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 Table of Contents  
CASE REPORT
Year : 2023  |  Volume : 9  |  Issue : 1  |  Page : 31-34

Sciatica masquerading parathyroid carcinoma


1 Department of Speciality Surgery, Vandhana Hospital, Chennai, Tamil Nadu, India
2 Department of General Surgery, Rajiv Gandhi Government General Hospital, Chennai, Tamil Nadu, India
3 Department of General Medicine, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu, India

Date of Submission14-Jun-2022
Date of Acceptance25-Jan-2023
Date of Web Publication17-Mar-2023

Correspondence Address:
Dr. A Karthick Ramalingam
Department of General Medicine, Sree Balaji Medical College and Hospital, Chennai, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijam.ijam_53_22

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  Abstract 


Primary hyperparathyroidism is detected in the Western world in an asymptomatic stage due to routine calcium screening. In India, patients are still diagnosed with various symptoms such as bone disease and renal disease and only 5.6% are in the asymptomatic stage. A 48-year-old female came with complaints of right hip pain of 1-month duration. She had low back pain a year ago radiating to both thighs. The pain was not suggestive of inflammatory arthritis, and the autoimmune profile was negative. The pain progressively worsened, and the patient became bedridden. General examination revealed left-sided neck swelling that moved with deglutition. Right lower-limb movement was restricted by severe pain. Magnetic resonance imaging of the lumbar spine revealed a diffuse altered signal intensity involving the entire spine, bilateral pelvis, and femur with hypointense on both T1 and T2 images suggestive of marrow infiltrative disorder. Her serum calcium was 12.7 mg/dl and serum phosphorus was 1.9 mg/dl. The patient's alkaline phosphatase was 2919 IU/L. Her parathyroid hormone level was 2095 pg/ml. The patient was diagnosed to have hyperparathyroidism. 99mTc-sestamibi parathyroid single-photon emission computed tomography–computed tomography was suggestive of left superior parathyroid adenoma. The surgical endocrinologist did a left superior parathyroidectomy. Postoperatively, she developed hypocalcemia. It was managed with intravenous calcium infusion, oral calcium, and oral calcitriol. Six weeks after surgery, the pain in the right hip decreased significantly, and she was able to walk without support. Histopathological examination of the removed parathyroid was suggestive of parathyroid carcinoma.
The following core competencies are addressed in this article: Practice-based learning and improvement, Medical knowledge.

Keywords: Hyperparathyroidism, marrow infiltrative disorder, parathyroidectomy


How to cite this article:
Nellaiappar P, Hussain Z, Parvathy N A, Venkatesh N S, Ramalingam A K. Sciatica masquerading parathyroid carcinoma. Int J Acad Med 2023;9:31-4

How to cite this URL:
Nellaiappar P, Hussain Z, Parvathy N A, Venkatesh N S, Ramalingam A K. Sciatica masquerading parathyroid carcinoma. Int J Acad Med [serial online] 2023 [cited 2023 Mar 27];9:31-4. Available from: https://www.ijam-web.org/text.asp?2023/9/1/31/371890




  Introduction Top


The classical manifestations of primary hyperparathyroidism (PHPT) include generalized bone disease, kidney stones, nephrocalcinosis, gastrointestinal, cardiovascular, neuromuscular, and neuropsychiatric symptoms.[1] PHPT is detected in the Western world in an asymptomatic stage due to routine calcium screening. In India, patients are still diagnosed with various symptoms such as bone disease 77%, renal disease 36%, and asymptomatic only 5.6%. The majority of hyperparathyroidism is due to parathyroid adenoma (89.1%).[2] The patient can present with bone pain alone without fracture for a few months before developing a fracture. Here, we report one such case.


  Case Report Top


A 48-year-old female was admitted to the hospital with complaints of right hip pain of 1-month duration. The patient told that she was comfortable a year ago after which she developed pain in both the hips that were not associated with joint swelling. The patient had no history of fever, dysuria, diarrhea, rash, or photosensitivity. She had low back pain a year ago radiating to both thighs. She denied pain or swelling in any other joint. The evaluation was done in another hospital where records showed that she was negative for rheumatoid factor and antineutrophilic antibody. Serum uric acid level was found to be normal. Magnetic resonance imaging (MRI) lumbar spine was done a year ago and showed L3-L4 canal narrowing. The patient was conservatively managed at that time. The patient's bilateral hip pain progressively worsened over the next 7 months. She had a slip and fall 3 months before admission after which pain increased in her right hip. She was not able to sleep at night due to pain in her right hip. She started using calipers to walk for a month but eventually became bedridden due to pain in her right hip. The patient had no history of fever, loss of weight, or appetite. The patient had no comorbid illness. She has two children both delivered by cesarean section. She attained menopause a year ago. General examination revealed left-sided neck swelling that moved with deglutition. It was firm in consistency and without any tenderness. Her blood pressure was 130/80 mmHg, and her pulse rate was 86 beats/min. Motor system examination did not reveal any weakness except for the fact that right lower-limb movement was restricted by severe pain in the right hip. The pain was not radiating to the thigh. Her sensory system examination was normal.

