Coccidioidomycosis of female adnexa

COCCIDIOIDOMYCOSIS

OF FEMALE

LOWELL C. WORMLEY, M.D., LAZARUS MANOIL, Phoenix,

T

HE purpose of presenting this report is the extreme rarity of coccidioidomycosis of the femaIe adnexa. In the past onIy two cases of dissemination of this condition have been reported, one by H. Jacobson’ and another by Page and Boyers. 2 In both cases reported complete recovery foIIowed tota remova of the infected adnexa. The case reported herein may we11 be a third such case. Epidemiolog),. Coccidioidomycosis is endemic in the southwestern United States, particuIarIy in southern Arizona and the San Joaquin VaIIey of California. However, coccidioida1 infections have been reported from the Argentina-Uruguay area and in other parts of the South American continent. IsoIated cases have been reported from Hawaii and ItaIy. Pathogenesis. Infection by the fungus, Coccidioides immitis, usually takes pIace by inhaIation and the primary infection occurs in the Iung. However, abrasions sustained in the infested desert areas may resuIt in skin infection. C. immitis is a diphasic organism, and in its infectious stage it is represented by chIamydospores which are highIy infectious, cIing to clothing and may be transmitted quite a distance. They Iie in the dust and in the shrubs in the foothiIIs of the mountains. The chlamydospores represent the saprophytic or infectious phase of the fungus; the parasitic or spheruIar phase is a non-infectious phase or what we actuaIIy see in the tissues and body Auids which have aIready become infected by the chIamydospores. When infection takes place through the Iungs by inhalation, it may be demonstrated roentgenologicalIy aIthough physical findings may be negative. However, there will be no suspicion of coccidioidomycosis unti1 there is evidence of dissemination of the fungus elsewhere in the body. Dissemination to other parts of the body through the bIood stream from the Iungs is not too frequent. CharIes E. Smith of Stanford University states that in not more than one infection in I ,000 does

ADNEXA*

M.D. AND MAURICE ROSENTHAL, M.D.

Arizona the fungus disseminate, but when it does it is usuaIIy disastrous. It is believed that coId abscesses of the skin and skin Iesions are more the result of dissemination of primary puImonary infection rather than primary infection of the skin itself. SeveraI cases of dissemination of the infection to the bones, joints, skin and meninges have been reported. Diagnosis. Coccidioidomycosis once suspected may be diagnosed by the intradermal use of 0.1 cc. of I : IOO diIution of coccidioidin. The reaction is similar to the Mantoux reaction and is read thirty-six hours later. The cornpIement fixation test is diagnostic and also very vaIuabIe in following the course of the disease. The titer of complement fixation rises with the severity of the infection so that in disseminated Infection it may be compIete in I :2~6 serum diIution. It is thus vaIuabIe in warning of impending dissemination of the infection. CASE

REPORT

The following is submitted as the third case of coccidioidomycotic infection of femaIe adnexa on record: The patient was a twenty-three year oId colored femaIe seen with a compIaint of severe postmenstrual pain and a profuse, white, thick vagina1 discharge for the past year. Premenstrua1 pains had graduaIIy increased in severity in the weeks preceding admission to the hospital. Onset of menses was at nineteen years of age and the periods had been regular, every twenty-eight days, and of three to four days’ duration. The patient had not had menstrua1 pains prior to the present iIIness. She had been married two years and had had no pregnancies. About eighteen months prior to admission, whiIe picnicking she had been thrown off a horse into a piIe of dust and sagebrush. Soon after this fall two “kernels” developed, one in the right supracIavicuIar region and the other over the left externa1 malleolus. These Iesions were treated by a IocaI physician who incised and drained one; the other opened

* From the Surgical and Pathology Departments, St. Monica’s Hospital, Phoenix, Ark 958

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spontaneousIy. After this the patient recovered and improved in general heahh. However, severa months Iater she began to have a profuse vaginal discharge associated with peIvic pain for which she was given peniciIIin by her IocaI physician. As the pelvic pains persisted, further examination discIosed adnexaI disease for which surgery was advised. She was admitted to St. Monica’s HospitaI ApriI 9, 1949, and was operated upon two days Iater. The patient gave a history of having had n.easIes and chickenpox in childhood. She had had an appendectomy at the age of thirteen years. There were no other serious iIInesses or operations. On physica examination there was a smaI1 area at the site of former drainage of a carbuncIe on the right side of the neck just above the cIavicIe. The abdomen reveaIed a lower right rectus scar. There was diffuse, noduIar, tender fuIness in the lower abdomen and tenderness on deep paIpation over the symphysis biIateraIIy. BimanuaI examination of the peIvis reveaIed the cervix to be norma in size and consistency. There was a fluctuant, tender mass in the Ieft fornix. There was a nodular, hard mass in the posterior peIvis which appeared to be fixed and difhcuIt to move. IvlanipuIation of the cervix was very painfu1 to the patient. Th e extremities were norma except for a scar on the externa1 maIIeoIus of the Ieft ankIe. At this time a diagnosis was made of Ieft tubo-ovarian abscess and fibroid uterus. Laboratory work reveaIed the folIowing: Kahn test, negative; urine, negative; red blood ceIIs, 4,100,ooo; hemogIobin 77 per cent; white bIood ceIIs, I 2,650; basophiIes, I ; eosinophiIes 2; stabs, 6; segmented, 46; Iymphocytes, 43; and monocyctes, 2. There was moderate hypochromia. X-ray examination of the chest reveaIed no pathoIogic condition of the Iungs. On ApriI I I, 1949, the patient was prepared for surgery contempIating an abdomina1 and a vagina1 approach. A midline incision was made from the symphysis pubis to and sIightIy to the left of the umbiIicus. The rectus fascia was incised mesiaIIy and the muscIe separated in the midline. The peritoneum was opened and the abdomina1 cavity exposed. With the patient in the Trendelenburg position the omentum was carried down over the smaI1 intestines and packed away from the pelvis. The pelvis revealed a Iarge mass in the midline. It was covered with visceral peritoneum. The visceral

