Atraumatic Fractures Research Articles

Four levels pedicle fractures in a renal failure patient: A case report and literature review

Lumbar pedicles are wider than thoracic pedicles and L5 is considered the widest, which requires high-energy trauma to fracture. It is rare to have atraumatic lumbar pedicle fractures. However, it is reported mainly in predisposed patients, like those with contralateral spondylolysis, degenerative spondylolisthesis, previous spine surgery, stressful activities, and metabolic bone disease. We are reporting a rare case of a 37-year-old renal failure male patient who is an office worker who started dialysis only 1 month before the presentation. After a fall from the bed, the patient was found to have four levels pedicle fractures. We aimed to increase the awareness that lumbar pedicle fractures can coexist in patients with renal failure and increase the suspicion index that can promote early diagnosis and effective management.

Does Zoledronic Acid Provide a Good Clinical Outcome in Patients With Chronic Back Pain Associated With Vertebral Osteoporosis?

Background Osteoporosis is a chronic, progressive, systemic condition of the skeletal tissue that is characterized by reduced bone density, microarchitecture deterioration, and fragile bones, making osteoporotic fractures or fragility fractures more likely to occur. This condition often remains asymptomatic and undiagnosed until it presents with fragility fractures. The condition is associated with a significant socioeconomic burden with disability, morbidity, and mortality. Therefore, early diagnosis, as well as treatment, is needed to prevent fractures. Intravenous zoledronic is an effective bisphosphonate with high patient compliance due to once-yearly dosing. The present study aims to determine whether zoledronic acid effectively treats chronic back pain in people with osteoporosis. Materials and methods Seventy patients above the age of 60 years presented with complaints of chronic low back aches to the outpatient department of orthopedics, R L Jalappa Hospital & Research Centre attached to Sri Devaraj Urs Medical College. The study was conducted between November 2016 and November 2018. Results All the patients found excellent clinical improvement following zoledronic acid infusion in early and long-term follow-ups. Additionally, it was found that zoledronic acid's effectiveness was excellent, with significant improvement in bone mineral density (BMD), T-score, and Z-score. Conclusion Early diagnosis and treatment of vertebral osteoporosis is the most important factor in preventing fragility fractures. Zoledronic acid, an antiresorptive drug with better compliance, is very effective in controlling low back pain, improving bone mineral density, and preventing the occurrence of atraumatic fragility fractures. With all the above factors, zoledronic acid is a preferable bisphosphonate for the treatment and prevention of osteoporosis compared to other modalities of treatment of osteoporosis.

Atraumatic acetabular fracture after generalized seizure during hemodialysis: A case report

Atraumatic acetabular fracture after generalized seizure during hemodialysis: A case report

Treating 'osteoporosis': a near miss in an unusual case of FGF-23-mediated hypophosphataemic osteomalacia.

We present the case of a 60-year-old female who developed repeated atraumatic stress fractures. She was initially diagnosed with osteoporosis based on her dual-energy X-ray absorptiometry (DXA) scan bone mineral density (BMD) T-scores and started on denosumab therapy. Secondary osteoporosis screen revealed abnormal myeloma screen and low serum phosphate levels. It was thought that the patient had multiple myeloma with associated Fanconi-related tubular dysfunction. However, fibroblast growth factor-23 (FGF-23) levels were grossly elevated, making Fanconi syndrome unlikely. The patient was subsequently diagnosed with two separate conditions, namely cardiac amyloid light-chain (AL) amyloidosis and FGF-23-related hypophosphataemia, likely due to tumour-induced osteomalacia. This case highlights the importance of excluding osteomalacia as a cause of low BMD and checking FGF-23 levels in the workup for hypophosphataemia. Tumour-induced osteomalacia is a difficult diagnosis as the tumour is often small and slow growing. Imaging may fail to identify a tumour, and treatment therefore consists of calcitriol and phosphate replacement. Tumour-induced osteomalacia should be suspected in the adult presenting with new-onset hypophosphataemia, elevated FGF-23 levels and isolated renal phosphate wasting. Serum phosphate is not part of the routine chemistry panels. Routinely checking phosphate levels prior to initiating antiresorptive therapy is warranted. DXA cannot distinguish low bone mineral density due to osteoporosis from osteomalacia. Antiresorptive therapy should be avoided in osteomalacia due to the risk of clinical and radiographic deterioration.

