Annals of African Medicine
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Table of Contents
CASE REPORT
Year : 2015  |  Volume : 14  |  Issue : 1  |  Page : 62-64  

Aspergillosis of the bone


1 Department of Orthopaedics and Trauma Surgery, University of Maiduguri, Maiduguri, Borno State, Nigeria
2 Department of Histopathology, College of Medicine, University of Maiduguri, Maiduguri, Borno State, Nigeria

Date of Web Publication7-Jan-2015

Correspondence Address:
T M Dabkana
Department of Orthopaedics and Trauma Surgery, College of Medical Sciences, University of Maiduguri, Maiduguri, Borno State
Nigeria
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/1596-3519.148745

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   Abstract 

Aspergillosis of the long bones has not been reported. Those of the bones of the paranasal sinuses and ear canal have been reported but rare. A young woman reported to us with history of discharging sinuses around the right knee and recent fracture of the right femur. Despite all efforts, she ended up losing the whole limb from the hip. When a patient with Aspergillosis of the long bones presents late, amputation may be the best option. Early diagnosis will prevent this.

   Abstract in French 

Résumé
L'aspergillose des os longs n'a pas été signalé. Ceux des os des sinus paranasaux et conduit auditif ont été signalés, mais rares. Une jeune femme nous signalé avec histoire de décharger les sinus autour du genou droit et une récente fracture du fémur droit. Malgré tous les efforts, elle a fini par perdre la branche entière de la hanche. Quand un patient présentant l'aspergillose des os longs présente tardivement, l'amputation peut être la meilleure option. Un diagnostic précoce permettra d'éviter cela.
Mots-clés: Adjacentes des os longs, aspergillose, enfance, patient immunocompétent

Keywords: Adjacent long bones, aspergillosis, childhood, immunocompetent patient


How to cite this article:
Dabkana T M, Pindiga UH, Mayun AA, Nggada HA. Aspergillosis of the bone. Ann Afr Med 2015;14:62-4

How to cite this URL:
Dabkana T M, Pindiga UH, Mayun AA, Nggada HA. Aspergillosis of the bone. Ann Afr Med [serial online] 2015 [cited 2020 Oct 20];14:62-4. Available from: https://www.annalsafrmed.org/text.asp?2015/14/1/62/148745


   Introduction Top


Although usually harmless inhabitants of the body (upper respiratory tract, skin, etc.), Aspergillus species may rarely be associated with human infection, being the second most common opportunistic fungal infection. Aspergillus fumigatus is by far the most common pathogen, but other species (Aspergillus niger, Aspergillus flavus and Aspergillus nidulans) have also been implicated. All these cause opportunistic infections and usually in existing body cavities. [1] These infections can however invade other tissues such as the liver, kidneys, spleen, etc., in the immunosuppressed (drugs for transplant patients, AIDS) patient. Infection of long bones has not been reported.


   Case Report Top


ZZR, a 26-year-old house wife reported to UMTH Maiduguri in 2009 with 17 years history of swelling around the right knee with no prior history of trauma. The swelling has been discharging a dark colored substance for the past 5 years. She had three operations to the knee in private hospitals prior to presentation, with no improvement. In 2008, she fell down and fractured the right femur and treated it at a traditional bone setter's place with no improvement.

On examination, right knee was found to be grossly swollen, tender with reduced range of move. There were multiple scars around the knee. An X-ray revealed a malunited supracondylar fracture of the right femur. There were cystic lesions in the distal third of the femur and proximal third of the tibia [Figure 1]. She was booked for open reduction and angle plating. While awaiting admission, she developed multiple discharging sinuses around the knee. She was then treated as a case of chronic osteomyelitis and was lost to follow-up for 1 year. In October 2010, she reported to Borno Orthopedic Centre (BOC) Maiduguri, with 2 weeks history of the sudden increase in the swelling with multiple discharging sinuses. She had also developed a fixed flexion deformity of the right knee. X-ray revealed features of COM of the distal half of the femur and proximal third of the tibia.
Figure 1: X-ray before above knee amputation

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She was admitted for sequestrectomy. Microscopy of the discharge yielded Gram-negative bacilli, but culture yielded no bacterial growth, Retroviral Studywas negative. Erythrocyte sedimentation rate (ESR) =121 mm/1 h, packed cell volume (PCV) = 30% and white blood cells (WBC) of 4.8 × 109/L. Urinalysis was normal. On the operating table, the marrow cavity of the distal half of the right femur and proximal third of the tibia were found to be filled with a dark substance (like iron fillings) [Figure 2], and the cortices of the bones infiltrated and paper thin. The patella was also involved. Consent was then sought from the husband and father for above knee amputation. This was done and postoperative X-rays revealed no residual pathology in the femoral stump.

