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CASE REPORT
Year : 2013  |  Volume : 10  |  Issue : 1  |  Page : 26-28

Association of autoimmune thyroiditis and Sjogrens syndrome: Case based review


1 Department of Endocrinology, ESI Hospital, Sanathnagar, Hyderabad, India
2 Department of Biochemistry, Prime Hospital, KPHB, India
3 Department of Endocrinology, Osmania General Hospital, Hyderabad, India

Date of Web Publication10-Jan-2013

Correspondence Address:
Babul H Reddy
Department of Endocrinology, Flat No-507, Emerald Block, My Home Jewel, Madinaguda, Hyderabad - 500 049, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0973-0354.105844

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  Abstract 

Sjogrens syndrome is a slowly progressing autoimmune disease, primarily affects exocrine glands, in addition non-exocrine glands affected as well. The coexistence of Sjogrens syndrome and autoimmune thyroiditis is frequent and suggests a common genetic or environmental factor predisposition with similar pathogenic mechanisms. We herein report three cases of this association, who presented to us with different manifestations of Sjogrens syndrome.

Keywords: Anti Ro and anti La antibodies, hypothyroidism, sjogrens syndrome


How to cite this article:
Reddy BH, Guntaka M, Nagesh SV, Ramesh J. Association of autoimmune thyroiditis and Sjogrens syndrome: Case based review. Thyroid Res Pract 2013;10:26-8

How to cite this URL:
Reddy BH, Guntaka M, Nagesh SV, Ramesh J. Association of autoimmune thyroiditis and Sjogrens syndrome: Case based review. Thyroid Res Pract [serial online] 2013 [cited 2017 Mar 23];10:26-8. Available from: http://www.thetrp.net/text.asp?2013/10/1/26/105844


  Introduction Top


Primary Sjogrens syndrome is a slowly progressing autoimmune disease characterized by lymphocytic infiltration of the exocrine glands, mainly the lacrimal and salivary glands, resulting in impaired secretory function. [1] The syndrome can be presented either alone (as primary Sjögren's syndrome) or in the context of underlying connective tissue disease (as secondary Sjögren's syndrome). An association between primary Sjogren's syndrome with autoimmune thyroiditis has been reported. [2],[3] However, controlled studies looking at this association have been few, and the results have not been uniform. [4]

We are herein present three cases of this association, who presented to us with different manifestations of Sjogrens syndrome. Then we review the literature systematically about this association.


  Case Reports Top


Case 1

A 27-year-old lady known hypothyroid for 3 years, presented to our hospital for an evaluation of her recurrent parotitis for 3 years. She was delivered 1 year back, now on 75 μg of levothyroxine. There were no symptoms of hypothyroidism. She was evaluated in the lines of mumps, parotid calculi and HIV. There was no history of fever, joint pain, skin rash, photosensitivity, dry eyes or dry mouth. There is no family history of a similar illness. O/E She had bilateral parotid swelling, presence of grade II diffuse goiter. Her vitals and systemic examination was normal. Investigations: Hemoglobin 10 g% (11-15), total leukocyte count-5,100/ (4- 11,000), Erythrocyte Sedementation Rate 25 (9- 14), Fasting blood sugar 79 mg% (80-110), sr.creatinine-0.8 mg/dl (0.8-1.4), corrected serum calcium-9.0 mg/ dl (8.4-10.4), T3- 1.2 ng/ml (0.6-2.00), T4-8.4 mcg/ml (5-12), Thyroid stimulating hormone 2.9 miU/ml (0.3-5.5), Anti Thyroid Peroxidase Ab more than 1,200, Anti nuclear antibody positive, minor salivary gland biopsy: Lymphocytic infiltration around acini and ducts suggestive of Sjogrens syndrome, Anti Ro, La antibodies-strongly positive, Shirmer test negative.

After treatment patient is doing well with no similar complaints for the past 1 year.