The patient was provisionally diagnosed to have a right hip fracture. X-ray of the right hip did not reveal any fracture. Because of previous lumbar canal stenosis, magnetic resonance (MR) of the lumbar spine with the screening of the right hip was done. MR of the lumbar spine revealed a disc bulge at L3-L4 causing spinal canal narrowing and mild compression over the thecal sac. MR also revealed a diffuse altered signal intensity involving the entire spine, bilateral pelvis, and femur with hypointense on both T1 and T2 images suggestive of marrow infiltrative disorder. The patient's hemogram had anemia with hemoglobin of 11.5 g/dl. Her serum calcium was 12.7 mg/dl and serum phosphorus was 1.9 mg/dl. The patient's alkaline phosphatase was elevated at 2919 IU/L. The patient thyroid function test was suggestive of a euthyroid state. Because of hypercalcemia and hypophosphatemia with increased alkaline phosphatase,(intact Parathyroid Hormone) iPTH level was done. Because of MR lumbar spine report suggestive of a marrow infiltrative disorder, serum protein electrophoresis and urine for Bence Jones protein were done. Her parathyroid hormone level was 2095 pg/ml. Protein electrophoresis was normal, and urine Bence Jones protein was negative. The patient was diagnosed to have hyperparathyroidism. The patient's renal function test was normal, and hence, tertiary hyperparathyroidism was ruled out. The patient was diagnosed to have PHPT. The patient was hydrated with normal saline because of hypercalcemia along with loop diuretics and serial serum calcium level was done and her calcium levels were normalized.

99mTc-sestamibi parathyroid single-photon emission computed tomography–computed tomography was done to localize the hyperactive parathyroid gland. It was suggestive of left superior parathyroid adenoma [Figure 1] and [Figure 2]. Dual-energy X-ray absorptiometry scan was done, and it showed a total body bone mineral density (BMD) of 5.1, a right hip T score of −5.6, a left hip T score of −4.8, and a lumbar spine T score of −5.2. After evaluating for comorbid illness, correcting hypercalcemia medically, and obtaining surgical fitness, the surgical endocrinologist did left superior parathyroidectomy and was sent for histopathological examination. Postoperatively, she developed hypocalcemia. It was managed with intravenous calcium infusion, oral calcium, and oral calcitriol. Serial calcium monitoring was done, and hypocalcemia was corrected. Apart from hypocalcemia, the postoperative period was uneventful, and she was discharged on the 7th postoperative day
Figure 1: Left superior parathyroid swelling

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Figure 2: Left superior parathyroid gland hyperactivity 99mTc-sestamibi parathyroid

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Six weeks after surgery, the patient's pain in the right hip decreased significantly, and she was able to walk without support. Her Histopathological Examination (HPE) was suggestive of parathyroid carcinoma.


  Discussion Top


Parathyroid hormone maintains serum calcium within a narrow range. It has multiple effects on bone. Chronic exposure to high parathyroid hormone levels results in osteoclast-mediated bone resorption with increased fracture risk. Our patient had bilateral hip pain with low back pain along with an MRI lumbar spine that was suggestive of lumbar canal stenosis. We thought of metabolic bone disorder after she developed a debilitating unilateral right hip pain. Due to a lack of awareness and lack of routine calcium screening, delayed presentation is seen in the Indian scenario.[3] Bone disease and palpable neck mass as seen in our patient were more commonly seen in subjects with pancreatitis and PHPT than in those with milder parathyroid disease.[4] Solitary parathyroid adenoma remained the most common cause of PHPT with left inferior glands being affected more frequently as seen in another study from India.[5],[6] The higher prevalence of Vitamin D deficiency in India is correlated with large parathyroid adenoma.[7] Our patient had left superior parathyroid enlargement, and histopathology showed that it is parathyroid carcinoma which is rare. The prevalence of parathyroid carcinoma associated with PHPT is about 2%–3% in the Indian population. Parathyroid carcinoma is usually characterized by the presence of gross capsular and vascular invasion, cellular and nuclear pleomorphism, and a high mitotic index with or without lymph node metastasis.[8] BMD improves significantly after parathyroidectomy within the 1st year following surgery.[9],[10]

