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Torsion of the Appendix Testis (Hydatid of Morgagni)1
TORSION OF THE APPENDIX TESTIS (HYDATID OF MORGAGNI) 1 ALEXANDER RANDALL
This interesting clinical-pathological entity is being presented for several reasons: (1), to bring the reported cases up to date (end of 1937) since the excellent presentation of Dix in 1931; (2), to include additional cases; (3), to try to correct the earlier statements that the iesion occurred only in childhood and adolescence; (4), to accentuate the varying pathology; and (5), because the American medical literature seems peculiarly silent on observations of the condition, whose prompt diagnosis and surgical treatment will save prolonged suffering. History. Colt is credited with publishing in 1922 the first accurate description of an operated case; and his presentation immediately brought case reports from Shattock and from Walton, each of whom had operated upon such a case in 1918 and 1913 respectively. Shattock's observation remains the only one where 2 hydatids on a single testis were each undergoing acute torsion. Ombredanne in 1913 also saw the lesion in his fifth reported case, but confused the picture with torsion testis (of which he was writing), though he specifically stated that testicular torsion was not present in this case. The constant publications of Mouchet since 1923 give to him the credit for focusing attention on the clinical picture, the true pathology, the ultimate significance and the necessity of prompt surgical treatment. He has personally observed 13 accredited cases (Dix). In 1931 Dix published all cases to the end of 1930, and tabulated 42 cases then in the world's literature. He added 3 cases in which the clinical picture was similar, but the causative factor lay in torsion of other vestigial structures. To this total of 45 he added his 2 personal cases (1 vestigial) and 6 additional unpublished ones from Mouchet (personal communication), bringing the grand total to 53, of which 49 were torsion of the appendix testis, and 4 of other vestigial structures as follows: 2 of the appendix of epididymis, 1 of the organ of Giraldes, and 1 of the vas aberrans of Haller. The literature from 1931 to 1937, inclusive, I have found to contain 17 reported cases of simple torson of the appendix testis and 1 case of 1 Read before annual meeting of American Association of Genito-Urinary Surgeons, Atlantic City, May 3, 1938.
715
716
ALEXANDER RANDALL
torsion of a cyst of the vas aberrans. This brings the total in the literature to 71, of which 66 are true torsion of the hydatid of Morgagni (appendix testis) and 5 of other vestigial structures. To this list I.wish to add 2 cases of torsion of the appendix testis; so that the tabulation to date records : cases
Torsion of the appendix testis (Morgagni) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 68 Torsion of the appendix epididymis .. .. ..... . .. . .. . . .. .... ... . . . . . ... . ... 2 Torsion of the organ of Giraldes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1 Torsion of the vas aberrans. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2 Total. .... . . . . ... ... . ... . .. . . .. . . . .. . . . .. ... . .. ..... . . . . . .. . .. .. .... 732
That there have been only 2 articles in the American literatureFoshee (2 cases) and Rhodes (2 cases)-seems worthy of especial remark, and probably many have been unappreciated and simply considered epidido-orchitis of undertermined origin. Such is a pity, as prolonged, or recurrent, invalidism has been repeatedly observed and reported. Anatomy. Gross anatomy recognizes that the sessile hydatid of Morgagni is the most constantly present rudimentary structure of that group which owe their origin to vestigial remains of the Mtillerian duct, or to embryonic faults of development. Situated on the anterior face of the superior pole of the testis, it lies free of the overhanging globus major of the epididymis. Paradoxically, it is more often pedunculated than sessile, and its proper title is the appendix testis. The appendix epididymis, often called the stalked hydatid, is less constantly present, and arises from the tubules of the epigenitalis that do not form tubtili collectivi. It is said to contain plain muscle in its stalk. The paradidymis, or organ of Giraldes, is composed of a group of . rudimentary tubules within the structures of the first portion of the cord, and lesions therefrom appear separate from both the testis and the epididymis. These tubules represent embryonic remnants of the paragenitalis, or vestiges of the Wolffian body. The vasa aberrantia of Haller are located in the middle of the epididymo-testicular groove, and are again embryonic elements that have failed to consummate their total development, and have their origin probably from the Wolffian duct. 2 In a brief discussion of both torsion testis and torsion of the appendix testis Sorrel reports that he has observed 7 cases of torsion of the hydatid of Morgagni and 2 cases of torsion of the organ of Giraldes, giving the ages from "4½ to 13 years," but without recording any further data. Including these, as Dix did those of Mouchet, the grand total becomes 82, with torsion of the appendix testis alone represented by 75 cases.
TORSION OF THE APPENDIX TESTIS
717
It is of interest to recall that similar vestigial remnants occur in the female . An hydatid of Morgagni, properly termed the appendix vesiculosa, is found below the fimbriated end of the Fallopian tube, but here it represents the vestigial remains of the Wolffian duct. Putzudonedder presents a complete discussion of the anatomy and pathology of the female hydatid, while Henrich reports a case of torsion in this hydatid and quotes 3 further cases from the literature. Microscopically the hydatid of Morgagni is covered with simple cylindrical epithelium and is composed of loose fibrous tissue, with blood vessels and some plain muscle fibers, and usually there are found remnants of a duct (Mi.illerian), lined by ciliated epithelium. Pathology. It is a bit confusing, as one reads the brief case reports, to classify each case clearly, and to determine whether torsion was present or not. To begin with, repeated attacks, with evidence of detorsion, have been recognized-Guillemin and Grandineau, Roche, Tavernier and Garin. This represents the "formes frustes" of Mouchet. Secondly, the term "infarction" is used-Guerin, Blauchat, Foshee-a distinction one would consider difficult to make, even microscopically, after 5 days' duration, as in one of Foshee's cases. Thirdly, the diagnosis of fibroma has been given-Salmon and Contiades-and, again, fibroma of the hydatid without torsion-Salmon, Mosinger and Contiades, and Glass. Cyst of the hydatid- Meyer-Wildisen- 6½ cm. in diameter is reported. This confusion, however, is of academic interest only, and for the clinician the picture remains very much the same, and the therapy indicated is aways operative interference. It has been necessary to exclude from the series those cases which do not present acute torsion as the major clinical-pathological condition, and to include only those where this factor of torsion is present, or where repeated attacks with detorsion have been recognized. Clinical picture. The clinical picture (table 1) has some confusing points. The onset is practically always sudden, with unilateral acute pain. The pain is rarely so severe or so continuous as that accompanying acute infectious epididymitis, but palpation is distressing, and in children the pain is probably deceptively exaggerated. A confusing symptom is a concomitant redness and edema of the scrotum at the onset of the attack, and in children especially there is a slight elevation of temperature that makes one think of an infectious process. Ruling out a urethritis, mumps, tuberculosis, or other general disorders, clears this side of the diagnostic picture.
