|Year : 2016 | Volume
| Issue : 4 | Page : 300-302
A rare fatal case of disseminated cutaneous blastomycosis in a child
Amit Shekhar1, Pradeep Kumar Gupta2, Pratik Gahalaut1, Nitin Mishra1
1 Department of Dermatology, Shri Ram Murti Smarak Institute of Medical Sciences, Bareilly, Uttar Pradesh, India
2 Department of Pediatrics, Shri Ram Murti Smarak Institute of Medical Sciences, Bareilly, Uttar Pradesh, India
|Date of Web Publication||7-Oct-2016|
69, Silver Estate, Bareilly - 243 006, Uttar Pradesh
Source of Support: None, Conflict of Interest: None
We report a rare fatal case of disseminated blastomycosis in an otherwise healthy 12-year-old child. He presented with multiple verrucous, hyperkeratotic suppurating plaques, and multiple sinuses discharging thick pus over the face, chest, and upper and lower limbs for the last 8 months. Clinical diagnosis was confirmed by histopathology which showed thick-walled, clustered refractile yeast cells with broad-based unidirectional budding resembling hyphae. Although he was vigorously treated by intravenous amphotericin-B and itraconazole, the child succumbed to death within 2 weeks of presentation. This case is being reported for its rarity and fatal outcome. Besides, in the present case, widespread cutaneous spread occurred without any other systemic involvement.
Keywords: Cutaneous blastomycosis, deep fungal infection, itraconazole
|How to cite this article:|
Shekhar A, Gupta PK, Gahalaut P, Mishra N. A rare fatal case of disseminated cutaneous blastomycosis in a child. Indian J Paediatr Dermatol 2016;17:300-2
|How to cite this URL:|
Shekhar A, Gupta PK, Gahalaut P, Mishra N. A rare fatal case of disseminated cutaneous blastomycosis in a child. Indian J Paediatr Dermatol [serial online] 2016 [cited 2020 May 27];17:300-2. Available from: http://www.ijpd.in/text.asp?2016/17/4/300/187898
| Introduction|| |
Blastomycosis is caused by a dimorphic fungus Blastomyces dermatitidis. Recently, a few cases have been reported from India. Only 3–11% of all reported cases are aged <20 years. Cutaneous forms are almost always secondary to pulmonary disease. We report a rare fatal case of disseminated cutaneous blastomycosis without pulmonary involvement in pediatric age group.
| Case Report|| |
A 12-year-old boy presented to the casualty with 8 months history of multiple, verrucous, hyperkeratotic, and suppurating plaques over the face, chest, and limbs. At the time of admission, there were multiple hyperkeratotic and scaly plaques of varying sizes on the face, neck, chest, hands, and legs. His lesions started as a plaque on the left knee joint which did not respond to Anti tubercular treatment. At the time of admission, there were multiple suppurating plaques seen on the base of neck, chest, hands, and legs. He had Grade 3 malnutrition as per the WHO classification with a height of 130 cm and 20 kg.
Clinical examination [Figure 1] showed multiple ulcers of size ranging from 1 cm × 5 cm on the base of the neck, B/L forearms, and legs. Thick suppurative pus was exuding from some of these ulcers. The edges were ill-defined and the floor had pus and granulation tissue on examination. Ulcers were mobile and nontender.
|Figure 1: Verrucous plaques on the tip of nose and the base of neck and jaw |
Click here to view
Respiratory system and cardiovascular system examination were insignificant. Abdominal examination failed to reveal any organomegaly, ascites, or fluid thrill. The child was conscious, alert but irritable and disoriented to time, place, and person. His reflexes were normal. His blood culture was negative, but pus culture showed heavy growth of Escherichia coli. Hemogram revealed a low hemoglobin of 6.6 g/dl, total leukocyte count of 25,500/mm 3, differential leukocytes count of neutrophils 87%, lymphocytes 13%, platelet count 530,000/mm 3, erythrocyte sedimentation rate 40 mm/h, serum alkaline phosphatase 129 IU/L, serum glutamate pyruvate transaminase18 IU/L, serum glutamate-oxaloacetate transaminase 38 IU/L, serum proteins 4.6 g/dl, albumin 1.4 g/dl, albumin/globulin ratio 0.4, serum uric acid 9.7 mg/dl, blood urea nitrogen 23 mg/dl, serum sodium 114 mmol/L and serum potassium 4.8 mmol/L. HIV test was negative. Chest radiograph revealed normal radiographic findings. Cerebrospinal fluid obtained from lumbar puncture was within normal limits; Koh mount showed broad-based budding organisms on tissue scrapings from ulcer margins. Gram staining showed Gram-negative cocci. Blood culture was sterile. Provisional clinical diagnosis of blastomycosis was confirmed by histopathology report.
