| Abstract|| |
Progressive disseminated histoplasmosis (PDH) caused by Histoplasma capsulatum generally develops in immunocompromised patients or those on long-term systemic corticosteroid therapy. Here, we report a case of PDH in an immunocompetent patient, which is a rare occurrence. A 56- year-old male patient, whose work often led him to caves, now reported with a low-grade fever and altered sensorium. Investigations revealed hyponatremia, bilateral adrenal enlargement, and mediastinal lymphadenopathy. A rare complication of the manifestations of histoplasmosis in an immunocompetent patient, in the form of ring lesions in the brain, came to light during the clinical investigations. The patient developed generalized tonic–clonic convulsions and needed care for the critical condition 2 weeks into treatment. This case report traces the clinical course, histological and serological findings, and response to amphotericin B therapy in the patient.
Keywords: Immunocompetent patient, progressive disseminated histoplasmosis (PDH), ring brain lesions
|How to cite this article:|
Agarwal VK, Prusty B S, Pereira KR. An uncommon presentation of progressive disseminated histoplasmosis. Ann Trop Med Public Health 2016;9:279-82
|How to cite this URL:|
Agarwal VK, Prusty B S, Pereira KR. An uncommon presentation of progressive disseminated histoplasmosis. Ann Trop Med Public Health [serial online] 2016 [cited 2021 Apr 14];9:279-82. Available from: https://www.atmph.org/text.asp?2016/9/4/279/184798
| Introduction|| |
The first description of histoplasmosis was by an American physician named Samuel Taylor Darling in Panama, who described the disseminated form of the disease in a case from Martinique, France. Initially, the disease was attributed to miliary tuberculosis. Progressive disseminated histoplasmosis (PDH) infection is caused by Histoplasma capsulatum, a dimorphic saprophytic fungus, residing in the soil, and found abundantly in caves. Infection to humans is mainly caused by inhalation of mycelial fragments and conidia., In India, a few cases have been reported most commonly from the eastern regions and in the soil of the Gangetic plains., There exists a spectrum of signs and symptoms of histoplasmosis that are dependent on the immune status of the patient and the intensity of exposure. Therefore, the patient may have an asymptomatic presentation or could present with PDH. Patients, when brought to hospitals could have respiratory failure, shock state, multiorgan dysfunction, and effects on liver, spleen, adrenals, mucocutaneous regions, and bone marrow. Statistics reveal that 30-50% of the patients with PDH present with asymptomatic adrenal gland involvement, out of which few patients present with adrenal insufficiency. We report a case of PDH in an immunocompetent patient presenting with bilateral adrenal mass associated with hypoadrenalism and coincident with cerebral ring enhancing lesion, which is seldom associated with histoplasmosis.
| Case Report|| |
A 56-year-old male patient, geologist by profession, was admitted to the hospital with altered sensorium. The patient was presently working in Hyderabad, Telangana, India and had a history of frequent work visits to caves in Visakhapatnam, Andhra Pradesh over the past 3 years. The patient presented with low-grade fever, accompanied by chills, and complained of anorexia and weight loss since 6 months. There was a spurt of hiccups since the past 2 days prior to admission. The patient was a habitual smoker, and smoked one packet of cigarettes every day since the last 30 years.
On examination, the patient appeared to be incoherent, slipping in and out of consciousness and had a thinly-built body (body mass index of 18.0). The patient's spleen was enlarged. On laboratory evaluation, the patient was found to have hyponatremia, with serum sodium level of 109 mEq/L. There was clubbing of fingers but without lymphadenopathy, anemia, or jaundice. The patient's pulse rate was recorded as 76 beats/min, blood pressure as 110/70 mm of Hg, respiratory rate as 23 breaths/min, and an oxygen saturation (Spo2) of 93% on room air. The other electrolyte levels were found normal. Further tests revealed leukopenic total leukocyte count (TLC) 3,300/min with adequate platelets and hemoglobin and hypothyroidism with thyroid stimulating hormone (TSH) 13.98 units. The patient's chest X-ray imaging was within normal limits. Ultrasound of the abdomen revealed splenomegaly, bilateral suprarenal space-occupying lesions suggestive of bilateral adrenal masses. Right 6.2 cm × 4.7 cm, left 7.5 cm × 4.3 cm with few central cystic lesions [Figure 1].
