Successful allogeneic bone marrow transplantation for myelodysplastic syndrome complicated by severe pulmonary alveolar proteinosis.
Pulmonary alveolar proteinosis (PAP) is a rare disorder characterized by the abnormal accumulation of alveolar surfactant protein in alveolar spaces. We report herein a rare case of myelodysplastic syndrome (MDS-RAEB) complicated by severe PAP, and successful allogeneic bone marrow transplantation (BMT) for both disorders. An unrelated BMT was planned for a 48-year-old male with advanced MDS-RAEB. Just before the initiation of the conditioning regimen for unrelated BMT in March 2007, he developed dyspnea. A diagnosis of PAP was made based on findings of chest X-ray, CT scanning, and the fluid obtained by bronchoalveolar lavage. To improve his dyspnea and improve BMT safety, whole lung lavage (WLL) was performed twice, with the partial improvement of PAP. Unrelated allogeneic BMT was performed in September 2007. We had to perform a third WLL because of the worsening of PAP on day 26 after BMT. Despite many infectious complications after BMT, GVHD was relatively mild. PAP had almost disappeared 6 months after BMT. He was well with favorable hematopoiesis 20 months after the BMT without any specific treatment. There has been no report of an MDS patient with PAP in whom 3 WLL procedures were performed before and after allogeneic BMT.
Tabata
Sumie
S
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Shimoji
Sonoko
S
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Murase
Kimihiko
K
Department of Pulmonary Medicine, Kobe City Medical Center General Hospital, Kobe, Japan.
Takiuchi
Yoko
Y
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Inoue
Daichi
D
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Kimura
Takaharu
T
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Nagai
Yuya
Y
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Mori
Minako
M
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Togami
Katsuhiro
K
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Kurata
Masayuki
M
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Ito
Kiminari
K
Department of Hematology, Institute of Biomedical Research and Innovation, Kobe, Japan.
Hashimoto
Hisako
H
Department of Hematology, Institute of Biomedical Research and Innovation, Kobe, Japan.
Matushita
Akiko
A
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Nagai
Kenichi
K
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan.
Takahashi
Takayuki
T
Department of Hematology and Clinical Immunology, Kobe City Medical Center General Hospital, 4-6 Minatojima-Nakamachi, Chuo-ku, Kobe, 650-0046, Japan. sonata53@kcgh.gr.jp.
D016026:Bone Marrow Transplantation; D018893:Bronchoalveolar Lavage; D003131:Combined Modality Therapy; D006801:Humans; D008297:Male; D008875:Middle Aged; D009190:Myelodysplastic Syndromes / Q000150:complications* / Q000628:therapy*; D011649:Pulmonary Alveolar Proteinosis / Q000150:complications* / Q000000981:diagnostic imaging / Q000628:therapy*; D012720:Severity of Illness Index; D014057:Tomography, X-Ray Computed; D014184:Transplantation, Homologous
Science. 1994 Apr 29;264(5159):713-6
8171324
N Engl J Med. 1958 Jun 5;258(23):1123-42
13552931
Bone Marrow Transplant. 2006 Oct;38(8):581-2
16953205
Mayo Clin Proc. 1987 Jun;62(6):499-518
3553760
Am J Physiol. 1996 Apr;270(4 Pt 1):L650-8
8928826
Am J Respir Crit Care Med. 2002 Jul 15;166(2):215-35
12119235
J Clin Invest. 1997 Nov 1;100(9):2211-7
9410898
Am J Respir Crit Care Med. 1994 Mar;149(3 Pt 1):788-94
8118651
Chron Respir Dis. 2006;3(3):149-59
16916009
Am J Hematol. 2002 Jun;70(2):154-7
12111790
Arch Pathol Lab Med. 2003 Feb;127(2):235-8
12562244
Blood. 1998 Aug 15;92(4):1097-103
9694696
Bone Marrow Transplant. 1997 Sep;20(6):507-10
9313886
Am J Hematol. 2006 Feb;81(2):107-9
16432866
Radiology. 1999 Apr;211(1):155-60
10189465
Am J Respir Crit Care Med. 2005 May 15;171(10):1142-9
15735059
J Exp Med. 1999 Sep 20;190(6):875-80
10499925