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18. Topliss DJ, White EL, Stockigt JR. Significance of thyrotropin excess
in untreated primary adrenal insufficiency. J Clin Endocrinol Metab
1980;50:52-6.
19. Barnett AH, Donald RA, Espiner EA. High concentrations of
thyroid stimulating hormone in untreated glucocorticoid deficiency:
Indication of primary hypothyroidism? Br Med J (Clin Res Ed)
1982;285:172-3.
20. Chopra IJ, Williams DE, Orgiazzi J, Solomon DH. Opposite effects of
dexamethasone on serum concentrations of 3,3,5- triiodothyronine
(reverse T3) and 3,5,3-triiodothyronine (T3). J Clin Endocrinol
Metab 1975;41:911.
21. Drucker D, Shumak S, Angel A. Schmidts syndrome presenting with
intrauterine growth retardation and postpartum Addisonian crisis.
Am J Obstet Gynecol 1984;149:229.
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Figure 1: Hypertrichosis

DOI:
10.4103/2230-8210.95747

A tale of nonhormonal hairs


Sir,
Porhphyria cutanea tarda (PCT) is a hepatic porphyria
in which the activity of the heme synthetic enzyme
uroporphyrinogen decarboxylase is deficient. It may be
sporadic (80%) or familial (20%). Hypertrichosis can
occur in PCT without the classical skin manifestations of
blistering and thickening of the skin, making the diagnosis
difficult.[1]
A 35-year-old lady presented with history of excessive
growth of hair over the face and the hands for the past
10 years [Figure 1]. She was evaluated multiple times for
the hormonal status and which was always normal. On
detailed history, she gave history of itching and burning
sensation of sun-exposed areas on exposure to sunlight.
Physical examination showed thick terminal hair over
the face and the forearms [Figure 2] and below the knee
[Figure 3]. Hairs over the chest, abdomen, lower back, and
pubic area were normal. She also had thickening of the skin
over the fingers, terminal onycholysis, and absorption of
the digits [Figures 4 and 5]. Based on the clinical history
and physical findings, the diagnosis of porphyria was
thought of and under ultraviolet light, acidified urine
showed coral pink fluorescence of uroporphyrins. In
view of the age of onset, absence of family history, and
elevated uroporphyrins, a final diagnosis of PCT type
1 was made. The patient was managed with therapeutic
phlebotomies and low-dose hydroxychloroquine and had
a 75% improvement in symptoms after 1 year.

Figure 2: Hypertrichosis over sun-exposed area

Figure 3: Hypertrichosis over the leg

Porphyrias are due to altered activity of specific enzymes


of the heme biosynthetic pathway. Out of the porphyrias,
X-linked protoporphyria, congenital erythropoietic

Indian Journal of Endocrinology and Metabolism / May-Jun 2012 / Vol 16 | Issue 3

483

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Letters to the Editor

Figure 4: Blisters over the skin, reabsorption of fingers, and


pseudoscleroderma

Figure 5: Cicatricial conjunctivitis

porphyria, PCT, hepatoerythropoietic porphyria,


hereditary co-proporphyria, variegate porphyria (VP), and
erythropoietic protoporphyria have skin manifestations,
which is due to the accumulation of photoactive porphyrins
in the skin.

(125 mg) twice weekly is usually effective when repeated


phlebotomies are contraindicated.[1]

Cutaneous photosensitivity with vesicles and bullae are


common. Thickening, scarring, and calcification of skin
and reabsorption of terminal parts of the digits happens,
which resembles sclerodermathe pseudoscleroderma
appearance.[1,2]

PCT should be considered as a diagnostic possibility in


patients undergoing evaluation for excessive hair growth.
The typical location of hypertrichosis is the sun-exposed
areas, sparing other androgen-dependent areas, other
features, such as photosensitivity, skin blisters, cola-colored
urine, and examination of urine under ultra violet light
helps to differentiate this condition from other endocrine
causes of excessive hair growth.
Rajeev Philip, Prem P. Patidar,
Praveen Ramachandra, Keshav K. Gupta

