10.5826/dpc.1303a123
text/xml
2160-9381
2802
en
Sudheer Kumar Arava
https://creativecommons.org/licenses/by-nc/4.0
Dermatology Practical & Conceptual
doxycycline
Saxophone Penis Secondary to Lichen Simplex Chronicus Responding to Low Dose of Methotrexate and Doxycycline
Text.Serial.Journal
https://dpcj.org/index.php/dpc
Research Letter
2023-07-31
2022-09-01
2023-07-31
2023-08-24
e2023123
Dermatol Pract Concept
Sudheer Kumar Arava
2023/07/31
10.5826/dpc.1303a123
e2023123
Department of Pathology, All India Institute of Medical Sciences, New Delhi, India
2160-9381
doxycycline
en
e2023123
Ju T, Vander Does A, Mohsin N, Yosipovitch G. Lichen Simplex Chronicus Itch: An Update. Acta Derm Venereol. 2022;102:adv00796. DOI: 10.2340/actadv.v102.4367. PMID: 36250769. PMCID: PMC9677261.
Saxophone Penis Secondary to Lichen Simplex Chronicus Responding to Low Dose of Methotrexate and Doxycycline
https://dpcj.org/index.php/dpc/article/download/2802/2250
                 Saxophone Penis Secondary to Lichen
                 Simplex Chronicus Responding to Low
                 Dose of Methotrexate and Doxycycline
              Bhavya Swarnkar1, Somesh Gupta1, Neetu Bhari1, Sudheer Kumar Arava2


1 Departments of Dermatology and Venereology, All India Institute of Medical Sciences, New Delhi, India
2 Department of Pathology, All India Institute of Medical Sciences, New Delhi, India



Key words: Saxophone penis, lichen simplex chronicus, methotrexate, doxycycline
Citation: Swarnkar B, Gupta S, Bhari N, Kumar Arava S. Saxophone penis secondary to lichen simplex chronicus responding to low dose
methotrexate and doxycycline. Dermatol Pract Concept. 2023;13(3):e2023123. DOI: https://doi.org/10.5826/dpc.1303a123
Accepted: December 19, 2022; Published: July 2023
Copyright: ©2023 Swarnkar et al. This is an open-access article distributed under the terms of the Creative Commons Attribution-
NonCommercial License (BY-NC-4.0), https://creativecommons.org/licenses/by-nc/4.0/, which permits unrestricted noncommercial use,
distribution, and reproduction in any medium, provided the original authors and source are credited.
Funding: None.
Competing interests: None.
Authorship: All authors have contributed significantly to this publication.
Corresponding author: Dr Somesh Gupta, Department of Dermatology, All India Institute of Medical Sciences,
Ansari Nagar East, New Delhi-110029 Phone number: 9868899120 E-mail: someshgupta@hotmail.com




Introduction                                                                  He had bilateral inguinal hernia for the past 6-7 years.
                                                                              Skin biopsy showed psoriasiform epidermal hyperplasia
Our case is interesting because of a rare presentation of ano-           with parakeratosis and focal presence of a granular layer.
genital lichen simplex chronicus (LSC) as a saxophone penis              There was perivascular and peri-eccrine infiltrate with some
and a good response to anti-inflammatory agents like doxy-               interstitial spill of lymphocytes, histiocytes, plasma cells, and
cycline and methotrexate.                                                eosinophils. Sclerosis of collagen was noted in the dermis.
                                                                         Overall, features were suggestive of sclerosing mixed inflam-
Case Presentation                                                        matory tissue reaction (Figure 1 B and C).
                                                                              Peripheral smear for microfilaria, Mantoux test, PCR for
A man in his 60s presented with complaints of severe itching             chlamydia (urethral and rectal), Venereal Disease Research
over genitalia and scrotal skin thickening (lichenification)             Laboratory (VDRL) test, and stool for occult blood were
associated with overlying depigmented macules for the past               negative.
5 years. He also complained of deformity of the penis asso-                   He was diagnosed as a case of saxophone penis and
ciated with deviation in urine stream during micturition for             scrotal thickening secondary to LSC and was advised to
the past 3 months (Figure 1A).                                           avoid scratching and treated with scrotal support, low dose
    He had been diagnosed with LCS, vitiligo, and                        methotrexate 7.5 mg/week (0.1 mg/kg/week), doxycycline
non-specific pruritus by different physicians and had taken              100 mg once daily along with mid potency topical steroid
intermittent oral antihistamines and topical steroid and anti-           once daily, emollients, and antihistamines. Methotrexate
fungals in the past with minimal improvement.                            and doxycycline were added because of the severity of the