Case 2

A 45-year-old peri-menopausal female known hypothyroid for 8 years on regular treatment with LT4, came with the history of dryness of mouth and eyes for the past 2 years, associated with history of weight loss. No h/o photosensitivity, rash or arthralgias; O/E she had pallor, no goiter, vitals as well as systemic examination was normal. Investigations: Hemoglobin-11 g% (11-15), total leukocyte count-7,500/cmm (4-11,000), ESR-15 (9-14), FBS-87 mg% (80-110), sr.creatinine-1.0 mg/dl (0.8-1.4), T3-1.0 ng/ml (0.6-2.00), T4-6.4 mcg/ml (5-12), TSH-8.3 miU/ml (0.3-5.5). Anti TPO Ab 600, ANA positive, minor salivary gland biopsy: Lymphocytic infiltration around acini and ducts suggestive of Sjogrens syndrome, Anti Ro, La antibodies strongly positive, Shirmer test is positive. After treatment, patient is doing well with no resolution of symptoms.

Case 3

A 40-year-old female patient who is a known hypothyroid for 15 years and on LT4. (On irregular treatment) History of hypokalemic paralysis since 4 years on oral potklor solution and h/o of on and off episodes of weakness when she skips potklor solution. The fresh episode occurred on 3 rd August 2011. She was admitted in Acute Medical Care and she recovered with potklor. The past episodes used to last for less than 24 h. h/o nocturia since 4 years, h/o myalgias with difficulty in walking. No h/o renal stone disease. No history of oral ulcers, arthralgias, dryness of mouth or dry eyes. No history of laxative or diuretic usage.

On examination Body Mass Index was 16 kg/m 2 , Goitre: Grade 2, firm, non-tender, pallor was noted, Vitals were normal, Heart and lung examination normal, examination of CNS power in all groups in UL: 4/5 and power in all groups in LL: 4-/5 rest of CNS examination was normal.

Investigations: Hemoglobin-8 g% (11-15), total leukocyte count-5,800/cmm (4-11,000), ESR-28 (9-14), FBS-90 mg% (80-110), sr.creatinine-0.8 mg/dl (0.8-1.4), corrected serum calcium-9.4 mg/dl (8.4-10.4), TSH-98 miU/ml (0.3-5.5), Anti TPO Ab more than 1,200, ANA positive, Liver function test TSB: 0.8 mg/dl, SGPT: 15 U/L, Alk phosphatase: 80 u/l, ECG: U waves, Na + 135 Meq/L (135- 155), K + 2.9 Meq/L (3.5- 5.5), 24 h urinary potassium: 975 meq/day (0-15meq/day), Urine pH: 6.55, ABG: pH 7.27, PO 2 105.0 mm/Hg, PCO 2 29.9 mm/ Hg, Hco 3 15 mmol/L.

Ultra Sonogram abdomen: grade I echotexture of kidney, prominent pyramids surrounded by thin echogenic rim suggestive of Renal tubular acidosis Minor salivary gland biopsy: lymphocytic infiltration around acini and ducts suggestive of Sjogrens syndrome, Anti Ro, La antibodies-strongly positive.

Nephrology consultation was taken diagnosed as Sjogrens syndrome with Distal renal tubular acidosis advised T. POTRATE M and prednisolone. Along with 150 μg of Thyroxine patient is doing well, with no fresh episode for the past 1 year.


  Discussion Top


Primary Sjogrens syndrome is a disease of exocrine glands presenting with manifestations related to dry eyes and dry mouth. Non-exocrine organ systems may also be involved, including skin, lung, gastrointestinal tract, central and peripheral nervous system, muscular skeletal apparatus, and the kidney. [5]

The co-existence of Sjogrens syndrome and autoimmune thyroiditis is frequent and suggests a common genetic or environmental factor predisposition with similar pathogenic mechanisms. PSS was 10 times more frequent in patients with autoimmune thyroid disease, and autoimmune thyroiditis was 9 times more frequent in pSS. [6] Therefore, Sjogrens syndrome should be studied in patients with thyroid disease and vice versa.