PSAT222 Muscle Weakness, Bone Pain, and Stress Fractures: When a Fracture is NOT JUST a Fracture: A Case Report

Abstract Case A previously healthy 46-year-old male presented to orthopedic clinic with bilateral hip pain, and progressive weakness without history of trauma requiring cane for ambulation. X-ray and MRI evaluation revealed stress fractures of left subtrochanteric femur and right basicervical femoral neck, both requiring surgery. In hospital blood work revealed elevated total alkaline phosphatase 167 units/L, mild hypocalcemia 8.5 mg/dL, and low 25-hydroxy Vitamin D 18 ng/mL. The patient was started on calcium/vitamin D supplements upon discharge. Initial endocrinology outpatient work-up revealed low phosphorus level of 2.1 mg/dl and low 1, 25 OH vitamin D of 5.9 pg/ml (19.9–79.3). Bone mineral density revealed normal spine bone density, and 1/3 radius osteopenia (T score -1.1). Physical exam revealed profound proximal weakness with inability to rise independently from sitting position. Follow-up of low phosphorous level demonstrated an elevated FGF-23 level of 186 RU/mL (normal < 180) and elevated fractional excretion of phosphorus 16% (expect < 5% with hypophosphatemia), indicating inappropriate renal phosphate wasting. The patient was started on Calcitriol and phosphorus. Before above evaluation was completed the patient had an ED visit with complaint of small mass just below left knee associated with mild pain and warmth. Ultrasound and MRI showed a complex cyst which was aspirated and showed cystic appearing mass suggestive of hematoma. This was treated with antibiotic course. Due to suspicion of tumor-induced osteomalacia, octreotide scan (Indium-111 pentetreotide) performed showed focal increased activity in the medial left knee. MRI showed a 26 mm cystic lesion within the subcutaneous tissue along the inferomedial aspect of the left knee, which correlated with the site of radiotracer uptake on the octreotide scan. The patient underwent left knee soft tissue tumor resection, and pathology demonstrated phosphaturic mesenchymal tumor. Follow-up several weeks postoperatively showed normalization of phosphorus and FGF-23 level. The patient's mobility and weakness improved significantly within a few months. Discussion Tumor-induced osteomalacia is a rare paraneoplastic condition characterized by chronic hypophosphatemia secondary to excessive tumoral FGF 23 production. Typical presenting symptoms are profound muscle weakness, bone pain, and fractures. Diagnosis is often delayed for several years, as phosphorus levels are not included in a routine metabolic panel. Considering high morbidity of this condition including significant mobility limitation requiring use of assistive devices, healthcare provider vigilance is paramount as this condition is potentially curable if the offending tumor is detectable and amenable to surgical resection. This case illustrates the importance of consideration of an underlying metabolic bone disease in patients with unexplained atraumatic fractures and hypophosphatemia. Presentation: Saturday, June 11, 2022 1:00 p.m. - 3:00 p.m.

ODP125 Soft Bones in Crohn's: Tumor-induced osteomalacia and inflammatory bowel disease

