Journal of the
American Academy of
688 Correspondence
Fi~'. 1. Note diminution of lesions on sun-exposed skin,
maculopapular eruption, most prominently observed on the
trunk, axilla, and upper extremities, and a relative diminution
of these lesions in the sun-exposed skin. In the sun-exposed
areas there were decreases in the size, number, ancl pigmen-
tation of the individual urticaria pigmentosa lesions. Labo-
ratory and radiologic studies failed to detect any evidence of
systemic involvement by mast cells. Skin biopsies from in-
volved and uninvolved skin revealed histopathologic changes
consistent with urticaria pigmentosa. Mast cell counts, uti-
lizing toluidine blue stains, were six to twelve and thirty to
fifty cells per high-power field in uninvolved and involved
skin, respectively.
Because UV radiation affects the mast cell popula-
tion, psoralens plus ultraviolet A (PUVA) was tried and
found to be all effective treatment modality for urticaria
pigmentosa.I-6 PUVA not only decreased the clinical
symptoms, such as itching and urticaria, but also de-
creased urinary histamine and histamine metabolite ex-
cretion, as well as the number of cutaneous mast cells.
Vellabriffa et aP noted that after PUVA two patients
had almost complete fading of the macules. They also
noted that there was a regional, site-dependent variation
in the PUVA response; i.e., shaded lesions on the inside
of the legs resolved more slowly.
The findings in our patient parallel those observed
in urticaria pigmentosa patients treated with PUVA,
i.e., diminution of number, size, and pigmentation of
lesions in UV-exposed sites. However, this phenome-
non in our patient occurred in response to natural sun-
light alone. Additionally, his decrease in symptoms,
such as itching, paralleled his sunlight exposure during
the summer. We found no other reports of urticaria
pigmentosa having a similar distribution or of natural
sunlight having a beneficial effect on urticaria pigmen-
tosa. Our patient may be unique to the literature, but
we believe the phenomenon is not uncommon. Grad-
ually increasing the exposure of patients with urticaria
Dermatology
pigmentosa to natural sunlight with concomitant anti-
histamine therapy should be considered as an inexpen-
sive alternative treatment for patients with urticaria pig-
mentosa.
Gordon Vire, MD., L. Jackson Roberts, M.D.,
and Lloyd E. King, Jr., M.D., Ph.D.
Division of Dermatology, Department of Medicine
Vanderbilt University Medical School and
Veterans Administration Medical Center
Nashville, TN 37212
REFERENCES
1. Granerus G, Roupe G, Swanbeck G: Decreased urinary
metabolites after sucessful PUVA treatment of urticaria
pigmentosa. J Invest Dermatol 76: 1-3, 198 J.
2. Christophel's E, I-Ionigsmann 1-1, Wolff W, Langner A:
PUVA treatment of urticaria pigmentosa. Br J Dermatol
98:701-702, 1978.
3. Vellabriffa D, Eady RAJ, James MP, et al: Photochemo-
therapy in the treatment of urticaria pigmentosa. HI' J Der-
matoll09:67-75, 1983.
4. Kolde G, Frosch PJ, Czarnetzki BM: Response of cuta-
neous mast cells to PUVA in patients with urticaria pig-
mentosa: I-listomorphometric, ultrastructural, and bio-
chemical investigations. J Invest Dermatol 83: 175-178,
1984.
5. Valtonen EJ: The effect of ultraviolet radiation of some
spectral wavebands on the mast cell count in the skin. An
experimental study on mice. Acta Pathol Microbiol Scand
(Suppl) 15:99, 1961.
6. Eady RA, Cowen T, Marshall TF, et al: Mast cell popu-
lation density, blood vessel density and histamine content
in normal skin. Br J Dermatol 100:623-633, 1979.
Acquired cutaneous Iymphangiectasias
secondary to scarring from scrofuloderma
To the Editor:
An unusual case of acquired lymphangiectasias that
clinically simulated herpes zoster is presented. To our
knowledge, this is the first report of acquired lymphatic
dilatation resulting from dermal and subcutaneous scar-
ring caused by past involvement with scrofuloderma.
A cutaneous lymphangioma consists of dilated lym-
phatic channels that may be found in the superficial
dermis and may extend into the subcutaneous fat to the
level of the deep fascia. Cutaneous lymphangiomas rep-
resent rare skin lesions, the majority of which are of
congenital origin. Occasionally, they arise from ob-
struction of lymphatic flow from an extrinsic cause, and
thus they are more appropriately termed lymphangiec-
tasias. I -4 One such case is reported.