As in our first case, pSS can present with recurrent parotitis in a subgroup of pediatric patients. [6] As Sjogrens syndrome is a progressive one, classical symptoms may follow in due course of time. Even though, there are no classical symptoms of sicca, one should suspect the possibility of this entity in underlying hypothyroidism.

Like in our second case, if the patient is present with classical features of Sjogrens syndrome, the diagnosis will be a straight forward.

As in our third case, pSS can present with RTA. The spectrum of renal disease includes interstitial nephritis, which can be manifest as distal RTA, proximal RTA, tubular proteinuria, nephrogenic diabetes insipidus, glomerular diseases, or renal failure. [7] Hypokalemia is the most common electrolyte abnormality in patients with dRTA. The mechanisms of distal RTA induced hypokalemia includes decreased distal tubular Na delivery, secondary hyperaldosteronism, defective H-K ATPase, and bicarbonaturia. [8] Even though hypokalemic paralysis is a rare complication of RTA secondary to pSS, [9] it responds very well with treatment.

Therapy includes topical agents to improve moisture and decrease inflammation. Systemic therapy includes steroidal and non-steroidal anti-inflammatory agents, disease-modifying agents and cytotoxic agents to address extraglandular manifestations involving lung, heart, kidneys, skin, hematological, nervous and lymphoproliferative disorders.


  Conclusion Top


In order to improve Quality Of Life of hypothyroid patients, high degree of suspicion about Sjogrens syndrome is needed even in the absence of classical sicca manifestations. As this association is not uncommon and responds well to treatment.

 
  References Top

1.Fox RI. Sjögren's syndrome. Lancet 2005;366:321-31.  Back to cited text no. 1
    
2.Karsh J, Pavlidis N, Weintraub BD, Moutsopoulos HM. Thyroid disease in Sjögren's syndrome. Arthritis Rheum 1980;23:1326-9.  Back to cited text no. 2
    
3.Hansen BU, Ericsson UB, Henricsson V, Larsson A, Manthorpe R, Warfvinge G. Autoimmune thyroiditis and primary Sjögren's syndrome: Clinical and laboratory evidence of the coexistence of the two diseases. Clin Exp Rheumatol 1991;9:137-41.  Back to cited text no. 3
    
4.Punzi L, Ostuni PA, Betterle C, De Sandre P, Botsios C, Gambari PF. Thyroid gland disorders in primary Sjögren's syndrome. Rev Rhum Engl Ed 1996;63:809-14.  Back to cited text no. 4
    
5.Bossini N, Savoldi S, Franceschini F, Mombelloni S, Baronio M, Cavazzana I, et al. Clinical and morphological features of kidney involvement in primary Sjögren's syndrome. Nephrol Dial Transplant 2001;16:2328-36.  Back to cited text no. 5
    
6.Baszis K, Toib D, Cooper M, French A, White A. Recurrent parotitis as a presentation of primary pediatric Sjögren syndrome. Pediatrics 2012;129:e179-82.  Back to cited text no. 6
    
7.Goules A, Masouridi S, Tzioufas AG, Ioannidis JP, Skopouli FN, Moutsopoulos HM. Clinically significant and biopsy-documented renal involvement in primary Sjögren syndrome. Medicine (Baltimore) 2000;79:241-9.  Back to cited text no. 7
    
8.Jara LJ, Navarro C, Brito-Zerón Mdel P, García-Carrasco M, Escárcega RO, Ramos-Casals M. Thyroid disease in Sjögren's syndrome. Clin Rheumatol 2007;26:1601-6.  Back to cited text no. 8
    
9.Aygen B, Dursun FE, Dogukan A, Ozercan IH, Celiker H. Hypokalemic quadriparesis associated with renal tubular acidosis in a patient with Sjögren's syndrome. Clin Nephrol 2008;69:306-9.  Back to cited text no. 9
    




 

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