Yeungnam Univ J Med Search

CLOSE


J Yeungnam Med Sci > Epub ahead of print
Tabata, Matsui, Miyamoto, Omori, Tabata, and Majima: Transient osteoporosis of the hip with a femoral neck fracture during follow-up: a case report

Abstract

We report a case of transient osteoporosis of the hip with a femoral neck fracture found during follow-up. A 53-year-old man presented with left hip pain without trauma. The pain did not improve after 2 weeks and he was brought to our hospital by ambulance. Magnetic resonance imaging (MRI) of the left hip joint showed diffuse edema in the bone marrow, which was identified by low signal intensity on T1-weighted images, high signal intensity on T2-weighted images, and increased signal intensity on short tau inversion recovery. This edema extended from the femoral head and neck to the intertrochanteric area. He was diagnosed with transient osteoporosis of the left hip. Rest gradually improved his pain; however, 3 weeks later, his left hip pain worsened without trauma. X-ray, computed tomography, and MRI results of the hip joint demonstrated a left femoral neck fracture, and osteosynthesis was performed. Differential diagnoses included avascular necrosis of the femoral head, infection, complex regional pain syndrome, rheumatoid arthritis, leukemia, and other cancers. Transient osteoporosis of the hip generally has a good prognosis with spontaneous remission within a few months to 1 year. However, a sufficient length of follow-up from condition onset to full recovery is necessary to avoid all probable complications such as fractures.

Introduction

Transient osteoporosis of the hip (TOH) is a rare disease that was first reported in 1959 in three pregnant women who had strong unilateral or bilateral hip pain [1]. TOH generally occurs in middle-aged men and pregnant women in their last trimester [2-4]. Its etiology is unknown; however, it is suspected that ischemia in the femoral head could be involved in the onset of TOH [5-7]. Differential diagnosis includes avascular necrosis of the femoral head, infection, complex regional pain syndrome, rheumatoid arthritis, leukemia, and other cancers. TOH is conventionally a disease with a good prognosis and spontaneous remission within a few months to 1 year. We report a case of TOH where a femoral neck fracture later occurred without trauma, and osteosynthesis was performed.

Case

Ethical statements: Written informed consent was obtained from the patient for this case report. This study was approved by the Institutional Review Board (IRB) of Musashino General Hospital (IRB No: 12).
A 53-year-old man presented with left hip pain without trauma that had persisted for 2 weeks. After the pain did not subside, he was brought to our hospital by ambulance. He had schizophrenia and epilepsy resulting from a previous head injury. He drank approximately 1.5 L of beer every day and had smoked 20 cigarettes per day for 37 years.
On the first admission, he had difficulty walking due to severe left hip pain. The blood examination was normal. His left hip joint demonstrated painful limitations in passive range of motion. Patrick’s test was positive.
X-ray and computed tomography (CT) revealed no obvious fracture (Fig. 1). Magnetic resonance imaging (MRI) showed diffuse edema in the bone marrow, which was identified by low signal intensity on T1-weighted images, high signal intensity on T2-weighted images, and increased signal intensity on short tau inversion recovery (STIR). This edema extended from the femoral head and neck to the intertrochanteric area (Figs. 2, 3).
We prescribed rest and non-weight bearing for him; however, he did not follow our instructions due to his psychological condition. His pain gradually improved and he was discharged from our hospital using crutches.
Three weeks later, his left hip pain worsened without trauma and he visited our hospital again. X-ray, CT scan, and MRI of the hip joint demonstrated a left femoral neck fracture (Garden stage Ⅱ) (Figs. 46), and osteosynthesis (Prima Hip Screw System, Japan Medical Dynamic Marketing, Inc., Tokyo, Japan) was performed (Fig. 7).