Abstract Introduction Bone loss is common in patients with inflammatory bowel disease (IBD) and is usually attributed to Vitamin D malabsorption, treatment with glucocorticoids or chronic inflammation. We report a case of bone loss, hypophosphatemia, and atypical fractures in a patient with IBD who was ultimately diagnosed with tumor-induced osteomalacia (TIO), a rare paraneoplastic syndrome of chronic hypophosphatemia and impaired vitamin D metabolism. Clinical Case A 39-year-old male presented for evaluation of low bone density, hypophosphatemia, and multiple stress fractures. His medical history includes Crohn's disease in biochemical remission on adalimumab and bilateral hip avascular necrosis. There was no history of significant steroid use or known family history of metabolic bone disease. He was previously highly active as a modern dancer. Over the course of 3 years, he developed progressive diffuse bony pain and he became wheelchair-bound. A whole body bone scan visualized multiple age-indeterminate atraumatic fractures, including bilateral metatarsal fractures and numerous rib fractures. Dual energy x-ray absorptiometry showed significantly low bone mass for age (Z-scores –3.4, -3.1, -3.7 at the L1-L4 spine, total right femur and right femoral neck, respectively). Physical exam was notable for a soft, subcutaneous 8 cm mass posterior to the left paraspinal muscle at the level of T12, which had previously been diagnosed as a lipoma. Laboratory evaluation revealed phosphorus 1 mg/dL (2.5-4.5 mg/dL), calcium 9.1 mg/dL (8.6-10.2 mg/dL), PTH 35 pg/mL (15-65 pg/mL), c-telopeptide 592 pg/mL (38-724 pg/mL), bone specific alkaline phosphatase 114.5 mcg/L (4-27 mcg/L), vitamin D-1,25 8.9 pg/mL (19.9-79.3 pg/mL), and vitamin D-25 34 ng/mL (normal >30 ng/mL). Fibroblast growth factor-23 (FGF-23) level was 1200 RU/mL (44-215 RU/mL), consistent with TIO. Thyroid function, dexamethasone suppression test, testosterone and IGF-1 were normal. Ga-DOTATATE PET scan demonstrated that the paraspinal soft tissue mass was strongly somatostatin avid. The patient was treated with oral calcitriol and phosphate replacement. He underwent a biopsy of the left paraspinal mass, which was consistent with a bland epithelioid and spindle cell neoplasm, most suggestive of a phosphaturic mesenchymal tumor. He is scheduled for a radical resection of the soft tissue mass. Conclusion To date, there are no published case reports of TIO with coexisting IBD. Inflammatory cytokines, such as tumor necrosis factor alpha, have been shown to positively correlate with FGF-23 levels, and FGF-23 has been documented to be elevated during IBD flares. Given this, the presence of IBD may portend a worsened metabolic bone prognosis in patients who simultaneously develop FGF-23 secreting tumors. Presentation: No date and time listed

Revision of a Failed Primary Total Hip Arthroplasty following Excessive Reaming with a Medial Cup Protrusion.

Background and Objectives: Atraumatic intrapelvic protrusion of the acetabular component following excessive reaming of the acetabulum with a far medial positioning of the cup is a rare, but serious complication of a total hip arthroplasty (THA). This study analyzes the factors contributing to this uncommon complication and presents the outcome after the revision surgery using the Ganz reinforcement ring combined with a bone graft and plating of the posterior column and/or screws for the anterior column. Materials and Methods: A retrospective case series study with seven patients (four males, mean age 76 ± 10 years (60–86)) that underwent a revision THA within 24 ± 17 days (5–60) after an atraumatic periprosthetic acetabular fracture with a medial cup protrusion was performed. All fractures were reconstructed with a Ganz reinforcement ring and bone graft with a mean follow-up of 1.7 ± 1.7 years (0.5–5). Radiographs were evaluated for the following: (i) cup positioning immediately after the primary THA and the revision surgery, (ii) cup migration in the follow-up, and (iii) fracture healing. Results: The position of the acetabular component as assessed on the postoperative radiographs after the index surgery and before the complete medial cup protrusion showed a cup placement beyond the ilioischial line indicative of a fracture of the medial wall. The revision surgery with the reconstruction of the medial wall with a Ganz reinforcement ring combined with a bone graft restored in the presented cases the center of rotation in the horizontal direction with a statistical significance (p < 0.05). During the follow-up, there was no aseptic loosening with the relevant cup migration or significant change in the position of the acetabular cup at the final follow-up (p > 0.05) after the revision. All seven fractures and bone grafts realized a bone union until the latest follow-up. Conclusions: Following excessive reaming, the acetabular component was placed too far medially and resulted in an intrapelvic cup protrusion. An unstable cup following a fracture of the medial wall was evident on the immediate postoperative radiographs. In the case of the medial wall perforation with an intrapelvic cup protrusion after the primary THA, the reconstruction with a Ganz reinforcement ring was a successful treatment option resulting in the fracture healing and a stable cup positioning. Surgeons should be aware of that rare and probably underreported complication and restore the anatomic center of rotation by treating the defect intraoperatively.

