Oral Medicine

Gangrenous stomatitis (cancrum oris): Clinical features, etiologic

factors, and complications
M. L. Chindia*/S. W. Guthua*/S. S. Kimaro**/J. Moshy***

Abstract Gangrenous stomatitis (cancrum oris) is a lesion involving the orofacial structures
that is primarily seen in areas where the socioeconomic standards are iow and there
is poor hygiene. The general clinical features, associated etiologic Jactors. and
ensuing complications in eight consecutive cases diagnosed between 1991 and
1995 are presented and discussed. (Quintessence Int ¡997,28:277-281.)

the disease within the tissues where the associated

Clinical relevance
Among the economically deprived communities,
whose residents constitute a majority of the world
population, gangrenous stotnatitis remains a eom-
mon clinical condition. Although specific know-
ledge on the pathogenesis of this disease remains
elusive, it is important that clinicians caretùlly
document all cases eticountered.
Introduction
Gangrenous stomatitis <CS) is primarily a lesion
involving the orofacial structures.' Although it has
been classified into three types-labi o mental, buccal,
and labiomaxillary'-there are many cases in which the
lesions only involve extensive neerosis ofthe gingivae
and the tooth-bearing bone. In such cases, it is
comtnon to encounter extensive sequestration ofthe
alveolus in the mandible or maxilla without perforation
through the buccal or labial tissues. In such situations,
the defense mechanisms ofthe body manage to contain
* Faculty of Dental Sciences. University of Nairobi, Kenya.
*' Deceased.
" • Faculty of DentUtry, University of Dar-es-Salaam. Tanzania.
Reprint requests: Dr Mark L. Chindia. Faculty of Denial Sciences.
University of Nairobi, PO Box I9íi7(í, Nairobi, Kenya.
causative microorganisms have established themselves.
Because the disease is usually well established
before any medical and dental help is sought, it is
understandable that description ofthe initial stages of
GS may have been inaccurate.' This disease is ob-
served primarily in areas where the social standards are
low and hygiene is poor."" Childhood cases are common
mainly in developing countries, while adult eases are
rare and reported predominantly in industrialized
countries,^ where the disease is eonsidered to be only a
clinical curiosity.'
This article reviews the general clinical characteristics,
associated etiologic factors, and ensuing complica-
tions encountered in eight consecutive cases of GS,
Cases
Patients and methods
Eight consecutive patients who presented between
1991 and 1995 at the joint oral and maxillofacial con-
sultation clinics ofthe Kenyatta National Hospital and
the Department of Dental Surgerj'. University of Nai-
robi ( including one patient examined at the Muhimbili
Medieal Centre, Dar-es-Salaam) were clinically eval-
uated for the general characteristics of GS. Routine
radiographie examination was also performed to
assess the ensuing dentoalveolar complications and
any other craniofacial complications. Routine hémato-
logie investigations were performed for those patients
who underwent surgery.

277
Quintessence International Volume 28, Number 4/1997
Chmdia et al

Table I General characteristics of palients presenting with gangrenous stomatitis

