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1 Kobe University Repository : Kernel タイトル Title 著者 Author(s) 掲載誌 巻号 ページ Citation 刊行日 Issue date 資源タイプ Resource Type 版区分 Resource Version 権利 Rights DOI JaLCDOI URL Bilateral Hip Fractures in a Patient with Osteomalacia Ishikawa, Hitoshi / Hirata, Soichiro / Simooku, Kiyoshi / Ishikawa, Makoto / Isobe, Takashi / Andoh, Yoshihiro Bulletin of allied medical sciences Kobe : BAMS (Kobe),10: Departmental Bulletin Paper / 紀要論文 publisher PDF issue:
2 Bilateral Hip Fractures in a Patient with Osteomalacia Hitoshi Ishikawa l, Soichiro Hirata], Kiyoshi Simooku 2, Makoto Ishikawa 3, Takashi Isobe l and Yoshihiro Andoh 3 We are reporting the case of a sixty-five-year old patient with bilateral sequential femoral neck fractures whose underlying pathological condition was osteomalacia. An initial radiological skeletal survey revealed multiple Looser's zone in the costal ribs, metacarpal bones, metatarsal bones, and bilateral femoral necks. Along with these roentgenographic findings, decreased serum levels of 25-0H vitamin D, calcium and phosphorus, remarkably high levels of alkaline phosphatase and parathyroid hormone (PTH), we concluded the disgnosis of osteomalacia. His left hip fracture was treated with open reduction and internal fixation. Finally we emphasized that any patient with bilateral fractures or pseudofractures should be suspected of having a metabolic bone disease. Key Words Osteomalacia, Vitamin D, Bilateral hip fractures, Post - gastrectomy syndrome. INTRODUCTION Osteomalacia is characterized by accumulation of increased amounts of unmineralized osteoid and a decrease in the rate of bone formation. The radiographic findings in osteomalacia in adults are those of generaized dimineralization with increased trabecular markings. The diagnostic radiologic features are pseudofractures (Looser's zone), which are linear radiolucencies that are perpen- FaCUlty of Health Science l and Department of Orthopaedic Surgerl, Kobe University School of Medicine 2, Kobe, and Ishikawa Hospital 3, Himeji, Japan dicular to the bone surface and most commonly found symmetrically distributed in the ribs, long bones, lateral scapular margins, and pelvic rami (1,2). In elderly patients with hip fractures many investigators have reported a significant incidence of osteomalacia (3-8). The pathogenesis of these lesions has been variously attributed to local pressure from nutrient arteries or to muscle attachment, which increased local stress (1,2). Thus, the detection of osteomalacia is important since it may influence subsequent management of the femoral fracture. Weare reporting the case of a patient with bilateral sequential femoral neck fracture associated with osteomalacia. The purposes of the present report are to review the case, and to raise the caution that all cases of bilateral fractures of femoral neck in elderly should be investigated for underlying pathology. Vol. 10, 1994 Bulletin of Allied Medical Sciences, Kobe 121
3 H. Ishikawa et al. CASE REPORT A sixty-five-year old man was first seen by the orthopaedic service at our institute December 5, Physical examination revealed an lean man with pale face. He complained of pain in bilateral chest wall, hands, feet and hips. He denied a specific injury to any region which he complained pain. He also complained of easy fatigability, malaise, and muscle weakness of the limbs. Five year before being seen by us the patient had received a subtotal gastrectomy, because of the early stage of gastric cancer. Since that time the patient had developed a malabsorption syndrome, and had lost a great deal of body weight. This seemed to be due to poor eating habits, fear of eating of the dumping syndrome and malabsorption. He denied the use of antacid, aniticovulsants, or glucocorticoids. Examination of the chest, hands, feet revealed tenderness to palpation. A skeletal survey in December 1992 revealed multiple Looser's zone in the costal ribs, metacarpal bones, metatarsal bones (Figure 1) and bilateral femoral necks (Figure 2). Pertinent laboratory data (normal values) at this time included the followings: RBC 379X10 4,WBC 8700, Hg 11.9 g/dl, serum Ca 3.7 mg/dl ( mg/di), serum P 2.9 mg/dl ( mg/di) ; unnary Ca 0.05 g/day ( gl day), unnary P 0.49 gl day ( g/day), serum GOT 15, CPT 11, BUN 15.2 and serum alkaline phosphatase 2167 U/I ( U II) which was mostly from bone fraction (Figure 3). Plasma level of Figure 1. Multiple Looser's zone in December Costal ribs (A), metatarsal bones (B), matacarpal bone (C). Arrows indicate the Looser's zone. 122 Bulletin of Allied Medical Sciences, Kobe