Sagittal Imbalance May Lead to Higher Risks of Vertebral Compression Fractures and Disc Degeneration—A Finite Element Analysis

Sagittal balance is an important clinical parameter of the spine for its normal function. Maintenance of the sagittal balance is crucial in the clinical management of spinal problems. Three different finite element models with spinal alignments based on Schwab's classification were developed: (1) Balanced/Normal model (sagittal vertical axis [SVA]= 0 mm, lumbar lordosis [LL]= 50°, thoracic kyphosis [TK]= 25°, pelvic incidence [PI]= 45°, pelvic tilt [PT]= 10°, sacral slope [SS]= 35°); (2) Balanced with compensatory mechanisms/Flatback model (SVA= 50 mm, LL= 20°, TK= 20°, PI= 45°, PT= 30°, SS= 15°); and (3) Imbalanced/Hyperkyphotic model (SVA= 150 mm, LL= -5°, TK= 25°, PI= 45°, PT= 40°, SS= 5°). All 3 models were subjected to the follower loads simulating bodyweight/muscular contractions along with the moments to simulate flexion, extension, lateral bending, and axial rotation. The maximum cortical vertebral stress, annular stress, and intradiscal pressure (IDP) were calculated and compared. The results showed that the hyperkyphotic model had higher stresses in the vertebrae (25% higher), the annulus fibrosus (48% higher) and the IDP (8% higher) than the normal models in flexion. The segments near the thoracolumbar junction (T10-L1) showed the highest increase in the vertebral body stress, the annulus fibrosus stress, and the IDP. This study showed that the imbalance in sagittal alignment might be responsible for disc degeneration and atraumatic vertebral fractures at the thoracolumbar regions, supporting clinical findings.

Pedicle fracture after successfully posterior spinal fusion for adult idiopathic scoliosis

Study Design: A Case report.Objective: To report a case of atraumatic pedicle fracture for idiopathic scoliosis after posterior spinal fusion.Summary of Background Data: Pedicle fracture after posterior spinal fusion for idiopathic scoliosis is rare, with only few cases reported in the literature.Conclusions: Pedicle fracture in idiopathic scoliosis after posterior spinal fusion is a rare long-term complication. In this case report, we report successful treatment of a fracture.

Failure of VariAx® Distal Radial Locking Plate: Fractographic Analysis and Report of Two Cases

We describe two cases of atraumatic fracture of VariAx® distal radial locking plate following its use in distal radial osteotomies. The plates fractured through an unused screw hole at the level of the osteotomy. Scanning electron microscopy was used to evaluate the fracture surfaces. This confirmed that there had been fatigue failure and did not find evidence of abnormal composition or structure of the metal. These cases are important because they highlight that this new and innovative implant can be prone to failure, and demonstrate the requirement for adequate surgical technique and metalwork protection following surgery.

Uncommon presentation of bilateral subtrochanteric insufficiency fractures in young male associated with hypogonadism: A case report

Hypogonadism in men is a well-recognized cause of secondary osteoporosis. It is characterized by insufficient production of androgen, testosterone, and sperms. Testosterone deficiency is the key factor for insufficiency fractures in men, resulting from normal loading on an osteoporotic bone. In this report, we are presenting a case of a 27-year-old male known to have hypogonadotropic hypogonadism, who suffered from both femoral atraumatic subtrochanteric fractures that had been existing for a considerable time. The fractures interfered with the patient’s walking and daily activity. It is worth reporting this case because most of the proximal femur insufficiency fractures mentioned in the literature correlated with prolonged alendronate therapy or were post-bariatric surgery.