TABLE
!.-Clinical findings -.:i .....
PHYSICAL SIGNS
00
YEAR NO.
OF PUBLI·
OBSERVER
AGE
TESTIS AFFECTED
DUJ1.ATION
Local conditlon Temp.
CATION
Appearance of scrotum
-- - -
Cord
Palpation of testis
days
1
1931
Guilleminet
12
Left
Normal
Left
Repeated attacks. Many days 5
2
1932
Foshee
16 mo.
3 4
1932 1933
5 6 7 8 9 10 11 12 13
1933 1934 1935 1935 1935 1935 1935 1935 1935
14
1936
Foshee Guillemin and Grandineau Inouye Chiariello Blauchat Guerin Moncalvi Rhodes Rhodes Roche Rochet and Pouyanni Grasso
8 10½
Left Left
3 14
33 12
Left Left
1
4½ 9 39 11 14 15
Left Right Left Left Right Right
1 3 28 2 2 3
11
Right
3
37.3
15 16 17
1936 1936 1936
18 19
Normal
Normal
Tender at upper pole
98 .8 39.5
Double size and red Slight thickening Swollen and red Normal Swollen Normal
Enlarged, firm and tender Firm and tender Swollen, not tender
39 37.4
Red and swollen
Normal
Painful Swollen
Swollen and red Testis in canal Swollen Red and swollen Normal Tender and unilateral swelling Oedema and red
Swollen
100
38.2 Normal 100 99
Normal Normal
Tender Tender, swollen Not palpable Very tender Normal Tender Swollen and tender. Epid. not enlarged Double size Enlarged Tender
4 11 13
Left Left Left
4 7 Repeated attacks
Normal Normal Normal
Swollen Swollen and red Slight oedema
1938
Olascoaga Olascoaga Tavernier and Garin Randall
Thick and tender Normal Very painful Normal
34
Left
Normal
Normal
Normal
Tender
1938
Rosenblum
12
Right
Repeated attacks, 3 days 3
Red and oedematous
Normal
Tender at upper pole
99
I t-,j ~
':d
~
~
r'
TORSION OF THE APPENDIX TESTIS
719
Complete torsion of the testis presents greater difficulty; though this condition is almost uniformly of greater symptomatic severity, and, fortunately, this differential diagnosis is not essential since prompt operative interference is the treatment of choice in both conditions. In cases of a few days' duration the diagnosis becomes easier, and unless edema persists, or a secondary hydrocele collection interferes, the spot tenderness at the superior pole of the testis and just under the globus major of the epididymis is diagnostic. With rare exception, general systemic symptoms are absent, and though fever is usual at onset, chill, nausea, vomiting, etc., are rarely recorded. Inouye's patient, who had 14 attacks in 16 years, is of interest, in that frequency of urination regularly occurred with each attack. Roche likewise reports urinary frequency and the unique radiation of the pain down the thigh; while the case reported by Tavernier and Garin was long undiagnosed, as his pain in numerous attacks was regularly across the lower abdomen, until the last one, when the testicular character of the pain was first recognized. As Dix says, when the condition is thought of, diagnosis presents no insuperable difficulties, and it has been made correctly and without reserve in a number of the published cases. It is certain, however, that many cases of this lesion are unrecognized, and drag out a long and painful course before resolution and a natural cure occur. Three cases in Dix' tabulation show the lesion in ectopic testes; and one in my tabulation (Moncalvi) had both testes ectopic and palpable in the groins, with the right one involved in a torsion of the hydatid. Age. The early reports made the condition one of infancy, childhood and adolescence, and Mouchet wrote in 1925, "I am unable to explain why torsion of the hydatid of Morgagni presents itself exclusively in the infant and adolescent." Probably the absence of infectious conditions of the genitalia in this young group brought the diagnosis more readily to mind. In Dix' statistics 27 of the 42 cases (64 per cent) he collected from the literature occurred in boys between the ages of 11 and 14, and only 3 were past their twentieth birthday. The 6 cases of Mouchet, as yet unpublished, were of unrecorded age. In my additional 19 cases, 3 were older than 20 years. As changes prior to and at puberty are thought to influence this torsion, it would be proper to take those past 16 years as a criterion; and this shows that in the total series of 73 cases, 11 (15 per cent) were post-puberal. Etiology. The cause is still unknown. Certainly infection plays no part. Trauma has been discussed; sudden or violent cremasteric reflex
720
ALEXANDER RANDALL
response suggested; but too many cases occur during ordinary play or occupation to allow such to stand. A few have occurred during sleep, as in torsion testis. It is suggested, apropos of the marked frequency during adolescence, that increased vascularity with testicular growth and development may play an important role. Certainly those in whom the hydatid has a greater pedicle development become potentially more liable; and my Case 1 demonstrates this, in that the hydatid from the opposite testis was far from sessile. Case 1. R. B., age 23, single, consulted me December 16, 1936, complaining of repeated attacks of acute left testicular pain of only 10 to 15 minutes' duration. His first attack was 3 months before, on September 14, 1936, following a game of tennis, which he played in rather tight "shorts." He awoke at 5 o'clock the following morning with severe left testicular pain and a generalized ache over the entire lower left abdomen, with radiation down the left thigh. At 9 o'clock, he consulted a physician, who advised a suspensory, and his pain was gone by 3 in the afternoon. Since this primary attack he had worn a suspensory regularly, and had had but 1 other attack of moderate severity. At times exercise would develop an ache, and at others really strenuous exercise would cause him no discomfort. Long standing would cause an ache to develop, at times growing to be a constant annoyance. Three days before the consultation an attack of acute pain had lasted 2 hours, and then subsided entirely. He was otherwise enjoying excellent health, and planned to be married in 3 months. As his fiancee lived elsewhere, sexual excitement had played no part in the attacks. An examination was completely negative: there was no pathological change in the testis or the epididymis, and no hernia or varicocele. It was considered a case of torsion and detorsion of testis, and an operation for bilateral fixation was advised. Operation, December 18, 1936: Gas anesthesia; incision