Treatment of Cutaneous Leishmaniasis With Antimony Sodium Gluconate Walter A. Koerber, Jr, MD;
Marilyn C. Koehn, MD;
Leishmaniasis, while uncommon in the United States, is found frequently in other parts of the world. We present a case of cutaneous leish-
maniasis and describe cal and histological and treatment.
diagnosis, clinicharacteristics,
Report of a Case 45-year-old man vacationed
A with his wife on the Yucatan Peninsula in Mexico in March 1975. Approximately nine days after leaving, they both noticed that "bites" on the arms had evolved into crusted papules. The man, the more symptomatic of the two, was seen by one of us (M.K.) in August 1975. A biopsy specimen showed a focus of marked ulceration in which the upper portion of the dermis and connective tissue
completely replaced by amorphous eosinophilic material. The dermis showed various-sized aggregates of histiocytes and multinucleated giant cells that were surrounded by a moderate to sharp infiltration of lymphocytes. Also in the dermis were well-marginated tubercles, some of which were obscured by the surrounding lymphocytic infiltration. were
Cultures on Sabouraud's agar with chlorand cycloheximide, tubed-egg medium, and NNN (Novy MacNeil-Nicolle) media were negative, after three separate isolation attempts. Routine bacterial cul¬ tures, Gram stains, acid-fast stains, and multiple Giemsa stains of aspirate from the ulcer were also noncontributory.
amphenicol
From the Department of Dermatology, Stanford (Calif) University Medical Center. Reprint requests to the Department of Dermatology, Stanford University Medical Center, Stanford, CA 94305 (Dr Jacobs).
Paul H.
Jacobs, MD; Betty J. Russell, MS
Serum for direct agglutination titers submitted to the National Communi¬ cable Disease Center (NCDC) in Atlanta on Dec 5,1975. Results were as follows: Leishmania trópica, 1:256 (1:128, diagnostic); L donovani, 1:16 (1:32, diagnostic); and L braziliensis, 1:32 (1:64, diagnostic). A 24hour Montenegro skin test showed an erythema of 15 x 17 mm, a 48-hour test showed an erythema of 54 x 42 mm and an induration of 18 x 18 mm, and a 70-hour test showed an induration of 30 x 25 was
mm.
Because of the continued pain and of the patient's lesions, the decision was made to begin active treat¬ ment with antimony sodium gluconate (sodium stibogluconate, Pentostam, Great
enlargement
Britain). The generally accepted dosage for Pen¬ tostam is 0.1 ml/kg per injection, with a maximum of six ml (600 mg of antimony) per injection.1 The patient received 600 mg of Pentostam intramuscularly for ten days,
three ml in each buttock at a concentration of 100 mg/ml. The only side effect he experienced was a slight discomfort at the sites of injection. Values and results from the following laboratory tests, which were performed before and after treatment, were within normal limits: complete blood count and
differential; urinalysis; ECG; BUN; creatinine; total bilirubin; and a chest x-ray
film. Four days after treatment began, the ulcération on the elbow became more active, with increased erythema and crust¬ ing. Two days later this activity began to subside, and the lesions slowly healed. One month later the ulcération and crusting were gone, and the patient was left with a scar.
Solitary Nevus Lipomatosus Cutaneus Superficialis of the Knee Venkateshiah
The is is
Sathyanarayana, MD, Stanley Weitzner,
solitary nodular form of nevus lipomatosus cutaneus superficial-
cases
The site in the 21 in the literature was
uncommon.
reported
From the
Department of Pathology, Universi-
ty of Mississippi Medical Center, Jackson.
Reprints
not available.
MD
the upper thigh in eight cases,1-3 the buttock in seven cases,1-3 the lower back in two cases,1 and the axilla,1 arm,1 ear,4 and scalp5 in one case each. There is no apparent sex predilection for this condition. The age when the lesion was first noted was available for 16 patients, and it varied from 195
Downloaded From: http://archderm.jamanetwork.com/ by a University of Pittsburgh User on 06/03/2015
Comment Because of limited demand, Pento¬ stam has not been licensed in this country and must be restricted by federal law to investigational use only.1 The NCDC has obtained permis¬ sion from the Food and Drug Admin¬ istration to supply physicians with the drug for therapy of leishmaniasis. Reactions to pentavalent antimony compounds are not severe, as a rule. Antimony sodium gluconate, due in part to its rapid excretion, seems to be the least toxic of the commonly used
pentavalent antimony compounds. Side effects include gastrointestinal disturbance, jaundice, albuminuria, weakness, dermatitis, cough, pneumo¬ nia, and changes in the ECG.1 Most of the above experiences with
Pentostam are in the treatment of kala azar. Many of the previously described signs and symptoms of Pentostam therapy may occur in kala azar. Cahill reported no side effects in six cases of patients with kala azar treated with intravenous Pentostam.2 Pentostam has also been well toler¬ ated by children under two years of age.1 Our patient experienced no side effects other than transient pain at the site of injection. References 1. Informational Material for Physicians: Pentostam (Sodium Antimony Gluconate): Protocol
Atlanta, Center for Disease Control. 2. Cahill KM: Leishmaniasis in the Sudan Republic: Infection in American personnel. Am J Trop Med Hyg 13:794-799, 1964. #53.
