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  Table of Contents    
CASE REPORT  
Year : 2023  |  Volume : 66  |  Issue : 1  |  Page : 196-198
Vertebromedular histoplasmosis: About a case of transverse myelitis in a young and immunocompetent adult


1 Department of Morphological Sciences and Pathological Anatomy, Faculty of Medicine and Biomedical Sciences, The University of Ngaoundere, Ngaoundéré, Adamawa; Garoua Military Hospital, Garoua, Cameroon
2 Department of Radiology and Medical Imaging, Faculty of Medicine and Biomedical Sciences, The University of Ngaoundere, Ngaoundéré, Adamawa, Cameroon
3 Department of Morphological Sciences and Pathological Anatomy, Faculty of Medicine and Biomedical Sciences, The University of Ngaoundere, Ngaoundéré, Adamawa; Garoua Regional Hospital, Garoua, Cameroon
4 Department of Morphological Sciences and Pathological Anatomy, Faculty of Medicine and Biomedical Sciences, The University of Yaounde I, Yaounde, Centre Region, Cameroon

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Date of Submission11-Aug-2021
Date of Acceptance17-Sep-2021
Date of Web Publication18-Jan-2023
 

   Abstract 


Systemic mycosis raging endemic, histoplasmosis has an increasing incidence with the advent of HIV-AIDS. The authors report a case of vertebra-medullar histoplasmosis mimicking tuberculous spondylodiscitis in a patient without proven immunosuppression. This was a patient who spent 3 years in a highly endemic area. He presented with signs of transverse myelitis. MRI of the lumbar spine showed a para-vertebral tumor lesion that partially infiltrated the spinal cord. The positive diagnosis of histoplasmosis was made by histomorphology, and symptoms were reduced with administration of itraconazole. These findings show the need for a proper coding of the management of patients living or having stayed in histoplasmosis endemic areas, whether they are immunocompromised or not.

Keywords: Histoplasmosis, immunocompetent, itraconazole, Pott's disease

How to cite this article:
Ngo Pambe CJ, Aminou M, Ngaroua, Mendimi Nkodo JM. Vertebromedular histoplasmosis: About a case of transverse myelitis in a young and immunocompetent adult. Indian J Pathol Microbiol 2023;66:196-8

How to cite this URL:
Ngo Pambe CJ, Aminou M, Ngaroua, Mendimi Nkodo JM. Vertebromedular histoplasmosis: About a case of transverse myelitis in a young and immunocompetent adult. Indian J Pathol Microbiol [serial online] 2023 [cited 2023 Feb 2];66:196-8. Available from: https://www.ijpmonline.org/text.asp?2023/66/1/196/367990





   Introduction Top


Caused by Histoplasma capsulatum var capsulatum (Hcc) and Histoplasma capsulatum var duboisii (Hcd), histoplasmosis is an endemic systemic mycosis in sub-Saharan Africa and some American countries, as well as in Asia and Oceania. Although with its probably underestimated prevalence, its incidence has increased somewhat with the advent of HIV-AIDS, making it an opportunistic infection. Thus, when faced with a diagnosis of histoplasmosis, the patient should be ruled out as being immunosuppressed to HIV.[1],[2] Although Hcc is ubiquitous, the Hcd is found exclusively in Africa. Despite the increasing incidence of histoplasmosis in nonendemic areas, the diagnosis of isolated, central nervous system forms remains difficult and the prognosis is often poor.[2],[3]


