Ann Thorac Cardiovasc Surg 2012; 18: 465–467 doi: 10.5761/atcs.cr.11.01728

Case Report Pulmonary in Patient with Anorexia Nervosa: Case Report

Akira Mogi, MD, PhD, Takayuki Kosaka, MD, PhD, Ei Yamaki, MD, and Hiroyuki Kuwano, MD, PhD

A 31-year old female with anorexia nervosa was referred to the Department of General Sur- gical Science at Gunma University for a surgical resection of a pulmonary aspergilloma. The patient had received treatment for anorexia nervosa at the Department of of the Hospital of Gunma University Graduate School of . A showed an infiltrative shadow with apical pleural thickening in the left upper lung field. A contrast enhanced computed tomography showed an irregular mass shadow with cavity formation that involved spherical clusters in the left upper lobe. The patient was diagnosed with pulmo- nary aspergilloma by serological studies and radiological features. A pulmonary segmentec- tomy of the left apical segment (S1 + 2) through a lateral thoracotomy was successfully per- formed. She had an uneventful postoperative recovery, and the final histopathological exami- nation confirmed the diagnosis of pulmonary aspergilloma. This is a rare case study of a young female patient with anorexia nervosa who developed pulmonary aspergilloma.

Keywords: pulmonary aspergilloma, anorexia nervosa, surgical resection

Introduction arises in a preexisting cavity in the lung, and the fungus does not generally invade the surrounding lung paren- Aspergillus is a ubiquitous soil-dwelling organism chyma. On the other hand, few anorexia nervosa patients found in organic debris, dust, compost, foods, spices, and have experienced infectious diseases, such as pulmonary rotted plants. It causes a variety of clinical syndromes in aspergilloma, despite the existence of malnutrition. We the lung, ranging from aspergilloma in patients with lung herein report a rare case of a young woman with anorexia cavities to chronic necrotizing on those who nervosa who developed pulmonary aspergilloma. are mildly immunocompromised or have chronic lung disease.1) Pulmonary aspergilloma, a disease caused by Case Report fungal mycelia, can occur within a cavity, usually within the parenchyma of the lung. An aspergilloma usually A 31-year-old female presented to an internist with chest pain, palpitation, and dyspnea, which occurred sub- Department of General Surgical Science, Gunma University sequent to a cold. She had received outpatient treatment Graduate School of Medicine, Maebashi, Gunma, Japan for anorexia nervosa in the Department of Psychiatry at Gunma University Hospital for 5 years. She was severely Received: July 8, 2011; Accepted: November 8, 2011 Corresponding author: Akira Mogi, MD, PhD. Department of underweight, and her height and weight were 160 cm and General Surgical Science, Gunma University Graduate School of 32 kg, respectively. Her calculated body mass index Medicine, 3-39-22, Showa-machi, Maebashi, Gunma 371-8511, (BMI) was 12.5. Even with a cadaverous body, general Japan conditions were satisfactory. She was referred to the Email: [email protected] ©2012 The Editorial Committee of Annals of Thoracic and Department of because a chest radio- Cardiovascular . All rights reserved. graph showed an infiltrative shadow in the left upper lung

Ann Thorac Cardiovasc Surg Vol. 18, No. 5 (2012) 465 Mogi A, et al.

Fig. 2 Chest computed tomography shows a nodular shadow with cavity formation and an intracavi- tary mass.

Fig. 1 Chest radiography shows an irregular infil- tration in the left upper lung field and apical pleural thickening. field (Fig. 1). A contrast-enhanced computed tomography showed an irregular mass shadow with cavity formation that involved spherical clusters in the left upper lobe (Fig. 2); the lesion was suspected to pulmonary aspergilloma. Bronchoscopy for a transbronchial lung biopsy (TBLB) could not be performed due to her intense pharyngeal re- flex. Serological analyses revealed that the serum asper- gillus antigen was negative, but the serum anti-aspergillus antibody was positive. Other serological and laboratory Fig. 3. A microscopic finding shows multiple fungal studies, including tumor markers such as CEA, SCC, and hyphae with dichotomous branching (Grocott’s NSE, did not show any abnormalities. In addition, bacte- silver stain: original magnification, × 200). rial studies of sputum showed a normal flora. Therefore, she was also diagnosed with pulmonary aspergilloma and was referred to our department for surgical treatment. stain and Alcian blue-periodic acid-Schiff. These hyphae Written informed consent concerning the operation were uniform and regularly separated, and their branch- was obtained from the patient and her family before sur- ing was dichotomous (Fig. 3). Therefore, the final patho- gery. The patient underwent a segmentectomy of the left logical diagnosis was pulmonary aspergilloma. apical segment (S1 + 2) through the lateral thoracotomy The patient had an uneventful recovery and was dis- of the fourth intercostal space. Because the lesion tightly charged on postoperative day 4. She has been monitored infiltrated the adjacent parietal pleura, extrapleural dis- for 7 months as an outpatient without any clinical symp- section was performed to prevent incomplete resection. toms of recurrence, and a chest radiograph produced no Macroscopically, the lesion, which was 3.5 cm in di- abnormal findings. ameter, existed in S1 + 2 of the left upper lobe, and its cavity was filled with fragile materials. Microscopically, Discussion the cavity was surrounded with fibrous walls associated with inflammatory cell infiltration and lymphoid follicles. Pulmonary aspergilloma is the most common form of Intracavitary materials attached to the cavity wall con- pulmonary involvement due to Aspergillus. The aspergilloma tained hyphae positively stained with Grocott’s silver consists of masses of fungal mycelia, inflammatory cells,

