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Mycetoma of the scalp: report of a case
412
TRANSACTIONS OFTHEROYALSOCIETY OFTROPICAL MEDICINE ANDHYGIENE
Mycetoma King Faisal
of the scalp:
(1986), 80, 412-414
report
of a case
J. T. ANIM AND NAFISA 0. EL-GAALI University and King Fahd Teaching Hospital, P.O. Box 2114, Dammam 31451, Saudi Arabia Abstract
Mycetoma of the scalp in a lo-year-old boy is reported. This is the first report of this infection in the Eastern Province of Saudi Arabia. The causative agent, Actinomadura madurue, has already been described in the Kingdom and shown by other workers to affect other parts of the body. The probable pathogenesis is discussed and the need to increase clinical awareness of the condition stressed. Introduction
Case Report
Mycetoma commonly affects the extremities, especially the lower. Involvement of the head and neck is rare but in endemic areas, such as the Sudan, cranial and mandibular involvement have been reported (HICKEY, 1956; MAHGOUB & MURRAY, 1973; GUMAA et aE., 1975). Involvement of the paranasal sinuses has also been described in a Congolese child (MUNYUNGA-KASENGULU et al., 1971). Although the diseaseis known in Saudi Arabia (KUBBA & SATIR, 1984; MAHGOUB,1983), involvement of the head and neck has not been documented. Mycetoma in children is also uncommon, but has been reported in endemic areas (MUNYUNGA-KASENGULU et al.. 1971: MAHGO~B, 1976). We here report a case of involvement of the temporal region of the scalp by mycetoma in a lo-year-old boy, to our knowledge the first report from the Eastern Province of Saudi Arabia.
Fig. 1. CT
scanshowing
soft tissue
T.A.A., a lo-year-old boy, presented to the teaching hospital of King Faisal University in Al-Kobar, complaining of persistent left-sided headache of several days duration, dizziness and fever. He had had a swelling in the left temporal region five months previously which was drained in another hospital. At the time of presentation the left temporal swelling had recurred. Examination revealed tenderness over a fluctuant swelling in the left temporo-parietal region of the scalp. No other lesions were noted elsewhere in the body. X-ray of the skull showed soft tissue swelling in the area with mild reactive changes on the outer surface of the temporal bone but no obvious bone destruction. This was confirmed by CT scan (Fig. 1). No lesions were seen intracranially. A diagnosis of left temporal osteomyelitis was made. In view of the persistent headacheand fever, the soft tissue masstogether with
III~SS in the left temporo-parietal region
with mild periosreal reaction.
J. T. ANIM AND N. 0. EL-GAALI
Fig. 2. An abscess containing a colony of micro-organisms (H & E x 200).
attached temporalis muscle and the underlying flap of temporal bone was excised. Specimens were sent for histological and microbiological examination. Histology revealed chronic suppurative inflammatory tissue with focal abscessescontaining large colonies of micro-organisms. These stained deeply purple with haematoxylin and eosin (Fig. 2) and were positive with Gram’s stain. The delicate filamentous structure of the organism was demonstrable. The morphological and staining characteristics were consistent with Acrinomadura madurue. No culture studies were performed to isolate the organism and the grain characteristics were not studied becausethe diagnosis was not suspected at the time of examination. The temporalis muscle showed interstitial inflammatory changes with focal fibrosis and the underlying bone also showed inflammatory changes in the periosteum but no bone destruction or involvement by the organism. Discussion
The occurrence of mycetoma in Saudi Arabia has been noted by several workers (MAHGOUB, 1983; KUBBA & SATIR, 1984; BUTLER & MACKEY, 1980).
