An Official publication of The Asian Congress of Neurological Surgeons (AsianCNS)

Search Article
Home About us Editorial board Search Ahead of print Current issue Archives Submit article Instructions Advertise Subscribe Contacts Login  Facebook Tweeter
  Users Online: 5735 Home Print this page Email this page Small font sizeDefault font sizeIncrease font size  

   Table of Contents      
CASE REPORT
Year : 2021  |  Volume : 16  |  Issue : 3  |  Page : 582-586

A severe case of brain myiasis: Treatment rationale and review of literature


1 Department of Neuroscience, Università di Padova, Padova, Italy
2 Department of Neurosurgery, Aulss 2 Marca Trevigiana, Treviso, Italy

Date of Submission03-Dec-2020
Date of Decision01-Feb-2021
Date of Acceptance26-Mar-2021
Date of Web Publication14-Sep-2021

Correspondence Address:
Dr. Enrico Giordan
Department of Neurosurgery, Aulss 2 Marca Trevigiana, Via Piazzale 1, Treviso, Treviso 31100
Italy
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ajns.AJNS_521_20

Rights and Permissions
  Abstract 

Cerebral myiasis is a rare condition caused by a parasitic infestation of fly larvae feeding on the host's necrotic or living tissue. Only 16 cases of cerebral myiasis have been published. We presented the case of a 72-year-old man with a neglected infestation of an extensive ulcerative cancer of the scalp. A large cranial lesion, with exposed brain and dura mater and severe Sarcophaga carnaria maggot infestation, was evident. We gently removed the maggots and covered the defect with thick gauze and sodium hypochlorite solution dressing. We additionally present a review of the literature to highlight shared features and suggestions for care management. In all cases, there was an absence of fatal meningitis and encephalitis, which is surprising given the open skull erosion with prolonged cortical exposure and points to the protective effects of larvae wound infestation.

Keywords: Brain, cerebral infestation, larvae, myiasis, neglected


How to cite this article:
Curzi C, Bartoletti V, Canova G, Giordan E. A severe case of brain myiasis: Treatment rationale and review of literature. Asian J Neurosurg 2021;16:582-6

How to cite this URL:
Curzi C, Bartoletti V, Canova G, Giordan E. A severe case of brain myiasis: Treatment rationale and review of literature. Asian J Neurosurg [serial online] 2021 [cited 2021 Dec 4];16:582-6. Available from: https://www.asianjns.org/text.asp?2021/16/3/582/325950


  Introduction Top


Myiasis in humans is caused by a parasitic infestation of fly larvae feeding on the host's tissues.[1] Most cases occur in tropical and subtropical regions.[2] Myiasis in higher-income nations is typically associated with travel or immigration. Additional factors, such as hygiene, diabetes, immunocompromised status, and delay in seeking medical attention, may contribute.[3]

Cerebral localization of myiasis is exceedingly rare,[2],[3],[4] and the involvement of a large area of brain tissue can result in very severe manifestations. Here, we describe a rare case of cerebral myiasis in Northeast Italy and present a literature review to highlight the pathological features of and suggestions for managing such occurrences.


  Procedure Top


In 2017, a 72-year-old male was diagnosed with right frontal basal cell cancer. The cancer was treated weekly with topical agents. Unfortunately, due to improper nursing care and missed clinical appointments, the patient developed a right frontal extensive ulcerative lesion.

In June 2019, the patient was found stuporous with a large cranial lesion with exposed brain and severe maggot infestation [Figure 1]a.
Figure 1: (a) Severe frontal maggot infestation. (b) Larvae debridement with saline-sodium hypochlorite solution. (c) Removing necrotic and pathologic bone and skin tissue from defects margins

Click here to view


The patient lived alone and was resistant to nursing care. He had been advised to seek medical attention repeatedly but had always refused. Maggot infestation went unrecognized by relatives. The patient was likely demented, which may explain his refusal of care and loss of awareness.

Vital signs were normal, but he was febrile (38.2°C/100.8°F). Complete blood cell count revealed a white blood cell count of 15.1 K/μL with 91% neutrophils [Supplementary Video 1] [Additional file 1]. Physical examination revealed bilateral frontal scalp and cranial erosion (13 cm × 11 cm) [Figure 1]b. The edges of the skin were hypertrophied and erythematous, and the dura mater was exposed. A cluster of approximately 100 maggots was found at the center of the defect, without apparent cerebrospinal fluid (CSF) leakage. A computed tomography (CT) scan [Figure 2] revealed an extensive frontal bony defect with brain exposure.
Figure 2: (a) Sagittal bone computed tomography scan. (b) Coronal bone computed tomography scan. (c) Axial computed tomography scan. It is evident the severe and profound destruction of the scalp layer done by the basal cell carcinoma

Click here to view


We urgently cleaned the exposed wound to debride the maggots, obtain tissue specimens, and protect brain integrity. An infectious disease specialist and a plastic surgeon were contacted, and a literature search was completed to plan the best approach. (Cerebral myiasis video).

