Elsevier

Bone

Volume 31, Issue 1, July 2002, Pages 96-101
Bone

Original article
Intravenous pamidronate in the treatment of transient osteoporosis of the hip

https://doi.org/10.1016/S8756-3282(02)00812-8Get rights and content

Abstract

The aim of this study was to evaluate the efficacy of intravenous pamidronate in patients with transient osteoporosis of the hip (TOH). Thirteen men and three women (mean age 38.3 years, range 30–49) were recruited. The diagnosis was made by means of radiographs, bone scintigraphy, and magnetic resonance imaging (MRI). Pamidronate (45 mg) was intravenously administered three times, once every third day. The outcome measures included a clinical assessment using a pain visual analog scale (VAS; range 0–100), and the WOMAC functional impairment score (FUI; range 0–100). The bone mineral density (BMD) of the total hip and femoral neck was measured using dual-energy X-ray absorptiometry (DXA). Clinical assessments were made before treatment (T0) and 1 month later (T1), and the densitometric measurements at T0, and then after 2 (T2) and 4 months (T4). A further MRI scan was made 3 months after treatment. In comparison to the unaffected side, there was a significant decrease at T0 in the BMD of both the total hip (median 16.6%, range 8.5%–29.1%, p < 0.00001) and femoral neck (median 22.5%, range 12.0%–34.2%, p < 0.00001). By T1, both VAS and FUI had decreased significantly (p < 0.00001). By T2, the total hip and femoral neck BMD had increased by 10.9% (range 2.7%–23.6%, p < 0.00001) and 12.3% (range 7.8%–26.9%, p < 0.00001), respectively, and all patients were asymptomatic. By T3, the MRI findings had normalized in all patients and, at T4, there was a further increase in BMD. None of the patients experienced symptom relapse during the follow-up of 39.5 ± 17.7 months. These results suggest that a short course of pamidronate is effective in treating TOH, and leads to a prompt and long-lasting recovery.

Introduction

Transient osteoporosis of the hip (TOH), also referred as bone marrow edema syndrome (BMES), describes a clinical entity of unknown etiology characterized by disabling hip pain that typically affects middle-aged men and women in the third trimester of pregnancy. The clinical course is usually characterized by an abrupt or gradual onset of pain, with functional disability increasing to a maximum within 4–8 weeks. This is followed by a period of some weeks or months during which the level of pain remains stable before a regression phase in which the pain gradually subsides. As described by Lequesne,22 the patients show a typical disproportionate functional disability in relation to a minimal restriction of movement, with pain only being elicited at the extremes of motion. A focal loss of radiodensity involving the femoral head and neck, and sometimes the trochanter, becomes apparent on plain radiographs 4–8 weeks after the clinical onset; however, bone scintigraphy radiotracer uptake and bone marrow edema, as displayed by magnetic resonance imaging (MRI), are seen earlier and appear just a few days after appearance of symptoms.

Since the first description of the disease by Curtiss and Kincaid,9 the clinical, roentgenographic,20, 23, 31, 32 scintigraphic,14 and MRI features6, 39 of TOH have been extensively reported in the literature under various names (e.g., transitory demineralization, migratory osteolysis, algodystrophy of the hip), and all investigators have described it as self-limiting with spontaneous resolution 6–24 months after a conservative approach, such as restricted weight-bearing and symptomatic treatment.

Various therapeutic approaches have been adopted in an attempt to shorten the clinical course, but there is no general agreement about their efficacy, because validated methods of objectively assessing clinical improvement have rarely been used. It has been reported that an invasive procedure, such as core decompression, will significantly reduce pain within a few weeks,16 but some investigators consider operative intervention unnecessarily aggressive and dangerous.15, 41

Over the last 10 years, a number of studies have reported positive results after the treatment of reflex sympathetic dystrophy (RSD) with bisphosphonates. Clodronate,38 pamidronate,8, 11, 24 and alendronate1 have all proved to be efficacious in the management of RSD, which has many scintigraphic, histopathological, and MRI similarities with TOH.

We assessed the therapeutic efficacy of a short intravenous pamidronate course in patients with TOH by means of clinically validated methods and X-ray absorptiometry, which proved to be helpful in the follow-up of the disease, and allowed a quantitative evaluation of the bone involvement.37

Section snippets

Patients

Between February 1995 and March 2000, 16 consecutive patients with TOH (13 men and 3 women, mean age 38.3 years, range 30–49) were recruited after having been referred to our Rheumatology Day Hospital by the Orthopedic and Rheumatological Outpatient Services and the Emergency Department. All patients complained of pain in the hip on weight-bearing; four reported pain at rest (sitting and lying in bed) and two had nocturnal pain. None reported recent trauma or illness or any other risk factors

Results

Fifteen patients completed the study: one patient refused to undergo the second MRI scan 3 months after the end of treatment, but regularly underwent BMD measurements and answered when interviewed by phone. Assuming that the onset of the disease occurred when the pain began, the mean duration of disease at entry was 8 ± 3.8 weeks (median 7). As a predisposing factor other than pregnancy, a history of unusual activity before onset of the symptoms23 was reported by two patients (a long walk and a

Discussion

Because of its rarity and the difficulty in making an early diagnosis (radiographs in the first weeks after clinical onset are often unrevealing), TOH is not widely recognized and therefore possibly underdiagnosed. As we found, no laboratory test, including bone markers, provides any useful diagnostic clues.20 About 300 cases have so far been reported in the literature, but there is still controversy about the etiology, pathophysiology, and outcome of the disease, and there is disagreement as

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