In their analysis of 10 casescollected over a three-year period no involvement of the head and neck was noted by KUBBA & SATIR (1984). Only one case each has been recorded by HICKEY (1956) and GUMAA et ~1. (1975) and in both the causative agent was Madurella mycehnatis. The involvement of the scalp in our case, therefore, is somewhat unusual. The pathogenesis of the scalp diseaseis not clear but direct inoculation of
which stain deeply purple with haematoxylin
and eosin
the micro-organism into scratches and abrasions sustained from leaves and branches bearing the organisms is a possibility (Satir, 1984; personal communication). In support of this mechanism is the fact that mycetoma in endemic areas, is common among agricultural workers and in adult males who are more likely to become exposed to the organisms. No definite history of exposure could be obtained in our cases. Of the ten cases reported by KUBBA & SATIR (1984), four were due to A. madurue, as in our case. The remaining six were due to M. mycetomatis. The organisms causing the disease vary with different localities (GuMAA~~u~., 1975;M~~~0~~,1976,1977) and it is necessary to study the distribution of organisms for each locality. The value of the study of grain characteristics and culture of the organism has been stressedmainly for selecting the right treatment, by various workers (KUBBA & SATIR, 1984; MAHGOUB, 1973, 1976 and 1977). Mycetoma is becoming increasingly recognized in Saudi Arabia. The prevalence of the disease has probably been underestimated and it may be more common in the southern part of the Kingdom where infectious diseases are known to be more common (El-Hassan, 1984;personal communication). Only a nationwide survey can determine whether or not the diseaseis endemic in Saudi Arabia. It is thus important to consider this diseasein the differential diagnosis of chronic inflammatory conditions of any part of the body, including the head and neck, in Saudi Arabia. This will ensure that appropriate
414
MYCETOMA OF THE SCALP
examination with the naked eye is made both at the time of surgery and during pathological examination and that culture studies are performed to characterize the infecting organism. This will undoubtedly aid in the choice of the appropriate chemotherapeutic agents in the treatment of the disease as has been stressed by earlier workers (MAHGOUB, 1976, 1973).
Kubba, A. & Satir, A. A. (1984). Mycetoma in the Eastern Province of Saudi Arabia. Saudi Medical Journal, 5, 147-151. Mahgoub? E. S. (1976). Medical management of mycetoma. Bulletm of the World Health Organization,
54, 303-310.
Butler, P. G. & Mackey, I. (1980). Medical Treatment of Mycetoma. Proceedings of the Fourth Saudi Medical Conference, Dammam, King Faisal University, pp. 190192. Gumaa, S. A., Satir, A. A., Shehata, A. H. & Mahgoub, E. S. (1975). Tumor of the mandible caused by Madurella
Mahgoub, E. S. (1977). Mycosis of the Sudan. Transactions of the Royal Society of Tropical Medicine and Hygiene, 71, 184-188. Mahgoub, E. S. (1983). As quoted by: Kubba, A. & Satir, A. A. (1984). Saudi Medical Journal, 5, 147-151. Mahgoub, E. S. & Murray, I. G. (1973). Mycetoma. London: William Heinemann Medical Books. Munyunga-Kasengulu, C., Bastin, J. P., Gatti, F. & Vanbreuseghem, R. (1971). Invasion of paranasal sinuses by Madurella mycetomi in a Congolese child. Annales de la Soci& Belge de Mldicine Tropicale, 51, 247-254.
Hickey, B. B. (1956). Cranial maduromycosis. Transactions of the Royal Society of Tropical Medicine and Hygiene, 50, 393-396.
Accepted for publication
References
mycetomi. American Journal Hygiene, 24, 471-474.
FELLOWSHIP
of Tropical
OF
THE
Medicine
and
ROYAL AND
SOCIETY HYGlENk
OF
TROPICAL
20th March,
1983.
hlE,DICINl:
All registered medical and veterinary practitloncrs, nurse< :~nd o111~~1.~ intcrcstcd in xicntific pursuits relating to tropical medicine, whose qualifications arc dcc~ned hnti\lactor!, hy tllc Cou~rc~i, arc cllgihlc for election as Fellows of the Socictv.
