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\begin{document}

\title{Actinomycetoma distal radius mimic osteosarcoma, case report of rare
presentation.}



\author[1]{Hassan Elbahri}%
\author[2]{Adnan Alnaser}%
\author[3]{Sawsan  Babiker}%
\author[3]{Alaa Mohamed }%
\affil[1]{International University of Africa}%
\affil[2]{University of Gezira Faculty of Medicine}%
\affil[3]{University of Khartoum Faculty of Medicine}%


\vspace{-1em}



  
  \date{May 25, 2024}


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\selectlanguage{english}
\begin{abstract}
Various conditions may mimic osteosarcoma. The differential diagnosis of
osteosarcoma is broad. A proper history, examination, diagnostic imaging
studies, and pathological analysis are essential to establish the
precise diagnosis. This case report serves to illustrate the
significance of recognizing similarities and other rare features to
prevent late or misdiagnosis.%
\end{abstract}%



\sloppy


\textbf{Introduction}

Osteosarcoma is commonest primary malignant bone tumor of unintelligible
origin, with peak age at 10-19 years old(1). The distal radius is a rare
site for osteosarcoma; it was reported less than \textless{}1\% (2).
Between all the bone tumors, osteosarcoma have the widest variety in
clinical presentation, imaging findings, morphological and histological
features. The mainstream treatment is combination of chemotherapy and
surgical treatment which improved the overall prognosis in the last two
decades (3-4). However, mycetoma is a chronic cutaneous and subcutaneous
painless swelling caused by two major types of pathogens, either
bacterial type (actinomycotic) or fungus type (eumycotic) {[}5{]}. It is
the most common neglected disease of human in tropical and subtropical
areas among low socioeconomic individuals {[}5,6{]}. It involves the
skin and connective tissue. Painless destructive nature, late
presentation, poor response to treatment and high recurrence rate are
characteristic features of this disease. Among all the endemic
countries, Sudan have the highest incidence {[}6-8{]}. The chronic
inflammatory granuloma, various deformaties, disabilities and high
morbidity rate are the commonest known complications of mycetoma, the
disease can be potentially limb threatening in its late stage
{[}9,10{]}. Clinically, mycetoma starts as a small painless lump which
increase in size gradually, forming multiple sinuses with seropurulent
discharge and eventually multicolored grains appear {[}11{]}. Meticulous
Clinical assessment , radiological and histopathological analysis of
such lesion is important for a presice diagnosis and management,
especially when dealing with unclear osteo-articular lesion. Management
of mycetoma predominantly depends on the etiological agent, site and
extent of the disease {[}12{]}. This case review serves to exemplify the
importance of recognizing the similarities between these 2 lesions and
other rare features that will help differentiate between osteosarcoma
and mycetoma, also to highlight and demonstrate the destructive features
of bone involvement by mycetoma that usually associate with inadequate
assessment, and the impact of delayed presentation or late diagnosis to
the overall outcome.

\textbf{Case presentation}

A 40 year old female farmer from Sudan referred to our orthopedic
oncology clinic at Ibrahim Malik Teaching Hospital --Sudan, complaining
of chronic right distal forearm pain, swelling and numbness for two
years, it started with small painless lump that over period of time
increased in size gradually, recently her quality of life and functional
status had been massively affected due to increasing pain intensity,
increase symptoms anguish and constant decrease in range of motion at
the wrist joint. Patient were ill but not pale, no loss of weight
neither constitutional symptoms were noted, vital signs were within
normal range. No Sinuses were noted throughout the course of
presentation. Conventional xray show right distal radius mixed lytic and
sclerotic bone lesion with sunburst periosteal reaction and soft tissue
swelling {[}fig1{]} . Initially and with inadequate assessment, she was
suspected with osteosarcoma distal radius, thus she was referred. No
advance imaging requested, or biopsies were taken. Since then the
deterioration in her general condition had been growing steadily.
Painful activity of daily living agonized the patient, swelling and
limitation of movement on the affected side had been worsening
dramatically. Furthermore she ended by using daily pain killer for long
time. On clinical examination there was obvious right distal forearm
swelling and tenderness mainly at the radial and dorsal side of the
forearm, the mass was irregular in shape, hard in consistency measured
about 7*3 cm, with mild temperatures asymmetry compared with adjacent
skin, no sinuses were identified lymphatic streaks. Hematological
investigations were unremarkable, X-ray revealed the same findings and
MRI of right wrist show right distal forearm swelling (enhancement)
incasing distal neurovascular bundle , pre-tumor edema , no skip
lesions{[}fig2-3{]}. CT chest \& chest X-ray were normal, no evidence of
metastases. At this point the decision was to take core biopsy under
general anasthesia. Histopathology analysis reports multiple grains of
streptomyces somaliensis surrounded by granulasion tissues with gaint
cell granuloma, the features of actinomycetoma consistent with
streptomyces somaliensis{[}fig4{]} . Second surgery i.e. wide local
excision and fixation was done and second biopsy were taken, confirming
the diagnose.