The patient was positioned supine and tilted into a Trendelenburg position, using gravity to aid in removing the maggots and checking for spontaneous CSF leakage [Figure 1]c. After the removal of the larvae, the surface was moisturized with a 50:50 sodium hypochlorite sterile saline solution.

After maggot removal, the bone was debrided until it obtained a normal aspect. The dura mater was gently scratched to remove any debris. The sagittal sinus was not touched to avoid bleeding, and the infiltrate tumor was debulked where possible [Figure 3]a. In the absence of signs of CSF leakage, we avoid dura substitutes to lower the risk of infection. The exposed cortex was covered with patches of fibrin and collagen matrix. The defect was eventually covered with a sodium hypochlorite solution dressing and fat gauzes [Figure 3]b.
Figure 3: (a) Debrided defect. Remnants of the superior sagittal sinus are visible in the center. (b) Dressin g the defect with fatty gauze

Click here to view


We administered intravenous broad-spectrum antibiotics. Urine and blood cultures were negative, and the fever resolved rapidly. There were no signs of new larvae development. The maggots were classified as belonging to the Sarcophaga carnaria species. Microbiological and CFS cultures were normal. After defervescence, the patient was discharged to a medical ward, and plastic reconstruction was planned. Unfortunately, the patient refused further intervention and died two months later.

See proposed video for case presentation. The case is described and narrated extensively in the video provided with the paper (Cerebral myiasis video).


  Discussion Top


Both primary and secondary cerebral myiasis are exceedingly rare conditions, especially in high-income countries.[1],[2],[5] In primary myiasis, the larvae penetrate the skin via a puncture wound, while in the secondary variety, fly eggs are laid into a skin ulcer.[4] Benign or malignant dermatological or traumatic conditions have been associated with secondary myiasis, but rarely as extensively as presented here[3],[4],[5],[6] [Table 1].
Table 1: Cases of cerebral myiasis reported in the literature

Click here to view


Cerebral myiasis is rare, with only 16 published cases, and may lead to life-threatening complications.[2],[3],[4],[7],[8],[9],[10],[11],[12],[13],[14],[15] The majority of known cerebral myiasis cases in higher-income countries were consequent to neglected skin tumors,[3],[14],[15] while in lower-income countries, they were mostly attributable to inadequate wound care.[4],[9],[16]

Almost all cerebral myiasis cases were found close to the scalp surface in frontal regions.[3],[8],[14],[15],[16],[17] Rarely, intraparenchymal involvement with consequent hematoma formation or abscess development was found.[10],[13],[17] Symptom presentation may be delayed from a few days to years, particularly among patients with dementia. The larvae may penetrate the brain and spread via CSF to the subependymal space, where it can remain undetected for years.[10] Deep parenchymal involvement and older age [4],[14],[16],[18],[19] are associated with decreased survival.[10],[16],[17],[18],[19]

Proper management requires a multidisciplinary team (i.e., infectious specialists, radiologists, neurosurgeons, and plastic surgeons). Among patients with a confirmed calvarium erosion, a contrast-enhanced CT or magnetic resonance imaging (MRI) is recommended to rule out venous sinuses' integrity or for phlogistic processes.[6] MRI imaging may also help assess the integrity of arachnoidal layers or signs of infection. Furthermore, CSF sampling is recommended for cytological and microbiological evaluation.

Treatment relies on larvae removal, debridement of necrotic/malignant tissue, and reconstruction of the defect. In the literature, several agents or drugs are proposed for larvae removal.[1],[15] In the case of massive erosion and brain exposition, gentle irrigation with saline solution and mechanical removal is suggested.[2],[9],[14],[15] A sodium hypochlorite solution, both intraoperatively and as a dressing, is also recommended. Intravenous broad-spectrum antibiotics (>6 weeks) are mandatory to control and prevent secondary infection. In addition, in cases with brain or dural sinus invasion, it is better to limit the debulking maneuvers to visible lesions to avoid additional damage.[4] Further interventions, such as reconstruction surgeries, should be considered only after myiasis resolution.

