Twisted hematosalpinx

TWISTED

HEMATOSALPINX*

ROBERT

TAUBER,

Philadelphia,

T

HE

term hematosalpinx ~-as long understood to designate any tube filled with blood. l’hen Veit came forward with the suggestion that tubal pregnancies as we11 as bIood tubes due to new growth shouId be excluded, a view which is now generally accepted. Thus hematosalpinx may be divided into the folIowing three groups according to origin: (I j cases in which the condition is due to a congenital obstruction of the genital cana (gynatresiaj; (2) cases attributabIe to bleeding from a ruptured bIood vesse1 into a previously occluded tube (as in saIpingitis, hemorrhagic inflammation, typhoid fever or phosphorus poisoning); and (3) cases attributable to a severe disturbance in blood supply due to torsion or twisting of the tube, either normaI or diseased. Heynemann is of the opinion that torsion of the normal tube is not too “uncommon” and stresses the importance of keeping this possibility in mind. In 1918 Schweitzer pubIished a case presenting isolated torsion of a normal tube; simiIar cases have been described by Michael, Hansen, Scheid and Neugebauer. The literature contains simiIar reports by Rueder, AuIhorn, Norris, Roger, Stark, Herff, Hirst, Barton and Branch, NeeI, Lear and Virnig, Auvray, Ansbach-Prager, Eunike and others as we11 as reports on cases involving compIete amputation of the twisted tube by such authorities as Kolb, Ruppold, Ries (biIateraI), Barrett-Lash and Anderson. In 1927 before the Obstetrical Society of Vienna the writer presented a case of a twisted tube in pregnancy. Many a case of this kind is probably overIooked for the reason that diagnosis is often diflicuIt. The writer believes, therefore, that it might be usefu1 to describe the condition * From the GynecoIogicaI

AlaJr,

1949

M.D.

Pennsylvania

in some detail and to submit a report on a recent, particularly iIIuminating case. The literature mentions the Etiology. various factors generaIIy heId responsible for torsion of a tube. As a rule, however, no clear distinction is made between the genera1 condition, the immediate cause and the twisting forces themselves. It wouId seem timeIy, therefore, to attempt to cIarify this question of the etioIogy of torsion and above al1 to estabhsh the difference between predisposing factors and twisting forces. The predisposing factors may be divided into two categories: (I) those directly related to pathologic conditions generalI?; in the abdomen (e.g., adhesion, displacement of organs); and (2) those directI? related to the part which is twisted. Moreover, the twisting forces themseIves can operate in two distinct ways: (I ) from without (e.g., a blow on the abdomen or some extraordinary movement of the body as in sports, or dancing) with repercussions in the part in question; and (2) from within the abdomen (e.g., a growing tumor or intestina1 peristalsis). In each case the predisposing factors and twisting forces shouId be studied separateIy. The foIlowing, which Iists the factors and circumstances IikeIy to favor torsion, is designed to faciIitate determination of the etioIogy in a given case by making it somewhat easier to recognize both the predisposing factors and the twisting forces at play. PREDISPOSING FACTORS I. PathoIogic condition of the tube itself: (A) Enlargement and elongation Anspach) Congenital Infantilism (Schweitzer) Acquired Hydrosalpinx

Service of the Graduate SchooI of Medicine, University PhiIadeIphia, Pa.

661

(Rokitansky,

of Pennsylvania,

662

Tauber-HematosaIpinx

Hematosalpinx Pyosalpinx Ectopic pregnancy (B) AbnormaI mobility due to long and membranaceous mesosalpinx CongenitaI (Michel, Roger, Davies, Barrett) Acquired Pregnancy (Aulhorn, Tauber) Ovarian or parovarian tumor (Tauber) z. Pathologic conditions eIsewhere in the abdomen: (A) Adhesions (Rokitansky) due to previous operations or acute or chronic infIammafions (B) DispIacement of the uterus causing narrowing of the pedicle (Barrett) (C) Tumor formation TWISTING

FORCES

I. Extraperitoneal

2.