Discussion

TOH generally affects middle-aged men and pregnant women in their last trimester [2-4]. It is usually bilateral in pregnant women and unilateral in middle-aged men [8]. The cause and pathogenesis of TOH remain unclear; however, the most dominant theory implicates femoral head ischemia due to venous obstruction as the cause [5-7]. On examination, bone biopsies of patients with TOH showed increased numbers of erythrocytes, suggesting the presence of venous stasis [5]. Furthermore, Orth and Anagnostakos [9] reported that decreased concentrations of fibrinolytic agents and increased concentrations of thrombophilia markers in TOH patients could potentially cause venous obstruction. Risk factors for TOH include trauma, a history of steroid use, consumption of alcohol, smoking, low testosterone levels, low vitamin D levels, osteogenesis imperfecta, hypothyroidism, and hypophosphatasia [10-13]. In this case, smoking and alcohol were mentioned as risk factors.
MRI is the most powerful tool for the diagnosis of TOH and usually shows low signal intensity on T1-weighted images, high signal intensity on T2-weighted images, and increased signal intensity on STIR, reflecting bone marrow edema.
TOH typically has a good prognosis with spontaneous remission occurring within a few months to 1 year. Methods for joint preservation, including restricted weight bearing, are the first-line therapy. As alternative treatments, the use of bisphosphonates to inhibit bone resorption and the use of teriparatide as an osteoanabolic agent were reported to provide successful outcomes [14-17]. Additionally, hyperbaric oxygen therapy was reported to potentially accelerate recovery in TOH patients, although the effects were not statistically significant [18]. More recently, out of 15 TOH cases, 10 underwent hip drilling for core decompression while five underwent conservative therapy. An investigation of the time to full recovery revealed that hip drilling required a median of 5.8 weeks while conservative therapy required 48.3 weeks, demonstrating the validity of hip drilling as a treatment modality [19].
In the present case, regarding our recommendations to avoid bearing weight, the patient was nonadherent due to his poor mental health; therefore, it was assumed that the constant stress of weight bearing caused the femoral neck fracture. In fact, the fracture pattern in this case originated in the lower part of the femoral neck and is considered to be a pathological condition similar to insufficient fracture secondary to TOH. Interestingly, Hadji et al. [20] demonstrated that out of 52 TOH patients, 12.1% sustained a hip fracture. Therefore, for patients with a high likelihood of fracture, it is necessary to provide thorough education, such as instructions not to apply a load to the lower limbs during a specified period. Prophylactic osteosynthesis may also be considered for patients expected to have a high likelihood of fracture. Considering these factors, a sufficient follow-up period from condition onset to full recovery is necessary to avoid as many complications as possible.

Notes

Conflicts of interest

No potential conflict of interest relevant to this article was reported.

Funding

None.

Author contributions

Conceptualization, Supervision: YT, TM; Data curation: YT, SM, MM; Formal analysis: SM, MM, KO, YT, TM; Investigation: YT; Writing-original draft: YT; Writing-review & editing: all authors.

Fig. 1.
X-ray and computed tomography (CT) reveal no obvious fractures. (A) X-ray anterior view. (B) CT coronal view. (C) CT axial view.
jyms-2022-00479f1.jpg
Fig. 2.
The magnetic resonance imaging coronal views of the left transient osteoporosis of the hip. (A) Short tau inversion recovery image. (B) T2-weighted image. (C) T1-weighted image.
jyms-2022-00479f2.jpg
Fig. 3.
Magnetic resonance imaging (MRI) axial view of the left transient osteoporosis of the hip. MRI shows diffuse edema in the bone marrow, extending from the femoral head and neck to the intertrochanteric area. (A) Short tau inversion recovery image. (B) T2-weighted image. (C) T1-weighted image.
jyms-2022-00479f3.jpg
Fig. 4.
The left femoral neck fracture (arrows). It is difficult to find the fracture line of the femoral neck, by either X-ray or computed tomography (CT). (A) X-ray anterior view. (B) X-ray lateral view. (C) CT axial view. (D) CT coronal view.
jyms-2022-00479f4.jpg
Fig. 5.
Magnetic resonance imaging coronal view of the left femoral neck fracture (arrows). (A) Short tau inversion recovery image. (B) T1-weighted image.
jyms-2022-00479f5.jpg
Fig. 6.
Magnetic resonance imaging (MRI) axial view of the left femoral neck fracture (arrows). MRI shows a clear fracture line in the left femoral neck. (A) Short tau inversion recovery image. (B) T1-weighted image.
jyms-2022-00479f6.jpg
Fig. 7.
Osteosynthesis with three Prima Hip screws (Japan Medical Dynamic Marketing, Inc., Tokyo, Japan). (A) X-ray anterior view. (B) X-ray lateral view.
jyms-2022-00479f7.jpg

