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 of the Neck: Scrofuloderma 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.

Report of a Case | A woman in her 80s was referred for surgical B treatment of a cervical . 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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4. Degitz K. Detection of mycobacterial DNA in the skin. Etiologic insights and Figure 2. Growth of 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 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 1 and Hygiene, University of Luebeck, Luebeck, Germany (von Thomsen, tent with kwashiorkor 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- [email protected]). 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. 3 1990;29(9):666-668. effect on NKH. 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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