   Observation Top


This was a 48-year-old man who consulted for progressively increasing back pain that did not respond to the usual analgesics. The pain exacerbated when changing position. He had paresthesia and paraparesis of the lower limbs. His medical history was unremarkable. However, there was a notion of a stay of more than 3 years in rural Gambia. Magnetic resonance imaging (MRI) of the dorsal spine showed a heterogeneous, poorly limited, and osteolytic lesion at the vertebral epicenter with intracanal extensions. The lesion infiltrated the paravertebral muscle tissue and extended 3 cm under the skin. There was a spinal cord compression [Figure 1] and [Figure 2]. The diagnostic hypothesis was that of a malignant tumor with a differential diagnosis of Pott's disease. The pathological examination involved heterochromatic soft tissues and bone fragments. On microscopy, a granulomatous reaction with necrosis was observed. Numerous isolated elements or doublets surrounded by a clear halo were present. These elements were extracellular and intracytoplasmic in multiple multinucleated giant cells. Periodic Acid Schiff staining revealed a double-contoured wall. On Gomori-Grocott staining, these elements acquired a black hue. Ziehl-Nielsen staining was negative [Figure 3]. The microscopic features and the stay in a highly endemic area confirmed the diagnosis of Histoplasmosis and the specy Hcd. Daily administration of 200 mg of itraconazole for 12 months resulted in a significant reduction in symptoms at month six. On the follow-up MRI at 8 months, there was only a 2-cm-long scar opposite the initial lesion [Figure 4].
Figure 1: MRI scan without (a) and with (b) contrast medium showing a poorly limited and infiltrating lesion opposite T4, T5 and T6

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Figure 2: MRI scan showing compression (a) and discontinuity (b) of the spinal cord

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Figure 3: (a) HE, Gx20 stain; granulomatous reaction with necrosis and haemorrhage (star) with participation of multinucleated giant cells (triangle). (b) PAS stain, Gx40; highlighting the double-contoured wall of H. capsulatum var. duboisii surrounded by a clear halo. (c) Gomori Grocott stain, Gx40; black stain of yeast. Highlighting of the 8 (arrow) or lemon (triangle) morphology. (d) Ziehl-Nielsen stain, Gx40; Absence acid fast bacilli

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Figure 4: Control MRI at 8 months, under itraconazole. Note the recovery of the continuity of the spinal cord (a) and the significant regression of the size of the lesion (b and c).

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   Discussion Top


African histoplasmosis usually occurs in immunocompetent individuals who have spent long periods of time in highly endemic areas, as in the case of the patient described, although it can develop in immunocompromised individuals with CD4 counts below 200/mm3. Moreover, epidemiological data concerning this condition are not well understood, hence the underestimation of its frequency.[1],[4] According to some authors, 50–90% of immunocompetent individuals infected with Histoplasma sp remain asymptomatic.[5]

The patient presented here is male and immunocompetent. The current data suggest a male predominance of the condition.[6] He had a localized form of the disease, with double involvement of vertebra and spinal cord, suggesting tuberculosis or malignancy. African histoplasmosis is responsible for large localized or disseminated forms of the disease. The most frequent manifestations are cutaneous. Bone manifestations are less frequent and are part of osteoarticular disorders. They generally concern flat bones. Vertebral involvement often leads to the diagnosis of tuberculosis. Malignant tumor was evoked here because of the radiological aspect. Involvement of the central nervous system is also rare; the majority of cases reported in the literature are cerebral and cerebello-medullary, which may or may not be associated with cognitive disorders, intracranial hypertension, hydrocephalus, and meningitis.[7] Cerebello-medullary involvement is rare and associated with Hcc. Spinal cord involvement is exceptional and very few cases have been reported in the literature.[8]

Numerous advances have been made in diagnosis. These include serological techniques for the detection of antibodies and antigens by Enzyme Immunoassay or Enzyme Linked Immunoassay. These are frequently supplemented by histopathology and sometimes culture for a definitive diagnosis.[9] Molecular biology techniques such as Polymerase Chain Reaction (PCR) and Fluorescence In Situ Hybridisation (FISH) offer improved sensitivity, specificity, and diagnostic speed as noninvasive techniques. This is particularly true for cardiac and central nervous system locations. Unfortunately, they are expensive and not widely used in sub-Saharan Africa; whereas the majority of countries are developing, although located in endemic areas. Moreover, in most reported African histoplasmosis, the diagnosis was made by histology and culture; only in five countries (Tanzania, Benin, South Africa, Egypt, and Uganda) was serology reported as being used to make a diagnosis, and in three of the cases, the samples were processed in Western countries.[10] On this basis, in Africa, histopathology remains the gold standard for the diagnosis of histoplasmosis. Treatment is long and relies on the administration of antifungal molecules, notably amphotericin B and itraconazole. They are accompanied by a progressive disappearance of neurological symptoms in cases of isolated localizations. The patient presented here had a localized form of the condition. He showed a progressive regression of clinical signs at month 6 of daily administration of 200 mg of itraconazole. According to some authors, localized forms often have a good prognosis in contrast to disseminated forms.[10]


