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C OPYRIGHT Ó 2021 BY T HE J OURNAL OF B ONE AND J OINT S URGERY, I NCORPORATED

Tension-Sided Femoral Neck Stress Fracture


in an Adolescent with Vitamin D Deficiency
and Osteomalacia
Downloaded from http://journals.lww.com/jbjscc by k38zdtHxkv4GqUtrpBilxmqdJ33q2Na+36qVoYuWTEZ6YFqRPbzQU55uRYh1x4ne+3ogVPI8KCwzGHNQ2jW7jxocTJFXjSs8sfhveTzYyYmf/BgP5lRkaUy2C3ks40l03ZJbomxmNEQ= on 01/05/2022

A Case Report
Neeraj Vij, BS, Ashish S. Ranade, MBBS, MS (Ortho), Supriya Gupte, MBBS, MD, Gauri A. Oka, MBBS, MPH, and
Mohan V. Belthur, MD, FRCSC, FRCS (Tr & Orth)

Investigation performed at Deenanath Mangeshkar Hospital and Research Center, Pune, Maharashtra, India

Abstract
Case: An adolescent girl presented with groin pain without any history of trauma. Imaging showed a tension-sided stress
fracture of the femoral neck. Vitamin D deficiency (VDD), and raised alkaline phosphatase and parathyroid hormone levels
were found. Pain relief was not achieved with nonoperative treatment. Considering the risk of fracture progression and
displacement, the fracture was fixed with cannulated cancellous screws. Fracture healed without any complications.
Conclusion: A tension-sided femoral neck stress fracture, with VDD and osteomalacia, is a rare cause of hip pain in an
adolescent. Surgical fracture stabilization is necessary in addition to medical treatment.

F
emoral neck stress fractures (FNSFs) are rare in children1. had persisted despite activity modification and rest. The patient
The FNSF could be of compression side or tension side, had no known medical conditions. She attained menarche at 12
with the latter being uncommon2,3. FNSFs are generally years with regular cycles. The patient’s height was 141 cm, and
believed to be predisposed by athletic activity or metabolic weight was 46 kg (body mass index of 23.1 kg/m2). Physical
bone diseases4-6. Vitamin D deficiency (VDD) may affect the examination revealed a normal-appearing right lower
bone architecture that may predispose to a stress fracture7. extremity. The right hip terminal flexion was mildly painful.
Fracture associated with metabolic bone diseases is either a Internal and external rotations were pain-free, and the range of
medial compression-type FNSF or a complete neck femur motion was symmetrical in both the hips. There was no
fracture5,6. To the best of our knowledge, there is no report of a obligatory external rotation.
tension-sided FNSF associated with VDD and osteomalacia in The radiographs showed a transverse fracture involving
an adolescent. the superior side of the femoral neck (tension-type fracture)
In contrast to compression-type fractures, tension-sided (Fig. 1 and Fig. 2). The fracture was incomplete and non-
FNSFs carry the risk of progression to complete fracture and displaced. The MRI demonstrated a partial fracture involving
displacement at the fracture site. We report a case of an adolescent the superior part of the femoral neck, minimal effusion in the
girl with a tension-sided FNSF with osteomalacia and VDD. hip joint, and edema in the femoral neck (Figs. 3 and 4). There
The patient and her parents were informed that data were no radiological signs of osteomalacia. Radioisotope bone
concerning the case would be submitted for publication, and scan was not performed. Blood tests revealed vitamin D levels
they provided consent. (25(OH) vitamin D) 6 ng/dL (reference range, 20-50 ng/dL),
serum ionic calcium 4.64 mg/dL (reference range, 4.4-5.5 mg/dL),
Case Report serum calcium 8.4 mg/dL (reference range, 8.4-10.2 mg/dL),

A 15-year-old girl presented with complaints of right hip


pain. There was no history of trauma or sports activity.
The pain was present for the past 8 months. The pain was rated
serum phosphorous 2.75 mg/dL (reference range, 2.3-4.7), and
serum alkaline phosphatase 321 IU/L (reference range, 47-149
IU/L). Bone-specific alkaline phosphatase was not performed.
as mild and increased with activities such as climbing stairs and Liver and renal function tests, and serum electrolytes were
Disclosure: The Disclosure of Potential Conflicts of Interest forms are provided with the online version of the article (http://links.lww.com/JBJSCC/B540).
Keywords: stress fracture, femoral neck, adolescent, hip pain, osteomalacia, hypovitaminosis D, pediatric, vitamin D deficiency, tension-sided femoral
neck stress fracture

