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SURVIVAL AFTER RUPTURE OF AN ANEURYSM OF THE SPLENIC ARTERY DURING PREGNANCY.

By GAVIN BOYD, M.B., F.R.C.S.(Ed.), F.R.C.O.G. Royal Maternity Hospital, Belfast.

A R E V I E W of the l i terature on this subject down to 1953 has been published by Tomsykoski et al. 17 In their paper 27 cases are collected f rom previous publications and one new case is added.

Out of these 28 cases there were two survivals. The first of these, re- corded by MacLeod and Maurice, 1° was a case of a primigravida, aged 22, who, at the 28th week of pregnancy suddenly developed symptoms of an internal haemorrhage which proved to be due to rup ture of the splenic a r t e ry near the hilum. A splenectomy was per formed and five days la ter the pat ient was delivered of a macerated foetus. Subsequently she had a normal pregnancy. No actuM aneurysmal dilatation was observed.

The record of the second case to survive was published by Gillam 5 and was that of a primigravida, aged 26. At the 40th week of her pregnancy she developed epigastric pain and vomiting; labour was induced and a dead foetus delivered. Four teen days la ter symptoms recurred and a spleneetomy was performed. Although no definite aneurysm was located, bleeding was controlled and the pat ient made a successful recovery.

A th i rd case has been recorded by Gallagher and Hudson 4, br inging the total number of repor ted cases to 29 ,and of survivals to three. In this case the patient, aged 32, was at the 20th week of pregnancy when she was suddenly seized with intense pain in the left hypochondrium. A laparotomy was performed and bleeding was seen to be originating in a lateral rup tu re of the splenic ar tery. A splenectomy was performed. Eleven days later she had a spontaneous miscarriage of a macerated foetus and then went on to make an un in te r rup ted recovery.

In this issue I now repor t a fu r the r case of the survival of a pat ient who had been under my personM care. Although, as in the cases of survival recorded above, no aneurysm was seen at operation or subse- quently on examination of the spleen, I consider it unlikely that the haemorrhage could have originated from any other lesion. The recovery may be a t t r ibuted very largely to the administrat ion under pressure of an intra-aort ic blood transfusion of one pint which, given when the abdomen was about to be closed and at a time when the pat ient seemed moribund, was followed by a dramatic revival.

As this is the four th repor ted case of rup tu red splenic aneurysm in pregnancy, in which recovery has taken place, and as it concerns the first pat ient to whom an intra-arter ial t ransfusion has been given in the Royal Materni ty Hospital, Belfast, the facts seem worthy of publication.

Case Report. A primig.avida, aged 22, a t tended the antenatal clinic of the Royal ~VIaternity

Hospital. B~lfast. from the 20th week of her pregnancy until the 38th week. Apart from slight oedema of her ankles no abnormality was noted at any of her visits. Her Y~rassermann reaction was negative.

On l l t h Jlmo, 1954, during the 38th week of her pregnancy, she was sent b y her doctor into ho~pltal suffering from abdominal pain. She s ta ted that she had felt pain across the upper abdomen for 6 hours and tha t she had vomited once. On examination no obvious reason for her complaint w a s revealed. The pulse rate was 80. the blood pressure 130/80 and the temperature normal. There was a vague tenderness in the

A N E U R Y S M OF S P L E N I C A R T E R Y D U R I N G P R E G N A N C Y 517

u p p e r a b d o m e n , b u t n o r igidi ty . T h e u t e r u s w a s en la rged to t h e e n s i f o r m proees s, t h e p r e s e n t a t i o n w a s a v e r t e x a n d t h e foeta l h e a r t w a s heard .

E i g h t e e n h o u r s a f t e r a d m i s s i o n , i.e. 24 h o u r s a f t e r t h e onse t o f s y m p t o m s , t h e p a t i e n t s u d d e n l y co l lapsed w i t h a c u t e a b d o m i n a l pain . She l ay on he r le f t s ide w i t h he r leg s d r a w n up , a n d w a s unwi l l ing to t u r n on to her back.

O n e x a m i n a t i o n she p r e s e n t e d t h e typ ica l s igns of p ro found shock. The pu l s e w a s n o t coun tab le , t h e blood p re s su re w a s n o t recordab]e a n d t he foeta l h e a r t w a s n o t heard . A blood t r a n s f u s i o n w a s s t a r t e d a n d m o r p h i n e (gr. ½) was a d m i n i s t e r e d , b u t v e r y l i t t le i m p r o v e m e n t , i f any , w a s n o t e d e v e n a f te r two p i n t s o f b lood h a d b e e n given.

