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Table of Contents   
CASE REPORT  
Year : 2014  |  Volume : 7  |  Issue : 4  |  Page : 199-201
Fulminant cranio-cerebral invasive aspergillosis in a patient with phytophotodermatitis


1 Department of Surgery, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha, Maharashtra, India
2 Department of ENT, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha, Maharashtra, India
3 Department of Dermatology, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha, Maharashtra, India

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Date of Web Publication5-Mar-2015
 

   Abstract 

Cranio-cerebral invasive aspergillosis of sino-nasal involving the skull base is rare and associated with a very high mortality due to its peculiarly fulminant clinical course. We present an unusual case of 55-year-old diabetic female who initially treated for phytophotodermatitis, but later on she developed massive invasive cerebral aspergillosis. Advances in diagnosis and management of aspergillosis with improved and early detection and more effective therapy have promised some success in the management of these devastating infections.

Keywords: Aspergillosis, fungus, phytophotodermatitis, skull base

How to cite this article:
Agrawal A, Baisakhiya N, Singh AL. Fulminant cranio-cerebral invasive aspergillosis in a patient with phytophotodermatitis. Ann Trop Med Public Health 2014;7:199-201

How to cite this URL:
Agrawal A, Baisakhiya N, Singh AL. Fulminant cranio-cerebral invasive aspergillosis in a patient with phytophotodermatitis. Ann Trop Med Public Health [serial online] 2014 [cited 2019 Sep 22];7:199-201. Available from: http://www.atmph.org/text.asp?2014/7/4/199/150113

   Introduction Top


Cranio-cerebral invasive aspergillosis of sino-nasal involving the skull base is rare and associated with a very high mortality due to its peculiarly fulminant clinical course. [1],[2],[3],[4] We present an unusual case of a diabetic female who initially treated for phytophotodermatitis, but later on she developed massive invasive cerebral aspergillosis.


   Case Report Top


A 55-year-old female managed for erythematous, vesicular, pruritic, and bullous skin lesions on her scalp, face, upper chest, abdomen, and extremities, which were developed over 24 h [Figure 1]a-d]. With a diagnosis of phytophotodermatitis, she received steroid and antihistaminics at a peripheral hospital. She was a known diabetic. She was apparently alright for next 7 days that she noticed rapid deterioration of vision in both the eyes, and when she presented to us, she was completely blind. Neurological examination was suggestive of bilateral cortical blindness. Fundus was normal. Higher mental functions, motor, and sensory examination was normal. A magnetic resonance imaging of the brain showed extensive infarction of both the occipital lobes, and diffuse signal changes in the clivus and occipital bone and extensive lesions involving basal cisterns and encasing the major vessel [Figure 2]a-c]. There was diffuse enhancement of the blood vessels [Figure 3]. Magnetic resonance venogram showed diffuse narrowing of the major vessels that resulted in the diffuse cerebral infarcts [Figure 4]. A diagnosis of fungal infection was suspected. Biopsy from the sphenoid sinus was negative however the tests for immunoglobulin E specific for Aspergillus was positive. The patient was started on antifungal therapy (amphotericin B and metronidazole) without any improvement in the clinical condition.
Figure 1: (a-d) Hyperpigmented patches and macules present on the exposed parts of the body

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Figure 2: (a-c) Magnetic resonance imaging brain showing diffuse signal changes in the clivus, occipital bone and bilateral infarction involving the occipital lobe

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Figure 3: Computed tomography scan showing diffuse enhancement of the major vessels and erosion of the skull base

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Figure 4: Magnetic resonance venogram showing diffuse narrowing of the major vessels probably the cause of neurological status

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   Discussion Top


Phytophotodermatitis is a phototoxic reaction characterized by skin eruption that results from the interaction of radiant energy from the sun (ultraviolet-A radiation-wavelengths of 320-400 nm) with photosensitizing compounds found in many plant varieties, including several isomers of psoralens (furocoumarins). [5],[6] Phytophotodermatitis is a clinical diagnosis and typical lesions with unusual configurations suggest the diagnosis. [6] The acute phase of phytophotodermatitis is characterized by erythematous plaques, vesicles, and bullae, and over a period the lesions may become erythematous or appear as hyperpigmented patches. The skin eruptions may appear hours to days after exposure, and the exposed parts of the body are, usually, involved. [6] There is no specific treatment for this reaction however in severe cases, analgesics, antihistamines for itching, wet compresses and topical steroids may be used. Blistered areas need to be kept clean to prevent secondary infection. The hyperpigmentation fades over time as it does in sunburned skin. [5],[6] As the patient was a farmer she would have been exposed to furocoumarins as well as fungus through handling of plants while working in the fields and also in present case administration of steroids in a known diabetic resulted in further immnocompromised state and infection with Aspergillus. The temperate climate of our country and constant exposure to the high spore content of pathogenic Aspergillus species in the "mouldy" work environment [7] posed a high risk to the patient. Cerebral infarction associated with skull base aspergillosis can mimic like any other causes of cerebrovascular disease. [2],[3],[4] Vascular invasion in cerebral aspergillosis can lead to thrombosis, infarction, hemorrhage, or mycotic aneurysms. [8],[9] In its severe form, the hyphal elements of the organisms can occlude the veins (frequently in basal ganglia, thalamus, corpus callosum, and cortical or subcortical localizations) [9],[10],[11] or lenticulostriate and thalamoperforating arteries [12] leading to infarction. Imaging is helpful to suspect a diagnosis of aspergillosis. [1] As in present case detection of the fungus may not be possible in biopsy; however, the diagnosis can be suspected based on serology. [2] Radical surgical excision followed by aggressive antifungal chemotherapy is the mainstay of treatment for invasive aspergillosis. [3] However, once the intracranial invasion of infection occurs; it is difficult to control with conventional antifungal (possibly due to difficulties in the transport of antifungal agents through the hyphal element into the brain tissues) and surgical therapies and the mortality is very high. [13] Advances in diagnosis and management of aspergillosis with improved and early detection and more effective therapy have promised some success in the management of these devastating infections. [14],[15]