JBJS Case Connect 2021;11:e20.00787 d http://dx.doi.org/10.2106/JBJS.CC.20.00787


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Fig. 1 Fig. 2
Fig. 1 Anteroposterior radiographic view of the pelvis demonstrating a tension-sided (lateral) fracture of the right femoral neck. Fig. 2 Frog pelvic view
showing a stress fracture along the anterior aspect neck of the femur.

within the normal range. The parathyroid hormone (PTH) Nonoperative treatment with non–weight-bearing
level was 276.5 pg/mL (reference range, 10-76 pg/mL) sug- ambulation and vitamin D 60,000 IU once every week for
gestive of compensatory secondary hyperparathyroidism. Total 10 weeks along with calcium 500 mg daily for 12 weeks was
leukocyte count was 8,700/mm3 (reference range, 4,000- started. The patient continued to have persistent pain and
10,000/mm3), and CRP was 0.5 mg/L (reference range, 0-3 mg/L). difficulty in maintaining non–weight-bearing ambulation.
We did not do bone mineral density studies or bone biopsy. Considering inadequate pain relief with nonoperative mea-
The patient was also evaluated by a pediatric endocrinologist. sures and the risk of progression of the tension-sided FNSF to
The patient was diagnosed to have a tension-sided FNSF with complete fracture and displacement, a decision was made to
VDD and osteomalacia. The diagnosis of osteomalacia was stabilize the fracture surgically. The fracture was stabilized with
made based on the criteria by Uday and Högler [high alkaline two 6.5-mm partially threaded, cannulated cancellous screws
phosphatase, high PTH, low dietary calcium intake, and/or to achieve compression across the fracture site (Fig. 5). Post-
low serum 25OH (<30 nmol/L)]8. The differential diagnoses operatively, the patient was allowed to ambulate toe-touch
considered in a case of chronic right hip pain for 8 months in weight-bearing with crutches for 6 weeks and then transitioned
an adolescent were infectious causes (sclerosing osteomyeli- to full weight-bearing over the next 6 weeks.
tis), inflammatory arthropathy, slipped capital femoral The patient recovered well with no complications and
epiphysis, delayed traumatic presentation, and osteoid oste- became pain-free at 2 weeks after the surgery. After completion
oma. In the given case, the imaging confirmed the diagnosis of treatment for VDD, a repeat biochemical profile at
of a tension-sided FNSF. 5 months showed normalization of vitamin D levels and

Fig. 3 Fig. 4
Fig. 3 Axial and coronal MRI T2 images at presentation demonstrating a tension-sided femoral neck stress fracture. The axial view shows edema and
capsular effusion. Fig. 4
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30-month follow-up, the patient remains pain-free. We con-


tinue to keep close follow-up.

Discussion

W e present an unusual case of a tension-sided FNSF in an


adolescent girl with VDD and osteomalacia. Pediatric
FNSFs are rare, and a majority are associated with overuse in
sports1,3,4. Repetitive cyclical loading of the hip joint leads to a
stress fracture. Other causes of FNSFs include osteomalacia,
VDD, and rickets5,6. The FNSFs are classified into compression-
type (fracture along the inferomedial side of the femoral neck)
and tension-type (a vertical fracture along the superior side of
the femoral neck)2.
Children with FNSFs present with hip or referred knee
pain with or without a limp. There might not be a history of
increased sports activity before the onset of symptoms10. Clinical
examination shows a slight limitation of hip movements. A
radiograph may show linear sclerosis or callus formation in the
femoral neck; however, it may be absent during the initial few
weeks. A stress fracture may progress with continued activity to a
complete fracture and also may become displaced.
Diagnosing FNSFs in a child is challenging, given the
minimal findings on clinical examination and subtle changes in the
radiographs. MRI is the key investigation to achieve early diagnosis.
Although most of the FNSFs tend to heal with nonoperative
Fig. 5 treatment, fractures involving the tension side are at increased risk
Postoperative anterior-posterior radiographic view of the pelvis after per- of fracture completion or displacement. Treatment should aim to
cutaneous placement of two 6.5-mm screws. avoid this complication. The presence of a tension-sided fracture
or fracture with hip joint effusion is the risk factor for developing a
alkaline phosphatase levels and resolution of secondary complete fracture, and fracture stabilization is recommended.
hyperparathyroidism. Thereafter, calcium 250 mg daily and Tension-sided stress fracture or fracture with involvement of >50%
vitamin D 400 IU were prescribed based on the recommen- of the femoral neck is at high risk of displacement and is recom-
dation of Khadilkar et al9. The PTH and other biochemical mended to undergo surgical stabilization11,12.
parameters remained normal at 30-month follow-up. Radi- Laboratory investigations to rule out metabolic causes
ographs obtained at the 30-month follow-up showed healed should include vitamin D (25OH) levels, serum calcium,
fracture without any signs of osteonecrosis (Figs. 6 and 7). At serum alkaline phosphatase, and serum PTH levels, in addition

