Introduction
Nasal rhinosporidiosis refers to a chronic granulomatous disease of the
nose that is caused by Rhinosporidium seeberi.1-4 The
disease affects both human beings and animals and is reported more
commonly in hot tropical climates even though it is endemic in India and
Sri Lanka.1,5-8 On the other hand, sporadicity has
been reported elsewhere such as Brazil, Africa and
Argentina.6,7,9,10 Despite the disease being very rare
in Tanzania, there are some countries that have reported an appreciable
number of cases.8
Regarding race and sex predilection of rhinosporidiosis, there is no
racial predominance reported and in terms of sex of predominance, males
are more affected than females with the male to female ratio being
4:1.1,9-11 The disease also affects those individuals
aged 15-40 years.1,2,12
The mode of transmission of rhinosporidiosis may be by direct contact
with spores and this can be through dust, infected clothing and swimming
in stagnant water.1,8,12-14
The diagnosis of nasal rhinosporidiosis is usually established by
observing the characteristics of the implicated etiological agent in
nasal tissue biopsies like sporangia. The sporangia when examined in
nasal tissue biopsies may be visible at variable stages of maturation.
On the other hand, rhinosporidiosis has a tendency of mimicking other
nasal masses as it presents like a polypoidal
mass.1,7,15-17 Therefore a high index of suspicion by
clinicians is of paramount importance in instituting the management of
patients with nasal masses particularly in this era where the disease is
on surge.
The variable clinical presentation of nasal rhinosporidiosis includes an
indolent nasal growth, nasal obstruction, intermittent epistaxis, nasal
itching and sneezing and yellowish foul smelling nasal discharge
accompanied with blood-stained purulent nasal
discharge.1,9,10
The recommended treatment of choice for nasal rhinosporidiosis remains
to be surgical excision of the nasal mass despite the reported high
recurrence rate.3,16,18 Despite being amenable to
surgery, there are reported deaths in patients who are not
immunocompetent.19 Whenever accessible electro
cauterization of the base of the excised site should be done though
cryosurgery can also be used.20 Systemic therapy with
dapsone serves as an important adjuvant therapy.1,20,21
To the best of our knowledge, this is perhaps the second reported case
of nasal rhinosporidiosis in Central Tanzania and the first case of its
unique nature due to synchronicity with inverted papilloma.
Being a rare benign tumor, inverted papilloma was described initially in
1854 by Ward and in 1855 by Billroth.20,22 This tumor
accounts for 0.5-4% of all nasal tumors.20,23 The
disease tends to peak in the 5th to
6th decade 23 with male to female
ratio being between 3:1 and 10:1.20 It is exceptional
in the pediatric population by being very rare as only very few cases
have been reported.24 Though inverted papilloma is
known to be a benign tumor, yet it is characterized by its possibility
of undergoing malignant transformation to carcinoma, local
aggressiveness, high risk of synchronous or metachronous malignancy and
has the propensity of local recurrence especially if incomplete surgical
excision is entertained.22-24
The commonest location for inverted papilloma is the lateral nasal wall
and the paranasal sinuses. There are other rare anatomical sites being
involved by inverted papillomas outside the sinonasal tract such as
skull base 25, nasolacrimal duct 26,
oropharynx 27.28 and
nasopharynx.29,30
The exact etiology of inverted papilloma is still debated to date thus
unknown. There is a laid hypothesis that Human papillomavirus may be
implicated in the development of inverted papillomas; particularly type
11.31-35 There are other proposed aetiological agents
like chronic inflammation, allergy and occupational
exposures.23,33 The treatment of choice remains to be
surgical excision of the nasal mass which can be done endoscopically
whenever equipment permits. 23,26 Despite adequate
treatment of an inverted papilloma, recurrence rate may be as high as 30
– 60 % of cases.20
We are therefore reporting a paediatric patient who presented with a
synchronous nasal rhinosporidiosis and inverted papilloma and was
managed by endoscopic surgical excision of the nasal mass and kept on
oral dapsone for 6 months postoperatively.