including two cases that occurred in the fourth decade and four cases each that occurred in the third, fifth, and sixth decades.1,3 The clinical diagnosis was of a variety of benign lesions, most commonly nevocellular nevus and fibroepithelial papilloma. A case of solitary nevus lipomatosus cutaneus superficialis, which is, to our knowledge, the first case to be present since birth and on the knee, is to 641 years,
reported.
Report
of
a
Case
underwent excision of 19-year-old an asymptomatic nodule on the left knee that had been present since birth. The A
man
Urticaria
Pigmentosa in Identical
form is difficult. Wilson-Jones et al1
Twinslatter
agree that the
solitary form in the may represent a forme fruste of the typical plaque form.5 Although adipose tissue in the dermis may suggest several other lesions, a plentiful, normal appearance of the dermis, absence of nevus cells, and no central core of adipose tissue in our case exclude diagnoses of focal dermal hypoplasia, nevus pigmentosus et lipo¬ matosus, and skin tag, respectively. We believe that solitary nevus lipo¬ matosus cutaneus superficialis is probably more common than is sug¬ gested by the paucity of cases in the
Fig 1—Islands of mature adipose tissue, often vascularized, In upper and middermis; increase of subpapillary capillar¬ ies; and epidermis with crypt-like indenta¬ tion and focal, irregular, elongation of rete ridges (hematoxylin-eosin, x100).
lesion was soft to rubbery, skin-colored, and measured 15 x 6 x 6 mm. The results of the remainder of the physical examina¬ tion were not remarkable. Microscopically, the nodule showed scat¬ tered small islands of mature adipose tissue in normal-appearing dermal colla¬ gen. The adipose tissue was primarily in the upper half of the lesion and in places close to the papillary dermis. Small vascu¬ lar channels, primarily capillaries, were increased in the subpapillary dermis and collections of adipose tissue. The slightly acanthotic epidermis had an occasional crypt-like indentation and irregular elon¬ gation of some rete ridges (Fig 1 and 2). There was no continuity of dermal and subcutaneous adipose tissue. Nevus cells were absent. Collagen fibers and elastic tissue appeared to be normal with trichrome and elastic van Gieson stains. There was absence of metachromasia with toluidine blue stain, and there was no appreciable increase of ground substance with alcian blue stain.
Urticaria
areas
literature.
The
small
number of
reported cases may be owing to a lack of recognition and confusion of the condition with pedunculated lipofibroma.
Angel Arafiles, MD, provided the clinical permission to report the case.
data
and the
Fig 2.—Vascular Island of mature adipose tissue in superficial dermis and epidermis with irregular acanthosis and elongated rete ridge (hematoxylin-eosin, x 500). Comment tissue in the dermis distin¬
Adipose guishes nevus lipomatosus cutaneus superficialis from other nevoid anom¬ alies. The adipose tissue is considered to be derived from the dermal perivascular mesenchymal tissue. Solitary nevus lipomatosus cutaneus superfi¬ cialis may occur in any site, although to account for its predilection for the thigh and buttock in cases of the more common, typical, multinodular plaque
Pigmentosa in Identical
References 1. Wilson-Jones E, Marks R, Pongsehirun D: Nevus superficialis lipomatosus: A clinicopathological report of twenty cases. Br J Dermatol 93:121-133, 1975. 2. Nikolowski W: \l=U"\berNaevus lipomatodes cutaneus superficialis (Hoffmann-Zurhelle). Dermatol Wochenschr 122:735-741, 1950. 3. Kurihara Y, Yasyhara M, Minami H, et al: Nevus lipomatosus cutaneus superficialis (Hoffmann-Zurhelle). Skin Research 9:48-57, 1968. 4. Knoth W: \l=U"\berNaevus lipomatosus cutaneus superficialis Hoffmann-Zurhelle und \l=u"\ber Naevus naevocellularis partim lipomatodes. Dermatologica 125:161-173, 1962. 5. Weitzner S: Solitary nevus lipomatosus cutaneus superficialis of scalp. Arch Dermatol 97:540-542, 1968.
