Coccidioidomycosis Presented As a Pelvic Mass in an Otherwise Healthy Female

Available online at www.annclinlabsci.org 585 Annals of Clinical & Laboratory Science, vol. 45, no. 5, 2015 Coccidioidomycosis Presented as a Pelvic Mass in an Otherwise Healthy Female

Frances Compton, Jamie Everett, Audrey Wanger, and Songlin Zhang

Department of Pathology and Laboratory Medicine, The University of Texas Health Science Center at Houston, Houston, TX, USA

Abstract. We present a case of a 32 year old female with a past medical history of hypertension who presented with several years of chronic back pain and was ultimately diagnosed with isolated pelvic coccidioidomycosis. She was initially seen by gynecologic oncology for assessment of possible metastatic cancer by image study, but a cytopathologic diagnosis of coccidioidomycosis lead to a cancellation of the planned surgery and extensive antifungal treatment managed by the infectious disease team. She had no known previous pulmonary disease or immunodeficiency. Pelvic coccidioidomycosis without known pulmonary disease is very rare, and disseminated infection typically only occurs in those who are severely immunocompromised. Our case presented with several years of back pain and a pelvic mass mistaken for possible malignancy by image study.

Introduction about 1% of cases, and it is often associated with some form of immunodeficiency [1]. Spread to the Coccidioidomycosis, or valley fever, is a systemic genitourinary tract is even more uncommon and fungal infection which primarily causes communi- often reflects a particularly severe disseminated dis- ty acquired pneumonia. The disease is caused by ease process [2]. Coccidioides immitis, a dimorphic fungus which is endemic to the southwestern United States [1]. Case Report Infection is usually caused by inhalation of the ar- throconidia during the dry season of the endemic We present a case of a 32 year old female with a past region. The lungs are the most frequent site of entry medical history of hypertension who presented with several years of chronic back pain. Imaging revealed an 8 and infection, but disseminated disease can occur cm paraspinal process causing destruction to the lower in extrathoracic organs such as skin, soft tissue, lumbar spine and sacrum (Figure 1). She was referred to bone, and meninges [1]. Once in tissue, the di- gynecologic oncology for resection of the possibly malig- morphic fungus transforms into endosporulating nant mass, and pathology was requested to perform im- spherules which eventually break open to release mediate assessment of fine needle aspiration of the mass more spherules. The double-walled spherules con- prior to surgery. Two passes of CT-guided fine needle taining endospores can be easily detected histologi- aspiration of the patient’s pelvic mass were performed by cally by examining hematoxylin and eosin stains of the radiologist, and the initial interpretation revealed the tissue, but other stains such as periodic acid- profuse purulent material with marked acute inflamma- Schiff and Grocott-Gomori methenamine silver tion. A portion of the pelvic aspirate was triaged to microbiology for culture, and a cellblock was prepared can also be used for identification [2]. Disseminated from the remaining aspirate. The scheduled next day coccidioidomycosis is quite rare as it occurs in surgery was canceled after discussion with surgical gynecologic oncology. Address correspondence to Frances Compton, MD; Department of Pathology and Laboratory Medicine, The University of Texas Smears and sections from the cellblock showed many Health Science Center at Houston, 6431 Fannin Street, MSB 2.262, Houston, Texas, 77030, USA; phone: 817 475 2968; e mail: oval to round fungal organisms with a size range of 10- [email protected] 50 micrometers (Figures 2 and 3). There were also

multiple empty cysts and cysts containing endospores. The diagnosis of coccidioidomycosis was rendered based on the classic fungal morphology as well as a positive Grocott-Gomori methenamine silver stain (Figure 4). The diagnosis was also confirmed by microbiology culture. The patient was referred to the infectious disease team, and she was subsequently started on 2 weeks of intravenous amphotericin B. Further questioning revealed that she had visited Arizona several years ago, but she has no history of immunosuppression. Unfortunately, she sustained an episode of acute renal failure in response to the antifungal treatment and was switched to itraconazole on day 10 of her treatment. Treatment plan for her was to continue itraconazole for 12 months and to fol- low up with the infectious disease team for any necessary Figure 1. CT abdomen/pelvis with large, destructive treatment in the future. mass. Discussion