D1 MTE1 Case 2: Young male with chronic hepatitis and bony deformity

19 years old gentleman presented with deformity of lower limbs noticed two years back. Significant past h/o jaundice at 8 years of age, h/o fracture of right femur at 17 years of age following fall from bicycle and an atraumatic fracture at the same site one year later. He underwent cataract surgery one year back where he was found to have bilateral renal stone disease with elevated creatinine. He had short stature with scoliosis and apparent genu valgum on right. Biochemical evaluation showed CrCl of 40 ml/min/m2 with increased liver enzymes. He had hypophosphatemia with low-normal s.calcium, elevated ALP, elevated PTH (242 pg/ml) and normal 25(OH)D3. Urine evaluation showed impaired acidification along with glycosuria and phosphaturia in the presence of systemic acidosis and normoglycemia. Skeletal survey showed generalized rarefaction; DXA showed osteopenia. MBD secondary to RTA was considered. Ultrasound showed coarsened liver echotexture. Suspecting Wilson's disease, s.ceruloplamin and 24hrs urinary copper was done but was normal and KF ring was absent. Therefore, liver biopsy was done suggesting chronic hepatitis with dry weight copper of 127.23 mcg/gm (normal < 45 mcg/g) which was <4ULN needed for diagnosis. Can the diagnosis of Wilson's disease be made from the above findings or else, any alternative diagnosis to be considered?

Reverse total shoulder arthroplasty in wheelchair-dependent patients: a matched cohort study

BackgroundShoulder function in wheelchair-dependent patients is critical for preserving independence and quality of life due to lower extremity impairment. The purpose of this study was to report the revision rate, as well as clinical and radiological outcome in wheelchair-dependent patients treated with reverse total shoulder arthroplasty (RTSA) and to compare them to an ambulating population. MethodsProspectively obtained data of 21 primary RTSAs in 17 wheelchair-dependent patients (5 male, 12 female) with a median age of 72.4 years (range: 49-80) and a minimum follow-up of 2 years were analyzed retrospectively. Revision rate, clinical (Subjective Shoulder Value = SSV, relative Constant-Murley Score = rCS, wheelchair user’s shoulder pain index = WUSPI) and radiological (glenoid loosening, scapular notching, glenoid inclination) outcome, as well as implant-related parameters (baseplate peg length, glenosphere size, bony augmentation), were compared with a 2:1 matching cohort of 42 ambulating patients (10 male, 32 female) with a median age of 72.5 years (range: 56-78). ResultsThe revision rate was 9.5% in both cohorts. In the wheelchair cohort, two shoulders had to be revised due to a complete baseplate dislocation. In the matching cohort, four shoulders had to be revised due to one prosthetic dislocation, one traumatic and one atraumatic scapular spine fracture with glenoid baseplate dislocation, and one fracture of the greater tuberosity. Median preoperative SSV and rCS did not differ significantly between cohorts. Postoperative SSV was also comparable (wheelchair: median 70 (range: 10-99) vs. matching: median 70 (30-100), p = n.s.). Relative CS was significantly lower in the wheelchair cohort (65% vs. 81.4%, P = .004). Median postoperative WUSPI was 35 points (range: 13-40) for difficulty and 0 points for pain (range: 0-29). The highest difficulty and pain were found for ‘hygiene behind the back’ and ‘propulsion of wheelchair up a ramp or on uneven surface’. Glenoid loosening, scapular notching, and postoperative baseplate inclination did not differ significantly between cohorts. In the wheelchair cohort, glenoid autograft augmentation (38.1% vs. 7.1%, P = .002) and implantation of baseplates with longer pegs were performed more often (≥ 25mm: 38.1% vs. 7.1%, P = .004). ConclusionRTSA is a valuable therapeutic option for the treatment of advanced OA or irreparable rotator cuff tears in wheelchair-bound patients with high patient satisfaction. Postoperatively, poorer function and a higher rate of baseplate dislocations might be anticipated compared to ambulating patients.

The Contribution of Deleterious Rare Alleles in ENPP1 and Osteomalacia Causative Genes to Atypical Femoral Fracture.