Biopsy [Figure 2] and [Figure 3] showed upper and lower dermal nodular dense tuberculoid and suppurative granulomatous infiltrate made up of lymphocytes, plasma cells, histiocytes, and epithelioid cells with occasional Langhans and foreign body giant cells and small collections of neutrophils. Overlying epidermis shows moderate spongiotic psoriasiform change. Clusters of large, variably sized thick walled brown colored refractile yeast cells with broad based unidirectional budding were observed. At places, rows of such yeast cells resemble hyphae. Periodic acid–Schiff (PAS) stain for fungi was positive.
|Figure 2: Thick brown-colored refractile yeast cells in clustered pattern with broad-based unidirectional budding (H and E, ×40)|
Click here to view
|Figure 3: Upper and lower dermal nodular granulomatous infiltrates (H and E, ×10)|
Click here to view
Thereafter, an aggressive treatment was started to control the disseminated blastomycosis. The patient was started on oral itraconazole 5 mg/kg/day. Intravenous (I.V.) cefoperazone and sulbactam combination was started at the dose 20 mg/kg/day along with I.V. vancomycin 3 mg/kg/day dose. His skin lesions improved. He was also given three packed blood transfusions over a period of 7 days. The child lost consciousness on the 4th day when he was started on injection amphotericin B 5 mg/kg/body weight. Unfortunately, he succumbed to death on the day 15th of admission.
| Discussion|| |
Blastomycosis is a relatively uncommon chronic granulomatous disease even in the endemic regions, more so in India. It is rarely diagnosed in children. In children, lung involvement is followed by skin, bone, and central nervous system (CNS) in descending order of incidence. However, in the present case, there was no lung involvement. Diagnostic delays are not uncommon and often result in increased morbidity and mortality.
Primary cutaneous blastomycosis has infrequently been reported after dog bites also; such patients may not disease clinically active pulmonary as in our present case. However, the parents denied any history of dog bite in the present patient.
Blastomycosis is caused by inhalation of conidia of dimorphic fungi B. dermatitidis although direct inoculation can occur. Skin is the most frequent site (40–80%) of dissemination of pulmonary blastomycosis followed by bone (10–50%), genitourinary tract (10–30%), and CNS (1–5%). In our case, skin seems to be the primary site of inoculation. Disseminated blastomycosis occurs more frequently in immunocompromised individuals. To the best of our knowledge, this is the second case of disseminated blastomycosis from India in pediatric age group. In our case, there was no lung and CNS involvement and disease appeared limited to skin.
Diagnosis of blastomycosis may be confirmed through identification of yeasts of B. dermatitidis in body fluids and tissues. Although culture is the gold standard method for the detecting fungus, it is positive in only 3.2% cases. Even in the absence of positive culture, the case can be diagnosed by histopathological examination and fine needle aspiration cytology of the lesions. Yeast forms can be seen in hematoxylin and eosin-stained sections but are better visualized by methenamine silver stain or PAS stain.
For children with mild to moderate infection, oral itraconazole, at a dosage of 10 mg/kg/day (to a maximum of 400 mg orally per day) for 6–12 months, is recommended. Serum levels of itraconazole should be determined after the patient has received this agent for at least 2 weeks to ensure adequate drug exposure.
For children with severe blastomycosis, lipid formulation AmB, at a dosage of 3–5 mg/kg/day, is recommended for initial therapy. Itraconazole, 10 mg/kg/day (up to 400 mg per day) as step-down therapy, is recommended for a total of 12 months. Despite antifungal therapy, the mortality rate of blastomycosis associated acute respiratory distress syndrome is still 50–89%.
| Conclusion|| |
Early detection and diagnosis of a suspected case of blastomycosis and prompt treatment with antifungal agents are the key to the successful management of blastomycosis. This case is being reported for its rarity and the fact that dissemination over skin may appear without any lung involvement in pediatric age group.
The authors wish to thank Dr. Uday Khopkar, MD for reviewing the histopathological slide for this case in academic interest.
Financial Support and Sponsorship
Conflicts of Interest
There are no conflicts of interest.
| References|| |
Dhamija A, D'Souza P, Salgia P, Meherda A, Kothiwala R. Blastomycosis presenting as solitary nodule: A rare presentation. Indian J Dermatol 2012;57:133-5.
Mason AR, Cortes GY, Cook J, Maize JC, Thiers BH. Cutaneous blastomycosis: A diagnostic challenge. Int J Dermatol 2008;47:824-30.
Rao GR, Narayan BL, Durga Prasad BK, Amareswar A, Sridevi M, Raju B. Disseminated blastomycosis in a child with a brief review of the Indian literature. Indian J Dermatol Venereol Leprol 2013;79:92-6.
Lemos LB, Guo M, Baliga M. Blastomycosis: Organ involvement and etiologic diagnosis. A review of 123 patients from Mississippi. Ann Diagn Pathol 2000;4:391-406.
Garvey K, Hinshaw M, Vanness E. Chronic disseminated cutaneous blastomycosis in an 11-year old, with a brief review of the literature. Pediatr Dermatol 2006;23:541-5.
Chapman SW, Dismukes WE, Proia LA, Bradsher RW, Pappas PG, Threlkeld MG, et al.
Clinical practice guidelines for the management of blastomycosis: 2008 update by the Infectious Diseases Society of America. Clin Infect Dis 2008;46:1801-12.
Lahm T, Neese S, Thornburg AT, Ober MD, Sarosi GA, Hage CA. Corticosteroids for blastomycosis-induced ARDS: A report of two patients and review of the literature. Chest 2008;133:1478-80.
[Figure 1], [Figure 2], [Figure 3]