of right scrotum and testicular tunics (normal side). The testis was delivered, and appeared normal throughout; except that the hydatid testis appeared double normal in size, 1½ cm. long, and from its extremity there hung a gelatinous mass fully 1 cm. in length. This abnormality, which had been symptomless, was excised, and the pedicle cauterized with the high-frequency cautery. The wound was closed without drainage, after scarifying the testicular surface and taking two catgut sutures to include it while closing its tunica. A similar scrotal incision was made on the left, where, on opening the tunica vaginalis, this startling picture was found: The stem of the appendix testis was fully 8 mm. in length, twisted 1½ turns clockwise, and banded a dark red at the point of greatest constriction. Proximal and distal to this point many tiny cysts could be seen. Pendant from this twisted stem was a tonguelike gelatinous portion, fully 2½ cm. in length and approximately 8 mm. in
TORSION OF THE APPENDIX TESTIS
721
thickness. It hung so that its lower extremity was well below the middle of the testicular body. This was excised, and the wound closed without drainage, as on the opposite side. Cure has been permanent. Microscopic sections (fig. 1) showed an organ for the most part devoid of its epithelial covering, though in a few places remnants were found, and consisted of low cuboidal cells occurring in single layers in some places, but in others 2 to 3 cells deep. Underneath the surface there was abundant stroma, composed of loose connective tissue which was in a state of fairly advanced degeneration, with only an occasional cell nucleus taking the hematoxylin
FIG. 1. Case 1. Microscopic section through area of torsion of pedicle of left hydatid
stain. There was quite marked edema, and areas showing deposits of hemosiderin, indicating old hemorrhage. Only here and there were seen a few red blood cells lying in blood vessels with degenerated walls. There was some evidence of proliferation of fibroblasts and a few round cells. Scattered through the stroma were seen numerous tortuous, irregular spaces, whose walls contained large deposits of calcium salts, and whose lumina were for the most part empty, except for necrotic tissue and fragments of calcium deposition. These spaces might have been the remnants of ductal structures. The picture suggests that this organ had been deprived of its normal blood supply for considerable time, and that the pathologic changes noted above were secondary to strangulation.
722
ALEXANDER RANDALL
I wish to record the following case, but as no operation was performed, verification is lacking. Case 2. A. B., age 34, married, asked me to see him at home in October 1934. Twelve days before he was suddenly seized, while at his desk, with acute right testicular pain. It was so severe that he was hurried home, and required morphia to quiet him. The following day it was still exceedingly tender on any motion, but eased under some local applications, plus heat. The day I saw him he had been out of bed for the first time, and was comfortable with a supporter, though the testis was still sensitive to pressure. As 4 physicians had made 4 different diagnoses (epididymitis, hernia, gonorrhea and appendicitis), he was concerned about his condition. An examination revealed a perfectly normal scrotum, testicle and epididymis; there was neither hernia nor history of urethritis. Palpating the right testis, one could feel with the greatest ease a small, firm mass, the size of a grain of wheat, on the anterior testicular surface just below the head of the epididymis. Pressure thereon reproduced identically the pain of complaint. Operation was advised, but declined when assured that it could not be cancer, and that in all probability the hydatid was then dead and would trouble him no more with acute pain. Case 3. 3 A child, 12 years of age, was seen February 10, 1938, 3 days after the onset of an acute painful swelling of the right scrotum and its contents, and with a temperature of 99°F. An examination showed the swelling to be limited to the upper half of the scrotum and to the globus major of the epididymis. The scrotal skin was reddened, and palpation elicited acute pain. The cord was free of any involvement in the picture, and torsion was not demonstrable. Actually, the testicular body could be gently palpated without acute pain being elicited. A diagnosis of torsion testis was made, and immediate operation advised and accepted. Operation, February 13, 1938: Gas anesthesia. An incision was made through the scrotal walls and tunica vaginalis, which exposed a normal testis and slightly congested epididymis. The hydatid of Morgagni was of a deep purple color, verging on gangrene, and the size of a large pea. It appeared stalked, with the distal two-thirds dry and verging on necrosis. No definite torsion was demonstrated, though looked for, but the appearance was so characteristic that it was considered that recent detorsion had occurred. It was excised, and no bleeding ensued. The wound was closed in layers with fine catgut. Recovery was prompt and cure established. Microscopic sections showed extensive hemorrhagic extravasation and quite generalized cellular necrosis. 3 Personal communication from Dr. Philip Rosenblum, and to be published later. It was presented by him before the joint meeting of the Philadelphia and New York Urological Societies, April 21, 1938.
TABL E
2.-0perative findings APPENDIX OF TESTIS
NO .
OBSERVER
DAY OF ATTACK
CORD
FLUID CONTENTS
TESTIS
EPIDIDYMIS
Size
Color
Turns
Direction
days
1
Guilleminet
Many
Normal
Serum
Normal
Normal
2 3
Foshee Foshee
5 3
Oedema Oedema
Normal Twice normal
Oedema
Clear yellow
4 5 6
Guillemin and 3rd attack Grandineau in 14da. Inouye 15 Chiariello 3
Normal
Brown
7 8 9
Blauchat Guerin Moncalvi
Slightly hemorrhagic
10
Rhodes
11 12 13
2 2 3
14
Rhodes Roche Rochet and Pouyanni Grasso
Congested Hemorrhagic Congenital Serohemorhernia rhagic Normal 4 oz. hemorrhagic Normal Yellow Normal Yellow Normal
3
Normal
15
Olascoaga
4
Normal
16 17
Olascoaga Tavernier and Garin Randall Rosenblum
7
Normal Normal
18 19
1 3
Normal
28
3 3
Normal Normal
Serohemorrhagic Amber Amber Serohemorrhagic Amber Amber
Grain of wheat 1cm. 1 cm.
