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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 9  |  Issue : 3  |  Page : 435-438

A case report of skeletal fluorosis leading to cervical compressive myelopathy and a review of literature


Department of Medicine, All India Institute of Medical Sciences, Bhopal, Madhya Pradesh, India

Date of Submission24-Jun-2022
Date of Acceptance23-Aug-2022
Date of Web Publication29-Sep-2022

Correspondence Address:
Dr. Abhishek Singhai
Department of Medicine, All India Institute of Medical Sciences, Bhopal 462020, Madhya Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/mgmj.mgmj_92_22

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  Abstract 

Fluorosis is a public health problem that is caused by excess intake of fluoride through ground/deep bore water. It gets deposited in the bones, teeth, and soft tissues. Fluoride in the human body acts as a “double-edged sword.” Fluoride is beneficial in small amounts but toxic in large amounts. People who have consumed 10–20 mg of fluoride per day for more than 10–20 years may develop crippling skeletal fluorosis. Stiffness and pain in the major joints, including the neck, back, hips, and knees, reduce mobility. The bone structure may change and ligaments may calcify in extreme cases, resulting in muscular weakness and pain. Here, we have reported a case of chronic quadriparesis due to compressive cervical myelopathy. After extensive workup, diagnosis of skeletal fluorosis was made.

Keywords: Fluorosis, myelopathy, public health, quadriparesis


How to cite this article:
Singhai A, Mishra VN, Ingle V. A case report of skeletal fluorosis leading to cervical compressive myelopathy and a review of literature. MGM J Med Sci 2022;9:435-8

How to cite this URL:
Singhai A, Mishra VN, Ingle V. A case report of skeletal fluorosis leading to cervical compressive myelopathy and a review of literature. MGM J Med Sci [serial online] 2022 [cited 2022 Nov 29];9:435-8. Available from: http://www.mgmjms.com/text.asp?2022/9/3/435/357493




  Introduction Top


Fluorosis is a public health problem that is caused by excess intake of fluoride through drinking water and food, rarely by inhalation. It gets deposited in the bones, teeth, and soft tissues. Fluorosis-induced dental effects appear much earlier than skeletal effects in persons exposed to high levels of fluoride.[1] Ingestion of fluoride after 6 years of age will not cause dental fluorosis. According to the World Health Organization, the safe limit of fluoride consumption is 1.5 parts per million (ppm) or milligram (mg) per liter. Fluoride in the human body acts as a “double-edged sword.” Fluoride is beneficial in small amounts but toxic in large amounts.[2] People who have consumed 10–20 mg of fluoride per day for more than 10–20 years may develop crippling skeletal fluorosis. Fluoride accumulates in the bone over a long period in skeletal fluorosis. Stiffness and pain in the major joints, including the neck, back, hips, and knees, reduce mobility. The bone structure may change and ligaments may calcify in extreme cases, resulting in muscular weakness and pain. As the bones become stiffer, the condition becomes increasingly painful and can lead to impairment and permanent disability.[3] Known fluoride belts in the world include one that stretches from Syria through Jordan, Egypt, Libya, Algeria, Sudan, and Kenya and another that stretches from Turkey through Iraq, Iran, Afghanistan, India, northern Thailand, and China. In 2014, fluorosis was reported in 230 districts in 19 Indian states, affecting over 60 million people nationwide. Rajasthan is most affected by fluorosis, followed by Telangana, Andhra Pradesh, Bihar, Madhya Pradesh, and Uttar Pradesh.[4]

Here, we have reported a case of skeletal fluorosis leading to compressive cervical myelopathy.


  Case history Top


A 43-year-old male, a daily wage worker, belonging to a lower socio-economic class, from Chandala village of Chhatarpur district of Madhya Pradesh, India presented with a history of low backache and neck pain since 10 years, which was insidious onset, gradually progressive associated with the history of morning stiffness, and more during the winter season when compared with other seasons. It was associated with multiple large and small joint pain, for which he was taking over-the-counter analgesics. Last year he had a burning sensation over both upper and lower limbs. Gradually, he developed proximal as well as distal weakness in the upper limb, later on involved both lower limbs within 3 months. He also developed overflow urinary incontinence (suggestive of the neurogenic bladder) and constipation over the last 1 month. There was no history of smoking, exposure to coal smoke, or use of rock salt. In his family, he had five brothers, all of them had chronic backache and arthralgia, and they walk by bending forward.

