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  In this Article
  Case Description
  Autopsy Findings
  Histopathology
  Discussion
  Conclusion
   References
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Table of Contents    
NI FEATURE: JOURNEY THROUGH THE EONS - COMMENTARY
Year : 2019  |  Volume : 67  |  Issue : 3  |  Page : 643-647

Autopsy study of a case of skeletal fluorosis (1977)


1 Department of Neurology, PSG Institute of Medical Science and Research, Coimbatore, Tamil Nadu, India
2 Department of Neurology, MGR Medical University and Mercury Hospital, Pantheon Road, Egmore, Chennai, Tamil Nadu, India
3 Department of Neurosurgery, Institute of Neurology and Apollo Hospitals, Chennai, Tamil Nadu, India
4 Department of Neuropathology, Madras Medical College, Chennai, Tamil Nadu, India

Date of Web Publication23-Jul-2019

Correspondence Address:
Dr. Madakasira Bheemarao Pranesh
Ananda Nilaya, 11, 5th Street, Tatabad, Coimbatore - 641 012, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.263254

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How to cite this article:
Pranesh MB, Arjundas G, Kalyanaraman S, Bharati R S. Autopsy study of a case of skeletal fluorosis (1977). Neurol India 2019;67:643-7

How to cite this URL:
Pranesh MB, Arjundas G, Kalyanaraman S, Bharati R S. Autopsy study of a case of skeletal fluorosis (1977). Neurol India [serial online] 2019 [cited 2019 Aug 24];67:643-7. Available from: http://www.neurologyindia.com/text.asp?2019/67/3/643/263254




Endemic fluorosis is prevalent in several states of India. Punjab and Andhra Pradesh have large belts of hyper-endemic areas, and more recently, endemic areas have been described from Tamil Nadu, Karnataka, Rajasthan and Uttar Pradesh.

Fluorosis in India is due to chronic fluoride intoxication, mainly through drinking of fluoride containing water. This affects the teeth and skeleton and also produces neurological complications. Dental fluorosis is the earliest manifestation of this condition, followed in the course of time by skeletal changes. These have been extensively described by several earlier workers.[1],[2],[3],[4],[5],[6],[7],[8],[9],[10] However, the neurological complications of fluorosis are, fortunately, less common and so far only 209 cases have been reported in Indian literature. Only a few detailed autopsy reports are available in Indian literature. We are presenting a detailed autopsy study of a patient who presented with crippling skeletal and neurological fluorosis.


  Case Description Top


An adult male subject, aged 40 years (MIN. NO.3049/77), was admitted to the Institute of Neurology, General Hospital, Madras with severe spasms of all four limbs. Two years ago, he fell down prone while walking and immediately developed flaccid tetraplegia with loss of sensations over the trunk, limbs and also developed urinary incontinence. He later developed spasticity and urinary bladder recovered its function. He was admitted at that point of time to the Coimbatore Medical College Hospital where a diagnosis of 'skeletal fluorosis with cervical myeloradiculopathy' was made. He was given serpentine (aluminium meta silicate), 100 mg daily orally, for nearly sixteen months. As the neurological condition had become worse, he was referred to the Institute of Neurology, Madras. The patient came from a highly endemic area of fluorosis, i.e., Pollachi Taluk of Coimbatore District (Tamil Nadu). His brother and daughter were victims of skeletal fluorosis with no neurological complications.

His nutritional state was moderate, and he was like one piece of rigid wood, with all the four limbs adducted and flexed [Figure 1]. He had signs of cervical myeloradiculopathy with wasting confined mainly to the shoulders and hands. The neck was rigid. He had a chest expansion of just 1 cm but the lungs were clear. The heart was normal. The blood pressure was 120/80 mm of mercury. He required help for all daily activities.
Figure 1: Four limbs adducted and flexed

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The skeletal survey showed the classic evidence of fluorosis in the form of dense bones and ligament calcification [Figure 2]. The haemogram was normal. Blood urea was 20 mg%. The two-hour post–prandial blood sugar level was 108 mg/dl. Serum alkaline phosphatase was 20 KA units. Urinary fluoride content was 4.1 mg/l (normal range: 0.2-1.1 mg/l). The fluoride content in bone ash was 0.252% (normal: 0.045%). Urine did not darken on standing. Cisternal cerebrospinal fluid (CSF) protein content was 36 mg% and was acellular. CSF venereal disease research laboratory (VDRL) test was non-reactive. The cisternal myelogram with myodil showed marked median bars and hold up of the dye at the level of third cervical vertebra [Figure 3]a and [Figure 3]b.
Figure 2: X Ray of radius and ulna showing thickening of the bone and calcification of interosseous membrane. The distal ends of the bones are osteoporotic

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Figure 3: (a and b) The cisternal myelogram with myodil showed marked median bars and hold up of the dye at the level of third cervical vertebra

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As the patient had a relentlessly progressive spinal cord compression, a cervical laminectomy was decided upon. At surgery, the supra-spinous ligaments were found to be calcified. Laminectomy was done from the second cervical vertebra to the sixth cervical vertebra. The laminae felt soft to cut. The dura was found to be compressed all around by the bone. The dura was not opened. Immediately on removal of the laminae, the cord bulged up as if it was being released from incarceration. Post-operatively, the neurological deficit did not improve and he developed pulmonary complications and unfortunately died on the seventh post-operative day inspite of all efforts.


  Autopsy Findings Top


The cervical cord was bulging. The spinal canal all through the cervical and thoracic regions was like a slit and the cord was compressed severely anteroposteriorly, the maximum compression being in the cervical region. The laminae were cut without much resistance. The lung on the right side showed consolidation of the upper and middle lobes. The brain, pituitary gland, thyroid gland, parathyroid gland and viscerae appeared normal.

