Akihito Tanabe

Hideaki Kaneto

Kenji Kohara

Kohei Kaku

Maiko Takai

Masashi Shimoda

Ryo Shigemoto

Shinji Kamei

Shintaro Irie

Tomoatsu Mune

Yurie Hirata

PubMed

Crossref

Acta Diabetologica

Hamman’s syndrome triggered by the onset of type 1 diabetes mellitus accompanied by diabetic ketoacidosis

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LETTER TO THE EDITOR
Hamman’s syndrome triggered by the onset of type 1 diabetes
mellitus accompanied by diabetic ketoacidosis
Shinji Kamei1 • Hideaki Kaneto1 • Akihito Tanabe1 • Ryo Shigemoto1 •
Shintaro Irie1 • Yurie Hirata1 • Maiko Takai1 • Kenji Kohara1 • Masashi Shimoda1 •
Tomoatsu Mune1 • Kohei Kaku1
Received: 18 May 2016 / Accepted: 23 May 2016 / Published online: 13 June 2016
 The Author(s) 2016. This article is published with open access at Springerlink.com
Dear Editor,
Hamman’s syndrome, which is also known as spontaneous
pneumomediastinum, could be induced in various situa-
tions such as labor and delivery [1]. The potential mech-
anism for Hamman’s syndrome is as follows: The
vulnerability of alveolar walls by some reason increases air
pressure, which leads to alveolar rupture. Then, spilled air
peels the connective tissue and finally enters into medi-
astinal space through pulmonary hilum. Hamman’s syn-
drome is one of the relatively rare pulmonary diseases, and
there are few reports showing the case of Hamman’s syn-
drome triggered by the onset of type 1 diabetes mellitus
accompanied by diabetic ketoacidosis. However, recently
we experienced three cases of young male subjects with
Hamman’s syndrome triggered by the onset of type 1
diabetes mellitus accompanied by diabetic ketoacidosis.
These cases are very rare, and there were many similarities
among these three cases.
Clinical characteristics of these three cases were as fol-
lows: age (years old): 29, 20, 20 (case 1, case 2, case 3);
gender: male, male, male; HbA1c (%): 12.0, 12.3, 6.9;
plasma glucose level (mg/dL): 1298, 1543, 783; pH: 6.89,
7.10, 7.24; total ketone body concentration in blood (lmol/
L): 1662, 20,960, 15,500; acetoacetic acid (lmol/L): 734,
5080, 2960; 3-hydroxybutyric acid (lmol/L): 928, 15,880,
12,540; WBC (/mL): 37,370, 27,590, 7050; CRP (mg/dL):
4.01,\0.03, 0.76, respectively. Type of diabetes in all three
subjects was type 1A, and the autoantibodies of the pan-
creas were positive: anti-GAD antibody (U/mL): 157.7, 3.0,
1.5 (case 1, case 2, case 3); anti-IA-2 antibody (U/mL): 2.6,
5.6,\0.4, respectively. Marked reduction in body weight
was observed: BMI on admission (kg/m2): 25.1, 13.7, 19.0;
BMI before the onset of type 1 diabetes (kg/m2): 29.3, 17.2,
21.7; recent weight loss (kg/duration): 10 kg/1 month,
11 kg/2 weeks, 8 kg/1 week, respectively. All three cases
experienced vomiting very frequently, and case 1 and 2, but
not case 3, experienced chest pain. On physical examina-
tion, subcutaneous emphysema was observed in all three
cases. In chest X-ray, pneumomediastinum was observed in
these three cases (red arrows) (Fig. 1). Also, in computed
tomography, such pneumomediastinum was confirmed in
these three cases (red circles) (Fig. 1). In case 1, pneumonia
image was observed in lower right lung field (blue arrows
and blue circle). In addition, subcutaneous emphysema was
observed in all cases.
We immediately started antibiotics therapy in order to
prevent the progression of inflammation. Mediastinal
emphysema image in chest X-ray disappeared in several
days after starting the therapy. The number of days
required for the disappearance of emphysema image in
these three cases was 4, 9 and 5 days, respectively. The
number of days until the complete recovery was 15, 9 and
6 days, respectively. There were no sequelae and no
recurrence of emphysema at all in all cases.
It is thought that in cases with diabetic ketoacidosis air
pressure could be increased by Kussmaul’s breathing or
