Recurrent Abscesses of The Neck: Scrofuloderma: Bacterium Tuberculosis PCR Were Negative. A Culture Was Not

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Letters

1. McKenzie WS, Rosenberg M. Iatrogenic subcutaneous emphysema of dental


and surgical origin: a literature review. J Oral Maxillofac Surg. 2009;67(6): Figure 1. Clinical Images of the Patient
1265-1268.
A
2. Romeo U, Galanakis A, Lerario F, Daniele GM, Tenore G, Palaia G.
Subcutaneous emphysema during third molar surgery: a case report. Braz Dent
J. 2011;22(1):83-86.
3. Arai I, Aoki T, Yamazaki H, Ota Y, Kaneko A. Pneumomediastinum and
subcutaneous emphysema after dental extraction detected incidentally by
regular medical checkup: a case report. Oral Surg Oral Med Oral Pathol Oral
Radiol Endod. 2009;107(4):e33-e38.
4. Sujeet K, Shankar S. Images in clinical medicine: prevertebral emphysema
after a dental procedure. N Engl J Med. 2007;356(2):173.
5. Hsu HL, Chang CC, Liu KL. Subcutaneous emphysema after dental
procedure. QJM. 2011;104(6):545.

Recurrent Abscesses of the Neck:


Scrofuloderma
Tuberculosis of the skin has become a rare disease in indus-
trialized countries. Polymerase chain reaction (PCR) is a pow-
erful diagnostic tool for mycobacterial infections of the skin,
but it can fail, as demonstrated in this case.

B
Report of a Case | A woman in her 80s was referred for surgical
treatment of a cervical abscess. Similar abscesses erupted in
the cervical region over the course of 2 years (Figure 1A).
A needle aspiration biopsy was performed on a node at the
left side of the neck, which measured 2 cm. The histopatho-
logic report described a minor nonspecific inflammatory
reaction, not suggestive of infection. Findings of the Myco-
bacterium tuberculosis PCR were negative. A culture was not
performed. Two months later, the whole nodule was
excised, including the adjacent inflamed skin. The resulting
defect, with a diameter of 7 cm, was closed with a rotary-
transposition flap. During this intervention, the thoracic
nerve was injured resulting in an elevation palsy of the left
arm. The histopathologic report of the excised tissue again
showed a nonspecific inflammatory reaction; no microbio-
logical analysis was conducted. A, Multiple cervical abscesses recurred in the 2 years prior to presenting for
care. B, A cold abscess at the neck was opened and drained to gain a specimen
At presentation, the patient had puckered scars scattered for histologic and microbiological analysis; multiple puckered scars are visible on
over the neck in addition to an unusual “cold abscess” the neck.
(Figure 1B). The clinical appearance was suggestive of scrofu-
loderma. Results of the Mendel-Mantoux test were positive (di-
ameter, 20 mm), as were those from the interferon-γ release picin, 600 mg/d, was initiated. After 2 months, the regimen was
assay. However, PCR findings from the skin biopsy specimen reduced to isoniazid and rifampicin. After 4 months of the re-
and abscess material were negative for M tuberculosis. Histo- duced regimen, all skin lesions had healed completely, leav-
logically, no acid-fast bacilli could be detected by Ziehl- ing scars, and sonography revealed no remaining abscesses.
Neelsen staining. Treatment was well tolerated, and at 24-month follow-up, no
Cervical sonography and magnetic resonance tomogra- new nodules had evolved.
phy revealed multiple abscesses in the lateral muscle loge.
Chest radiography excluded pulmonary tuberculosis. Labo- Discussion | From 1% to 2% of tuberculosis cases are cutane-
ratory work showed an elevated level of C-reactive protein (115 ous tuberculosis (CTB).1 Tuberculosis cutis colliquativa, also
mg/L; normal, <5 mg/L) but no other pathological findings. (To known as scrofuloderma, is the most common CTB subtype
convert C-reactive protein to nanomoles per liter, multiply by in Europe.2 Scrofuloderma is a subcutaneous form of CTB
9.524.) manifesting with cold abscesses most commonly on the
After 19 days, M tuberculosis was cultivated from the skin neck that spreads from underlying lymph nodes. Infection
specimen (Figure 2). The strain was sensitive to isoniazid, rif- can also involve joints, bones, and epididymis.3 The same
ampicin, pyrazinamide, ethambutol, and streptomycin. quadruple antibiotic therapy is used as in pulmonary tuber-
Classic quadruple treatment with isoniazid, 300 mg/d; culosis. Before treatment is begun, possible multidrug resis-
pyrazinamide, 1500 mg/d; ethambutol, 1200 mg/d; and rifam- tance should be excluded.

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Letters

4. Degitz K. Detection of mycobacterial DNA in the skin. Etiologic insights and


Figure 2. Growth of Mycobacterium tuberculosis in Liquid Culture diagnostic perspectives. Arch Dermatol. 1996;132(1):71-75.
5. Abdalla CM, de Oliveira ZN, Sotto MN, Leite KR, Canavez FC, de Carvalho CM.
Polymerase chain reaction compared to other laboratory findings and to clinical
evaluation in the diagnosis of cutaneous tuberculosis and atypical mycobacteria
skin infection. Int J Dermatol. 2009;48(1):27-35.
6. Puri P, Ramam M, Ramesh V. Comparison of culture systems for the isolation
of mycobacteria in cutaneous tuberculosis and their drug susceptibility
patterns. Int J Dermatol. 2009;48(9):1017-1018.

