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OJHAS Vol. 8, Issue 3: (2009
Jul-Sep) |
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Subcutaneous Phycomycosis in a Child |
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Manjiri R Naniwadekar, Professor, Department of Pathology,
Sunil V Jagtap, Associate Professor, Department of Pathology,,
Balkrishna
P Nikam, Lecturer, Department of Dermatology,
Kajal D Sanghavi, Assistant Lecturer, Dept. of Pathology,
Krishna Institute of Medical Sciences Deemed University, Karad.
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Address For Correspondence |
Professor, Department Of Pathology,
Krishna Institute of Medical Sciences Deemed University,
Karad, India.
E-mail:
manjiri61@gmail.com |
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Naniwadekar MR, Jagtap SV, Nikam BP, Sanghavi KD. Subcutaneous Phycomycosis in a Child Online J Health Allied Scs.
2009;8(3):14 |
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Submitted: Aug 14, 2009; Accepted:
Sep 10, 2009 Published: Nov 15, 2009 |
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| Abstract: |
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Subcutaneous phycomycosis is a rare entity. We hereby
report a case of subcutaneous phycomycosis in 18 months old female child who presented with a painless, non-tender
swelling on the thigh. Skin biopsy showed eosinophilic granuloma lying deep in the subcutaneous tissue, with sparse
hyphae. Culture on Sabouraud's dextrose agar showed characteristic colonies. Patient was started on oral potassium iodide.
The swelling was completely resolved after one month of treatment.
Key Words: Subcutaneous phycomycosis,
Subcutaneous zygomycosis, Basidiobolus ranarum, Potassium iodide
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Subcutaneous phycomycosis
(subcutaneous zygomycosis) is one of the deep fungal infections caused by zygomatous species which is normally
found in the tropical countries.[1] Basidiobolus ranarum, a saprophytic fungus can cause a variety of clinical
manifestations including subcutaneous zygomycosis, gastrointestinal zygomycosis and occasionally an acute systemic
illness. Subcutaneous zygomycosis is the commonest presentation.[2] It is a granulomatous infection of the skin and
subcutaneous tissues characterized by the formation of fluctuant firm and non-tender swellings, generally on the
extremities and trunk.[3] It is predominantly a disease of early childhood.[4] Definitive diagnosis of subcutaneous
phycomycosis can be made on histopathological study.[5] Oral potassium iodide can completely cure the disease.[1]
In India, so far only a few cases have been described.
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An 18 months old female was brought to dermatology
clinic with complaints of a swelling over the left thigh for three months duration. Initially the swelling was
small, measuring two centimeter in diameter. After two months, the swelling increased in size, measuring eight
centimeter in diameter. Her general, physical and systemic examination and routine laboratory investigations were
normal. X-ray of the thigh revealed a soft tissue swelling without any bone involvement. On examination, the
swelling was non-tender with stony hard consistency, smooth rounded edges, raised up by finger underneath it.
(Fig 1). The overlying skin was normal. A diagnosis of subcutaneous phycomycosis was suspected on the basis of
clinical features.
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Figure 1: The swelling over thigh, with smooth rounded edges, raised up by finger underneath it |
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Skin biopsy was taken. The Haematoxylin and eosin stained sections revealed granulomatous
inflammation, with numerous eosinophils, lymphocytes and occasional giant cell lying deep in
the dermis and subcutaneous tissue and largely replacing fat.(Fig 2) An eosinophilic material ,
suggestive of 'Splendore - Hoepplie' phenomenon was seen. Gomori’s methenamine silver stain for
fungus showed occasional wide aseptate hyphae.(Fig 3) Areas of fibrosis were noted. A 10% potassium
hydroxide wet mount of the tissue revealed broad, thin walled and aseptate hyphae. Necrotic tissue
from biopsy material was cultured on Sabouraud's dextrose agar, which showed furrowed, creamy brown,
heaped up, radially folded colonies after three days incubation at 25°C.(Fig
4) On performing lactophenol cotton blue wet mount of the culture growth, numerous aseptate hyphae and numerous
smooth walled zygospores with characteristic conjugation beaks were observed.(Fig 5) The fungus
was identified as Basidiobolus ranarum. Patient was started on oral potassium iodide, three drops,
three times a day and gradually increased upto five drops three times a day for one month. After
one week of potassium iodide treatment, oral itraconazole in a dose of 100mg/day was added and was
continued for one month. The swelling was completely resolved after one month of treatment. |
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Fig 2:
Photomicrograph shows granulomatous inflammation, with numerous
eosinophils, lying deep in the dermis and subcutaneous tissue and
largely replacing fat. (Haematoxylin and eosin X100) |
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Fig 4:(Above) Furrowed, creamy brown ,
heaped up, radially folded colonies after three days incubation at
25°C cultured on Sabouraud's dextrose agar
Fig 5: (Left) Lactophenol cotton blue wet mount of the culture
growth showing numerous aseptate hyphae and numerous smooth walled zygospores with characteristic
conjugation beaks. |
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Fig 3:
Photomicrograph shows occasional wide aseptate hyphae. (Gomori’s
methenamine silver stain X400) |
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Subcutaneous phycomycosis was first described in
Indonesia in 1956.[4] It is the commonest clinical form of Basidiobolomycosis, and is endemic in South India.
