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Journal of Orthopaedics, Trauma and Rehabilitation 24 (2018) 77e79

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Journal of Orthopaedics, Trauma and Rehabilitation

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Case Report

Mucormycosis Vertebral Osteomyelitis毛霉菌脊椎骨髓炎

Chan Chun Fung*, Wong Martin Chung Ting, Cheung Ka Kin, Cheng Hung On,Yim Wing Ngai, Wen EleanorDepartment of Orthopedic and Traumatology, Tuen Mun Hospital, Tuen Mun, Hong Kong

a r t i c l e i n f o

Article history:Received 21 December 2016Received in revised form29 December 2016Accepted 31 December 2016

Keywords:fungal infectionsmucormycosisosteomyelitisspinal infections

* Corresponding author. E-mail: johnchancf@yahoo

http://dx.doi.org/10.1016/j.jotr.2016.12.0022210-4917/Copyright© 2018,HongKongOrthopaedic AssociatCC BY-NC-ND license (http://creativecommons.org/licenses/by

a b s t r a c t

Fungal vertebral osteomyelitis by mucormycosis is extremely rare. We report a 16-year-old patient withnewly diagnosed acute lymphoid leukaemia who developed extensive vertebral osteomyelitis caused bymucormycosis. He was treated with three-level subtotal spondylectomy and long-term oral antifungalmedications. He had no neurological deficit after the operation.

中 文 摘 要

由毛霉菌引致的真菌性脊椎骨髓炎是非常罕見的。我們執告一例16歲剛診斷急性淋巴白血病患者由毛霉菌引

起的廣泛脊髓骨髓炎。治療包括三節次全脊椎切除術及長期口服抗真菌藥物。手術後，沒有神經功能缺損。

Introduction

Mucormycosis infection is notorious for its highmortality rate inmany reported series, especially when developed as a disseminatedinfection. The 90-day mortality ranged from 20% to 58%.1 Itcommonly occurs in immunocompromised patients, especially inpatients with haematological malignancy, organ transplant, ordiabetes mellitus. Cerebral, maxillary sinus, and pulmonary in-fections are relatively common sites of mucormycosis infection.Pulmonary and disseminated infections are associated with veryhighmortality rates. Their diagnosis is difficult and usually delayed.Spinal infection bymucormycosis is extremely rare; there are only afew cases reported in the literature.

We report a case of mucormycosis with spinal infection, rapidlyprogressing from pulmonary to paraspinal and finally vertebral oste-omyelitis. The patient was successfully treated with three-level sub-total spondylectomywith instrumentationandantifungalmedication.

Case Report

A 16-year-old Chinese boy, suffering from newly diagnosed acutelymphoblastic leukaemia,was started on chemotherapy for 1month.He developed left-sided chest painwith high fever andwas admittedto our hospital. His X-ray scan was unremarkable. Meanwhile, thecontrast computed tomography scan showed severe left-side pleural

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effusion (Figure 1) and left-side paraspinal soft tissue shadow fromT8 to L1 level (Figure 1). Several tiny air pocketswere also seen inT10to T12 bodies. The spinal canal was spared (Figure 2).

Pleural fluid culture could not identify any causative microor-ganism, neither bacterial, Mycobacterium, nor fungal.

Antibiotic and antifungal medication was initiated as empiricaltreatment of his infection, yet our patient did not respond to thetreatment and his fever persisted. An operation was performed fordebridement of left side paraspinal soft tissue through a posteriorapproach to T11eT12. Only the left side of lamina and paraspinalsoft tissue was exposed. We found that the left-side paraspinal softtissue was a necrotic lesion, whereas the muscle remained healthy(Figure 3). There was no pus or any collection in the infected area.The left side costotransverse joint was also subluxed.

The histology report confirmed a Rhizopus species infection,whereas bacterial culture and rapid polymerase chain reaction re-sults for tuberculosis were negative.

Our patient was treated with amphoteric B after the initialdebridement after a discussion with the paediatrician and themicrobiologist. Two weeks after the first operation, the patientdeveloped sepsis and neurological deficits, left quadriceps powergrading 2/5, right quadriceps 4/5, bilateral ankles dorsiflexion 4/5.Repeated contrast imaging showed that there was extensivelyincreased infection of T10 to T12 vertebral bodies.

An urgent operation was performed. First, an open thoracotomywith partial lobectomy to excise the infected pulmonary tissue wasdone by a cardiothoracic surgeon. Dissection of the anterior part of

aedic Surgeons. Published by Elsevier (Singapore) Pte Ltd. This is an openaccess articleunder the

Figure 1. Severe left sided pleural effusion.

Figure 2. Involvement of vertebral body while spinal canal was spared.

Figure 3. Healthy paraspinal muscle.

Figure 4. Extensive necrosis of vertebral body.

C.F. Chan et al. / Journal of Orthopaedics, Trauma and Rehabilitation 24 (2018) 77e7978

infected T10 to T12 bodies was performed through the same tho-racotomy. Then the patient was placed in a prone position, andpedicle screws were inserted via the posterior approach.