Ochre Blue-black Blue-black
3
Clockwise
8mm.
Black
11.2
Clockwise
1½ cm. Pea
Dark red Black
1
Clockwise
Haddetorsioned
.., 0 ...rn ~
Enlarged
~ ~
~
Congested Ectopic in canal Normal
Congested Oedema
1cm. Small
Blue Red
Normal
3cm.
Necrotic
Normal Normal Inverted
Normal Normal
2½ cm. Cherry
Bluish Purple red Black
2 2 1
Normal
Normal
Large pea
Black blue
+
Double normal Normal Normal
Normal Normal
Normal Normal
Normal Normal
I
+
~
Clockwise Clockwise Counterclockwise
~
;
rn
Enlarged Enlarged Double normal 2cm. Large pea
Red Violet Red Deep purple
Many times 1½
~
Clockwise
~
~
724
ALEXANDER RANDALL
Torsion of other vestigial structures. In this period from 1931 to 1937, inclusive, the available literature contains 2 reports of this character:
Case 1 (Patch): Report of a case of torsion of a cyst arising from the vas aberrentia. Case 2 (Sorrel): Reports two cases of torsion of the organ of Giraldes.2 Treatment. The operation is so simple, and so devoid of any serious complications, that it should always be advised. In that I was able to demonstrate so clearly, in my Case 1, the bilateral development of unusually long hydatids, it would seem advisable to operate on both sides in any given case. The :fixation of the testis to the incision in the tunica vaginalis, when sewing up, I should likewise advise as a step to prevent subsequent torsion testis, as these 2 conditions have been reported as occurring simultaneously (table 2). The question of conservative- non-operative- treatment is ill-advised: (1), because no local treatment relieves the distress; (2), because detorsion and recurrent attacks are possible; (3), because the nonoperative recovery can take longer than the operative removal and convalescence; and (4), we have the suggestion of Contiades and Merigot, who report 2 cases where they felt evidence to exist that a hydrocele collection was secondary to a small foreign body found within the tunica vaginalis, and in each case the history gave evidence that torsion of the hydatid had preceded the development of the hydrocele. CONCLUSIONS
Two additional cases of torsion of the appendix testis are reported; bringing the total to sixty-eight cases in the literature to the end of 1937.3 It is of moment that only 2 American authors have reported cases. It is assumed that the diagnosis is being missed, and masquerading under a faulty interpretation of the symptoms and signs. Treatment is surgical and, as a preventive step, should be performed on both sides. Medical Arts Bldg., Philadelphia, Pa. REFERENCES BLAUCHAT: Bordeaux Chir., 6: 159, 1935. CmARIELLO: Pediatria, 44: 1104, 1934. COLT: Brit. J. Surg., 9: 464, 1922. CONTIADES AND MERIGOT: Ann. d'Anat. Path. Med. Chir., 9: 795, 1932. DIX: Brit. J. Urol., 3: 245, 1931.
TORSION OF THE APPENDIX TESTIS
725
Doc: Ztschr. f . Urol. Chir., 33: 486, 1931. - -: Ztschr. f. Urol., 26: 408, 1932. FOSHEE : J. A. M.A., 99: 389, 1932. , GLASS : Quoted by Salmon, Mosinger and Contiades. GUERIN: Bordeaux Chir., 6: 157, 1935. GurLLEMIN AND GRANDINEAU : Rev. med. de !'est, 61: 826, 1933. Gu1LLEMINET: Lyon Chir., 23: 821 , 1931. GRASSO: Arch. Ital. d. Chir., 43: 221, 1936. HENRICH: Amer. J . Obst., 21: 121, 1931. INOUYE : Abst. Jap. J . Derm. and Urol., 33: 47, 1933. MEYER-WILDISEN : Beitr. z. klin. Chir., 164: 47, 1936. MoNCALVI: J. d'urol. med. et chir., 36: 501, 1933. MoucHET : Presse med., 31: 485, 1923. MoucHET: Bull. et mem. de la Soc. Nat. de Chir., 61: 431, 1925. OLASCOAGA : Semana med., 2: 1484, 1936. OMBREDANNE: Bull. et mem. de la Soc. Nat. de Chir., 39: 779, 1913. PATCH: Brit. J. Urol., 6: 122, 1933. PUTZUDONEDDER : Pathologica, 29: 98, 1937. RHODES: New Eng. J. Med., 213: 1005, 1935. ROCHE: Clin. J., 64: 96, 1935. ROCHET AND POUYANNI: Bordeaux Chir., 6: 135, 1935. SALMON, MosINGER AND CONTIADES : Ann. de Anat. and Path., 10: 490, 1933. SALMON AND CONTIADES: J. d'urol. med. et chir., 37: 412, 1934. SHATTOCK: Lancet, 1: 693, 1922. SORREL: Bull. et mem. de la Soc. Nat. de Chir., 61: 1270, 1935. TAVERNIER AND GARIN : Lyon Chir., 33: 598, 1936. WALTON: Brit. J. Surg., 10: 151, 1922. The following references could not be obtained for review; or the given data was incomplete or in error, as they could not be found in the Index Medicus: ALLENDE : Rev. de chir. de Bs., 1931. CONSTANTINI : Soc. de Chir. d'Alger, Jan. 19, 1933. DIAz, CASTRO, BONNECARRERE : Med. Soc. Urug. d . Urol., 2: 36, 1936. GAMBOA y NEIDELMAN : Semana med., 1931. SERANTES, MONSERRAT AND GARCIA : Revista Argentina de Urol., p. 196, 1936.