I m m e d i a t e l a p a r o t o m y w a s u n d e r t a k e n a n d t h e a b d o m i n a l c a v i t y w a s f o u n d to c o n t a i n a v e r y large q u a n t i t y o f f resh blood a n d blood clot . A c lass ica l C a e s a r e a n sec t ion w a s p e r f o r m ed a n d a s t i l lborn m a l e foe tus , we igh ing 6 lb. 8 oz., was del ivered.

I f w a s t h e n d iscovered t h a t t h e b leeding w a s c o m i ng f rom the region o f t h e sp leen a n d a d iagnos i s of r u p t u r e d a n e u r y s m of t h e splenic a r t e ry w a s m a d e , a l t h o u g h no a n e u r y s m w a s def ini te ly located. Access to t h e sp leen w a s ob ta ined b y e x t e n d i n g t h e incis ion a l m o s t to t h e ens i fo rm process ; a s p l e n e c t o m y w a s p e r f o r m e d a n d t he b leed ing control led. T h e u ter ine w o u n d w a s t h e n qu ick ly su tu red .

A t t h i s s t a g e t h e p a t i e n t s e e m e d m o r i b u n d . There w a s only a n occas iona l g a s p a n d t h e aort ic pu l se w a s weak. Troub le w a s exper ienced w i t h t h e b lood t r a n s f u s i o n as t h e needle h a d c o m e o u t of t h e v e i n a n d al l t h e o t h e r ve ins were comple t e ly col lapsed.

A t r a n s f u s i o n needle w a s p u s h e d in to t h e a o r t a a t t h e level of t he 3rd l u m b a r v e r t e b r a a n d one p i n t o f b lood w a s g iven u n d e r pressure . T h e r e sponse w a s i m m e d i a t e . O n c o m p l e t i o n of th i s t r a n s f u s i o n t h e needle w a s w i t h d r a w n a n d a finger p laced over t he needle punc tu re . W h e n t h e finger w a s r e m o v e d a s m a l l h a e m a t o m a formed, necess i t a t - ing t h e re-appl ica t ion of f inger p ressure . Some m i n u t e s la te r t he p r e s su re w a s r e l e a sed for a second t i m e ; all a p p e a r e d to be well. T h e a b d o m e n w a s closed. On c o m p l e t i o n o f t h e opera t ion t h e p a t i e n t ' s b lood p r e s s u r e w a s 85/40.

A n o t h e r p i n t of b lood w a s t h e n g i v e n in to a ve in , b r ing ing t h e t o t a l a m o u n t o f b lood a d m i n i s t e r e d to 5 p in t s . T h e blood p ressu re w a s n o w [00/60.

a course o f penici l l in a n d s t r e p t o m y c i n w a s s u b s e q u e n t l y g i v e n a n d t h e conva l e scence w a s sa t i s fac tory , un t i l , on t h e 8 t h d a y o f t h e p u e r p e r i n m , a deep v e n o u s t h r o m b o s i s deve loped in t h e lef t leg. T h i s w a s t r e a t e d w i t h hepar in followed by pheny l i n -daned ione (" D i n d e v a n "). U l t i m a t e l y , five w e e k s a f te r t h e opera t ion w h e n t h e v e n o u s t h r o m b o s i s h a d resolved, she w a s d i s c h a r g e d f rom hosp i t a l fit a n d well .

Six weeks a f te r d i scharge f rom hosp i t a l she w a s seen a t t h e p o s t - n a t a l clinic a n d he r cond i t ion w a s f o u n d to be s a t i s f ac to ry .

O n t h e 27 th J a n u a r y , 1956, t h e p a t i e n t h a d a s p o n t a n e o u s de l ivery o f a n o r m a l m a l e ch i ld we igh ing 7 lb. 3 oz.

Pathology : T h e sp leen w a s of n o r m a l size a n d appearance . There was m u c h b lood a n d b lood clot obscur ing t h e hi lar s t r u c t u r e s . No a n e u r y s m w a s found. The re w a s n o his to logical les ion to a c c o u n t for t h e haemor rhage . T h e splenic s u b s t a n c e w a s n o r m a l a p a r t f rom s inuso ida l d i la ta t ion .

Discussion. Rupture of the splenic artery with or without aneurysmal formation

is an uncommon condition. Hil l and Inglis 7 have collected 245 cases of which 9 survived.

The various factors which may determine or contribute to the forma- tion of an aneurysm are as fol lows : - -

(1) A congenital defect in the arterial wall, which usually occurs at the point where the artery branches. This explains why the lesion may be multiple, as in the case reported by Tennent and Starritt, 1~ and why rupture can occur without format ion of an aneurysm, as in the case reported by Gallagher and Hudson. 4

(2) Arteriosclerosis. Sherlock and Learmonth 14 found atheromatous degeneration in 46 per cent. of their cases. Owens and Coffey 11 found it in 60 per cent. of their cases, and noted that this condition could localise itself to the splenic artery. I t is unlikely, however, that arterio- sclerosis causes splenic arterial aneurysm in pregnancy.