Subsequently, a three-level subtotal spondylectomy in a piece-meal manner through left-side costotraversectomy was performed.There was an extensive necrosis of the vertebral bodies; the cancel-lous bone was dry and was greyish-green in appearance (Figure 4),which was typical for Rhizopus species infection. Debridement andcurettage of the infected vertebrae was performed until bonebleeding was noted. A titanium cage impacted with healthy autolo-gous bone graft was placed on the left side from T10 to T12.

After the operation, the patient was given a course of antifungalmedications. Serial imaging and blood test showed the infectionwas resolved.

At 2 years after the operation, he has residual back pain withoutlower limb symptoms (Figure 5).

Discussion

Mucormycosis is caused by fungi of the order Mucorales and thefamily Mucoraceae. Rhizopus, Mucor, and Lichtheimia are the mostcommon genera that cause 70e80% of all cases of mucormycosis.2

Their infection can lead to high morbidity and mortality rates inboth immunocompromised and immunocompetent patients.Infection occurs through the characteristic process of angioinvasionby the fungus, with subsequent vessel thrombosis and tissue ne-crosis. These hinder the leucocytes and antifungal medicationsfrom reaching the infected foci effectively. Hence, radical debride-ment of the infected tissue is usually required for mucormycosisinfection, a devastating condition owing to the need for radicaldebridement or excision.

The diagnosis of mucormycosis infection is usually delayedbecause of difficulties in identifying the pathogen. Cultural isola-tion yield ranged from 50% to 71% only,2 and there were no readilyavailable nonculture methods, including blood test to facilitate thediagnosis of mucormycosis. In our patient, the diagnosis ofmucormycosis infection was confirmed by histology.

Figure 5. Post-operative X-ray.

C.F. Chan et al. / Journal of Orthopaedics, Trauma and Rehabilitation 24 (2018) 77e79 79

To our knowledge, this is the first described case of direct in-vasion of the paraspinal space from disseminated mucormycosislung infection that progressed as vertebral osteomyelitis.

Case reports on mucormycosis infection in the limb are morereadily available in the literature; many of the patients end up withamputation for the treatment of the disease. Holtom et al reported acase of mucormycosis of the tibia after a Steinmann pin insertion, inwhich the patient ended up with an above-the-knee amputation.3

Wilkins et al reported a patient with mucormycosis infection inthe femur after anterior cruciate ligament construction, inwhich thepatient required multiple debridement and ended up with a meg-aprothesis.4 Moran et al reported a series of seven cases of immu-nocompetent patients with mucormycosis infection followingmotor vehicle accidents with gross contamination or farming in-juries. Multiple debridement was required, and amputation wasperformed in four out of seven patients despite the fact that all ofthem were immunocompetent to start with.5 Jones et al reported acase of successful salvage of the forearm after mucormycosisinfection in a 6-month-old child after liver transplantation.6

Mucormycosis causing spinal osteomyelitis is extremely rare.There have been only two such cases reported in English literature.The first reported case was a post-mortem diagnosis by Buruma in1979. He reported a patient with a history of laryngectomy withpartial hypopharyngectomy owing to an underlying malignancy,who developed neck pain and muscle weakness. The patient subse-quently passed away because of massive pulmonary embolism, andthe autopsy result showed vertebral osteomyelitis due to mucor-mycosis.7 The second case was reported by Chen et al. They reportedan immunocompetent patient who acquired an iatrogenic infectionafter lumbar disc puncture and radiofrequency nucleoplasty. Thepatient was treated with repeated radical debridement withoutinstrumentation while long-term antifungal therapy was given.8

Frazier et al reported a series of 11 patients with spinal fungalinfection not limited to mucormycosis. In their report, the averagedelay of diagnosis was 99 days. Nine out of 11 patients wereimmunocompromised. Eight patients developed paralysis second-ary to the infection. They recommended routinely performingfungal cultures whenever a spinal infection is suspected.9

In our patient, the success of treatment could be attributed tothe early identification of the pathogen, early prescription of sys-temic antifungal agent, and a radical three-level subtotal spondy-lectomy. Clinical and radiological clearance of the disease wasachieved in our patient.

Conclusion

Mucormycosis infection is a rare infection that is notoriouslydifficult to treat. It can be a rare cause of spinal osteomyelitisespecially in immunocompromised patients. We recommendroutine fungal culture in suspected spine infections followed byradial debridement and systemic antifungal therapy after thediagnosis is confirmed.

Conflict of interest statement

None of the authors has received or will receive benefits forpersonal or professional use from a commercial party relateddirectly or indirectly to the subject of this article.

References

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8. Chen F, Lü G, Kang Y, et al. Mucormycosis spondylodiscitis after lumbar discpuncture. Eur Spine J 2006 Mar;15(3):370e6. Epub 2005 Nov 18. PubMed PMID:16328227; PubMed Central PMCID: PMC3489305.

9. Frazier DD, Campbell DR, Garvey TA, et al. Fungal infections of the spine. Reportof eleven patients with long-term follow-up. J Bone Joint Surg Am 2001 Apr;83-A(4):560e5. PubMed PMID: 11315785.