forces (SeIIheim, Hansen, Rieder) (A) Trauma FaII; bIow to the abdomen (B) Active movement of the whoIe body (a) Sudden or constant movements (b) Continuous or repeatedIy equa1 movements (C) Increased action of the abdominal pressure (a) by lifting heavy weights (b) on coughing IntraperitoneaI forces Traction of shrinking adhesions Traction or pressure of adjacent neoplasms (Tauber) of growing pregnant uterus (Barrett, Tauber) of dispIaced organs IrreeuIar mowing of the organ itself which influences -its bzance &chweitzer, Payr) Resistance in growing to a certain direction by the presence of vesseis, ligaments, etc. IntestinaI peristaIsis (Schweitzer) Tuba1 peristaIsis AIternate distension and evacuation of the bIadder Increasing of the abdomina1 pressure by straining Hemodynamic mechanism (Payr), activated by: Menstruation (MicheI, Norris, Roger, Schweitzer) Constipation (Stark, Schweitzer) Coitus (Schweitzer) Prolapsing uterus (Barrett-Lash) Adhesion (present case)

Opinions on the twisting forces are SelIheim stresses the sharply divided. extraperitonea1 forces and believes that most cases are due to these forces aIone. On the other hand, Payr’s interesting theory of hemodynamic torsion is compIetely different. After extensive studies on modeIs and cadavers he was the first to induce intra-abdominal torsion of the pedicIe of artificial tumors in the omentum of animaIs. He arrived at the folIowing concIusion :

Arteries have a firm and rigid wall and act as a support for the pedicIe. The veins are much longer and often tortuous. The elasticity of their waIIs aIIows an enormous increase in their capacity. Compression or kinking of the pedicIe activates this development. The thin-waIled veins are far more prone to obstruction than the more resistant arteries. The veins receive bIood from the capiIIaries without resistance. In this way the veins are congested and increase in length as well as in transverse diameter and uItimateIy form a concave bow toward the artery. The connective tissue between the artery and the vein, however, limits the bow formation and forces the vein to form a spiral around the artery. This Ieads to torsion of the pedicIe. Torsion brought about by unequa1 pressures in the bIood vesseIs of the pedicle is termed hemodynamic torsion. DIAGNOSIS

AND

DIFFERENTIAL

DIAGNOSIS

Diagnosis of a twisted tube is aIways a difficuIt matter. The cIinica1 picture is not suffrcientIy characteristic; as a rule, therefore, the true nature of the condition is not discovered unti1 the patient is being operated on, generahy under the diagnosis of acute appendicitis. The twisted tube must be differentiated from ureter stone, ectopic pregnancy, acute appendicitis and salpingitis. Both a ureter stone and a twisted tube are associated with repeated attacks of severe pain radiating to the back and to the symphysis. Urologic studies and x-ray examination wiI1 generaIIy serve to exchrde a ureter stone. In the differentiation of a twisted tube from an ectopic pregnancy the vagina1 examination is of IittIe heIp. The adnexa1 mass cannot be outIined definiteIy because of the patient’s inabiIity to reIax due to severe pain. The absence of IIuid in the cuI-de-sac of DougIas does not excIude ectopic pregnancy and the presence of fItrid might be interpreted as an exudate or evaded bIood from a twisted tube. SmaII amounts of fluid without buIging of the American

Journal of Surgery

Tauber-Hematosalpinx posterior vaginal waII can hardly serve as a basis for the diagnosis. The red bIood count is of no diagnostic value because it can be normaI or subnorma in both conditions. OnIy the history remains; this may be very characteristic of ectopic pregnancy although in rare occasions ectopic pregnancies are encountered in the presence of normaI menstruation. Both acute appendicitis and a right twisted tube show severe pain in the right lower abdomen. The pain in appendicitis may be somewhat higher up but this finding is too vague to serve as the basis for a differentia1 diagnosis. The diffIcuIty is further increased by the fact that the bIood count shows a high increase in the number of white blood ceIIs and a high percentage of polymorphonucIear Ieukocytes in appendicitis as we11 as in cases of a twisted tube because of the peritonea1 irritation. A history of \.omiting in both conditions is another symptom IikeIy to Iead to the wrong diagnosis. The discovery of a soft, IongitudinaI mass on the side of the uterus, by vagina1 examination after administration of a sedative or in anesthesia, wilI excIude appendicitis or at Ieast suggest that appendicitis is most unIikeIy. Acute saIpingitis can be excIuded safeIy by the absence of high temperature. Moreover, saIpingitis is generaIIy biIatera1 and commonIy presents other signs of acute inflammation on the externa1 genitaIia, such as vuIvovaginitis, urethritis, infection Gf Bartholin’s gIands and puruIent discharge. CASE