After two months from operation the patient's condition improved and
referred to mycetoma center in Soba hospital for medications {[}fig5{]}
.

\textbf{Discussion and conclusion}

Actinomycetoma Osteomyelitis radius is very rare condition to be
reported. Destructive bone pattern by mycetoma species, in situ grains
formation and Unequivocal distinction between actinomycetoma and various
manifestations of osteosarcoma either in painful event or numbness at
late stage are extremely rare findings to be seen, also the unique
periosteal reaction in form of sunburst ray appearance can also be the
source of significant diagnostic challenges, when it happens the risk of
limb loss by amputation will be very high. Initial Painless nature of
the lesion, low socioeconomic status and low level of education are the
reason drives Sudanese patients to present late.

As mentioned above the treatment of mycetoma predominantly depends on
the infective agent, site and extent of infection{[}12{]}. Until
recently in Sudan, amputation was the only available treatment for
mycetoma, as extremely high recurrence rate prevent optimum therapeutic
results to be reached.

Actinomycetoma (bacterial type) is usually treated with medications only
as it shows relative response to medical treatment in early stages. For
fungal type (eumycetoma), a combination of aggressive surgical and
medical treatment (anti-fungal agents) are the gold standard as drug
resistant prevent solely medical treatment {[}13{]}.

Actinomycetoma infective agents are difficult to confirm. Hence,
accurate assessment should include proper clinical history and
examination, radiological evaluation by expert radiologist and
Orthopedist, pathological analysis of the affected area with a Tru-Cut
needle biopsy and immunohistochemistry to avoid problems of inadequate
specimens commonly associated with incisional biopsy {[}14{]}. Overall
outcome can be optimized significantly after precisely identification of
the causative organism and extended post treatment follow up.

Unfortunately due to the late presentation, aggressive features of bone
and surrounding structures Invasion found in our patient and fear of
high recurrence rate, surgical approaches in combination with

Medical treatment were decided. Many Sudanese patients undergo many
operations with several Prolonged medical regimens to enable adequate
response. Nevertheless, surgical options for mycetoma Treatment in Sudan
range from wide local excision to amputation of the affected limb,
correct wide local Excision and bloodless field by expertise during
surgery are paramount essential to excellence the Outcome {[}15{]}.

The post treatment recurrence rate is high, range between 25-50\%. Age,
duration, site and extent of Involvement and previous history of
mycetoma recurrence are predictors of overall outcome. Thereby, Surgical
operation considers the best treatment option to minimize the risk of
recurrence especially if Done properly as mentioned above{[}16{]}.

