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Case Report
Open Access Peer-reviewed

Lymphocytic Colitis Associated to Systemic Lupus Erythematosus in a Young Woman

Salem Bouomrani , Souad Yahyaoui, Mouna Guermazi, Nesrine Regaïeg
American Journal of Medical Case Reports. 2019, 7(2), 24-26. DOI: 10.12691/ajmcr-7-2-3
Received December 11, 2018; Revised January 14, 2019; Accepted February 17, 2019

Abstract

Introduction: Gastrointestinal symptoms are common in systemic lupus erythematosus (SLE) and are often difficult to interpret. Specific lupus involvement (lupus enteritis) typically affects the jejunum and ileum, whereas colonic and rectal involvement remains rare. Lymphocytic colitis (LC) associated with SLE remains exceptional and unusual. We report an original observation of LC occurring in a 35-year-old lupus patient. Case report: 35-year-old woman, having in her medical history a Hashimoto thyroiditis diagnosed for four years and a SLE diagnosed for three years, consulted for chronic fluid diarrhea, not improved by symptomatic treatment, and associated for two weeks with rectorrhagia. Somatic examination, basic biological tests, stool culture and parasitological stool examination were without abnormalities. Colonoscopy revealed a diffuse minimal inflammatory colitis interesting the entire colon, and the pathological examination of multiple biopsies led to the diagnosis of isolated LC. She was treated with 5-amino-salicylate with good evolution. Conclusion: Our observation is, to our knowledge the third reporting the association of LC with SLE; it is characterized in addition by the young age of occurrence. This association once again comforts the hypothesis of a common pathophysiology to these two diseases mainly involving genetic predisposition, HLA susceptibility, immune disorders, and environmental factors.

1. Introduction

Gastrointestinal symptoms are common in systemic lupus erythematosus (SLE) 1 and are often difficult to interpret: specific lesion of lupus? Infectious complication? Adverse effect of treatment? Or association with other dysimmune diseases? 1, 2.

Gastrointestinal manifestations can be objectified in up to 50% of Lupus patients 3; Specific lupus involvement (lupus enteritis, described for the first time in 1980 by Hoffman BI and Katz WA 4), however, remains exceptional and unusual 5. It typically affects the jejunum and ileum, whereas colonic and rectal involvement remains rare 5, 6. Indeed, only 11 among 288 patients with LES in the Iwamuro M et al series, had colic involvement (3.81%) 6.

The mechanism involved is usually gastrointestinal vasculitis and/or gastrointestinal microthromboses, particularly if associated antiphospholipid antibody syndrome 7, 8. The most serious complication of this lupus colitis is colonic perforation, which is often fatal 7, 8.

More recently, other types of colitis have been reported in association with SLE suggesting other pathophysiological mechanisms 9; the association SLE and lymphocytic colitis (LC) remains exceptional and unusual 10, 11.

We report an original observation of LC occurring in a 35-year-old lupus patient.

2. Case Report

35-year-old woman, having in her medical history a Hashimoto thyroiditis diagnosed for four years and a SLE diagnosed for three years.

The diagnosis of SLE with secondary antiphospholipid syndrome was retained based on the following data: distal polyarthritis without joint deformities or destruction, photosensitivity, malar erythema (vespertilio), right femoral vein thrombosis, leucopenia at 3 100/mm3, positive anti-nuclear autoantibodies at 1/640, positive native anti-NDA autoantibodies, and positive antiphospholipid autoantibodies (anti cardiolipin and anti-β2 glucoprotein1). She was treated with systemic corticosteroids, anticoagulant, and hydroxychloroquine with a favorable course. His Hashimoto's thyroiditis was stabilized with 125 μg/day of thyroxine.

She consulted for fluid diarrhea that had been evolving for two months, not improved by symptomatic treatment, and associated for two weeks with rectorrhagia.

The somatic examination was without abnormalities, the patient was particularly apyretic and without skin or joint active lesions of lupus disease. The abdomen was supple, depressible, painless on palpation, and without lymphadenopathy or visceromegaly.

The biological tests were within normal limits: erythrocyte sedimentation rate, C-reactive protein, serum protein electrophoresis, hemoglobin, leukocytes, platelets, calcemia, creatinine, ionogram, total cholesterol, triglycerides, liver enzymes, thyroid stimulating hormone, and glucose. Stool culture and parasitological stool examination were negative.

