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Cutaneous Complication of Chronic Fatigue: An Answer From the Horse’s Mouth

Cutaneous Complication of Chronic Fatigue: An Answer From the Horse’s Mouth Chronic fatigue syndrome (CFS) is defined as new-onset fatigue lasting at least 6 months not substantially alleviated by rest and detrimental to professional and social functioning.1 Diagnosis also requires at least 4 of the following: impairment of short-term memory or concentration, sore throat, muscle pain, multijoint pain without swelling or redness, headaches distinct from any experienced before, and disproportionate malaise after exertion.1 Nonetheless, self-diagnosis is common, and many patients seek alternative treatments.2 One consequence of the use of alternative treatment is morbidity owing to delayed recognition of reversible causes of fatigue.2 Herein, however, we report a case of self-diagnosed CFS in which morbidity stemmed directly from the alternative treatment pursued. Report of a Case A 46-year-old man presented to his dermatologist with chronic, focally scarred lesions on his scalp. The differential diagnosis included erosive dermatitis, infection, excoriation, chemical irritation, and dissecting cellulitis vs other scarring alopecias. A punch biopsy of the right vertex was performed. Histopathologic examination revealed a nodular to diffuse mixed inflammatory infiltrate with neutrophils and histiocytes surrounding the deep portion of follicles and infiltrating the subcutis (Figure, A). Purulent, hemorrhagic scale crust covered the lesion. Figure. View LargeDownload Silver and modified acid-fast stains reveal mycobacterial remnants among inflammatory cells. A, Deep, mixed inflammatory infiltrate is revealed by hematoxylin-eosin staining (original magnification ×10). Plump rods in the central clearing are revealed by Warthin-Starry (B), partial acid-fast (C), and Fite (D) stains (original magnification ×40 for panels B-D). Periodic acid–Schiff and gram stains failed to reveal fungus and bacteria, respectively. However, within the inflammation were clusters of tiny rods scarcely visible on hematoxylin-eosin–stained sections. They were stained weakly by Warthin-Starry (Figure, B) and Gomori methenamine silver stains and strongly by partial (or Coates) and Fite acid-fast stains (Figure, C and D). They were not stained by Ziehl-Neelsen acid-fast stain. A diagnosis of suppurative granulomatous dermatitis and panniculitis with partially acid-fast microorganisms was made. An infectious disease consult was also recommended regarding antimicrobial coverage for atypical mycobacteria and filamentous fungi pending identification by culture. Our subsequent recommendation of a second biopsy procedure prompted the patient to reveal that his symptoms arose after a trip to Mexico for treatment of self-diagnosed CFS. This treatment involved scalp injections of Equimune (Bioniche Life Sciences Inc, Belleville, Ontario, Canada), a commercial “immunostimulant” for horses.3 The principal ingredient of Equimune is a proprietary “mycobacterium cell wall fraction.”3 Thus, the rods were identified as the presumably noninfectious mycobacterial remnants of either an intrinsically partially acid-fast organism or, more likely, a typically acid-fast organism rendered less so by processing and destabilization of the mycolic acid fraction in the Equimune. Comment Equimune is advertised as a treatment for equine respiratory disease complex, a cadre of bacterial and viral respiratory infections in horses.3 Though outrageous on its face, the administration of Equimune to a human patient was likely motivated by a rich but controversial literature documenting altered cellular immunity in patients with CFS.4 In fact, our observations confirmed Equimune's potential as an immunologic adjuvant, though the effect was local and somewhat tragic. Interestingly, Equimune's manufacturer has initiated clinical trials in humans of mycobacterial cell wall DNA complexes for bladder cancer refractory to therapy with BCG.5 This development raises the possibility that the exotic, cross-species therapy reported herein may rematerialize as a local, off-label application. Our case demonstrates that injudicious use of an adjuvant might result in not only a severe adverse reaction but also an erroneous histopathologic diagnosis, proof positive that nonvalidated therapies might have consequences worse than simple disappointment. Correspondence: Dr Finn, Department of Pathology and Laboratory Medicine, University of North Carolina Hospitals, CB No. 7525, Brinkhous-Bullitt Building, Chapel Hill, NC 27599 (afinn@unch.unc.edu). Financial Disclosure: None reported. References 1. Centers for Disease Control and Prevention, Chronic fatigue syndrome: the revised case definition (abridged version). http://www.cdc.gov/cfs/cfsdefinitionHCP.htm. Accessed November 18, 2007 2. Hurel SJAbuiasha BBaylis PH et al. Patients with a self diagnosis of myalgic encephalomyelitis. BMJ 1995;311 (7000) 329PubMedGoogle ScholarCrossref 3. Equimune I.V. http://www.drugs.com/vet/equimune-i-v.html. Accessed November 18, 2007 4. Lyall MPeakman MWessely S A systematic review and critical evaluation of the immunology of chronic fatigue syndrome. J Psychosom Res 2003;55 (2) 79- 90PubMedGoogle ScholarCrossref 5. Bioniche Life Sciences Inc, Mycobacterial cell wall-DNA complex in treatment of BCG-refractory patients with non-muscle invasive bladder cancer. http://clinicaltrials.gov/ct/show/NCT00406068?order=1. Accessed November 18, 2007 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Archives of Dermatology American Medical Association