 
   References Top

1.
Siddiqui AA, Bashir SH, Ali Shah A, Sajjad Z, Ahmed N, Jooma R, et al. Diagnostic MR imaging features of craniocerebral aspergillosis of sino-nasal origin in immunocompetent patients. Acta Neurochir (Wien) 2006;148:155-66.  Back to cited text no. 1
    
2.
Ueki Y, Kazuta T, Naitou E, Hayashi M, Tanaka K, Mizutani T, et al. A case of CNS aspergillosis developing orbital apex syndrome and causing mycotic aneurysm and the subsequent cerebral infarction. Rinsho Shinkeigaku 2002;42:761-5.  Back to cited text no. 2
    
3.
Dubey A, Patwardhan RV, Sampth S, Santosh V, Kolluri S, Nanda A. Intracranial fungal granuloma: Analysis of 40 patients and review of the literature. Surg Neurol 2005;63:254-60.  Back to cited text no. 3
    
4.
Hurst RW, Judkins A, Bolger W, Chu A, Loevner LA. Mycotic aneurysm and cerebral infarction resulting from fungal sinusitis: Imaging and pathologic correlation. AJNR Am J Neuroradiol 2001;22:858-63.  Back to cited text no. 4
    
5.
Arnold HL, Odom RB, James WD. Andrews' Diseases of the Skin. 8 th ed. Philadelphia: WB Saunders Co.; 1990. p. 35-7.  Back to cited text no. 5
    
6.
Goskowicz MO, Friedlander SF, Eichenfield LF. Endemic "lime" disease: Phytophotodermatitis in San Diego county. Pediatrics 1994;93:828-30.  Back to cited text no. 6
    
7.
Murthy JM, Sundaram C, Prasad VS, Purohit AK, Rammurti S, Laxmi V. Sinocranial aspergillosis: A form of central nervous system aspergillosis in South India. Mycoses 2001;44:141-5.  Back to cited text no. 7
    
8.
Ashdown BC, Tien RD, Felsberg GJ. Aspergillosis of the brain and paranasal sinuses in immunocompromised patients: CT and MR imaging findings. AJR Am J Roentgenol 1994;162:155-9.  Back to cited text no. 8
    
9.
DeLone DR, Goldstein RA, Petermann G, Salamat MS, Miles JM, Knechtle SJ, et al. Disseminated aspergillosis involving the brain: Distribution and imaging characteristics. AJNR Am J Neuroradiol 1999;20:1597-604.  Back to cited text no. 9
    
10.
Miaux Y, Ribaud P, Williams M, Guermazi A, Gluckman E, Brocheriou C, et al. MR of cerebral aspergillosis in patients who have had bone marrow transplantation. AJNR Am J Neuroradiol 1995;16:555-62.  Back to cited text no. 10
    
11.
Guermazi A, Gluckman E, Tabti B, Miaux Y. Invasive central nervous system aspergillosis in bone marrow transplantation recipients: An overview. Eur Radiol 2003;13:377-88.  Back to cited text no. 11
    
12.
Keyik B, Edgüer T, Hekimoglu B. Conventional and diffusion-weighted MR imaging of cerebral aspergillosis. Diagn Interv Radiol 2005;11:199-201.  Back to cited text no. 12
    
13.
Nabika S, Kiya K, Satoh H, Mizoue T, Araki H, Oshita J. Local administration of amphotericin B against aspergilloma in the prepontine cistern - Case report. Neurol Med Chir (Tokyo) 2007;47:89-92.  Back to cited text no. 13
    
14.
Streppel M, Bachmann G, Arnold G, Damm M, Stennert E. Successful treatment of an invasive aspergillosis of the skull base and paranasal sinuses with liposomal amphotericin B and itraconazole. Ann Otol Rhinol Laryngol 1999;108:205-7.  Back to cited text no. 14
    
15.
Gottfredsson M, Steingrímsdóttir H. Disseminated invasive aspergillosis in a patient with acute leukaemia. Acta Biomed 2006;77 Suppl 2:10-3.  Back to cited text no. 15
    

Top
Correspondence Address:
Amit Agrawal
Department of Surgery, Division of Neurosurgery, Datta Meghe Institute of Medical Sciences, Sawangi (Meghe), Wardha - 442 004, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1755-6783.150113

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