infancy is widely recognized1-5; however, no consensus has yet been reached as to the remodeling potential of a long bone to correct rotational deformities4,6,7. To the best of our knowledge, the present report is the first to document the long-term radiographic and functional follow-up of a patient who had proximal humeral epiphysiolysis with marked rotational deformity due to birth trauma. Our patient was informed that data concerning the case would be submitted for publication.

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Case Report

Agirl weighing 3420 grams was delivered after thirty-nine weeks of gestation to a primiparous mother in an obstetrics clinic. The baby presented in the double footling breech position. The right upper extremity was forcibly manipulated because it was caught by the umbilical cord during the vaginal delivery. On the day following birth, the infant did not move the right arm and cried on passive movement of that extremity. On the third day after birth, a radiograph made at the obstetrics clinic revealed a fracture of the right clavicle (Fig. 1). Because the mother later noticed that the child had a limitation of adduction and internal rotation of the right shoulder, the infant was examined on the thirteenth day after birth at an orthopaedic clinic and radiographs made there showed callus around the proximal aspect of the humerus.

The patient was examined at our clinic on the following day. She maintained the right shoulder in an abducted and externally rotated position, and a deep cleft was visible in the skin of the lateral shoulder. A hard mass was palpable in the anteromedial aspect of the shoulder, and the humeral head was palpable under the acromion. An osseous mass, 2.5 cm in diameter, was noted at the middle of the clavicle, and an elastic mass, 2 cm in diameter, was noted in the mid-portion of the sternocleidomastoid muscle. No neurologic deficit was present. Arthrography showed that the humeral head was in its normal position in relation to the glenoid (Fig. 2). The infant was diagnosed as having epiphysiolysis of the proximal aspect of the right humerus, a clavicular fracture, and myogenic torticollis due to birth trauma.

The fractured bone had completely united without any limitation of adduction seven weeks after the patient was born. The right shoulder, with the arm at the side, had passive external rotation to 180° (the left had 100°) and 0° of internal rotation. When the child was one year and two months old, marked rotational deformity was still seen on the radiographs (Fig. 3). When the girl was one year and nine months old, the right shoulder, with the arm at the side, had passive external rotation to 160° (the left had 90°) and internal rotation to the level of L1 (to the level of T3 on the left). With the arm in 90° of abduction, the right shoulder had 180° of passive external rotation (the left had 120°) and 0° of internal rotation (90° on the left). Thereafter, the range of external rotation with the arm at the side gradually diminished and the range of internal rotation increased, but there was no appreciable change in the range of external or internal rotation with the arm in 90° of abduction. When the patient was twelve years old, the right shoulder, with the arm at the side, had 120° of external rotation (the left had 90°) and internal rotation to the level of T9 (to the level of T3 on the left). With the arm in 90° of abduction, the right shoulder had 140° of external rotation (the left had 105°) and 5° of internal rotation (the left had 60°). Humeral head retroversion was 80° on the right side (37° on the left) as measured with use of computed tomography scans according to the method of Symeonides et al.8. When the patient was sixteen years old, the right shoulder, with the arm at the side, had 120° of external rotation (the left had 90°) and internal rotation was to the level of T8 (to the level of T2 on the left). With the arm in 90° of abduction, the right shoulder had 140° of external rotation (the left had 100°) and 10° of internal rotation (the left had 70°). When the patient was twenty years old, the range of motion did not differ from that achieved when the patient was sixteen years of age; there was no evidence of excess laxity in any direction, the load-and-shift test and the apprehension test both yielded negative results, and the patient remained entirely free from limitations of daily activities.

Humeral retroversion, measured on computed tomography scans, was 77° on the right (30° on the left) at both the examination when she was sixteen years old and when she was twenty years old. The difference in torsion between the two humeral diaphyses was measured on the computed tomography scans that had been made when the patient was sixteen years old (Figs. 4-A and 4-B). This measurement demonstrated that the affected humerus was rotated uniformly throughout the central 50% of the diaphysis. Throughout the follow-up period, the humerus was consistently shorter on the right side, although the difference never exceeded 20 mm clinically or radiographically.