This interesting clinical-pathological entity is being presented for several reasons: (1), to bring the reported cases up to date (end of 1937) since the excellent presentation of Dix in 1931; (2), to include additional cases; (3), to try to correct the earlier statements that the iesion occurred only in childhood and adolescence; (4), to accentuate the varying pathology; and (5), because the American medical literature seems peculiarly silent on observations of the condition, whose prompt diagnosis and surgical treatment will save prolonged suffering. History. Colt is credited with publishing in 1922 the first accurate description of an operated case; and his presentation immediately brought case reports from Shattock and from Walton, each of whom had operated upon such a case in 1918 and 1913 respectively. Shattock's observation remains the only one where 2 hydatids on a single testis were each undergoing acute torsion. Ombredanne in 1913 also saw the lesion in his fifth reported case, but confused the picture with torsion testis (of which he was writing), though he specifically stated that testicular torsion was not present in this case. The constant publications of Mouchet since 1923 give to him the credit for focusing attention on the clinical picture, the true pathology, the ultimate significance and the necessity of prompt surgical treatment. He has personally observed 13 accredited cases (Dix). In 1931 Dix published all cases to the end of 1930, and tabulated 42 cases then in the world's literature. He added 3 cases in which the clinical picture was similar, but the causative factor lay in torsion of other vestigial structures. To this total of 45 he added his 2 personal cases (1 vestigial) and 6 additional unpublished ones from Mouchet (personal communication), bringing the grand total to 53, of which 49 were torsion of the appendix testis, and 4 of other vestigial structures as follows: 2 of the appendix of epididymis, 1 of the organ of Giraldes, and 1 of the vas aberrans of Haller. The literature from 1931 to 1937, inclusive, I have found to contain 17 reported cases of simple torson of the appendix testis and 1 case of 1 Read before annual meeting of American Association of Genito-Urinary Surgeons, Atlantic City, May 3, 1938.
715
716
ALEXANDER RANDALL
torsion of a cyst of the vas aberrans. This brings the total in the literature to 71, of which 66 are true torsion of the hydatid of Morgagni (appendix testis) and 5 of other vestigial structures. To this list I.wish to add 2 cases of torsion of the appendix testis; so that the tabulation to date records : cases
Torsion of the appendix testis (Morgagni) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 68 Torsion of the appendix epididymis .. .. ..... . .. . .. . . .. .... ... . . . . . ... . ... 2 Torsion of the organ of Giraldes . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1 Torsion of the vas aberrans. . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2 Total. .... . . . . ... ... . ... . .. . . .. . . . .. . . . .. ... . .. ..... . . . . . .. . .. .. .... 732
That there have been only 2 articles in the American literatureFoshee (2 cases) and Rhodes (2 cases)-seems worthy of especial remark, and probably many have been unappreciated and simply considered epidido-orchitis of undertermined origin. Such is a pity, as prolonged, or recurrent, invalidism has been repeatedly observed and reported. Anatomy. Gross anatomy recognizes that the sessile hydatid of Morgagni is the most constantly present rudimentary structure of that group which owe their origin to vestigial remains of the Mtillerian duct, or to embryonic faults of development. Situated on the anterior face of the superior pole of the testis, it lies free of the overhanging globus major of the epididymis. Paradoxically, it is more often pedunculated than sessile, and its proper title is the appendix testis. The appendix epididymis, often called the stalked hydatid, is less constantly present, and arises from the tubules of the epigenitalis that do not form tubtili collectivi. It is said to contain plain muscle in its stalk. The paradidymis, or organ of Giraldes, is composed of a group of . rudimentary tubules within the structures of the first portion of the cord, and lesions therefrom appear separate from both the testis and the epididymis. These tubules represent embryonic remnants of the paragenitalis, or vestiges of the Wolffian body. The vasa aberrantia of Haller are located in the middle of the epididymo-testicular groove, and are again embryonic elements that have failed to consummate their total development, and have their origin probably from the Wolffian duct. 2 In a brief discussion of both torsion testis and torsion of the appendix testis Sorrel reports that he has observed 7 cases of torsion of the hydatid of Morgagni and 2 cases of torsion of the organ of Giraldes, giving the ages from "4½ to 13 years," but without recording any further data. Including these, as Dix did those of Mouchet, the grand total becomes 82, with torsion of the appendix testis alone represented by 75 cases.
TORSION OF THE APPENDIX TESTIS
717
It is of interest to recall that similar vestigial remnants occur in the female . An hydatid of Morgagni, properly termed the appendix vesiculosa, is found below the fimbriated end of the Fallopian tube, but here it represents the vestigial remains of the Wolffian duct. Putzudonedder presents a complete discussion of the anatomy and pathology of the female hydatid, while Henrich reports a case of torsion in this hydatid and quotes 3 further cases from the literature. Microscopically the hydatid of Morgagni is covered with simple cylindrical epithelium and is composed of loose fibrous tissue, with blood vessels and some plain muscle fibers, and usually there are found remnants of a duct (Mi.illerian), lined by ciliated epithelium. Pathology. It is a bit confusing, as one reads the brief case reports, to classify each case clearly, and to determine whether torsion was present or not. To begin with, repeated attacks, with evidence of detorsion, have been recognized-Guillemin and Grandineau, Roche, Tavernier and Garin. This represents the "formes frustes" of Mouchet. Secondly, the term "infarction" is used-Guerin, Blauchat, Foshee-a distinction one would consider difficult to make, even microscopically, after 5 days' duration, as in one of Foshee's cases. Thirdly, the diagnosis of fibroma has been given-Salmon and Contiades-and, again, fibroma of the hydatid without torsion-Salmon, Mosinger and Contiades, and Glass. Cyst of the hydatid- Meyer-Wildisen- 6½ cm. in diameter is reported. This confusion, however, is of academic interest only, and for the clinician the picture remains very much the same, and the therapy indicated is aways operative interference. It has been necessary to exclude from the series those cases which do not present acute torsion as the major clinical-pathological condition, and to include only those where this factor of torsion is present, or where repeated attacks with detorsion have been recognized. Clinical picture. The clinical picture (table 1) has some confusing points. The onset is practically always sudden, with unilateral acute pain. The pain is rarely so severe or so continuous as that accompanying acute infectious epididymitis, but palpation is distressing, and in children the pain is probably deceptively exaggerated. A confusing symptom is a concomitant redness and edema of the scrotum at the onset of the attack, and in children especially there is a slight elevation of temperature that makes one think of an infectious process. Ruling out a urethritis, mumps, tuberculosis, or other general disorders, clears this side of the diagnostic picture.