References

1. Curtiss PH Jr, Kincaid WE. Transitory demineralization of the hip in pregnancy: a report of three cases. J Bone Joint Surg Am 1959;41-A:1327–33.
crossref pmid
2. Van Wagenen K, Pritchard P, Taylor JA. Transient osteoporosis of the hip: a case report. J Can Chiropr Assoc 2013;57:116–22.
pmid pmc
3. Ma FY, Falkenberg M. Case reports: transient osteoporosis of the hip: an atypical case. Clin Orthop Relat Res 2006;445:245–9.
crossref pmid
4. Asadipooya K, Graves L, Greene LW. Transient osteoporosis of the hip: review of the literature. Osteoporos Int 2017;28:1805–16.
crossref pmid pdf
5. Rosen RA. Transitory demineralization of the femoral head. Radiology 1970;94:509–12.
crossref pmid
6. Plenk H Jr, Hofmann S, Eschberger J, Gstettner M, Kramer J, Schneider W, et al. Histomorphology and bone morphometry of the bone marrow edema syndrome of the hip. Clin Orthop Relat Res 1997;(334):73–84.
crossref
7. Koo KH, Jeong ST, Jones JP Jr. Borderline necrosis of the femoral head. Clin Orthop Relat Res 1999;(358):158–65.
crossref
8. Manara M, Varenna M. A clinical overview of bone marrow edema. Reumatismo 2014;66:184–96.
crossref pmid pdf
9. Orth P, Anagnostakos K. Coagulation abnormalities in osteonecrosis and bone marrow edema syndrome. Orthopedics 2013;36:290–300.
crossref pmid
10. Diwanji SR, Cho YJ, Xin ZF, Yoon TR. Conservative treatment for transient osteoporosis of the hip in middle-aged women. Singapore Med J 2008;49:e17–21.
pmid
11. Daniel RS, Farrar EK, Norton HR, Nussbaum AI. Bilateral transient osteoporosis of the talus in pregnancy. Osteoporos Int 2009;20:1973–5.
crossref pmid pdf
12. Jennings PE, O’Malley BP, Griffin KE, Northover B, Rosenthal FD. Relevance of increased serum thyroxine concentrations associated with normal serum triiodothyronine values in hypothyroid patients receiving thyroxine: a case for “tissue thyrotoxicosis”. Br Med J (Clin Res Ed) 1984;289:1645–7.
crossref pmid pmc
13. Dunstan CR, Evans RA, Somers NM. Bone death in transient regional osteoporosis. Bone 1992;13:161–5.
crossref pmid
14. Flores-Robles BJ, Sanz-Sanz J, Sanabria-Sanchinel AA, Huntley-Pascual D, Andréu Sánchez JL, Campos Esteban J, et al. Zoledronic acid treatment in primary bone marrow edema syndrome. J Pain Palliat Care Pharmacother 2017;31:52–6.
crossref pmid
15. Emad Y, Ragab Y, El-Shaarawy N, Rasker JJ. Transient osteoporosis of the hip, complete resolution after treatment with alendronate as observed by MRI description of eight cases and review of the literature. Clin Rheumatol 2012;31:1641–7.
crossref pmid pdf
16. Kibbi L, Touma Z, Khoury N, Arayssi T. Oral bisphosphonates in treatment of transient osteoporosis. Clin Rheumatol 2008;27:529–32.
crossref pmid pdf
17. Schapira D, Braun Moscovici Y, Gutierrez G, Nahir AM. Severe transient osteoporosis of the hip during pregnancy: successful treatment with intravenous biphosphonates. Clin Exp Rheumatol 2003;21:107–10.
pmid
18. Yagishita K, Jinno T, Koga D, Kato T, Enomoto M, Kato T, et al. Transient osteoporosis of the hip treated with hyperbaric oxygen therapy: a case series. Undersea Hyperb Med 2016;43:847–54.
pmid
19. Bashaireh KM, Aldarwish FM, Al-Omari AA, Albashaireh MA, Hajjat M, Al-Ebbini MA, et al. Transient osteoporosis of the hip: risk and therapy. Open Access Rheumatol 2020;12:1–8.
crossref pmid pmc
20. Hadji P, Boekhoff J, Hahn M, Hellmeyer L, Hars O, Kyvernitakis I. Pregnancy-associated transient osteoporosis of the hip: results of a case-control study. Arch Osteoporos 2017;12:11.
crossref pmid pdf
TOOLS
Share :
Facebook Twitter Linked In Google+ Line it
METRICS Graph View
  • 0 Crossref
  •    
  • 464 View
  • 17 Download
Related articles in
J Yeungnam Med Sci


ABOUT
ARTICLE CATEGORY

Browse all articles >

BROWSE ARTICLES
FOR AUTHORS AND REVIEWERS
Editorial Office
170 Hyeonchung-ro, Nam-gu, Daegu 42415, Korea
Tel: +82-53-640-6832    Fax: +82-53-651-0394    E-mail: jyms@yu.ac.kr                

Copyright © 2022 by Yeungnam University College of Medicine, Yeungnam University Institute Medical Science.

Developed in M2PI

Close layer
prev next