REPORT

J. G., a tlventy-eight year oId woman, was admitted to the Graduate Hospital on December 23, 1946. She compIained of severe pain in the right Ion-er abdomen and was unabIe to walk in an upright position. She had one six year old chiId, with no miscarriages. Menstruation began at the age of fifteen, was reguIar every tkventy-eight days lasting 3 to 6 days, was moderate in quantity and was associated with sIight cramps on the first day. The last menstruation, which was of norma duration and quantity, was on December g, 1946. The patient had undergone a Ieft saIpingo-oophor.Vay,

1ygc)

663

ectomy, February 17, 1942, because of an ectopic pregnancy. For the past two years the patient complained of pain in the right side of the abdomen and the back a11 the way down to the inguina1 region. The pain came in attacks, the first of which made its appearance two years previously. An x-ray examination ~vas done in another hospitaI where she was informed that she had a ureter stone. She said that the stone dissoIved after she took medicine. For the past year she was treated every month by dilatation of the ureter. There was a second major attack on Easter, 1916. Again she was hospitalized and treated by ureter diIatation. In November, 1946, a third attack brought her back to the same hospita1 where x-ray stucIies and dilatation of the~ureter were carried out. The patient was in permanent pain ever since and was unable to walk or stand upright. On December 17, 1946, a fourth attack Ied to hospitalization. S-ray studies and uroIogic examination were completeIy negative. The patient \vas discharged from the hospita1 but was never free of duI1 pain in the abdomen. On December 22, 1946, she suffered an attack of severe pain accompanied by vomiting. Her family physician sent her to the Graduate Hospital. On admission the patient’s temperature was IOO.I’F. and her p&e, 92. The right lower abdomen was so extremely tender that she couId not toterate the sJightest touch. After administration of a sedative a carefu1 vaginal examination was performed. The folIowing findings were noted: uterus of norma size, in body axis, firm and of Iimited mobility; the right adnexaJ area was extremeJy tencler; a soft mass between the right uterine edge and the symphysis could J>e palpated but it was impossibJe to outline the mass definiteIy because of the pain and the patient’s resistance. The chart of the previous operation bore the information that a Jeft saIp;ngo-oophorectomy had been done because of an ectopic pregnancy, the latter having been determined by the microscopic finding of large cells and syncytia1 masses in the faJIopian tube. No diagnosis couIc1 be given after the first examination after admission. Fctopic pregnancy on the right side, a ureter stone, appendicitis and acute saIpingitis were under discussion. The family physician informed us that a uroIogic examination, incIuding an x-ray study performed a few days previously in another

664

FIG. I. Specimen of the removed right hematosaIpinx and the band-like adhesion which extended across the pelvis. After fixation the specimen was cut in several segments to allow histologic studies in order to exclude inflammation and ectopic pregnancy.

hospital, had been completely negative. On the strength of this information the diagnosis of a ureter stone was excIuded. Ectopic pregnancy seemed most unlikely in view of the history of perfectly normal menstruation. The temperature of I 00. I OF. did not suggest an acute process in the appendix or the adnexa1 area. The bIood count reveaIed the following: 4,400,000; hemogIobin, 75 per erythrocytes, neutrocent (12.5 Gm.) ; Ieukocytes, 13,900; phiIes, 88 per cent; Iymphocytes, IO per cent; and mononucIears, 2 per cent. The bIood count pointed to acute appendicitis but the vagina1 findings suggested an adnexal disease. The fact that the temperature was onIy sIightIy elevated together with the comparatively low puIse certainly seemed to excIude any acute inflammation. Therefore, whiIe no definite diagnosis had been made, we decided to resort to surgery in the expectation of finding some pathoIogic condition in the adnexal area. The abdomen was opened by midline incision. On the right side there was a bluishbIack adnexal mass the size of a Iemon which proved to be the enIarged right tube. The mass was caught by a band-shaped adhesion some 7 cm. in Iength which extended across the inlet of the peIvis from the top of the bIadder to the sigmoid and which probabIy had been caused by the previous operation. The tuba1 mass was twisted, making four times a compIete turn of 360 degrees. The ovary was abnormaIIy far from the tube. There was no free blood in the abdomina1 cavity. SaIpingectomy was performed and the band-shaped adhesion was removed. The cecum was inspected but no