   Conclusions Top


In Africa, the diagnosis and management of histoplasmosis remain poorly codified. Although rare, localizations of the disease in the central nervous system can occur in both immunocompetent and immunocompromised subjects. The mastery of medical and personal history and systematic histological analysis of lesions in patients who live or have lived in endemic areas would be a major asset to avoid diagnostic and therapeutic erraticism.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Develoux M, Amona FM, Hennequin C. Histoplasmosis caused by Histoplasma capsulatum var. duboisii: A comprehensive review of cases from 1993 to 2019. Clin Infect Dis 2021;73:e543-9.  Back to cited text no. 1
    
2.
Gonzalez HH, Rane M, Cioci A, Goodman S, Espinosa PS. Disseminated central nervous system histoplasmosis: A case report. Cureus 2019;11:e4238. doi: 10.7759/cureus. 4238.  Back to cited text no. 2
    
3.
Hamada M, Tsuji S. [Central nervous system histoplasmosis]. Brain Nerve 2009;61:129-34.  Back to cited text no. 3
    
4.
Cipriano A, Neves-Maia J, Lopes V, Fleming CE, Ferreira MA, Bathay J. African histoplasmosis in a Guinea Bissau patient with HIV-2: Case report and review. J Mycol Med 2020;30:100904.  Back to cited text no. 4
    
5.
Diadie S, Diatta B, Ndiaye M, Gaye M, Sow D, Ndiaye MT, et al. Histoplasmose multifocale à Histoplasma capsulatum var. duboisii chez un Sénégalais de 22ans sans immunodépression prouvée. J Mycol Med 2016;26:265-70.  Back to cited text no. 5
    
6.
Darré T, Saka B, Mouhari-Touré A, Dorkenoo AM, Amégbor K, Pitche VP, et al. Histoplasmosis by Histoplasma capsulatum var. duboisii observed at the Laboratory of Pathological Anatomy of Lomé in Togo. J Pathog 2017;2017:e2323412. doi: 10.1155/2017/2323412.  Back to cited text no. 6
    
7.
Paccoud O, Nervo M, Alanio A, Garcia-Hermoso D, Bougnoux M-E, Mateo J, et al. Cerebral histoplasmosis caused by Histoplasma capsulatum var. duboisii in a patient with no known immunodeficiency. J Travel Med 2021;28. doi: 10.1093/jtm/taaa167.  Back to cited text no. 7
    
8.
Rivierez M, Heyman D, Brebion A, Landau-Ossondo M, Desbois N, Vally P. [Spinal cord histoplasmoma. A case report]. Neurochirurgie 2002;48:44-8.  Back to cited text no. 8
    
9.
Azar MM, Hage CA. Laboratory diagnostics for histoplasmosis. J Clin Microbiol 2017;55:1612-20.  Back to cited text no. 9
    
10.
Oladele RO, Ayanlowo OO, Richardson MD, Denning DW. Histoplasmosis in Africa: An emerging or a neglected disease? PLoS Negl Trop Dis 2018;12:e0006046. doi: 10.1371/journal.pntd. 0006046.  Back to cited text no. 10
    

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Correspondence Address:
Christiane J Ngo Pambe
Faculty of Medicine and Biomedical Sciences of Garoua, The University of Ngaoundere, BP: 26, Garoua
Cameroon
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpm.ijpm_812_21

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