(3) Syphil is does not as a rule affect a peripheral artery, and in this case the Wassermann and Kahn reactions were negative.

(4) A mycotic aneurysm fol lowing bacterial endocarditis has never been reported during pregnancy.

518 IRISH JOURNAL OF MEDICAL SCIENCE

(5) Splenomcgaly, which calls for an increase in the size of the splenic artery.

It has been pointed out by Lennie and Sheehan 9 that aneurysm of the splenic artery is commoner in women, whereas aneurysm of the renal artery is commoner in men. It is fur ther pointed out that ruptured aneurysm of the splenic artery is commoner in pregnancy than apart from pregnancy, and usually takes place between the 7th and 9th months of gestation.

The reason for rupture, however, during pregnancy is not very clear. Pressure and trauma are possible causes. Sheehan and Falkiner 13 point out that during pregnancy a considerable number of patients show splenomegaly associated with anaemia or accidental haemorrhage. Splenomegaly causes the splenic artery to enlarge, with increase of strain on an aneurysm or on any defect present in the wall. Neither anaemia nor accidental haemorrhage was present in the case presented. In pregnancy there is frequently a splenomegaly which may be physiologieal in origin.

Possibly two factors are involved; a congenital defect in the artery wall and changes in the vessel wall due to splenomegaly. The speed with which these changes occur may decide whether or not an aneurysm will form. A rapid increase in the size of the artery would cause a rupture of the wall even without aneurysm formation, while a slow increase in size would give time for the formation of an aneurysmal sac.

It has also been suggested that aneurysms of the cerebral vessels are more likely to rupture during pregnancy. Sullivan et al. ~5 report that 2 per cent. of sudden obstetric deaths are due to the rupture of a con- genital cerebral aneurysm. They postulate that the sudden decline of hormonal influences affects blood vessel tone and makes rupture of an aneurysm more likely after the 36th week.

I t is obvious, therefore, that the factors determining the rupture of congenital aneurysms are very obscure; it has even been noted that cerebral aneurysms for no known reason show an increased tendency to rupture in the spring of the year.

I t has been observed that an aneurysm of the splenic artery frequently ruptures in two stages separated by several hours or days. Owens and Coffey 11 report a " double rupture " in 21 per cent. of 204 cases examined, and in the case reported here the interval between the two stages was 24 hours. In the first stage there is a small leakage of blood which ~orms a retroperitoneal haematoma around the aneurysm. During this stage a diagnosis might be made, and might be confirmed by arteriography. Usually, however, the diagnosis is not made until the second stage, when there is a severe haemorrhage which bursts into the lesser sac or into the peritoneal cavity. Frequently the condition at this stage is thought to be a concealed accidental haemorrhage.

On laparotomy, however, the presence of blood in the peritoneal cavity suggests a ruptured aneurysm. Lennie and Sheehan 9 state that the aneurysm may be in the splenic, the renal, the hepatic or the superior mesenterie arteries. If it is in the splenic artery they advise immediate Caesarean section to obtain better access, followed by a division of the greater omentum below the stomach in order to expose the splenic artery. A ligature may be passed around the artery proximal to the aneurysm,

ANEURYSM OF S P L E N I C A R T E R Y DURING P R E G N A N C Y 519

and it is suggested by these authors that the spleen may be left to a t rophy if the patient 's condition does not warrant splenectomy.

Hill and Inglis, however, report a ease 7 in which an aneurysm of the splenic a r t e ry was discovered unexpectedly dur ing a part ial gastrectomy for duodenal ulcer in a male patient aged 46. The splenic a r te ry was ligated on either side of the aneurysm and the sac excised. Par t ia l gastrectomy was then performed, af ter which it was seen that the blood supply to the spleen, presumably through the vasa brevia, was unimpaired. The spleen was left in situ and the patient made a good recovery. Aird 1 reports a case in which the splenic ar te ry was ligated af te r it had been eroded by a peptic ulcer. At a subsequent gastrectomy the spleen appeared to be normal.

The question of intra-arterial transfusion has been extensively explored in recent publications. Seeley and Nelson TM have published a compre- hensive review of the l i terature and mention tha t Landois in 1879 first re fer red to the possibility of this type of transfusion. Its value over intravenous transfusion seems to lie in its effect on the heart, due to increased intra-aortic pressure causing improvement in the coronary blood flow.