appendix couId be detected. The appendix probably had been removed in the course of the first operation but no mention of the fact appeared on the chart. The postoperative course was uneventfu1 and the patient Ieft the hospita1 on the tenth day after surgery. She had no complaints and said she feIt perfectly weI1. PathoIogicaIIy, the faIIopian tube was a bluish-black, retort-shaped, twisted mass showing acute circuIatory changes. (Fig. I.) The Iength of the tube was IO cm. and its greatest diameter was 4.5 cm. The peritonea1 surface was smooth and the mass was semi-soIid. The uterine end was conica and the two Ioops formed by the torsion were gIued together. The fimbriotic end was cIosed. OnIy a few smaI1 residua of the fimbriae were present. Section of the tube reveaIed a thick, dark red waI1. The Iumen was fiIIed with ctotted bIood and bIoody fluid; IongitudinaI folds were visibIe after remova of the contents. HistoIogicaIIy, the muscIe fibers of the tuba1 waI1 were separated by masses of red bIood ceIIs and by edema. The vesseIs were markedIy diIated. There was extensive thrombosis of the veins. The endosaIpinx was infiltrated with bIood; the epithelium was flattened and Iifted away from the underIying Iayers in some areas. At a few points there was no trace of epitheIia1 tissue. The waI1 contained many ceIIs fiIIed with bIood pigment. No evidence of inflammation and ectopic pregnancy couId be found. The pathoIogic disturbance in this patient was an originaIIy norma right tube which had undergone acute axia1 torsion resuIting in stranguIation with consequent hemorrhagic changes. The rapid deveIopment of the edema together with the thrombosis of the veins gave the pedicIe a pIastic consistency which made detorsion and restitution impossibIe. COMMENTS

This case is especiaIIy iIIuminating from the diagnostic viewpoint. Even a previous appendectomy did not prevent the usua1 error of taking appendicitis into serious consideration. The patient did not know that the appendix was out and, as mentioned, the appendectomy was not recorded on the oId chart. The possibihty of a ureter stone couId be excIuded here in view of the American

Journal of Surgery

complete urologic examination performed in another hospital onIy a few days before admission. However, it is amazing to note that this patient was treated repeatedIy in the same hospital for a stricture of the right ureter and for a ureter stone two years previously. She had never seen the stone nor had its presence ever been demonstrated by x-ra)‘. It is highIy probable, therefore, that the various attacks which led to the repeated hospita1 visits were due to torsion of the tube and that the attacks of pain were due to the pathoIogica1 condition of the tube rather than to some dubious ureter stone. This, however, can be considered proved beyond doubt only if the attacks do not recur in the future. Ectopic pregnancy was not definitely excluded but seemed most unlikely in view of the history of perfectIy normal menstruation. The patient reported no irreguiarity; both the duration and the amount were normal and constant as far as could be ascertained. However, the history of a pre\,ious ectopic pregnancy on the left side did arouse the suspicion that the case might possibly be one of ectopic pregnant! on the right side. The difhculties involved in making the diagnosis of a twisted tube underscore the importance of considering the possibiIity of acute salpingitis as well as of a twisted tube in any case of questionable appendicitis in a temaIe patient. Attacks of pain always point to the diagnosis of twisted pedicle. This symptom, which is generaIl? recognized as pathognomonic in ovarian cl-sts, is often neglected in the diagnosis of a twisted tube so long as there is no o\-arian pathology. However, the histor\ of repeated attacks of pain seems to be more significant diagnosticalIy than al1 the other symptoms and constant awareness of this fact wilt prev;ent many errors and enhance the chances of a correct diagnosis of twisted tube before the abdomen is opened. As for the etioIogy of the case under discussion, inspection of the tabIes wiI1 show that there were predisposing factors here tlirectIy related to pathologic conditions in .\ tar-,

