Int J Gynaecol Obstet 16: 34^37, 1978
Multiple Spontaneous Fractures of the Pelvis and Femoral Neck During Pregnancy Subrata K. Chatterjee, Biswajit Ghosh and Biren Das Department of Obstetrics and Gynaecology, Vivekananda Institute of Medical Science, Ramakrishna Mission Seva Pratisthan, Calcutta, India
ABSTRACT Chatterjee SK, Ghosh B, Das B (Dept of Obstetrics and Gynaecology, Vivekananda Institute of Medical Science, Ramakrishna Mission Seva Pratisthan, Calcutta, India). Multiple spontaneous fractures of the pelvis andfemoral neck during pregnancy. Int) Gynaecol Obstet 16: 34-37, 1978. This paper presents the case report of a pregnant northern Indian woman who sustained multiple spontaneous fractures of the pelvis and femoral neck due to severe osteomalacia, without any resulting skeletal deformity. Osteomalacia is endemic in the northern part of India, but spontaneous fracture from the disease is extremely rare.
INTRODUCTION A spontaneous fracture can be described as a fracture which occurs in a patient who is mentally healthy and emotionally stable and who presents with no history of violence, trauma or twisting strain to the lower limb (6). There have been few reported cases of spontaneous fracture of the pelvis or femoral neck, but a m o n g those reported, three distinct groups of fractures can be identified. T h e first group includes pathologic fractures with demonstrable bone destruction. These fractures usually occur as a result of bony neoplasms, Paget's disease, hyperparathyroidism and fibrous bony dysplasia (13). T h e second group includes fatigue or stress fractures which usually occur in young adults engaged in prolonged and strenuous physical activity (4, 8). T h e third group of fractures occurs most frequently in elderly subjects as a result of senile osteoporosis, particularly following irradiation for pelvic malignancy (5). Spontaneous fracture is extremely unusual in osteomalacia and is particularly rare in young patients (1, 6). Sporadic cases have been reported in elderly patients, particularly where osteoporosis is also a contributing factor (1, 11, 13).
Int J Gynaecol Obstet 16
T h e present paper reports a rare case of severe osteomalacia during pregnancy, with multiple spontaneous fractures of the pelvis and femoral neck but without any resulting skeletal deformity.
CASE R E P O R T H.K., a 28-year-old Muslim woman, gravida 5, was pregnant and at term when she was admitted to the hospital as an emergency patient in March 1976. She presented with a history of severe low back pain of two weeks' duration followed by a sudden inability to walk which lasted for three to four days. Four previous pregnancies ended in uneventful term deliveries, the most recent of which occurred three years before this admission. H.K. belonged to a disadvantaged socioeconomic group, but there was nothing in her past history that was relevant to her present condition. H.K. was of average build, her blood pressure at admission was 120/70 mm Hg, and there was no edema. Her right hip joint and pubes were markedly tender, but her left hip was not. Her right lower limb was shortened by 2 cm and was rotated laterally. Movement, especially abduction, was restricted. Her left lower limb was free and there was no tenderness over the spine. Her uterus was of term size with a nonengaged cephalic presentation. Fetal heart sounds were audible. A detailed pelvic examination could not be performed due to severe tenderness on the forepelvis. H.K.'s hemoglobin level was 7.5 gm/100 ml, her total red blood cell count was 3.6 million/mm 3 and her total and differential leukocyte counts were within normal range. Her erythrocyte sedimentation rate was 40 m m / h r , and examination of her urine and stool samples revealed no abnormality. Fasting blood sugar and urea tests were normal and one Venereal Disease Research Laboratories test was negative. H.K.'s blood group was 0 Rhesus negative, with a negative Coombs'
Spontaneous fracture during pregnancy
35
Fig. 1. Antenatal radiograph showing cephalic presentation and multiple fractures of the rarefied pelvis and right femoral neck (arrows).
