* ,ChtourouKhalil ,GuermaziFadhel ,AyadiAli ,HadrichInes ,MahfoudhAbdelmajid ,HachichaMongia CheikhrouhouFatma ,MakniFattouma ,SellamiHayet ,SellamiAmira Disseminatedaspergillosiswiththyroidandbonelocalization:Acasereport

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CASE REPORT

Disseminated aspergillosis with thyroid and bone localization: A case report

Cheikhrouhou Fatma

a

, Makni Fattouma

a

, Sellami Hayet

a

, Sellami Amira

a

, Hadrich Ines

a

, Mahfoudh Abdelmajid

b

, Hachicha Mongia

b

, Chtourou Khalil

c

, Guermazi Fadhel

c

, Ayadi Ali

a,

*

aLaboratory of Parasitology and Mycology, Habib Bourguiba UH, Sfax, Tunisia

bPediadric Ward of Sfax Hedi Chaker UH, Sfax, Tunisia

cNucleic Medicine Ward of Habib Bourguiba UH, Sfax, Tunisia

Received 12 September 2008; received in revised form 9 January 2009; accepted 12 January 2009 Available online 9 March 2009

1. Introduction

Disseminated aspergillosis has a poor prognosis.

Extra-pulmonary involvement is rare and occurs at an advanced stage and represents an ominous sign for immunocompromised patients. We describe an

original case of disseminated aspergillosis (pulmon- ary, thyroid and vertebral).

2. Case reports

A 13-year-old girl, from Sfax (south of Tunisia), was hospitalized for cervical tumefaction, pain in the left knee with deterioration of the general state.

She had recurrent pulmonary infections and bron- chial dilatation at the age of 8 and 10 years. After 1

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KEYWORDS Aspergillus flavus;

Thyroid;

Bone;

Disseminated aspergillosis

Summary We describe an original case of disseminated aspergillosis (pulmonary, thyroid and vertebral) in a 13-year-old girl from Tunisia. The patient had presented a thyroid node and a knee mass. The scintigraphy confirmed the cold thyroid node and an increased bone uptake in the left knee, the 5th left coast, the femur and the hip.

The histological and mycological examinations concluded to an aspergillosis with Aspergillus flavus. The outcome was rapidly fatal.

Few reports have been published on thyroid aspergillosis localization. This major mycosis occurs in children with septic family granulomatosis whose forecast remains dark.

#2009 Elsevier Ireland Ltd. All rights reserved.

* Corresponding author at: Fungal and Parasitic Molecular Biol- ogy Laboratory, School of Medicine, Sfax, Tunisia.

Tel.: +216 74247130; fax: +216 74247130.

E-mail address:ali.ayadi@rns.tn(A. Ali).

1871-4048/$ — see front matter # 2009 Elsevier Ireland Ltd. All rights reserved.

doi:10.1016/j.pedex.2009.01.005

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year, the patient had presented a left thyroid node without hyperthyroidism.

The clinical examination showed a thyroid node and a left knee mass. The X-ray visualized a multiple bone lysis (Fig. 1) in the femur and the knee. The scintigraphy showed abnormal uptake in the left lobe of thyroid (Fig. 2), a decrease in perfusion in the right lower lobe of the lung (Fig. 3) and an increased bone uptake in the left knee, the 4th and 5th left coast, the femur and the hip (Fig. 4).

The neoplasic origin was evoked, and the patient had a thyroidectomy. Extensive necrosis

with infiltration of fungal hyphae was observed in the thyroid gland (Fig. 5). The histological and mycological examinations concluded to aspergil- losis with Aspergillus flavus. The Aspergillus anti- genemia was positive.

She was treated with Amphotericin B (30 mg/day) combined with Itraconazole (300 mg/day). She also developed a femur and tibia osteomyelitis (Figs. 1 and 6). Aggressive surgical debridement was done.

Culture from intra-operative bone tissue grewed A.

flavus. The magnetic resonance imaging showed extension of infiltrative process into the mediasti- num, the lung and pleural cavity with vertebral body destruction (Fig. 7). The evolution was marked by cervical abscess and multiorgan deterioration. The outcome was rapidly fatal.

