TY - GEN
T1 - Work related acute leukemia and mucor mycosis in a boat-builder
AU - Magnavita, Nicola
AU - Placentino, Roberta Anna
AU - Chiusolo, Patrizia
AU - Fiorini, Alessia
AU - Laurenti, Luca
AU - Sica, Simona
PY - 2002
Y1 - 2002
N2 - mcreased risks for leukemia and lymphoma have been suggested in studies of workers exposed to styrene in the rubber and
plastics industry.l Reportlng of anecdotal cases, with complete occupational histories, might contribute to the recognition of
new causes of leukemia.
In the August 1994 a 47-years-old caucasian man, boat-builder, was admitted to our Division of Hematology complaining of
weakness, fever, purpura, and gingival hypertrophia. He had been exposed for 21 years to styrene in fiber-reinforced plastic
shipyards as /aminator. He sprayed styrene with a gun on ship hull, provided with half-facepiece air-purifying or full-face airsupplied
respirators which he shared with his co-workers. Hematologic examination showed severe leucocitosis 80x109/L
(blasts 29%), severe anemia (Hb 8.4 g/dL), and thrombocytopenia (PLTs 38x109/L). Bone marrow aspirate showed 65%
blasts and 30% of monocyte cells. Blasts were of medium size with loose chromatin, thin cytoplasmatic granulations
peroxidase-positive and sometimes with single Auer roads. Flow cytometric immunophenotyping demonstrated expression of
HLA-DR, CD13, CD33, CD11b, CD14. Cytogenetic analysis showed a normal male kariotype as 46XY. A diagnosis of
Myelomonocitic Acute Leukemia (FAB M4) was made. A complete remission was induced by standard chemoterapy (ICE
protocol). After chemoterapy the patient showed sepsis by Pseudomonas aeruginosa successfully treated with antibiotic
therapy. During aplastic period, at the left maxillary bone occurred massive necrosis of mucosa and bone. Mucosa biopsy
identified hyphae of Mucormycosis. The diagnosis was confirmed by hystological examination of mascellar and alveolar bones
during maxillary surgery. Fungal infections are frequently seen in immunosuppressed patients with neutropenia or prolonged
impaired T-celi function, such as bone marrow transplant recipients. Aside from common Candida and Aspergillus species,
rare fungi like Mucor may be observed. Anecdotal cases of mucormycosis pneumonia are reported in patients with
hematological neoplasms and iatrogenic immunosuppression after treatment.2-4 Mucor hyphae.were later demonstrated in the
filter of the mask that he shared with his co-workers. Microbial growth o n respirator filters from improper storage In humid
environments has been reported.s Our patie.nt, however, was the sole case of mucormycosis in the boat building factory.
Antimycotical therapy with Amphotericine-B (total dose 1.34 g) was started with resolution of mycotical infection after 2
months. In the October 1995, persisting complete remission and resolution of mycotical infection, he underwent to
consolidation chemotherapy. After 4 months the patient was in complete remission, so that explantation of bone marrow
followed by autologous bone-marrow transplantation was done. Actually the patient is in continuous complete remission.
Severa! recent studies of the reinforced plastics industry, where high exposure to styrene occurs, have suggested that
workers exposed to styrene have increased mortality from lymphatic and hematopoietic cancer. A historical cohort study
conducted in Denmark, Finland, Italy, Norway, Sweden and the United Kingdom involving >40,000 workers suggested an
association between hematological malignancies and time elapsed since first occupational exposure to styrene, and a two-fold
risk 20 years after first exposure, even lf mortality from the lymphatic and haematopoietic tlssues was non elevated,6 A
Danish nested case-referent study found a 2.5-fold increased risk for myeloid leukemia wlth clonai chromosome aberrations
among workers wlth styrene exposure.7 These results do not exclude the posslbility that styrene causes leukemia. Within the
boat-making industry, hu/1/amination job ranks higher in exposure to styrene than other jobs, and frecjuently exceeds the
NIOSH-recommended time-weighted averl:lge standard (50 ppm).B Evidence of exposure, and epidemiologica! data support
AB - mcreased risks for leukemia and lymphoma have been suggested in studies of workers exposed to styrene in the rubber and
plastics industry.l Reportlng of anecdotal cases, with complete occupational histories, might contribute to the recognition of
new causes of leukemia.