Surgical intervention unfortunately associates with high rate of
morbidity and disability among Mycetoma patients in Sudan. Postoperative
extended follow up, physiotherapy of the nearby joints and Adherence to
antibiotics regimen regarding to the protocol are essential for better
clinical and functional Outcomes and to avoid the permanent
disabilities.(17)

In conclusion, actinomycetoma Osteomyelitis radius is rare condition.
Clinicians in endemic areas must Consider mycetoma osteomyelitis as a
differential diagnosis when they are dealing with vague atypical
Musculoskeletal destructive lesions. Multidisciplinary team and Triple
assessment including clinical, Radiological and histopathological
correlation are extremely important to prevent misdiagnosis, surgical
Treatment in combination with medical treatment followed by regular
clinical and radiological follow up Can be limb saving procedure in such
cases. Finally, treatment of mycetoma osteomyelitis cases is case By
case individually according to the predictors of post-operative
recurrence.(17)

\textbf{Reference}

1.Messerschmitt PJ, Garcia RM, Abdul-karim FW, Greenfield EM, Getty PJ.
Osteosarcoma. J Am Acad Orthop Surg. 2009;17:515--527.

2.Unni KK, editor. Dahlin's Bone Tumors: General Aspects and Data on
11,087 Cases. 5\textsuperscript{th} edition. Lippincott-Raven;
Philadelphia, PA: 1996. Pp. 143--183.

3. Wadhwa N. Osteosarcoma: Diagnostic dilemmas in histopathology and
prognostic factors. Indian Journal of orthopaedics. 2014
Jun;48(3):247-54.

4.Eckardt JJ, Grogan TJ. Giant cell tumor of bone. Clin Orthop Relat
Res. 1986;204:45--58.

5.Zijlstra EE, Van De Sande WW, Fahal AH. Mycetoma: a long journey from
neglect. PLoS Negl Trop Dis. 2016;10(1):e0004244.

6.Develoux M, Dieng MT, Kane A, Ndiaye B. Management of mycetoma in
West-Africa. Bull Soc Pathol Exot. 2004;96(5):376--82.

7.Fahal AH. Mycetoma thorn on the flesh. Trans R Soc Trop Med Hyg.
2004;98(1):3--11.

8.Fahal AH, Hassan MA. Mycetoma. Br J Surg. 1992;79(11):1138--41.

9.Bonifaz A, Tirado-S\selectlanguage{ngerman}ánchez A, Calderón L, Saúl A, Araiza J, et al.
Mycetoma: experience of 482 cases in a Single center in Mexico. PLoS
Negl Trop Dis. 2014;8(8):e3102.

10.Ahmed AO, van Leeuwen W, Fahal A, van de Sande W, Verbrugh H, et al.
Mycetoma caused by Madurella mycetomatis: a neglected infectious burden.
Lancet Infect Dis. 2004;4(9):566--74.

11.Nenoff P, van de Sande WW, Fahal AH, Reinel D, Schöfer H. Eumycetoma
and actinomycetoma---an Update on causative agents, epidemiology,
pathogenesis, diagnostics and therapy. J Eur Acad Dermatol Venereol.
2015;29(10):1873--83.

12.Welsh O, Al-Abdely HM, Salinas-Carmona MC, Fahal AH. Mycetoma medical
therapy. PLoS Negl Trop Dis. 2014;8(10):e3218.

13.Abd El Bagi ME. New radiographic classification of bone involvement
in pedal mycetoma. AJR Am J Roentgenol. 2003;180(3):665--8.

14.Ahmed AA, van de Sande W, Fahal AH. Mycetoma laboratory diagnosis:
Review article. PLoS Negl Trop Dis. 2017;11(8): e0005638.

15.Bakhiet SM, Fahal AH, Musa AM, Mohamed ESW, Omer RF, Ahmed ES, El
Nour M, Mustafa ERM, Sheikh A, Rahman ME, Suliman SH, El Mamoun MAG, El
Amin HM. A holistic approach to the mycetoma Management. PLoS Negl Trop
Dis. 2018;12(5):e0006391.

16.Wadal A, Elhassan TA, Zein HA, Abdel-Rahman ME, Fahal AH. Predictors
of post-operative mycetoma Recurrence using machine-learning algorithms:
the mycetoma research center experience. PLoS Negl Trop Dis.
2016;10(10): e0005007.

17.Awad A, Alnaser A, Abd-Elmaged H, Abdallah R, Khougali HS. Eumycetoma
Osteomyelitis Calcaneus in Adolescent; report of case and literature
review. BMC Infectious Diseases. 2021 Dec;21(1):1-4.\selectlanguage{english}
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