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Discussion

Birth trauma occurs frequently during breech extractions from primiparous mothers9. Upper-limb birth trauma occurs more often on the right side of the infant's body, with the clavicle being the most frequently fractured bone10. Proximal humeral epiphysiolysis, which is the most frequent shoulder injury11, is usually a Salter-Harris type-I injury12; the only exception, to our knowledge, was a patient with a type-II pattern reported by Jones et al.13.

An early, correct diagnosis of proximal humeral epiphysiolysis is often difficult to make because ossification of the proximal humeral epiphysis is visualized radiographically in only 50% of newborns at two weeks after birth14. Displacement of the proximal metaphysis in relation to the glenoid may be the only radiographic finding15,16. Because callus becomes demonstrable on radiographs within five to seven days after the injury, bone injury is often first diagnosed at that point9,17. Arthrography15,16,18, ultrasonography16,19, and magnetic resonance imaging20 are useful for making an early diagnosis. In our patient, the radiographic evidence of callus and the arthrographic verification of the position of the humeral head made it possible to establish the diagnosis.

Fractured long bones in children are widely recognized to have a high potential to remodel an angular deformity. An angular deformity is corrected independently in the physis according to the Hueter-Volkmann law and in the fracture region according to Wolff's law2. In a clinical study on radial fractures, Gandhi et al.1 noted that correction of an angular deformity is most rapid and most complete when there is separation at the distal epiphysis, where three-fourths of longitudinal humeral growth takes place. The proximal physis of the humerus contributes 80% of the longitudinal growth of that bone21, so fractures at that site exhibit considerable remodeling potential5. However, the ability of long bones to correct a rotational deformity remains undefined. Although no conclusion has yet been drawn from extensive clinical studies on fractures of the immature femoral shaft4,6,7, Murray et al.4, in a report on an experimental investigation demonstrating a helical growth pattern at the physis, stated that rotational deformities were able to correct as a result of this helical pattern.

The fracture in our patient resulted in at least 80° of excessive retroversion of the humerus at seven weeks, evidenced by the fact that external rotation of the affected shoulder was 80° greater than that of the unaffected shoulder. The retroversion of the humerus on the affected side, which was measured on computed tomography scans performed when the patient was sixteen and twenty years of age, was 47° greater than that on the unaffected side. Consequently, the amount of correction of the rotational deformity that was achieved with growth was 33°.

Krahl noted that retroversion of the humerus diminishes physiologically by 9° from the time of birth until adulthood22, which indicates that retroversion decreases normally with growth. Edelson23 reported that humeral retroversion in children reaches adult values by the age of sixteen years. The osseous correction of rotational deformity that we observed in our patient appears to represent, in part, the growth-associated physiological decrease in the retroversion of the humerus. On the basis of the uniform change in humeral shaft torsion that was observed on computed tomography scans made when the patient was sixteen years of age, it appears that the osseous remodeling proceeded almost uniformly during growth.

Siegel et al.24, who followed patients after in situ fixation of a slipped capital femoral epiphysis, suggested that soft-tissue stretching might account for the recovery of motion despite only minimal change in the osseous structure. In our patient, stretching of the adjoining soft tissues might have affected the improvement in motion to some extent as the residual excessive retroversion did not interfere with daily activities. Nevertheless, the soft-tissue stretching did not generate any shoulder instability.

The case of our patient serves to emphasize that surgical intervention is not required for a rotational deformity resulting from proximal humeral epiphysiolysis due to birth trauma; rather, rotational remodeling of the humerus and stretching of the adjoining soft tissues with growth can provide adequate compensation. ▪

The authors did not receive grants or outside funding in support of their research for or preparation of this manuscript. They did not receive payments or other benefits or a commitment or agreement to provide such benefits from a commercial entity. No commercial entity paid or directed, or agreed to pay or direct, any benefits to any research fund, foundation, educational institution, or other charitable or nonprofit organization with which the authors are affiliated or associated.

Investigation performed at the Department of Orthopedic Surgery and Sports Medicine, School of Medicine, Keio University, Tokyo, Japan

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