1c)q)

the tube itself as we11 as elsewhere in the abdomen. There was a firm adhesion across the pelvis due to the previous operation. The ovary was abnormaIIy far from the tube, showing that the right mesosaIpinx was extremely Iong, forming a pedicle which was we11 predisposed for torsion. The tube itself was abnormaIIy long, probably a congenita1 condition. It seems very likely that there was aIso an elongation of the other tube and that this brought on the ectopic pregnancy which was operated upon two years ago. The patrent gave no history of trauma. There was no recohection of any fall or b1ow to the abdomen which could be construed as an extraperitonea1 twisting force but, of course, active and extraordinary movements of the whoIe body and increased abdominal pressure can never be excluded compIeteIy. In this case the intraperitoneal forces and, most particuIarIy, the factor of tubal peristaIsis seem to be of greater importance. It requires no stretch of the imagination to picture the probabIe sequence of events here. Since the Iong tube on the elongated mesosaipinx had an exceptionally wide the tube was caught in the bandrange, shaped adhesion across the pelvis; the band exerted pressure on the tube, blocking the circulation and thus activating the twisting forces for hemodynamic torsion. Torsion, in turn, brought on the pathologic changes. It may be presumed that these changes took the following course: the peritoneal Iayers of the tube were glued together; congestion increased more and more; the mucous membrane became edematous; and blood evaded into the Iumen; the frmbriatic end was inverted as a result of the extremely swohen condition of the mucosa (a mechanism described by Opitz) ; the tube, finally, cIosed up and the colIection of bIood in the tube formed the hematosaIpinx. In this case, therefore, it would seem that a previousIy norma tube had been twisted and that the hematosaIpinx subsequentIy deveIoped. The hematosaIpinx became another predisposing

666

Tauber-HematosaIpinx

factor favoring the subsequent torsions, The twisting forces of the Iater torsions were probabIy a combination of the hemodynamic mechanism and extra- and other intraperitonea1 forces. SUMMARY

A brief review of seIected cases of twisted tube was foIIowed by a few words concerning the significance of this condition. An attempt was made to dispe1 some of the confusion surrounding the etioIogic probIems by drawing a sharp distinction between the predisposing factors and the twisting forces. TabIes Iisting such factors and forces were submitted in the hope that this condensed information might faciIitate determination of the etioIogy in a given case. The diffIcuIties invoIved in making the diagnosis were pointed out and some directions were given as to the manner of differentiating between this condition and other entities presenting simiIar symptoms. An enIightening case observed by the writer was reported in detai1 notably for the purpose of iIIustrating the diffcuIty in estabIishing the differentia1 diagnosis. The pathoIogy and the deveIopment of the pathoIogic changes in the norma tube were discussed. REFERENCES ANDERSON, H. E. Torsion of apparently normal ovary, and spontaneous amputation of faIIopian tube during adoIescence. Am. J. Obst. u Gynec., 49: 2839 1945. ANSPACH, B. M. The torsion of tuba1 enIargements with a specia1 reference to pyosaIpinx. Am. J. Obst. u Gynec., 66: 553, rgrz. AULHORN. F. Soontane StieItorsion normaler Adnexe in de; Schwangerschaft Zentralbl. J. Gyniik., 34: 538, 1910. AUVRAY, H. Un cas de torsion spontanei: de Ia trompe et de I’ovaire normaux. Bull. Sot. d’obst. et de \ ~JWWC., I: 727, 1912. BARRE, H. W. and LASH, A. F. Spontaneous amputation and subsequent acute torsion of the normal Ieft faIIopian tube and hbroma of the right ovary. West J. Surg., 51: 135, 1943. BARTON, F. E. and BRANCH, C. F. A uniIatera1 hydrosaIpinx with torsion without invoIvement of the

