Clinical and Histopathological Features and a Unique Spectrum of Organisms Significantly Associated with Chronic Granulomatous Disease Osteomyelitis during Childhood

Galluzzo, Maria; Hernández, C; Dávila, María Teresa García de; Pérez, Laura; Oleastro, M.; Zelazko, Marta; Rosenzweig, Sergio D.

doi:10.1086/527446

Cited by 30 publications

(28 citation statements)

References 21 publications

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“…Infections of long bones in children with CGD developed most frequently via hematogenous dissemination from a pulmonary focus. Our findings of incidence and pathogen distribution in CGD-affected patients with Aspergillus osteomyelitis are in accordance with other published reports [157, 154, 152, 160]. Our findings also are consistent with the case series by Dotis and Roilides who reported 46 cases of Aspergillus osteomyelitis complicating CGD, where the median age was 8 years and overall mortality was 37% [152].…”

Section: Discussionsupporting

confidence: 92%

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Aspergillus osteomyelitis: Epidemiology, clinical manifestations, management, and outcome

Gamaletsou

Rammaert

Bueno

et al. 2014

Journal of Infection

128

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Background The epidemiology, pathogenesis, diagnosis, and management of Aspergillus osteomyelitis are not well understood. Methods Protocol-defined cases of Aspergillus osteomyelitis published in the English literature were reviewed for comorbidities, microbiology, mechanisms of infection, clinical manifestations, radiological findings, inflammatory biomarkers, antifungal therapy, and outcome. Results Among 180 evaluable patients, 127 (71%) were males. Possible predisposing medical conditions in 103 (57%) included pharmacological immunosuppression, primary immunodeficiency, and neutropenia. Seventy-three others (41%) had prior open fracture, trauma or surgery. Eighty (44%) followed a hematogenous mechanism, 58 (32%) contiguous infections, and 42 (23%) direct inoculation. Aspergillus osteomyelitis was the first manifestation of aspergillosis in 77%. Pain and tenderness were present in 80%. The most frequently infected sites were vertebrae (46%), cranium (23%), ribs (16%), and long bones (13%). Patients with vertebral Aspergillus osteomyelitis had more previous orthopedic surgery (19% vs 0%; P=0.02), while those with cranial osteomyelitis had more diabetes mellitus (32% vs 8%; P=0.002) and prior head/neck surgery (12% vs 0%; P=0.02). Radiologic findings included osteolysis, soft-tissue extension, and uptake on T2-weighted images. Vertebral body Aspergillus osteomyelitis was complicated by spinal-cord compression in 47% and neurological deficits in 41%. Forty-four patients (24%) received only antifungal therapy, while 121(67%) were managed with surgery and antifungal therapy. Overall mortality was 25%. Median duration of therapy was 90 days (range, 10–772 days). There were fewer relapses in patients managed with surgery plus antifungal therapy in comparison to those managed with antifungal therapy alone (8% vs 30%; P=0.006). Conclusions Aspergillus osteomyelitis is a debilitating infection affecting both immunocompromised and immunocompetent patients. The most common sites are vertebrae, ribs, and cranium. Based upon this comprehensive review, management of Aspergillus osteomyelitis optimally includes antifungal therapy and selective surgery to avoid relapse and to achieve a complete response.

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Section: Discussionsupporting

confidence: 92%

“…Among 339 screened cases, there were 180 well-described cases of Aspergillus osteomyelitis in the English literature that fulfilled criteria for inclusion in the database over a study period from 1947 to 2013 [1–160]. …”

Section: Resultsmentioning

confidence: 99%

Aspergillus osteomyelitis: Epidemiology, clinical manifestations, management, and outcome

Gamaletsou

Rammaert

Bueno

et al. 2014

Journal of Infection

128

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“…Unfortunately, there is limited guidance in the current literature with respect to when specific cultures should be performed for children with musculoskeletal infection. While it is tempting to send material for a wide array of culture types to avoid missing unusual organisms, the routine use of low-yield cultures may be costly and unnecessary 8,11,12 . It is also possible that the results of certain cultures may be misleading due to the identification of contaminants.…”

Section: Discussionmentioning

confidence: 99%

Microbiological Culture Methods for Pediatric Musculoskeletal Infection

Section

Gibbons

Barton

et al. 2015

The Journal of Bone and Joint Surgery

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“…In a brief report published by Galluzzo et al [48], 14 CGD patients were compared with control subjects to determine osteomyelitis-related markers suggestive of CGD. Generally, multifocal osteomyelitis and/or simultaneous other organ involvement were the most relevant and statistically significant clinical findings suggesting CGD as an underlying disease.…”

Section: Osteomyelitismentioning

confidence: 99%

Invasive Fungal Infections in Patients with Chronic Granulomatous Disease

Henriet

Verweij

Holland

et al. 2012

Advances in Experimental Medicine and Biology

103

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Invasive fungal infections are a major threat for chronic granulomatous disease (CGD) patients. The present study provides a comprehensive overview of published invasive fungal infections in the CGD host through an extensive review of epidemiological, clinical, diagnostic and therapeutic data. In addition to the often mild clinical presentation, the currently used diagnostics for invasive aspergillosis have low sensitivity in CGD patients and cannot be easily translated to this non-neutropenic host. Aspergillus fumigatus and A. nidulans are the most commonly isolated species. A. nidulans infections are seldom reported in other immunocompromised patients, indicating a unique interaction between this fungus and the CGD host. The occurrence of mucormycosis is mainly noted in the setting of treatment of inflammatory complications with immunosuppressive drugs. Candida infections are infrequently seen and do not cause mucocutaneous disease but do show an age-dependent clinical presentation. The CGD patient is susceptible to a wide range of fungal pathogens, indicating the need to determine the causative fungus, often by invasive diagnostics, to guide optimal and rational treatment. This review summarizes current understanding of invasive fungal infections in patients with CGD and will serve as a starting point to guide optimal treatment strategies and to direct further research aimed at improving outcomes.

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Clinical and Histopathological Features and a Unique Spectrum of Organisms Significantly Associated with Chronic Granulomatous Disease Osteomyelitis during Childhood

Cited by 30 publications

References 21 publications

Aspergillus osteomyelitis: Epidemiology, clinical manifestations, management, and outcome

Aspergillus osteomyelitis: Epidemiology, clinical manifestations, management, and outcome

Microbiological Culture Methods for Pediatric Musculoskeletal Infection

Invasive Fungal Infections in Patients with Chronic Granulomatous Disease

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