Case report. A 68-year-old black woman presented to one
of us (R. A. J.) with a long-term history of translucent thick-
walled "vesicles," up to 5 mm in diameter, arranged in a
Volume 14
Number 4 Correspondence 689
April, 1986

linear pattern on the right lateral aspect of the trunk (Fig. 1).
The lesions were not present at birth and reportedly waxed
and waned in size. There was no focal swelling or pain.
Tzanck smear was negative for multinucleated epithelial giant
cells. The remainder of the physical examination was unre-
markable.
Past medical history was significant for tuberculous lymph-
adenitis involving the right axillary nodes, which resulted in
scrofuloderma of the overlying skin from contiguous spread.
This condition resolved without treatment.
Biopsy of one of the lesions showed dilated endothelial-
lined spaces in the superficial dermis. These contained lymph
and scattered lymphocytes (Fig. 2). The patient did not desire
treatment.
Discussion. Acquired lymphangioma or lymphan-
giectasia is the terminology ascribed to lesions that clin- Fig. 1. Clinical photograph showing axillary scarring
ically consist of one or multiple groups of translucent and linear vesicles on the superolateral aspect of the
"vesicles," which represent acquired lymphatic dila- chest wall.
tations secondary to an identifiable external cause. IHis-
tologically, dilated lymphatic channels are present in
the superficial and mid dermis, but only one, or oc-
casionally two, dilated lymphatics are seen in the deep
dermis. 5 The overlying epidermis can display varying
degrees of hyperkeratosis, acanthosis, and papilloma-
tosis, and it may appear to enclose the ectatic lymphatic
channels. 5 These may contain scattered lymphocytes
and variable numbers of red cells, imparting a purplish
tinge to the lesions. Lymphangiectasias have been de-
scribed in postmastectomy lymphedema4 and secondary
to scarring from surgical procedures of various kinds. 6•7
They have also been reported in association with anom-
alous deep lymphatics, 8 radiation therapy, 1 cutaneous
trauma/' w recurrent infections, * and even sclero-
derma. 2 Fig. 2. Low-power photomicrograph showing dilated
To our knowledge, this is the first reported case of endothelial-lined lymphatic spaces containing lymph
lymphangiectasia secondary to scarring from scrofu- and lymphocytes covered by epidermis. (Hematoxylin-
loderma, although lesions on the vulva have been de- eosin stain; XSO.)
scribed in association with tuberculosis. 3 These oc-
curred after repeated therapeutic incisions aimed at eral authors.I.II-13 They are generally associated with
draining suppurative tuberculous inguinal lymph nodes. more extensive involvement of the deep dermis and
This case was also associated with considerable lymph- subcutis5 and have a significantly higher recurrence rate
edema. In our patient, the scarring was more superficial after treatment by simple excision. II
and thus did not significantly involve main lymphatic In summation, lymphangiectasias represent a rare
channels, as evidenced by the absence of limb swelling cutaneous condition that results from lymphatic ob-
clinically. struction caused by a spectrum of scarring processes.
Cutaneous lymphangiomas have been classified ac- The presence of associated lymphedema is variable.
cording to distinctive clinical and histopathologic fea- Our case is also remarkable in that the linearly arranged
tures. 5 Lymphangioma circumscriptum (localized and vesicles simulated herpes zoster clinically.
classical types) and cavernous lymphangioma are
Mario Di Leonardo, B.A., and
thought to be congenitally derived hamartomas by sev-
Richard A. Jacoby, M.D.
Hahnemann University School of Medicine
*Jadassohn J: Handbuch der haul-und geschlechlskrankheiten. III. Division of Dermatology
2:352-366, 1969. Broad & Vine Streets, Philadelphia, PA 19102

690 Correspondence
REFERENCES
I. Fisher I, Orkin M: Acquired lymphangioma (lymphan-
giectasias). Report of a case. Arch Dermatol 101:230-
234, 1970.
2. Tufanelli DL: Lymphangiectasis due to scleroderma.
Arch Dermatol 3:1216, 1975.
3. Heuvel NVD, Stolz E, Notowicz A: Lymphangiectasias
of the vulva in a patient with lymph node tuberculosis.
Int J DeImatol 18:65-66, 1979.
4. Plotnick H, Richfield D: Tuberous lymphangiectatic var-
ices secondary to radical mastectomy. Arch Derl11atol
74:466-468, 1956.
5. Lever WF, Shaumburg-Levcr G: Histopathology of the
skin, ed6. Philadelphia, 1983, J. B. Lippincott Co., pp.
631-633.
6. Russell B: Lymphangioma circumscriptum and keloids.
Bd Dermatol 63:158-159, 1951.