Many different arteries have been used for this purpose, but the radial and the femoral arteries are the two most often chosen. The aorta, how- ever, has undoubted advantages when the abdomen has been opened, as in this case.

Brown s describes his experiences in 165 cases in which hypotension was obtained pre-operatively in neurosurgieal cases by exsanguination. Sub- sequently the blood was replaced and it was found that intra-arterial replacement produced a more rapid response than intravenous replace- ment. I t is his view that intra-arterial transfusion restores the efficiency of the heart, and he has noted that 100 ml.-200 ml. of blood given in 30 seconds can have a dramatic effect. Haxton 6 describes a method of inserting a special needle into the aorta from the lumbar region in order to administer an intra-aortic transfusion and points out that the aorta will not go into spasm or become the seat of obstructive thrombosis, dangers which may accompany a transfusion into the radial artery.

Horton e t al . 8 describe the technique in detail for transfusion into the radial ar tery and advocate a prel iminary brachial plexus or stellate ganglion block, using 20 ml. of I per cent. lignocaine hydrochloride. Oncc the cannula is ia positio11 2 ml. of 2 per cent. procaine is injected into the artery, following which the blood is pumped in at 50 mm. Hg. When a sufficient quanti ty of blood has been given (usually about 1-2 litres), a fu r the r 2 ml. of 2 per cent. procaine is injected. Obviously, if this type of transfusion is contemplated, preparat ions must be made beforehand ready for the moment when a sudden emergency arises.

In the case described no preparat ions had been made. When it was obvious that the pat ient ' s condition had become desperate a transfusion needle was pushed into the aorta and blood pumped in by means of a sphygmomanometcr bulb attached to the bottle of blood.

Judging from the result in this cas~ it seems that there is a place for intra-arterial transfusion in obstetrics, but in the author 's opinion the procedure should be reserved for those very collapsed patients who do not respond to the more orthodox intravenous transfusion.

520 I R I S H J O U R N A L OF M E D I C A L S C I E N C E

Summary. A case of rup tu red splenic ar ter ia l aneurysm dur ing p regnancy is

presented. Twenty nine cases of this condition are to be found in the li terature, with the survival of three. This case raises the total to thir ty, with the survival of four.

An impor tan t fac tor in the recovery of the pa t ien t was thought to be the t ransfus ion of one p in t of blood under pressure into the aorta.

In t ra -a r te r ia l t ransfusions are discussed.

I am grateful to ~¢Ir. H. L. Hardy-Greet, into whose wards this pat ient was admit ted for his permission to publish the ease. I am alice grateful to Mr. T. Kennedy and Dr. F. Grant, for their assistance at the time of the operation, and to Dr. H. Graham for administering an excellent anaesthetic.

I am indebted to Dr. J. E. :Morrison for his help and criticism in the preparation of this paper.

References. 1. Aird, Jam. (1946). Brit. J. Surg., 33, 385. 2. Brown, A. S. (1953). Lancet, ii, 745. 3. Chalmers, J . A. (1949). Brit. J. Surg., 37, 86. 4. Gallagher, H. W. and Hudson, K. (1954). Brit. Meal. J . , ~i, 1209. 5. Gillam, J. :F. E. (1942). lbld., i, 69. 6. Haxton, H. A. (1953). Lancet, i, 622. 7. Hill, 1~. M. and Inglis A. (]955). •rit. J . Surg., 42, 408. 8. Horton, J. A. G., Inkster, J. S., :Mackenzie, A. and Pask, E. A. (1953}. Brit. Med.

J . , ii, 1294. 9. Lennie, R. A. and Sheehan, H. L. (1942). J. Obst. Gyn. Brit. Emp. , 49, 426.

I0. ~VIacLeod, D. and :Maurice, T. (1940). Lancet, i, 924. 11. Owens, J. C. and Coffey, 1~. J . (1952). Surg. Gyn. Obst., 97, 313. 12. Seeley, S. F. and Nelson, 1~. M. (1952). Internat. Abstr. Surg., 94, 3, 209. 13. Sheehan, H. L. and Falkiner, N. :M. (1948). Brit. Med. J . , ii, 1105. 14. Sherlock, S. V. P. and Learmonth, J. R. (1942). Bri. J. Surg., 30, 151. 15. Sullivan, C. L., ~¢Iinkel, H., Camobel, E. and Graham, J. H. (1953}. Postgrad Med.

14, 4, 329. 16. Tennent, R. A. and Starri t t , A. (1950). Glozgow Med. J . , 31, 465. 17. Tomsykoski, A. J . , Stevens, R. C., Izzo, P. A. and Rodriquez, C. E. (1953). Am. J .

Obat., 66, 1264.