test. A straight x-ray of the abdomen (posterior/ anterior view) showed fractures of the right femoral neck, the superior pubic rami and the left inferior pubic ramus (Fig. 1). A healthy male weighing 2500 gm was delivered by elective cesarean section on the day following H.K.'s admission. T u b a l ligation was performed. Repeat radiologic examination on the eighth postoperative day revealed multiple fractures of the pelvis and the right femoral neck, with gross demineralization of the bones but without any skeletal deformity (Fig. 2). Laboratory evaluations revealed that H.K.'s serum calcium was 8.4 mg/100 ml (normal = 9.5-10.5 mg/100 ml), her inorganic phosphate was 3.1 mg/100 ml (normal = 3.5-4.5 mg/100 ml), her alkaline phosphatase was 47.5 King-Armstrong units (KAU) (normal = 6-12 KAU), and she had a normal serum protein and a slightly altered albumin-globulin ratio. Tests performed on urine samples revealed that her 24-hour excretion of calcium and inorganic phosphate were 88 mg (normal = 100-300 mg) and 460 mg (normal = 800-1200 mg), respectively. A provisional diagnosis of severe osteomalacia was made. The patient was treated with bedrest, blood transfusion and parenteral hematinics. Calcium gluconate (5.0 gm) and calciferol (1.25 mg) were administered daily by mouth. Four weeks after delivery, H.K.'s femoral neck
fracture was treated by osteotomy and pin-plating with closed adductor tenotomy. Histopathologic examination of bone samples taken from the right iliac crest during the operation revealed a markedly rarefied bone with very little calcification and some areas where calcification was entirely absent. T h e marrow spaces were filled with a reactive marrow hyperplasia and patches of fibrosis. There was marked osteoblastic activity of the bony lamellae with new osteoid-like formation (Fig. 3). H.K. was discharged from the hospital after a stay of five months and advised to continue oral calcium and vitamin D (2.0 g m / d a y and 0.5 m g / day, respectively) for another three months. At follow-up examination six weeks later, H.K. was in sound health and had no walking disability (Fig. 4), and her blood biochemistry was within normal limits.
DISCUSSION Osteomalacia has become an exceedingly rare disease in countries of the developed world, but it is still endemic in the northern parts of India and China (7, 9, 12). T h e reported incidence during pregnancy in the endemic areas of India is between 1% and 3% (9, 10); in China it is about 10% (7). Clark et al (2) and Swan and Cooke (11) observed
IntJ Gynaecol Obstei 16
36
Chalterjee et al
Fig. 2. Radiograph on the eighth postnatal day shows grossly demineralized bones, with fractures of the right femoral neck, the superior pubic rami and the left inferior pubic ramus (arrows).
ÍWW*
»J "V
~
•i
•
• •• 'i
'
Í \ v Fig. 3. Photomicrograph of histologic section from osteomalacic bone showing increased osteoid seams and osteoblastic proliferation (HE, x 100).
that most cases of osteomalacia during pregnancy detected in Britain in recent years were among immigrants from northern India and that the majority of their blood relatives also suffered from
hit J Gynaecol Obstet 16
Fig. 4. Radiograph 22 weeks after internal fixation of the femoral neck and treatment of osteomalacia shows united fractures and dense mineralized bones.
subclinical disorders (2). T h e important causes of the disease in the endemic areas of India are a dietary deficiency of calcium and vitamin D, the inadequate absorption of these elements from the gut due to a predominantly cereal diet containing excess phytic acid and a lack of exposure to sunlight
Spontaneous fracture during pregnancy
due to the custom that requires women to be heavily veiled in public (2, 9, 12). In addition, some patients may have resistance to the action of vitamin D (11). Occasionally, pregnancy and the puerperium may precipitate symptoms from a subclinical state (3). All of the factors described above were present in the case presented. H.K. responded rapidly to the oral administration of calcium and vitamin D, indicating that her osteomalacia was nutritional and caused by a lack of sunlight which prevented calciferol from being formed in the skin. Pregnancy probably precipitated the condition which culminated in the occurrence of spontaneous fractures. Generalized bone aches followed by characteristic bony deformities are the usual first signs of the disorder. T h e patient reported here first experienced severe aching pains in her hips and back, followed by the inability to walk. She sustained multiple spontaneous fractures without any resulting skeletal deformity, an occurrence which is extremely rare. Apart from the possibly unique entity, the case also points toward the prevalence of nutritional bone disease among northern Indian people and the necessity for community surveys. Unfortunately, the other family members of the patient could not be examined.