Colonoscopy revealed a colonic mucosa that was discreetly congestive and inflammatory, without ulceration, erosion, tumor lesions, diverticula or varicose veins. The appearance was that of a diffuse minimal inflammatory colitis interesting the entire colon (Figure 1).

Pathological examination of multiple biopsies led to isolated lymphocytic colitis. There were no micro-thromboses, granulomas or associated colonic vasculitis. She was treated with 5-amino-salicylate with good evolution.

3. Discussion

LC is a relatively new, rare, clinico-pathological entity that is still little known and often under-diagnosed in current medical practice. Its prevalence varies from 5.7/100,000 to 12.9/100,000 depending on the series and countries 12, 13. It integrates with collagenous colitis in the nosological framework of microscopic colitis 12, 13, 14.

The etiology of these colitis is not yet known. It appears to be multifactorial involving genetic predisposition, immune dysfunction, drug intake, infections, and environmental factors 12, 13, 14.

These colitis typically occur in the elderly, with an average age at diagnosis between 50 and 70 years 12, 14 and subjects over 65 years old appear to be 5.6 times more exposed to these colitis compared with those under 45 years of age 15.

The main associations of LC are with thyroid disorders, celiac disease, and diabetes mellitus 12, 13, 14. These associations suggest common genetic and immune factors 12, 13.

The association of microscopic colitis with SLE was reported for the first time by Desmoulins F et al in 1991 9; since only a few sporadic cases have been reported, mainly with collagenous colitis 16, 17, 18. The hypothesis of a non-hazardous association is comforted by the observation of Al-kandari A et al, reporting SLE associated with collagen gastritis in a 47-year-old woman 19.

The association of CL and SLE remains exceptional and unusual; only two observations have been reported: a patient in the series of 104 microscopic colitis of Chande N et al, 30 of whom already had autoimmune associations (0.96%) 10, and a second sporadic case reported by Hegazi MO and al 11.

The LC could thus be considered, for some authors, as a particular clinical presentation of lupus disease 11.

In addition, the presence in our observation of Hashimoto's thyroiditis is a further argument in favor of the non-hazardous association of these three diseases which shares a common dysimmunitary signature.

4. Conclusion

Our observation is, to our knowledge the third reporting the association of LC with SLE; it is characterized in addition by the young age of occurrence.

This association once again comforts the hypothesis of a common pathophysiology to these two diseases mainly involving genetic predisposition, HLA susceptibility, immune disorders, and environmental factors.

This association deserves to be well known by hospital practitioners because of its particular therapeutic and prognostic implications.

Conflicts of Interest

None.

References

[1]  Luman W, Chua KB, Cheong WK, Ng HS. Gastrointestinal manifestations of systemic lupus erythematosus. Singapore Med J. 2001; 42(8): 380-4.
In article      PubMed
 
[2]  Witt M, Zecher D, Anders HJ. Gastrointestinal manifestations associated with systemic lupus erythematosus. Eur J Med Res. 2006; 11(6): 253-60.
In article      PubMed
 
[3]  Jaspersen D. Gastrointestinal manifestations of systemic lupus erythematosus: symptoms, diagnosis and differential diagnosis. Fortschr Med. 1992; 110(10): 167-9.
In article      PubMed
 
[4]  Hoffman BI, Katz WA. The gastrointestinal manifestations of systemic lupus erythematosus: a review of the literature. Semin Arthritis Rheum. 1980; 9: 237-247.
In article      View Article
 
[5]  Smith LW, Petri M. Lupus enteritis: an uncommon manifestation of systemic lupus erythematosus. J Clin Rheumatol. 2013; 19(2): 84-6.
In article      View Article  PubMed  PubMed
 
[6]  Iwamuro M, Okada H, Kato J, Tanaka T, Sada KE, Makino H, et al. Indications for colonoscopy in patients with systemic lupus erythematosus. Hepatogastroenterology. 2013; 60(128): 1940-4.
In article      PubMed
 
[7]  Zizic TM, Shulman LE, Stevens MB. Colonic perforations in systemic lupus erythematosus. Medicine (Baltimore). 1975; 54(5): 411-26.
In article      View Article
 
[8]  Ho MS, Teh LB, Goh HS. Ischaemic colitis in systemic lupus erythematosus--report of a case and review of the literature. Ann Acad Med Singapore. 1987; 16(3): 501-3.
In article      PubMed
 