Research Letter | Dermatol Pract Concept. 2023;13(3):e2023123                                                                            1
disease (architectural distortion), their proven efficacy, po-         some are non-infective like congenital hereditary lymphoe-
tent anti-inflammatory action, good safety, and cost-effective         dema, malignant infiltration of lymph nodes, LSC [3].
profile. The patient was referred to the surgery department                Kumaran et al gave the hypothesis of the pathogenesis
for the management of inguinal hernia.                                 of the saxophone penis by mentioning that long-standing
    There was a significant improvement in the deformity               inflammation leads to fibrosis of lymphatics, which run
of the penis, thickening of the scrotal skin, deviated urinary         along the dorsal penile vein as well as surrounding connec-
stream, and itching after 3 months of therapy (Figure 1D).             tive tissue. Due to poor blood supply of the dorsal aspect of
                                                                       the penis, contraction of that tissue occurs leading to dorsal

Discussion and Conclusions                                             bending of the penis. Corpora cavernosa is attached to ischial
                                                                       tuberosity but bulbospongiosus and glans penis don’t have
Complications of genital LSC are clitoral or labial hyper-             such attachment leading to their diversion upwards. The
trophy, fibrosis, and scarring that can occur due to vigorous          ventral part (vascular part) hence becomes a dependent part
scratching leading to architectural distortion [1].                    leading to excessive fluid extravasation leading to edema of
    The inguinal hernia has been hypothesized to cause LSC             ventral prepuce which further pushes the glans upwards and
by inducing itching due to the occlusive effect [2]. This might        outwards, accentuating the dorsal curvature, forming a sax-
be one of the triggers for the development of LSC in our case.         ophone penis [4]. This hypothesis holds true in our case as
    There are various other causes of saxophone penis. Some            well, as chronic inflammation due to LSC might have led to
of them are infective like lymphogranuloma venereum (LGV),             the fibrosis of lymphatics and surrounding connective tissue
chlamydia trachomatis infection (non-LGV), filariasis and              (shown in the biopsy image) leading to a saxophone penis.




    Figure 1. (A) Thickened deformed (saxophone) penis and scrotal thickening with depigmented macules. (B) Psoriasiform hyper-
    plasia of the epidermis with parakeratosis and focal presence of a granular layer (H&E:40X). (C) Interstitial infiltrate of lympho-
    histiocytes, plasma cells, and eosinophils. Sclerosis of collagen was noted in the mid and deep dermis (H&E: 100x). (D) Significant
    improvement with correction of deformity of penis and decrease in scrotal thickening after 3 months of treatment.



2                                                                           Research Letter | Dermatol Pract Concept. 2023;13(3):e2023123
   Treatment includes treatment of suspected trigger like             Recommended treatment options for severe cases include
hernia, cutting off the itch-scratch-itch cycle, emollients,       nemolizumab, dupilumab, Janus kinase inhibitors like to-
antihistamines, anti-inflammatory agents like doxycycline,         facitinib, focused ultrasound, and transcutaneous electrical
methotrexate, topical and oral corticosteroids etc [5].            nerve stimulation [6].
   Moisturizers containing polidocanol have been found                We should be aware of such a complication of LSC and
to reduce non-histaminergic itch in placebo-controlled             its management to prevent unnecessary apprehension about
double-blind trials. Humectants like urea, lactic acid are not     genital malignancy.
recommended due to their irritant potential. Short-term high
potency topical corticosteroids are considered first-line ther-
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Research Letter | Dermatol Pract Concept. 2023;13(3):e2023123                                                                        3
