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Pleural mesothelioma in a couple of brothers.
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MedLine Citation:
PMID:  24872671     Owner:  NLM     Status:  PubMed-not-MEDLINE    
Abstract/OtherAbstract:
Malignant mesotheliomas of the pleura, epithelial type, were observed in two brothers. Both the patients had histories of severe exposure to asbestos, having worked as insulators. The latency periods in the two cases were 26 and 38 years, respectively. Available literature data suggest that mesothelioma occurrence among blood-related people is favored by a genetic predisposition.
Authors:
Claudio Bianchi; Tommaso Bianchi
Publication Detail:
Type:  Journal Article    
Journal Detail:
Title:  Indian journal of occupational and environmental medicine     Volume:  17     ISSN:  0973-2284     ISO Abbreviation:  Indian J Occup Environ Med     Publication Date:  2013 Sep 
Date Detail:
Created Date:  2014-05-29     Completed Date:  2014-05-29     Revised Date:  2014-06-02    
Medline Journal Info:
Nlm Unique ID:  9815775     Medline TA:  Indian J Occup Environ Med     Country:  India    
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Languages:  eng     Pagination:  122-3     Citation Subset:  -    
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Journal Information
Journal ID (nlm-ta): Indian J Occup Environ Med
Journal ID (iso-abbrev): Indian J Occup Environ Med
Journal ID (publisher-id): IJOEM
ISSN: 0973-2284
ISSN: 1998-3670
Publisher: Medknow Publications & Media Pvt Ltd, India
Article Information
Copyright: © Indian Journal of Occupational and Environmental Medicine
open-access:
Print publication date: Season: Sep-Dec Year: 2013
Volume: 17 Issue: 3
First Page: 122 Last Page: 123
PubMed Id: 24872671
ID: 4035608
Publisher Id: IJOEM-17-122
DOI: 10.4103/0019-5278.130865

Pleural mesothelioma in a couple of brothers
Claudio Bianchiaff1
Tommaso Bianchiaff1
Center for the Study of Environmental Cancer, Italian League against Cancer, Monfalcone, Italy
Correspondence: For Correspondence: Dr. Claudio Bianchi, Center for the Study of Environmental Cancer, Italian League against Cancer, Hospital of Monfalcone, 34074 Monfalcone, Italy. E-mail: legatumori1@interfree.it

INTRODUCTION

A number of reports have been devoted to familial mesothelioma.[1, 2, 3, 4, 5] The meaning of such an occurrence remains controversial. The recent International Agency for Research on Cancer (IARC) monograph on asbestos suggests that familial mesothelioma is a rather rare event.[6] However, various data are at odds with such view. We describe a couple of brothers with pleural mesothelioma. Both the patients had histories of severe occupational exposure to asbestos.


CASE REPORT
Case 1

A 45-year-old man was admitted to a hospital in central Italy in August 2000 for right pleural effusion. Three-months later, a diagnosis of malignant mesothelioma, epithelial type, was made on the basis of multiple pleural biopsies obtained at right thoracotomy. Hyaline pleural plaques were also visible. Partial right pleurectomy was performed on December 2000. In the following years, the patient was treated by chemotherapy. In May 2003, a progression of the disease was documented. Involvement of spinal cord and large subcutaneous neoplastic infiltration coexisted. The patient died in June 2003.

In the period 1974-1981, the patient had intermittently worked as an insulator at a sodium carbonate factory for a total of about 3 years. Moreover, in the period 1977-1986, he was intermittently employed in various sugar refineries for a total of nearly 3 years.

Case 2

A 56-year-old man, the brother of the Case 1, was admitted to a hospital in northern Italy in March 2006. A thoracic computed tomography (CT) showed two nodules adjacent to pleura in the right lung. One year previously the patient had been treated with oral anticoagulants for pulmonary thromboembolism. At the right videothoracoscopy, multiple neoplastic pleural nodules were visible, associated with hyaline pleural plaques. At the histological examination, malignant mesothelioma, epithelial type, was diagnosed. Despite chemotherapy with cysplatin and alimta, thoracic CT showed severe progression of the neoplasia. In March 2007, the patient was treated with mediastinal radiotherapy for a mediastinal syndrome. He died in April 2007.

The patient had worked as an insulator for some 20 years (1967-1986). His work took place in thermoelectric plants and chemical industries in Italy as well as in foreign countries (Algeria, Libya, Saudi Arabia).

The two patients were born in central Italy. Their parents came from a town of Sicily; some degree of blood relationship existed between the parents, they being second cousins.