[9]  Desmoulins F, Lévy P, Boccaccio F, Thulliez M, Christoforov B. Collagenous colitis associated with systemic lupus erythematosus: first case. Gastroenterol Clin Biol. 1991; 15(4): 368.
In article      PubMed
 
[10]  Chande N, Driman DK, Reynolds RP. Collagenous colitis and lymphocytic colitis: patient characteristics and clinical presentation. Scand J Gastroenterol. 2005; 40(3): 343-7.
In article      View Article  PubMed
 
[11]  Hegazi MO, Owayed SF, Mourou M, Joneja M, Mashankar A. Lymphocytic enterocolitis in systemic lupus erythematosus. Saudi J Gastroenterol. 2009; 15(4): 274-6.
In article      View Article  PubMed  PubMed
 
[12]  Pardi DS. Diagnosis and Management of Microscopic Colitis. Am J Gastroenterol. 2017; 112(1): 78-85.
In article      View Article  PubMed
 
[13]  Chetty R, Govender D. Lymphocytic and collagenous colitis: an overview of so-called microscopic colitis. Nat Rev Gastroenterol Hepatol. 2012; 9(4): 209-18.
In article      View Article  PubMed
 
[14]  Park T, Cave D, Marshall C. Microscopic colitis: A review of etiology, treatment and refractory disease. World J Gastroenterol. 2015; 21(29): 8804-10.
In article      View Article  PubMed  PubMed
 
[15]  Williams JJ, Kaplan GG, Makhija S, Urbanski SJ, Dupre M, Panaccione R, et al. Microscopic colitis-defining incidence rates and risk factors: a population-based study. Clin Gastroenterol Hepatol. 2008; 6(1): 35-40.
In article      View Article  PubMed
 
[16]  Heckerling P, Urtubey A, Te J. Collagenous colitis and systemic lupus erythematosus. Ann Intern Med. 1995; 122(1): 71-2.
In article      View Article  PubMed
 
[17]  Bachevalier F, Lederlin P, Laugros A, Durr JF, Le Quang D. Collagenous colitis associated with systemic lupus erythematosus and circulating anticoagulant syndrome. Rev Med Interne. 1997; 18(11): 908-9.
In article      View Article
 
[18]  Alikhan M, Cummings OW, Rex D. Subtotal colectomy in a patient with collagenous colitis associated with colonic carcinoma and systemic lupus erythematosus. Am J Gastroenterol. 1997; 92(7): 1213-5.
In article      PubMed
 
[19]  Al-Kandari A, Al-Alardati H, Sayadi H, Al-Judaibi B, Mawardi M. An unusual case of collagenous gastritis in a middle- aged woman with systemic lupus erythromatosis: a case report. J Med Case Rep. 2014; 8: 278.
In article      View Article  PubMed  PubMed
 

Published with license by Science and Education Publishing, Copyright © 2019 Salem Bouomrani, Souad Yahyaoui, Mouna Guermazi and Nesrine Regaïeg

Creative CommonsThis work is licensed under a Creative Commons Attribution 4.0 International License. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/

Cite this article:

Normal Style
Salem Bouomrani, Souad Yahyaoui, Mouna Guermazi, Nesrine Regaïeg. Lymphocytic Colitis Associated to Systemic Lupus Erythematosus in a Young Woman. American Journal of Medical Case Reports. Vol. 7, No. 2, 2019, pp 24-26. http://pubs.sciepub.com/ajmcr/7/2/3
MLA Style
Bouomrani, Salem, et al. "Lymphocytic Colitis Associated to Systemic Lupus Erythematosus in a Young Woman." American Journal of Medical Case Reports 7.2 (2019): 24-26.
APA Style
Bouomrani, S. , Yahyaoui, S. , Guermazi, M. , & Regaïeg, N. (2019). Lymphocytic Colitis Associated to Systemic Lupus Erythematosus in a Young Woman. American Journal of Medical Case Reports, 7(2), 24-26.
Chicago Style
Bouomrani, Salem, Souad Yahyaoui, Mouna Guermazi, and Nesrine Regaïeg. "Lymphocytic Colitis Associated to Systemic Lupus Erythematosus in a Young Woman." American Journal of Medical Case Reports 7, no. 2 (2019): 24-26.
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  • Figure 1. Colonoscopy: moderate inflammation of the colonic mucosa at different parts of the colon (transverse, right, and left colon)
[1]  Luman W, Chua KB, Cheong WK, Ng HS. Gastrointestinal manifestations of systemic lupus erythematosus. Singapore Med J. 2001; 42(8): 380-4.
In article      PubMed
 
