Paraneoplastic relapsing polychondritis and Sweet syndrome coexisting in a patient with myelodysplasia

Calistru, Ana Maria; Lisboa, Cármen; Azevedo, Filomena

doi:10.4103/0378-6323.86505

Cited by 11 publications

(10 citation statements)

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“…There is probably a common trigger that links the two diseases. The autoimmune basis is more and more raised, but it part in the pathogenesis of paraneoplastic RP has not yet been obvious [13,[31][32][33][34]. Of note, treatment may induce remission of the two diseases.…”

Section: Check For Updatesmentioning

confidence: 99%

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2016

J Dermatol Res Ther

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Some case reports and series presumed a link between malignancy and relapsing polychondritis. The most frequent described neoplasms have been hematological disorders especially myelodysplastic syndromes. Less commonly have also been reported solid neoplasms as colorectal cancer like our patient. We suggest that patients with RP should be monitored more carefully even in remission for early detection of cancer.On admission to our department, physical examination revealed thickening of both external ears with erythema of the auricles with a saddle-nose. Fine rales over both lower lung fields were heard. Ophtalmological examination revealed bilateral anterior uveitis.Computed tomography of the chest disclosed stenosis of the trachea, swelling of cartilages and bilateral main bronchus (Figure 1).All the laboratory data was with the normal range. Antinuclear antibody was negative. Auricle biopsy showed a lymphocytic infiltration around the blood vessels, which is compatible with RP. Given that our patient has recurrent chondritis of auricles, nasal cartilage and the upper respiratory tract, recurrent uveitis, the diagnosis of RP was made and the patient was treated with Prednisone: 30 mg/day for 3 weeks and the dose of prednisone was tapered with no relapse.He was admitted 3 years after for acute rectal bleeding. The pulse rate was 110 per minute, and his blood pressure was 110/70 mmHg. He stated that he lost 10 Kg in weight 2 months before. Besides, he complained of lower abdominal pain and bloody diarrhea.Laboratory studies revealed anemia at 6.9 g/dl, the leukocyte count: 5690/mm 3 , the platelets count: 423000/mm 3 . Serum electrolytes, creatinine and liver tests were with the normal range. The stool culture was negative.Tumor markers were checked: PSA were normal but he had marked elevation of CEA, CA19-9 markers of up to 325 ng/ml. The patient was transfused. Abdominal computed tomography revealed a thickness of the descending colon wall, a liver mass on the right lobe and a necrotic adenopathy (Figure 2). Colonoscopy revealed an ulcer formation and bleeding from the sigmoid colon. Colic tumor specimen showed undifferentiated adenocarcinoma. An extended right colectomy was performed. A subsequent chemotherapy was administrated. He was discharged and still receiving Prednisone: 10 mg/day without flare-up of RP.

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confidence: 99%

“…Of note, treatment may induce remission of the two diseases. Many authors suggest that the RP and cancer may be induced by a T-cell defect [3,32,35,36]. Hall R, et al suggest that MS dendritic cells are activated and may lead to B-cell hyperplasia with abnormal antibodies production, and probable genetic error inducing neoplasm [15].…”

Section: Check For Updatesmentioning

confidence: 99%

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2016

J Dermatol Res Ther

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“…Calistru AM et al in a retrospective analysis of RP cases associated with Sweet syndrome found that 60% of cases were associated with malignancy or premalignant conditions and MDS was the leading cause occurring in 85% cases. 3 Recently Haque MA et al reported a case of RP from Bangladesh. 10 But our patient was unique in that he had associated neurological features and MDS.…”

Section: Case Reportmentioning

confidence: 99%

“…1,2 RP can be isolated or secondary to malignancy, connective tissue diseases or other autoimmune diseases. 3,4 Neurological involvement is uncommon. Here we describe a case of RP who first presented to us because of facial palsy.…”

Section: Introductionmentioning

confidence: 99%

Facial Palsy as Presenting Feature of Relapsing Polychondritis in Association with Myelodysplastic Syndrome

et al. 2015

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“…The relapsing polychondritis [1] is an acquired disease of unknown etiology, chronic course, and tendency to recur. It is characterized by inflammation and degeneration of cartilage and can result in deformities such as floppy ear and saddle nose.…”

Section: Introductionmentioning

confidence: 99%

Relapsing Polychondritis Associated with Myelodysplastic Syndrome

Mahfoudhi¹,

Mamlouk²,

Rym³

et al. 2015

OJCD

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A 62-year-old man had arthritis and an infiltrated purpuric eruption of the legs. He presented then a cyanosis and aches of the left ear's pinna with a voice disturbance and an inflammatory aspect of the nose. He had an anemia and a thrombopenia. The infectious and auto-immune research was negative. The laryngo-tracheal scan revealed calcifications of the larynx, the trachea and the initial portion of bronchi. Moreover, the biopsy from ear's pinna cartilage confirmed the diagnosis of chondritis and peri-chondritis. He was thus considered to have the association with relapsing polychondritis and a myelodysplastic syndrome. The patient was treated with corticosteroids and iterative transfusions. The articular and skinny patient's symptoms completely resolved. The anemia and the thrombopenia were partially improved. The relapsing polychondritis can be considered as paraneoplastic complication in the context of a myelodysplastic syndrome or as a fortuitous association. The mechanism of such an association remains unknown.

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Paraneoplastic relapsing polychondritis and Sweet syndrome coexisting in a patient with myelodysplasia

Cited by 11 publications

References 4 publications

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Facial Palsy as Presenting Feature of Relapsing Polychondritis in Association with Myelodysplastic Syndrome

Relapsing Polychondritis Associated with Myelodysplastic Syndrome

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