TABLE
!.-Clinical findings -.:i .....
PHYSICAL SIGNS
00
YEAR NO.
OF PUBLI·
OBSERVER
AGE
TESTIS AFFECTED
DUJ1.ATION
Local conditlon Temp.
CATION
Appearance of scrotum
-- - -
Cord
Palpation of testis
days
1
1931
Guilleminet
12
Left
Normal
Left
Repeated attacks. Many days 5
2
1932
Foshee
16 mo.
3 4
1932 1933
5 6 7 8 9 10 11 12 13
1933 1934 1935 1935 1935 1935 1935 1935 1935
14
1936
Foshee Guillemin and Grandineau Inouye Chiariello Blauchat Guerin Moncalvi Rhodes Rhodes Roche Rochet and Pouyanni Grasso
8 10½
Left Left
3 14
33 12
Left Left
1
4½ 9 39 11 14 15
Left Right Left Left Right Right
1 3 28 2 2 3
11
Right
3
37.3
15 16 17
1936 1936 1936
18 19
Normal
Normal
Tender at upper pole
98 .8 39.5
Double size and red Slight thickening Swollen and red Normal Swollen Normal
Enlarged, firm and tender Firm and tender Swollen, not tender
39 37.4
Red and swollen
Normal
Painful Swollen
Swollen and red Testis in canal Swollen Red and swollen Normal Tender and unilateral swelling Oedema and red
Swollen
100
38.2 Normal 100 99
Normal Normal
Tender Tender, swollen Not palpable Very tender Normal Tender Swollen and tender. Epid. not enlarged Double size Enlarged Tender
4 11 13
Left Left Left
4 7 Repeated attacks
Normal Normal Normal
Swollen Swollen and red Slight oedema
1938
Olascoaga Olascoaga Tavernier and Garin Randall
Thick and tender Normal Very painful Normal
34
Left
Normal
Normal
Normal
Tender
1938
Rosenblum
12
Right
Repeated attacks, 3 days 3
Red and oedematous
Normal
Tender at upper pole
99
I t-,j ~
':d
~
~
r'
TORSION OF THE APPENDIX TESTIS
719
Complete torsion of the testis presents greater difficulty; though this condition is almost uniformly of greater symptomatic severity, and, fortunately, this differential diagnosis is not essential since prompt operative interference is the treatment of choice in both conditions. In cases of a few days' duration the diagnosis becomes easier, and unless edema persists, or a secondary hydrocele collection interferes, the spot tenderness at the superior pole of the testis and just under the globus major of the epididymis is diagnostic. With rare exception, general systemic symptoms are absent, and though fever is usual at onset, chill, nausea, vomiting, etc., are rarely recorded. Inouye's patient, who had 14 attacks in 16 years, is of interest, in that frequency of urination regularly occurred with each attack. Roche likewise reports urinary frequency and the unique radiation of the pain down the thigh; while the case reported by Tavernier and Garin was long undiagnosed, as his pain in numerous attacks was regularly across the lower abdomen, until the last one, when the testicular character of the pain was first recognized. As Dix says, when the condition is thought of, diagnosis presents no insuperable difficulties, and it has been made correctly and without reserve in a number of the published cases. It is certain, however, that many cases of this lesion are unrecognized, and drag out a long and painful course before resolution and a natural cure occur. Three cases in Dix' tabulation show the lesion in ectopic testes; and one in my tabulation (Moncalvi) had both testes ectopic and palpable in the groins, with the right one involved in a torsion of the hydatid. Age. The early reports made the condition one of infancy, childhood and adolescence, and Mouchet wrote in 1925, "I am unable to explain why torsion of the hydatid of Morgagni presents itself exclusively in the infant and adolescent." Probably the absence of infectious conditions of the genitalia in this young group brought the diagnosis more readily to mind. In Dix' statistics 27 of the 42 cases (64 per cent) he collected from the literature occurred in boys between the ages of 11 and 14, and only 3 were past their twentieth birthday. The 6 cases of Mouchet, as yet unpublished, were of unrecorded age. In my additional 19 cases, 3 were older than 20 years. As changes prior to and at puberty are thought to influence this torsion, it would be proper to take those past 16 years as a criterion; and this shows that in the total series of 73 cases, 11 (15 per cent) were post-puberal. Etiology. The cause is still unknown. Certainly infection plays no part. Trauma has been discussed; sudden or violent cremasteric reflex
720
ALEXANDER RANDALL
response suggested; but too many cases occur during ordinary play or occupation to allow such to stand. A few have occurred during sleep, as in torsion testis. It is suggested, apropos of the marked frequency during adolescence, that increased vascularity with testicular growth and development may play an important role. Certainly those in whom the hydatid has a greater pedicle development become potentially more liable; and my Case 1 demonstrates this, in that the hydatid from the opposite testis was far from sessile. Case 1. R. B., age 23, single, consulted me December 16, 1936, complaining of repeated attacks of acute left testicular pain of only 10 to 15 minutes' duration. His first attack was 3 months before, on September 14, 1936, following a game of tennis, which he played in rather tight "shorts." He awoke at 5 o'clock the following morning with severe left testicular pain and a generalized ache over the entire lower left abdomen, with radiation down the left thigh. At 9 o'clock, he consulted a physician, who advised a suspensory, and his pain was gone by 3 in the afternoon. Since this primary attack he had worn a suspensory regularly, and had had but 1 other attack of moderate severity. At times exercise would develop an ache, and at others really strenuous exercise would cause him no discomfort. Long standing would cause an ache to develop, at times growing to be a constant annoyance. Three days before the consultation an attack of acute pain had lasted 2 hours, and then subsided entirely. He was otherwise enjoying excellent health, and planned to be married in 3 months. As his fiancee lived elsewhere, sexual excitement had played no part in the attacks. An examination was completely negative: there was no pathological change in the testis or the epididymis, and no hernia or varicocele. It was considered a case of torsion and detorsion of testis, and an operation for bilateral fixation was advised. Operation, December 18, 1936: Gas anesthesia; incision