Case Sex Age (yl Presenting features Medieal history Remarks

Stunted growth; gross scarifi-
cation and deficiency of lower
lip; complete trismus
Appeared healthy; extensive
necrotic right palatal shelf
wilh u large oronasal fistula
Appeared healthy; gross
cutaneous scarification over
the right mandibular body;
extensive necrosis ofthe right
alveolar and basal mandible;
marked trismus
Stunted growth; rough, scaly
skin and thin, sparse hain
necrotic alveolar and basal
maxillary bone in Ihe entire
molar area
Stunled growth; sparse, thin
hair; extensive necrosis ofthe
right alveolar, basal, and
palatal shelf with cutaneous
extension
M Emaciated and jaundiced;
massive abdominal Burkitt's
lymphoma; evident hepato-
splenomegajy
Stunted growth; ihin, sparse
hair; gross loss of tissues and
extensive necrosis of the left
maxilla
Crossly emaciated and stunt-
ed growth; severe gingival in-
flammation; blue-black discol-
oration of the right upper lip
from the angle area with
buccal advancement
Reeurreni malarial attacks
and diarrhea ai age 18 months
Recurrent fevers and diarrhea
at age I year; several malarial
attacks
Recurrent fevers, malarial
attacks, and diarrhea at
1 year
Skin condition developed in
infancy; recurrent fevers and
diarrhea in early childhood
Recurrent malarial attacks
since infancy; occasional
diarrhea
Recurrent fevers and malaria!
attacks in most of childhood
Recurrent fevers and malarial
attacks in infancy and most of
childhood
Pancytopenia secondary to
acute myelocytic leukemia
(M II), revealed by blood
chemistry and marrow exami-
nation
Parents are rural peasants
Single mother; ehild under
eare of rural grandmother
most of (he time
Refiigee-camp victim by age
I year
Father is a semiskilled railway
worker supporting a family of
six
Parents reside in a semislum
area without specific employ-
ment
Stays witb both parents; father
a semiskilled worker
Stays with both parents, who
have no specific employment
Single mother; under the care
of rural grandmother

Findings In general, radiographie examination revealed ex-

The patients included five girls and three boys with a
mean age of 5.1 years. The general characteristics of all
the patients are presented in Table 1. Figures 1, 2, ,1,
and 4 depict the clinical appearance ofthe patients in
cases 1, 2, 7, and 8, respectively. The patient in case 8
exhibited the classic skin color change during the acute
phase ofthe condition (Eig 4),
tensive bone necrosis adversely affecting permanent
tooth germs and craniofacial development. In additioti
to the unsightly lip deficiency and scarification,
ankylosis of the temporomandibular joint is an affiic-
tion that causes much misery for the victim, interfering
with mastication and digestion of food, denying the
body the benefit of a balanced diet, and preventing

