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Year : 2017  |  Volume : 60  |  Issue : 4  |  Page : 612-613
Addison's darling crisis


1 Hepatology and Liver Transplant Medicine, PVS Memorial Hospital, Cochin, Kerala, India
2 Gastroenterology, PVS Memorial Hospital, Cochin, Kerala, India
3 Interventional and Diagnostic Radiology, PVS Memorial Hospital, Cochin, Kerala, India
4 Pathology, VPS Lakeshore Hospital, Cochin, Kerala, India

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Date of Web Publication12-Jan-2018
 

How to cite this article:
Philips CA, Augustine P, Kumar L, Mahadevan P. Addison's darling crisis. Indian J Pathol Microbiol 2017;60:612-3

How to cite this URL:
Philips CA, Augustine P, Kumar L, Mahadevan P. Addison's darling crisis. Indian J Pathol Microbiol [serial online] 2017 [cited 2019 Sep 20];60:612-3. Available from: http://www.ijpmonline.org/text.asp?2017/60/4/612/222987




A 48-year-old nonalcoholic male, teacher by profession, with insulin-dependent diabetes mellitus type 2 and controlled glycemic status presented to our intensive care unit with altered mental status, multiple episodes of nonbilious vomiting followed by hypotension for 1 day. One week prior, he complained of loss of appetite and fatigue, without loss of weight, skin changes or jaundice and gastrointestinal bleeds. Nine months prior, he had brought some excavated land and started cultivation activity near his village. Clinical examination revealed a pale, disoriented, dehydrated patient in shock, not responsive to 2 L fluid boluses over an hour. After mechanical ventilation, blood and imaging workup was initiated. Hemogram and liver biochemistries were essentially normal and renal function tests were suggestive of severe hyponatremia (108 mmol/L, normal 135–145 mmol/L) and hyperkalemia (6.8 mmol/L, 3.5–5.0 mmol/L). Random serum cortisol was in the low normal range (3.8 μg/dL; afternoon-2–18 μg/dL) and tests for blood-borne viruses, sputum samples for acid-fast bacilli, and fungal stains were negative as was blood and urine cultures. Contrast computed tomography imaging of the chest was normal and that of the abdomen revealed asymmetrically enlarged bilateral heterogenous adrenal glands, a guided biopsy from which revealed extensive caseation [[Figure 1]a, arrow], and necrotizing granulomatous inflammation [[Figure 1]b, dashed arrows] with multinucleated giant cells. Periodic acid–Schiff [[Figure 2]a, arrows] and Grocott's methenamine silver stains [Figure 2]b showed numerous ~4 mm, ovoid, uninuclear, intracellular, and extracellular yeast forms with thin walls and narrow-based budding, consistent with Histoplasma species. Mucicarmine and acid-fast stains were negative for Cryptococcus and Mycobacterium, respectively. Two distinct subspecies of Histoplasm a capsulatu m are known, Histoplasm a capsulatu m var. capsulatu m and Histoplasma capsulatum var. duboisii, the latter is unknown outside Africa and is common where bat and bird droppings accumulate over time (such as excavation sites and caves).[1]H. capsulatum (Darling's disease or Caver's disease) most commonly affects the lung. Adrenal involvement, commonly associated with lung, liver, cutaneous, or digestive tract involvement, presents customarily as chronic fatigue syndrome, incidental bilateral or unilateral adrenomegaly, fever of unknown origin, unexplained weight loss or chronic adrenal insufficiency.[2] Addisonian crisis (differentials – acute hypoglycemia, acute hypopituitarism, septic shock, etomidate toxicity during ventilation) as the index presentation is quite rare and isolated adrenal gland involvement is even rare still, both of which were seen in our patient.[3],[4] Early diagnosis and differentiation from other caseating and necrotizing granulomatous diseases (tuberculosis, cryptococcosis, and lymphomas) is of utmost importance. The current Infectious Diseases Society of America guidelines recommend itraconazole for mild-to-moderate infection and liposomal amphotericin B for overwhelming acute pulmonary histoplasmosis, chronic pulmonary histoplasmosis, and all forms of progressive disseminated histoplasmosis and other acute life-threatening presentations of the disease. Monitoring of itraconazole levels should commence 2 weeks after the start of treatment with levels >10 mcg/mL related to toxicity.[5] Our patient was started on weight-based liposomal amphotericin B along with intravenous hydrocortisone (tapering dose) planned to be continued for 2 weeks, followed by oral itraconazole for 1 year duration. He was extubated on the 5th day after admission and is currently recovering uneventfully.
Figure 1: Biopsy from the adrenal gland lesion showing extensive caseation (arrows, a [H and E, ×10] and b [H and E, ×20]) with necrotizing granulomatous inflammation (dashed arrows A [H and E, ×10]) with multinucleated giant cells

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Figure 2: Biopsy of adrenal lesion showing numerous ovoid, uninuclear, intracellular and extracellular yeast forms with thin walls and narrow-based budding, consistent with Histoplasma species (a, arrows, [Periodic acid Schiff, ×40] and b, Grocott's methenamine silver stain, ×40)

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   References Top

1.
Wong P, Houston S, Power B, Lalor E, Bain VG. A case of histoplasma capsulatum causing granulomatous liver disease and addisonian crisis. Can J Gastroenterol 2001;15:687-91.  Back to cited text no. 1
    
2.
Larbcharoensub N, Boonsakan P, Aroonroch R, Rochanawutanon M, Nitiyanant P, Phongkitkarun S, et al. Adrenal histoplasmosis: A case series and review of the literature. Southeast Asian J Trop Med Public Health 2011;42:920-5.  Back to cited text no. 2
    
3.
Hu AS, Hu AS, Hu CH. Histoplasmosis with addisonian crisis: Call for bird control. Med J Malaysia 2015;70:104-5.  Back to cited text no. 3
    
4.
Bhansali A, Das S, Dutta P, Walia R, Nahar U, Singh SK, et al. Adrenal histoplasmosis: Unusual presentations. J Assoc Physicians India 2012;60:54-8.  Back to cited text no. 4
    
5.
Turashvili G, Cunningham KS. Bilateral adrenal histoplasmosis in a man with chronic alcoholism. J Microbiol Immunol Infect 2016;49:797-8.  Back to cited text no. 5
    

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Correspondence Address:
Cyriac Abby Philips
Philip Augustine Associates, PVS Memorial Hospital Campus, Kaloor, Cochin - 682 017
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_717_16

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