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CASE REPORT
Year : 2009  |  Volume : 27  |  Issue : 2  |  Page : 161-163
 

Pulmonary botryomycosis in a patient with down syndrome


1 Department of Surgery, King Fahad Specialist Hospital - Dammam, PO Box 15215, Dammam, 314 44, Saudi Arabia
2 Department of Pediatrics, King Fahad Specialist Hospital - Dammam, PO Box 15215, Dammam, 314 44, Saudi Arabia
3 Department of Medicine, King Fahad Specialist Hospital - Dammam, PO Box 15215, Dammam, 314 44, Saudi Arabia
4 Department of Intensive Care, King Fahad Specialist Hospital - Dammam, PO Box 15215, Dammam, 314 44, Saudi Arabia

Date of Submission20-Aug-2008
Date of Acceptance05-Oct-2008

Correspondence Address:
F A Al-Rabee
Department of Surgery, King Fahad Specialist Hospital - Dammam, PO Box 15215, Dammam, 314 44
Saudi Arabia
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0255-0857.49434

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 ~ Abstract 

Pulmonary botryomycosis is a rare chronic, pyogranulomatous infection affecting the lung parenchyma. We describe here the clinical and histopathological findings of pulmonary botryomycosis reported for the first time in a Down syndrome female who required prolonged intensive care. This case has other different unique aspects. It is the first case to present with empyema, the second case involving the right lower lobe and the first case managed by decortication.


Keywords: Botryomycosis, decortication, Down syndrome, empyema


How to cite this article:
Al-Rabee F A, Hayajneh W A, Shorman M, Al-Hubail R. Pulmonary botryomycosis in a patient with down syndrome. Indian J Med Microbiol 2009;27:161-3

How to cite this URL:
Al-Rabee F A, Hayajneh W A, Shorman M, Al-Hubail R. Pulmonary botryomycosis in a patient with down syndrome. Indian J Med Microbiol [serial online] 2009 [cited 2019 Oct 22];27:161-3. Available from: http://www.ijmm.org/text.asp?2009/27/2/161/49434


Botryomycosis was first described in a horse lung in 1870. [1] It is a rare chronic, pyogranulomatous infection caused mainly by Staphylococcus aureus and occasionally by other bacteria such as Pseudomonas aeruginosa , Proteus sp.,  Escherichia More Details coli , Serratia marcescens ,  Neisseria More Details sp.,  Moraxella More Details sp., Propionibacterium sp. and others. [2] The term botryomycosis is derived from the Greek words botryose (bunch of grapes) and mycosis (fungus). [3] Histologically, the appearance consists of densely packed microorganisms surrounded by eosinophilic material and is similar to that of actinomycosis with characteristic granule formation. [4],[5] Two forms exist; a relatively common cutaneous form (75%) and a rare visceral one (25%). [3] The lung is rarely the primary location of this disease, with only 23 reported cases in the English literature. We describe here a case of pulmonary botryomycosis in a Down syndrome female. To our knowledge, this may be the first case of botryomycosis to be reported from Saudi Arabia or in a Down syndrome patient.


 ~ Case Report Top


A 23-year-old Saudi Down syndrome female presented to the emergency room on 29 January 2008 with a 1-month history of fever, productive cough and worsening dyspnea. She had received medical care at different centres without any improvement. There was no history of recent travel or exposure to sick contacts. Past medical history was unremarkable apart from being diagnosed with Down syndrome and cognitive developmental delay. Physical examination revealed a sick-looking, febrile (38.5C) female with tachypnea, tachycardia and hypotension. Oxygen saturation was 82% at room air. There was nearly absent breathing sounds over the right side of the chest. Chest radiograph revealed near total opacification of the right lung with hydropneumothorax, lung collapse and contralateral mediastinal shift [Figure 1]. A chest computed tomography (CT) scan [Figure 1] revealed right-sided massive loculated collection associated with air-fluid levels, complete collapse of the right lung and contralateral mediastinal shift. There was also an anterior mediastinal loculated collection compressing the great vessels. Intravenous fluids and piperacillin-tazobactam were administered immediately along with other supportive measures. Initial laboratory results showed a very high erythrocyte sedimentation rate (138mm/h), elevated blood leukocytes (17.6 10 9 /L) and low haemoglobin (9gm/dL). She had low serum albumin (19g/L) and elevated serum creatinine. The patient underwent immediate decortication (empyemectomy, multiple abscesses drainage, partial parietal pleurectomy and removal of the constricting peal) through the right posterolateral thoracotomy with lung isolation anaesthesia. The right lower lobe was found to be completely hepatized. Post-operatively, the patient required mechanical ventilation for 17 days and the antimicrobial regimens were modified to include vancomycin, meropenem and levofloxacin over variable periods due to persistent fever. The patient continued to improve clinically and a repeated chest CT scan [Figure 1] showed dramatic improvement. She was discharged after 40 days from surgery and she completed another month of oral levofloxacin. Bacterial, mycobacterial and fungal cultures of pleura, pleural fluid, blood and sputum failed to show any growth. Histopathologic examination of the lung tissue revealed dense nodular and mixed acute and chronic inflammatory infiltrates. Several eosinophilic granules were noticed with embedded cores of gram-positive cocci [Figure 2].