Excessive hair growth in porphyrias predominantly


occurs over the sun-exposed areas sparing the other
androgen-dependent areas, offering a diagnostic clue.
The hypertrichosis can sometimes be so extreme that the
affected patients are called monkeys children.[3]
PCT is the most common porphyrias and presents usually
after puberty with skin lesions.
The precipitating factors for this disease includes alcohol
abuse, smoking, estrogen, hepatitis C, and hemochromatosis
gene (HFE) mutations.[1]
Diagnosis: Plasma porphyrins are increased in patients
with porphyrias causing blistering skin lesions. The
fluorescence spectrum of plasma and urine can distinguish,
erythropoietic protoporphyria, and PCT.[1] A predominance
of uroporphyrin and heptacarboxyl porphyrin in urine,
producing coral pink fluorescence is diagnostic of PCT.
Treatment: Phlebotomies to reduce ferritin levels is the
treatment of choice.[4] Deferoxamine, an iron chelator,[5]
or low-dose hydroxychloroquine (100 mg) or chloroquine
484

Department of Endocrinology, LLRM Medical College Campus,


Meerut, Uttar Pradesh, India
Corresponding Author: Dr. Rajeev Philip,
G10, PG Hostel, LLRM Medical College Campus, Garh Road,
Meerut, Uttar Pradesh 250 004, India.
E-mail: endollrm@yahoo.com

References
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5.

Anderson KE. The porphyrias. In: Lee G, Andrew IS, editors.


Goldmans Cecil Medicine. 24th ed. Philadelphia: Saunders, Elsevier;
2012. p. 1367-9.
Grossman ME, Bickers DR, Poh-Fitzpatrick MB, Deleo VA, Harber
LC. Porphyria cutanea tarda. Clinical features and laboratory findings
in 40 patients. Am J Med 1979;67:277-86.
Peters HA, Gocmen A, Cripps DJ, Bryan GT, Dogramaci I.
Epidemiology of hexachlorobenzene-induced porphyria in Turkey:
Clinical and laboratory follow-up after 25 years. Arch Neurol
1982;39:744-9.
Lundvall O. Phlebotomy treatment of porphyria cutanea tarda. Acta
Derm Venereol Suppl (Stockh) 1982;100:107-18.
Rocchi E, Gibertini P, Cassanelli M, Pietrangelo A, Borghi A,
Pantaleoni M, et al. Iron removal therapy in porphyria cutanea tarda:
Phlebotomy versus slow subcutaneous desferrioxamine infusion. Br
J Dermatol 1986;114:621-9.

Indian Journal of Endocrinology and Metabolism / May-Jun 2012 / Vol 16 | Issue 3

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Letters to the Editor


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DOI:
10.4103/2230-8210.95754

Hyperandrogenemia due to
ingestion of Butea superba
Sir,
Androgen or testosterone is an important masculine
hormone. It helps construct the male appearance.
Hyperandrogenemia is majorly described in of females
with hirsutism,[1] insulin resistance[2] and polycystic ovarian
syndrome.[3] However, hyperandrogenemia in males is not
frequently mentioned. In this short article, the authors
report a case of hyperandrogenemia due to ingestion
of Butea superba, a herb found in South East Asia. This
is an interesting case of hyperandrogenemia induced by
an external source. The effect of phytoandrogens is also
discussed.
The patient was a Thai single male, aged 35 years, without
any underlying disease (his basic annual laboratory
checkup showed normal results). On presentation,
the chief complaint of this patient was a feeling of
increased sexual drive. He gave the history of no use of
narcotic and regular intake of vitamin and nutritional
supplementation. Physical examination revealed no
significant abnormality. Laboratory investigations were
performed which showed increased dihydrotestosterone
(1512 pg/mL, reference value 250990 pg/mL). The results
of other sexual hormone related investigations in this case
included dehydroepiandrosterone sulfate 328 g/dL, free
testosterone 1.7% and sex hormone binding globulin 43.24
nmol/L [no data on follicle stimulating hormone (FSH)
and luteinizing hormone (LH) levels]. The diagnosis of
hyperandrogenemia in this case was therefore arrived at.
Further investigation to find the source of androgen in
this case was performed. With complete history taking, the
clinical nutritionist could define an important problematic
food component, B. superba, a local herb. The patient gave
additional information that he had just taken this locally
made capsule of this herb for a few weeks because he was
suffering from hair loss. This patient was advised to stop
ingestion of this herb, and follow-up after 1 week revealed
that the patient had no feeling of increased sexual drive
and dihydrotestosterone had decreased to normal level.

