404
￭ S-721 ￭
Primary Pyoderma Gangrenosum with ARDS and Multiple Hepatosplenic Abscess
대구파티마병원, 류마티스 내과
*
이유진, 김건우, 한승우
Pyoderma gangrenosum (PG) is a rare neutrophilic ulcerative skin disease of unknown etiology. The most common presentation of PG is an in- flammatory papulo-pustular skin lesion that progresses to a painful ulcer and is able to involve almost every organ. We present a case of acute respira- tory distress syndrome (ARDS)-like pulmonary involvement in primary PG with multiple hepatosplenic abscess. A 85-year-old woman presented with ulcerative skin lesions at both lower legs and right arm was diagnosed as PG with aseptic hepato-splenic abscess (Figure 1, 2) and took corticosteroid and azathioprine, which showed a good response. Eight months later, she was admitted complaining of a short of breath. She had hypertension, angina pectoris, and type 2 diabetes mellitus. Based on the decreased oxygenation of Pao2/Fio2 below 200 mmHg and bilateral infiltration on chest radiograph, she was diagnosed as ARDS by severe bilateral pneumonia in immunocompromised host (Figure 2A). Although intravenous piperacillin/tazobactam and ciprofloxacin was administered, her symptom and radiograph has worsened (Figure 2B). The poor response to antibiotics, repeatedly negative cul- ture results and newly developed hepatic abscess lead us the possibility of pulmonary involvement of PG. We administered a steroid pulse therapy and consequent high-dose steroid, and then her symptom and radiograph improved dramatically (Figure 2C). The dysregulation of the immune system, es- pecially uncontrolled activation of neutrophils may contribute to PG and the pulmonary involvement can be a consequence of an alveolar injury which causes release of pro-inflammatory cytokines. When a patient has a neutrophil-dominant ulcerative non-infectious skin lesion, the possibility of PG should be one of the list of differential diagnosis and keep an eye on the presence of systemic involvement including pulmonary and hepato-splenic lesion.
￭ S-722 ￭
Development of IgA nephropathy in a Patient Receiving Infliximab for Crohn's Disease
성균관대학교 의과대학 내과학교실1, 영상의학교실2, 한양대학교 의과대학 병리학교실3
*
유태경
1, 박동일
1, 이규백
1, 권헌주
2, 정유민
3, 권유현
1, 안중경
1Infliximab is a chimeric tumor necrosis factorα inhibitor that is approved for the treatment of Crohn disease, psoriasis, and rheumatoid arthritis.Various complications associated with TNFα inhibitor have been reported. We report a case of IgA nephropathy developed after treatment with infliximab in a patient with Crohn’s disease. A 28 year-old female patient visited our hospital for gross hematuria during treatment with infliximab. She did not have any kidney disease at the time of the diagnosis of Crohn's disease. There was no interval change of extent of Crohn’s disease on colonoscopy and Computed tomography.. (Figure 1A: small round ulcer in terminal ileum, 1B: deformity and mild stricture due to ulcer scar on the terminal ileum,Figure 2A : CT performed this year, 2B: CT performed 2 years ago). She was diagnosed with IgA nephropathy at renal biopsy.(Figure 3A: Mild mesangial en- largement due to increase of cells and matrix with hyaline nodules, consistent with IgA nephropathy PAS,x400 3B: The interstitum showing mild fib- rosis and edema with infiltration of lymphocytes. PAS, ×200, 3C: Portion of a glomerulus from a patient showing electron dense mesangial depos- its(arrows). EM, ×8000, 3D: Immunofluorescence preparation of a glomerulus demonstrating mesangial deposits of IgA.) Her symptom is getting better after infliximab cessation, without steroid treatment. To our knowledge, this is the first case of IgA nephropathy caused by infliximab therapy in a pa- tient with Crohn’s disease in our country.
Table 1. Comparison of previously reported cases of IgA nephropathy induced by infliximab treatment
Case 1 [1] Case 2 [2] Case 3 [3] Case 4 [4] Case
Age/Sex 56/M 61/M 60/M 38/M 28/F
Underlying disease Psoriasis Psoriasis Psoriasis Crohn’s disease Crohn’s disease
Drug Infliximab Adalimumab Infliximab Infliximab Infliximab
Duration of drug use 10month 18 month 12 month 24 month 60 month
Treatment for renal disease
Cessation of infliximab ACE inhibitor, salt-free diet, ustekinumab (for psoriasis, unknown effect for nephrop- athy), no steroid treatment.
Cessation of adalimumab, prednisolone 3weeks (initial 1mg/kg, tapered
10mg/week).
Cessation of in- fliximab,
deflazacort 9mg/day.
Glucocorticoids (dose unknown).
ACE inhibitors, fish oil.
Cessation of in- fliximab,
no steroid treatment.
Time to recovery 15month 3weeks 3weeks 6month 3month
ACE, angiotensin converting enzyme
1. Nicolas Kluger MD AD-TM, Didier Bessis MD, Marie-Francoise Servel MD, Georges Mourad MD. Psoriasis-associated IgA nephropathy under infliximab therapy. International Journal of Dermatology 2014;54:e79-e80.
2. Sinniah SSWaR. Adalimumab (TNF-α Inhibitor) Therapy Exacerbates IgA Glomerulonephritis Acute Renal Injury and Induces Lupus Autoantibodies in a Psoriasis Patient. Case Reports in Nephrology 2013;2013:4.
3. Dae Hong Kim MD, Ji Hyun Lee, M.D., Tae Yoon Kim, M.D. Development of Renal Disorder in a Patient Receiving Infliximab (Remicade ®) for Psoriasis. Korean Journal of Dermatology 2014;52:494~497.
4. Lisbeth Selby MD, Cynthia Harris, M.D., Willem deVilliers, M.D. Crohn's disease, infliximab treatment and IgA nephropathy. The American Journal of Gastroenterology 2003;98:S199.