 ~ Discussion Top


Pulmonary botryomycosis is a rare entity that is usually associated with variable levels of immune dysfunction as in patients with diabetes, cystic fibrosis, human immunodeficiency virus infections and others. Other risk factors were also reported in literature as post-operative stress, foreign body, steroid intake, periodontal abscesses, alcoholism and malnutrition. [3],[6],[7] We present here a case of pulmonary botryomycosis in a Down syndrome patient. Apparently, this is the first case to be reported from Saudi Arabia. The diagnosis of botryomycosis in our patient was mainly established by histopathological examination of lung parenchyma, which revealed multiple eosinophilic granules surrounding cores of dense collections of gram-positive cocci [Figure 2]. This appearance is similar to the previously described classic Splendore-Hoeppli reaction, where abscesses usually contain dense colonies of bacteria. [8] The failure to grow bacteria from the patient's specimens was probably due to long courses of oral antimicrobials and pre-surgical intravenous piperacillin-tazobactam. This case presents several unusual patterns about pulmonary botryomycosis. It is the first case reported in a Down syndrome patient, which might be related to the already described immune dysfunction in these patients. [9] Unlike the other reported cases of pulmonary botryomycosis that usually presented with discrete lung masses, this patient presented with empyema and clinical sepsis. Other less common radiological variations include cavitatory lesions, consolidations or diffuse infiltrations. Review of the literature revealed that this is the second case to involve the right lower lobe. [2] Early surgical intervention was crucial for survival of our patient. Moreover, the patient was spared any lung resection, which is the usual practice in most of the reported cases. [10]

In summary, this case has different unique aspects. It is the first reported case in a Down syndrome patient, the first case to present with empyema, the second case involving the right lower lobe and the first case managed by decortication. It is justified to consider the possibility of botryomycosis, even though it is rare, in cases of undiagnosed lung masses and infections.

 
 ~ References Top

1.Bollinger O. Mycosis der Lunge beim Pferde. Virchows Arch Pathol Anat 1870;49:583-6.  Back to cited text no. 1    
2.Speir WA Jr, Mitchener JW, Galloway RF. Primary pulmonary botryomycosis. Chest 1971;60:92-3.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]
3.Katapadi K, Pujol F, Vuletin JC, Katapadi M, Pachter BR. Pulmonary botryomycosis in a patient with AIDS. Chest 1996;109:276-8.  Back to cited text no. 3  [PUBMED]  [FULLTEXT]
4.Simantov A, Chosidow O, Fraitag S, Feuilhade de Chauvin M, Prost C, Ravisse P, et al . Disseminated cutaneous botryomycosis: An unexpected diagnosis after 20 years duration. Clin Exp Dermatol 1994;19:259-61.  Back to cited text no. 4  [PUBMED]  
5.Waisman M. Staphylococcic actinophytosis (botryomycosis): Granular bacteriosis of the skin. Arch Dermatol 1962;86:525-9.  Back to cited text no. 5  [PUBMED]  
6.Saadat P, Ram R, Sohrabian S, Vadmal MS. Botryomycosis caused by Staphylococcus aureus and Pneumocystis carinii in a patient with acquired immunodeficiency disease. Clin Exp Dermatol 2008;33:266-9.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]
7.Katznelsen D, Vawter GF, Foley GE, Shwatchman H. Botryomycosis: A complication of cystic fibrosis, report of 7 cases. J Pediatr 1964;65:525-39.  Back to cited text no. 7    
8.Dail DH, Hammer SP, editors. In: Pulmonary pathology. New York: Springer Verlag; 1988. p. 160-2.  Back to cited text no. 8    
9.Hamurcu Z, Demirtas H, Patiroglu T, Kumandas S, Imamoglu N, Ozkul Y, et al . Age-dependent decreases in mitogen-stimulation level and RNA content in peripheral blood mononuclear cells of Down syndrome patients. Cytometry B Clin Cytom 2007;72:43-8.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]
10.Vasishta RK, Gupta N, Kakkar N. Botryomycosis-a series of six integumentary or visceral cases from India. Ann Trop Med Parasitol 2004;98:623-9.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]


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