7. Weakley DR, Juhlin EA: Lymphangiectases and lymph-
angiomata. Arch Dcrmatol Syph 64:574-578, 1961.
8. Palmer LC, Strauch WG, Welton WA: Lymphangioma
circumscriptum. A case with deep lymphatic involve-
ment. Arch Dermatol 114:394-396, J 978.
9. Zufall R: Lymphangiectasis of penis. Urology 19:53,
1982.
10. Caro W: Tumors of the skin, in Moschella S, Pillsbury
DM, Hurley HJ: Dermatology, cd, I, Philadelphia, 1975,
W, B. Saunders Co., pp. l389-1391.
II, Peachey RDG, Lim CC, Whimster rw: Lymphangioma
of the skin. A review of 65 cases. Br J Dcrmatol 83:519-
527, 1970,
12. Godart S: Embryological significance of lymphangioma.
Arch Dis Child 41:204-206,1966.
13. Whimstcr IW: The pathology of lymphangioma circum-
scriptum. Br J Dermatol 94:473-486, 1976,
Flagellate pigmentation from bleomycin
To the Editor:
We were very interested in the paper by Fernandez-
Obregon et al entitled "Flagellate Pigmentation From
Intrapleural Bleomycin, a Light Microscopy and Elec-
tron MicroScopy Study" (J AM ACAD DERMATOL
13:464-468, 1985). The authors report a case of flag-
ellate pigmentation following an intrapleural injection
of 30 mg of bleomycin, the lowest dose reported here-
tofore to produce this particular side effect. We have
recently observed flagellate pigmentation in a 51-year-
old patient who had a unique intravenous injection of
15 mg of bleomycin for diagnostic purposes. lOur pa-
tient presented with an acute striated urticarial reaction
24 hours after the injection. The inflammatory com-
ponent of the striae disappeared spontaneously, but flag-
ellate pigmentation appeared and persisted for over a
month. The biopsy specimen taken 48 hours after the
injection showed pigmentary incontinence as well as
intense edema and perivascular infiltration of mono-
nuclear and polymorphonuclear cells.
Journal of the
American Academy of
Dermatology
Besides its well-known therapeutic effects, bleo-
mycin is also used for diagnostic scaiming, in particular
in the setting of lung cancer. 2 .* This is the only case of
flagellate pigmentation we observed in our institution
over 18 months while 300 patients had diagnostic scan-
nings.
Our case further emphasizes Fernandez-Obregon
et al' s conclusions that bleomycin may produce flag-
ellate pigmentation even at very low doses. Bleomycin
is inactivated by an enzyme, hydrolase, in every organ
except lung and skin,3 which may be related to the
frequency of pulmonary and dermatologic side effects.
B. S. Polla, M.D.,** Y. Merot, M.D.,
J. H. Saunzt, M.D., and D. Slosman, MD.
RespiratOlY Division, Clinique de Dermatologie
Hopital Cantonal Universitaire, Genevcl, Switzerland,
and Brookhaven National Laboratory, Long Island,
Upton, NY IJ973
*Slosman D, Polla B, Egeli R, et al: Cobalt" bleomycin scanning
for lung cancer detection: A prospective study in thoracic surgery.
Nuclear Medicine Communications 6:235-244, 1985.
**Present address: Arthritis Unit, Massachusetts General Hospital,
Boston, MA 02114.
REFERENCES
1. Palla L, Merot Y, Siosman D, et al: Dcrmite lineairc et
pigmentogene aprcs scintigraphic a la bleomycine. Ann
Dermatol Venereal. (In press.)
2. Nieweg DE, Beekhuis H, Piers DA, et al: "Co-bleomycin
and b7Ga-citrate in detecting and staging lung cancer. Tho-
rax 38:16-21, 1983.
3. Dantzig PI: Immunosuppressive and cytotoxic drugs in
dermatology. Arch DermatolllO:393-406, 1974.
Rules for reprint requests
To the Editor:
I wish to suggest that new rules of etiquette are in
order for those who request reprints of journal articles.
Since we live in an age in which almost everyone has
ready access to photocopying equipment, the provision
of reprints is a courtesy that borders on a luxury. Those
who send reprints are really providing those who re-
quest them with glossy high-quality copies that contain
no more information than an ordinary photocopy would.
Individuals who receive copies of articles are prob-
ably being spared the inconvenience of a trip to a med-
ical library with its attendant search for a particular
article of interest. They are also able to avoid the ritual
of standing before a copying machine that mayor may
not be coin-activated. In any case, those who send
reprints to their colleagues are providing a courte~y at