|Figure 1: Ultrasound of the abdomen: An abdominal ultrasound was conducted and splenomegaly was observed. Seen in the image are bilateral suprarenal space occupying lesions suggestive of bilateral adrenal masses. Right 6.2 cm × 4.7 cm, left 7.5 cm × 4.3 cm with few central cystic lesions|
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An 18 fluorodeoxyglucose positron emission tomogram (18 FDG-PET) scan showed increased uptake in mediastinal lymph nodes, bilateral adrenal mass lesions [Figure 2], and non-FDG ring-enhancing lesion in the brain [Figure 3].
|Figure 2: Bilateral adrenal mass lesions: An 18 flurodeoxyglucose positron emission tomography scan showed bilateral adrenal mass lesions|
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|Figure 3: Non FDG ring enhancing lesion in the brain: The 18 fluorodeoxyglucose positron emission tomogram revealed non-FDG ring enhancing brain lesion|
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Computed tomography (CT)-guided adrenal biopsy showed well-formed granulomatous tissue with multinucleated giant cells, epithelial histiocytes, and lymphocytic mononuclear inflammatory infiltrate. Many round-to-oval periodic acid-Schiff (PAS) positive fungal organisms were observed, which was suggestive of histoplasmosis [Figure 4] and [Figure 5].
|Figure 4: CT-guided adrenal biopsy: CT-guided adrenal biopsy showed many round to oval PAS positive fungal organisms, which was suggestive of histoplasmosis|
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|Figure 5: Histology of CT-guided adrenal biopsy: Well-formed granulomatous tissue with multinucleate giant cells, epithelial histiocytes, and lymphocytic mononuclear inflammatory infiltrate were observed|
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The diagnosis of PDH, involving the brain, mediastinal lymph nodes, and bilateral adrenal glands, was made.
The hyponatremia was corrected with intravenous (IV) fluids and salts and for PDH, IV liposomal amphotericin B 3 mg/kg body weight was administered. The patient was human immune deficiency virus (HIV) negative and the cluster of differentiation (CD4) counts were within normal limits.
After 2 weeks of amphotericin B treatment, despite maintaining normal electrolytes, the patient developed an episode of generalized tonic–clonic seizures (GTCS) and was treated with lorazepam, followed by fosphenytoin. A magnetic resonance imaging (MRI) of the brain was done that showed the same findings as the PET scan [Figure 6].
|Figure 6: MRI of the brain: An MRI of the brain was conducted when the patient developed an episode of generalized tonic–clonic seizures after 2 weeks of amphotericin B treatment. The findings of the MRI were similar to those observed in the PET scan |
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After 3 weeks of treatment with liposomal amphotericin B, the patient developed hypotension. The serum sodium level was recorded to be 119 meq/L, and potassium level was recorded to be 6.1 meq/L. The patient was managed with IV fluids. The patient's serum cortisol at 8 am was 3.64 µcg/dL (normal 4.3-22.4) and an initial hydrocortisone therapy was started and then changed to oral prednisolone 7.5 mg/day. The patient's plasma renin level was 3.22 ng/dL, which is the upper limit of the normal range.
After 4 weeks of liposomal amphotericin B treatment, oral itraconazole 200 mg twice daily was started and amphotericin B was stopped.
| Discussion|| |
In the present case, the patient hails from Kolkata (Gangetic delta of East India), West Bengal, India from where most of the cases of PDH, have been reported in India.,,
Our patient presented with low-grade fever, weight loss, generalized weakness anorexia, and bilateral adrenal enlargement. In addition, the patient had adrenal insufficiency, as diagnosed by low serum cortisol, hyponatremia (119 meq/L), hyperkalemia (6.1 meq/L) that continued, despite 3 weeks of treatment with liposomal amphotericin B.
18 FDG-PET scan revealed intensely increased uptake in both the adrenals, mediastinal lymph nodes, and non-FDG enhancing ring lesions in the brain. After 2 weeks of treatment, the patient developed GTCS, which was due to existing non-FDG enhancing ring lesion in the brain. A ring enhancing lesion in the brain in a PDH patient is uncommon when the patient is an immunocompetent adult.
The patient was treated with liposomal amphotericin B for 4 weeks with a dose of 3 mg/kg body weight and followed by oral itraconazole twice daily that was advised for 1 year. The patient showed dramatic clinical response to treatment.
| Conclusion|| |
This case report will significantly help the medical fraternity by bringing to light the possibility of brain lesions associated with histoplasmosis. Further, clinicians can be alerted to the possibility of finding the signs and symptoms of histoplasmosis in immunocompetent patients, contrary to the longstanding view of its predominant association with immunocompromised patients.
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Conflicts of interest
There are no conflicts of interest.
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Karishma Rosann Pereira
Yashoda Hospital, Malakpet, Hyderabad, Telangana
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]