Dermatitis and Dangerous Diets:


A Case of Kwashiorkor
Although uncommon, kwashiorkor continues to occur in de-
veloped nations. A recent case highlights the fact that such oc-
currences are typically the result of well-meaning dietary re-
striction in the setting of nutritional ignorance. Telltale skin
and hair changes should prompt a thorough dietary history and
appropriate dietary intervention.
Microscopic results from a liquid culture stained with Ziehl-Neelsen showing
acid-fast M tuberculosis. The cord factor arrangement typical of M tuberculosis Report of a Case | A young boy presented with a 1-year history
can be seen. The positive result was obtained 19 days after the specimen was
of progressive skin, hair, and nail changes after institution of
collected. The arrow indicates a single bacterium.
a low-protein diet recommended by an outside physician as
therapy for his nonketotic hyperglycinemia (NKH). Examina-
Cutaneous tuberculosis can be caused by consuming cow tion revealed generalized hypopigmentation with numerous
milk contaminated with Mycobacterium bovis or by droplet in- erythematous and denuded patches over his trunk, arms, and
fection with M tuberculosis. The correct diagnosis is often sig- legs. Desquamation in a flaking or “paint-chip” pattern was
nificantly delayed because CTB is not routinely considered in prominent on the upper and lower extremities (Figure 1). His
the differential diagnosis or because investigations fail to re- abdomen was distended with dependent edema over the sa-
veal the presence of M tuberculosis.4 crum and extremities. His hair was pale yellow and brittle with
Our case illustrates that scrofuloderma, though a rare dis- patches of alopecia. Fingernails were thin and brittle with dis-
ease in industrialized countries, should still be considered in tal nail plate splitting.
the differential diagnosis of unusual abscesses and nodules of Laboratory levels were measured as follows: total pro-
the neck. Skin testing and interferon-γ release assay can sup- tein, 5.4 g/dL (normal, 5.7-8.2 g/dL); albumin, 2.8 g/dL (nor-
port the clinical diagnosis. Since PCR has been shown to have mal, 3.2-4.8 g/dL); and prealbumin, 8 mg/dL (normal, 10-40
a limited sensitivity and specificity (eg, 88% sensitivity and 83% g/dL). Aspartate transaminase and alanine transaminase lev-
specificity5), there is a risk of failure to detect mycobacteria els were elevated at 76 U/L (normal, <11-34 U/L) and 55 U/L (nor-
in skin samples by relying solely on PCR. Therefore, PCR should mal, 10-49 U/L), respectively. (To convert total protein and al-
always be accompanied by culture.6 bumin to grams per liter, multiply by 10; to convert prealbumin
to milligrams per liter, multiply by 10; to convert aspartate
Ozan Haase, MD transaminase and alanine transaminase to microkatals per li-
Alexander J. von Thomsen, MD ter, multiply by 0.0167.) Levels of alkaline phosphatase, total
Detlef Zillikens, MD bilirubin, iron, phosphorous, magnesium, and stool alpha-1 an-
Werner Solbach, MD titrypsin were within normal limits. Values for zinc, vitamins
Birgit Kahle, MD A, K, and E and 1,25-vitamin D were above or within refer-
ence ranges.
Author Affiliations: Department of Dermatology, University of Luebeck, Our patient’s clinical and laboratory findings were consis-
Luebeck, Germany (Haase, Zillikens, Kahle); Institute of Medical Microbiology
and Hygiene, University of Luebeck, Luebeck, Germany (von Thomsen, tent with kwashiorkor1 secondary to dietary protein restric-
Solbach). tion intended as therapy for NKH, a rare disease of glycine me-
Corresponding Author: Ozan Haase, MD, Department of Dermatology, tabolism causing accumulation of glycine in the cerebrospinal
University of Luebeck, Ratzeburger Allee 160, 23538 Luebeck, Germany (ozan fluid and leading to subsequent N-methyl-D-aspartate recep-
.haase@uksh.de).
tor excitotoxic effects or overstimulation of glutamate recep-
Published Online: April 23, 2014. tors in the central nervous system. These excitotoxic effects
doi:10.1001/jamadermatol.2013.10175.
manifest clinically as intractable seizures, severe mental retar-
Conflict of Interest Disclosures: None reported.
dation, and permanent neurologic disease.2 Glycine is a non-
1. Puri N. A clinical and histopathological profile of patients with cutaneous
essential amino acid produced via numerous catabolic path-
tuberculosis. Indian J Dermatol. 2011;56(5):550-552.
ways; therefore, dietary restriction of glycine has no therapeutic
2. Sehgal VN, Wagh SA. The history of cutaneous tuberculosis. Int J Dermatol.
1990;29(9):666-668. effect on NKH.3 Our patient’s skin changes improved rapidly
3. Lai-Cheong JE, Perez A, Tang V, Martinez A, Hill V, Menagé HduP. Cutaneous with increased dietary protein. Figure 2 demonstrates resolu-
manifestations of tuberculosis. Clin Exp Dermatol. 2007;32(4):461-466. tion of desquamation and erosions at 1-month follow-up.

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