There was a surprising absence of meningitis or encephalitis in the setting of an open skull erosion with prolonged cortical exposure in all available reports. Maggot infestation may have reduced bacterial infection risk by protecting the tissue surface area.[3],[9],[15] Although the patient was febrile, there was no evidence of systemic infection. This may be attributable to the beneficial effects of wound infestation with larvae, which were first used medically during the American Civil War.[15] The larval activity might have prevented a secondary bacterial infection by eating dead tissues, leading to more prolonged survival despite delayed treatment.[9],[14]

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
  References Top

1.
Sesterhenn AM, Pfützner W, Braulke DM, Wiegand S, Werner JA, Taubert A. Cutaneous manifestation of myiasis in malignant wounds of the head and neck. Eur J Dermatol 2009;19:64-8.  Back to cited text no. 1
    
2.
Piña-Tornés AA, Salvador-Fernández CL, Lindao-Camacho R, González-Longoria-Boada LB, Vintimilla-Burgos NP, Selles Almarales M. Massive cutaneous myiasis mimicking brain invasion. Case report and literature review. MÉD.UIS. 2016;29:145-53.  Back to cited text no. 2
    
3.
Cheshier SH, Bababeygy SR, Higgins D, Parsonnet J, Huhn SL. Cerebral myiasis associated with angiosarcoma of the scalp: Case report. Neurosurgery 2007;61:E167.  Back to cited text no. 3
    
4.
Aggarwal A, Maskara P. Maggots in the brain: Sequelae of ignored scalp wound. World Neurosurg 2018;109:115-6.  Back to cited text no. 4
    
5.
Gabriel JG, Marinho SA, Verli FD, Krause RG, Yurgel LS, Cherubini K. Extensive myiasis infestation over a squamous cell carcinoma in the face. Case report. Med Oral Patol Oral Cir Bucal 2008;13:E9-11.  Back to cited text no. 5
    
6.
Zhou X, Kambalame DM, Zhou S, Guo X, Xia D, Yang Y, et al. Human Chrysomya bezziana myiasis: A systematic review. PLoS Negl Trop Dis 2019;13:e0007391.  Back to cited text no. 6
    
7.
Gilly R, Lapras C, Mamelle JC, Challamel MJ, Ghilhot JH, Nicolas A, et al. Hypodermic migrant myiasis with intracerebral hematoma. Apropos of a case in a 7-year-old child. Pediatrie 1976;31:67-75.  Back to cited text no. 7
    
8.
Navarro JN, Alves RV. Postoperative cerebral myiasis: A rare cause of wound dehiscence in developing countries. Surg Neurol Int 2016;7:69.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Giri SA, Kotecha N, Giri D, Diyora B, Nayak N, Sharma A. Cerebral myiasis associated with artificial cranioplasty flap: A case report. World Neurosurg 2016;87:661.e13-6.  Back to cited text no. 9
    
10.
Marco de Lucas E, Díez C, Gutiérrez A, Montiaga F, Arnáiz J, Mandly AG, et al. Unusual MRI findings in a patient with history of frontal fracture and skin infestation by fly larvae, as a possible sign of intracerebral myiasis. Clin Neurol Neurosurg 2008;110:725-8.  Back to cited text no. 10
    
11.
Zucoloto S, Rossi MA. Facial myiasis with spreading to the cranial vault. Rev Bras Med 1971;28:13-6.  Back to cited text no. 11
    
12.
Froomin LL. Intradural cyst of parasitic origin (myiasis clinic). Zh Ushn Nos Gorl Bolezn 1939;16:427-33.  Back to cited text no. 12
    
13.
Kalelioğlu M, Aktürk G, Aktürk F, Komsuoğlu SS, Kuzeyli K, Tiğin Y, et al. Intracerebral myiasis from Hypoderma bovis larva in a child. Case report. J Neurosurg 1989;71:929-31.  Back to cited text no. 13
    
14.
Terterov S, Taghva A, MacDougall M, Giannotta S. Posttraumatic human cerebral myiasis. World Neurosurg 2010;73:557-9.  Back to cited text no. 14
    
15.
Arbit E, Varon RE, Brem SS. Myiatic scalp and skull infection with diptera Sarcophaga: Case report. Neurosurgery 1986;18:361-2.  Back to cited text no. 15
    
16.
Rossi MA, Zucoloto S. Fatal cerebral myiasis caused by the tropical warble fly, Dermatobia hominis. Am J Trop Med Hyg 1973;22:267-9.  Back to cited text no. 16
    
17.
Pouillaude JM, Dupont J, Gilly R, Lapras C. Intracerebral myiasis in a child. Pediatr Radiol 1980;10:121-3.  Back to cited text no. 17
    
18.
Tauziede-Espariat A. Infections of the Central Nervous System: Pathology and Genetics, 2020. Chapter 49: Brain myiasis. doi: 10.1002/9781119467748.ch49]. [doi: 10.1002/9781119467748.ch49].  Back to cited text no. 18
    
19.
Semenov PV. A case of penetration of Hypoderma lineatum de Villers larva into the human brain. Med Parazitol (Mosk) 1969;38:612-3.  Back to cited text no. 19
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1]



 

Top
 
 
  Search
 
<
Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
Access Statistics
Email Alert *
Add to My List *
* Registration required (free)  

 
  In this article
   Abstract
  Introduction
  Procedure
  Discussion
   References
   Article Figures
   Article Tables

 Article Access Statistics
    Viewed282    
    Printed4    
    Emailed0    
    PDF Downloaded45    
    Comments [Add]    

Recommend this journal