Intermittent Hydrarthrosis

INTERMITTENT HYDRARTHROSIS RoBERT

I.

HILLER,

M.D., F.A.C.S.,

THE

MILWAUKEE,

Wm.

subject of intermittent hydrarthrosis has been summarized by Schlesinger,J·• Bierring,o Pulawski,6 and Berger.7 The first case was reported by Perrin in 184!!• In 1921 Bierring summarized the literature and found a total of 77 cases. When Bergt;r reported his ea~e of allergic eti.ology in 1939, he was able to find a total of 105 case,-. The average age incidence is between 20 and 45 year~, hut cases in much younger an' who had attacks of joint swelling every 13 days. 'l'hen• had h<"en a history of two attack:' of rheumatic fever. There was no response to therapy. Miller and Lewinn reported a case of a woman of :3tl, who responded to typhoid vaccine intravenously as a form of nonspecific therapy; and which was, therefore, con tddered to be due to anaphylaxis. Quincke 's edema wa~ con:;idered the cause in soiw· eases by f-1chlesinger.l He found eases in which the attach were associated with either sialorrhea, edema of the glottis, or diarrhea. In one family, he found several member' afflicted with intermittent hydrarthrosis. Quincke's edema wa~ preRent in two affected members. Blood dyscrasia, probably purpura, was pre~ent in memherH of one of th•' afflicted families. Reimann and Angelides'" also collected a group of cases of periodk arthralgia in five generations of one family. Several of the ea~es were in males. N(Jit<· of the women were relieved during pregnane.'' and many of the l'ases were desf'rihHd a:c ~howing "black and blue spots" about the joints involved. Weismarm·Netterr:; reporwd a case of a woman 5ti years old, whose effusion appeared with marked regularity with her periods. He made careful ehemical analysis of fluirl rernon'd from the knee joint. He also Rtudied the blood chemistry. He obtained a complete cure after seven injectim" of ergotamine tartrate following no results with thyroid, O\'arian extract. atropine. Adrenalin, alkalization with sodium bicarbonate, addiiif•ation with acid phosphat£>. inj<:><· tions of peptone, autohemotherapy, Atophan, and intravenous calcium chloride. 'rhe .fir;d by several authors, In l!J:~~ Rhehldorf14 reported two interesting eases: one started with intermittent hydrarthrosis wiih the onset of menses at the age of 1:i yean,. A.t tlw age of 10, the paitent had had diphtheria witlt heart failure. With the on~ei of menses. ftui•l appeare
209

210

Hli,LEH

i\m. I

of 40 who had had intermittent hydrarthi'OHis ~ince the age of HI. She had no t)ft'u8ion •luring 11ve pregnancies. Both knees were atreded. X-rays were negative. Her blood count anti sedimentation rate were normaL The administration of estrogenie hormone caused disappearance of the effusion. Howevn, nne can draw uo definite conclusions regarding the therapeutic value of estrogen in this second case because the amount com· monly administered in 1932 was quite small. Schlesinger2 believed that there was a definite relationship between effusions and ovarian dysfunrtion because they both fnL quently disappeared with the onset of tlw menopause. Some of his cases were treate~tosterone pellets beneath the skin of the abdominal mlll mu~e
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Volume 64 Number I

INTERMITTJ<~NT

HYDRARTHROHIK

211

and mouth disclosed no abnormalities. 'i'he tonsils had been removed. 'fhe thyroid was just palpable. There was no bruit and the cervical lymph nodes were not enlarged. Th<' breasts were normal. The lungs were clear. The heart had a regular rhythm. No murmurs were noted. The rate was n.ormal. 'fhc abdomen presented no tenderne~
Suprapatellar circumference Transpatellar circumference Infrapatellar circumference