Basidiobolus ranarum is a saprophytic fungus present in soil, decaying fruit and vegetable matter as well as in the gut
of amphibians and reptiles. It can cause a variety of clinical manifestations including subcutaneous zygomycosis,
gastrointestinal zygomycosis and occasionally an acute systemic illness. Subcutaneous zygomycosis is the commonest
presentation reported from many tropical countries including India.[2] However cases have not been reported from Maharashtra
and north India. Traumatic implantation is probably the mode of entry like in other subcutaneous mycoses. In the past,
clinical isolates of Basidiobolus were classified as B. ranarum, B. meristosporus and
B. haptosporus. But recent taxonomic
studies indicate that all human pathogens belong to B. ranarum.[2]
It is a granulomatous infection of the skin and subcutaneous tissues characterized by the formation
of firm and non-tender swellings, generally on the extremities, trunk and rarely other parts of the body.[3]
The disease usually occurs in children, less often in adolescents and rarely in adults. Males are much more
frequently affected than females.[3] In the study by Lal S. et al, the sites of involvement included leg,
thigh, buttock, shoulder, upper arm, thorax and abdominal wall and three patients had itching in the lesions
and three patients were having fever.[6] In the present case there were no complaints, other than the
swelling. The swelling may be lobulated with rounded edges and can be raised up by inserting the fingers
underneath it.[1] In the present case also, the swelling could be lifted up by inserting the fingers
beneath it. The possibility of mucormycosis could be ruled out by morphology of hyphae and absence of the
features such as vessel involvement and thrombosis. Also the patient was in good general health with
subcutaneous and painless swelling with intact overlying skin.
Laboratory diagnosis is based on histopathology. Histologically, Basidiobolomycosis is associated with
eosinophilic infiltration, which was also the case in our patient. This has been postulated to be due to a
predominant Th2 type of immune response with release of cytokines like IL-4 and IL-10 which in turn are
helpful in recruiting eosinophils to the affected site.[2] The other histological features are extensive
dermal and subcutaneous fibrosis and large zygomatic hyphae. The hyphae appear as short sections of broad
hyphae without septae. Some may be surrounded by an eosinophilic material which is called as 'Splendore –
Hoepplie' phenomenon.[4] In the present case, the clinical and histopathologic features of skin lesion were
characteristic of basidiobolus infection. Culture on Sabouraud's dextrose agar and lactophenol cotton blue wet
mount confirmed the diagnosis of Basidiobolomycosis.
Most patients with Basidiobolomycosis respond very well to oral potassium iodide therapy as also to azoles
particularly itraconazole.[2] In the present case also the patient responded well to potassium iodide and the
swelling was completely resolved after one month of treatment. Prasad PV et al have observed that there was a
relapse of the lesion in their patient after an excisional biopsy and have commented that surgery may hasten the
spread of infection.[1]
To summarize, subcutaneous phycomycosis is a chronic fungal infection of the children, with a few
reports from south India. The clinical and histopathologic features of skin lesion are characteristic of
Basidiobolus infection. Clinical history, eosinophilic granuloma on histopathology and special stains for
Basidiobolus help in clinching the diagnosis. Culture on Sabouraud's dextrose agar and lactophenol cotton blue wet
mount are diagnostic. Surgical excision should be avoided as the patients respond well to oral potassium iodide.
- Prasad PV, Paul EK, George RV, Ambujam S, Viswanthan P. Subcutaneous phycomycosis in a child.
Indian J Dermatol Venereol Leprol 2002;68:303-4
- Sujatha S, Sheeladevi C, Khyriem
AB, Parija SC, Thappa DM. Subcutaneous zygomycosis caused by Basidiobolus ranarum - a case report.
Indian Journal of Medical Microbiology 2003; 21(3):205-6
- Gugnani HC. A review of zygomycosis due to
Basidiobolus ranarum. Eur J Epidemiol. 1999;15(10):923-9
- Burkitt DP, Wilson A M, Jelliffe DB. Subcutaneous phycomycosis.
A review of 31 cases seen in Uganda.
Brit Med J 1964;1: 1669-72
- Chandrasekhar HR, Shashikala P, Haravi R, Kada. Subcutaneous phycomycosis.
Indian J Dermatol Venereol Leprol 1998;64:89-90
- Lal S, Baruah MC, Padiyar NV et al. Clinicopathological study of
subcutaneous phycomycosis.
Indian J Dermatol Venereal Leprol 1984;50:245-248.
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