Reply

J. A. Bosch3 and S. Marsal

Internal Medicine Service and
1 Rheumatology Unit, Hospital General i Universitari Vall d’Hebron, Barcelona, Spain

We thank Drs Filiz and Dikensoy for their interest in our study. They present a female case of Behçet's disease (BD) with pulmonary artery aneurysms and massive haemoptysis 1 month after the normal delivery of her third child. She died 18 months later. Because of that they suggest the possibility of a relationship between pregnancy and vascular manifestations of BD.

As the authors concede in the title, their presentation is of a very rare manifestation of an infrequent disease which occurred in a puerperium period. On account of this we think that it is bold to infer any relationship between BD and pregnancy. Nevertheless, other points should be made.

The male/female ratio in BD is 1.03 and the mean age at onset is 25.6 yr [1], a common age for childbearing. If a correlation existed between BD and pregnancy we would expect more vascular complications in women with BD. However, it is known that male patients have vascular involvement 5.02 times more frequently than female patients [1]. Tüzün et al. [2] published 24 patients with arterial aneurysms. All were male. Yassine et al. [3] reported five male patients with BD and aneurysms of the pulmonary artery. Other individual cases of lung involvement in males have been reported.

In this particular case, we wonder what happened in her previous two pregnancies. We must assume that they occurred when the patient already had BD manifestations because she was suffering ‘ulcers for several years’ and she was relatively young for having a third child. Presumably both previous pregnancies, labours, puerperium and newborns developed properly. The absence of an association between BD and pregnancy could be suggested from this observation.

Also, it is mentioned that the patient had BD manifestations until 1 yr prior to admission. So, as we suggest in our study, this case is one more example showing that the clinical manifestations of BD are not consistently worsened during pregnancy.

Another important consideration is that the formation of pulmonary artery aneurysms carries a particularly poor prognosis and as the authors say ‘it is life threatening due to the increased risk of aneurysm rupture which often results in massive haemoptysis’. Obviously, the evolution presented here does not differ substantially from the expected evolution in BD male patients or even in non-pregnant BD female patients.

It is known that both pregnancy and labour increase the pulmonary blood pressure. In this case, the haemoptysis occurred 1 month after labour when the pulmonary blood pressure had returned to normal levels. We do not know when the vascular lesion develops or the mechanisms implicated in the rupture.

The therapeutic solution for pulmonary artery aneurysms is complex and good results have been published in relation to surgery and embolization [46]. In our series of 79 patients with BD, two male patients presented with arterial aneurysms (2.5%). One patient had an aneurysm of the hepatic artery and died on post-operative week 1. The second patient had an iliac artery aneurysm and is now well after 9 months of embolization.

We are conscious of the difficulties in studying the relationship between pregnancy and BD. In our study, we tried to advance knowledge in this interesting field since we, like other authors, were tempted to associate the two. But in view of our results and the epidemiological data, we do not believe that so far any correlation between pregnancy and vascular manifestations of BD can be established.

Notes

3 Correspondence to: J. A. Bosch. Back

References

  1. Gürler A, Boyvat A, Türsen U. Clinical manifestations of Behçet's disease: an analysis of 2147 patients. Yonsei Med J 1997;38:423–7.[Medline]
  2. Tüzün H, Besirli K, Sayin A et al. Management of aneurysms in Behçet's syndrome: an analysis of 24 patients. Surgery 1997; 121:150–6.[ISI][Medline]
  3. Yassine N, el Meziane A, Alaoui-Yazidi A, Bartal M. Aneurysms of the pulmonary artery in Behçet disease: Apropos of 5 new cases. Rev Pneumol Clin 1997;53:42–8.[Medline]
  4. Tüzün H, Hamurydan V, Yildirim S et al. Surgical therapy of pulmonary arterial aneurysms in Behçet's syndrome. Ann Thorac Surg 1996;61:733–5.[Abstract/Free Full Text]
  5. Sasaki Sh, Yasada K, Takigami K, Shiiya N, Matsui Y, Sakuma M. Surgical experiences with peripheral arterial aneurysms due to vasculo-Behçet's disease. J Cardiovasc Surg (Torino) 1998;39:147–50.[ISI][Medline]
  6. Mouas H, Lortholary O, Lacombe P et al. Embolization of multiple pulmonary arterial aneurysms in Behçet's disease. Scand J Rheumatol 1996;25:58–60.[ISI][Medline]
Accepted 8 September 1999





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