A Case of Acute Biphenotype Leukemia after Diagnosis of Pulmonary Alveolar Proteinosis

Hong-Ryeol Cheong; Ha-rin Lee; Su-Bum Park; Hyo-Jeong Kim; Na-Ri-A Lee; Jae-Hoon Cheong; Mi-Ra Kim; Young-Jin Choi; Joo-Seop Chung; Goon-Jae Cho

doi:10.5045/kjh.2009.44.2.117

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Cheong, Lee, Park, Kim, Lee, Cheong, Kim, Choi, Chung, and Cho: A Case of Acute Biphenotype Leukemia after Diagnosis of Pulmonary Alveolar Proteinosis

Case Report

Korean J Hematol 2009;44(2):117-121.

Published online: 20 June 2009

DOI: https://doi.org/10.5045/kjh.2009.44.2.117

A Case of Acute Biphenotype Leukemia after Diagnosis of Pulmonary Alveolar Proteinosis

Hong-Ryeol Cheong, M.D., Ha-rin Lee, M.D., Su-Bum Park, M.D., Hyo-Jeong Kim, M.D., Na-Ri-A Lee, M.D., Jae-Hoon Cheong, M.D., Mi-Ra Kim, M.D., Young-Jin Choi, M.D., Joo-Seop Chung, M.D., Goon-Jae Cho, M.D.

Department of Internal Medicine, College of Medicine, Pusan National University, Busan, Korea

Correspondence to：Goon-Jae Cho, M.D. Department of Internal Medicine, College of Medicine, Pusan National University 1-10, Ami-dong, Seo-gu, Busan 602-739, Korea Tel: ＋82-51-240-7225, Fax: ＋82-51-254-3127 E-mail: chyj@pusan.ac.kr

Received 17 March 2009 Revised 14 May 2009 Accepted 17 May 2009

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

We present the case of a 34-year-old man with acute biphenotype leukemia that co-expressed B-lymphoid and myeloid antigen after the diagnosis of pulmonary alveolar proteinosis (PAP). The diagnosis of PAP was established by Periodic Acid-Schiff reaction staining on the Video Associated Thoracoscope guided lung biopsy and biphenotype leukemia was revealed by immunohistochemical stains of the blasts harvested from the bone marrow biopsy. Supposedly, PAP follows a hematologic malignancy, yet this case shows the reverse sequence.

Keywords: Pulmonary alveolar proteinosis, Acute biphenotype leukemia, B-lymphoid, Myeloid

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Fig. 1.

VATS lung biopsy. VATS biopsy showed many homogeneous, eosinophilic diastase-resistant PAS reactive material deposition in intra alveolar space (H&E ×400).

Fig. 2.

Computed tomography done before (A) and after (B) whole lung lavage.

Fig. 3.

BM aspiration. BM aspiration showed several undifferentiated blasts.

Fig. 4.

Cytogenetic study showed BCR/ABL1 gene rearrangement.

Fig. 5.

Plain chest X-ray and computed chest tomography at 2^nd admission. X-ray showed interval increasing extent of crazy paving appearance and ground glass opacity in both lungs.

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