The patient had hypocalcemia in the postoperative period due to hungry bone syndrome. The hungry bone syndrome is a complication of parathyroidectomy. A sudden decrease in parathyroid hormone levels results in calcium uptake by demineralized bone resulting in hypocalcemia. Increased alkaline phosphatase levels, hypercalcemia, severe osteoporosis, and elevated parathyroid hormone levels are the risk factors present in our case for hungry bone syndrome. Pradeep et al. reported that, out of the 100 consecutive PHPT patients who underwent parathyroidectomy, 79 suffered early symptomatic hypocalcemia and 92 had biochemical hypocalcemia after parathyroidectomy.[11] Hypocalcemia after parathyroidectomy is milder in patients with PHPT than in patients with secondary hyperparathyroidism.[12] Patients with decreased serum Vitamin D levels are more prone to develop hypocalcemia than in patients with normal Vitamin D level following parathyroidectomy.[13]


  Conclusion Top


In patients coming with joint pain, disorders of calcium metabolism such as hyperparathyroidism and Vitamin D deficiency should also be considered a differential diagnosis apart from inflammatory and noninflammatory arthritis.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

Research quality and ethics statement

The authors attest that this case report was determined to not require the Institutional Review Board/Ethics Committee review, and the corresponding protocol/approval number is not applicable. The authors certify that they have obtained appropriate patient consent. The patient has given the consent for the clinical information to be reported in the journal.



 
  References Top

1.
Dandurand K, Ali DS, Khan AA. Primary hyperparathyroidism: A narrative review of diagnosis and medical management. J Clin Med 2021;10:1604.  Back to cited text no. 1
    
2.
Pradeep PV, Jayashree B, Mishra A, Mishra SK. Systematic review of primary hyperparathyroidism in India: The past, present, and the future trends. Int J Endocrinol 2011;2011:921814.  Back to cited text no. 2
    
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Harinarayan CV, Gupta N, Kochupillai N. Vitamin D status in primary hyperparathyroidism in India. Clin Endocrinol (Oxf) 1995;43:351-8.  Back to cited text no. 3
    
4.
Bhadada SK, Udawat HP, Bhansali A, Rana SS, Sinha SK, Bhasin DK. Chronic pancreatitis in primary hyperparathyroidism: Comparison with alcoholic and idiopathic chronic pancreatitis. J Gastroenterol Hepatol 2008;23:959-64.  Back to cited text no. 4
    
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Shah VN, Bhadada S, Bhansali A, Behera A, Mittal BR. Changes in clinical & biochemical presentations of primary hyperparathyroidism in India over a period of 20 years. Indian J Med Res 2014;139:694-9.  Back to cited text no. 5
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6.
Kapur MM, Agarwal MS, Gupta A, Misra MC, Ahuja MM. Clinical & biochemical features of primary hyperparathyroidism. Indian J Med Res 1985;81:607-12.  Back to cited text no. 6
    
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Paul TV, Thomas N, Seshadri MS, Oommen R, Jose A, Mahendri NV. Prevalence of osteoporosis in ambulatory postmenopausal women from a Semiurban region in Southern India: Relationship to calcium nutrition and vitamin D status. Endocr Pract 2008;14:665-71.  Back to cited text no. 7
    
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Bhansali A, Masoodi SR, Reddy KS, Behera A, das Radotra B, Mittal BR, et al. Primary hyperparathyroidism in North India: A description of 52 cases. Ann Saudi Med 2005;25:29-35.  Back to cited text no. 8
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Khan AA, Hanley DA, Rizzoli R, Bollerslev J, Young JE, Rejnmark L, et al. Primary hyperparathyroidism: Review and recommendations on evaluation, diagnosis, and management. A Canadian and international consensus. Osteoporos Int 2017;28:1-19.  Back to cited text no. 9
    
10.
Khedr A. Skeletal manifestations of hyperparathyroidism. In: Anatomy, Posture, Prevalence, Pain, Treatment and Interventions of Musculoskeletal Disorders: IntechOpen; 2018.  Back to cited text no. 10
    
11.
Pradeep PV, Mishra A, Agarwal G, Agarwal A, Verma AK, Mishra SK. Long-term outcome after parathyroidectomy in patients with advanced primary hyperparathyroidism and associated vitamin D deficiency. World J Surg 2008;32:829-35.  Back to cited text no. 11
    
12.
Anwar F, Abraham J, Nakshabandi A, Lee E. Treatment of hypocalcemia in hungry bone syndrome: A case report. Int J Surg Case Rep 2018;51:335-9.  Back to cited text no. 12
    
13.
Unsal IO, Calapkulu M, Sencar ME, Hepsen S, Sakiz D, Ozbek M, et al. Preoperative vitamin D levels as a predictor of transient hypocalcemia and hypoparathyroidism after parathyroidectomy. Sci Rep 2020;10:9895.  Back to cited text no. 13
    


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