4 Bilateral hip fractures with osteomalacia Figure 2. Bilateral Looser's zone of the femoral neck (arrows). Figure 4. Radiograph of bilateral hip taken in September, 1993 (-) Normal serum (+) 3 2 (- ) Patient serum ~ +) Figure 3. Electrophoresis pattern of Alkaline phosphatase of the patient hydroxy-vitamin D (25-0HD) was 5 (10-55 ng/l), and dihyodroxy-vitamin D was 16 (20-76 pg/mi). Parathyroid hormone (PTH) was remarkably elevated as 138 pg/ml (10-65 pg/i). The diagnosis of ostemalacia due to postgastrectomy syndrome was made. The patient began taking calcitonin, 10 unit weekly and 25-0H vitamin D 1 microgram a day. Instead of these medication described above, patient had continued to complain bone pain all regions where he had symptoms initially. The laboratory data were not improved after six months of treatment. In June, 1993, it was revealed that serum Ca 3.9, serum P 2.9, alkaline phosphatase 1962, and plasma level of 25 -OH vitamin D 8, OH2 vitamin D 20, and PTH was 230 pg/dl. In September 19, 1993, the patient had considerable difficulty when walking with one cane or getting in and out of a chair. Roentgenograms of both hips demonstrated that cortical psudofractures at site of insertion of psoas tendon into lesser Vol. 10,
5 H. Ishikawa et al. Figure 5. Radiograph of left hip in November 20,1993. trochanter, consistent with Looser's zone (Figure 4, arrows). There was marked limitation of motion in both hips. The patient was only allowed to walk with crutches. In November 20, the patient complained of severe left hip pain which revealed the displacement fracture of the femoral neck of left side (Figure 5). The patient was treated with open reduction and internal fixation using a dynamic hip screw and side plate (Figure 6). During surgery, the histological specimens of bone and call us tissue were obtained. The bone tissue specimen demonstrated that large amounts of osteoid tissue. His postoperative course was uneventful and he returned to a full weight-bearing status on left side. However he still have fear of fracture on right hip at present time. Figure 6. The patient was treated with internal fixation using a dynamic hip screw and side plate. DISCUSSION In osteomalacia, a normal amounts of bone matrix is present. However there is deficient or delayed mineralization of this tissue (9). Although an association between osteomalacia and femoral neck fracture in the elderly has previously been reported (3-8), a recent survey showed that the prevalence of osteomalacia is now very low (10,11). The declining frequency of osteomalacia of the elderly may reflect the improvement of nutrition, but circulating concentration of 25 hydroxy vitamin D are low in this age group (12,13). Therefore it seems paradoxical in view of the accepted role of vitamin D in the maintenance of calcium and skeletal homeostasis. Laboratory data obtained at his initial visit to us 124 Bulletin of Allied Medical Sciences, Kobe