Pelvic coccidioidomycosis is a very rare presentation of the systemic fungal infection, and there have been very few case reports describing similar infections. Furthermore, the disease has a wide range of presentations according to the degree and location of infection. Some of the patients described in previous case reports with pelvic coccidioidomycosis presented with complaints of pelvic or abdominal pain [3,4] while other patients were diagnosed inci- dentally [5]. Our patient presented with chronic lower back pain as the infection had already invad- ed the sacrum and lower spine. One of the above- Figure 2. Pap stain (magnification x 400) of fine needle mentioned case reports of pelvic coccidioidomyco- aspiration showing many cocci spherules of varying sizes. sis was, much like our case, preliminarily diagnosed with a pelvic malignancy [3]. The patient presented with abdominal distention and pain, and was found to have an elevated CA-125 level as well as moder- ate ascites on sonogram. She was given the provi- sional diagnosis of ovarian cancer, and she elected to have a total abdominal hysterectomy with bilat- eral salpingectomy [3]. This patient, similar to ours, had an initial diagnosis of malignancy followed by an unexpected diagnosis of coccidioidomycosis after pathological analysis.

While few case reports of pelvic coccidioidomycosis exist, there are even fewer cases of isolated pelvic coccidioidomycosis documented. One case from 1976 describes a case of a patient with isolated coc- Figure 3. Hematoxylin and eosin stain (magnification x cidioidomycosis of the uterus in which no known 400) of cell block prepared from fine needle aspirations showing cocci spherules containing endospores. primary lung infection was found [5]. The authors Pelvic Coccidioidomycosis 587 Indeed, Coccidioides species has been described as “one of the great imitators” as it has a history of unusual manifestations ranging from pericarditis to peritonitis [8]. Therefore, even more rare presentations such as pelvic and genital tract infections should never be discounted.

References

1. Malo J, Luraschi-Monjagatta C, Wolk DM, Thompson R, Hage CA, Knox KS. Update on the diagnosis of pulmonary coccicioidomycosis. Ann Am Thorac Soc 2014; 11 (2): 243-253. 2. Welsh O, Vera-Cabrera L, Rendon A, Gonzalez G, Bonifaz A. Coccidiomycosis. Clinics in Dermatology 2012; 30: 573-591. 3. Micha JP, Goldstein BH, Robinson PA, Rettenmaier MA, Brown JV. Abdominal/Pelvic Coccidioidomycosis. Figure 4. Grocott-Gomori methenamine silver stain Gynecologic Oncology 2005; 96: 256-258. (magnification x 400) of cell block prepared from fine 4. Saw EC, Smale LE, Einstein H, Huntington RW. Female needle aspiration showing positive-staining cocci. Genital Coccidoidomycosis. Obstetrics and Gynecology 1975; 45 (2): 199-202. theorized that the route of infection could have 5. Hart WR, Prins RP, Tsai JC. Isolated coccidioidomycosis of the uterus. Human Pathology 1976; 7 (2): 235-239. been a primary lung infection, which subsequently 6. Huntington R, Waldmann W, Sargent J, O’Connell H, Wybel healed, but they favored a primary uterine infec- R, Croll D. Pathologic and clinical observations on 142 cases tion. This is due to the fact that the patient had of fatal coccidioidomycosis with necropsy. In: Coccidioidomycosis: Papers from the Second Symposium on multiple child-births in an endemic area [5]. Our Coccidioidomycosis. (Ajello L, Ed.) The University of Arizona patient does have a history of travel to Arizona, an Press, Tucson, 1967; pp. 143-167. endemic region, as well as history of a cesarean sec- 7. Smith G, Hoover S, Sobonya R, Klotz SA. Abdominal and Pelvic Coccidioidomycosis. Am J Med Sci 2011; 341 (4): tion. While primary pulmonary infection is far 308-311. more common than genital tract infection, there is 8. Crum-Cianflone NF, Truett AA, Teneza-Mora N, Maves RC, Chun HM, Bavaro MF, Hale BR. Unusual presentations of also documentation that even severely disseminated coccidioidomycosis: A case series and review of the literature. coccidioidomycosis does not typically invade the Medicine 2006; 85: 263-277. genital tract. One study of 142 autopsies of fatal coccidioidomycosis found that even in those patients who died of disseminated coccidioidomycosis; there was no involvement of the female genital tract [6]. Although only 33 of those 142 cases were women, this study still clearly demonstrates the in- frequency of this case presentation.

Despite the fact that our patient had visited an endemic region for coccidioidomycosis, she does not have a medical history of immunosuppression to explain the cause of dissemination. Although disseminated coccidioidomycosis is typically related to an immunocompromised state, it is not required. A paper which discussed six unusual cases of abdominal and pelvic coccidioidomycosis stated that five out of the six cases they reported had no known cause for dissemination [7]. The only immunocompromised patient in the case series was a patient with a previous solid-organ transplant. This paper further uncovered the immensity of this disease.