Fig. 6 Fig. 7
Fig. 6 Anteroposterior and frog pelvic radiographs obtained at 30-month follow-up showing the united femoral neck stress fracture. Fig. 7
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to other investigations. Osteomalacia is characterized by low neck femur fracture may lead to several complications such as
vitamin D level, raised alkaline phosphatase, normal or low osteonecrosis, nonunion, or coxa vara. To avoid these com-
serum calcium level, and raised serum PTH level in an indi- plications, timely diagnosis and treatment are important.
vidual after skeletal maturity. In a child with open physis, the A tension-sided FNSF with VDD and osteomalacia could
same laboratory picture is seen in rickets. Various diagnostic be a rare cause of hip pain in an adolescent. The metabolic causes
criteria have been described including several biochemical should be ruled out while treating an FNSF in an adolescent. n
markers and bone biopsy findings8.
A high prevalence of VDD (>80%) has been reported in
Indian school-going children (age group, 6-17 years)13. This
deficiency was believed to be a result of multiple factors such as Neeraj Vij, BS1
dietary deficiency, lack of sun exposure, and genetic factors13,14. Ashish S. Ranade, MBBS, MS (Ortho)2
There are no guidelines regarding the type of questions a clinician Supriya Gupte, MBBS, MD3
should ask to help screen for VDD in an otherwise healthy- Gauri A. Oka, MBBS, MPH4
appearing adolescent. Also, there is insufficient evidence for Mohan V. Belthur, MD, FRCSC, FRCS (Tr & Orth)5
pediatric healthcare providers to recommend which VDD risk 1University of Arizona College of Medicine, Phoenix, Arizona
factors should be used for screening in children and adolescents15.
An association between osteomalacia and FNSFs in ado- 2Blooming Buds Center for Pediatric Orthopaedics, Deenanath
lescents has been reported5,6. A study has reported a compression- Mangeshkar Hospital and Research Center, Erandwane, Pune,
type FNSF in a 14-year-old girl with VDD. The patient was treated Maharashtra, India
nonoperatively, and 18 months later, the fracture healed5. In
3Department of Pediatric Endocrinology, Deenanath Mangeshkar
another report, a complete and displaced femoral neck fracture in
association with low levels of vitamin D has been described6. Hospital and Research Center, Erandwane, Pune, Maharashtra, India
Notably, neither of these reports demonstrated any radiological 4Research Consultant, Deenanath Mangeshkar Hospital and Research
features of osteomalacia on pelvic radiographs or MRI. This Center, Erandwane, Pune, India
probably suggests that the pathological process within the bones
that predisposes to pediatric FNSFs may precede the development 5Department of Pediatric Orthopaedics, Phoenix Children’s Hospital,
of the radiologic features. The only reported case of a tension- Phoenix, Arizona
sided FNSF is of a 14-year-old boy. There was no metabolic
abnormality in this patient, and the fracture was believed to have E-mail address for A.S. Ranade: ranadea2@gmail.com
developed after participating in a football camp. This fracture was ORCID iD for N. Vij: 0000-0002-7214-0411
treated surgically using cannulated compression screws3. ORCID iD for A.S. Ranade: 0000-0003-0925-6427
The tension-sided FNSF tends to be unstable, given their ORCID iD for S. Gupte: 0000-0003-0594-1558
anatomic position and forces leading to a higher propensity to ORCID iD for G.A. Oka: 0000-0003-3425-8512
develop a complete fracture with displacement3. A displaced ORCID iD for M.V. Belthur: 0000-0001-5279-0591

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