repeated vomiting [2, 3]. Therefore, we think that repeated
vomiting might have triggered the onset of Hamman’s
syndrome in these three subjects. In addition, decrease in
pulmonary surfactant observed with diabetic ketone aci-
dosis could lead to the vulnerability of alveolar walls [4].
Managed by Antonio Secchi.
& Shinji Kamei
s.kamei@med.kawasaki-m.ac.jp
1 Department of Diabetes, Metabolism and Endocrinology,
Kawasaki Medical School, 577 Matsushima,
Kurashiki 701-0192, Japan
123
Acta Diabetol (2016) 53:1067–1068
DOI 10.1007/s00592-016-0871-z
Therefore, although speculative, such phenomena might
have been also involved in the onset of Hamman’s syn-
drome in these cases.
In conclusion, we should be aware of the possibility that
Hamman’s syndrome is induced by the onset of type 1
diabetes mellitus accompanied by diabetic ketoacidosis.
Since such pneumomediastinum sometimes becomes very
severe and leads to a fatal outcome [5], we think it would
be better to perform computed tomography for many sub-
jects with diabetic ketoacidosis.
Author contributions S.K. researched data and wrote the manu-
script. H.K. wrote and reviewed/edited the manuscript. A.T., R.S.,
S.I., Y.H., M.T., K.Ko. M.S., T.M. and K.Ka. researched data and
contributed to the discussion.
Compliance with ethical standards
Conflict of interest The authors declare that there is no conflict of
interest associated with this manuscript.
Ethical standard All procedures followed were in accordance with
the ethical standards of the responsible national committee on human
experimentation and with the Helsinki Declaration of 1975, as revised
in 2008.
Human and animal rights All procedures performed in studies
involving human participants were in accordance with the ethical
standards of the national research committee and with the 1964
Helsinki declaration and its later amendments or comparable ethical
standards.
Informed consent Informed consent was obtained from the reported
patient.
Open Access This article is distributed under the terms of the
Creative Commons Attribution 4.0 International License (http://crea
tivecommons.org/licenses/by/4.0/), which permits unrestricted use,
distribution, and reproduction in any medium, provided you give
appropriate credit to the original author(s) and the source, provide a
link to the Creative Commons license, and indicate if changes were
made.
References
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stage of labor. Gynecol Obstet Invest 67:70–72
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LW, Wood BP (1989) Radiological case of the month. Pneumo-
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hyperpnea. Am J Dis Child 143:93–94
3. Pooyan P, Puruckherr M, Summers JA, Byrd RP Jr, Roy TM
(2004) Pneumomediastinum, pneumopericardium, and epidural
pneumatosis in DKA. J Diabetes Complicat 18:242–247
4. Sahebjami H, Vassallo CL, Wirman JA (1978) Lung mechanics
and ultrastructure in prolonged starvation. Am Rev Resp Dis
117:77–83
5. Kaneki T, Kubo K, Kawashima A, Koizumi T, Sekiguchi M, Sone
S (2000) Spontaneous pneumomediastinum in 33 patients: yield of
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Respiration 67:408–411
case 1 case 2 case 3 Fig. 1 Chest X-ray (upper
panels) and computed
tomography (middle and lower
panels) in three cases with
Hamman’s syndrome triggered
by the onset of type 1 diabetes
mellitus accompanied by
diabetic ketoacidosis.
Pneumomediastinum is
observed in these three cases
(red arrows and red circles). In
case 1, pneumonia image is
observed in lower right lung
field (blue arrows and blue
circle)
1068 Acta Diabetol (2016) 53:1067–1068
123


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Hamman’s syndrome triggered by the onset of type 1 diabetes mellitus accompanied by diabetic ketoacidosis

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