Image Quiz

Flexion Contracture and Cyclic Hip Pain in a Twenty-Three-Year-Old Woman1

A twenty-three-year-old woman was seen because of a twenty-month history of stiffness and pain in the right hip. The first symptom was stiffness in the hip, followed by the onset of pain in the right groin approximately two months later. The pain was cyclical in nature and always occurred twenty-four to forty-eight hours after the onset of her menstrual period. It lasted for ten to twelve days and then gradually subsided until her next monthly period. The pain and stiffness progressively worsened, resulting in marked external rotation and flexion of the extremity and a limp during walking. The pain was exacerbated by activity but was also present at rest and had been unresponsive to several trials of nonsteroidal anti-inflammatory medications. The patient had no history of trauma. She worked as a radiographic technician and was otherwise healthy, with her only medications being oral contraceptives for the treatment of dysmenorrhea. She had a smoking history of eight cigarettes per day for the past six years.

She was seen by an orthopaedic surgeon approximately one year after the onset of symptoms. She was noted to have marked limitation of internal rotation of the hip. Radiographs were suggestive of an old slipped capital femoral epiphysis, which was thought to explain the external rotation and flexion contracture. She was referred to a gynecologist for further evaluation of the pain. The findings of the pelvic examination were unremarkable. A pelvic ultrasound study revealed normal findings. An intravenous pyelogram did not reveal any abnormalities of the kidneys or ureters. An exploratory laparoscopy revealed papillary lesions on the surface of the right ovary. These lesions were biopsied, and the pathologic diagnosis was consistent with reactive fibrosis.

Three months later, the patient consulted another orthopaedic surgeon, who noted tenderness at the posterior-superior iliac spine and the adductor origins from the pubis as well as a fixed external rotation contracture of the hip. Magnetic resonance imaging revealed diffuse hypertrophy of the right obturator internus muscle and signal changes consistent with hemorrhage. A variety of nonsteroidal anti-inflammatory medications did not relieve the symptoms. Two months later, the patient was evaluated by a third orthopaedist, who noted that the patient stood and walked comfortably with the hip externally rotated approximately 90° during gait and that she had an external rotation contracture of 30° when lying in the supine position. Abduction was limited to 20° on the right, compared with 70° on the left. The right hip had a 30° flexion contracture, with flexion to 80°. The range of motion of the right knee was normal. A repeat gynecologic examination and Pap smear were negative.

The patient was then referred to our institution for further evaluation and treatment. At that time, she was found to walk with the right lower extremity externally rotated approximately 45°. The hip was fixed in 45° of external rotation and 5° of abduction with no internal rotation, external rotation, abduction, or adduction from this position. The right hip had a flexion arc of 30° to 70°. Straight-leg raising did not elicit pain. The girth of the right thigh was 2 cm smaller than that of the left thigh when measured 5 cm proximal to the superior pole of the patella. Sensory and vascular examinations of the lower extremities revealed normal findings. The motor examination revealed normal findings except for mild weakness of hip flexion, which was classified as grade 4 (of 5). No palpable masses or lymphadenopathy was detected.

Radiographs showed demineralization of the right femoral head and neck and external rotation of the right hip (Fig. 1). Laboratory studies revealed a hemoglobin level of 13.1 g/dL, a white blood-cell count of 7.9 × 109/L, a platelet count of 254 × 109/L, an erythrocyte sedimentation rate of 11 mm/hr, a negative test for antinuclear antibodies, a C-reactive protein level of 1.9 mg/dL, a rheumatoid factor level of <20 IU/mL, and an anti-DS-DNA level of 4 IU/mL. A triple-phase technetium-99 bone scan revealed hyperemia of the soft tissues of the medial aspect of the proximal part of the right thigh, and the bone images showed focal areas of uptake in the right ilium, the right ischium, and the inferior part of the right acetabulum. These findings were interpreted as being consistent with infiltrative disease or increased bone-remodeling secondary to bone stress.


Fig. 1

Fig. 2
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What is the diagnosis?