The annual subscription payable is f30 The Transactions and the current Year Hooh of the Swietv arc posted regularly, Surface Post, to every Fellow ivhose subscription is nut in xxar. Further
information
may be obtained from: The Honorary Secretaries, Koyal Society of Tropical Medicine Manson House, 26 Portland Place London WlN 4EY
and Hygiene,
by Accclcrated
TRANSACTIONS OFTHEROYALSOCIETY OFTROPICAL MEDICINE ANDHYGIENE
Mycetoma King Faisal
of the scalp:
(1986), 80, 412-414
report
of a case
J. T. ANIM AND NAFISA 0. EL-GAALI University and King Fahd Teaching Hospital, P.O. Box 2114, Dammam 31451, Saudi Arabia Abstract
Mycetoma of the scalp in a lo-year-old boy is reported. This is the first report of this infection in the Eastern Province of Saudi Arabia. The causative agent, Actinomadura madurue, has already been described in the Kingdom and shown by other workers to affect other parts of the body. The probable pathogenesis is discussed and the need to increase clinical awareness of the condition stressed. Introduction
Case Report
Mycetoma commonly affects the extremities, especially the lower. Involvement of the head and neck is rare but in endemic areas, such as the Sudan, cranial and mandibular involvement have been reported (HICKEY, 1956; MAHGOUB & MURRAY, 1973; GUMAA et aE., 1975). Involvement of the paranasal sinuses has also been described in a Congolese child (MUNYUNGA-KASENGULU et al., 1971). Although the diseaseis known in Saudi Arabia (KUBBA & SATIR, 1984; MAHGOUB,1983), involvement of the head and neck has not been documented. Mycetoma in children is also uncommon, but has been reported in endemic areas (MUNYUNGA-KASENGULU et al.. 1971: MAHGO~B, 1976). We here report a case of involvement of the temporal region of the scalp by mycetoma in a lo-year-old boy, to our knowledge the first report from the Eastern Province of Saudi Arabia.
Fig. 1. CT
scanshowing
soft tissue
T.A.A., a lo-year-old boy, presented to the teaching hospital of King Faisal University in Al-Kobar, complaining of persistent left-sided headache of several days duration, dizziness and fever. He had had a swelling in the left temporal region five months previously which was drained in another hospital. At the time of presentation the left temporal swelling had recurred. Examination revealed tenderness over a fluctuant swelling in the left temporo-parietal region of the scalp. No other lesions were noted elsewhere in the body. X-ray of the skull showed soft tissue swelling in the area with mild reactive changes on the outer surface of the temporal bone but no obvious bone destruction. This was confirmed by CT scan (Fig. 1). No lesions were seen intracranially. A diagnosis of left temporal osteomyelitis was made. In view of the persistent headacheand fever, the soft tissue masstogether with
III~SS in the left temporo-parietal region
with mild periosreal reaction.
J. T. ANIM AND N. 0. EL-GAALI
Fig. 2. An abscess containing a colony of micro-organisms (H & E x 200).
attached temporalis muscle and the underlying flap of temporal bone was excised. Specimens were sent for histological and microbiological examination. Histology revealed chronic suppurative inflammatory tissue with focal abscessescontaining large colonies of micro-organisms. These stained deeply purple with haematoxylin and eosin (Fig. 2) and were positive with Gram’s stain. The delicate filamentous structure of the organism was demonstrable. The morphological and staining characteristics were consistent with Acrinomadura madurue. No culture studies were performed to isolate the organism and the grain characteristics were not studied becausethe diagnosis was not suspected at the time of examination. The temporalis muscle showed interstitial inflammatory changes with focal fibrosis and the underlying bone also showed inflammatory changes in the periosteum but no bone destruction or involvement by the organism. Discussion
The occurrence of mycetoma in Saudi Arabia has been noted by several workers (MAHGOUB, 1983; KUBBA & SATIR, 1984; BUTLER & MACKEY, 1980).