On examination, his vitals and general examination were normal. Examination of oral cavity and teeth examination was normal. Central nervous system examination revealed normal higher mental functions, normal cranial nerves, normal muscle bulk, increased tone in all four limbs, 2/5 power in upper limbs, 3/5 power in lower limbs, poor hand grip, exaggerated all deep tendon reflexes, and extensor plantar reflex. On sensory examination, pain, touch, temperature, and vibration were impaired. On examination of neck, restriction of movement was there; flexion, extension, and lateral movement were involved. Restricted movement of the back was also present. Finally, a provisional diagnosis of compressive cervical myelopathy with bowel and bladder incontinence with sensory involvement was made. His routine investigations were done and results were as follows: hemoglobin 13.5 g/dL, total leucocyte count 4850/µL, platelets 1.8 lakhs/µL, erythrocyte sedimentation rate 12 mm/h, creatinine 0.69 mg/dL, alkaline phosphatase 185 U/L, total protein 6.73 g/dL, total calcium 9.02 mg/dL, magnesium 2.19 mg/dL, phosphate 3.79 mg/dL, aspartate aminotransferase 34 U/L, alanine aminotransferase 31 U/L, total bilirubin 0.62 mg/dL, serum sodium 133 mmol/L, serum potassium 3.8 mmol/L, serum chloride 100 mmol/L, and uric acid 5.47 mg%; urine microscopy was normal. Further investigations showed tests for human immunodeficiency virus-negative, human leucocyte antigen B27-negative, and rheumatoid factor-negative; C-reactive protein was 2.1 mg/dL. Magnetic resonance imaging (MRI) of the spine was done which was suggestive of the severe ossified posterior longitudinal ligament (PLL) in the upper cervical spine (C2–C4) causing compressive myelopathy, along with calcification, thickening of anterior longitudinal ligament, ligamentum flavum, and multilevel bridging anterior and posterior osteophytes [Figure 1]. Diffuse sclerotic marrow changes involving all the bones in the spine, bilateral ribs, and pelvic bones were present [Figure 2] and [Figure 3]. Diffuse skeletal fluorosis was suspected. Water fluoride level of well water, from where he was taking water, was estimated in the State Research Laboratory of Bhopal; it came out to be 2.5 mg/l. Next, we did his skeletal survey which was suggestive of diffuse bony sclerosis with increased bone density, presence of osteophytes, compact bone thickening, and periosteal bone formation [Figure 4]. The patient was transferred to the neurosurgery ward for further management. He underwent partial C2–C6 laminectomy along with C3–C6 bilateral lateral mass screw rod fixation. After 7 days of surgery, we removed the foley catheter and the patient was able to void normally. Postoperatively, his limb power significantly improved (grade 4 power) and he was able to walk without support. His sensory symptoms persisted. He was discharged under stable condition after 10 days of surgery with the advice of safe drinking water.
Figure 1: MRI spine showing ossified posterior longitudinal ligament in upper cervical spine (C2–C4) along with calcification, thickening of anterior longitudinal ligament and ligamentum flavum, and multilevel bridging anterior and posterior osteophytes

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Figure 2: Thoracolumbar spine suggestive of increased bone density, fluffy osteophytes, and degenerative changes

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Figure 3: Pelvic bones showing increased bone density present with degenerative changes

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Figure 4: Skeletal survey showing diffuse bony sclerosis with increased bone density, presence of osteophytes, compact bone thickening, and periosteal bone formation

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  Discussion Top


Advanced skeletal fluorosis can progress to the point where it produces crippling deformities and neurological complications. Radiculomyelopathy, the neurological symptom of skeletal fluorosis, is caused by mechanical compression of the spinal cord and nerve roots caused by osteophytosis, a sclerosed vertebral column, and ossified ligaments. As the condition advances, vascular impairment and ischemic injury to the spinal cord and spinal roots may occur. The cervical cord is affected first in skeletal fluorosis, followed by the dorsal cord. Restricted spinal movement is frequently linked to neurological impairments. Higher cerebral function deficits or cranial nerve palsies are relatively uncommon in skeletal fluorosis. The ossification of PLL and calcification of the interosseous membrane of the forearm, a radiographic sign of fluorosis, should suggest the diagnosis of fluorosis. In our case, the spinal cord compression revealed chronic fluorosis, and the compression was caused by posterior vertebral ligament calcification in the cervical region. When the history was reviewed in our patient, he was taking drinking water from the well and pump well since childhood.