Atypical femoral fractures (AFFs) are very rare atraumatic or mild trauma fractures in the subtrochanteric region or femoral shaft. Some unique genetic variants in Asian populations might confer susceptibility to AFF, since the incidence of AFFs is higher in Asian populations. Because rare variants have been found to be causative in some diseases and the roles of osteomalacia causative genes have not been reported, we investigated rare variants in genes causing abnormal mineralization. Exome sequencing was performed to detect variants in gene coding and boundary regions, and the frequencies of deleterious rare alleles were compared between Japanese patients with AFF (n = 42) and controls of the 4.7KJPN panel of Tohoku Medical Megabank by whole genome sequencing (n = 4773). The frequency of the deleterious rare allele of ENPP1 was significantly increased in AFF (P = .0012, corrected P [Pc] = .0155, OR 4.73, 95% CI 2.15-10.40). In multigene panel analysis, the frequencies of deleterious rare alleles of candidate genes were increased in AFF (P = .0025, OR 2.72, 95% CI 1.49-4.93). Principal component analysis of bone metabolism markers identified a subgroup of patients with AFF with higher frequencies of deleterious rare alleles in ENPP1 (P = 4.69 × 10-5, Pc = .0006, OR 8.47, 95% CI 3.76-19.09) and the candidate genes (P = 1.08 × 10-5, OR 5.21, 95% CI 2.76-9.86). AFF is associated with genes including ENPP1 that cause abnormal mineralization, suggesting that osteomalacia is an underlying condition predisposing to AFF and that higher incident rates of AFFs in Asian populations might be explained by the genetic risk factors including ENPP1.

Clinical features of methotrexate osteopathy in rheumatic musculoskeletal disease: A systematic review

BackgroundThere is growing evidence from case reports that methotrexate (MTX) therapy may impair bone metabolism in individual patients leading to low bone mass, atraumatic stress fractures and immobilizing bone pain – referred to as ‘MTX osteopathy’. However, the clinical features, risk factors and treatment options of this condition are still elusive. MethodsA systematic review was conducted according to PRISMA guidelines. Two databases (MEDLINE, Embase) were searched for published cases of MTX osteopathy in patients with rheumatic musculoskeletal diseases (RMD). Data from the included publications were extracted and descriptive statistical analysis was performed. ResultsWe report data from 32 studies describing 80 adult RMD patients with stress fractures in MTX osteopathy. Most cases were found in elderly women with longstanding RMD, especially rheumatoid arthritis (72.5%). MTX osteopathy commonly presented as stress fracture of the distal tibia (51.3%), calcaneus (35.0%) and proximal tibia (27.5%), mimicking arthritis in some cases. Although a majority of the patients met the densitometric criteria for osteoporosis (58.1%), typical osteoporotic fractures (e.g., vertebral fractures) were rarely seen. Patients frequently suffered from bilateral (55.0%), multiple (71.3%) and recurrent fractures (25.0%). Fractures mainly occurred at low to moderate doses of MTX therapy (45.0%). It should be noted that half (48.8%) of the patients did not receive systemic steroid therapy for at least 3 years. ConclusionsLow-dose MTX therapy in RMD may result in atraumatic stress fractures of the lower extremity that can mimic arthritis. MTX osteopathy is characterized by a pathognomonic type of stress fractures with band- or meander-shaped appearance along the growth plate.

Parkinson and atraumatic sternum fracture: a case report

Parkinson and atraumatic sternum fracture: a case report

Test yourself answer to question: atraumatic fracture of an abnormal humerus

Test yourself answer to question: atraumatic fracture of an abnormal humerus

Fibroblast Growth Factor-23 Secreting Tumour Induced Osteomalacia

Tumour Induced Osteomalacia (TIO) caused by a Fibroblast-Growth- Factor-23 (FGF-23) secreting tumour is a rare paraneoplastic disorder. Patients often present with non-specific complaints of pain and stiffness however findings of hypophosphataemia and inappropriately suppressed 1,25 dihidroxyvitamin D are almost pathognemonic of this condition. We present a previously healthy 51-year-old man who presented with an 18 month history of body aches, hypophosphataemia, and an incidental finding of atraumatic foot fractures on x-ray. Technetium bone scan identified diffuse skeletal pathologic fractures. He was further found to have persistent hypophosphataemia, suppressed 1-25-dihidroxyvitamin-D, renal phosphate wasting with reduced Tubular Reabsorption of Phosphate (TRP), and an FGF-23 level more than twice the upper limit of normal. Fluorodeoxyglucose (FDG) Positron Emission Tomography (PET), Gallium-68 Dotatate PET and magnetic resonance imaging further identified and characterised a 40 mm FDG and Dotatate-avid lesion in the right third rib. Histopathological examination of a biopsy specimen revealed phosphaturic mesenchymal tumour with positive FGF-23 immunohistochemical staining arising in a background consistent with osteomalacia. After tumour resection, the patient’s hypophosphataemia, reduced TRP and elevated FGF- 23 normalised and he made an excellent recovery with rehabilitation. This case report illustrates the importance of a diagnosis of hypophosphataemia in a patient with musculo-skeletal aches and alerts clinicians of the differential diagnosis of TIO.