37
REFERENCES 1. Chalmers J, Conacher W D H , G a r d n e r DL, Scott PJ: Osteomalacia—a common disease in elderly women. J Bone Joint Surg |Br] 4973:403, 1967. 2. Clark F, Simpson W, Young J R : Osteomalacia in immigrants from the Indian subcontinent in Newcastle upon T y n e . Proc R Soc M e d 65:478, 1972. 3. Felton D J C , Stone W D : Osteomalacia in Asian immigrants during pregnancy. Br Med J 7:1521, 1966. 4. Gibbens J E : March fracture of the neck of the femur. J Bone Joint Surg 27:162, 1945. 5. Jeffery C C : Spontaneous, fractures of the femoral neck. J Bone Joint Surg [Br] « f l : 5 4 3 , 1962. 6. Jeffery C C : Spontaneous fractures of the femoral neck. O r t h o p Clin North Am 5:713, 1974. 7. Maxwell J P , Miles L M : Osteomalacia in China. J Obstet Gynaecol Br C o m m o n w 32:433, 1925. 8. Miller LF: Bilateral stress fracture of the neck of the femur. J Bone J o i n t Surg [Am] 32/1:695, 1950. 9. Mukherjee CL: Osteomalacia. In Obstetrics a n d Gynaecology in the Tropics a n d Developing Countries (ed J B Lawson, DB Stewart), p 29. Edward Arnold Publishers, London, 1967. 10. Scott AC: Osteomalacia in India. Indian J Med Res 4:140, 1916. 11. Swan C H J , Cooke W T : Nutritional osteomalacia in immigrants in an urban community. Lancet 2:456, 1971. 12. Vaishnava H P , Rizvi SNA: Nutritional osteomalacia in immigrants in an urban c o m m u n i t y (letter to the editor). Lancet 2:1147, 1971. 13. Wilson J N : Fractures a n d J o i n t Injuries, p 1233. Churchill Livingstone, London, 1976.
ACKNOWLEDGMENT The authors are grateful to the Secretary of Ramakrishna Mission Seva Pratisthan, for his permission to publish hospital data. We also wish to express our thanks to Dr S. Punja and Dr B. Sen, for allowing us access to the patient whose case report is presented in this paper.
Address for reprints: Subrata K. Chatterjee P-599 Keyatala Rd Calcutta 700 029 India
IntJ Gynaecol Obstet 16
Multiple Spontaneous Fractures of the Pelvis and Femoral Neck During Pregnancy Subrata K. Chatterjee, Biswajit Ghosh and Biren Das Department of Obstetrics and Gynaecology, Vivekananda Institute of Medical Science, Ramakrishna Mission Seva Pratisthan, Calcutta, India
ABSTRACT Chatterjee SK, Ghosh B, Das B (Dept of Obstetrics and Gynaecology, Vivekananda Institute of Medical Science, Ramakrishna Mission Seva Pratisthan, Calcutta, India). Multiple spontaneous fractures of the pelvis andfemoral neck during pregnancy. Int) Gynaecol Obstet 16: 34-37, 1978. This paper presents the case report of a pregnant northern Indian woman who sustained multiple spontaneous fractures of the pelvis and femoral neck due to severe osteomalacia, without any resulting skeletal deformity. Osteomalacia is endemic in the northern part of India, but spontaneous fracture from the disease is extremely rare.
INTRODUCTION A spontaneous fracture can be described as a fracture which occurs in a patient who is mentally healthy and emotionally stable and who presents with no history of violence, trauma or twisting strain to the lower limb (6). There have been few reported cases of spontaneous fracture of the pelvis or femoral neck, but a m o n g those reported, three distinct groups of fractures can be identified. T h e first group includes pathologic fractures with demonstrable bone destruction. These fractures usually occur as a result of bony neoplasms, Paget's disease, hyperparathyroidism and fibrous bony dysplasia (13). T h e second group includes fatigue or stress fractures which usually occur in young adults engaged in prolonged and strenuous physical activity (4, 8). T h e third group of fractures occurs most frequently in elderly subjects as a result of senile osteoporosis, particularly following irradiation for pelvic malignancy (5). Spontaneous fracture is extremely unusual in osteomalacia and is particularly rare in young patients (1, 6). Sporadic cases have been reported in elderly patients, particularly where osteoporosis is also a contributing factor (1, 11, 13).
Int J Gynaecol Obstet 16
T h e present paper reports a rare case of severe osteomalacia during pregnancy, with multiple spontaneous fractures of the pelvis and femoral neck but without any resulting skeletal deformity.
