Severe inflammation may be caused by hyperferritinemia of pseudo-pseudo Meigs' syndrome in lupus patients: two cases rep

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CASE BASED REVIEW

Severe inflammation may be caused by hyperferritinemia of pseudo-pseudo Meigs' syndrome in lupus patients: two cases reports and a literature review Sang Yeob Lee & Sung Won Lee & Won Tae Chung

Received: 27 June 2013 / Accepted: 31 July 2013 / Published online: 20 August 2013 # Clinical Rheumatology 2013

Abstract Pseudo-pseudo Meigs' syndrome or Tjalma syndrome is a rare manifestation of patients with systemic lupus erythematosus defined by the presence of ascites, pleural effusions, and an elevated cancer antigen 125 level. In this case, we described a patient with lupus who presented with sudden impaired renal function and subsequent development of massive ascites with marked high serum ferritin. Hyperferritinemia is one of the abnormal laboratory findings in severe inflammation with ferritin functioning as an inflammatory marker. However, its correlation with lupus activity remains unclear. Therefore, a review of the literature regarding pseudo-pseudo Meigs' syndrome associated with lupus and high ferritin level in this disease was carried out. Keywords CA-125 . Lupus . Meigs' syndrome

as PPMS [5]. Ural et al. described a patient presenting with a photosensitive rash, detectable double-stranded DNA (dsDNA), and massive ascites along with an elevated serum CA-125 level; she was diagnosed with PPMS secondary to SLE [6]. Bes and Soy also described a case of a patient with lupus manifested by hydrothorax, ascites, and an elevated serum CA-125 but it is unclear to what degree drug therapy contributed to her symptom complex [7]. In this paper, we aim to present two patients, diagnosed as SLE, who had high serum ferritin which was detected during an anemia workup; PPMS was diagnosed in these patients subsequently. PPMS completely resolved in these patients with high-dose steroid therapy. These cases show high serum ferritin that results from severe inflammation; severe inflammation may be the cause of PPMS in lupus. We also discuss two similar cases reported in the literature and review the clinical features of PPMS of lupus.

Introduction Pseudo-pseudo Meigs' syndrome (PPMS) or Tjalma syndrome is a rare manifestation of patients with systemic lupus erythematosus (SLE), defined by the presence of ascites, pleural effusions, and an elevated cancer antigen 125 (CA125) level. The triad of hydrothorax, ascites, and a benign ovarian fibroma is known in the USA as Meigs' syndrome [1]. By contrast, pseudo-Meigs' syndrome refers to pleural effusion, ascites, and a malignant tumor either an ovarian teratoma or a uterine leiomyoma [2, 3]. Tjalma described the first case report of a lupus patient with ascites, hydrothorax, and an elevated CA-125 [4]. In 2005, Schmitt et al. published a case report of a patient with similar features identifying this entity S. Y. Lee : S. W. Lee : W. T. Chung (*) Department of Internal Medicine and Rheumatology, Dong-A University Hospital, 3 Ga 1, Dong-daeshin dong, Seo-Gu, Busan, South Korea e-mail: [email protected]

Case 1 A 29-year-old female patient was admitted to our depa