In skeletal fluorosis, spinal cord compression in the cervical region may be due to: (a) cervical canal stenosis; (b) localized calcified ligamentum flavum; (c) anterior compression by osteophytes; (d) ossification of PLL; and (e) a combination of these lesions. Ossified PLL is very common in skeletal fluorosis and was found in 37 of the 80 people over the age of 40 years in whom cervical spine skiagrams were taken in an endemic village.[5] There are case reports in which there is cervical cord compression due to ossification of the posterior longitudinal ligament at the cervical level. They have been diagnosed based on the epidemiological data associated with radiographic findings.[6] A similar case has also been reported in the village of Dhar district of Madhya Pradesh.[4] There is another case report in which a 54-year-old male developed quadriparesis due to fluorosis causing ossification of the posterior longitudinal ligament, resulting in spinal cord compression.[7]

After excluding all other possible causes of osteosclerotic bone diseases, based on history, family history, and laboratory and radiological findings, we concluded that the patient is having skeletal fluorosis which is causing cervical compressive myelopathy.

Assessment of fluoride concentrations in different districts of Madhya Pradesh showed the highest value of fluoride (14.20 ppm) in Seoni district, followed by 13.86 ppm fluoride in Jhabua district. Other districts having high fluoride levels in water are Gwalior, Shivpuri, and Vidisha. Compared with other areas, the Chhatarpur district water supply has normal fluoride content.[8] In our case, the patient was taking water from the pump well in which the fluoride content was 2.5 mg/L, which is more than the permissible limit and is the reason for fluorosis.

Wang et al.[9] studied 23 cases of skeletal fluorosis between 1993 and 2003 in China. Imaging study findings showed that all the cases have ossification of ligamentum flavum together with ossification of many other ligaments and interosseous membranes, i.e., interosseous membranes of the forearm in 18 of 23 (78.3%), the leg in 14 of 23 (60.1%), and the ribs in 11 of 23 (47.8%) patients.


  Conclusion Top


Skeletal fluorosis is a rare toxic osteopathy causing skeletal abnormalities and neurological complications depending on its severity. In India, there are states where fluorosis is not endemic still patients present with neurological complications due to skeletal fluorosis. Patients who present with chronic backache and polyarthritis should be screened during the initial stage to prevent the debilitating disability. Since it is an incurable disease and no definite treatment is available, prevention is the only way to avoid disability due to fluorosis.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given consent for his images and other clinical information to be reported in the journal. The patient understands that his names and initials will not be published, and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Ethical consideration

Obtaining the approval/clearance to undertake the proposed study on “A case report of skeletal fluorosis leading to cervical compressive myelopathy and review of the literature” has been exempted by the Institutional Ethics Committee vide their letter of June 26, 2022.

Financial support and sponsorship

None.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
World Health Organization. Water, Sanitation, Hygiene, and Health. Geneva: WHO; 2022. Available from: https://www.who.int/teams/environment-climate-change-and-health/water-sanitation-and-health/burden-of-disease/other-diseases-and-risks/fluorosis. [Last accessed on 30 September 2021].  Back to cited text no. 1
    
2.
Del Bello L Fluorosis: An ongoing challenge for India. Lancet Planet Heal 2020;4:e94-5.  Back to cited text no. 2
    
3.
Onipe T, Edokpayi JN, Odiyo JO A review on the potential sources and health implications of fluoride in groundwater of sub-Saharan Africa. J Environ Sci Health A Tox Hazard Subst Environ Eng 2020;55:1078-93.  Back to cited text no. 3
    
4.
Khairnar MR, Dodamani AS, Jadhav HC, Naik RG, Deshmukh MA Mitigation of fluorosis—A review. J Clin Diagn Res 2015;9:ZE05-9.  Back to cited text no. 4
    
5.
Reddy DR Neurology of endemic skeletal fluorosis. Neurol India 2009;57:7-12.  Back to cited text no. 5
    
6.
Gassara Z, Ben S, Feki A, Hedi M, Fourati H, Akrout R, et al. Spinal cord compression as the presenting feature of skeletal fluorosis. Authorea2021;5:1-4.  Back to cited text no. 6
    
7.
Reddy KBK, Naik RR, Vasavi B, Goud CK Case report: Fluorosis presenting as quadriparesis. IOSR-JDMS 2019;18:64-70.  Back to cited text no. 7
    
8.
Saxena KL, Sewak R Fluoride consumption in endemic villages of India and its remedial measures. Int J Eng Sci Invent 2015;4:58-73.  Back to cited text no. 8
    
9.
Wang W, Kong L, Zhao H, Dong R, Zhou J, Lu Y Thoracic myelopathy caused by ossification of ligamentum flavum of which fluorosis as an etiology factor. J Orthop Surg Res 2006;1:10.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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