In their analysis of 10 casescollected over a three-year period no involvement of the head and neck was noted by KUBBA & SATIR (1984). Only one case each has been recorded by HICKEY (1956) and GUMAA et ~1. (1975) and in both the causative agent was Madurella mycehnatis. The involvement of the scalp in our case, therefore, is somewhat unusual. The pathogenesis of the scalp diseaseis not clear but direct inoculation of
which stain deeply purple with haematoxylin
and eosin
the micro-organism into scratches and abrasions sustained from leaves and branches bearing the organisms is a possibility (Satir, 1984; personal communication). In support of this mechanism is the fact that mycetoma in endemic areas, is common among agricultural workers and in adult males who are more likely to become exposed to the organisms. No definite history of exposure could be obtained in our cases. Of the ten cases reported by KUBBA & SATIR (1984), four were due to A. madurue, as in our case. The remaining six were due to M. mycetomatis. The organisms causing the disease vary with different localities (GuMAA~~u~., 1975;M~~~0~~,1976,1977) and it is necessary to study the distribution of organisms for each locality. The value of the study of grain characteristics and culture of the organism has been stressedmainly for selecting the right treatment, by various workers (KUBBA & SATIR, 1984; MAHGOUB, 1973, 1976 and 1977). Mycetoma is becoming increasingly recognized in Saudi Arabia. The prevalence of the disease has probably been underestimated and it may be more common in the southern part of the Kingdom where infectious diseases are known to be more common (El-Hassan, 1984;personal communication). Only a nationwide survey can determine whether or not the diseaseis endemic in Saudi Arabia. It is thus important to consider this diseasein the differential diagnosis of chronic inflammatory conditions of any part of the body, including the head and neck, in Saudi Arabia. This will ensure that appropriate
414
MYCETOMA OF THE SCALP
examination with the naked eye is made both at the time of surgery and during pathological examination and that culture studies are performed to characterize the infecting organism. This will undoubtedly aid in the choice of the appropriate chemotherapeutic agents in the treatment of the disease as has been stressed by earlier workers (MAHGOUB, 1976, 1973).
Kubba, A. & Satir, A. A. (1984). Mycetoma in the Eastern Province of Saudi Arabia. Saudi Medical Journal, 5, 147-151. Mahgoub? E. S. (1976). Medical management of mycetoma. Bulletm of the World Health Organization,
54, 303-310.
Butler, P. G. & Mackey, I. (1980). Medical Treatment of Mycetoma. Proceedings of the Fourth Saudi Medical Conference, Dammam, King Faisal University, pp. 190192. Gumaa, S. A., Satir, A. A., Shehata, A. H. & Mahgoub, E. S. (1975). Tumor of the mandible caused by Madurella
Mahgoub, E. S. (1977). Mycosis of the Sudan. Transactions of the Royal Society of Tropical Medicine and Hygiene, 71, 184-188. Mahgoub, E. S. (1983). As quoted by: Kubba, A. & Satir, A. A. (1984). Saudi Medical Journal, 5, 147-151. Mahgoub, E. S. & Murray, I. G. (1973). Mycetoma. London: William Heinemann Medical Books. Munyunga-Kasengulu, C., Bastin, J. P., Gatti, F. & Vanbreuseghem, R. (1971). Invasion of paranasal sinuses by Madurella mycetomi in a Congolese child. Annales de la Soci& Belge de Mldicine Tropicale, 51, 247-254.
Hickey, B. B. (1956). Cranial maduromycosis. Transactions of the Royal Society of Tropical Medicine and Hygiene, 50, 393-396.
Accepted for publication
References
mycetomi. American Journal Hygiene, 24, 471-474.
FELLOWSHIP
of Tropical
OF
THE
Medicine
and
ROYAL AND
SOCIETY HYGlENk
OF
TROPICAL
20th March,
1983.
hlE,DICINl:
All registered medical and veterinary practitloncrs, nurse< :~nd o111~~1.~ intcrcstcd in xicntific pursuits relating to tropical medicine, whose qualifications arc dcc~ned hnti\lactor!, hy tllc Cou~rc~i, arc cllgihlc for election as Fellows of the Socictv.
The annual subscription payable is f30 The Transactions and the current Year Hooh of the Swietv arc posted regularly, Surface Post, to every Fellow ivhose subscription is nut in xxar. Further
information
may be obtained from: The Honorary Secretaries, Koyal Society of Tropical Medicine Manson House, 26 Portland Place London WlN 4EY
and Hygiene,
by Accclcrated