of right scrotum and testicular tunics (normal side). The testis was delivered, and appeared normal throughout; except that the hydatid testis appeared double normal in size, 1½ cm. long, and from its extremity there hung a gelatinous mass fully 1 cm. in length. This abnormality, which had been symptomless, was excised, and the pedicle cauterized with the high-frequency cautery. The wound was closed without drainage, after scarifying the testicular surface and taking two catgut sutures to include it while closing its tunica. A similar scrotal incision was made on the left, where, on opening the tunica vaginalis, this startling picture was found: The stem of the appendix testis was fully 8 mm. in length, twisted 1½ turns clockwise, and banded a dark red at the point of greatest constriction. Proximal and distal to this point many tiny cysts could be seen. Pendant from this twisted stem was a tonguelike gelatinous portion, fully 2½ cm. in length and approximately 8 mm. in
TORSION OF THE APPENDIX TESTIS
721
thickness. It hung so that its lower extremity was well below the middle of the testicular body. This was excised, and the wound closed without drainage, as on the opposite side. Cure has been permanent. Microscopic sections (fig. 1) showed an organ for the most part devoid of its epithelial covering, though in a few places remnants were found, and consisted of low cuboidal cells occurring in single layers in some places, but in others 2 to 3 cells deep. Underneath the surface there was abundant stroma, composed of loose connective tissue which was in a state of fairly advanced degeneration, with only an occasional cell nucleus taking the hematoxylin
FIG. 1. Case 1. Microscopic section through area of torsion of pedicle of left hydatid
stain. There was quite marked edema, and areas showing deposits of hemosiderin, indicating old hemorrhage. Only here and there were seen a few red blood cells lying in blood vessels with degenerated walls. There was some evidence of proliferation of fibroblasts and a few round cells. Scattered through the stroma were seen numerous tortuous, irregular spaces, whose walls contained large deposits of calcium salts, and whose lumina were for the most part empty, except for necrotic tissue and fragments of calcium deposition. These spaces might have been the remnants of ductal structures. The picture suggests that this organ had been deprived of its normal blood supply for considerable time, and that the pathologic changes noted above were secondary to strangulation.
722
ALEXANDER RANDALL
I wish to record the following case, but as no operation was performed, verification is lacking. Case 2. A. B., age 34, married, asked me to see him at home in October 1934. Twelve days before he was suddenly seized, while at his desk, with acute right testicular pain. It was so severe that he was hurried home, and required morphia to quiet him. The following day it was still exceedingly tender on any motion, but eased under some local applications, plus heat. The day I saw him he had been out of bed for the first time, and was comfortable with a supporter, though the testis was still sensitive to pressure. As 4 physicians had made 4 different diagnoses (epididymitis, hernia, gonorrhea and appendicitis), he was concerned about his condition. An examination revealed a perfectly normal scrotum, testicle and epididymis; there was neither hernia nor history of urethritis. Palpating the right testis, one could feel with the greatest ease a small, firm mass, the size of a grain of wheat, on the anterior testicular surface just below the head of the epididymis. Pressure thereon reproduced identically the pain of complaint. Operation was advised, but declined when assured that it could not be cancer, and that in all probability the hydatid was then dead and would trouble him no more with acute pain. Case 3. 3 A child, 12 years of age, was seen February 10, 1938, 3 days after the onset of an acute painful swelling of the right scrotum and its contents, and with a temperature of 99°F. An examination showed the swelling to be limited to the upper half of the scrotum and to the globus major of the epididymis. The scrotal skin was reddened, and palpation elicited acute pain. The cord was free of any involvement in the picture, and torsion was not demonstrable. Actually, the testicular body could be gently palpated without acute pain being elicited. A diagnosis of torsion testis was made, and immediate operation advised and accepted. Operation, February 13, 1938: Gas anesthesia. An incision was made through the scrotal walls and tunica vaginalis, which exposed a normal testis and slightly congested epididymis. The hydatid of Morgagni was of a deep purple color, verging on gangrene, and the size of a large pea. It appeared stalked, with the distal two-thirds dry and verging on necrosis. No definite torsion was demonstrated, though looked for, but the appearance was so characteristic that it was considered that recent detorsion had occurred. It was excised, and no bleeding ensued. The wound was closed in layers with fine catgut. Recovery was prompt and cure established. Microscopic sections showed extensive hemorrhagic extravasation and quite generalized cellular necrosis. 3 Personal communication from Dr. Philip Rosenblum, and to be published later. It was presented by him before the joint meeting of the Philadelphia and New York Urological Societies, April 21, 1938.
TABL E
2.-0perative findings APPENDIX OF TESTIS
NO .
OBSERVER
DAY OF ATTACK
CORD
FLUID CONTENTS
TESTIS
EPIDIDYMIS
Size
Color
Turns
Direction
days
1
Guilleminet
Many
Normal
Serum
Normal
Normal
2 3
Foshee Foshee
5 3
Oedema Oedema
Normal Twice normal
Oedema
Clear yellow
4 5 6
Guillemin and 3rd attack Grandineau in 14da. Inouye 15 Chiariello 3
Normal
Brown
7 8 9
Blauchat Guerin Moncalvi
Slightly hemorrhagic
10
Rhodes
11 12 13
2 2 3
14
Rhodes Roche Rochet and Pouyanni Grasso
Congested Hemorrhagic Congenital Serohemorhernia rhagic Normal 4 oz. hemorrhagic Normal Yellow Normal Yellow Normal
3
Normal
15
Olascoaga
4
Normal
16 17
Olascoaga Tavernier and Garin Randall Rosenblum
7
Normal Normal
18 19
1 3
Normal
28
3 3
Normal Normal
Serohemorrhagic Amber Amber Serohemorrhagic Amber Amber
Grain of wheat 1cm. 1 cm.
