Saudi Journal of Gastroenterology
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CASE REPORT Table of Contents   
Year : 2001  |  Volume : 7  |  Issue : 1  |  Page : 40-41
Esophageal carcinoma presenting as seronegative symmetrical synovitis with pitting edema


College of Medicine, King Saud University & King Khalid University Hospital, Riyadh, Saudi Arabia

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Date of Submission22-Dec-1999
Date of Acceptance06-Mar-2000
 

How to cite this article:
Al-Arfaj AS. Esophageal carcinoma presenting as seronegative symmetrical synovitis with pitting edema. Saudi J Gastroenterol 2001;7:40-1

How to cite this URL:
Al-Arfaj AS. Esophageal carcinoma presenting as seronegative symmetrical synovitis with pitting edema. Saudi J Gastroenterol [serial online] 2001 [cited 2019 Oct 14];7:40-1. Available from: http://www.saudijgastro.com/text.asp?2001/7/1/40/33414


Remitting seronegative symmetrical synovitis with peripheral edema (RS, PE) was first described by McCarly in 1985 as an abrupt onset of seronegative symmetrical synovitis with peripheral edema occurring in elderly persons [1] . Other reports large number was associated with other conditions such as polymyalgia rheumatica [6] , sarcoidosis [6], vasculitis [7],[8] , hematological diseases [9] and solid followed [2],[3],[4],[5],[6] . Although some cases occurred alone, a tumors [10]. All of the solid tumors reported have been adenocarcinomas [10] . We report a case of RS, PE preceding by weeks, the discovery of squamous cell carcinoma of the esophagus. Review of literature indicates that this is the first case where such association has been described.


   Case report Top


A seventy-three year-old Saudi gentleman was admitted with three weeks history of symmetrical polyarthitis the small and large joints. He also complained of fever, sweating, and anorexia for 10 days prior to admission. He denied dysphagia, hematemesis or melena. He was started on rifampin and doxycycline elsewhere on the on the presumptive diagnosis of  Brucellosis More Details and continued on the treatment for seven days with no improvement. When admitted, he was ill, pale without jaundice or lymphadenopathy. His temperature was 39C, pulse 114, BP120/70. He had eruptive xanthomatas on face, clubbing of the fingers and moderate pitting edema of the hands and feet. His joints examination showed active symmetrical synovitis of the wrists, elbows, knees and ankles with flexor tenosynovitis of the hands and feet, and signs of old trauma to his left 5 th finger and right 1 st metatarsophalangeal joint.

Laboratory investigations showed hemoglobin of 9.1(g/cl) (range 13.0 - 18.0 g/dl) WBC 21. 1x 108/L (range 4-11lx 10 a /L), platelet 793 x 10 a /L (range 140-450 x 10 8 /L), erythrocyte sedimentation rate (ESR) of 35 mm/hr (range 0-10 mm/hr),  Brucella More Details serology repeatedly negative, Brucella blood culture negative, stool for occult blood positive. Antinuclear antibodies, rheumatoid factors and HIV antibodies - were all negative. Synovial fluid analysis showed WBC of 37250, 80% of which were neutrophils but cultures, gram stains, TB culture, TB stain, brucella culture, crystals were all negative. His serum ferritin was 1842. X- rays of his joints were normal apart from signs of old trauma of right MTP and left 5 th digit. Ultrasound of abdomen was normal. He was diagnosed as remitting seronegative symmetrical synovitis with pitting edema (RS 3 , PE) and was given one injection of methylprednisolone acetate in his right knee and started on prednisolone 7.5 mg daily, sulfasalazine 500 mg twice daily and diclofenac 50 mg twice daily and also ferrous fumarate 200 mg three times a day.

After predrisolone, his symptoms improved remarkably. While in hospital, he developed supraventricular tachycardia, which responded to verapamil 40 mg TID. He later was discharged home in a good condition. He was seen in out patient clinics six weeks later in a good condition with only mild right knee effusion. His hemoglobin at this time was 13.9 g/dl. He attended the clinic once again one month later, where his main complaint this time was anorexia and weight loss of 4 kg over the past three months. He was admitted one month later with melena, anoxia and a further weight loss of 3 kg in 1 month. His temperature was 37.4, pulse 84/min, BP 125/60. A small left cervical lymph node (1 cm diam.) was present in his left supraclavicular region. His hemoglobin dropped to 4.5 g/dl and he was transfused four units of blood. On this occasion, he agreed to undergo upper gastrointestinal endoscopy, which revealed the presence of 4x4 cm mass at gastroesophageal junction. Multiple biopsies revealed a moderately differentiated squamous cell carcinoma probably of esophageal origin. Computed tomography scan of the chest showed right paratracheal lymph node enlargement. A CT of the abdomen showed enlarged four paraotic lymphnodes. He was treated with palliative radiotherapy to his extensive esophageal carcinoma. He experienced a brief period of relief, but returned few weeks later with dysphagia and vomiting which was treated symptomatically. He died few weeks later.


