Case Report
A 28-year-old young lady with ulcerative colitis since
2019, diagnosed based on colonoscopic appearance and
biopsy and maintained on mesalazine 1 g twice daily.
She had also a cerebral venous thrombus two years ago
. The patient had been in remission for 3 years. She
presented to the emergency department with
progressively constitutional symptoms as fever and
asthenia, diffuse myalgias with Symmetric proximal
muscle weakness and non erosive polyarthritis for the
last three weeks but no cutaneous lesions also no
diarrhea corresponding to “mild” clinical ulcerative
colitis in the classification of Truelove and Witts.
Laboratory tests showed the following: white cell count
of 1200/ mm3 and hemoglobin of 9.5 g/dl, platelets count
of 181,000/ mm3. sedimentation rate of 73 mm/hr and
CRP, ALT,ALP, renal tests, electrolytes and Thyroid test
were normal range. AST, CPK were mildly elevated .
Hepatitis serologies, HIV serology, Urine culture and
Blood cultures were all negative.
Patient had an ECG , US Doppler of lower extremities
and CT Scan which were reported as normal. Her
colonoscopy was found in endoscopic remission.
Autoimmune serologies revealed ANA homogeneous
pattern with titer of 1:640 ,anti nRNP (1:25) and anti Ku
(1:102) were positive.
EMG of the quadriceps and deltoid muscles revealed
abundant low-amplitude and short-duration potentials
indicating a myopathy.
Left lower leg muscle biopsy showed the presence of
macrophages and activated lymphocytes cells in muscle
fibers with mononuclear cell invasion around non-
necrotic muscle fibers in endomysial areas without
vacuolated, necrotic fibers.
During the course of her hospitalization, she was
started on prednisone (1 mg/kg/day) for 4 to 8 weeks
which dramatically improved her symptoms, followed by
slow taper to low dose prednisone (5 to 10 mg) over 6
months; subsequent attempts are expected to wean off
prednisone within 12 months. Her treatment was
combined with azathioprine (2 mg/kg/day) to reduce the
side effects of the glucocorticoid and to boost the
immunosuppressive effect. She was followed up and her
disease was in remission for almost a year.
Discussion
UC is a chronic systemic inflammatory condition
primarily involving the mucosa of the colon associated
with relapsing and remitting episodes. It is also
associated with multiple extra-intestinal manifestations
with prevalence of approximately 25% during the
course of the disease [1, 2]. Polymyositis is a
particularly rare EIM of UC. The earliest case of
myositis associated with UC was reported by Oshitani
et al [5].
The review of literature shows only a few cases
describing an association of ulcerative colitis and
inflammatory myositis [1, 4], most of them during the
acute relapse of the disease, but the activity of bowel
disease does not seem to be essential for occurrence
or progression of myositis [2]. In addition, the interval
from diagnosis of IBD and occurrence of myositis was
considered. It was found that the majority of patients
developed myositis 5 years to 10 years after the onset
of IBD. In our case report, the interval was 3 years.
We report a case of polymyositis according to criteria
of Bohan and Peter, in a young patient who was in
remission from UC on long-term with mesalamine. The
diagnosis of myositis is usually based on the presence
of symptoms typical of the disease and positivity of
two out of three assessments: muscular histology,
EMG and serum markers of myolysis such as CPK,
LDH. Some authors have highlighted the usefulness of
magnetic resonance imaging (MRI) [3].
Myositis can be either a direct consequence of disease
activity of the bowel (e.g., rhabdomyolysis, electrolyte
imbalance, and dehydration) or be an immune-
mediated response as in our patient [3, 4].
Understanding the pathogenesis of immune-mediated
EIM can help with early recognition and adequate and
aggressive management, which will contribute to
complete recovery and faster remission [4].
Treatment of myositis is usually based on the oral
administration of steroids, either alone or in
association with immunosuppressants such as
azathioprine, methotrexate, the efficacy of these
agents in the treatment of UC supports the hypothesis
of a common pathogenetic mechanism of myositis and
IBD. The use of biological agents has significantly
changed the management of myositis in IBD patients
resistant to conventional treatment [2, 3]. Nonetheless,
in our case such treatment has not been used yet.
Conclusion:
This case report indicated that the diagnosis of
polymyositis should be considered in ulcerative
colitis patients complaining of myalgia or muscular
weakness. This disease activity is not necessary for
the onset of myositis. Furthermore, early recognition
of these extra-intestinal manifestations should be
helpful in guiding therapy that will reduce overall
morbidity in affected patients. Both diseases
responded well to corticosteroids and azathioprine.
27 ÈME CONGRÈS NATIONAL DE LA SAMI &
10 ÈME CONGRÈS FRANCO-MAGHRÉBIN DE MÉDECINE INTERNE
References
1- Polymyositis of the skeletal muscles as an extraintestinal complication in quiescent ulcerative colitis. Voigt E, Griga T, Tromm A, Int J Colorectal Dis. 1999 Dec
2- Recurrent Inflammatory Myositis as an Extra-Intestinal Manifestation of Dormant Ulcerative Colitis in a Patient on Long-Term Mesalamine. Naramala S, Konala V Case Rep Gastrointest Med.
3-Occurrence of myositis in a patient with ulcerative colitis and Hashimoto's thyroiditis--case report and review of the literature. Paoluzi OA, Int J Colorectal Dis. 2006 Jul
4-Dermatomyositis: A Rare Extra-intestinal Manifestation of Ulcerative Colitis Chang Hyun Park et al Journal of Rheumatic Diseases Vol. 23, No. 3, June, 2016
5- A case of dermatomyositis followed by multiple mononeuritis with the history of ulcerative colitis and Basedow's disease Oshitani H et al . J Kyorin Med Soc 1981;
Introduction
Ulcerative colitis is a chronic systemic large bowel
inflammatory condition with an incidence of 2.2 to 14.3
cases per 100,000 person-years [1]. Extra-intestinal
manifestation accounts for 25% of patients with
inflammatory bowel disease, also their occurrence
depends on disease activity and a combination of
factors [2]. The review of literature reported only a few
cases of inflammatory myopathy in association with UC.
We report an unusual presentation of polymyositis in a
young patient with quiescent UC on long-term
mesalamine therapy.
Keywords: inflammatory bowel disease (IBD), Ulcerative Colitis,
Extraintestinal manifestation (EIM), polymyositis
Polymyositis as an Extra-Intestinal Manifestation of Ulcerative
Colitis in a young patient on Long-Term Mesalamine
D. Merdaci M.Benkaouha I.Maalam D.Azizi
Department of Internal Medicine kais hospital