Ochre Blue-black Blue-black
3
Clockwise
8mm.
Black
11.2
Clockwise
1½ cm. Pea
Dark red Black
1
Clockwise
Haddetorsioned
.., 0 ...rn ~
Enlarged
~ ~
~
Congested Ectopic in canal Normal
Congested Oedema
1cm. Small
Blue Red
Normal
3cm.
Necrotic
Normal Normal Inverted
Normal Normal
2½ cm. Cherry
Bluish Purple red Black
2 2 1
Normal
Normal
Large pea
Black blue
+
Double normal Normal Normal
Normal Normal
Normal Normal
Normal Normal
I
+
~
Clockwise Clockwise Counterclockwise
~
;
rn
Enlarged Enlarged Double normal 2cm. Large pea
Red Violet Red Deep purple
Many times 1½
~
Clockwise
~
~
724
ALEXANDER RANDALL
Torsion of other vestigial structures. In this period from 1931 to 1937, inclusive, the available literature contains 2 reports of this character:
Case 1 (Patch): Report of a case of torsion of a cyst arising from the vas aberrentia. Case 2 (Sorrel): Reports two cases of torsion of the organ of Giraldes.2 Treatment. The operation is so simple, and so devoid of any serious complications, that it should always be advised. In that I was able to demonstrate so clearly, in my Case 1, the bilateral development of unusually long hydatids, it would seem advisable to operate on both sides in any given case. The :fixation of the testis to the incision in the tunica vaginalis, when sewing up, I should likewise advise as a step to prevent subsequent torsion testis, as these 2 conditions have been reported as occurring simultaneously (table 2). The question of conservative- non-operative- treatment is ill-advised: (1), because no local treatment relieves the distress; (2), because detorsion and recurrent attacks are possible; (3), because the nonoperative recovery can take longer than the operative removal and convalescence; and (4), we have the suggestion of Contiades and Merigot, who report 2 cases where they felt evidence to exist that a hydrocele collection was secondary to a small foreign body found within the tunica vaginalis, and in each case the history gave evidence that torsion of the hydatid had preceded the development of the hydrocele. CONCLUSIONS
Two additional cases of torsion of the appendix testis are reported; bringing the total to sixty-eight cases in the literature to the end of 1937.3 It is of moment that only 2 American authors have reported cases. It is assumed that the diagnosis is being missed, and masquerading under a faulty interpretation of the symptoms and signs. Treatment is surgical and, as a preventive step, should be performed on both sides. Medical Arts Bldg., Philadelphia, Pa. REFERENCES BLAUCHAT: Bordeaux Chir., 6: 159, 1935. CmARIELLO: Pediatria, 44: 1104, 1934. COLT: Brit. J. Surg., 9: 464, 1922. CONTIADES AND MERIGOT: Ann. d'Anat. Path. Med. Chir., 9: 795, 1932. DIX: Brit. J. Urol., 3: 245, 1931.
TORSION OF THE APPENDIX TESTIS
725
Doc: Ztschr. f . Urol. Chir., 33: 486, 1931. - -: Ztschr. f. Urol., 26: 408, 1932. FOSHEE : J. A. M.A., 99: 389, 1932. , GLASS : Quoted by Salmon, Mosinger and Contiades. GUERIN: Bordeaux Chir., 6: 157, 1935. GurLLEMIN AND GRANDINEAU : Rev. med. de !'est, 61: 826, 1933. Gu1LLEMINET: Lyon Chir., 23: 821 , 1931. GRASSO: Arch. Ital. d. Chir., 43: 221, 1936. HENRICH: Amer. J . Obst., 21: 121, 1931. INOUYE : Abst. Jap. J . Derm. and Urol., 33: 47, 1933. MEYER-WILDISEN : Beitr. z. klin. Chir., 164: 47, 1936. MoNCALVI: J. d'urol. med. et chir., 36: 501, 1933. MoucHET : Presse med., 31: 485, 1923. MoucHET: Bull. et mem. de la Soc. Nat. de Chir., 61: 431, 1925. OLASCOAGA : Semana med., 2: 1484, 1936. OMBREDANNE: Bull. et mem. de la Soc. Nat. de Chir., 39: 779, 1913. PATCH: Brit. J. Urol., 6: 122, 1933. PUTZUDONEDDER : Pathologica, 29: 98, 1937. RHODES: New Eng. J. Med., 213: 1005, 1935. ROCHE: Clin. J., 64: 96, 1935. ROCHET AND POUYANNI: Bordeaux Chir., 6: 135, 1935. SALMON, MosINGER AND CONTIADES : Ann. de Anat. and Path., 10: 490, 1933. SALMON AND CONTIADES: J. d'urol. med. et chir., 37: 412, 1934. SHATTOCK: Lancet, 1: 693, 1922. SORREL: Bull. et mem. de la Soc. Nat. de Chir., 61: 1270, 1935. TAVERNIER AND GARIN : Lyon Chir., 33: 598, 1936. WALTON: Brit. J. Surg., 10: 151, 1922. The following references could not be obtained for review; or the given data was incomplete or in error, as they could not be found in the Index Medicus: ALLENDE : Rev. de chir. de Bs., 1931. CONSTANTINI : Soc. de Chir. d'Alger, Jan. 19, 1933. DIAz, CASTRO, BONNECARRERE : Med. Soc. Urug. d . Urol., 2: 36, 1936. GAMBOA y NEIDELMAN : Semana med., 1931. SERANTES, MONSERRAT AND GARCIA : Revista Argentina de Urol., p. 196, 1936.