Twins
Alan S. Rockoff, MD
Urticaria neous
pigmentosa (UP), or cutamastocytosis, is the most
mast cell disease of childhood. It produces no visceral lesions or systemic symptoms and usually improves by adolescence.1 During the last 100 years, the literature has reported cases of various forms of mastocytosis in 40 families in which more than one member was affected.2 Among these cases were 11 common
Accepted for publication Oct 24, 1977. Department of Pediatrics, Mt Sinai Hospital, Hartford, Conn. Dr Rockoff is now with the Department of Dermatology, Boston University School of Medicine. Reprint requests to the Department of Dermatology, Boston University School of Medicine, 75 E Newton St, Boston, MA 02118. From the
one set of triplets. pairs of identical twins and the triplets were concordant for the disorder, while two identical twin pairs and
sets of twins and
Seven
the two fraternal sets were discordant. We report an eighth set of identical twins who both have UP, and we discuss the genetics of this condition.
Report of Cases Twins 1 and 2 were black, male infants who were seen at the age of 4 months. Their parents were not related, and there was no family history of skin disease. The delivering obstetrician recalled that there was one placenta, but the obstetrician had not noted whether the amniotic sac or chorion were single or double. Neither twin had vomiting, persistent
diarrhea, or flushing. At the age of 2 months, hyperpigmented lesions developed behind the right ear of twin 1. These later spread to cover his entire body, including scalp and soles. About a week after his rash began, an eruption of identical description and distribution developed on twin 2. Physical examination revealed welldeveloped infants, each with brown hair and eyes. Neither had lymphadenopathy or hepatosplenomegaly. Each had numerous reddish-brown, 2- to 4-mm papules on the trunk, extremities, palms, soles, and scalp. Several of these papules were surrounded by a pink halo, while the halo developed around others after being rubbed (positive Darier's sign). Punch biopsy specimens of back lesions
from each twin showed nests of cells in the upper dermis that were identified as mast cells by toluidine blue staining.
Downloaded From: http://archderm.jamanetwork.com/ by a University of Pittsburgh User on 06/03/2015
Marilyn C. Koehn, MD;
Leishmaniasis, while uncommon in the United States, is found frequently in other parts of the world. We present a case of cutaneous leish-
maniasis and describe cal and histological and treatment.
diagnosis, clinicharacteristics,
Report of a Case 45-year-old man vacationed
A with his wife on the Yucatan Peninsula in Mexico in March 1975. Approximately nine days after leaving, they both noticed that "bites" on the arms had evolved into crusted papules. The man, the more symptomatic of the two, was seen by one of us (M.K.) in August 1975. A biopsy specimen showed a focus of marked ulceration in which the upper portion of the dermis and connective tissue
completely replaced by amorphous eosinophilic material. The dermis showed various-sized aggregates of histiocytes and multinucleated giant cells that were surrounded by a moderate to sharp infiltration of lymphocytes. Also in the dermis were well-marginated tubercles, some of which were obscured by the surrounding lymphocytic infiltration. were
Cultures on Sabouraud's agar with chlorand cycloheximide, tubed-egg medium, and NNN (Novy MacNeil-Nicolle) media were negative, after three separate isolation attempts. Routine bacterial cul¬ tures, Gram stains, acid-fast stains, and multiple Giemsa stains of aspirate from the ulcer were also noncontributory.
amphenicol
From the Department of Dermatology, Stanford (Calif) University Medical Center. Reprint requests to the Department of Dermatology, Stanford University Medical Center, Stanford, CA 94305 (Dr Jacobs).
Paul H.
Jacobs, MD; Betty J. Russell, MS
Serum for direct agglutination titers submitted to the National Communi¬ cable Disease Center (NCDC) in Atlanta on Dec 5,1975. Results were as follows: Leishmania trópica, 1:256 (1:128, diagnostic); L donovani, 1:16 (1:32, diagnostic); and L braziliensis, 1:32 (1:64, diagnostic). A 24hour Montenegro skin test showed an erythema of 15 x 17 mm, a 48-hour test showed an erythema of 54 x 42 mm and an induration of 18 x 18 mm, and a 70-hour test showed an induration of 30 x 25 was
mm.