[2]  Witt M, Zecher D, Anders HJ. Gastrointestinal manifestations associated with systemic lupus erythematosus. Eur J Med Res. 2006; 11(6): 253-60.
In article      PubMed
 
[3]  Jaspersen D. Gastrointestinal manifestations of systemic lupus erythematosus: symptoms, diagnosis and differential diagnosis. Fortschr Med. 1992; 110(10): 167-9.
In article      PubMed
 
[4]  Hoffman BI, Katz WA. The gastrointestinal manifestations of systemic lupus erythematosus: a review of the literature. Semin Arthritis Rheum. 1980; 9: 237-247.
In article      View Article
 
[5]  Smith LW, Petri M. Lupus enteritis: an uncommon manifestation of systemic lupus erythematosus. J Clin Rheumatol. 2013; 19(2): 84-6.
In article      View Article  PubMed  PubMed
 
[6]  Iwamuro M, Okada H, Kato J, Tanaka T, Sada KE, Makino H, et al. Indications for colonoscopy in patients with systemic lupus erythematosus. Hepatogastroenterology. 2013; 60(128): 1940-4.
In article      PubMed
 
[7]  Zizic TM, Shulman LE, Stevens MB. Colonic perforations in systemic lupus erythematosus. Medicine (Baltimore). 1975; 54(5): 411-26.
In article      View Article
 
[8]  Ho MS, Teh LB, Goh HS. Ischaemic colitis in systemic lupus erythematosus--report of a case and review of the literature. Ann Acad Med Singapore. 1987; 16(3): 501-3.
In article      PubMed
 
[9]  Desmoulins F, Lévy P, Boccaccio F, Thulliez M, Christoforov B. Collagenous colitis associated with systemic lupus erythematosus: first case. Gastroenterol Clin Biol. 1991; 15(4): 368.
In article      PubMed
 
[10]  Chande N, Driman DK, Reynolds RP. Collagenous colitis and lymphocytic colitis: patient characteristics and clinical presentation. Scand J Gastroenterol. 2005; 40(3): 343-7.
In article      View Article  PubMed
 
[11]  Hegazi MO, Owayed SF, Mourou M, Joneja M, Mashankar A. Lymphocytic enterocolitis in systemic lupus erythematosus. Saudi J Gastroenterol. 2009; 15(4): 274-6.
In article      View Article  PubMed  PubMed
 
[12]  Pardi DS. Diagnosis and Management of Microscopic Colitis. Am J Gastroenterol. 2017; 112(1): 78-85.
In article      View Article  PubMed
 
[13]  Chetty R, Govender D. Lymphocytic and collagenous colitis: an overview of so-called microscopic colitis. Nat Rev Gastroenterol Hepatol. 2012; 9(4): 209-18.
In article      View Article  PubMed
 
[14]  Park T, Cave D, Marshall C. Microscopic colitis: A review of etiology, treatment and refractory disease. World J Gastroenterol. 2015; 21(29): 8804-10.
In article      View Article  PubMed  PubMed
 
[15]  Williams JJ, Kaplan GG, Makhija S, Urbanski SJ, Dupre M, Panaccione R, et al. Microscopic colitis-defining incidence rates and risk factors: a population-based study. Clin Gastroenterol Hepatol. 2008; 6(1): 35-40.
In article      View Article  PubMed
 
[16]  Heckerling P, Urtubey A, Te J. Collagenous colitis and systemic lupus erythematosus. Ann Intern Med. 1995; 122(1): 71-2.
In article      View Article  PubMed
 
[17]  Bachevalier F, Lederlin P, Laugros A, Durr JF, Le Quang D. Collagenous colitis associated with systemic lupus erythematosus and circulating anticoagulant syndrome. Rev Med Interne. 1997; 18(11): 908-9.
In article      View Article
 
[18]  Alikhan M, Cummings OW, Rex D. Subtotal colectomy in a patient with collagenous colitis associated with colonic carcinoma and systemic lupus erythematosus. Am J Gastroenterol. 1997; 92(7): 1213-5.
In article      PubMed
 
[19]  Al-Kandari A, Al-Alardati H, Sayadi H, Al-Judaibi B, Mawardi M. An unusual case of collagenous gastritis in a middle- aged woman with systemic lupus erythromatosis: a case report. J Med Case Rep. 2014; 8: 278.
In article      View Article  PubMed  PubMed