   Discussion Top


This patient presented with what is called RS, PE as manifested by the abrupt onset of symmetrical arthritis accompanied by peripheral pitting edema, a negative rheumatoid factor and good response to corticosteroids as described by others [1],[2],[3] .

At presentation, in addition to his arthritis, he had fever, sweating and anorexia, which were interpreted to be brucellosis as it is common in our locality. These systemic symptoms have been described in up to 7.4% of Olive et al cases [9] . The response to steroid in this case was good initially but as the primary disorder progressed, it began to lose effect as described by others [10] . This is the first report of carcinoma of esophagus to be associated with RS 3 PE in which it also preceded the discovery of carcinoma. It also represents the first case of squamous cell carcinoma presenting as RS 3 PE as all the previously reported cases have been associated with hematological or adenocarcinomas. The prognosis of the condition depends on the primary disease, which as in this case and others is usually bad [10] .

In conclusion, RS 3 PE can be the initial presentation of more seriously hidden malignancy and if detected early and treated, may lead to good outcome as reported by Tada et al [11] . Unfortunately, the primary disease in our case was far advanced by the time of its discovery.

 
   References Top

1.McCarly DJ, O'Duffy JD, Pearson L, et al. Remitting seronegative symmetrical synovitis with pitting RS 3 PE syndrome. JAMA 1985; 254:2763-7.  Back to cited text no. 1    
2.Chaunal D, le Parc, JM. The syndrome of seronegative symmetrical synovitis with pitting edema (RS3 PE syndrome): A unique form of arthritis in the elderly? Report of four additional cases. J Rheumatol 1989; 16: 1211-3  Back to cited text no. 2    
3.Russel EB, Hunter JB, Pearson I Et al Remitting seronegative symmetrical synovitis with pitting edema. 13 additional cases. J. Rheumatol 1990; 17: 633-9.  Back to cited text no. 3    
4.Pariser KM, Canoso JJ. Remitting seronegative symmetrical synovitis with pitting edema. Two cases of RS 3 PE J Rheumatol 1991; 18: 1260-2  Back to cited text no. 4    
5.Salvarani C, Gabriel S, Hunder GG. Distal extremity swelling with pitting edemain polymyalgia rheumatica. Report of nineteen cases. Arthritis Rheum 1996; 39(1) 7380.  Back to cited text no. 5    
6.Cantini F, Niccoli L, Oliviveri I, et al. Remitting distal lower extremity swelling with pitting edemain acute sarcoidosis (letter). Ann Rheum Dis 1997; 56: 565-6.  Back to cited text no. 6    
7.Billy T, Navaux F, Lassoued S. RS, PE syndrome revealing a periarteritis nodosa (letter). Rev Rheum E Eng Ed 1995; 62:53-8.  Back to cited text no. 7    
8.Ika RW, Blairus M: Corticosteroid responsive puffy hands and occult vasculitic neuropathy. RS,PE plus? J Rheumatol 1993, 20: 205-6.  Back to cited text no. 8    
9.Olive A, Del Blanco J, Pons M, et al: Tena X and the Catalan group for the study of RS,PE. The clinical spectrum of remitting seronegative symmetrical synovitis with pitting edema. J Rheumatol 1997; 24:333-6.  Back to cited text no. 9    
10.Sibitia J, Friess S, Schaeverbeka TH et al. Remitting seronegative symmetrical synovitis with pitting edema (RS 3 PE): A form of paraneoplastic polyarthritis? J Rheumatol 1999 Jan, 26: 115-20.  Back to cited text no. 10    
11.Tada Y, Sato H, Yoshizaw S, et al. Remitting seronegative symmetrical synovitis with pitting edemaassociated with gastric carcinoma. J Rheumatol 1997;24:974-5.  Back to cited text no. 11    

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Correspondence Address:
Abdulrahman S Al-Arfaj
P.O. Box 34471, Riyadh 11468
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


PMID: 19861766

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