The specimen was sent to the Pathology Department of the University of Maiduguri Teaching Hospital and the result came back to be aspergillosis [Figure 3]. She was placed on tioconazole and discharged from the hospital after 3 weeks, doing very well. Three months later (January 2011) patient came back to BOC with pain in the stump. The stump was found to be swollen and tender. X-ray showed erosive lesions in the femoral stump. Tioconazole was re-instituted and Flagyl added. The swelling and the pain subsided and the patient was referred to the mycologist at University of Maiduguri Teaching Hospital.

Eight months later, patient was referred to the Orthopedic Department of the hospital by the mycologist with history sudden increase in the swelling of the stump and discharging sinuses. Examination revealed ill looking female, pale but afebrile. The stump was swollen with two discharging sinuses. X-ray showed almost total destruction of the femoral stump.
Figure 2: Dark substance in bone marrow first surgery

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Figure 3: Histology picture after above knee amputation

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Investigation revealed an ESR of 121 mm/1 h, PCV = 30%, WBC of 4.8 × 109/L and platelet count of 190 × 109/L, HIV test was still negative. Patient was then booked for disarticulation at the hip. On the table, the femoral stump was infiltrated by the same dark looking substance up to the neck [Figure 4]. This was removed and sent for histology as before. The result was the same, aspergillosis. She was placed on systemic tioconazole and discharged 3 weeks postoperative. She has so far done well and no other part of the body is showing signs of the disease.
Figure 4: The same dark substance seen during disarticulation

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   Discussion Top


Of the 350 strains of aspergillosis spp., only four strains: A. fumigatus, A. flavus, A. niger, and A. nidulans are known to cause human disease. Most of the bones affected are those around the respiratory tract and ear sinuses. [2],[3],[4],[5] The organism being airborne, usually in the immunocompromised; AIDS or transplant patients on immunosuppressive drugs, and patients with prolonged neutropenia secondary to cytotoxic chemotherapies and maxillary bone implants are also at the highest risk of invasive aspergillosis. [1],[5] A. fumigatus is by far the most common cause of human disease. Aspergillosis of the skin has however been reported in the immunocompetent patient. [6] In general, aspergillosis of the bone is very rare even in the immunocompromised, accounting for about 1.8% in the most literature. [7]

The azoles have remained the mainstay of treatment of invasive aspergillosis. Amphotericin-B is also used, but has side-effects. Voriconazole, [7],[8] a new azole, is still an investigational drug. We hope to institute systemic amphotericin B at the earliest sign of dissemination of disease in this patient. She has so far done well.


   Conclusion Top


Aspergillosis of the bone in the immunocompetent is very rare, [7] usually affecting the bones of the paranasal sinuses and ear canal. It is even rarer when long bones are affected in a healthy patient. Treatment is by surgery and systemic azoles and or amphotericin-B. Close follow-up of the patients is important. Early diagnosis will avoid limb ablation.

 
   References Top

1.
Grossman M. Aspergillosis of bone. Br J Radiol 1975;48:57-9.  Back to cited text no. 1
[PUBMED]    
2.
Ederies A, Chen J, Aviv RI, Pirouzmand F, Bilbao JM, Thompson AL, et al. Aspergillosis of the Petrous Apex and Meckel′s Cave. Skull Base 2010;20:189-92.  Back to cited text no. 2
    
3.
Knipping S, Holzhausen HJ, Koesling S, Bloching M. Invasive aspergillosis of the paranasal sinuses and the skull base. Eur Arch Otorhinolaryngol 2007;264:1163-9.  Back to cited text no. 3
    
4.
Freudenstein D, Koerbel A, Beschorner R, Tatagiba M. Successful treatment of Aspergillus granuloma involving the cavernous sinus and the middle fossa by using surgery and voriconazole. Case illustration. J Neurosurg 2007;106:511.  Back to cited text no. 4
    
5.
Sato FR, Sawazaki R, Berretta D, Moreira RW, Vargas PA, de Almeida OP. aspergillosis of the maxillary sinus associated with a zygomatic implant. J Am Dent Assoc 2010;141:1231-5.  Back to cited text no. 5
    
6.
Tahir C, Garbati M, Nggada HA, Yawe EH, Abubakar AM. Primary cutaneous aspergillosis in an immunocompetent patient. J Surg Tech Case Rep 2011;3:94-6.  Back to cited text no. 6
    
7.
Mouas H, Lutsar I, Dupont B, Fain O, Herbrecht R, Lescure FX, et al. Voriconazole for invasive bone aspergillosis: A worldwide experience of 20 cases. Clin Infect Dis 2005;40:1141-7.  Back to cited text no. 7
    
8.
Clinical Trials.gov (NCT00001646.) Voriconazole Vs Amphotericin-B in treatment of invasive aspergillosis. Available from: http://www.clinicaltrials.gov/ct2/sow/NCT00001648.  [Last accessed on 2008 Mar 03].  Back to cited text no. 8
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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