278 Quintessence International Volume 28, Number


Fig 1 Grcss scarification following deslruction of the
lower lip There was consequent ankylosis in Ihe right
temporomandibular joint.
Fig 3 Gross necrosis of Ihe left maxilla and the associated
soft tissues.
participation in the pleasures traditionally associated
with the culinary arts.^
Discussion
Aithough the hallmark of case studies may lie in the
outcome of overall management, inadequate health
facilities in many industrially developing countries
often lead to failure of optimal treatment and grossly
inconsistent follow-up of cases. Indeed, in many of
these countries, surgical specialties do not even exist. '
The purpose of this article, therefore, is to present the
prevailing status with regard to the etiologic factors
associated with GS. In addition, the range of morbidity
that may be experienced and the reconstructive
Quinfessence international Volume 28, Number 4/1997
Chlndía et al
Fig 2 Extensive necrosis of the righl palatal shelf and
ma>;illary process wilh eventual oronasoculaheous fistula.
Frg 4 Characteristic early GS lesion, which is typically
blue-black in color, that culminates ih the exfoliation ot the
necrotio tissues.
surgical challenges that should be anticipated by
attending clinicians are also discussed. Although GS
may only be a clinical curiosity in economically
developed countries, presentation of these cases is still
important because innovations in reconstructive sur-
gery may öfter improved management strategies.
Notwithstanding the long list of etiologic factors
proposed in the literature, the present cases clearly
showed that GS is essentially a disease of poverty
among children in an underdeveloped economy. This
disease shall continue to present more frequently in the
tropical nonindustrialized regions under the current
socioeconomic constraints. Over the years, diverse
reports have associated the development of GS with a
significant increase in oral fusospirochetal popula-
279
Chindia el al
tions. Hi3wever, il has not been possible to experimen-
tally demonstrate the specific role oflliese organisms
in the initiaiion and propagation of tliis disease.
Cnrrent reviews oftlic etiology of GS indicate that a
wide range of microorganisms may be involved in the
pathogenesis of the condition. Those that have been
frequently implicated include:
1. Eusiforniis fusiforinis
2. Nonhemolytic streptococcus
3. Stapiiyioc()ccu.\ aiireus
4. Treponenia vincentii
5. Bacteroides asacchnroiyticus
6. Anaerobic organisms (known to produce the
destructive collagenase enzyme)
7. Eiisobacterium neirophonnn (generally found in
oral necrotic lesions)
Although it is apparent that poor oral hygiene is a
significant factor, it is also possible that deficiencies of
vitamins ana minerals may coexist, particularly in
children; these may jointly cause an alteration in the
intercellular substances and also a decrease in mucous
secretion, thereby favoring microbial growlh. To eluci-
date the significance of these various factors in the
course of the disease, further specific investigations
would have to be performed at certain stages.
The early clinical features of GS are characteristi-
cally indistinguishable from those of acute necrotizing
ulcerative gingivitis. When the development of gingival
inflammation with ulcération is followed by edema of
the affected area, which may extend into the neigh-
boring tissues, the early features of GS are imminent.
Apparently, the overwhelming bacterial toxins and
enzymes, produced by the large variety of microorga-
nisms, act to amplify the inflammatory response,
which may then rapidly damage the vascular network
ofthe affected area. This is followed by the character-
istic blue-black orocutaneous coloration. Soon after
this stage, tissue breakdown occurs. Clinically, there-
fore, there are two eariy. clearcut phases of GS, which
may be categorized as the ulcerative and necrotic
phases.
Evidently, GS makes its first appearance intraorally
and spreads outward very rapidly to involve adjacent
structures. If early medical intervention is achieved,
tissue destruction may involve mainly ihe oral mucosa
and alveolar and basal bones with minimal destruction
of extraoral structures. Many clinicians believe that
once the gangrenous area has begun to develop, a
certain amount of tissue will be iost no matter what
form of treatment is undertaken. '
280
If tissue necrosis is confined to the intraoral
structures, meticulous conservative oral hygiene pro-
cedures should be instituted and time should be
allotted for any sequestra to separate. Thereafter,
carefully planned sequestrectomy should be performed
lo salvage any closely associated viable structures.
When there is extensive involvement ofthe extraoral
tissues, similar management should be undertaken,
and reconstruction may be attempted only when the
GS lesion has completely healed and any debilitating
underlying condition has been effectively controlled.
Diagnosis of the disease in the acute phase ean
usually be made quite easily if there is a history of
recent or recurrent debilitating illness in a ehild
preceding the appearance ofa foul-smelling orofaeial
mutilating necrosis. The very short duration during
which extensive tissue necrosis occurs should distin-
guish GS from the somewhat rare lesions of leprosy
and llingal infection, it is notable that GS lesions may
respond fairly promptly to antibiotic trealment. while
any other similar lesions will respond only after the
precipitating condition has been corrected.
The relationship between GS and an underling
debilitating condition, such as malnutrition, dehydra-
tion, blood dyscrasia, cliemotherapeutics, and infec-
tious diseases is well documented.^ As exemplified by
the present cases, undernutrition and recurrent infec-
tive diseases are, undoubtedly, the most salient etiolog-
ic factors in the onset of GS. A tropical environment
characterized by heavy parasitemia caused by concur-
rent infestation with a variety of protozoa! organisms,
such as malaria and kala-azar, has a devastatingly
depressive effect on the general immunity of children.
Situations in which an underlying immunosuppres-
sive condition, such as chronic myelocytic leukemia,
may be lurking (case 8) underscore the need for
comprehensive examination of each patient. This
crucial requirement is. however, often hampered by the
costs involved.
The seriousness ofthe clinical problems associated
with GS is underscored by the tact that even with
appropriate antibiotic therapy, time-consuming and
repeated surgery is the only answer for established
cases. Furthermore, the resuh is, at best, less satisfac-
tory in terms of the subsequent quality of life for the
victim.'^ For the patient in case 1, for instance, surgical
procedures would involve resection of the ankylosis.
costochondral bone grafting to effect mandibular
growth, tissue expansion to lessen the microstomia,
and distant flap operations to reconstruct the deficient
lower lip for optitnal function and esthetics. These
Quintessence International Volume 28. Numbet