Because of the continued pain and of the patient's lesions, the decision was made to begin active treat¬ ment with antimony sodium gluconate (sodium stibogluconate, Pentostam, Great
enlargement
Britain). The generally accepted dosage for Pen¬ tostam is 0.1 ml/kg per injection, with a maximum of six ml (600 mg of antimony) per injection.1 The patient received 600 mg of Pentostam intramuscularly for ten days,
three ml in each buttock at a concentration of 100 mg/ml. The only side effect he experienced was a slight discomfort at the sites of injection. Values and results from the following laboratory tests, which were performed before and after treatment, were within normal limits: complete blood count and
differential; urinalysis; ECG; BUN; creatinine; total bilirubin; and a chest x-ray
film. Four days after treatment began, the ulcération on the elbow became more active, with increased erythema and crust¬ ing. Two days later this activity began to subside, and the lesions slowly healed. One month later the ulcération and crusting were gone, and the patient was left with a scar.
Solitary Nevus Lipomatosus Cutaneus Superficialis of the Knee Venkateshiah
The is is
Sathyanarayana, MD, Stanley Weitzner,
solitary nodular form of nevus lipomatosus cutaneus superficial-
cases
The site in the 21 in the literature was
uncommon.
reported
From the
Department of Pathology, Universi-
ty of Mississippi Medical Center, Jackson.
Reprints
not available.
MD
the upper thigh in eight cases,1-3 the buttock in seven cases,1-3 the lower back in two cases,1 and the axilla,1 arm,1 ear,4 and scalp5 in one case each. There is no apparent sex predilection for this condition. The age when the lesion was first noted was available for 16 patients, and it varied from 195
Downloaded From: http://archderm.jamanetwork.com/ by a University of Pittsburgh User on 06/03/2015
Comment Because of limited demand, Pento¬ stam has not been licensed in this country and must be restricted by federal law to investigational use only.1 The NCDC has obtained permis¬ sion from the Food and Drug Admin¬ istration to supply physicians with the drug for therapy of leishmaniasis. Reactions to pentavalent antimony compounds are not severe, as a rule. Antimony sodium gluconate, due in part to its rapid excretion, seems to be the least toxic of the commonly used
pentavalent antimony compounds. Side effects include gastrointestinal disturbance, jaundice, albuminuria, weakness, dermatitis, cough, pneumo¬ nia, and changes in the ECG.1 Most of the above experiences with
Pentostam are in the treatment of kala azar. Many of the previously described signs and symptoms of Pentostam therapy may occur in kala azar. Cahill reported no side effects in six cases of patients with kala azar treated with intravenous Pentostam.2 Pentostam has also been well toler¬ ated by children under two years of age.1 Our patient experienced no side effects other than transient pain at the site of injection. References 1. Informational Material for Physicians: Pentostam (Sodium Antimony Gluconate): Protocol
Atlanta, Center for Disease Control. 2. Cahill KM: Leishmaniasis in the Sudan Republic: Infection in American personnel. Am J Trop Med Hyg 13:794-799, 1964. #53.
including two cases that occurred in the fourth decade and four cases each that occurred in the third, fifth, and sixth decades.1,3 The clinical diagnosis was of a variety of benign lesions, most commonly nevocellular nevus and fibroepithelial papilloma. A case of solitary nevus lipomatosus cutaneus superficialis, which is, to our knowledge, the first case to be present since birth and on the knee, is to 641 years,
reported.
Report
of
a
Case
underwent excision of 19-year-old an asymptomatic nodule on the left knee that had been present since birth. The A
man
Urticaria
Pigmentosa in Identical
form is difficult. Wilson-Jones et al1
Twinslatter
agree that the
solitary form in the may represent a forme fruste of the typical plaque form.5 Although adipose tissue in the dermis may suggest several other lesions, a plentiful, normal appearance of the dermis, absence of nevus cells, and no central core of adipose tissue in our case exclude diagnoses of focal dermal hypoplasia, nevus pigmentosus et lipo¬ matosus, and skin tag, respectively. We believe that solitary nevus lipo¬ matosus cutaneus superficialis is probably more common than is sug¬ gested by the paucity of cases in the
Fig 1—Islands of mature adipose tissue, often vascularized, In upper and middermis; increase of subpapillary capillar¬ ies; and epidermis with crypt-like indenta¬ tion and focal, irregular, elongation of rete ridges (hematoxylin-eosin, x100).