466 Ann Thorac Cardiovasc Surg Vol. 18, No. 5 (2012) Pulmonary Aspergilloma fibrin, mucus, and tissue debris, usually developing in a Conclusion preformed lung cavity. is the most frequent cause, but other examples are , bronchiectasis, This is a report of a rare case of young female patient neoplasms, , other fungal infec- with anorexia nervosa who developed pulmonary asperg- tions, cystic fibrosis, and invasive aspergillosis.1–4) Pulmo- illoma. It is considered that mild but chronic systemic nary aspergilloma is known to be an opportunistic infec- immunocompromise resulting from a long-term poor nu- tious disease that patients with immunocompromised or tritional status due to anorexia nervosa was the cause. malnutritional status develop. However, only two cases of Surgical treatment for pulmonary aspergilloma should be aspergilloma in patients with anorexia nervosa have been considered when patients are determined to be able to reported in the literature (as searched on Medline).5,6) Se- withstand the rigors of surgery, regardless of with or vere malnutrition is observed in individuals diagnosed without . with anorexia nervosa; however, few infectious diseases are found to occur concomitantly.7–9) In the case reported References here, we concluded that our young patient with moderate cachexia attributed to anorexia nervosa was immunocom- 1) Soubani AO, Chandrasekar PH. The clinical spectrum promised, which led to the Aspergillus infection. of pulmonary aspergillosis. Chest 2002; 121: 1988-99. Many patients with aspergilloma are asymptomatic, 2) Kauffman CA. Quandary about treatment of aspergil- lomas persists. Lancet 1996; 347: 1640. but the most frequent symptom is hemoptysis. Less com- 3) Zizzo G, Gastriota-Scaderberg A, Zarrelli N, et al. monly, patients develop chest pain, dyspnea, malaise, and Pulmonary aspergillosis complicating ankylosing wheezing. Fever is rare, except in the case of bacterial su- spondylitis. Radiol Med 1996; 91: 817-8. perinfection.1,4,10) In the case reported here, the patient 4) Glimp RA, Bayer AS. Pulmonary aspergilloma: diag- was afebrile without hemoptysis, but chest pain and dysp- nostic and therapeutic considerations. Arch Intern Med 1983; 143: 303-8. nea were observed. In asymptomatic patients, no treat- 5) Takushima M, Haraguchi S, Hioki M, et al. Video-as- ment is required, and observation is warranted in most sisted thoracic surgery for pulmonary aspergilloma in cases. On the other hand, patients with aspergilloma who patients with anorexia nervosa. J Nippon Med Sch experience symptoms of hemoptysis should undergo sur- 2004; 71: 333-6. gical treatment because there is a risk of sudden life- 6) Noter SL, Hendriks ER, Steup WH, et al. Aspergil- threatening hemoptysis and there is no effective alterna- loma of the lung due to aspiration during nasal tube 11) feeding. Gen Thorac Cardiovasc Surg 2009; 57: 169- tive medical . There is no consistent evidence 70. that aspergilloma responds to antifungal agents, and such 7) Hotta M, Nagashima E, Takagi S, et al. Two young fe- drugs rarely achieve the minimal inhibitory concentra- male patients with anorexia nervosa complicated by tions within the lung cavities.12) Surgical treatment ap- Mycobacterium tuberculosis infection. Intern Med pears controversial in this case because no hemoptysis 2004; 43: 440-4. 8) Bowers TK, Eckert E. Leukopenia in anorexia nervosa: had occurred, but the patient was symptomatic. The rea- lack of increased risk of infection. Arch Intern Med sons for surgical treatment in this case were as follows: 1) 1978; 138: 1520-3. she was young, and her pulmonary function was ade- 9) Marcos A. The immune system in eating disorders: an quately preserved; 2) her prolonged symptoms possibly overview. Nutrition 1997; 13: 853-62. interfered with dietary intake in the control of anorexia 10) Massard G, Roeslin N, Wilhm JM, et al. Pleuropulmo- nervosa; 3) the patient agreed to receive surgical treat- nary aspergilloma: clinical spectrum and results of surgical treatment. Ann Thorac Surg 1992; 54: 1159- ment after receiving detailed information about it. With 64. regard to the surgical treatment, segmentectomy of S1 + 11) Regnard JF, Icard P, Nicolosi M, et al. Aspergilloma: 2 in the left upper lobe was chosen in this case because A series of 89 surgical cases. Ann Thorac Surg 2000; the estimated resection margin from aspergilloma was 69: 898-903. sufficient, as indicated by the detailed information from 12) Pennington JE. Aspergillus lung disease. Med Clin North Am 1980; 64: 475-90. pre-operative three-dimensional computed tomography. Indeed, the complete resection of the lesion was proven by a post-operative pathological study.

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