6 Bilateral hip fractures with osteomalacia showed reduced serum calcium and phosphorus, highly elevated alkaline phosphatase, and a 24-hour urine collection demonstrated a tubular resorption of calcium and phosphorus were reduced. Serum vitamin D studies also showed markedly depressed level of these values. In a case presented here is a typical osteomalacia of postgastrectomy syndrome which revealed numerous Looser's zone in roentgen ography. Therefore we confirmed the diagnosis of osteomalacia due to vitamin D deficiency. Decreased levels of 25-0H-vitamin D and calcium along with remarkably high level of alkaline phosphatase and PTH, are consistent with the presence of vitamin D deficiency (14,15). Although we have tried to treat the patient with vitamin D regimen and calcitonin, serum levels of 25-0H vitamin D and 1-25 OH2 vitamin D were not improved. This seems to be a vitamin D resistent osteomalacia. This case is interesting because the fractures occurred bilaterally at the same level, with no history of trauma. Many peseudofractures, also known as Milkman's.pesedofractures, Looser's zones (line), or umbauzonen are present in axillary border of the scapula, femoral neck, pubic rami, matacarpal bones, matatarsal bones and ribs in the present case. Fracture of the femoral neck has several predisposing factors including trauma, increased age, race, and metabolic condition (16). Although histological study of the sections obtained from surgery showed extensive osteopenia wi th large quantities of excessive lamellar osteoid, there is no agreement on the possible contribution of histologic osteomalacia to the pathogenesis of femoral neck fractures in the elderly (1 7). The author's patient had clinical symptoms of generalized bone pain and muscle weakness which led to a decline in his ablility to ambulate. Thus the patient of osteomalacia should be treated with appropriate therapy such modalities as Vitamin D and calcium carbonate supplement. The fractures must be treated effectively; in our case the femoral neck tracture was treated with internal fixation using dynamic hip screw system. The purpose of this surgery was to provide for maintenance of reduction, to persistence of femoral head vascularity. Finally we should mention that although classical vitamin D deficiency is uncommom in Japan, a thorough dietary, medication and patient history is warranted in any patient with bilateral hip fractures in elderly, and we recommend the diagnosis of osteomalacia should be entertained. REFERENCES 1. Hahn TJ: Metabolic bone disease. In Textbook of Rheumatology (Eds Kelly WN, Harris ED Jr, Ruddy S, Sledge CB) pp, , WB Sauders Co, Philadelphia, Mankin HJ: Rickets, osteomalacia and renal osteodystrphy. An update. Orthop Clin North Am 21:81-96, Chalmers J, Barclay A, Davison AM, et al: Quantitative measurements of osteoid in health and Vol. 10,
7 H. Ishikawa et al. disease. Clin Orthop ReI Res 63: Sokoliff L: Occult osteomalacia in America (U.S.A): patients with fracture of the hip. Am J Surg Pathol 2: Doppelt SH: Vitamin D. rickets. and osteomalacia. Orthop Clin North Am 15: Tucker GS. Middha VP. Sural A. et al: Incidence of osteomalacia in fractures of the proximal end of femur. Injury 19: Jenkin DHR, Roberts JG. Webster D et al: Osteomalacia in elderly patients with fractures of the proximal neck: a clinicopathological study. J Bone Joint Surg 55B: Aaron JE. Gallagher JC. Anderson J. et al: Frequency of osteomalacia and osteoporosis in fractures of the proximal femur. Lancet i: Pitt M: Osteopenic bone disease. Orthop Clin North Am 14: Wilton TJ. Hosking DJ. Pawley E. et al: Osteomalacia and femoral neck fractures in the elderly patient. J Bone Joint Surg 69B: Campbell GA. Kemm JR. Hosking DJ. et al: How common is osteomalacia in the elderly. Lancet ii: Lips p. Netelenbos JC. Jongen MJM. et al: Histomorphometric profile and vitamin D status in patients with femoral neck fractures. Metab Bone Dis ReI Res 4; Francis RM. Peacock KM. Taylor GA. et al: Calcium malabsorption in elderly women with vertebral fractures. Clin Science 66: Kaplan FS. Soriano S. Fallon MD. et al: Osteomalacia in a night nurse. Clin Orthop ReI Res 205: Paice EW. Hoffbrand Bl: Nutritional osteomalacia presenting with plantar fascitis. J Bone Joint Surg 69B: Alberts KA. Dafmborn M. Hindmarsh J. et al: Radionuclide scintimetry for diagnosis of complication following femoral neck fractures. Acta Orthop Scand 55: Bulletin of Allied Medical Sciences. Kobe
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