CASE R E P O R T H.K., a 28-year-old Muslim woman, gravida 5, was pregnant and at term when she was admitted to the hospital as an emergency patient in March 1976. She presented with a history of severe low back pain of two weeks' duration followed by a sudden inability to walk which lasted for three to four days. Four previous pregnancies ended in uneventful term deliveries, the most recent of which occurred three years before this admission. H.K. belonged to a disadvantaged socioeconomic group, but there was nothing in her past history that was relevant to her present condition. H.K. was of average build, her blood pressure at admission was 120/70 mm Hg, and there was no edema. Her right hip joint and pubes were markedly tender, but her left hip was not. Her right lower limb was shortened by 2 cm and was rotated laterally. Movement, especially abduction, was restricted. Her left lower limb was free and there was no tenderness over the spine. Her uterus was of term size with a nonengaged cephalic presentation. Fetal heart sounds were audible. A detailed pelvic examination could not be performed due to severe tenderness on the forepelvis. H.K.'s hemoglobin level was 7.5 gm/100 ml, her total red blood cell count was 3.6 million/mm 3 and her total and differential leukocyte counts were within normal range. Her erythrocyte sedimentation rate was 40 m m / h r , and examination of her urine and stool samples revealed no abnormality. Fasting blood sugar and urea tests were normal and one Venereal Disease Research Laboratories test was negative. H.K.'s blood group was 0 Rhesus negative, with a negative Coombs'
Spontaneous fracture during pregnancy
35
Fig. 1. Antenatal radiograph showing cephalic presentation and multiple fractures of the rarefied pelvis and right femoral neck (arrows).
test. A straight x-ray of the abdomen (posterior/ anterior view) showed fractures of the right femoral neck, the superior pubic rami and the left inferior pubic ramus (Fig. 1). A healthy male weighing 2500 gm was delivered by elective cesarean section on the day following H.K.'s admission. T u b a l ligation was performed. Repeat radiologic examination on the eighth postoperative day revealed multiple fractures of the pelvis and the right femoral neck, with gross demineralization of the bones but without any skeletal deformity (Fig. 2). Laboratory evaluations revealed that H.K.'s serum calcium was 8.4 mg/100 ml (normal = 9.5-10.5 mg/100 ml), her inorganic phosphate was 3.1 mg/100 ml (normal = 3.5-4.5 mg/100 ml), her alkaline phosphatase was 47.5 King-Armstrong units (KAU) (normal = 6-12 KAU), and she had a normal serum protein and a slightly altered albumin-globulin ratio. Tests performed on urine samples revealed that her 24-hour excretion of calcium and inorganic phosphate were 88 mg (normal = 100-300 mg) and 460 mg (normal = 800-1200 mg), respectively. A provisional diagnosis of severe osteomalacia was made. The patient was treated with bedrest, blood transfusion and parenteral hematinics. Calcium gluconate (5.0 gm) and calciferol (1.25 mg) were administered daily by mouth. Four weeks after delivery, H.K.'s femoral neck
fracture was treated by osteotomy and pin-plating with closed adductor tenotomy. Histopathologic examination of bone samples taken from the right iliac crest during the operation revealed a markedly rarefied bone with very little calcification and some areas where calcification was entirely absent. T h e marrow spaces were filled with a reactive marrow hyperplasia and patches of fibrosis. There was marked osteoblastic activity of the bony lamellae with new osteoid-like formation (Fig. 3). H.K. was discharged from the hospital after a stay of five months and advised to continue oral calcium and vitamin D (2.0 g m / d a y and 0.5 m g / day, respectively) for another three months. At follow-up examination six weeks later, H.K. was in sound health and had no walking disability (Fig. 4), and her blood biochemistry was within normal limits.
DISCUSSION Osteomalacia has become an exceedingly rare disease in countries of the developed world, but it is still endemic in the northern parts of India and China (7, 9, 12). T h e reported incidence during pregnancy in the endemic areas of India is between 1% and 3% (9, 10); in China it is about 10% (7). Clark et al (2) and Swan and Cooke (11) observed
IntJ Gynaecol Obstei 16
36
Chalterjee et al
Fig. 2. Radiograph on the eighth postnatal day shows grossly demineralized bones, with fractures of the right femoral neck, the superior pubic rami and the left inferior pubic ramus (arrows).