In general, androgen plays an important role in the sexual


drive of males. Decreased androgen level is strongly related
to reduced sexual activity and decreased sexual drive.[4]
This is a common problem in old males[4] and in cases
with erectile dysfunction.[5] In males with the problem of
sexual desire, androgen investigation is a useful test.[6] In
this case report, the patient also complained of increased
sexual desire, which hmight have been due to exogenous
hyperandrogenemeia.. Similar to hyperestrogenemia caused
due to phytoestrogens, some herbal regimens might contain
phytoandrogens that can lead to hyperandrogenemia. In
this case, B. superba is the problematic external source
of excessive androgen. This plant is considered to be a
male potency herb. In animal models receiving this herb,
stimulation of sexual organ has been reported.[7,8] In case
of human beings, there is only one previous trial using this
herb for treating erectile dysfunction.[9] The effect of this
herb is comparable to that of sildenafil.[9] However, there
has never been any report on this herb in healthy males.
This is the first case report on hyperandrogenemia due to
ingestion of B. superba. With the widespread usage of local
herbs presently, the effects of herbs need to be considered.
Indeed, a previous report also mentioned genotoxicity due
to large dosage ingestion of B. superba.[10]
Kamon Chaiyasit, Viroj Wiwnaitkit1
Mahidol Nutrition Society, Mahidol University, Thailand, 1Hainan
Medical University, China; Joseph AyoBabalola, Nigeria
Corresponding Author: Dr. Kamon Chaiyasit,
Mahidol Nutrition Society, Mahidol University, Thailand.
E-mail: kamon.chaiyasit@gmail.com

References
1.
2.

3.

4.

5.

6.

7.

8.

Maroulis GB. Evaluation of hirsutism and hyperandrogenemia. Fertil


Steril 1981;36:273-305.
Barbieri RL, Ryan KJ. Hyperandrogenism, insulin resistance,
and acanthosis nigricans syndrome: A common endocrinopathy
with distinct pathophysiologic features. Am J Obstet Gynecol
1983;147:90-101.
Goodarzi MO, Dumesic DA, Chazenbalk G, Azziz R. Polycystic ovary
syndrome: Etiology, pathogenesis and diagnosis. Nat Rev Endocrinol
2011;7:219-31.
Davidson JM, Chen JJ, Crapo L, Gray GD, Greenleaf WJ, Catania
JA. Hormonal changes and sexual function in aging men. J Clin
Endocrinol Metab 1983;57:71-7.
Guay A, Jacobson J. The relationship between testosterone levels,
the metabolic syndrome (by two criteria), and insulin resistance in
a population of men with organic erectile dysfunction. J Sex Med
2007;4:1046-55.
Corona G, Petrone L, Mannucci E, Ricca V, Balercia G, Giommi
R, et al. The impotent couple: Low desire. Int J Androl 2005;28
Suppl 2:46-52.
Malaivijitnond S, Ketsuwan A, Watanabe G, Taya K, Cherdshewasart
W. Luteinizing hormone reduction by the male potency herb, Butea
superba Roxb. Braz J Med Biol Res 2010;43:843-52.
Malaivijitnond S, Ketsuwan AN, Watanabe G, Taya K, Cherdshewasart
W. Androgenic activity of the Thai traditional male potency

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