In the August 1994 a 47-years-old caucasian man, boat-builder, was admitted to our Division of Hematology complaining of
weakness, fever, purpura, and gingival hypertrophia. He had been exposed for 21 years to styrene in fiber-reinforced plastic
shipyards as /aminator. He sprayed styrene with a gun on ship hull, provided with half-facepiece air-purifying or full-face airsupplied
respirators which he shared with his co-workers. Hematologic examination showed severe leucocitosis 80x109/L
(blasts 29%), severe anemia (Hb 8.4 g/dL), and thrombocytopenia (PLTs 38x109/L). Bone marrow aspirate showed 65%
blasts and 30% of monocyte cells. Blasts were of medium size with loose chromatin, thin cytoplasmatic granulations
peroxidase-positive and sometimes with single Auer roads. Flow cytometric immunophenotyping demonstrated expression of
HLA-DR, CD13, CD33, CD11b, CD14. Cytogenetic analysis showed a normal male kariotype as 46XY. A diagnosis of
Myelomonocitic Acute Leukemia (FAB M4) was made. A complete remission was induced by standard chemoterapy (ICE
protocol). After chemoterapy the patient showed sepsis by Pseudomonas aeruginosa successfully treated with antibiotic
therapy. During aplastic period, at the left maxillary bone occurred massive necrosis of mucosa and bone. Mucosa biopsy
identified hyphae of Mucormycosis. The diagnosis was confirmed by hystological examination of mascellar and alveolar bones
during maxillary surgery. Fungal infections are frequently seen in immunosuppressed patients with neutropenia or prolonged
impaired T-celi function, such as bone marrow transplant recipients. Aside from common Candida and Aspergillus species,
rare fungi like Mucor may be observed. Anecdotal cases of mucormycosis pneumonia are reported in patients with
hematological neoplasms and iatrogenic immunosuppression after treatment.2-4 Mucor hyphae.were later demonstrated in the
filter of the mask that he shared with his co-workers. Microbial growth o n respirator filters from improper storage In humid
environments has been reported.s Our patie.nt, however, was the sole case of mucormycosis in the boat building factory.
Antimycotical therapy with Amphotericine-B (total dose 1.34 g) was started with resolution of mycotical infection after 2
months. In the October 1995, persisting complete remission and resolution of mycotical infection, he underwent to
consolidation chemotherapy. After 4 months the patient was in complete remission, so that explantation of bone marrow
followed by autologous bone-marrow transplantation was done. Actually the patient is in continuous complete remission.
Severa! recent studies of the reinforced plastics industry, where high exposure to styrene occurs, have suggested that
workers exposed to styrene have increased mortality from lymphatic and hematopoietic cancer. A historical cohort study
conducted in Denmark, Finland, Italy, Norway, Sweden and the United Kingdom involving >40,000 workers suggested an
association between hematological malignancies and time elapsed since first occupational exposure to styrene, and a two-fold
risk 20 years after first exposure, even lf mortality from the lymphatic and haematopoietic tlssues was non elevated,6 A
Danish nested case-referent study found a 2.5-fold increased risk for myeloid leukemia wlth clonai chromosome aberrations
among workers wlth styrene exposure.7 These results do not exclude the posslbility that styrene causes leukemia. Within the
boat-making industry, hu/1/amination job ranks higher in exposure to styrene than other jobs, and frecjuently exceeds the
NIOSH-recommended time-weighted averl:lge standard (50 ppm).B Evidence of exposure, and epidemiologica! data support
KW - leukemia
KW - mycosis
KW - occupational cancer
KW - leukemia
KW - mycosis
KW - occupational cancer
UR - http://hdl.handle.net/10807/111573
M3 - Other contribution
ER -