Cutaneous Complication of Chronic Fatigue: An Answer From the Horse’s Mouth

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Publisher
American Medical Association
Copyright
Copyright © 2008 American Medical Association. All Rights Reserved.
ISSN
0003-987X
eISSN
1538-3652
DOI
10.1001/archderm.144.9.1238
Publisher site
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Abstract

Chronic fatigue syndrome (CFS) is defined as new-onset fatigue lasting at least 6 months not substantially alleviated by rest and detrimental to professional and social functioning.1 Diagnosis also requires at least 4 of the following: impairment of short-term memory or concentration, sore throat, muscle pain, multijoint pain without swelling or redness, headaches distinct from any experienced before, and disproportionate malaise after exertion.1 Nonetheless, self-diagnosis is common, and many patients seek alternative treatments.2 One consequence of the use of alternative treatment is morbidity owing to delayed recognition of reversible causes of fatigue.2 Herein, however, we report a case of self-diagnosed CFS in which morbidity stemmed directly from the alternative treatment pursued. Report of a Case A 46-year-old man presented to his dermatologist with chronic, focally scarred lesions on his scalp. The differential diagnosis included erosive dermatitis, infection, excoriation, chemical irritation, and dissecting cellulitis vs other scarring alopecias. A punch biopsy of the right vertex was performed. Histopathologic examination revealed a nodular to diffuse mixed inflammatory infiltrate with neutrophils and histiocytes surrounding the deep portion of follicles and infiltrating the subcutis (Figure, A). Purulent, hemorrhagic scale crust covered the lesion. Figure. View LargeDownload Silver and modified acid-fast stains reveal mycobacterial remnants among inflammatory cells. A, Deep, mixed inflammatory infiltrate is revealed by hematoxylin-eosin staining (original magnification ×10). Plump rods in the central clearing are revealed by Warthin-Starry (B), partial acid-fast (C), and Fite (D) stains (original magnification ×40 for panels B-D). Periodic acid–Schiff and gram stains failed to reveal fungus and bacteria, respectively. However, within the inflammation were clusters of tiny rods scarcely visible on hematoxylin-eosin–stained sections. They were stained weakly by Warthin-Starry (Figure, B) and Gomori methenamine silver stains and strongly by partial (or Coates) and Fite acid-fast stains (Figure, C and D). They were not stained by Ziehl-Neelsen acid-fast stain. A diagnosis of suppurative granulomatous dermatitis and panniculitis with partially acid-fast microorganisms was made. An infectious disease consult was also recommended regarding antimicrobial coverage for atypical mycobacteria and filamentous fungi pending identification by culture. Our subsequent recommendation of a second biopsy procedure prompted the patient to reveal that his symptoms arose after a trip to Mexico for treatment of self-diagnosed CFS. This treatment involved scalp injections of Equimune (Bioniche Life Sciences Inc, Belleville, Ontario, Canada), a commercial “immunostimulant” for horses.3 The principal ingredient of Equimune is a proprietary “mycobacterium cell wall fraction.”3 Thus, the rods were identified as the presumably noninfectious mycobacterial remnants of either an intrinsically partially acid-fast organism or, more likely, a typically acid-fast organism rendered less so by processing and destabilization of the mycolic acid fraction in the Equimune. Comment Equimune is advertised as a treatment for equine respiratory disease complex, a cadre of bacterial and viral respiratory infections in horses.3 Though outrageous on its face, the administration of Equimune to a human patient was likely motivated by a rich but controversial literature documenting altered cellular immunity in patients with CFS.4 In fact, our observations confirmed Equimune's potential as an immunologic adjuvant, though the effect was local and somewhat tragic. Interestingly, Equimune's manufacturer has initiated clinical trials in humans of mycobacterial cell wall DNA complexes for bladder cancer refractory to therapy with BCG.5 This development raises the possibility that the exotic, cross-species therapy reported herein may rematerialize as a local, off-label application. Our case demonstrates that injudicious use of an adjuvant might result in not only a severe adverse reaction but also an erroneous histopathologic diagnosis, proof positive that nonvalidated therapies might have consequences worse than simple disappointment. Correspondence: Dr Finn, Department of Pathology and Laboratory Medicine, University of North Carolina Hospitals, CB No. 7525, Brinkhous-Bullitt Building, Chapel Hill, NC 27599 (afinn@unch.unc.edu). Financial Disclosure: None reported. References 1. Centers for Disease Control and Prevention, Chronic fatigue syndrome: the revised case definition (abridged version). http://www.cdc.gov/cfs/cfsdefinitionHCP.htm. Accessed November 18, 2007 2. Hurel SJAbuiasha BBaylis PH et al. Patients with a self diagnosis of myalgic encephalomyelitis. BMJ 1995;311 (7000) 329PubMedGoogle ScholarCrossref 3. Equimune I.V. http://www.drugs.com/vet/equimune-i-v.html. Accessed November 18, 2007 4. Lyall MPeakman MWessely S A systematic review and critical evaluation of the immunology of chronic fatigue syndrome. J Psychosom Res 2003;55 (2) 79- 90PubMedGoogle ScholarCrossref 5. Bioniche Life Sciences Inc, Mycobacterial cell wall-DNA complex in treatment of BCG-refractory patients with non-muscle invasive bladder cancer. http://clinicaltrials.gov/ct/show/NCT00406068?order=1. Accessed November 18, 2007

Journal

Archives of DermatologyAmerican Medical Association

Published: Sep 15, 2008

Keywords: chronic fatigue syndrome,mouth

References