RIGHT

LEFT

15% inches 143,4 inches 12% inches

14% inches 14 inches 12 inches

After the attack subsided measurements of 14, 133,4, and 121,4 inches were noted on the right. The quadriceps muscle showed slight atrophy. At the Mount Sinai Hospital, in New York, where she was finally sent for a thorough investigation and for an opinion on the use of testosterone, the orthopedic consultant discovered a small Baker's cyst in the popliteal space of the right knee. He reoommended removal of this cyst if no success was achieved by other means. Examination by the consulting allergist disclosed negative sensitivity tests except for dust. The laboratory findings were as follows: Blood examination: hemoglobin 13.0 Gm., leukocytes 9,150 of which the polynuclears constituted: segmented 44 per cent, stab folms 8 per cent, lymphocytes 44 per cent, eosinophils 1 per cent, basophils 1 per cent, monocytes, 2 per cent. The sedimentation rate was 12 mm. in one hour, normal. The Kahn test for syphilis was negative. The Blood Wassermann test was negative. Blood uric acid was 2.0 mg. per 100 c.c. (n.ormal 2 to 3 mg.). The urine was clear, acid, with no albumin or sugar, specific gravity 1.030, and microscopic examination of a centrifuged specimen showed occasional epithelial cells, yeast, rare white blood celL The basal metabolic rate was minus 19 per cent, pulse 76. Electrocardiogram showed T4 semi-inverted, no significant abnormalities. X-rays of both knees showed no abnormality in the bones, joints, or adjacent soft tissues. Culture of the knee joint fluid showed no growth. A trial of x-ray therapy was suggested by one of the consultants. He also thought that the testosterone therapy could be tried. Upon her return from the examination at the Mount Sinai Hospital in New York, she was given one injection of 500 units of A.P.L. (chorionic gonadotropin) followed by aggravation of the swelling of the knee. On June 16, 1949, 25 mg. of testosterone propionate were given intramuscularly. It was repeated at intervals .of two weeks. Thyroid extract, llh grains daily, was taken orally by the patient. The injections were finally timed to precede ovulation and menstruation, and the swelling disappeared completely after a period of approximately three months. Injections were stopped completely on Feb. 20, 1950. There has been no recurrence .of the pain or !'welling. She has, however, continued to take the thyroid extract nearly e\'ery day. Discuss·ion.-Because the condition was aggravated by the administration of stilbestrol, the question of sensitivity of the patient to her own estrogen arose. However, swelling of the knee was present at ovulation time when the estr.ogen level is ordinarily high and also at menstruation when it should be low (Smith and Smith24). The swelling was absent during pregnancy when the estrogen level is very high, and during lactation when the level is very low ( Goldberger25). 'l'he patient has Rh-negative blood. She has two sisters, one Rh negative and one Rh positive. Her husband has Rh-positive blood. Her older child, a boy, has Rh-negative blood.

HlLLKH

Uonsequently the possibility of senHitization to her ehild 's blood tlm·ing her first pregnan<·: aM a factor in the etiology of the intermitU,IIt h.nlrarthrosiR is ruled out. Her ~ymploms disapp<>ared t.luring her seeond pn•gnauey with Hll Rh positive girl. Hinee the thyroid activity tluring ]m•gnancy and laetation should not vary appre· eiahly from the patient 'B nomml, the U8e of thy mid extraet eould not he eredited with her improvement, in spite of her minus ba~al metabolism. Tho te~tosteroue would. tlJf'r
Summary A case of intermittent hydrarthrosis l~ presented which responded to testoster
References l. Schlesinger, Hermann: Wien. klin. Wchnllchr. 39: 68, 1926.

2. Schlesinger, Hermann: Wien. klin. Wchnschr. 42: 832, 1929. Schlesinger, Hermann: Mtinehen. med. Weftnschr. 46: 1137, 1899. 4. Schlesinger, Hermann: Mitt. a.d. Grenzgeb. d. Med. u. Chir, Jena, lilOO, p. 441. 5. Bierring, W. L.: J. A.M. A. 77: 785, 1921. 6. Pulawski, A.: Wien. klin. Wchnschr. 27: 421. 19lcl. 7. Berger, Herbert: J. A. M.A. 112: 2402, 1939. 8. Melane, S., and Midana, A.: Minerva med. 2: 5I9, 1940. 9. Poirier, M.: Bull. Soc. path. exot. 35: 390, 1942. 10. Nielson, A. L.: J. A.M. A. 78: 507, 1922. 11. Miller, Joseph L., and Lewin, Philip: J. A.M. A. 82: 1177, 1924. 12. Reimann, Hobart A., and Angelides, A. P.: .r. A.M. A. 146: 713, 1951. 13. Weismann-Netter, R. J.: Bull. et m6m. Soc. med. d. hOp. de Paris 58: 185, 1937. 14. Rheindorf, G.: Miinehen med. Wchnschr. 79: 749, 1932. 15. Beveridge, T. F., and Howe, L. D.: J. Iowa M. Soc. 15: 314, 1925. 16. Schmitza, F., and Vogl, A.: Wien. klin. Wehnsehr. 45: 1455, 1932. 17. Alonza, L.: Minerva med. 2: 260, 1935. 18. Lievre, J. A.: Bull. et mem. Soc. med. de hop. de Paris 63: 183, 1947. 19. Rebierre, P.: Bull. et mem. Soc. med. de hop. de Paris 36: 476, 1913. 20. Krida, Arthur: J. Bone & Joint Surg. 15: 449, 1933. 21. Jl.fandl, Fritz: Zentralbl. f. Chir. 62: 445, 19:15. 22. Epstein, H. J., and Edeiken, Louis: Radiology 35: 360, 1940. 23. Pelner, Louis: Am. J. M. Se. 206: 498, 1043. 24. Smith, G. Van S., and Smith, 0. W.: New England J. Med. 215: 908, 1936. 25. Goldberger, M.A.: AM. J. 0BST. & GYNEC. 33: 1093, 1937. 26. Hellbaum, Arthur A., Ishmael, William K., Owens, J. N., Jr., Kuhn, .John I.,. and Duffy, Mary; Symposium on Steroids in Experimental and Clinical Practice, Philadelphia, 1951, The Blakiston Company.

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