The skeleton was almost double the size of normal, showed classic changes of fluorosis, and was chalky-white in colour [Figure 4].
Figure 4: The radius and ulna after the post-mortum showing thickening of the bone and calcification of interosseous membrane

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Various ligaments were calcified [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10]. The sagittal diameter of the spinal canal at C5 and C6 was just 5mm (normal is from 15 to 18 mm). The  Atlas More Details appeared normal. The spinal canal size, in the sagittal plane, at C2, C3 and C4 levels, could not be measured as a laminectomy had been done.
Figure 5: Fused vertebrae, chalky white with irregular bones. Note the fused axis with the C3 vertebrae

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Figure 6: The patient's femur bones with tibia, fibula and patella. Please note the increase in the size of the bones with rough edges at the attachment of the ligaments

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Figure 7: Specimen of the skull showing chalky-white bones with jaw and brown discoloration of teeth

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Figure 8: Base of skull showing irregular foramen magnum and rough ligamentous attachments

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Figure 9: The pelvic bones showing enormous thickening, roughness and calcification at the foramina

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Figure 10: The sacrotuberous ligament is densely calcified

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  Histopathology Top


The Ammon's horn and cerebellum showed anoxic changes [Figure 11]. Anterior horn cells of the spinal cord showed increased lipofusin pigment [Figure 12]; there was some amount of thickening of the meninges at the roots [Figure 13]. The right biceps muscle showed neurogenic atrophy. The thyroid gland showed occasional haemorrhage [Figure 14]. The kidney showed cloudy swelling of some glomeruli and tubules. The pituitary gland, parathyroids, adrenals and pancreas were normal. The liver, stomach and heart were normal.
Figure 11: Cerebellum showing anoxic changes

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Figure 12: Anterior horn cell showing increased lipofusion pigment

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Figure 13: Thickening of the meninges at the roots

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Figure 14: Haemorrhage in the thyroid

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  Discussion Top


Detailed autopsy studies in subjects of endemic fluorosis are scanty. Our patient's death was attributable to the rigid chest wall and the consequent pulmonary infection, resulting in respiratory failure. Although the patient had some amount of osteoporosis at the distal ends of the long bones and had soft bones suggestive of hyperparathyroidism, histologically the parathyroids appeared normal. Earlier workers had shown hyperparathyroidism in fluorosis. The haemorrhages in the thyroid gland are probably due to hypoxia.


  Conclusion Top


A man aged 40 years, had a relentlessly progressive compressive cervical myeloradiculopathy due to skeletal fluorosis. Following a cervical laminectomy, he developed pulmonary complications and unfortunately died. A detailed autopsy was done and this is discussed. Such autopsy studies are a few in the Indian Literature.

Acknowledgment

Our grateful thanks to the patient's family for their generosity and permission to conduct the autopsy and to conduct the skeletal study. We are thankful to Shri Venkataraman, Water analyst, King's Institute, Guindy, Madras, for estimating the fluoride content of urine and bone and to Dr. K.A.V.R. Krishnamachari, Deputy Director, National Institute of Nutrition, Hyderabad, for his helpful suggestions in preparing the manuscript. The patient was referred to Madras Institute of Neurology by late Prof T.K. Ganeshan, Professor of Medicine and Head of the Department of the Coimbatore Medical College.

Post script

Our thanks are due to Ebin Siby, Doctor of Pharmacy, Sri Ramakrishna Hospital, Coimbatore, for his secretarial help.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Reddy D R. Neurology of endemic skeletal fluorosis. Neurol India 2009;57:7-12.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
Hattab F. Fluoride in drinking water. Community Dent Oral Epidemiol 1980;8:211-211.  Back to cited text no. 2
    
3.
Ramesh M, Malathi N, Ramesh K, Aruna R, Kuruvilla S. Comparative evaluation of dental and skeletal fluorosis in an endemic fluorosed district, Salem, Tamil Nadu. J Pharm Bioallied Sci 2017;9:88.  Back to cited text no. 3
    
4.
Shruthi M, Santhuram A, Arun H, Kishore Kumar B. A comparative study of skeletal fluorosis among adults in two study areas of Bangarpet Taluk, Kolar. Indian J Public Health 2017;61:61.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Rawlani S, Rawlani S, Rawlani S. Assessment of skeletal and non-skeletal fluorosis in endemic fluoridated areas of Vidharbha Region, India: A survey. Indian J Community Med 2010;35:298.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Krishnamachari K.A.V.R. Some aspects of fluorosis in South India-A new approach to prevent the problem. Proceedings of the Symposium on Fluorosis. 1974;54:519-29.  Back to cited text no. 6
    
7.
Shortt H.E, McRobert GR, Barnard TW, Mannadi Nayar AS. Endemic fluorosis in the Madras presidency. Indian J Med Res 1937;25:553-68.  Back to cited text no. 7
    
8.
Singh A, Dass R. Heyrah, Jolly SS. Skeletal changes in endemic fluorosis. J Bone Jt Surg. 1962;443:806.  Back to cited text no. 8
    
9.
Jolly SS. Endemic fluorosis. Progress in clinical Medicine in India. Editor: Ahuja MMS. 1976:106.  Back to cited text no. 9
    
10.
Teotia SPS, Teotia M. Secondary hyperparathyroidism in patients with endemic skeletal fluorosis. Brit Med J 1973;1:637.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11], [Figure 12], [Figure 13], [Figure 14]



 

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