DISCUSSION

Both the aforementioned patients had severe exposure to asbestos, having worked as insulators. The exposure was different in duration (about 6 years in the case 1 and 20 years in the case 2). Latency periods also differed markedly, being unexpectedly short in the case 1 (26 years) and longer in the case 2 (38 years). In the case 2, the pulmonary thomboembolism presented by the patient in 2005 may be considered as the first manifestation of the tumor, given the low frequency of thromboembolism in the general population and the high incidence in mesotheliomas.[7]

The high incidence of malignant mesothelioma among insulators has been documented by many investigations.[8, 9, 10, 11, 12] There is no doubt that the two cases have to be considered as induced by asbestos. Nevertheless, the fact that only a relatively low percentage of people exposed to asbestos develop mesothelioma indicates that cofactors play a role in the genesis of asbestos-related mesothelioma.[13, 14] Mesotheliomas arising in blood-related people suggest a genetic predisposition to develop this rare malignancy. It is difficult to form an idea about the frequency of such an occurrence. In a large majority of the familial mesotheliomas reported in the literature, there is no reference to a denominator. In two studies conducted in Italy a denominator is reported, with different findings. In a series of 610 pleural mesotheliomas examined in the Trieste-Monfalcone area, Italy, the percentage of familial cases with blood relationship reached the value of 3.8%.[1] A percentage somewhat lower, 22 cases in a series of 1,954 pleural mesotheliomas, was observed by examining the data collected by three Italian Mesothelioma Registries.[2]

Recently, de Klerk et al., have investigated the familial aggregation of malignant mesothelioma in former workers and residents of Wittenoom, Western Australia.[5] More than 11,000 people have been followed. The researchers observed 369 family groups with at least one mesothelioma case and 25 cases of mesothelioma among relatives in the same families. The risk ratio for blood relatives was 1.9 (CI 1.3-2.9). After the authors, these findings suggest an important, but not large, genetic component in mesothelioma. In a study regarding 1,403 people hired at the shipyards of Monfalcone in the period 1950-59, 35 cases of mesothelioma were observed.[15] Of the 33 men with pleural mesothelioma in this group, four had a blood relative with the same tumor.


CONCLUSIONS

On the whole, the aforementioned data show that the occurrence of more mesothelioma cases among blood-related persons is not rare. Such occurrence may indicate a genetic predisposition to develop mesothelioma, even if asbestos exposure remains the principal cause.


Notes

Source of Support: Nil

Conflict of Interest: One of the authors (CB) has been asked to provide scientific information in criminal or civil court cases relates to asbestos diseases; he served as an expert for the court or for the plaintiff.

REFERENCES
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2. Ascoli V,Cavone D,Merler E,Barbieri PG,Romeo L,Nardi F,et al. Mesothelioma in blood related subjects: Report of 11 clusters among 1954 Italy cases and review of the literatureAm J Ind MedYear: 2007503576917407142
3. Bianchi C,Nemo A,Bianchi T. Familial mesothelioma: A puzzling issueEur J OncolYear: 2008131816
4. Kalogeraki AM,Tamiolakis DJ,Lagoudaki ED,Papadakis MN,Papadakis GZ,Agelaki SI,et al. Familial mesothelioma in first degree relativesDiagn CytopatholYear: 201341654722351650
5. de Klerk N,Alfonso H,Olsen N,Reid A,Sleith J,Palmer L,et al. Familial aggregation of malignant mesothelioma in former workers and residents of Wittenoom, Western AustraliaInt J CancerYear: 20131321423822858896
6. IARCIARC Monographs on the Evaluation of Carcinogenic Risks to HumansA review of Human carcinogens: Metals, arsenic, dusts, and fibresYear: 2012100CLyonWHO, IARC293
7. Nguyen D,Lee SJ,Libby E,Verschraegen C. Rate of thromboembolic events in mesotheliomaAnn Thorac SurgYear: 200881032818291192
8. Elmes PC,Simpson MJ. Insulation workers in Belfast. A further study of mortality due to asbestos exposure (1940-75)Br J Ind MedYear: 19773417480911687
9. Selikoff IJ,Seidman H. Asbestos-associated deaths among insulation workers in the United States and Canada, 1967-1987Ann N Y Acad SciYear: 19916431141809121
10. Järvholm B,Sandén A. Lung cancer and mesothelioma in the pleura and peritoneum among Swedish insulation workersOccup Environ MedYear: 199855766709924454
11. Ulvestad B,Kjaerheim K,Martinsen JI,Mowe J,Andersen A. Cancer incidence among members of Norwegian Trade Union of Insulation WorkersJ Occup Environ MedYear: 20044684914724482
12. Cocco P,Dosemeci M. Peritoneal cancer and occupational exposure to asbestos: Results from the application of a job-exposure matrixAm J Ind MedYear: 1999359149884740
13. Bianchi C,Bianchi T. Malignant mesothelioma: Global incidence and relationship with asbestosInd HealthYear: 2007453798717634686
14. Bianchi C,Bianchi T. Susceptibility and resistance in the genesis of asbestos-related mesotheliomaIndian J Occup Environ MedYear: 200812576020040979
15. Bianchi C,Bianchi T. Mesothelioma among shipyard workers in Monfalcone, ItalyIndian J Occup Environ MedYear: 2012161192323776320

Article Categories:
  • Case Report

Keywords: Asbestos, cofactors, familial mesothelioma, insulators.

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