Atraumatic thoracic spinal fracture mimicking herpes zoster neuralgia: a case report

BackgroundIntercostal neuralgia is most common in patients with herpes zoster, but it might be the initial symptom of serious diseases, such as atraumatic spinal fracture, which may lead to serious consequences if not diagnosed and treated early. Severe intercostal neuralgia is rarely reported as the first symptom of ankylosing spondylitis with atraumatic vertebral fractures.Case presentationA 70-year-old Chinese Han man previously diagnosed with ankylosing spondylitis presented to the hospital with intense intercostal pain without trauma. The patient was initially suspected of having herpes zoster neuralgia; however, he subsequently experienced numbness and weakness of both lower limbs as well as constipation. Thoracic vertebral fracture and compression of the spinal cord were detected with magnetic resonance imaging, and he underwent emergency posterior thoracic spinal canal decompression, and intercostal neuralgia was relieved after surgery. Spinal tuberculosis and tumors were later excluded by pathological examination and follow-up results. A 6-month postoperative follow-up showed that the weakness and numbness of the left lower limb had significantly improved, and his urinary function had recovered.ConclusionsPatients with ankylosing spondylitis could develop atraumatic spinal fractures. Severe intercostal neuralgia is an early indicator of spinal fractures, and spinal magnetic resonance imaging is crucial for the diagnosis.

Case of Juvenile Onset Hypophosphatasia Diagnosed in an Adult Patient

Background: Hypophosphatasia is a rare multisystem disease caused by mutations in genes encoding tissue nonspecific alkaline phosphatase, a key player in promoting bone mineralization1. Here we present a case of hypophosphatasia in a patient with history of recurrent fractures and dental caries since childhood. Case Report: Patient is a 52-year-old woman with history of multiple fractures who initially presented for follow up of osteoporosis following an atraumatic ankle fracture. Further questioning revealed a history of 16 atraumatic fractures since the age of 4, involving ankles, toes, and fingers. Several adult teeth had never developed requiring braces to fill in gaps at age 13, dental caries and tooth fractures involving the majority of her adult teeth. DEXA scan in 2019 revealed T score of -2.4 in the left femoral neck. Suspicion for hypophosphatasia in February 2019 following an ankle fracture and patient’s prior history prompted further workup, revealing low serum alkaline phosphatase levels of 29 and 32 (bone fraction 62 percent, liver fraction 38 percent), and Vitamin B6 levels elevated to 66.2. Remainder of workup, with Vitamin D, PTH, Magnesium, and Calcium was normal. A childhood history of multiple atraumatic fractures, various dental issues, with elevated Vitamin B6 and low serum alkaline phosphatase suggested Hypophosphatasia. As bisphosphonates are contraindicated in these patients due to their potential to reduce ALP, teriparatide was initiated. Discussion: Hypophosphatasia involves mutations in tissue nonspecific alkaline phosphatase, a key player in bone mineralization. In normal individuals, this enzyme dephosphorylates inorganic pyrophosphate (PPi), which otherwise inhibits bone mineralization. The mutated TNSALP leads to accumulation of PPi, and thereby unmineralized osteoid.1 Although individual presentations can vary, developmental abnormalities, such as delayed growth, early loss of primary or secondary teeth, or history of multiple fractures are characteristic. Due to the rarity of the disease, and its potential to be confused for more common bone and rheumatologic diseases, diagnosis is often delayed1. Patients in whom suspicion for hypophosphatasia is present, should undergo further testing with bone specific Alkaline phosphatase and Vitamin B6 which would be low and elevated, respectively and may be candidates for enzyme replacement therapy with bone-targeting recombinant alkaline phosphatase1. Traditional treatments such as bisphosphonates potentially decrease ALP and worsen disease, making accurate diagnosis all the more crucial. References1 Bishop N. Clinical management of hypophosphatasia. Clin Cases Miner Bone Metab. 2015;12(2):170–173.