ovary in a child 15 years of age. Arch. j. Pediur., 59: 356, 1942. EUNIKE, K. W. Isoliert torquierte normale Tube. Zentralbl. f. Gyniik., 46: 1484, rgzz. HANSEN, A. Tubentorsion mit HamatombiIdung und ihre AetioIogie. Zentralbl. J. C.yniik., 46: 707, rgzz. HEYNEMANN, T. HamatosaIpinx. In HaIban, J. and Seitz, L. Biologic und PathoIogie des Weibes. Berlin-Wien, 19.24. Urban and Schwarzenberg. MICHEL, B. Isoliert torauierte normaIe Tube. Zentralbl. J. Gyniik., 46: go5, 1922. NEEL, H. B., LEA A. and VIRNIG, M. P. Torsion of the uterine adnexa in childhood. Am. J. Obst. Ed Gynec., 45: 326, 1943. NORRIS, C. C. Torsion of the normal uterine appendages. Am. J. Obst., 63: 850, rgrr. NEUGEBAUER, F. Ein Beitrag zur Stieldrehung gesunder Adnexe im Kindesalter. Zentralbl. J. Gyniik., 46: 1961, 1922. OG~REK, P. Spontanabtrennungen der weiblichen Adnexe. Arch. J. GynGk., 102: 300, ,914. OPITZ, E. Beitrag zur Mechanik d. Tubenverschlusses. Zentralbl. J. Gyntik., 28: 1496, 1904. PAYR, E. Ueber d. Ursachen d. StieIdrehung intraperitonea1 geIegener Organe. Arch. J. klin. Cbir., 68: 501, 1902. PAYR, E. Weitere experimenteIIe and kIin. Beitrage zur Frage d. StieIdrehung intraperitoneaIer Organe u. GeschwiiIste. Deutscbe Ztscbr. J. Cbir., 85: 392, rgo6. PRAEGER, J. Ueber Stieldrehung d. Eileitergeschwtilste. Arch. j. Gyniik., 58: 579, 1899. REIFFENSCHEID. K. Die StieIdrehung d. Adnexe. HaIban, J.’ and Seitz, L. BioIogie- u. Pathologic d. Weibes. VoI. 3, p. 684. Berlin-Wien, 1924. Urban & Schwarzenberg. RIES. E. Snontaneous amoutation of both faIIoeian tubes. Am. Gynec. ti Obst. J., 16: 325, tgoo. ROGERS, L. Torsion of fallopian tubes. Brit. M. J., I : 778, 1925. v. ROKITANSKY, C. Ueber Abschniirungen d. Tuben u. Ovarien u. iiber StranguIation der Ietzteren durch Achsendrehung. Allg. Wien. med. Ztg., 5: g, 17, 25, 1860. RUDER, K. Drei FaIIe von StieIdrehung der Tube. Zentralbl. J. Gyniik., 45: 45, rgz I. RUPPOLT, F. Zur Kenntnis tiberzahliger Eierstocke. Arch. J. Gyniik., 47: 646, 1894. SCHEID, F. Weiterer Beitrag zur StieIdrehung d. Adnexe im kindlichen AIter. Zentralbl. J. Gyniik., 46: 1485, 1922. SCHWARTZ, J. Beitrag zum Vorkommen d. EiIeiterdrehung. Zentralbl. J. Gyniik., 46: 1959, 1922. SCHWEITZER, B. IsoIierte Torsion d. normalen Tube. Zentralbl. J. Gyniik., 42: 25, 1918. SELLHEIM, H. ErkILrung d. StieItorsion von Eierstockcysten. Arch. j. Gyniik., 117: 27, 1922. STARK, J. N. Acute torsion of norma appendages.with haematosaIpinx. J. Obst. @ Gynaec. Brit. Emp., Ig: 258, IgI I. TAUBER, R. StieIgedrehte Tube in der Schwangerschaft. Zentralbl. J. Gyntik., 3 I : 2813, 1927. VEIT, J. Handbuch d. GynBkoIogie, I. Audi. Wiesbaden, 1899. J. F. Bregmann.

American

Journal of Surgery