procedures not only are expensive but also require
meticulous coordination among the clinical specialists
involved if acceptable results are to be anticipated. The
presence of an underlying condition such as chronic
rayelocytic leukemia, which may run a somewhat
unpredictable course, further delays definitive surgical
case management.
Generally it is difficult to estimate the exact
morbidity and mortality rates arising from GS, In most
of Africa, the morbidity of GS is certainly considerable
because prompt management ofthe acute disease can
be grossly inadequate. In the present study, the patient
in case 8 succumbed to the underlying leukemia quite
rapidly. In such a case, the mortality should be more
directly attributed to leukemia than GS. On the whole,
the major causes of death among these patients are
dehydration, bronchopneumonia, toxemia, and other
complications arising from the underlying debilitating
disease. '
Evaluation ofthe current literature' indicates that
although survival of children afflicted by GS may be
high, adequate management ofthe ensuing complica-
tions involving severe tissue loss still demands critical
consideration. Extensive destruction of the dento-
alveolar processes and the adjacent orofacial structures
poses an intricate long-term challenge regarding ac-
ceptable functional and cosmetic reconstruction.
When an entire maxillary dentoalveolar process with
the palatal shelf is destroyed (see Figs 2 and 3),
restoration of balanced facial growth and adequate
dental function may hardly be achieved even with
optimal surgical facilities.
Most ofthe studies of cancrum oris in developing
countries have been preoccupied with specific local
factors and have almost completely neglected host
factors,^ It is now apparent that host factors need to be
examined more closely to unravel the exact pattern of
events that lead to the development of GS. In this way,
management would be aimed at eliminating those
spécifie etiologic factors that prevail before the condi-
tion develops. It is interesting that recurrent GS
Quintessence International Volume 28, Number 4/1997
Chindia et al
lesions appear to be extremely rare."* Whether this is
an indication that some form of immunity to GS
develops with the primary attack is a matter requiring
further investigation.
Currently, the primary treatment for acute GS
lesions involves the use of appropriate antibacterial
agentsi complete "on-loading" of parasites; treatment
of any underlying condition, such as leukemia; and the
restitution ofthe nutritional status. Thereafter, defini-
tive reconstructive surgical treatment may be contem-
plated.
Acknowledgments
The aulhors are iiiosl graleful ic llie Director of Ihe Keriyatla National
Hospital and the chairman ofthe dental unit for their permission to
presen! [he seven cases seen al the hospnal. Smeere thanks are also due to
Keiiah Mbujiua and Josephine Misare for preparing the manuscript
References
1. Prabhij SR, Praetorius F Mixed bacterial infections; Fusospirochae-
tal disease, tn: Prabhu SR. Wilson DF Daftarï DK, Johnson NW
(eds). Oral Diseases in the Tropics. Oxford, England: Oxford
University Press. 1972:269-270.
2. Stassen LFA, Batchelor AGG. Rennie JS. Moos KJ-. Cancrum oris
inanadultcaucasian female. BrJ Oral Maxillofac Surs iyS9;27:4t7-
422.
.1. Tempest MN. Cancrum oris. BrJ Surg I966;5.1:949-9i,<;.
4. Meneïes DM. The etiology and effects ofcancrumonsin children. J
Dent Child 1976:43:92-98.
; . Bcndt BJ. Padmos A, Harder tJ. McArthur PD. Noma. Report of
three adult cases. Aust J Dermaiol Í983;24; 115-111.
6. RoMe NL. Ankylosisof the temporomandibular joint. J R Cott Surg
hdinb 1982:27:67-79.
7 Nash ES, Cheng LHH, Smart K. Cancrum oris-tike tesion. BrJ Oral
Ma.iillofac Surg 1991;29:51-;3.
8. Enwonu.u CO. Infectious oral necrosis 1 cancrum oris) in Nigerian
children: A review. Community Dent Oral Epidemio! l985il3!l9O-
194.
9. Enwonwu CO. Epidemiológica! and biochemical studies of necro-
tiiing ulcerativo gingivitis and noma (cancrum uris) in Nigerian
children. Arch Oral Biol 1972;17:]357-1371.
10 Kaimenyi JT, üuthua SW. Residual facial deformity resulting from
cancrum oris: A case report. Easl Afr Med J 1994:71:476-478. G
281