December,

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Adnexa

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peritoneum and the Iower portion of the abdomina1 parieta1 peritoneum as we11 as the smaI1 intestines were covered with smaI1, anguIar, irregular noduIes approximateIy I to 3 mm. in diameter. The omentum was of a meaIy consistency, devoid of fat to a degree, and adherent to the anterior abdomina1 waI1. The smaI1 bowel was matted together and adherent to the uterus and the mass in the left peIvis by fairIy dense adhesions. The adhesions were freed and the mass was approached by doubIy Iigating the round Iigament, encircling the mass in the endopeIvic fascia by bIunt dissection. The mass was deIivered intact without rupture and Ieft a deep cavity, with moderateIy severe bIeeding, in the Ieft pelvis. Due to bIeeding in the cavity from the area where the tubo-ovarian mass had been, oxycel gauze was packed using three rolIs in the cavity. The appendix had been removed at a previous operation. The abdomen was closed in Iagers using No. oo chromic catgut to the peritoneum, No. 2 chromic catgut to the fascia and clips to the skin. No drainage was instituted. The patient was returned to the ward in good condition. The Iarge, thin-waIIed, tubo-ovarian mass was approximateIy the size of a grapefruit. The postoperative diagnosis was Ieft tubo-ovarian mass and possibIe tubercuIosis of the peIvic and abdomina1 peritoneum and the viscera. The postoperative course was uneventful. The patient was given antibiotics as a prophyIactic measure. Her temperature became norma in three days and she Ieft the hospita1 ten days postoperatively. When seen twice in a two-week interva1 foIIowing her discharge from the hospita1, her wound was soIid and there was no evidence of sinus formation. She was gaining weight and feeIing well, having no pain. The pathologic report reveaIed the foIlowing: Grossly, the specimen was comprised of a tubo-ovarian mass which measured 4 by 6 by I I cm. The ovary had been converted into a Iarge mass, cystic in nature, which on sectioning represented a muItiIocuIar cystic structure, a11 of which were fiIIed with thick, creamy, yeIIowish green, puruIent materia1. The waI1 of the cyst was thin. However, in some areas the waI1 measured 4 to 6 mm. in thickness. Sections through this area were aIso taken for histoIogic study. On the surface of this ovarian cystic structure there were remnants of a tube stiI1 discernibIe. Sections of these were taken for histologic study aIso. The tube was found to

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et al.-Coccidioidomycosis

I

of Female

Adnexa

2

FIG. I. Coccidioidal granuloma invoIving walI of faIIopian tube. FIG. 2. Note the endosporuIating spheruIe of C. immitis in giant cells.

he thickened. The muscuIar coat was thickened and the mucosa was swoIIen. The Iumen was somewhat narrowed. Representative sections were taken. MicroscopicaIIy, sections through the faIIopian tube reveaIed that the mucosa had been compIeteIy aItered and was no Ionger discernibIe. The inner Iining was repIaced by a chronic inflammatory reaction characterized by granuIoma in which “tubercuIar” Iesions existed. Within these “tubercuIar” Iesions and granuIomatus tissue characteristic endospores of C. immitis were found. The muscuIar coat was diffusely infiItrated with lymphocytes, pIasma ceIIs and an occasiona poIymorphonucIear Ieukocyte. The serosa was simiIarIy infltrated. Sections through the waI1 of the ovarian cystic structure showed that the inner Iining of the waI1 was aIso repIaced by chronic granuIomatous tissue which aIso presented numerous endospores of C. immitis. (Figs. I and 2.) The pathoIogic diagnosis was coccidioidomycosis of faIIopian tube and ovary. COMMENTS

The question of giving this patient additiona treatment foIIowing surgica1 intervention arises. Patietits in the past have been treated with coIIoida1 copper which seems to have been of benefit. Other patients have received x-ray

therapy, and stiI1 others injections of coccidioidin. One of the surgica1 cases reported the patient received no therapy whatsoever and apparentIy made an uneventful recovery. It is the opinion of the surgeon that this patient shouId not be given additiona therapy for fear that there may be a further dissemination of the infection. Review of the Iiterature reveals that two cases of coccidioidomycotic infection of the pelvic organs of the femaIe have been reported. We beIieve that the case presented is the third. According to other authors, when disseminated coccidioidomycosis has stabiIized itseIf, the predominating Iesions shouId be removed if amenabIe to surgery. The remova of the tissue invoIved may be sufhcient in the therapy of the individua1; however, subsequent foIIow-up studies of this and simiIar cases wi11 determine the choice of the method of treatment. REFERENCES

granuIoma. M. J. e? Rec., 130: 424, 498, 1929. 2. PAGE, ERNEST W. and BOYERS, L. hIORGAN. Coccidioidal peIvic inIIammatory disease. Am. J. I. JACOBSON,H. P. CoccidioidaI

Obsl. &+ Ginec., 50: 212, rg45.. 2. RUDDOCK. JOHN C. and HOPE, ROBT. B. Coccidioidal

peritonitis. J. A. M. A., I I;: 2054, 1939. 4. SMITH, CHARLES E. Coccidioidomycosis. M. Clin. North America, 27: 7go. ,943.

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Journal of SurgerJf