lesion was soft to rubbery, skin-colored, and measured 15 x 6 x 6 mm. The results of the remainder of the physical examina¬ tion were not remarkable. Microscopically, the nodule showed scat¬ tered small islands of mature adipose tissue in normal-appearing dermal colla¬ gen. The adipose tissue was primarily in the upper half of the lesion and in places close to the papillary dermis. Small vascu¬ lar channels, primarily capillaries, were increased in the subpapillary dermis and collections of adipose tissue. The slightly acanthotic epidermis had an occasional crypt-like indentation and irregular elon¬ gation of some rete ridges (Fig 1 and 2). There was no continuity of dermal and subcutaneous adipose tissue. Nevus cells were absent. Collagen fibers and elastic tissue appeared to be normal with trichrome and elastic van Gieson stains. There was absence of metachromasia with toluidine blue stain, and there was no appreciable increase of ground substance with alcian blue stain.
Urticaria
areas
literature.
The
small
number of
reported cases may be owing to a lack of recognition and confusion of the condition with pedunculated lipofibroma.
Angel Arafiles, MD, provided the clinical permission to report the case.
data
and the
Fig 2.—Vascular Island of mature adipose tissue in superficial dermis and epidermis with irregular acanthosis and elongated rete ridge (hematoxylin-eosin, x 500). Comment tissue in the dermis distin¬
Adipose guishes nevus lipomatosus cutaneus superficialis from other nevoid anom¬ alies. The adipose tissue is considered to be derived from the dermal perivascular mesenchymal tissue. Solitary nevus lipomatosus cutaneus superfi¬ cialis may occur in any site, although to account for its predilection for the thigh and buttock in cases of the more common, typical, multinodular plaque
Pigmentosa in Identical
References 1. Wilson-Jones E, Marks R, Pongsehirun D: Nevus superficialis lipomatosus: A clinicopathological report of twenty cases. Br J Dermatol 93:121-133, 1975. 2. Nikolowski W: \l=U"\berNaevus lipomatodes cutaneus superficialis (Hoffmann-Zurhelle). Dermatol Wochenschr 122:735-741, 1950. 3. Kurihara Y, Yasyhara M, Minami H, et al: Nevus lipomatosus cutaneus superficialis (Hoffmann-Zurhelle). Skin Research 9:48-57, 1968. 4. Knoth W: \l=U"\berNaevus lipomatosus cutaneus superficialis Hoffmann-Zurhelle und \l=u"\ber Naevus naevocellularis partim lipomatodes. Dermatologica 125:161-173, 1962. 5. Weitzner S: Solitary nevus lipomatosus cutaneus superficialis of scalp. Arch Dermatol 97:540-542, 1968.
Twins
Alan S. Rockoff, MD
Urticaria neous
pigmentosa (UP), or cutamastocytosis, is the most
mast cell disease of childhood. It produces no visceral lesions or systemic symptoms and usually improves by adolescence.1 During the last 100 years, the literature has reported cases of various forms of mastocytosis in 40 families in which more than one member was affected.2 Among these cases were 11 common
Accepted for publication Oct 24, 1977. Department of Pediatrics, Mt Sinai Hospital, Hartford, Conn. Dr Rockoff is now with the Department of Dermatology, Boston University School of Medicine. Reprint requests to the Department of Dermatology, Boston University School of Medicine, 75 E Newton St, Boston, MA 02118. From the
one set of triplets. pairs of identical twins and the triplets were concordant for the disorder, while two identical twin pairs and
sets of twins and
Seven
the two fraternal sets were discordant. We report an eighth set of identical twins who both have UP, and we discuss the genetics of this condition.
Report of Cases Twins 1 and 2 were black, male infants who were seen at the age of 4 months. Their parents were not related, and there was no family history of skin disease. The delivering obstetrician recalled that there was one placenta, but the obstetrician had not noted whether the amniotic sac or chorion were single or double. Neither twin had vomiting, persistent
diarrhea, or flushing. At the age of 2 months, hyperpigmented lesions developed behind the right ear of twin 1. These later spread to cover his entire body, including scalp and soles. About a week after his rash began, an eruption of identical description and distribution developed on twin 2. Physical examination revealed welldeveloped infants, each with brown hair and eyes. Neither had lymphadenopathy or hepatosplenomegaly. Each had numerous reddish-brown, 2- to 4-mm papules on the trunk, extremities, palms, soles, and scalp. Several of these papules were surrounded by a pink halo, while the halo developed around others after being rubbed (positive Darier's sign). Punch biopsy specimens of back lesions
from each twin showed nests of cells in the upper dermis that were identified as mast cells by toluidine blue staining.
Downloaded From: http://archderm.jamanetwork.com/ by a University of Pittsburgh User on 06/03/2015