The immunizing assessment of deficit could not be carried out for neither the patient nor her Fig. 1 X-ray: a femur and tibia bone lysis.

Fig. 2 Thyroid scintigraphy: abnormal uptake in the left lobe of thyroid.

Fig. 3 A decrease in perfusion in the right lower lobe of the lung.

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brother who died few years ago in septic shock with severe immunodeficiency.

3. Discussion

The incidence of Aspergillus infection has increased dramatically during the last decade [1]. However few reports have been published on extra-pulmonary aspergillosis and its clinical features have not been fully clarified [2]. The organ distribution was described by autopsy studies [1]. Osteomyelitis is the fourth most common site of infection for aspergillosis follow- Fig. 4 Bone scintigraphy: an increased bone uptake in the left knee, the 4th and 5th left coast, the femur and the hip.

Fig. 5 Histologic examination: extensive necrosis with infiltration of fungal hyphae.

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ing pulmonary, sinus and cerebral infections [3,4].

We have isolate A. flavus from thyroid and bone tissues. Frequency of thyroid localization varied

from 9% to 15% [2]. Aspergillus was the most common cause of fungal thyroiditis[5]. A majority of patients with thyroid aspergillosis remained asymptomatic as in our case. The thyroid node was suspected to be neoplasic. The diagnosis of thyroid fungi invasion is delayed in most cases and it is usually difficult to diagnose in the ante mor- tem period [1]. Aspergillus antigenemia is a key for early diagnosis and gallium scintigraphy might be useful to determine the site of fungal infection [6].

In the literature, invasive aspergillosis is usually caused by A. fumigatus and less commonly by A.

flavus[5,7].

Thyroiditis was diagnosed at autopsy as part of disseminated infection in a substantial num- ber of patients without clinical manifestations and laboratory evidence of thyroid dysfunction [5].

In our case, the immunizing assessment has not been carried. This type of major mycosis usually occurs in the children with septic family granulomatosis whose forecast remains always dark [8]. The extra pulmonary aspergillosis contributes to a high morbidity and a high mortality. Early recognition of these entities, prompt initiation of new, highly active antifungal therapies and adjunctive surgical manage ment may improve the prognosis of these conditions [1].

Fig. 6 MRI: femur and tibia osteomyelitis.

Fig. 7 MRI: infiltrative process and vertebral body destruction.

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References

[1] A. Hori, M. Kami, Y. Kishi, U. Machida, T. Mastumura, T.

Kashima, Clinical significance of extra–—pulmonary involve- ment of invasive aspergillosis: a retrospective autopsy-based study of 107 patients, J. Hosp. Infect. 50 (2002) 175—182.

[2] M. Hornef, J. Schopohl, C. Zietz, K. Hallfeldt, et al., Thyr- otoxicosis induced by thyroid involvement of disseminated Aspergillus fumigatus infection, J. Clin. Microbiol. 38 (2000) 886—887.

[3] L. Abu Jawdeh, R. Haider, F. Bitar, S. Mroueh, S. Akel, N.

Nuaryri-Salti, G.S. Dbaibo, Aspergillus vertebral osteomyeli- tis in a child with a primary monocyte killing defect: response to GM-CSF therapy, Br. Infect. Soc. (2000) 97—100.

[4] V. Sandeep, S. Manish, Spinal Aspergillus vertebral osteomye- litis with extradural abscess: case report and review of literature, Surg. Neurol. 61 (2004) 551—555.

[5] L.Z. Goldani, A.P. Zavascki, A.L. Maia, Fungal thyroiditis: an overview, Mycopathologia 161 (3) (2006) 129—139.

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Makuuchi, Aspergillus thyroiditis in a living donor liver trans- plant recipient, J. Infect. 53 (2006) 231—233.

[7] T. Mori, M. Matsumura, K. Yamada, et al., Systemic aspergil- losis caused by an aflatoxin-producing strain of Aspergillus flavus, Med. Mycol. 36 (1998) 107—112.

[8] J. Dotis, E. Roilides, Osteomyelitis due to Aspergillus spp. in patients with chronic granulomatous disease: comparison of Aspergillus nidulans and Aspergillus fumigatus, Int. J. Infect.

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