ÍWW*
»J "V
~
•i
•
• •• 'i
'
Í \ v Fig. 3. Photomicrograph of histologic section from osteomalacic bone showing increased osteoid seams and osteoblastic proliferation (HE, x 100).
that most cases of osteomalacia during pregnancy detected in Britain in recent years were among immigrants from northern India and that the majority of their blood relatives also suffered from
hit J Gynaecol Obstet 16
Fig. 4. Radiograph 22 weeks after internal fixation of the femoral neck and treatment of osteomalacia shows united fractures and dense mineralized bones.
subclinical disorders (2). T h e important causes of the disease in the endemic areas of India are a dietary deficiency of calcium and vitamin D, the inadequate absorption of these elements from the gut due to a predominantly cereal diet containing excess phytic acid and a lack of exposure to sunlight
Spontaneous fracture during pregnancy
due to the custom that requires women to be heavily veiled in public (2, 9, 12). In addition, some patients may have resistance to the action of vitamin D (11). Occasionally, pregnancy and the puerperium may precipitate symptoms from a subclinical state (3). All of the factors described above were present in the case presented. H.K. responded rapidly to the oral administration of calcium and vitamin D, indicating that her osteomalacia was nutritional and caused by a lack of sunlight which prevented calciferol from being formed in the skin. Pregnancy probably precipitated the condition which culminated in the occurrence of spontaneous fractures. Generalized bone aches followed by characteristic bony deformities are the usual first signs of the disorder. T h e patient reported here first experienced severe aching pains in her hips and back, followed by the inability to walk. She sustained multiple spontaneous fractures without any resulting skeletal deformity, an occurrence which is extremely rare. Apart from the possibly unique entity, the case also points toward the prevalence of nutritional bone disease among northern Indian people and the necessity for community surveys. Unfortunately, the other family members of the patient could not be examined.
37
REFERENCES 1. Chalmers J, Conacher W D H , G a r d n e r DL, Scott PJ: Osteomalacia—a common disease in elderly women. J Bone Joint Surg |Br] 4973:403, 1967. 2. Clark F, Simpson W, Young J R : Osteomalacia in immigrants from the Indian subcontinent in Newcastle upon T y n e . Proc R Soc M e d 65:478, 1972. 3. Felton D J C , Stone W D : Osteomalacia in Asian immigrants during pregnancy. Br Med J 7:1521, 1966. 4. Gibbens J E : March fracture of the neck of the femur. J Bone Joint Surg 27:162, 1945. 5. Jeffery C C : Spontaneous, fractures of the femoral neck. J Bone Joint Surg [Br] « f l : 5 4 3 , 1962. 6. Jeffery C C : Spontaneous fractures of the femoral neck. O r t h o p Clin North Am 5:713, 1974. 7. Maxwell J P , Miles L M : Osteomalacia in China. J Obstet Gynaecol Br C o m m o n w 32:433, 1925. 8. Miller LF: Bilateral stress fracture of the neck of the femur. J Bone J o i n t Surg [Am] 32/1:695, 1950. 9. Mukherjee CL: Osteomalacia. In Obstetrics a n d Gynaecology in the Tropics a n d Developing Countries (ed J B Lawson, DB Stewart), p 29. Edward Arnold Publishers, London, 1967. 10. Scott AC: Osteomalacia in India. Indian J Med Res 4:140, 1916. 11. Swan C H J , Cooke W T : Nutritional osteomalacia in immigrants in an urban community. Lancet 2:456, 1971. 12. Vaishnava H P , Rizvi SNA: Nutritional osteomalacia in immigrants in an urban c o m m u n i t y (letter to the editor). Lancet 2:1147, 1971. 13. Wilson J N : Fractures a n d J o i n t Injuries, p 1233. Churchill Livingstone, London, 1976.
ACKNOWLEDGMENT The authors are grateful to the Secretary of Ramakrishna Mission Seva Pratisthan, for his permission to publish hospital data. We also wish to express our thanks to Dr S. Punja and Dr B. Sen, for allowing us access to the patient whose case report is presented in this paper.
Address for reprints: Subrata K. Chatterjee P-599 Keyatala Rd Calcutta 700 029 India
IntJ Gynaecol Obstet 16
